American Journal of Case Reports最新文献

BACKGROUND Heparin-induced thrombocytopenia (HIT) is an immune-mediated reaction that occurs 5-10 days after heparin exposure. Antibodies against platelet factor 4 (PF4) activate platelets and lead to a hypercoagulable state, causing thrombotic complications, including deep venous thrombosis, pulmonary embolism, stroke, and myocardial infarction. Given the widespread use of prophylactic heparin in clinical practice, prompt recognition and management are crucial due to the associated high morbidity and mortality. CASE REPORT A 73-year-old woman with hypertension, hypothyroidism, depression, and anxiety presented with 6 days of nausea, vomiting, abdominal pain, constipation, weakness, and postural dizziness. She recently underwent cervical spine surgery and received prophylactic heparin. Laboratory findings showed normocytic anemia, mild leukocytosis, new-onset thrombocytopenia, and hyponatremia. Further evaluation revealed low morning cortisol, positive PF4 antibodies, and serotonin release assay (SRA), consistent with HIT. CT demonstrated bilateral adrenal enlargement and peripheral fat stranding, confirming adrenal infarction. Discontinuation of heparin and treatment with rivaroxaban, intravenous steroids, and hemodynamic support led to significant improvement. CONCLUSIONS HIT-induced adrenal infarction is uncommon and often overlooked due to its nonspecific presentation. This case emphasizes the importance of suspecting adrenal involvement in patients with prior heparin exposure and hypotension or electrolyte imbalance. Prompt anticoagulant adjustment, steroid replacement, and follow-up with hemodynamic monitoring can improve clinical outcomes.

BACKGROUND Secondary thoracoabdominal aortic replacement after a total arch repair is a complex procedure that has a risk of complications such as stroke and renal failure. Patients with Marfan syndrome are more at risk of distal aortic re-interventions. This report describes a 52-year-old woman with Marfan syndrome and a history of type A aortic dissection treated with total arch replacement and frozen elephant trunk implantation using a sutureless integrated stented graft (SISG), who presented 3 years later with a distal aortic dissection aneurysm requiring secondary thoracoabdominal aortic replacement. CASE REPORT A 52-year-old woman with Marfan syndrome underwent ascending aortic and total arch replacement with frozen elephant trunk implantation using a sutureless integrated stented graft (SISG) for acute type A aortic dissection 3 years earlier. One month before the current operation, thoracoabdominal aortic replacement was performed because follow-up imaging demonstrated false lumen expansion of the residual thoracoabdominal aortic dissection, with a maximal diameter exceeding 5.5 cm. Intraoperatively, significant false lumen thrombosis and obliteration were observed in the elephant trunk region. A 2-cm unstented graft segment at the distal end of the elephant trunk facilitated proximal anastomosis in the current surgery. Given the patient's Marfan syndrome, all branch vessels were reconstructed. Follow-up aortic computed tomography demonstrated satisfactory morphology, and the patient was discharged 2 weeks after surgery. CONCLUSIONS This case demonstrates successful staged repair of residual thoracoabdominal aortic dissection following initial total arch replacement using a SISG in a patient with Marfan syndrome. The sutureless graft facilitated secondary procedure by providing a suitable proximal landing zone, with complete false lumen thrombosis confirming effective primary repair and favorable outcomes.

BACKGROUND Esophagopleural fistula (EPF) is a rare but life-threatening complication following pneumonectomy, with a reported incidence of up to 1% and a mortality rate ranging from 49% to 63%. Management strategies vary depending on the fistula's characteristics and the patient's clinical status, encompassing conservative, endoscopic, and surgical approaches. CASE REPORT We report the case of a 65-year-old woman diagnosed with lung adenocarcinoma and N2 nodal involvement who received neoadjuvant chemo-immunotherapy, followed by surgical treatment with left pneumonectomy. In the early postoperative course, she developed fever and pleural contamination with food particles, leading to the diagnosis of EPF via methylene blue test and esophagogastroduodenoscopy (EGD). Initial endoscopic treatment with stent placement was attempted multiple times but its failure prompted the need for surgical intervention. After addressing the infectious and nutritional issues associated with the condition and optimization of the patient's clinical status, a complex 3-stage procedure was performed involving esophageal exclusion and reconstruction of the gastrointestinal tract using a retrosternally transposed gastric conduit. This was achieved through 3 surgical approaches: laparoscopy, left cervicotomy, and minilaparotomy. The postoperative course was uneventful, with successful reintroduction of oral intake. The esophagogram performed 3 months postoperatively showed a good caliber of the esophagogastric anastomosis and no leakage of contrast from the esophageal lumen. CONCLUSIONS Surgical treatment of large esophagopleural fistulas is indicated when endoscopic treatment fails. Furthermore, esophageal exclusion and bypass is the best course of action when the extent of the fistulous tract presents a problem for direct repair and the chest cavity presents difficulties.

BACKGROUND Empyema is the accumulation of infected fluid within the pleural cavity, sometimes accompanied by pneumothorax. Bacterial empyema is the most common. Tuberculous and fungal empyema are less common and can occur in immunocompromised patients. Empyema caused by mixed infection with both tuberculosis and fungal pathogens is even less common. CASE REPORT This report describes a 76-year-old male lung cancer patient admitted to the hospital with fever and cough. He was receiving tislelizumab immunotherapy before admission. Chest CT at admission revealed pneumonia. Following empirical antimicrobial therapy, the pneumonia showed no improvement. He refused bronchoscopy; therefore, a sputum sample was delivered for tNGS testing. Sputum tNGS testing indicated mixed infection with Acinetobacter baumannii, Stenotrophomonas maltophilia, Klebsiella pneumoniae, Streptococcus pneumoniae, Aspergillus fumigatus, Aspergillus flavus, and COVID-19. Following adjustment of the antimicrobial regimen based on pathogenetic findings, he developed empyema complicated by pneumothorax. A chest tube was inserted, resulting in improvement of empyema and pneumothorax symptoms. Bacterial, fungal, and Mycobacterium tuberculosis cultures of the pleural effusion were all negative. Further tNGS analysis of the pleural effusion revealed a mixed infection with Mycobacterium tuberculosis and Aspergillus fumigatus. The patient refused further treatment and died 5 days after discharge. CONCLUSIONS Diagnosis of tuberculous empyema and fungal empyema is challenging and the prognosis is poor. In patients with malignant tumors, particularly those receiving immunotherapy, the possibility of Mycobacterium tuberculosis infection and fungal infections should be fully considered when infections occur, and early diagnosis and treatment are essential.

BACKGROUND Actinomycosis is an uncommon bacterial infection caused by the commensal organism Actinomyces spp., which can become pathogenic upon tissue injury or disruption of the mucosal barrier. While primarily recognized for its cervicofacial and abdominopelvic presentations, actinomycosis can also involve the gallbladder and potentially the pancreas, posing diagnostic and therapeutic challenges due to its ability to mimic other conditions. CASE REPORT We present the case of a 26-year-old woman with a history of recurrent acute pancreatitis and cholecystitis, followed by the development of thrombotic microangiopathy. An atypical hemolytic-uremic syndrome panel revealed gene mutations previously associated with the onset of this condition. Computed tomography, endoscopic ultrasound, and abdominal ultrasound revealed focal enlargement of secondary ducts in the head of the pancreas. While no gallbladder wall thickening was observed, imaging demonstrated a substantial amount of heterogeneous, predominantly hypoechoic and amorphous content within the gallbladder, suggestive of biliary sludge. The patient subsequently underwent cholecystectomy, and histopathological examination of the gallbladder specimen confirmed the presence of Actinomyces spp., observed as sulfur granules and Gram-stained bacilli, as well as varying degrees of mucosal hyperplasia, transmural fibrosis, xanthomatous histiocytes, and Rokitansky-Aschoff sinuses. CONCLUSIONS Following the procedure and appropriate antimicrobial therapy, the patient experienced resolution of the infection, improvement in overall health status, and no new episodes of pancreatitis during the following year. We propose that underlying CFHR3 and CFI gene mutations and initial systemic inflammation precipitated the thrombotic microangiopathy-atypical hemolytic uremic syndrome, and facilitated Actinomyces colonization of the compromised gallbladder, ultimately leading to chronic gallbladder disease and recurrence.

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.