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This report presented a case of a Shepherd fracture of the lateral tubercle of the posterior process of the talus which was successfully managed by arthroscopic fixation. There are some inherent general features about the talus which may contribute to significant functional problems after injury including its complex anatomy, its involvement in multiple joints (tibiofibular, talocalcaneal and talonavicular), the large surface area that is covered by cartilage and its relatively poor vascularity . The functional outcome and the ability to return to the normal pre-injury level of activity can, however, be impaired in a significant number of cases. In this regard, Engelmann et al . have suggested that in those patients initially managed non-operatively (or where operative fixation was delayed), that there is a higher rate of complications which include chronic painful non-union, post-traumatic osteoarthritis and posterior ankle impingement. These patients have also been shown to have a greater need for secondary arthrodesis . | 4.054688 | 0.955078 | sec[2]/p[0] | en | 0.999996 | 39075516 | https://doi.org/10.1186/s13256-024-04652-7 |
A twenty-two-day-old, 3.65 kg male, presented to the emergency department (ED) due to concern for potential accidental flecainide overdose. The patient had been delivered by caesarean section at 36 weeks’ gestation after recognition of in utero supraventricular tachycardia (SVT). The SVT resolved spontaneously after birth. Two weeks later, the infant presented to an outside ED for evaluation of emesis and rapid heart rate. At that time, he was found to have SVT that did not respond to three doses of adenosine, 0.1 mg/kg once followed by 0.2 mg/kg twice. He was transferred to the regional neonatal intensive care unit for higher level of care. On arrival, he was noted to have a heart rate of 300 beats per minute; pediatric cardiology felt this represented atrial flutter. Echocardiography at that time showed a diminished ejection fraction of 17%. Synchronized electrical cardioversion at 1 J/kg was then used to successfully treat the dysrhythmia, restoring normal sinus rhythm after a single shock. | 3.894531 | 0.981445 | sec[0]/p[0] | en | 0.999998 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
During this hospitalization, amiodarone 9 mg/kg total and propranolol 2 mg/kg/day divided every 6 h were initially trialed at the recommendation of pediatric electrophysiology, along with a milrinone infusion due to the patient’s low ejection fraction. This regimen was ultimately unable to prevent recurrent episodes of SVT, which responded well to 0.2 mg/kg adenosine boluses. Consequently, oral flecainide 98 mg/m 2 /day (10 mg) divided every 12 h and propranolol 2.4 mg/kg/day (2 mg) divided every 6 h were used and successfully maintained durable sinus rhythm. | 3.871094 | 0.956055 | sec[0]/p[1] | en | 0.999998 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
The patient was discharged on oral flecainide 10 mg twice per day, and oral propranolol 2 mg four times per day. An electrocardiogram (ECG) obtained prior to discharge on the appropriate doses of flecainide and propranolol, demonstrated a rate of 111 beats per minute, PR interval of 106 ms, QRS interval of 80 ms, and QTc interval of 454 ms. At that time, the serum flecainide concentration was 0.37 µg/mL (therapeutic range 0.20-1.00). | 3.855469 | 0.961426 | sec[0]/p[2] | en | 0.999997 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
Several notable examples of flecainide’s narrow therapeutic index and potentially dangerous toxicity in very young pediatric patients have been reported in the literature. An 18-day-old received a two-fold dosing error (8 mg every eight hours instead of 4 mg every 8 h for four doses) due to a labeling error. Following the fourth dose, the neonate developed bradycardia without a palpable pulse. Following atropine administration, a wide complex tachycardia occurred that was successfully treated with sodium bicarbonate . A 23-month-old was found to have a new bifascicular block following a single, doubled-dose . A 48-day-old received nine doses of flecainide at 1.7 times the recommended upper pediatric dosing limit. Following the seventh dose, convulsions occurred, and following the ninth dose a wide complex tachycardia was recognized that resolved without intervention in three days . A four-month-old received six times the intended dose as a result of a syringe measurement error for multiple days. A sustained wide complex tachycardia with hemodynamic instability developed and was successfully treated with venoarterial extracorporeal membrane oxygenation (VA-ECMO) . A single, five-fold dosing error administered to a 7-month-old as a result of syringe measurement error caused pulseless ventricular tachycardia that was successfully treated with sodium bicarbonate boluses, lipid emulsion, and VA-ECMO . | 4.132813 | 0.897461 | sec[1]/sec[0]/p[1] | en | 0.999997 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
Upon hospital presentation after the dosing error was recognized, the patient appeared well. Vital signs and physical examination were normal. An ECG demonstrated a heart rate of 113, PR interval of 118 ms, QRS interval of 138 ms, and QTC interval of 513 ms. Laboratory testing was notable for a serum potassium of 5.7 mmol/L, magnesium 1.9 mg/dL, and calcium 11.3 mg/dL. Renal function and liver enzymes were within normal ranges. A flecainide concentration was drawn, but results were not rapidly available as it was a send-out test. | 3.638672 | 0.977051 | sec[2]/p[0] | en | 0.999996 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
The patient’s ECG is consistent with flecainide toxicity. Electrocardiography is the primary diagnostic tool for demonstrating the cardiotoxic effects of flecainide, regardless of clinical symptoms . The ECG shown in Fig. 2 demonstrates interval prolongation of the PR, QRS, and QTc intervals from therapeutic baseline . These are findings consistent with the known electrophysiologic effects of flecainide . It is valuable to recognize that prolongation of the PR, QRS, and QT intervals also occur at therapeutic dosing and the degree of prolongation is expected to correlate with increasing concentrations of flecainide . | 4.042969 | 0.563477 | sec[2]/sec[0]/p[0] | en | 0.999998 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
On review of potential alternative explanations to the described dosing error for the observed electrocardiographic changes, none were likely. The patient had been taken off of amiodarone prior to flecainide initiation and showed no signs of a drug interaction prior to discharge. Metabolic impairment of CYP2D6 would be expected to manifest electrocardiographic abnormalities more rapidly after flecainide initiation. No clinically significant electrolyte abnormalities were noted at the time of presentation. And finally, the patient had no known exposure to CYP2D6 inhibitors. The patient was breastfeeding and, to our knowledge, his mother was not on medications that could have passed to the patient via breast milk. Consequently, the described dosing error is the most plausible explanation for the patient’s ECG abnormalities. | 3.900391 | 0.920898 | sec[2]/sec[0]/p[2] | en | 0.999997 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
The beneficial effects of intravenous sodium bicarbonate generally manifest within minutes of administration and the clinical response can be dramatic. For example, an 18-day-old in cardiogenic shock from a wide complex tachycardia converted to a hemodynamically stable, narrow complex tachycardia after successive administration of two 10 mEq (5 mEq/kg) boluses . In one adult case, intravenous sodium bicarbonate restored spontaneous circulation by rapidly terminating flecainide-induced ventricular fibrillation . | 3.998047 | 0.558594 | sec[2]/sec[2]/p[2] | en | 0.999997 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
Although the efficacy of intravenous sodium bicarbonate is well established, it is not a panacea. Life-threatening toxicity despite aggressive sodium bicarbonate administration can occur. Notably, a 33-year-old developed ventricular tachycardia leading to cardiac arrest and initiation of extracorporeal circulatory support hours after ingestion despite treatment with a total of 850 mEq bolus and a 300 mEq per hour infusion of intravenous sodium bicarbonate . | 3.552734 | 0.875 | sec[2]/sec[5]/p[0] | en | 0.999995 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
The medical toxicology service was consulted (authors JS, AS) and recommended admission to the neonatal intensive care unit for close monitoring on telemetry. We did not recommend administration of activated charcoal or sodium bicarbonate, nor any other interventions at the time of admission. The patient had most recently ingested a therapeutic flecainide dose 2.5 h prior to presentation. As flecainide is rapidly absorbed, with peak plasma concentrations in 1 to 3 h, we felt the patient’s presentation likely represented post peak toxicity and we did not anticipate clinical worsening . Further, it would be challenging to administer activated charcoal to a neonate and require placement of a nasogastric tube, which would take significant additional time and thus be of limited clinical utility. | 3.400391 | 0.94873 | sec[3]/p[0] | en | 0.999997 | 38992232 | https://doi.org/10.1007/s13181-024-01018-9 |
Hypothyroidism is a common clinical condition that results from inadequate production of thyroid hormones or inadequate action of thyroid hormones in target tissues. If left untreated, hypothyroidism can lead to serious adverse effects on multiple organ systems. Acquired primary hypothyroidism is the most prevalent form worldwide with iodine deficiency and Hashimoto thyroiditis as the main causes ( 1 ). The clinical picture varies, with presentations ranging from asymptomatic to overt. Those with symptoms may present with fatigue, cold intolerance, and constipation ( 1 ). Complaints of muscle stiffness and myalgia may be elicited from patients with undiagnosed hypothyroidism ( 2 ). When measured, muscle enzymes may be elevated, but are usually less than 10 times the upper limit of normal ( 3 ), and rhabdomyolysis is rare ( 2 ). In most of the reported cases of rhabdomyolysis, a precipitating factor has been identified such as strenuous exercise ( 4 ) or the use of lipid-lowering drugs ( 5-8 ). Rhabdomyolysis associated with hypothyroidism without an obvious precipitating factor has been previously reported in the literature but mostly in adults ( 3 , 9-11 ). We describe the case of a prepubertal child presenting with rhabdomyolysis caused by profound primary hypothyroidism. | 4.191406 | 0.92627 | sec[0]/p[0] | en | 0.999998 | 39081695 | https://doi.org/10.1210/jcemcr/luae118 |
A 12-year-old boy with past medical history of asthma, exogenous obesity, and sleep apnea presented to the emergency room after an episode of severe painful muscle cramping preceded by a 3-month history of generalized muscle cramping. Initially, the muscle cramping was noted in the lower extremities, then gradually progressed to involve the lower back and chest. The patient was previously evaluated by his primary care pediatrician, who attributed the symptoms to growing pains and dehydration. He was instructed to increase water intake and start a multivitamin. His history was negative for increased physical exertion, environmental heat exposure, recent illnesses, illicit substances, or supplements. The patient was not on any daily medications. Review of systems was positive for cold intolerance, constipation, pale skin tone, and weight gain. The family did not report dark (tea-colored) urine. Review of his historical growth curve showed that his height had decelerated from the 78th percentile to the 49th percentile over the previous 2 years, while he had gained 45 pounds with a body mass index Z -score change from +2.25 to +2.5 over the same period. Family history was significant for autoimmune thyroiditis in his mother and maternal grandmother. | 3.777344 | 0.986328 | sec[1]/p[0] | en | 0.999997 | 39081695 | https://doi.org/10.1210/jcemcr/luae118 |
The patient appeared well in the emergency room with no evidence of goiter or edema. His neurologic and musculoskeletal examination was normal including no documented muscle tenderness. His vitals were normal except for a heart rate of 61. Initial laboratory evaluation in the emergency room was significant for a creatinine of 1.39 mg/dL (reference, 0.4-1 mg/dL; 122.88 µmol/L) and creatine kinase (CK) of 2056 U/L (reference, 0-300 U/L; 34.34 µkat/L) ( Table 1 ). Liver enzymes showed mildly elevated aspartate transaminase 62 U/L (reference, 5-50 U/L; 1.04 µkat/L) and normal alanine transaminase 28 U/L (reference, 5-50 U/L; 0.47 µkat/L). Urinalysis was normal and renal ultrasound was obtained, which showed no abnormalities. The patient was admitted for management of acute kidney injury (AKI) secondary to rhabdomyolysis and was started on intravenous fluid hydration. During his first day of admission, he was noted to have bradycardia with a heart rate of 45 bpm. Electrocardiogram showed bradycardia with intraventricular conduction delay. Because of the bradycardia, thyroid function testing were drawn that revealed a thyrotropin (TSH) level of 494 mIU/mL (reference, 0.40-6.00 mIU/mL) with low free thyroxine (T4) of less than 0.4 ng/dL (reference, 0.80-1.80 ng/dL; <5.15 pmol/L). Additional laboratory tests showed strongly positive thyroglobulin 51 U/mL (reference ≤ 4 U/mL; 51 kIU/L) and thyroid peroxidase antibodies 1467 U/mL . Thyroid ultrasound showed a solid and cystic nodule in the inferior left thyroid lobe measuring 1.2 × 1.4 × 1 cm. Laboratory work was also significant for dyslipidemia, mild normocytic anemia, hemoglobin 11.7 gm/dL (reference, 12.5-16.1 g/dL; 117 g/L), and low vitamin D level 14 ng/mL (reference, 30-100 ng/mL; 34.94 nmol/L). | 3.978516 | 0.975586 | sec[2]/p[0] | en | 0.999997 | 39081695 | https://doi.org/10.1210/jcemcr/luae118 |
Rhabdomyolysis is a rare complication of hypothyroidism in adults and even more rare in children. Most patients who develop rhabdomyolysis due to hypothyroidism have an inciting event such as heavy exercise, illness, or medications such as statins ( 6 ). Our patient did not have an inciting event but was symptomatic with myalgias over 3 months. This led to symptoms being attributed to viral illness and dehydration, thus delaying the diagnosis of rhabdomyolysis. Additional clinical signs of bradycardia led to further biochemical testing, revealing hypothyroidism as the etiology of rhabdomyolysis. Often with rhabdomyolysis secondary to hypothyroidism, the level of CK is noted to be less than 10 times the normal reference range ( 16 ). In this case, the CK was 6.8 times the normal reference range. | 3.996094 | 0.708496 | sec[5]/p[1] | en | 0.999997 | 39081695 | https://doi.org/10.1210/jcemcr/luae118 |
Primary treatment for hypothyroidism-induced rhabdomyolysis is levothyroxine repletion. Acutely, hydration will be important to treat symptoms, decrease the CK levels, and prevent worsening AKI. In this case, treatment of hypothyroidism addressed both the muscle pathology and renal dysfunction with a downtrend in creatinine and CK over weeks. Future episodes are unlikely with adequate thyroid hormone replacement. This case highlights that clinicians should consider thyroid function testing in patients with unexplained elevations in CK. A thorough history and tailored evaluation will aid in early diagnosis and optimal treatment. | 3.966797 | 0.73584 | sec[5]/p[3] | en | 0.999998 | 39081695 | https://doi.org/10.1210/jcemcr/luae118 |
After general anesthesia, the patient was placed in a supine position with the head extended backward as much as possible. The patient was prepared and draped in the usual sterile fashion. First, a supportive laryngoscope was used to lift and fully expose the epiglottis. Tetracaine (1%) was applied to the surface of the glottis for anesthesia, and a rigid bronchoscope was placed . High-frequency-assisted ventilation was performed through the side hole of the bronchoscope, and the foreign body was observed. Suitable foreign body forceps were then selected to quickly remove the foreign body, clean the secretions in the trachea, and explore the healthy side of the trachea to ensure that no foreign body remained. We used foreign body forceps during the surgery as shown in Figure 2 . We choose different forceps based on the actual situation during the surgical process. Conventional special types of foreign bodies usually choose forceps No.2 and No.4. Sharp or hard foreign bodies usually choose forceps No.1 and No.3. During this period, the vital signs of the child were closely monitored. | 3.751953 | 0.771973 | sec[1]/sec[1]/p[0] | en | 0.999996 | 39086627 | https://doi.org/10.3389/fped.2024.1395629 |
Our focus in this case report is on the t(11;14) mutation, a translocation that implicates CCND1 and immunoglobulin heavy locus (IgH) genes. These mutations are found in approximately 15-20% of MM cases and are associated with a favorable prognosis. Patients with this mutation exhibit a unique biological feature compared to other MM cell types, characterized by an increase in the presence of the B-cell lineage membrane protein CD20, higher levels of the B-cell receptor CD79a, and a decrease in the expression of both CD38 and CD56 . | 4.09375 | 0.702637 | sec[0]/p[2] | en | 0.999999 | 39087203 | https://doi.org/10.7759/cureus.63563 |
The patient was a 75-year-old male with a past medical history of diabetes mellitus, osteoporosis, and benign prostatic hyperplasia. He was referred by the oncologist for comprehensive diagnostic testing involving fluorescence in situ hybridization (FISH), a BM biopsy for histopathological diagnosis, as well as flow cytometry and analysis of the aspirate due to a recent diagnosis of MM. FISH testing confirmed significant hyperploidy involving trisomy 9 and an extra copy of CCND1, with concomitant trisomy 11q confirming a t(11;14) translocation (Table 1 ). | 3.839844 | 0.958008 | sec[1]/p[0] | en | 0.999997 | 39087203 | https://doi.org/10.7759/cureus.63563 |
We discussed the case of a 75-year-old male who was referred to the clinic by his oncologist for comprehensive diagnostic testing. Upon review of the patient's test results, it was evident that he possessed a t(11;14) translocation, which is common in 15-20% of patients with MM, and showed a rare elevation in his CD38 marker levels. The patient is currently being treated with daratumumab on a four-cycle assessment and response protocol, followed by tumor reassessment using CT imaging and response evaluation. The patient is on his fourth cycle and has yet to show any adverse effects to the treatment, and is tolerating it well. To the best of our knowledge, this is the first report of a case with MM t(11;14) subtype with elevated CD38 marker levels. We believe our findings will be beneficial to the scientific community. | 3.855469 | 0.978516 | sec[3]/p[0] | en | 0.999997 | 39087203 | https://doi.org/10.7759/cureus.63563 |
Herein, we report a case of bladder cancer metastatic to the bone marrow that presented as TMA. | 3 | 0.977051 | sec[0]/p[4] | en | 0.999998 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
The patient was admitted to our hospital’s urology department for further evaluation. Cystoscopy revealed a tumor in the right lateral bladder wall. Transurethral resection of the bladder tumor (TURBT) revealed high-grade urothelial cell carcinoma (grade III–IV) invading the muscularis propria with focal necrosis. Vascular and lymphatic invasion was highly suspected . | 3.316406 | 0.986816 | sec[1]/p[2] | en | 0.999996 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
A full-body CT scan was requested as the pelvic CT showed an enhancing asymmetric formation protruding from the right posterolateral bladder wall, adjacent to the right iliac vessels with no signs of invasion . The remaining CT scans did not reveal any significant findings or clear evidence of metastatic disease. However, the patient continued to experience bleeding, which resulted in acute anemia. The patient’s condition was complicated by worsening thrombocytopenia. The decision was made to admit the patient to the hematology department. A standard workup for hemolysis was performed ( Table 2 ). | 3.404297 | 0.986816 | sec[1]/p[3] | en | 0.999997 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
A peripheral blood smear was made after two days and, demonstrating the presence of schistocytes, reticulocytes, helmet cells, band neutrophils, and unencapsulated RBCs, which established the diagnosis of TMA . Nevertheless, the patient showed no signs of neurological or renal disorders. | 3.0625 | 0.959961 | sec[1]/p[4] | en | 0.999996 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
He was started on plasmapheresis with high-dose steroids for five days. Given the lack of response to previous treatment, a bone marrow aspiration from the iliac crest was performed. The aspiration smear revealed crowded nests of neoplastic cells with large hyperchromatic nuclei, which displayed a picture of metastatic carcinoma . Owing to these abnormal results, a bone marrow biopsy was performed. | 3.351563 | 0.982422 | sec[1]/p[5] | en | 0.999995 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
Pathological examination of the biopsy specimen revealed that the entire bone marrow was infiltrated by poorly differentiated carcinoma, most likely of urothelial origin, which established the diagnosis of advanced bladder cancer metastatic to the bone marrow (grade IV) . | 3.541016 | 0.729492 | sec[1]/p[6] | en | 0.999997 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
The patient was evaluated and considered to be a candidate for platinum–based chemotherapy regimen. Carboplatin was favored over cisplatin due to its lower rate of nephrotoxicity as the patient’s creatinine values increased from 0.6 mg/dl to 1.2 mg/dl after the first dose of cisplatin, and then the patient started on a first-line chemotherapy with gemcitabine on days 1 and 8 and carboplatin on day 8, repeated every 21 days for three cycles. Prophylactic granulocyte colony-stimulating factor (G-CSF) was administered once per cycle, and the results are shown in ( Table 3 ). | 3.851563 | 0.913574 | sec[1]/p[7] | en | 0.999996 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
Bladder cancer is the most frequent malignancy of the urinary tract , with approximately 20% of lesions being muscle—invasive . Muscle—invasive bladder cancer (MIBC) can also progress to metastatic bladder cancer, and metastasizes to the bone marrow . However, bone marrow infiltration by metastatic cancer can result in thrombocytopenia and MAHA. The occurrence of microangiopathic hemolytic anemia and thrombocytopenia in patients can be unexpected and alarming, and these abnormalities are typical clinical features of TMA syndromes . The occurrence of TMA has been documented in a number of cancers, including metastatic gastric cancer, ovarian cancer, prostate cancer, lung cancer, urothelial cancer, lymphoma, myeloproliferative neoplasm, and acute myeloid leukemia . TMA symptoms include renal impairment, neurological impairment, cardiac impairment, hemorrhage, venous thrombosis, and shock . The first step in the diagnostic pathway is the detection of TMA, which is defined as the presence of thrombocytopenia and MAHA. Thrombocytopenia is either absolute (platelet count <150 × 109/L) or relative (>25% reduction in platelet count from baseline). Microangiopathic hemolytic anemia is diagnosed when there are signs of anemia and hemolysis, such as schistocytes in a blood smear, increased lactate dehydrogenase, serum free hemoglobin, reticulocytosis, and diminished haptoglobin which may be a delicate, but nonspecific feature . In our case, there were no signs of neurological or renal disorders, and everything else indicated TMA. The diagnosis was made even though our patient did not have typical symptoms, where urinary bleeding from the bladder manifested by hematuria was the primary symptom. However, the full appearance of symptoms and signs occurs only in a small percentage of patients. Bone marrow aspiration is not required, but may facilitate differential diagnosis, especially in cases of TMA associated with cancer . Plasma exchange therapy (PEX) in combination with steroids is usually the primary treatment, but if clear cases of secondary TMA that are unresponsive to PEX are identified, only supportive care is provided, and further treatment is provided depending on the final diagnosis . In our case, the patient did not show any improvement after treatment with plasma exchange and steroids, which prompted us to treat the cancer with chemotherapy (gemcitabine and carboplatin). | 4.328125 | 0.647461 | sec[2]/p[0] | en | 0.999997 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
In this report, we present a rare case of metastatic bladder cancer that metastasized to the bone marrow, leading to secondary TMA syndrome that did not respond to standard treatment with plasma exchange and steroids. We were able to diagnose this condition and completely control the symptoms using chemotherapy with gemcitabine and platinum-based agents. | 3.826172 | 0.976563 | sec[3]/p[0] | en | 0.999997 | 39087090 | https://doi.org/10.1093/omcr/omae081 |
Primary central nervous system (CNS) lymphoma is a rare condition, accounting for 0.45 per 100,000 population . Diffuse large B cell lymphoma accounts for most primary CNS lymphomas, whereas primary CNS T cell lymphoma is relatively uncommon, accounting for 2% of all primary CNS lymphomas . Primary CNS lymphomas are generally characterized by clinical and neuroimaging findings consistent with those of single or multiple intracranial mass lesions. However, lymphomatosis cerebri (LC) is a rare subtype of primary CNS lymphoma, presenting with extensive white-matter lesions on magnetic resonance imaging (MRI) and nonspecific symptoms, such as cognitive decline and depression . Given the nonspecific clinical and neuroimaging characteristics, the diagnosis of LC often presents a considerable challenge, requiring careful consideration of various potential causes. CNS infections, inflammatory conditions, toxic insults, and metabolic disorders can imitate the radiological features observed in LC , leading to a delayed and incorrect diagnosis. The median overall survival in untreated patients was 1.97 months, while in patients receiving methotrexate-based therapies, it was 13.8 months. Particularly, given that the initiation of therapeutic measures during a favorable Karnofsky performance status is positively correlated with a favorable prognosis, early diagnosis and prompt intervention are deemed pivotal . In previous LC reports, the proportion of T cell type is 8.9%, and none of them was caused by human T cell leukemia virus type 1 (HTLV-1) [ 7 – 10 ]. Herein, we present the case of LC associated with adult T cell leukemia/lymphoma, aiming to contribute toward expediting the diagnostic process in such scenarios. | 4.304688 | 0.759766 | sec[0]/p[0] | en | 0.999998 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
A 49-year-old Japanese man with no history of physical or mental illness gradually developed symptoms of insomnia, anorexia, and weight loss approximately 1 month after being promoted at work and after an offer to be transferred at work 3 months before visiting our hospital. One month prior to his visit, he consulted an internist with complaints of poor concentration and somnolence during the day and was suspected of having depression. He was subsequently referred to a psychiatrist who prescribed duloxetine 20 mg/day. However, his spontaneity rapidly declined, and he required assistance with toileting and walking. The following week, he had difficulty talking, was judged to be comatose, and was subsequently admitted to a psychiatric hospital. At the time of admission, the patient could barely transfer himself to a wheelchair or open his eyes, and he displayed poor voluntary body movements and eye pursuit. He rapidly became bedridden. Intranasal injections of vortioxetine (a selective serotonin reuptake inhibitor) 10 mg/day, aripiprazole 3 mg/day, lorazepam 2 mg/day, and mirtazapine 15 mg/day were administered; however, the patient’s condition persisted with a Japan Coma Scale score of 200 points (equivalent to approximately six points on the Glasgow Coma Scale score ). He was suspected of having malignant catatonia, and despite being treated with intramuscular injections of lorazepam 2 mg/day and diazepam 20 mg/day, his symptoms did not improve. After 3 weeks of psychiatric inpatient treatment, he was referred to our hospital to exclude organic disease. | 3.71875 | 0.984375 | sec[1]/p[0] | en | 0.999994 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
At the time of admission, the patient had a Glasgow Coma Scale score of five points (E1V1M3) and presented the following vital signs: blood pressure, 153/106 mmHg; pulse rate, 106 beats per minute; respiratory rate, 21 breaths per minute; and oxygen saturation, 94% on room air. The general physical examination findings were normal. The neurological examination revealed the pupil size to be 3/3 mm, and light and eyelash reflexes to be absent bilaterally. The ocular position was predominantly left, and leftward horizontal nystagmus was observed in both eyes. Muscle tone was strong in the extremities, and tendon reflexes were enhanced, with right predominance in the upper body and left predominance in the lower body: brachioradialis +/+ , biceps ++ / + , triceps ++/+ , patellar +/++ , Achilles +/+ , ankle clonus −/+ , Babinski, Chaddock, Hoffman, and Trömner reflexes were absent, and there was a slight escape response to pain stimuli in all extremities. | 3.957031 | 0.977051 | sec[1]/p[1] | en | 0.999997 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
Blood tests revealed a white blood cell count of 17,420/μL, with differential percentages as follows: neutrophils 86.6%, lymphocytes 7.3%, monocytes 3.4%, eosinophils 1.2%, and basophils 0.6%. Red blood cell parameters (hemoglobin 15.2 g/dL, hematocrit 50.5%) and platelet count (297,000/μL) were within normal ranges. Biochemical and immunological tests showed a C-reactive protein level of 1.67 mg/L, with other data within normal limits. The syphilis, antinuclear antibody, copper, and vitamin B1 test results were also normal. Computed tomography (CT) of the head revealed symmetrical extensive hypoabsorption areas in the brain parenchyma. Head MRI showed marked diffusion abnormalities in the bilateral entorhinal to cerebral peduncles on diffusion-weighted imaging (DWI) . In addition, the extensive cerebral high-intensity lesion was observed on fluid-attenuated inversion recovery (FLAIR) imaging . A mildly elevated cerebrospinal fluid (CSF) cell count of 70/mm 3 , with a protein level of 52 mg/dL and glucose level of 56 mg/dL, was observed, suggesting encephalitis, neurosyphilis, drug-induced encephalopathy, CNS lupus, and malignant lymphoma. Electroencephalography mostly revealed generalized 1–2 Hz high-amplitude delta waves, with no apparent epileptic waves. On the third day of hospitalization, a May–Giemsa stain was performed on the peripheral blood, revealing the presence of flower cells . Subsequently, a cytopathological examination of CSF also revealed flower cells . Flow cytometric analysis of CSF showed a predominance of T cells. The immunophenotypic profile indicated that 82.6% of the cells were CD2 + , 76.3% were CD3 + , 73.5% were CD5 + , and 74.7% were CD7 + . The analysis also revealed the presence of both CD4 and CD8 T cells, with 42.4% and 37.2% positivity, respectively, resulting in a CD4/CD8 ratio of 1.1. These findings align with the expected T cell predominance in cases of adult T cell leukemia/lymphoma. The patient was also positive for HTLV-1 antibody; therefore, we suspected HTLV-1-related primary CNS lymphoma. The route of HTLV-1 infection remained unknown as the patient’s mother had no history of testing, there were no records of blood transfusions, and both the patient’s sexual history and history of substance abuse were inaccessible due to communication barriers. In this patient, the human immunodeficiency virus antibody test was negative. In random skin biopsies, no clustering of atypical lymphocytes within blood vessels was observed. In bone marrow aspiration and trephine biopsy, normocellular marrow was noted, with approximately 10% of small lymphocytes being CD3 + /CD20 − , although atypia was limited. Fig. 1 Head magnetic resonance imaging on admission day. a With diffusion-weighted imaging. b With fluid-attenuated inversion recovery imaging Fig. 2 Flower cells (arrow) from the patient’s peripheral blood using a 100× objective lens Fig. 3 Flower cells (arrow) from the patient’s cerebrospinal fluid using a 100× objective lens | 4.28125 | 0.889648 | sec[1]/p[2] | en | 0.999997 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
The clinical diagnosis of HTLV-1-associated primary CNS lymphoma was established based on high-level positivity for HTLV-1 antibody , positive spinal fluid cytology (flower cells), and absence of any apparent primary disease on bone marrow and skin biopsies and systemic contrast-enhanced CT . After consulting with a hematologist, we decided not to perform a brain biopsy or initiate aggressive treatment, including chemotherapy, as irreversible damage to the brainstem was suspected , and the neurological prognosis was deemed poor. The patient received palliative management while undergoing hydration via peripheral intravenous fluids. Although his consciousness was not restored, with the intervention of a palliative care team, including grief support for the family, he remained in a peaceful state accompanied by his family. Six weeks after admission to our hospital, the patient peacefully passed away. The pathological autopsy showed diffuse infiltration of T cell atypical lymphocyte-like cells into the brain tissue , and immunohistochemical analysis revealed that the lymphocytes were positive for CD3 . Evaluation of other organs, including the liver, spleen, lungs, and bone marrow, revealed no histological evidence of infiltration. Based on these findings, we made a definite diagnosis of HTLV-1-related primary CNS lymphoma. Fig. 4 Atypical T cell lymphocyte-like cells identified entirely in the patient’s brain tissue from the pathological autopsy using a 400× objective lens. a Hematoxylin and eosin stain demonstrating diffuse infiltration of atypical lymphocyte-like cells. b Immunohistochemical staining for CD3 in brain tissue indicating positive staining in lymphocyte-like cells | 4.074219 | 0.964844 | sec[1]/p[3] | en | 0.999996 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
We presented the case of a patient with relatively common LC along with depressive symptoms; however, the diagnosis was delayed due to anchoring bias and the patient’s work history before affliction. In general, primary CNS lymphoma due to adult T cell leukemia/lymphoma often presents as a mass ; however, LC, which is also a primary CNS lymphoma, does not present as a mass . Currently, there is no established definition for diagnosing LC. However, studies have adhered to the description of gliomatosis cerebri in the criteria established by the 2007 World Health Organization classification of tumors of the CNS. These criteria include: (1) T2-weighted image/FLAIR abnormal hyperintensity present in at least three cerebral lobes or three anatomical regions of the CNS and (2) non-enhancing lesions or lesions without nodular or mass enhancement visible on initial MRI scans . In addition, excluding the presence of concomitant systemic lymphoma or intravascular lymphoma is crucial. In the present case, the diagnosis of LC was confirmed using imaging and pathological findings. | 4.101563 | 0.934082 | sec[2]/p[0] | en | 0.999998 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
The diagnosis of LC was not made until approximately 3 months after symptom onset when the patient gradually developed impaired consciousness and gait disturbance and developed confusion. The importance of investigating organic causes in rapidly progressing psychiatric symptoms and dementia is often emphasized . Therefore, it must be said that there was a diagnostic delay in this case. However, even in a 2016 systematic review, the median time from onset to diagnosis of LC is 4.5 months . Generally, LC may be difficult to diagnose. This period may be shortened if LC is also added to the differential disease list at the onset of depressive symptoms and rapid progression of impaired consciousness and gait. | 3.759766 | 0.748047 | sec[2]/p[3] | en | 0.999997 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
Furthermore, confusion developed rapidly, but the treatment for catatonia with generalized muscle tension was continued, which delayed diagnosis. According to the Diagnostic and Statistical Manual of Mental Disorders, Fifth Edition, diagnostic criteria for catatonia, three or more of the following symptoms are required: stupor, waxy flexibility, catalepsy, mutism, posturing, negativism, stereotypes, mannerisms, grimacing, agitation, echopraxia, and echolalia . The patient met these criteria. Additionally, patients with catatonia and acute autonomic instability often require early therapeutic intervention with benzodiazepines or other drugs due to the increased risk of complications and death . Therefore, prompt therapeutic intervention was necessary as the condition rapidly deteriorated, and a poor response to the administered treatment should have prompted the consideration of alternative diagnoses. However, the patient’s history of depressive symptoms following work-related changes likely anchored the diagnosis , making the consideration of alternative diagnoses difficult. | 4.121094 | 0.879395 | sec[2]/p[4] | en | 0.999997 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
Additionally, upon admission, the case exhibited highly distinctive vital signs, as both systolic and diastolic blood pressure levels were elevated. Previous observational studies have revealed a consistent correlation between concurrent diastolic and systolic blood pressure levels and the presence of white matter lesions . While a causal relationship between these lesions and elevated blood pressure remains uncertain, it is plausible that they might underlie hypertension in this particular case, given the absence of other contributing factors. | 3.939453 | 0.764648 | sec[2]/p[6] | en | 0.999998 | 39080675 | https://doi.org/10.1186/s13256-024-04666-1 |
We present a complex case of a 79-year-old man who presented with severe hypercalcaemia arising from a parathyroid mass that produced excessively high levels of parathyroid hormone. This, along with the size of the mass, and the severity of his hypercalcaemia, raised the possibility of parathyroid carcinoma, a rare and notoriously difficult diagnosis to make on histological grounds . Two days following the administration of pamidronate disodium for his hypercalcaemia, he developed acute onset periorbital and orbital cellulitis with loss of visual acuity and ocular motility that responded well to systemic corticosteroids. | 3.929688 | 0.981934 | sec[0]/p[5] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
A 79-year-old man attended the geriatric rapid access clinic with a vague sense of ill health, reduced ambulation, and cognitive decline. His medical history was significant for a right inferior parathyroid adenoma excision for primary hyperparathyroidism in 2011, type 2 diabetes mellitus, and chronic kidney disease stage 3A (baseline creatinine of 160 umol/L). Unfortunately, he was lost to follow-up at a previous institution but was noted in retrospect to have mild persistent hypercalcaemia from 2014 to 2021 on routine blood testing, prior to the above presentation in the latter half of 2023. | 3.380859 | 0.98877 | sec[1]/p[0] | en | 0.999995 | 39087171 | https://doi.org/10.7759/cureus.63577 |
Systematic questioning and examination elicited no specific features. Screening blood tests revealed a corrected calcium level of 3.48 mmol/L, a plasma parathyroid hormone (PTH) level of 1,099 ng/L, a serum creatinine of 222 umol/L, and an estimated glomerular filtration (eGFR) of 23 mL/min (Table 1 ). Vitamin D, urinary calcium, thyroid function, urine metanephrines, and vasculitis screen were all unremarkable. | 3.808594 | 0.683105 | sec[1]/p[1] | en | 0.999998 | 39087171 | https://doi.org/10.7759/cureus.63577 |
Following an ear, nose, and throat doctor (ENT) consult, parathyroid gland ultrasonography showed a 3 cm x 1.9 cm x 3.4 cm mass adjacent to the lower pole of his left thyroid gland . It was heterogeneous and displayed increased vascularity. Its size and vascularity, along with the severity of his hypercalcaemia and hyperparathyroidism, raised concerns regarding parathyroid carcinoma. | 3.515625 | 0.967773 | sec[1]/p[3] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
Subsequent nuclear medicine parathyroid technetium 99m sestamibi (Tc-MIBI) scintigraphy and single photon emission computed tomography (SPECT) scans showed persistent focal abnormal increased uptake within the parathyroid mass . His hypercalcaemia persisted despite an increase in cinacalcet dose to 90 mg four times daily. He then received a slow infusion of intravenous bisphosphonate at a reduced dose (pamidronate disodium 30 mg) in light of his renal function. This normalised his calcium levels within 72 hours. | 3.916016 | 0.963867 | sec[1]/p[4] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
Additionally, 48 hours following his intravenous bisphosphonate, he developed right orbital pain with upper and lower lid oedema . This progressed over five days despite intravenous flucloxacillin and topical 1% chloramphenicol ointment. His symptoms then acutely worsened with severe pain, chemosis, reduced visual acuity (6/24 from a baseline of 6/12), and ocular motion restriction. His colour vision and intraocular pressure were normal, with no relative afferent pupillary deficits and retina or corneal abnormalities. He had no associated neurological or rhinological symptoms, with unremarkable nasal endoscopy. His antibiotics were escalated to intravenous ceftriaxone and metronidazole although he remained apyrexic. | 3.666016 | 0.983887 | sec[1]/p[5] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
His total white cell count was within a normal range (5.30x10 9 /L) with moderate elevation in C-reactive protein (72 mg/L). A contrast-enhanced CT scan of his orbits showed evidence of orbital and periorbital cellulitis, with associated proptosis and inflammation of his extraocular muscles . | 3.603516 | 0.977051 | sec[1]/p[6] | en | 0.999998 | 39087171 | https://doi.org/10.7759/cureus.63577 |
Histology of the parathyroid mass showed a well-circumscribed lobulated tumour composed predominantly of chief cells, with a partial surrounding pseudocapsule . It showed no evidence of thyroid, blood vessel, and lymphatic or perineural invasion. Overall appearances were consistent with parathyroid adenoma . Histology of the level six lesion showed a cyst lined by parathyroid chief cells, consistent in the right clinical context with a cystic parathyroid adenoma. | 4.011719 | 0.796387 | sec[1]/p[9] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
This complex and uncommon case has a number of widely generalisable learning points. Firstly, it underpins the importance of multidisciplinary work. During this patient's inpatient stay, he had input from geriatrics, acute medicine, nephrology, endocrinology, ENT, radiology, ophthalmology, and pathology for a number of significant concurrent issues. These included severe hypercalcaemia with acute kidney injury, a grossly elevated PTH level from a large parathyroid mass, acute orbital inflammation, and postoperative hungry bone syndrome. | 3.138672 | 0.974121 | sec[2]/p[0] | en | 0.999996 | 39087171 | https://doi.org/10.7759/cureus.63577 |
In our case, the aforementioned biochemical abnormalities prompted oncological surgery for his parathyroid mass. The identification of parathyroid tissue in level six in this patient does raise the possibility of multi-centric parathyroid cancer . Recurrence is usually heralded by a gradual increase in calcium and PTH levels, eventually leading to a hypercalcaemic crisis, hence the requirement for long-term follow-up . | 3.585938 | 0.933105 | sec[2]/p[11] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
This case highlights the importance of coordinated multidisciplinary care in the approach to medical complexity. We have described the successful treatment of a rare potentially severe complication of BP treatment, highlighting the importance of prompt ophthalmological review in patients with BP complaining of new ocular symptoms. | 3.220703 | 0.972168 | sec[3]/p[0] | en | 0.999997 | 39087171 | https://doi.org/10.7759/cureus.63577 |
We report a case of a 44-year-old woman, 69 kg for 1.76 m height, with a NET located in the terminal ileum and liver metastases who presented a CaS with a left sided CaHD. She was admitted at the hospital with class IV New York Heart association (NYHA) dyspnea and orthopnea due to severe mitral and aortic valve insufficiency. The patient also complained of non-specific abdominal pain. No flushing, tachycardia or diarrhea was reported. Upon physical examination, a grade 3/6 systolic and 3/6 diastolic murmur was identified. Electrocardiography showed sinus rhythm with left atrial and ventricular hypertrophy. Transthoracic echocardiography (TTE) and trans-esophageal echocardiography (TEE) showed left ventricle dilation with preserved global systolic function, a severe central jets of mitral regurgitation, a severe aortic incompetence (Image 1.) and a severe pulmonary hypertension (PH) with a moderate tricuspid insufficiency and a systolic PAP at 69 mmHg. The pulmonary function tests were normal. Upon admission, the blood results showed a NT-pro BNP level increased at 1549 pg/mL. The other laboratory parameters did not show any abnormalities. No degradation product of serotonin were measured at that time. The diagnosis was made following the positron emission tomography (PET-CT) and colonoscopy carried out in the presence of nonspecific symptomatology. Abdominal computer tomography confirmed a terminal ileum neoplasm, identified as the primary tumor with limited liver metastatic disease. Suspicion of CaS with left-sided CaHD secondary to NET of terminal ileum was raised and the patient was medicated with somatostatin (120 mg 1x/4 weeks) to provide relief of symptoms. Moreover, upon admission, and given the cardiac symptomatology, a treatment by inhibitor of ACE and diuretics was initiated. In this context surgery played a vital role in the treatment of the CaS, hence, the case was discussed in a multidisciplinary team. The indication for valvular surgery was discussed but given of the early oncological status of the primary lesion this strategy was not retained. | 3.996094 | 0.97998 | sec[1]/p[0] | en | 0.999996 | PMC11290185 | https://doi.org/10.1186/s12871-024-02648-w |
Terminal ileum resection by laparoscopy was declined for the safety reasons. Therefore, an open abdominal surgery was approved and scheduled. The surgery was conducted with the patient under general anesthesia (GA). The focus prior to surgery was to avoid carcinoid trigger such as emotional stress and anxiety. In this regard the patient received premedication with Alprazolam 0.5 mg. Standard anesthesia monitoring such as electrocardiogram, noninvasive blood pressure, pulse oximetry and end-tidal CO2 was used. Moreover, anesthesia depth monitoring (NeuroWave®) and cerebral oximetry (INVOS®) were carried out. Before induction a large intravenous catheter was placed, and the radial artery was cannulated. A balanced anesthetic technique was performed with 2 mg of midazolam, 10 mcg of sufentanil, 12 mg of etomidate and 80 mg of 2% lidocaine. To facilitate intubation, 50 mg of rocuronium for neuromuscular blocking was use. To avoid a carcinoid crisis in response to intubation a 50 mcg loading bolus of somatostatin was administered at induction followed by a continuous infusion rate at 100 mcg/h. The intubation was performed with a video laryngoscope (AirTrack®) to avoid excessive stimulation. The maintenance of the anesthesia was done with sevoflurane (End-Tidal Sevoflurane (EtSevo) 1.2–1.5). After induction, a central venous line was placed in the internal jugular vein for volume resuscitation or inotropic support. Our protocol agreed that in the event of hypotension, flushing or bronchospasm, an additional bolus of 50 mcg of somatostatin should be administered; after eliminating all other causes, bolus can be repeated 1x/5 min. With the possibility of increased the continuous infusion rate to maximum 300 mcg/h. Treatment of moderate hypotension would be done with phenylephrine or noradrenaline. A dobutamine support was ready to use in the event of refractory collapse. We also took care to exclude all histamine liberating drugs as morphine or atracurium. With regards to the perioperative and postoperative pain management, an ultrasound-guided transversus abdominal plan block with 0.25% levobupivacaine at the dose of 1 mg/kg was placed prior to surgical incision. The total duration of the surgery was 180 min and was well tolerated by the patient. She required two boluses of additional 50 mcg somatostatin each to treat hypotension due to tumor manipulation and 3 boluses of 50 mcg phenylephrine for moderate hypotension. Hemodynamics, heart rate and electrocardiography were continuously registered and remained stable. No intraoperative flushing, oedema or bronchospasm was noticed. At the end of the surgery the patient was awakened, extubated, and transferred to intensive care unit for surveillance. The somatostatin infusion was continued up to 12 h after the surgery. A piritramide pump provided additional postoperative analgesia. The patient did not experience any postoperative complications. She left the intensive care unit 2 days later. Fig. 1 Algorithm for management of carcinoid syndrome and carcinoid heart disease | 3.962891 | 0.920898 | sec[1]/p[1] | en | 0.999996 | PMC11290185 | https://doi.org/10.1186/s12871-024-02648-w |
Table 1 Clinical vignettes Scenario Reason for consultation Anamnese Interactive learning goals 1 Tooth extraction with risk factors An elderly person comes because their dentist wants 4 teeth extracted. Due to the preexisting conditions, the dentist or doctor will want this to be done in a clinic. The patient cannot say exactly why he/she should be treated in a clinic now and he/she does not know exactly his/her medication: “I trust my family doctor”. By taking a specific anamnesis, the risk factors for tooth extraction must now be determined and then a plan for the extraction must be carried out. On their own, the person hardly tells us anything about your preexisting conditions. - Building a sustainable doctor-patient relationship - Establishing a conversation with the patient so that relevant information can be obtained - The patient should understand why their previous illnesses are relevant 2 Sialolithiasis A patient comes to the outpatient clinic because he has been suffering from pain on the right side below the lower jaw for approximately 2 days and has also noticed an ever-increasing swelling there since this morning. He had already been to his dentist, but he could not find any cause of the swelling on the teeth. The swelling hurts significantly to the touch (and is located on the right below the mandibular bone in the soft tissue). When eating, the pain gets worse. She is concerned about the swelling, but she can swallow and breathe normally. - Building a sustainable doctor-patient relationship - Reassure the patient as she is very worried - Patients should feel that they are in good hands 3 Eagle-syndrom A patient has been suffering from pain in the neck area on the right side for about two years. He/she has already been to the dentist, family doctor, ENT doctor, neurologist and orthopedist several times, but they were unable to help. The patient is very desperate in view of the frequent visits to the doctor, which have not been able to provide an explanation for the symptoms and have not helped so far, and is now hoping to find out a cause and solution to the existing symptoms. - Building a sustainable doctor-patient relationship - Calming the unsettled patient - Patients should feel well received 4 Augmentation and implantation The patient comes to the hospital because the dentist has issued a referral for bone augmentation and subsequent implant placement. He/she has no remaining teeth in her lower or upper jaw. The dentures hold well, but there are difficulties when eating. The patient has a number of risk factors for implant treatment: smokes around 20–30 cigarettes a day, type 2 diabetes with an HbA1c of 12 and poor oral hygiene and prosthesis care. - Building a sustainable doctor-patient relationship - Calming an upset patient - The patient should leave the conversation feeling that the decision made is the best for them | 3.439453 | 0.928711 | sec[1]/sec[0]/p[2] | en | 0.999997 | 39080578 | https://doi.org/10.1186/s12909-024-05818-z |
Norplant is a type of subdermal contraceptive that uses a levonorgestrel-releasing, silastic subdermal implant to prevent pregnancy; subdermal contraceptive implants fall under the category of long-acting reversible contraceptives. They offer high effectiveness and low failure rates . Norplant was first developed by the Population Council's International Committee for Contraceptive Research in 1966 and approved for use in the United States by the Food and Drug Administration in 1990 . Each capsule contains 36 mg of dry crystalline levonorgestrel, for a total of 216 mg in six individual capsules . This progestin diffuses into the surrounding tissues, where it is absorbed by the circulatory system on a continuous basis and provides a contraceptive effect for up to five years. The silastic rods measure 4 cm x 0.22 cm. The recommended placement location is subdermal in the medial aspect of the nondominant upper arm . Due to issues with removal, Norplant was removed from the US market in 2002. The last insertions occurred in 2004 . Removal of the implants should be completed upon patient request, for medical indications, or at the end of five years by clinicians educated on the proper removal technique . Clinicians today may encounter patients with retained Norplant devices requesting removal. When removing such devices, careful attention should be paid to the initial placement location of the implant(s), time elapsed since implantation, and removal technique. Here we present a case that brings to light the complications of prolonged subcutaneous implants and a successful method of surgical extraction. | 4.234375 | 0.837402 | sec[0]/p[0] | en | 0.999999 | 39087147 | https://doi.org/10.7759/cureus.63585 |
A 51-year-old female presented to a gynecology practice for a Norplant removal 25 years after placement. She had lost a significant amount of weight during this time. She reported that the rods were palpable and uncomfortable. She had previously been unable to find a trained provider willing to remove the implants. Upon initial inspection, only two rods were visible , but all six were palpable . The risks and benefits of removal were discussed, and the patient provided informed consent to proceed with office removal. Labs were not drawn prior to implant removal. Lidocaine 1% (2 mL) was injected at the confluence , and lidocaine 1% (.5 mL) was injected under each rod in a tumescent manner to displace the implants superficially and numb the surrounding area; no further medications were administered during or after the procedure. A 4 mm incision was made with a #11 scalpel. Using pressure at the proximal end of the implant , the distal tip, encased in a fibrous sheath, was pushed toward the incision. The fibrous sheath was incised until the implant became visible . The implant was grasped and removed with forceps. All six 4 cm levonorgestrel implants were removed intact in this manner through the same incision . The incision was closed with one subcuticular stitch and a pressure dressing. There were no complications during or after the procedure. | 3.785156 | 0.979004 | sec[1]/p[0] | en | 0.999997 | 39087147 | https://doi.org/10.7759/cureus.63585 |
This case demonstrates an emerging complication of subdermal contraceptive implants associated with prolonged implantation. Norplant implants were removed from the US market in 2002; thus, current patients with these implants will likely face similar complications of removal. The most effective removal technique is also the safest and requires careful medical history taking and a physical exam, possibly with the addition of advanced imaging and a subspecialty referral. Our report shows the importance of recognizing the complications of prolonged Norplant implantation and the best technique for removal. Future research should systematically assess techniques for Norplant removal. | 3.814453 | 0.953613 | sec[3]/p[0] | en | 0.999996 | 39087147 | https://doi.org/10.7759/cureus.63585 |
A surgical aortic valve replacement (SAVR) considered successful by surgeons may be immediately offset by postoperative complications. Acute coronary obstruction after SAVR is rare but potentially fatal . Here, we present a rare life-threatening case of sudden obstruction of the right coronary artery (RCA) ostium six days after SAVR and describe its investigation and immediate management. Specifically, the patient in our case developed fatal ST-elevation myocardial infarction (STEMI) at a time when a patient is normally considered for discharge. Therefore, we discuss here the cause and treatment of this case. | 3.925781 | 0.960449 | sec[0]/p[0] | en | 0.999998 | 39087149 | https://doi.org/10.7759/cureus.63612 |
An 84-year-old woman was referred from a previous hospital with dyspnea and chest pain that has persisted for the past three days. Two months prior, an exercise stress electrocardiogram (ECG) performed at the hospital indicated ST-segment depression in the anterior thoracic lead. She had undergone percutaneous coronary intervention for proximal RCA (#1-#2) stenosis 13 years prior; however, all other past medical history was unremarkable. | 3.529297 | 0.987305 | sec[1]/p[0] | en | 0.999998 | 39087149 | https://doi.org/10.7759/cureus.63612 |
On admission, she was 155 cm tall and weighed 51 kg. The body surface area was 1.46 m 2 . Laboratory test results revealed no cardiac enzyme leakages. ECG showed a normal sinus rhythm and no significant ST changes . Transthoracic echocardiography revealed severe aortic valve stenosis (AS) (aortic valve area: 1.06 cm 2 , mean pressure gradient (PG): 53 mmHg) with a normal left ventricular ejection fraction (62%). No other valvular diseases were noted. Transesophageal echocardiography (TEE) revealed severe AS with an annular dimension of 20 mm, a sinotubular junction of 22 mm, and a sinus of Valsalva of 27 mm. Chest computed tomography (CT) showed significant calcification from the aortic root to the ascending aorta . The distance between the RCA ostium and the annulus was 1.06 cm . Coronary angiography (CAG) showed no in-stent restenosis in the RCA and no other significant stenosis or collateral vessel development. After discussion with the heart team and providing a detailed explanation to the patient, SAVR was selected instead of transcatheter aortic valve replacement because of the high possibility of valve annular rupture due to severe calcification. | 3.958984 | 0.973145 | sec[1]/p[1] | en | 0.999996 | 39087149 | https://doi.org/10.7759/cureus.63612 |
Surgery was performed via a median sternotomy. Cardiopulmonary bypass (CPB) was established between the ascending aorta and superior and inferior vena cava. Cardiac arrest was obtained by anterograde through the aortic root and retrograde cardioplegia. Aortotomy was performed approximately 3 cm above the origin of the RCA to avoid calcification. On observation, the aortic valve was tricuspid, with severe calcification from the cusps to the annulus and sinus of Valsalva. The calcifications were removed using a Cavitron Ultrasonic Surgical Aspirator (Cavitron, USA). After confirming the absence of Prosthesis-patient mismatch (effective orifice area index: 1.08 cm 2 /m 2 ), we implanted a 19-mm aortic bioprosthetic valve (INSPIRIS RESILIA; Edwards Lifesciences LLC, Irvine, USA). Although the patency of the RCA ostium was confirmed , we performed retrograde coronary perfusion. We determined that the return of the perfusion was insufficient; however, we decided to perform aortic de-clamping because we thought that bypassing RCA would be easier. As a result, she was easily weaned from the CPB, and TEE showed normal right ventricular function; therefore, the operation was terminated without CABG. | 3.96875 | 0.944336 | sec[1]/p[2] | en | 0.999998 | 39087149 | https://doi.org/10.7759/cureus.63612 |
The patient’s postoperative course was uneventful. She was extubated the day after surgery, and rehabilitation was initiated. However, on postoperative day (POD) 6, she complained of sudden chest pain and difficulty breathing at midnight. The ECG showed significant ST-segment elevation in the inferior wall, leading to bradycardia . Coronary CT angiography revealed that the RCA ostium was positioned to overlap the stent post of the bioprosthetic valve and the RCA was not contrasted . An emergency CAG was performed, but the catheter could not be engaged into the RCA ostium, and the RCA was not contrasted by aortography . Blood test results showed elevated cardiac enzyme levels and a prolonged preoperative prothrombin time/international normalized ratio of 2.32. Immediately after that, the patient experienced cardiogenic shock; therefore, percutaneous cardiopulmonary support and an intra-aortic balloon pump were inserted, and on-pump beating coronary artery bypass grafting (CABG) was performed emergently to the RCA (#3) using a saphenous vein graft (SVG). | 3.806641 | 0.980469 | sec[1]/p[3] | en | 0.999995 | 39087149 | https://doi.org/10.7759/cureus.63612 |
Postoperative cardiac function improved, and weaning from mechanical support devices was uneventful. There was no increase in the PG across the prosthetic valve (mean PG: 8.3 mmHg). Contrast-enhanced CT confirmed that the SVG was patent . However, weaning from the ventilator was difficult and tracheotomy was required. The patient also developed mediastinitis; therefore, an omental flap transposition was performed on POD 55. Finally, the patient was transferred to another hospital on POD 88 with difficulty weaning from the ventilator. At the time of this report, she is still undergoing rehabilitation at the hospital. | 3.244141 | 0.986816 | sec[1]/p[4] | en | 0.999995 | 39087149 | https://doi.org/10.7759/cureus.63612 |
Coronary ostial stenosis is a life-threatening postoperative complication of SVAR, with a reported complication rate of 1-5% in patients undergoing SAVR . Onset has been reported intraoperatively and within 3-6 months postoperatively . Shin et al. reported successful bare-metal stent placement for coronary ostial stenosis 22 months after SAVR . We believe that the present case is worth reporting because the complication occurred on POD 6. Particularly, the patient developed fatal STEMI at a time when she would normally be considered for discharge. Thus, the cause and treatment need to be thoroughly discussed. | 3.841797 | 0.821289 | sec[2]/p[0] | en | 0.999997 | 39087149 | https://doi.org/10.7759/cureus.63612 |
The possible mechanisms of intraoperative coronary ostial stenosis include improper positioning of the prosthetic valve, inappropriate aortotomy suture, calcific embolus, and spasm of the coronary arteries . Mechanical injury or direct trauma to vessels is usually caused by the use of a coronary perfusion cannula, which results in immediate dissection and myocardial infarction . There are reports of late development due to thromboembolism, fibrosis, and intimal thickening of the aortic root caused by turbulent flow around the prosthetic valve, as well as an immune response to the prosthetic valve . In the present case, the RCA had no collateral vessels, suggesting that blood flow from the coronary artery ostium was preserved in the early postoperative period. However, on the sixth POD, the patient suddenly developed coronary ostial obstruction. One possible cause of coronary artery ostial occlusion in the early postoperative period, especially in patients with highly calcified aorta as in this case, could be embolization of plaque. Another possible cause is a postoperative change in the morphology of the sinus of Valsalva, which may have led to occlusion of the coronary artery ostium. In any case, we believe that the proximity of the stent post of the prosthetic valve to the coronary artery ostium was one of the major risk factors. We believe that the position of the prosthetic valve should have been reviewed and root enlargement should have been considered. We anticipated the possibility of intraoperative RCA ostium occlusion and our policy was to perform CABG with SVG if an infarct was identified in the RCA region intraoperatively. However, the patient developed STEMI at an unexpected time. Based on this experience, we believe that patients with a narrow aortic annulus, a short distance from the annulus to the coronary artery ostium, or severe calcification, who are expected to have a high likelihood of coronary artery obstruction after SAVR, should undergo aggressive valve root enlargement with close attention to proper prosthetic valve positioning. An insufficient return of retrograde coronary perfusion, as in this case, may be an indicator of future coronary occlusion. However, it is difficult to prevent coronary ostium occlusion completely, and a sufficient follow-up period after SAVR is necessary to respond to fatal complications as soon as possible. If they do occur, prompt revascularization is required. | 4.199219 | 0.716797 | sec[2]/p[1] | en | 0.999996 | 39087149 | https://doi.org/10.7759/cureus.63612 |
We experienced a case of fatal STEMI six days after SAVR surgery that was resolved by emergent CABG. Coronary artery ostial stenosis after SAVR is a complication that can occur from the early to the late postoperative period. In this narrow annulus case, the bioprosthetic valve interfered with the coronary ostium and tended to embolize, which was thought to be a contributing factor to the coronary ostia occlusion. The occurrence of coronary artery ostia occlusion after SAVR should be prevented and monitored, and if it occurs, it should be revascularized as soon as possible. | 3.925781 | 0.960449 | sec[3]/p[0] | en | 0.999997 | 39087149 | https://doi.org/10.7759/cureus.63612 |
Ch“1” has a clear, complete fracture at approximately 67% of the original dorsoventral length of the bone. The original length of the bone was approximately 17.7 cm. The distal end is displaced to the left dorsolateral aspect of the bone and tilted 25° cranially. The two halves healed together in this position and a callus formed around the overlapping area, and slightly beyond that, both proximally and distally. | 3.515625 | 0.916016 | sec[3]/sec[2]/p[1] | en | 0.999996 | 39083447 | https://doi.org/10.1371/journal.pone.0306819 |
In Ch“2” the complete fracture is not as clear as in Ch“1”. This is most likely due to overpreparation, while not realizing the presence of the pathology and how it diverges from the normal anatomy. The fracture is positioned at approximately 66% of the dorsoventral length of the entire bone. The original length of the bone was approximately 17.6 cm. Similar to Ch“1”, the distal end is displaced to the left dorsolateral aspect of the bone and is tilted 25° cranially at the distal end. | 3.490234 | 0.519531 | sec[3]/sec[2]/p[2] | en | 0.999995 | 39083447 | https://doi.org/10.1371/journal.pone.0306819 |
A 37-year-old female patient came to our outpatient department of oral medicine and radiology for a routine dental checkup. A general examination revealed her vitals were stable. There was no history of harmful chewing or smoking tobacco. During the extraoral examination, a port-wine-colored macular patch, characterized by its irregular shape and margins, was only visible on the right side of the face. The macular patch extends superiorly from the right temple, close to the level of the right eyebrow, and extends inferiorly to the vermilion border on the right side of the upper lip. The macular patch extends anteriorly from the right lateral ala of the nose and extends posteriorly 2.5 cm from the tragus of the right ear . | 3.328125 | 0.987305 | sec[1]/p[1] | en | 0.999999 | 39086792 | https://doi.org/10.7759/cureus.63532 |
The purple-colored macular patches were consistent with the trigeminal nerve distribution. They were only on the right side of the face, following the first, namely the ophthalmic division of the trigeminal nerve (V1), and the second, the maxillary division of the trigeminal nerve (V2) branches of the dermatome supplied by the right trigeminal nerve. The patient had calculus in her mandibular anterior tooth region due to inadequate dental care. The positive diascopy test provisionally diagnosed a PWS involving the facial skin and gingiva. The differential diagnosis for this case includes pyogenic granuloma, which clinically occurs as a reactive growth in response to localized supra and sub-gingival calculus and bleeds on slight provocation. We performed an oral prophylaxis and referred the patient to a dermatologist for the management of PWSs on her face. | 3.707031 | 0.973633 | sec[1]/p[6] | en | 0.999997 | 39086792 | https://doi.org/10.7759/cureus.63532 |
The general differential diagnosis for PWSs or nevus flammeus includes rare congenital syndromes involving capillary abnormalities and pigmented lesions, with or without systemic symptoms, called phacomatosis pigmentovascularis. It was classified into four phenotypes (I: Nevus flammeus and nevus verrucous or pigmentosus; II: flammeus +/- anemic and Mongolian spots; III: flammeus +/- anemic and spilus; IV: flammeus +/- anemic and spilus with Mongolian spot) and subtypes. Happle recharacterized it as phacomatosis, cesioflamea, cesiomarmorata, spilorosea, and unclassified without or with systemic symptoms . Thus, a diagnosis of intraoral hemangioma with a PWS on the right side of the face was made. We referred her to consult a dermatologist for further treatment of the PWS. Informed consent was obtained in both the above-mentioned cases. | 3.904297 | 0.793457 | sec[1]/p[11] | en | 0.999997 | 39086792 | https://doi.org/10.7759/cureus.63532 |
Despite GP being well-documented in literature, the linkage of symptoms in this patient’s case with superior mesenteric artery (SMA) syndrome, pseudocyst development, and renal vein entrapment syndrome, also known as “nutcracker syndrome,” is significant. The interplay of these conditions appears to be correlated, offering a rare but valuable contribution to existing research and enriching international literature. To note, the incidence of pseudocysts in chronic pancreatitis is around 20%–40% . Specifically, there appear to be no reported cases describing these four conditions simultaneously. | 3.789063 | 0.567383 | sec[0]/p[1] | en | 0.999995 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
A 38-year-old male with gastroesophageal reflux disease, alcoholic pancreatitis, and a known duodenal fluid collection/pseudocyst (3.4 × 3.3 × 2.6 cm shown on computed tomography (CT) 5 days prior) presented with increasing mid-abdominal pain, nausea, and diarrhea after consuming alcohol and an unknown 30-pound weight loss over the prior 2–3-month period. The patient was admitted to the medicine floor for medical and surgical management. | 3.195313 | 0.989746 | sec[1]/p[0] | en | 0.999998 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
The CT abdomen on admission (day 1) showed that the fluid collection had enlarged from the previous CT, measuring 4.7 × 4.0 × 3.3 cm, and appeared to be within the wall of the duodenum and common bile duct while narrowing the descending duodenum. There was distension of the stomach and duodenum to the level of the SMA, indicative of SMA syndrome as seen in Figure 1A . CT incidentally revealed a coexisting nutcracker phenomenon, the extrinsic compression of the left renal vein by the SMA anteriorly . | 3.740234 | 0.96582 | sec[1]/p[1] | en | 0.999998 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
On day 7, gastroenterologist performed an esophagogastroduodenoscopy, which showed an obstructing bulge on the duodenal wall, and an endoscopic ultrasound (EUS) was performed for aspiration of a large periduodenal collection. The nasogastric tube in place was passed beyond the previously obstructed point and anchored. A repeat CT abdomen was scheduled in 4 weeks with possible nasogastric tube removal. However, the nasogastric tube was removed 3 days following discharge due to patient intolerance. | 3.03125 | 0.985352 | sec[1]/p[4] | en | 0.999997 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
The patient then returned to the Emergency Department 1 month following discharge with return of symptoms and similar sized fluid collection in the right upper abdomen measuring up to 4.5 cm. Repeat EUS was notable for small fluid collection (2.8 × 0.8 cm) around the second portion of the duodenum, which was too small for intervention. The patient has since had four additional Emergency Department visits for acute or chronic pancreatitis secondary to alcohol consumption. | 3.189453 | 0.990234 | sec[1]/p[6] | en | 0.999996 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
This atypical patient is unlike those commonly described in literature with GP. The patient was below the normal age range for those with GP and had significant complications including an encircling pseudocyst with severe gastric outlet obstruction. Loss of mesenteric fat pad with BMI of 17 kg/m 2 likely contributed to his SMA syndrome and nutcracker syndrome. Though it appears that these additional findings interplay and relate to his GP rather than are incidental, independent disease processes, a better understanding of the relationship between these disease etiologies allows for evidence-based treatment. | 3.625 | 0.970703 | sec[2]/p[0] | en | 0.999998 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
This patient with GP and SMA syndrome, pathologies not previously reported together in literature, showcases a poorly understood condition while stressing GP as a challenging diagnosis for patients with nonspecific symptoms while highlighting the challenges in pain management. Additionally, the simultaneous occurrence of SMA syndrome and nutcracker syndrome is rare as their coexistence has only been reported in a few cases, but these syndromes should be evaluated for by radiologists as potentially shared pathophysiology impacts patient management . | 3.794922 | 0.959961 | sec[2]/p[3] | en | 0.999998 | 39086504 | https://doi.org/10.1093/gastro/goae079 |
We present a case series of children and adolescents treated with dalbavancin for non-ABSSSI infections caused by Gram-positive pathogens. | 3.199219 | 0.870117 | sec[0]/p[2] | en | 0.999997 | 38958260 | https://doi.org/10.1093/jac/dkae212 |
The four patients with CLABSI received a single complete dose of dalbavancin (patient #6 received 15 mg/kg on day 1 and 7.5 mg/kg on day 8; while patients #1, #11 and #13 received a unique administration of 22.5 mg/kg). One adolescent patient (#7) received 1000 mg as an initial dose, followed by 15 500 mg doses on a weekly basis. Patient #10 received just one dose of 22.5 mg/kg, after which the treatment was discontinued due to a possible adverse effect. The remaining nine patients received two initial equal doses on days 1 and 8, followed by maintenance doses every other week initiated on day 28 , except for patient #12, whose treatment was stopped before maintenance due to adverse effects. Patients were discharged after the first dose of dalbavancin and subsequent ones were administered in the day hospital. | 3.716797 | 0.443115 | sec[2]/sec[2]/p[0] | en | 0.999998 | 38958260 | https://doi.org/10.1093/jac/dkae212 |
Two patients presented adverse events that led to the discontinuation of dalbavancin. Patient #10 presented fever and a diffuse confluent erythematous maculopapular rash, mostly affecting the trunk and without mucosal involvement 24 hours after the first administration of dalbavancin. He was also receiving captopril, diuretics, phenytoin, sedative medication, micafungin and ciprofloxacin. No more doses of dalbavancin were administered and he completed the treatment with linezolid. The patient did not need corticosteroids or antihistamines and the rash resolved within 36 hours. Patient #12 presented with one vomiting session after the first dose of the drug and several vomiting sessions, watery stools and abdominal discomfort following the second dose. We changed the antibiotic to oral cefadroxil. | 3.689453 | 0.970215 | sec[2]/sec[3]/p[0] | en | 0.999998 | 38958260 | https://doi.org/10.1093/jac/dkae212 |
Ectopic adrenocorticotropic secretion (EAS) constitutes a rare condition, responsible for less than 17% of all Cushing’s syndrome (CS) cases . It can result from excess hormonal secretion caused by small benign tumors or widespread metastatic disease. There are instances where the culprit lesion remains undetected even after conducting exhaustive investigations . Symptoms are mainly due to hypercortisolemia, which can cause a wide range of presentations including hypokalemia, hyperglycemia, hypertension, weight gain, abdominal striae, skin bruises, hirsutism, menstrual irregularities, decreased libido, facial plethora, and psychiatric manifestations. The diagnosis poses a challenge as it requires hormonal dynamic testing and radiological scans to accurately diagnose EAS and identify the ectopic source, which remains occult in approximately 20% of cases . EAS carries a high risk of mortality and morbidity if left untreated. Therefore, localizing the source of adrenocorticotropic hormone (ACTH) and removing the lesion contribute to disease remission in 80% of cases, with a better overall prognosis . In our case, we discuss an instance of EAS with an unusual presentation of acute severe peripheral edema and significant weight gain. | 4.230469 | 0.90918 | sec[0]/p[0] | en | 0.999997 | 39092370 | https://doi.org/10.7759/cureus.63619 |
A 76-year-old man was referred by the general practitioner (GP) to the Same Day Emergency Care Unit (SDEC) with worsening bilateral leg swelling of unknown cause, fatigue, dizziness, and a significant weight gain of about 10 kilograms (kg) over 10 days. His blood tests subsequently showed evidence of hypokalemia, which was exacerbated by diuretics prescribed by his GP to treat fluid overload. His past medical history included a left nephrectomy performed around 20 years ago for renal cell carcinoma, chronic obstructive pulmonary disease, hypothyroidism, and a history of active smoking (10 cigarettes per day for 40 years). | 3.427734 | 0.986328 | sec[1]/p[0] | en | 0.999999 | 39092370 | https://doi.org/10.7759/cureus.63619 |
Upon examination, he was found to have peripheral fluid overload in both upper and lower limbs, causing pitting edema. His lungs were clear on auscultation, heart sounds were normal, and neck veins were undetectable. The abdominal examination revealed abnormal purplish scar tissue on a previous nephrectomy scar and facial plethora. His systolic blood pressure readings ranged between 160 millimeters of mercury (mmHg) and 195 mmHg. A chest x-ray showed clear lung fields with a normal cardiac shadow. An electrocardiogram (ECG) showed sinus rhythm with an incomplete right bundle branch block. Pro-brain natriuretic peptide (pro-BNP) came back raised at 1,301 nanograms per liter (ng/L), and a cardiology opinion was sought regarding the possibility of a heart failure diagnosis. However, after a specialist review and an Echocardiography study, a heart failure diagnosis was ruled out. Other differential diagnoses included hepatic or renal causes, which were excluded due to normal renal and liver functions with no evidence of organ failure. | 3.884766 | 0.981445 | sec[1]/p[1] | en | 0.999995 | 39092370 | https://doi.org/10.7759/cureus.63619 |
At this stage, a random cortisol test was requested and came back raised at 1,607 nanomoles per liter (nmol/L). Based on the clinical picture, biochemical results, and after reviewing his medications list, an endogenous source of hypercortisolemia was suspected to be the cause of the presentation. Due to his previous cancer history, a computed tomogram (CT) scan of the chest, abdomen, and pelvis with contrast was arranged, which was queried for a 26-millimeter (mm) lesion in the right middle lobe of the lung . | 3.580078 | 0.978516 | sec[1]/p[2] | en | 0.999996 | 39092370 | https://doi.org/10.7759/cureus.63619 |
After detecting hypothyroxinemia, the magnetic resonance imaging (MRI) of the pituitary study was arranged to rule out the pituitary cause of the hormonal disturbances. The study showed a normal gland with no suspicious lesions. Further discussion in the endocrine multi-disciplinary team (MDT) meeting suggested the provisional diagnosis of an ectopic source of ACTH, causing ectopic CS. A decision was also made to start the patient on Metyrapone while waiting for further investigations. | 3.244141 | 0.97168 | sec[1]/p[5] | en | 0.999998 | 39092370 | https://doi.org/10.7759/cureus.63619 |
Additionally, the patient was referred to the respiratory team, who arranged a fluorine18-fluorodeoxyglucose positron emission tomogram CT (FDG PET CT) scan and CT-guided lung biopsy. The FDG-PET scan report indicated low-grade uptake in the lung lesion with slightly bulky adrenal glands with no discrete nodules (KS(SH1)) . | 3.072266 | 0.979492 | sec[1]/p[6] | en | 0.999998 | 39092370 | https://doi.org/10.7759/cureus.63619 |
On Metyrapone, gradually the patient’s symptoms improved markedly with a resolution of the peripheral edema. The patient’s weight decreased to 87 kg from 102 kg at presentation. There was restoration of normal blood pressure readings and anti-hypertensives were stopped with blood tests showing correction of the hypokalemia and normalization of the cortisol day profile (Table 3 ). | 3.216797 | 0.986328 | sec[1]/p[8] | en | 0.999998 | 39092370 | https://doi.org/10.7759/cureus.63619 |
A 57-year-old male without medical history came to our hospital due to intermittent low-grade fever for 10 days. His body temperature fluctuated between 37.5–38.0°C. On admission, physical examination revealed a temperature of 37.9°C and a heart rate of 94 beats per minute. A diastolic murmur could be heard in the second auscultation area of the aortic valve. Laboratory tests indicated an elevated erythrocyte sedimentation rate (ESR) to 35 mm/h, an increased white blood cell count to 13.56 × 10 9 /L, and a raised percentage of neutrophils to 72.9%. p-ANCA was positive and the anti-MPO antibody exceeded 400 RU/ml (normal value, <20.0 RU/ml). c-ANCA and anti-PR3 antibodies were both negative. Multiple blood cultures showed no bacterial infection, and acid-fast stain test of Mycobacterium tuberculosis was negative. Electrocardiogram showed sinus rhythm. Chest CT revealed a lobulated and spiculated mass in the apical segment of the left lung upper lobe, measuring approximately 4.8cm × 3.7 cm . Subsequently, the patient underwent CT-guided puncture biopsy of the mass in the left upper lobe. Pathological findings showed predominantly fibrous connective tissue with some alveoli showing organizing pneumonia . Echocardiography indicated thickening and enhanced echoes of the left and right coronary cusps of the aortic valve, with moderate echogenic masses observed on both valve leaflets. Aortic valve opening was limited and closure was poor . Color Doppler ultrasound showed accelerated blood flow through the aortic valve during systole, with a peak speed of 2.7 m/s, a pressure gradient of 29 mmHg, and moderate regurgitation flow signal during diastole . The patient was diagnosed with AAV based on laboratory and relevant imaging. Therefore, the patient was treated with prednisone (60 mg/day) and methotrexate (15 mg/week) for 10 days before being discharged. After discharge, the patient continued to take oral steroids combined with immunosuppressants for 2 months. Follow-up chest CT showed the disappearance of the pulmonary mass, and the organizing pneumonia had improved compared to before. Echocardiogram showed improvement in the thickening of the left and right coronary cusps of the aortic valve compared to the previous examination . p-ANCA and anti-MPO antibodies decreased significantly throughout the treatment process. Comparing the changes in aortic valve lesions before and after treatment, the thickening of the aortic valve of the patient was considered to be inflammatory and hyperplastic changes of the aortic valve due to AAV. After continuing treatment with steroids and immunosuppressants for half a year, the thickening of the aortic valve almost disappeared. | 4.089844 | 0.974121 | sec[1]/sec[0]/p[0] | en | 0.999996 | 39091355 | https://doi.org/10.3389/fcvm.2024.1434734 |
A 60-year-old female presented to our hospital with intermittent fever and cough for 2 months. 6 months before that, the patient came to our hospital with persistent high fever for 1 month. Tuberculosis antibody was positive and three sputum cultures did not reveal antacid bacilli. She underwent fiberoptic bronchoscopy with tissue biopsy. Pathological examination revealed scattered inflammatory cells, macrophages, and no malignant cells were seen. Chest enhancement CT showed multiple nodular, striated, and mass-like shadows in the left lng upper lobe and hilum area of the lung, with mild circumferential enhancement, as well as scattered nodular and striated foci in the rest of the lungs. 18 F-fluorodeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) scan showed abnormally high FDG uptake in the nodules of the upper lobe of both lungs, and also in scattered lymph nodes of the hilar and mediastinal regions. These findings suggested a high probability of pulmonary tuberculosis and then respiratory physicians initiated diagnostic anti-tuberculosis treatment. In addition, the patient was treated with prednisone (20 mg/day) for 15 days due to persistent hyperthermia. Subsequently, this patient was discharged from the hospital with normal body temperature, and she continued to receive 6 months of anti-tuberculosis treatment with standard therapeutic doses of oral isoniazid, rifampicin, ethambutol, and pyrazinamide. After admission, physical examination revealed a temperature of 38.4°C. Systolic murmur could be heard along the left sternal border at the second intercostal space. Auscultation of the lungs revealed coarse breath sounds, with no dry or wet rales heard. Laboratory tests showed an elevated ESR to 37 mm/h and an elevated ultrasensitive C-reactive protein to 4 μg/ml. c-ANCA was positive and the anti-proteinase 3 (PR3) antibody was elevated to 103.5 RU/ml (normal value, <20 RU/ml). Multiple blood and sputum cultures showed no bacterial infection. Antinuclear antibodies, antiphospholipid antibodies, phosphorus cancer-associated antigen, and p-ANCA were all normal. Chest enhancement CT showed no significant changes in lung lesions compared to six months earlier, and stenosis at the beginning of the left and right pulmonary arterial trunks . Echocardiography showed that the wall of the main pulmonary artery and the left and right pulmonary arteries were unevenly thickened, and the lumen was narrowed . Color Doppler showed a colorful mosaic pattern of blood flow in the main pulmonary artery and the left and right pulmonary arteries during systole, with systolic blood flow speeds of approximately 3.5 m/s, 3.0 m/s, and 3.3 m/s, respectively . Pulmonary artery CT angiography showed uneven thickness of the main pulmonary artery, thickening and narrowing of the proximal walls of the left and right pulmonary arteries, and occlusion of the branches of the upper left pulmonary artery . All these observations strongly supported the diagnosis of AAV, rather than the pulmonary tuberculosis diagnosed six months before. To validate the diagnosis, the patient underwent a CT-guided puncture biopsy of the left pulmonary nodule. Pathological findings showed interstitial fibrotic tissue proliferation and scattered few chronic inflammatory cell infiltrates, which further supported our diagnosis . The patient was treated with prednisone (50 mg/day) and cyclophosphamide (75 mg/day) for 15 days. Follow-up echocardiogram showed a reduction in stenosis of the main pulmonary artery and the right and left pulmonary arteries . This confirmed that hormonal and immunosuppressive treatments can improve pulmonary stenosis. Therefore, we concluded that the pulmonary artery stenosis in this patient was a change caused by AAV. In the subsequent treatment process, the patient's condition worsened, and she died from heart failure despite resuscitation efforts. | 4.089844 | 0.975098 | sec[1]/sec[1]/p[0] | en | 0.999995 | 39091355 | https://doi.org/10.3389/fcvm.2024.1434734 |
We reported two rare cases of AAV involving large vessels, both of which presented with fever. Chest CT scans in both cases revealed lung abnormalities, and etiological examinations were negative, indicating that the conditions were not infectious lesions. The first case involved a male in good health status previously. Ultrasound revealed an aortic valve lesion. Pathological findings of a puncture biopsy of a mass in the upper lobe of the left lung showed predominantly fibrous connective tissue with some alveoli showing mechanised pneumonia changes. Combined with serological ANCA testing (p-pattern by immunofluorescence and MPO antibodies detected by ELISA), the diagnosis was MPA with involvement of the aorta. After treatment, the aortic valve lesion was almost reversed and the lung mass disappeared, and the organizing pneumonia was better than before ( Table 1 ). With promptly diagnosis and quickly targeted therapeutic measures, the patient's condition was effectively controlled with remarkable efficacy. | 4.0625 | 0.961426 | sec[2]/p[2] | en | 0.999997 | 39091355 | https://doi.org/10.3389/fcvm.2024.1434734 |
The second case involved a 60-year-old woman with persistent fever. At the first time of admission six months earlier, although there was no direct pathogenetic evidence of tuberculosis, the chest enhancement CT and PET/CT findings were highly favorable for the diagnosis of tuberculosis. We treated her with diagnostic anti-tuberculosis treatment for a high incidence of tuberculosis in China ( 12 ). In addition, short-term, low-dose prednisone therapy, which also has a therapeutic effect on AAV, originally used for her persistent hyperthermia, therefore, her condition was under control and she was free of fever during the hospitalisation. After being discharged, the patient continued to receive anti-tuberculosis treatment for 6 months. At the second time of admission, ultrasonography and pulmonary artery CT angiography both showed wall thickening and luminal narrowing in the main pulmonary artery and proximal left and right pulmonary arteries. Pathological findings of puncture biopsy of the left lung nodule showed interstitial fibrotic tissue hyperplasia and scattered few chronic inflammatory cell infiltrations. Blood tests indicated positive c-ANCA and anti-PR 3 antibodies. Based on these findings, the patient could be diagnosed with GPA, and her condition had worsened compared to six months earlier, as the lesions had involved the main pulmonary artery and its primary branches. After receiving treatment with steroids combined with immunosuppressants, the condition of the pulmonary artery stenosis improved. She died of heart failure eventually because she did not receive timely treatment and was not adherent to prescription. Figure 5 is a timeline of the patient's diagnosis and treatment process. The patient's clinical manifestations lacked specificity, and the imaging findings were more likely to be tuberculosis; in addition, without enough knowledge of AAV, which is clinically rare, non-specialists could not rule out other non-tuberculous lung diseases and autoimmune diseases that start with pulmonary symptoms, resulting of the misdiagnosis and delay of the treatment. | 4.0625 | 0.975098 | sec[2]/p[3] | en | 0.999996 | 39091355 | https://doi.org/10.3389/fcvm.2024.1434734 |
These two case reports underscore the rarity, complexity and severity of AAV with large vessel involvement. The diagnosis of AAV involving large vessels requires a combination of multiple imaging methods and serological examination, which poses a great challenge. Understanding such rare presentations can aid clinicians in recognizing and diagnosing similar atypical cases more effectively. Although its prognosis is poor, vascular lesions can be reversed if treated early. Therefore, clinicians should diagnose AAV as early as possible, thoroughly evaluate the functional status of the affected organs, provide rational treatment, follow up closely, and adjust the treatment plan timely. | 3.992188 | 0.849121 | sec[3]/p[0] | en | 0.999999 | 39091355 | https://doi.org/10.3389/fcvm.2024.1434734 |
A man in his 70s came to our hospital complaining of weight loss, anorexia, and dysphagia. His medical history included hypertension and dyslipidemia, in addition to PD. He had mild dementia because his mimimental state examination score was 20. He had been taking medication for PD for 12 years, and his weight loss commenced in the last year of admission, with a loss of 10 kg in the last three months. In October of the same year, fatigue, dizziness, dysuria, and severe constipation developed along with a marked decrease in food intake. A few days before visiting our hospital, he visited a nearby hospital and underwent esophagogastroduodenoscopy and cranial magnetic resonance imaging; however, the results were negative. Blood tests revealed mild renal dysfunction (estimated glomerular filtration rate: 36.9 mL/min/1.73 m 2 ), mild inflammatory response (C-reactive protein: 1.24 mg/dL), and anemia (hemoglobin: 8.2 g/dL). He was admitted to our hospital for further examination and tests for weight loss, anorexia, and anemia were performed. Upon admission, a general physical examination revealed no significant abnormalities. However, a neurological examination revealed resting tremors, bladder and bowel incontinence, and orthostatic hypotension. In contrast, the mask-like facial appearance and muscle rigidity were very mild, even though his swallowing function significantly declined. He developed epileptic seizures and delirium on the morning of the 3rd day after amantadine and droxidopa was initiated for orthostatic hypotension. The total number of his medicines was 14 then ( Table 1 ). His epileptic seizures were considered as tonic–clonic, although he did not have status epileptics throughout the clinical course. The patient’s delirium was considered as a mixed type. Confusion Assessment Method (CAM) short form was applied and his condition met the CAM short form diagnostic criteria ( 9 ). The brain magnetic resonance imaging only showed mild cerebral atrophy, and the patient did not have any history of cerebrovascular diseases or seizures in the past. The electroencephalogram showed repetitive sharp waves and spike-and-wave complexes in the left frontal and temporal lobes, but any significant electrolytes imbalances were not observed including serum magnesium (2.3 mg/dL) and no magnesium supplement had been taken. Although the patient showed a mild loss of appetite, he did not have any infections including pneumonia or urinary tract infection. Because these results suggested intoxication from dopaminergic medicines, amantadine and droxidopa were discontinued. After discontinuation of amantadine and droxidopa, we increased the doses of levodopa (600 mg/day) and entacapone (600 mg/day) to 1,000 mg/day for each medicine. After these adjustments, his Parkinsonism was well controlled and no adverse reactions such as epileptic seizure, delusion or delirium were observed for half a year. Furthermore, his anorexia was improved but urinary symptoms or constipation was not changed drastically. In addition, he underwent a total colonoscopy for further examination, but no findings explaining the weight loss or any other issues were identified. Based on the patient’s clinical course, we concluded that his multiple manifestations were attributable to polypharmacy, including the use of anti-PD medicines. | 3.964844 | 0.978516 | sec[1]/p[0] | en | 0.999998 | 39091287 | https://doi.org/10.3389/fmed.2024.1415988 |
This patient, who was originally consuming 14 medicines ( Table 1 ), including six anti-PD medicines, presented with seizures and delirium after admission to the hospital. Based on the clinical course, the patient’s symptoms were speculated to have occurred due to elevated blood amantadine levels triggered by droxidopa. Considering the patient’s moderately impaired renal function, it is improbable that renal function alone caused amantadine intoxication, suggesting that polypharmacy also contributed. | 3.318359 | 0.98291 | sec[2]/p[4] | en | 0.999997 | 39091287 | https://doi.org/10.3389/fmed.2024.1415988 |
Polypharmacy is a serious issue in older adults, with overmedication leading to many unfavorable events. As multiple medicines regulate the balance of several neurotransmitters in PD, adding one agent could disrupt the homeostasis of the neuronal network. It may be useful to promote deprescribing in the everyday clinical practice rather than adding a medication for a new symptom or an adverse reaction. Considering the aforementioned information, treating patients with the minimum amount of medication whenever feasible is important. To achieve this goal, drug adjustments and prescriptions are recommended. In addition, patients with PD consuming multiple agents are at risk of developing amantadine intoxication due to drug interactions, even if the renal dysfunction is mild to moderate. This report emphasizes the risk of polypharmacy and suggests the importance of citing amantadine intoxication as a differential diagnosis in patients with PD and mild to moderate renal dysfunction who present with symptoms of seizures and/or delirium. | 4.140625 | 0.636719 | sec[2]/p[5] | en | 0.999996 | 39091287 | https://doi.org/10.3389/fmed.2024.1415988 |
Herein, we report a case of ruptured GO closed by the application of vacuum sealing drainage (VSD) combined with silver dressing and share the nutritional support strategy for congenital short small intestine. | 3.318359 | 0.974609 | sec[0]/p[2] | en | 0.999998 | 39091686 | https://doi.org/10.3389/fnut.2024.1421033 |
A 2-day-old girl with a 12 × 10 cm ruptured GO was transferred to our hospital on June 5, 2023. She was born vaginally with a birth weight of 3,000 g. Gestational age was 40 + 4 weeks. Apgar scores at 1 and 5 min were 10. The bowel was partially visible through the postnatally ruptured sac . The neonate had dyspnea and poor peripheral perfusion on examination owing to enteral fasting and evaporative fluid loss, characterized by decreased urine output, skin mottling, and prolonged capillary refill time. Radiography revealed polydactyly of the left foot and a right-sided thoracolumbar curve. After stabilization with fluid resuscitation, vasoactive drugs, antibiotics, and mechanical ventilation, pediatric surgeons placed a silo bag that night. The silo bag was anchored to the full layer of the abdominal wall with interrupted sutures so that pressures could be added post-operatively for reduction. The membrane of the sac was completely broken. There was severe adhesion and contamination on the intestines. Insensible water loss was increased from the intestines through the postnatally ruptured sac. The entire small bowel and colon, part of the stomach, and most of the liver were in omphalocele. The herniated portion of the liver was spherical. The length of the entire small intestine was 50 cm. Postoperatively, her intra-abdominal pressure was monitored, and she had a foley catheter to drain urine, thereby reducing the increase in intra-abdominal pressure caused by urinary retention. On the 10th postoperative day, the tension in the abdominal wound increased gradually, and the inferior suture ruptured with skin erythema and exudate. Additionally, she had recurrent fever with persistently elevated C-reactive protein (CRP) levels of up to 141 mg/L . | 3.867188 | 0.982422 | sec[1]/p[0] | en | 0.999996 | 39091686 | https://doi.org/10.3389/fnut.2024.1421033 |
Proper assessment and personalized wound repair strategy followed a multidisciplinary approach by an expert team, including neonatologists, plastic surgeons, pediatric surgeons, pediatric infectious disease specialist, enterostomal therapist, and radiologist. The protective wound material comprised three layers: the first layer was petroleum gauze, second layer was a carboxymethylcellulose silver dressing, and third layer was the closed VSD protective wound material; the VSD machine was connected. The VSD dressing was changed at the bedside 6 times over 38 days. CRP levels gradually decreased to normal without fever , the amount of liver exposure gradually decreased, and the wound gradually narrowed. A large amount of cellulosic exudate was observed on the surface of the antiadhesive petroleum gauze, which could be observed through the growth of granulation tissue and bled easily to touch , forming an intact granulation barrier. When the dressing was removed, the wound size was reduced to 2.3 × 2.6 cm; then, bovine basic fibroblast growth factor gel was applied to the wound to promote epithelialization. Finally, the abdominal wall defect was restored. On hospital days 28 and 47, alanine aminotransferase (ALT) and aspartate aminotransferase (AST) levels were markedly elevated . Other liver function tests, including bilirubin, albumin, international normalized ratio, and coagulation function, were normal. ALT and AST returned to normal levels within 1 week after weaning off VSD. Full enteral feeding was achieved within 19 days after parenteral nutrition and early trophic feeding. After 11 days of mechanical ventilation, she was successfully extubated. Through 58 days of hospitalization, the infant grew from 2,930 to 4,670 g, with an average growth of 30 g per day. At the last follow-up, the infant was 6 months old and breastfed exclusively for 3 months with normal growth and development. | 3.984375 | 0.960938 | sec[1]/p[1] | en | 0.999998 | 39091686 | https://doi.org/10.3389/fnut.2024.1421033 |
This patient developed markedly elevated serum ALT and AST levels. These suggested that the biochemical liver alterations were caused by hepatocellular injury. The etiologies of hepatocellular injury include acute viral hepatitis, ischemic hepatitis, septic shock, toxin/medication, metabolic disorders, and so on ( 29 ). This patient was tested for viruses, heavy metals toxin, and genetic metabolic disease, all of which were negative. Since the spherical shape of the liver was susceptible to injury by vacuum sealing drainage, the increased intra-abdominal pressure may have led to hepatic ischemia. In this case, the ALT and AST returned to normal ranges after withdrawing VSD. Therefore, we think that the elevated ALT and AST may be associated with hepatic ischemia/reperfusion injury. | 3.929688 | 0.817871 | sec[2]/p[2] | en | 0.999997 | 39091686 | https://doi.org/10.3389/fnut.2024.1421033 |
The mean small bowel length in a child with congenital short bowel is 57 cm ( 30 ). The patient’s small bowel length was only 50 cm, which was consistent with a congenital short bowel. It has been reported that the early onset of clinical signs, including such as vomiting and diarrhea, in children with congenital short bowel disease suggests a poor prognosis, and delayed presentation suggests a favorable outcome ( 12 ). This patient did not develop relevant symptoms early on, and full enteral feeding was eventually successfully established. The management goals for congenital short small bowel and short bowel syndrome caused by intestinal resection postoperatively are similar. During the early intestinal maladaptation period, parenteral nutrition support is required to gradually enhance intestinal adaptation and achieve enteral autonomy, while avoiding complications such as intestinal failure-related liver disease and catheter-related bloodstream infections ( 15 , 31 ). Breast milk is the first choice for enteral nutrition for children with short bowel syndrome; however, when breast milk is not available, some scholars recommend the use of hydrolyzed or amino acid milk because children with short bowel syndrome are prone to cow’s milk protein allergy ( 32 , 33 ). Breast milk was unavailable; therefore, we chose extensively hydrolyzed formulas (eHF) combined with active parenteral nutrition for early trophoblastic feeding. Owing to the low osmolality of eHF, its energy density (67–81 kcal/dL) could be increased with the use of additional milk powder. Moreover, eHF is more prone to inducing immune tolerance than the amino acid formula ( 34 ). After 19 days, she successfully transitioned to full enteral feeding by eliminating parenteral nutrition without complications such as intestinal failure-related liver disease or cow’s milk protein allergy. | 4.179688 | 0.551758 | sec[2]/p[3] | en | 0.999998 | 39091686 | https://doi.org/10.3389/fnut.2024.1421033 |
All surgeries were performed by one surgeon. Under general anesthesia, the patient was placed in the beach chair position. The fracture site was exposed via the previous incision. The fibrotic tissue in the fracture area was removed, and in the case of nonunion, the sclerotic bone and cyst were removed using a burr or curette. We used 3.5 mm superior locking compression plates (LCPs) for superior clavicle and anterior reconstruction (Depuy Synthes, MA, USA) in all patients. In addition, bone allograft was not performed, and the defect was filled using moldable allograft bone (S1-OXB, MedPark). Alignment was adjusted using reduction forceps, and the superior locking plate was fixed first. Using a 9−10-hole-long plate fixation, three locking and cortical screws were inserted at each end, and then, the bone graft was performed on the defect. Then, to fix the anterior aspect of the clavicle, the reconstruction plate was bent according to the bone curvature, and a screw was inserted . Fig. 1 A 63-year-old female who underwent ORIF for clavicle shaft fracture 10 months earlier was transferred due to plate and screw loosening with nonunion. A1 Plate and screw loosening after primary ORIF, A2 postoperative anteroposterior view of double plate fixation, A3 anteroposterior view 3 months after revision surgery, A4 anteroposterior view 24 months after revision surgery | 3.865234 | 0.873047 | sec[1]/p[2] | en | 0.999997 | 38598169 | https://doi.org/10.1007/s00590-024-03927-5 |
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