American Journal of Case Reports最新文献

BACKGROUND Multifocal acquired demyelinating sensory-motor neuropathy (MADSAM) is recognized as a variant of chronic inflammatory demyelinating polyneuropathy. The primary characteristics of MADSAM include multifocal sensory loss and muscle weakness, which are frequently asymmetrical and predominantly affect the upper limbs. Involvement of the lower limbs is less commonly observed in MADSAM. CASE REPORT A 27-year-old female patient presented with recurrent numbness and weakness in her left lower limb was admitted to our hospital. Her medical history included episodes of left peripheral facial paralysis and lower-limb numbness and weakness, which had previously improved after short-term oral steroid therapy. In addition to motor and sensory peripheral nerve impairment in the left lower limb, the neurological examination revealed atrophy of the tongue muscle and a leftward deviation of the tongue. Cerebrospinal fluid examination and magnetic resonance imaging indicated no abnormalities. Electromyography suggested demyelination of motor and sensory nerves in the left lower limb. Sural nerve biopsy demonstrated demyelination changes and axonal degeneration. A diagnosis of multifocal sensory and motor neuropathy was considered, and the patient was administered corticosteroids and tacrolimus. As the condition progressed, electromyography showed gradual involvement of both lower limbs, leading to the consideration of MADSAM. Despite treatment with corticosteroids and tacrolimus, the patient experienced relapse. Rituximab was initiated, resulting in symptoms improvement and reduced recurrence without adverse events. CONCLUSIONS Corticosteroids, plasma exchange, and immunoglobulins have been demonstrated to be effective treatments for CIDP. In our MADSAM case, rituximab proved effective when the patient did not respond to corticosteroids and tacrolimus. We propose that rituximab may serve as an alternative option for patients with MADSAM.

BACKGROUND Papillary thyroid carcinoma (PTC) is the most common form of thyroid cancer, while medullary thyroid carcinoma (MTC) and anaplastic thyroid carcinoma (ATC) are far less common. ATC is the most aggressive form, contributing to the majority of thyroid cancer-related deaths. While the coexistence of different pathological types of thyroid malignancies has been reported, including combinations of ATC, PTC, and poorly differentiated thyroid carcinoma, the detection of ATC within extrathyroidal metastatic lymph nodes remains exceedingly rare and of notable clinical significance. CASE REPORT We present a 68-year-old woman evaluated for a progressively enlarging right-sided neck mass which was apparat in clinical examination, with no other associated symptoms or signs. Nasopharyngoscopy was unremarkable. Imaging studies revealed a large cystic and necrotic mass in the right carotid space compressing the internal jugular vein. A core needle biopsy confirmed metastatic PTC. The patient underwent total thyroidectomy with right radical neck dissection. Histopathological evaluation reported a unique combination: a collision tumor composed of PTC, infiltrative follicular subtype, and MTC. Immunohistochemistry confirmed the diagnoses, and molecular analysis revealed a BRAF mutation. The patient was not a candidate for radioactive iodine therapy due to the aggressive nature of the anaplastic component. Thus, she was referred for targeted cancer therapy. CONCLUSIONS This case highlights the synchronous occurrence of PTC, MTC, and ATC in a single patient. The localization of anaplastic transformation solely to lymph nodes without thyroidal involvement underscores the importance of meticulous histopathological assessment. Comprehensive diagnostic workup and molecular profiling are critical in guiding treatment for such complex presentations.

BACKGROUND Acute graft-versus-host disease is a rare but highly fatal complication of liver transplantation, with a reported mortality rate exceeding 70%. Most patients are diagnosed at 3 to 6 weeks postoperatively. Early diagnosis remains challenging because of nonspecific symptoms and overlapping presentations of infections and drug reactions. CASE REPORT We report the case of a 48-year-old woman with end-stage liver disease secondary to primary biliary cholangitis who underwent deceased-donor liver transplantation. On postoperative day 16, she developed abrupt-onset anemia without bleeding, followed by a high-grade fever, erythematous rash, profuse diarrhea, and pancytopenia. Blood cultures and viral studies were negative, despite clinical deterioration. Punch biopsy of the trunk on postoperative day 20 confirmed acute graft-versus-host disease. She was treated with high-dose corticosteroids and enhanced immunosuppression but showed no improvement, meeting the criteria for steroid-refractory graft-versus-host disease. She died of multiorgan failure on postoperative day 30. This is one of the earliest biopsy-confirmed cases of graft-versus-host disease reported after deceased-donor liver transplantation. CONCLUSIONS This case highlights the importance of early clinical suspicion of graft-versus-host disease after liver transplantation, even before the full triad of symptoms manifests. Isolated anemia may be an early warning sign. Therefore, prompt histopathological confirmation via skin biopsy is essential. Current treatment strategies for steroid-refractory graft-versus-host disease are limited, underscoring the need for further therapeutic advances.

BACKGROUND In coronary artery bypass grafting, hyperthyroidism with autoantibodies, as seen in Basedow disease, is uncommon and rarely leads to severe thyrotoxicosis. However, surgery can trigger this condition, and documented cases have resulted in poor outcomes. We describe the case of an incidental hyperthyroidism finding on preoperative evaluation for coronary artery bypass grafting for triple-vessel coronary artery disease. CASE REPORT A 58-year-old man presented with exertional dyspnea, chest pain, and postprandial vomiting. Coronary angiography revealed severe triple-vessel disease. After admission, he experienced additional episodes of chest pain, which required continuous infusion of coronary vasodilators and strict bed rest. Preoperative screening showed severe hyperthyroidism. Thiamazole 80 mg/day and potassium iodide 50 mg/day were initiated but discontinued after 2 weeks because of leukopenia (white blood cell count, 2800×10⁹/L). Although thyroidectomy was considered, coronary artery bypass grafting was prioritized owing to cardiac risk concerns. Perioperative management included continuous hemodiafiltration dialysis, methylprednisolone (1000 mg/day), and slow plasma exchange, starting with surgery to prevent thyroid storm. Intravenous gamma globulin was administered 3 days before and after surgery. Free triiodothyronine and free thyroxine were normalized on postoperative days 3 and 7, respectively. The postoperative course was uneventful, and the patient remained symptom-free at 1-year follow-up. CONCLUSIONS Coronary artery bypass grafting with uncontrolled hyperthyroidism is prone to complications, and a thyroid storm can be fatal. Although surgery should be avoided in cases of uncontrolled hyperthyroidism, in rare cases in which urgent treatment is not feasible, a critical intervention as described in this report may prevent thyroid crisis.

BACKGROUND Filiform polyposis (FP) is a rare pseudopolyp associated with inflammatory bowel disease (IBD), characterized by elongated mucosal projections. It is found primarily in the colon and rectum, and presentation in the ileum is rare. Despite its benign nature, FP can mask underlying chronic intestinal inflammation. CASE REPORT A 52-year-old woman presented with loose stool but no other gastrointestinal symptoms. She was found to have a branched mucosal mass in the terminal ileum and diffuse ulcerations throughout the colorectum. Initial laboratory tests indicated anemia and elevated inflammatory markers. After 3 months of treatment with mesalazine and adjuvant drugs, the symptom of loose stool had improved, but the colonoscopy and imaging examination results show little change from the initial visit. Colonoscopy, imaging, and histopathology supported a diagnosis of Crohn's disease (CD). The ileal mass was identified as filiform polyposis. After multidisciplinary consultation, she was treated with Ustekinumab, leading to significant symptomatic and endoscopic improvement. The mass was subsequently resected via hybrid ESD and confirmed as an inflammatory polyp. CONCLUSIONS This case reveals that although a patient has no significant symptoms related to CD, FP located at the terminal ileum can initially manifest as CD, highlighting the need for through evaluation to detect underlying IBD. Multidisciplinary collaboration is essential for accurate diagnosis and management. FP can be the "tip of the iceberg" in hidden chronic intestinal inflammation, necessitating careful endoscopic and histologic assessment.

BACKGROUND Eisenmenger syndrome presents a unique challenge for the acute care surgeon. Even routine operations such as laparoscopic appendectomy or cholecystectomy become challenging due to the cardiopulmonary physiologic changes and anatomic anomalies associated with Eisenmenger syndrome. The care of these patients can be further complicated by the severity of disease, surgical complexity, and the abnormal anatomy associated with the syndrome. CASE REPORT A 35-year-old patient with Eisenmenger syndrome and abdominal heterotaxy presented with acute cholecystitis. She underwent percutaneous cholecystostomy tube placement during her index hospitalization, which was complicated by atrial fibrillation and a cerebrovascular accident due to air embolism. Three months after presentation, she underwent an uncomplicated open cholecystectomy. She was discharged on post-operative day 5, and her course was notable only for a superficial surgical site infection requiring incision and drainage and antibiotics. CONCLUSIONS Our experience managing acute cholecystitis in a patient with Eisenmenger syndrome, abdominal heterotaxy with interrupted inferior vena cava, and bilateral superior vena cava, highlights critical aspects of care of such patients in the context of routine acute surgical care. Pre-operative planning should include optimization of cardiopulmonary function, an individualized anesthetic plan to maintain systemic vascular resistance, and bailout maneuvers in the event of cardiovascular collapse, such as planning for extracorporeal membrane oxygenation with axillary cannulation in the case of our patient.

BACKGROUND Giant cell arteritis (GCA) is the most common large-vessel vasculitis in individuals over age 50 years. Although it typically affects extracranial branches of the carotid artery, central nervous system involvement is rare and can manifest with ischemic stroke. Links between GCA and malignancy have been reported, especially hematologic cancers, but paraneoplastic GCA associated with solid tumors remains exceptional and poorly understood. CASE REPORT We describe a 59-year-old man presenting with temporal headaches, right-hand paresthesia, monocular visual loss, and gait instability. Imaging revealed left thalamic ischemia and bilateral vertebral artery stenosis without atherosclerosis. Examination and Doppler ultrasound supported a diagnosis of GCA with associated polymyalgia rheumatica (PMR), although a temporal artery biopsy was negative. Inflammatory markers were only moderately elevated, consistent with reports of GCA cases with ischemic complications. PET-CT incidentally identified a mediastinal mass, confirmed as small-cell pulmonary neuroendocrine carcinoma. Symptoms improved with corticosteroids, but tapering below 15 mg/day caused relapse, requiring methotrexate. Despite partial oncologic response, brain metastases appeared 8 months later. Follow-up vascular imaging showed regression of arterial stenoses under corticosteroids, arguing against atherosclerosis and supporting the inflammatory nature of the lesions. GCA manifestations remained corticosteroid-dependent throughout follow-up. CONCLUSIONS The simultaneity of GCA and lung neuroendocrine carcinoma, persistence of corticosteroid dependence, and lack of remission despite oncologic response strongly suggest a paraneoplastic mechanism. This case emphasizes the need for heightened suspicion of underlying cancer in patients with atypical, biopsy-negative, or treatment-resistant vasculitis. Recognition of paraneoplastic GCA may improve early cancer detection and influence management decisions at the intersection of rheumatology and oncology.

BACKGROUND Paraduodenal hernia (PDH) is a rare internal hernia, accounting for 50% to 55% of internal hernias but only 0.2% to 0.9% of intestinal obstructions. Right-sided PDH is less common, occurring in approximately 25% of cases. CASE REPORT This case describes a 33-year-old pregnant woman at 20 weeks 6 days gestation who was admitted with a 15-day history of abdominal distension accompanied by nausea and vomiting. The patient had postprandial exacerbation of intermittent abdominal pain, bilious vomiting, and significant weight loss (5 kg), with a history of similar symptoms during previous pregnancies. Abdominal contrast-enhanced computed tomography revealed clustered bowel loops in the right upper quadrant and medial displacement of the superior mesenteric vein, which is consistent with right-sided PDH complicated by intestinal malrotation. After conservative treatment failed, the patient opted for pregnancy termination followed by laparoscopic surgery. Intraoperative exploration confirmed complete absence of fusion between the ascending mesocolon and the posterior peritoneum, resulting in a wide hernia defect through which bowel loops had herniated into the space lateral to the ascending duodenum. The procedure included reduction of herniated contents, adhesiolysis, and fixation of the ascending mesocolon. The patient's recovery was uneventful, and she was discharged on postoperative day 5. At 3-month follow-up, no abnormalities were noted. CONCLUSIONS Given its nonspecific clinical presentation, PDH is frequently misdiagnosed or diagnosed late. Abdominal computed tomography facilitates early diagnosis and timely intervention, while laparoscopic repair offers favorable outcomes.

BACKGROUND This report describes a case in which a 2-stage approach and minimally invasive vitrectomy successfully treated a giant retrobulbar hematoma caused by an ocular perforation injury. CASE REPORT A 22-year-old man sustained an ocular perforation injury from an iron wire. Emergency debridement and suturing of an anterior scleral laceration were performed. B-scan ultrasonography revealed a giant retrobulbar hematoma and posterior scleral laceration after the initial suturing procedure. Ten days later, minimally invasive vitrectomy was performed. During this procedure, intraocular hemorrhage was removed and vitreous traction was released. Laser photocoagulation was applied only to the edges of the posterior chorioretinal wound, and the surgery was completed with air tamponade. Three months after vitrectomy, the injured eye showed satisfactory recovery. B-scan ultrasonography confirmed resolution of the retrobulbar hematoma and closure of the posterior scleral wound. CONCLUSIONS A 2-stage, minimally invasive surgical approach for severe ocular perforation injury was performed at an appropriate time, yielding favorable therapeutic outcomes. In cases of posterior perforating injury of the globe, careful selection of surgical timing is essential; simple vitrectomy combined with air tamponade is safe and effective.

BACKGROUND Hiatal hernia (HH) is a common condition that can present diagnostic challenges when accompanied by complex comorbidities. While HH is associated with obesity and elevated intra-abdominal pressure, its occurrence with eating disorders is not as well documented. We report a rare case of symptomatic HH in a patient with concurrent obesity and bulimia nervosa, a combination not previously reported in the literature. CASE REPORT A 39-year-old woman presented with 2 years of postprandial vomiting, chest pain, dyspnea, and heartburn. Her medical history revealed self-induced vomiting for weight control that progressed to bulimia nervosa, resulting in weight loss of 44 kg. Initial treatments with proton pump inhibitors provided minimal relief. Comprehensive evaluation revealed type III paraesophageal HH (5 cm hernial orifice), grade 2 reflux esophagitis, anemia, and a gallbladder polyp. The patient underwent laparoscopic cruroplasty with Toupet fundoplication and cholecystectomy, combined with psychiatric management including cognitive-behavioral therapy and fluoxetine. At 3-month follow-up, complete symptom resolution was achieved. CONCLUSIONS This case highlights the importance of thorough history-taking in patients with atypical gastrointestinal symptoms and demonstrates that bulimia nervosa can contribute to HH development through repeated increases in intra-abdominal pressure. A multidisciplinary approach integrating surgical and psychiatric interventions is essential for successful management of HH when associated with eating disorders and other complex comorbidities.