Oxford Medical Case Reports最新文献

Vedolizumab is a gut selective anti-integrin monoclonal antibody with a very good and proven safety profile in the treatment of ulcerative colitis (UC). This case report highlights a rather unusual complication that every gastroenterologist ought to be aware of. It relates to the association of acute interstitial nephritis with vedolizumab usage in the management of UC. A 53-year-old man with liver cirrhosis due to Primary Sclerosing Cholangitis and UC was treated with vedolizumab following failed therapy with thiopurines and mesalazine. This led to improvement in his bowel function but his renal function steadily declined over subsequent months, in the presence of reactive urinary sediments. A subsequent renal biopsy demonstrated acute interstitial nephritis. Cessation of vedolizumab, along with high dose steroids treatment, led to near complete resolution of his renal failure.

Introduction: Brugada syndrome is a genetic disorder associated with sudden cardiac death (SCD) due to ventricular fibrillation (VF). The diagnosis relies on detecting a Type 1 ST-segment elevation in the right precordial leads, which often requires sodium channel blocker provocation for confirmation. Patient concerns: We present a case of a 55-year-old male bus driver who experienced out-of-hospital cardiac arrest due to VF. Diagnosis: After resuscitation, initial evaluations, including echocardiography and cardiac enzyme tests, were normal, showing no structural heart disease. The initial electrocardiogram displayed a Type 2 Brugada pattern. Intervention: A flecainide challenge test using oral flecainide (300 mg) revealed a Type 1 Brugada pattern, confirming Brugada syndrome. Outcomes: Despite mild hypokalemia, an implantable cardioverter defibrillator (ICD) was implanted due to the patient's high-risk occupation and VF history. Conclusion: This case underscores the importance of considering Brugada syndrome in unexplained VF cases and highlights the role of flecainide testing in diagnosis. A multidisciplinary approach, including thorough evaluation, timely ICD implantation, and patient education, is crucial for optimal outcomes, particularly for those in safety-critical professions. Recognizing Brugada syndrome and applying appropriate diagnostic and therapeutic measures are essential for preventing fatal arrhythmic events.

Learning points: This case underscores the importance of considering Brugada syndrome in patients with unexplained ventricular fibrillation, emphasizing the utility of the flecainide challenge test for accurate diagnosis when characteristic ECG findings are not evident.A multidisciplinary approach, including timely diagnosis, appropriate ICD implantation, and patient education, is critical in managing high-risk occupations to prevent catastrophic outcomes.

Azygous anterior cerebral artery (ACA) occlusion is a rare vascular anomaly that results in bilateral frontal infarction and carries a high morbidity rate. We present a 48-year-old male with no prior significant medical history who presented eight hours after sudden onset of right leg weakness. Initially, the National Institutes of Health Stroke Scale (NIHSS) score was four. Computed tomography angiography revealed a solitary azygous ACA with distal A2 occlusion. Mechanical thrombectomy was initially postponed due to mild deficits, but it was performed after clinical deterioration to a NIHSS score of eight. The procedure successfully recanalized the artery. Post-procedure magnetic resonance imaging confirmed bilateral ACA infarcts. The patient received medical therapy and rehabilitation, resulting in complete neurological recovery within three months. This case underscores the diagnostic difficulties associated with azygous ACA occlusion in patients presenting with fluctuating symptoms. Timely observation and intervention were pivotal in preventing severe disability.

Primary cardiac tumors are extremely rare. Myxomas are the most common, typically affecting middle-aged women. They arise in the left atrium in 75% of cases. Right atrial myxomas are less common. The coexistence of coronary artery disease is rare but can be complex. We report the case of an 80-year-old male smoker with peripheral arterial disease and stable angina, who was diagnosed with a right atrial mass seven years earlier but initially declined surgery due to fear of the procedure. He was admitted to our hospital with crescendo angina. Echocardiography revealed a large right atrial mass suggestive of myxoma. Coronary angiography demonstrated severe multi-vessel atherosclerotic coronary artery disease, including mid circumflex occlusion. After successful balloon angioplasty, he underwent combined myxoma resection and coronary artery bypass grafting. This case illustrates the uncommon but serious combination of myxoma and acute coronary syndrome, reinforcing the need for careful surgical planning to achieve a favorable outcome.

Background: Insulinomas are rare, typically benign pancreatic neuroendocrine tumors that cause endogenous hyperinsulinemic hypoglycemia. While usually diagnosed in middle age, their presence in young adults may suggest hereditary syndromes, such as MEN1. While semaglutide and other GLP-1 receptor agonists rarely cause hypoglycemia, they can unmask tumors secreting insulin.

Case presentation: A 25-year-old woman with obesity and polycystic ovary syndrome (PCOS) developed severe hypoglycemia (35 mg/dL) after starting semaglutide. She did not respond to IV dextrose. Labs showed hyperinsulinemia (77.5 mU/L) and elevated C-peptide (19.53 ng/mL). Imaging revealed a pancreatic tail mass, pituitary microadenoma, and ovarian teratoma, raising concern for MEN1. She underwent distal pancreatectomy, splenectomy, and right salpingo-oophorectomy. Glycemia normalized postoperatively. Pathology confirmed a grade 1 pancreatic neuroendocrine tumor (Ki-67 < 1%).

Conclusion: This case emphasizes that hypoglycemia occurring during GLP-1 receptor agonist therapy is not always a drug-related side effect but may result from unmasking of an underlying insulinoma.

A 40-year-old male presented with progressive backache; his workup revealed primary multifocal osseous Hodgkin's lymphoma (PMOHL), which is a very rare primary presentation of Hodgkin's lymphoma (HL). The common presenting symptoms include localized bone pain and tenderness with or without fever, weight loss or drenching night sweats. Given the rarity of the spine being affected in PMOHL and its symptoms overlapping with conditions like infectious or malignant causes, this case adds meaningful insight to existing literature so that delays in the diagnosis can be avoided. This case also signifies the need of repeat biopsy to prevent unnecessary delays and polypharmacy. Once diagnosis is made, adriamycin, bleomycin, vinblastine, and dacarbazine (ABVD) is still curative and cost-effective regimen in the cost constraint part of the world.

Introduction: Klippel-Trenaunay syndrome (KTS) is a complex and extremely rare congenital vascular syndrome. The disorder presents with a vascular malformation syndrome involving cutaneous capillaries and venous (hemangiomas and port-wine stains). Lymphatic anomalous development, with hyperplasia of soft tissue and bones, can also occur, which is due to overgrowth occurring as a result of somatic mutations.

Case presentation: We present a 12-year-old male child with a 12-year history of subcutaneous growths, initially painless. KTS was diagnosed in childhood and associated with a vascular malformation of the right thigh, leg, and foot, associated with hypertrophy in the ipsilateral lower leg. Further, the patient had the presence of extensive lymphangioma in the right leg, which co-existed with the vascular malformation. The patient was treated with a multi-disciplinary approach.

Discussion: KTS is a congenital disorder characterized by varicose veins, capillary malformations, and tissue hypertrophy. It may show GI bleeding, orthopedic problems, and possible complications such as splenic hemangiomas. The disorder can be managed using NSAIDs, embolization, sclerotherapy, and surgery for severe cases. Genetic testing and imaging, including MRI, are important in diagnosis and management planning.

Conclusion: In conclusion, KTS is a challenging disease due to its rarity, diagnostic complexity, and varied clinical manifestations. It can be presented with different manifestations and complications, hence makes it very difficult for clinicians to diagnose. For the growing awareness, new research can enhance the knowledge of the clinician about the disease and provide better care for the individuals afflicted by KTS.

A 43-year-old female patient having a known history of pulmonary sarcoidosis presented with a rapidly developing intracranial hypertension, followed by a state of drowsiness. Cerebral MRI revealed intense leptomeningeal contrast enhancement and thickening of the pituitary gland. The present report detailed the primary imaging abnormalities observed in the patient and highlighted the importance of considering a differential diagnosis within the context of the clinical picture The clinical approach supporting the inflammatory origin of the observed imaging abnormalities in this case is discussed. Furthermore, this case highlighted the rapid effect of cortiotherapy in improving the neurological dysfunctions associated with neurosarciodosis which can be life-threatening.