American Journal of Case Reports最新文献

BACKGROUND Tuberculosis involving the sternoclavicular joint is a rare manifestation of extrapulmonary tuberculosis. Involvement of the muscular and skeletal system in tuberculosis accounts for approximately 10% of extrapulmonary tuberculosis cases. Of these, only 1% to 2% involve infection of the sternoclavicular joint. The presentation of sternoclavicular joint tuberculosis is often insidious without systemic symptoms. Its diagnosis is very difficult because it mostly occurs with an atypical presentation, therefore requiring a multimodal diagnostic approach. Patients can require a combination of several tests, including imaging, polymerase chain reaction, microbiologic tests, histopathological tests, and acid-fast bacilli smear and culture to generate a reliable diagnosis. CASE REPORT We present a case of a 77-year-old woman originally from Tibet who had lived in India for several years and presented with localized pain and swelling over the left medial clavicle. Chest computed tomography demonstrated a lytic lesion involving the medial aspect of the left clavicle with a soft tissue mass. Biopsy of the soft tissue mass at the left clavicular head revealed granulomatous inflammation, and cultures confirmed the presence of acid-fast bacilli, Mycobacterium tuberculosis. The patient was successfully treated with a 12-month course of anti-tuberculous therapy. CONCLUSIONS Diagnosis of tuberculous involvement of the sternoclavicular joint is usually challenging and requires a multimodal diagnostic approach. A high index of suspicion is important, especially in patients with risk factors for tuberculosis exposure. Early diagnosis, early initiation, and appropriate duration of anti-tuberculous therapy can lead to successful treatment outcomes.

BACKGROUND Cardiac myxomas are the most common primary cardiac tumors in the general population but are exceedingly rare in orthotopic heart transplant recipients. Fewer than 5 cases have been reported. Their pathogenesis in the setting of chronic immunosuppression remains unclear. CASE REPORT A 57-year-old woman with end-stage heart failure due to coronary artery disease underwent an orthotopic heart transplant in August 2017 with use of the bicaval technique. Her postoperative course included immunosuppressive therapy with tacrolimus, mycophenolate mofetil, and prednisone, with 2 episodes of moderate rejection during the early post-transplant period. Early echocardiograms revealed no abnormalities. At a routine follow-up in May 2022, she was asymptomatic; however, transthoracic echocardiography detected a mobile left atrial mass attached to the interatrial septum. Transesophageal echocardiography confirmed a nodular lesion in the left atrium, measuring 1.6×2.4 cm, located in the region of the fossa ovalis. Cardiac computed tomography demonstrated a 2.5×1.6×1.5 cm contrast-enhancing lesion consistent with myxoma. The patient underwent surgical excision under extracorporeal circulation, including partial septal resection. Histopathology confirmed the diagnosis of myxoma. DNA profiling was inconclusive regarding donor or recipient origin. At 1-year follow-up, the patient remained asymptomatic, with preserved graft function and no recurrence. CONCLUSIONS This is the fourth reported case of myxoma developing in a transplanted heart. The case emphasizes the importance of vigilant long-term echocardiographic surveillance in heart transplant recipients and raises questions regarding the potential role of immunosuppression in benign cardiac tumorigenesis.

BACKGROUND Pleomorphic lipoma is a rare benign adipocytic tumor that can closely mimic, radiologically and histologically, malignant soft tissue neoplasms, particularly atypical lipomatous tumor or liposarcoma. Despite is benign nature, pleomorphic lipoma poses a diagnostic challenge because of its significant histologic overlap with malignant entities. Accurate diagnosis is essential to prevent unnecessary wide surgical resections or overtreatment. Recognition of its distinctive clinical, histologic, and immunohistochemical features is therefore critical. CASE REPORT A 68-year-old man presented with a progressively enlarging, painful mass in the left upper arm. Ultrasound demonstrated a lobulated subdermal soft tissue lesion measuring 4.7×1.0×2.5 cm with a small hypoechoic component, raising concern for an atypical lipomatous tumor. Surgical excision was performed for definitive diagnosis and symptom relief. Gross examination demonstrated a well-circumscribed, yellow, lobulated mass. Histopathologic evaluation showed a lipomatous lesion composed of atypical spindle cells with pleomorphism and multinucleation, intermixed with variably sized adipocytes within a fibrotic and myxoid stroma. Immunohistochemical studies were positive for CD34 and negative for MDM2 and CDK4, with loss of RB1 expression and a low Ki-67 proliferation index (<1%), confirming the diagnosis of pleomorphic lipoma. The postoperative course was uneventful, and the patient remained symptom-free on follow-up. CONCLUSIONS This case highlights the diagnostic challenges associated with pleomorphic lipoma due to its rarity and resemblance to malignant soft tissue tumors. It underscores the importance of thorough histopathologic evaluation and immunohistochemical profiling to establish an accurate diagnosis and avoid unnecessary aggressive treatment. Complete surgical excision remains essential for diagnosis and management.

BACKGROUND Systemic lupus erythematosus (SLE) commonly involves the kidneys, most often as lupus nephritis (LN), which affects up to 60% of patients who have SLE, and is a major cause of morbidity. IgA nephropathy (IgAN), the most common primary glomerulonephritis, follows a benign course but rarely coexists with SLE. This overlap creates a significant diagnostic dilemma because both conditions present with proteinuria, hematuria, and impaired kidney function. Whether this demonstrates a causal link or a simple coincidence remains debated. CASE REPORT We present the case of a young woman with diagnosed SLE, previously complicated by seizures, who presented with nephrotic-range proteinuria, edema, and hematuria, mimicking a LN flare. She met the EULAR/ACR criteria for SLE exclusive of renal involvement. However, renal biopsy revealed dominant mesangial IgA deposition without IgG or C1q, with an Oxford MEST-C score of M1E0S1T0C0, confirming IgAN. Early immunosuppression was administered, owing to the severity of the presentation. This resulted in marked improvement, with proteinuria decreasing significantly and stabilization of renal function on follow-up. CONCLUSIONS This case illustrates that severe IgAN can occur in patients with SLE and be clinically indistinguishable from an LN flare. Careful histopathological assessment, particularly immunofluorescence, is important to differentiate IgAN from classic LN, as management may differ. The aggressive presentation and favorable response to immunosuppression suggest that active SLE can exacerbate underlying IgAN. This co-occurrence raises questions about whether IgA-dominant nephritis in SLE is an incidental finding or is a linked pathogenesis.

BACKGROUND Gastrocolic fistula is a rare complication of partial gastrectomy that can present with weight loss, diarrhea, and feculent vomiting. Most cases documented in the literature have occurred following non-physiological anastomoses, such as Billroth II procedures. This report describes the case of a 60-year-old man with a gastrocolic fistula from the gastrectomy anastomosis to the transverse colon 14 years after a Billroth I (gastroduodenal) anastomosis. CASE REPORT A 60-year-old male patient presented with a 6-year history of recurrent diarrhea, which had worsened over the past 6 months and was accompanied by weight loss. Fourteen years prior, he had undergone a subtotal gastrectomy with Billroth I anastomosis for gastric perforation. Ultimately, meticulous combined gastroscopy and colonoscopy revealed the presence of a fistulous opening and colonic mucosa, establishing a diagnosis of gastrocolic fistula. Following comprehensive preoperative nutritional support, the patient underwent laparoscopic resection of the fistula tract and the affected gastric and colonic segments, with concomitant gastrointestinal reconstruction. Postoperatively, the patient recovered smoothly, with complete resolution of diarrhea symptoms and marked improvement in nutritional status. CONCLUSIONS This case highlights that gastrocolic fistula can present as an extremely late complication following gastrectomy, even after an anastomosis performed using the more physiologically approximating Billroth I technique. Clinicians should exercise heightened vigilance with patients with a history of gastric surgery (regardless of procedure type) presenting with unexplained chronic diarrhea. Proactive use of direct examination methods, such as endoscopy, is essential to prevent misdiagnosis of this rare yet curable condition.

BACKGROUND Rectovaginal fistulas (RVFs) typically present with vaginal flatus or fecal leakage, facilitating early diagnosis. However, atypical or occult presentations can obscure recognition. We report an unusual RVF manifesting as vaginal burning triggered by spicy food, resulting from capsaicin transfer through a fistulous tract obstructed by perineal coproliths - a rare mechanism that concealed classic symptoms. CASE REPORT A 52-year-old multiparous woman with prior pelvic surgeries experienced 2 years of vaginal burning exclusively after consuming spicy food, which was initially misdiagnosed as vaginitis. Symptoms worsened following a diarrheal episode. Physical examination revealed a 2-cm firm perineal mass. Pelvic MRI identified a low intersphincteric fistula (Parks classification), and ultrasound revealed calcification. Urinalysis showed microscopic hematuria (36.7 RBC/μL); other investigations were unremarkable. Surgical exploration exposed a 2-cm cavity between the rectum and vagina, obstructed by coproliths. After coprolith removal and seton placement, the patient's symptoms resolved. The postoperative course was uneventful, with complete symptom resolution by 2 weeks. Notably, the patient's recovery was achieved without stoma diversion, suggesting that a conservative staged approach may suffice for selected low-level, well-drained fistulas. A 1-year telephone follow-up with the patient confirmed sustained recovery, with normal bowel and urinary function. CONCLUSIONS This case highlights an exceptionally rare presentation of RVF with diet-triggered symptoms. Migration of capsaicin through an occult fistulous tract can cause isolated vaginal burning, while coprolith obstruction can mask classical signs and delay diagnosis. Recognition of such atypical, food-induced symptom patterns may guide earlier identification and individualized surgical management in patients with prior pelvic surgery.

BACKGROUND In adults, non-corrected truncus arteriosus type 1 with Eisenmenger syndrome is a rare congenital heart defect characterized by a single large artery from the heart that directs blood flow to the lungs, leading to irreversible pulmonary hypertension and a reversal of blood flow. This report describes the case of a 44-year-old woman with congenital Eisenmenger syndrome and unrepaired truncus arteriosus type 1 presenting with recurrent thromboembolic cerebral infarcts. CASE REPORT A 44-year-old woman presented with a 2-month history of recurrent neurological deficits, including aphasia, right-sided weakness, and vertigo. Magnetic resonance imaging (MRI) of the brain confirmed acute embolic infarcts, while computed tomography (CT) angiography ruled out large-vessel occlusion. Cardiac evaluation revealed a 15-mm perimembranous ventricular septal defect with a bidirectional shunt and a severely calcified truncal valve with moderate regurgitation. Imaging confirmed truncus arteriosus type 1, with the pulmonary arteries originating from the ascending aorta. The right ventricular systolic pressure was estimated at 114 mmHg, and baseline oxygen saturation (SpO₂) was 70-80%. Due to irreversible pulmonary vascular disease, she was not a candidate for surgical repair. A conservative management strategy was adopted, including dual antiplatelet therapy, sildenafil, macitentan, bisoprolol, and oxygen therapy. At the 6-month follow-up, the patient remained neurologically stable with her SpO₂ improved to 85-88%. CONCLUSIONS This case highlights the exceptional natural course of uncorrected truncus arteriosus in adulthood. It underscores that for patients with Eisenmenger physiology where surgical repair is not feasible, a multidisciplinary approach focusing on tailored antithrombotic and pulmonary vasodilator therapies is essential for stroke prevention and clinical stability.

BACKGROUND Hypercapnia is a known complication of laparoscopic surgery involving carbon dioxide (CO₂) pneumoperitoneum and is usually managed intraoperatively with ventilatory adjustments. However, delayed-onset postoperative hypercapnia is uncommon. Anemia impairs hemoglobin-mediated buffering and clearance of CO₂, potentially increasing vulnerability to postoperative respiratory acidosis. This report describes 2 cases of delayed-onset postoperative hypercapnia in anemic patients following total laparoscopic hysterectomy with CO₂ pneumoperitoneum, in the absence of subcutaneous emphysema. CASE REPORT Case 1: A 43-year-old woman with American Society of Anesthesiologists (ASA) physical status I and severe anemia (hemoglobin 7.3 g/dL) underwent laparoscopic hysterectomy. One unit of blood was transfused intraoperatively. Fifteen minutes after extubation, she experienced prolonged postoperative unresponsiveness. Arterial blood gas (ABG) showed significant respiratory acidosis with a partial pressure of arterial CO₂ (PaCO₂) of 88 mmHg. She needed reintubation and 2 hours of mechanical ventilation before full recovery. Case 2: A 45-year-old woman with ASA I and refractory anemia (hemoglobin 8.1 g/dL) underwent laparoscopic hysterectomy with intraoperative blood transfusion. Despite intraoperative hyperventilation for rising end-tidal CO₂ (>75 mmHg), she developed extended unresponsiveness following extubation. ABG detected severe hypercapnia (PaCO₂ 88 mmHg). She recovered using manual ventilation without reintubation. Neither patient showed signs of subcutaneous emphysema. CONCLUSIONS Delayed-onset hypercapnia can occur in anemic patients following laparoscopic surgery, despite standard ventilation protocols. Reduced hemoglobin-mediated CO₂ buffering, combined with ongoing CO₂ absorption after pneumoperitoneum may contribute to this presentation. Anemia should therefore be considered as a modifiable risk factor, and in high-risk patients, enhanced postoperative monitoring and individualized ventilation strategies should be considered.

BACKGROUND Nitrous oxide (N₂O) deactivates vitamin B12 and, when inhaled regularly, can lead to vitamin B12 deficiency, resulting in temporary or permanent peripheral neuropathy. This case report describes a young adult with N₂O-induced B12 deficiency, whose neurological symptoms were initially misattributed to a distracting traumatic injury from a motor vehicle collision (MVC). CASE REPORT We present a case of a previously healthy 24-year-old man who presented with low back pain and bilateral leg paresthesia that began 2 days after being a passenger in an MVC. There was no head trauma, loss of consciousness, or bowel or bladder dysfunction. Examination revealed an apraxic gait with a positive Romberg sign, while motor strength and sensation remained intact. Laboratory tests showed severely low vitamin B12 level. Diagnostic challenges included misattributing the symptoms to trauma from the MVC. MRI of the lumbar spine and CT scans of the head and spine were unremarkable. Follow-up questioning revealed recreational N₂O inhalation, which was sporadic over the preceding year and most recently occurred on the same night following the MVC. Intramuscular B12 provided rapid improvement in gait and paresthesias, thereby also offering support for a diagnosis of vitamin B12 deficiency secondary to N2O use rather than the initially presumed traumatic etiology. The patient was discharged within 24 hours without requiring admission. CONCLUSIONS This case underscores the importance of considering N₂O use in patients with unexplained neurological deficits, along with obtaining directed social history and serum B12 levels when this differential diagnosis is plausible. Early recognition, supplementation, and counseling can prevent lasting deficits.

BACKGROUND Upper-gastrointestinal (GI) bleeds refractory to endoscopic hemostasis can be treated with endovascular coil embolization. Transcatheter arterial embolization with endovascular coils is an effective and minimally invasive option with high success rates, and coil migration is a rare complication. This report describes the case of a 90-year-old woman with an upper-GI bleed managed with emergency transcatheter arterial embolization, presenting with an endovascular coil migration. CASE REPORT A 90-year-old woman presented to the emergency department (ED) with abdominal pain, dizziness, and diarrhea (day 0). Imaging revealed a duodenal bulb ulcer with a contained perforation, for which she underwent endovascular coil embolization on day 2. Thirteen days later, during an outpatient esophagogastroduodenoscopy (EGD), the endovascular coil was identified at the junction of the duodenal bulb and second portion of the duodenum. The endovascular coil was not removed due to risk of bleeding. At 6 weeks, she re-presented with mild symptoms and was managed conservatively. At 4 months, she remained asymptomatic and the endovascular coil had not passed. At 21-month follow-up, the endovascular coil remained unpassed, with intermittent symptoms, a finding that highlights the long-term management and challenges of this complication. CONCLUSIONS Due to the rarity of endovascular coil migration, there is no standard method for managing this complication. It is important to assess each patient individually, manage patient symptoms, and consult with interventional radiology and surgery as needed. This case shows that reporting such cases is essential to expand the limited literature, inform best practices, and guide clinicians faced with this rare complication.