AME Case Reports最新文献

Background: Acute transverse myelitis (ATM) is a pathological heterogenous inflammatory condition which causes acute or subacute spinal cord dysfunction. Diagnosis is challenging as the absence of magnetic resonance imaging (MRI) spinal cord lesions do no exclude this as a differential when patients present with well-defined dermatomal paraesthesia or myotome weakness. In approximately 40% of patient, there is a lack of MRI cord abnormalities. In addition, there are numerous of causes of ATM which contributes to diagnostic difficulty. The aim of this report is to highlight the challenges of diagnosing ATM, especially in the case of syphilis and cytomegalovirus.

Case description: A 56-year-old man with a background of high-risk promiscuous behaviour, presents with a symmetrical hand and feet desquamating rash with associated lumbago. Subsequently, he develops sensory impairment in his truncal region. Cerebrospinal fluid (CSF) findings revealed lymphocytosis and elevated protein however, MRI head and whole spine were normal. Together, the patient was diagnosed with ATM. Serology was positive for syphilis [Treponema pallidum haemagglutination assay (TPHA) and rapid plasma reagin (RPR)] as well as cytomegalovirus (CMV) immunoglobulin M (IgM) and immunoglobulin G (IgG), while human immunodeficiency virus (HIV) was negative. On first inspection, the most obvious diagnosis was deemed to be syphilis given the history and presentation with concurrent development of neurosyphilis. However, concurrent CMV IgM positivity obscures the underlying aetiology. He was treated with ceftriaxone and corticosteroids for syphilis, and doxycycline for concomitant chlamydia. At 4-month follow-up, CSF protein had improved and lymphocytosis resolved, although CMV IgM remained positive. His sensory deficits gradually improved, with complete recovery after 6 months, without antiviral therapy.

Conclusions: This case illustrates the diagnostic challenges of ATM when multiple infections coexist. While neurosyphilis was initially suspected, normal MRI findings and the persistence of CMV IgM suggest CMV myelitis as the more likely aetiology. Clinicians should consider CMV in the differential diagnosis of ATM even in immunocompetent patients, particularly when serology suggests recent infection but CSF polymerase chain reaction (PCR) and MRI findings are negative.

Background: Acute empyema presents a significant mortality risk in advanced-age populations, with rates reaching up to 20% in patients over 80 years of age. While initial treatment typically involves antibiotics and thoracic drainage, surgical intervention is often necessary in stage II empyema due to multiloculation and inadequate drainage. The renal (urea), age, fluid purulence, infection source, dietary (albumin) (RAPID) score has been proposed as both a prognostic tool and a potential surgical indicator, but its role in high-risk patients remains uncertain.

Case description: An 85-year-old male nursing home resident with an Eastern Cooperative Oncology Group performance status of 3 and comorbidities including type 2 diabetes, chronic kidney disease stage 3b, and cardiovascular history presented with fever and cough. Chest computed tomography revealed encapsulated pleural effusion with multiloculation, consistent with acute stage II empyema secondary to aspiration pneumonia. Initial antibiotic therapy and thoracic drainage were ineffective. Despite high-risk conditions and a RAPID score of 6, semi-elective two-port video-assisted thoracoscopic surgery (VATS) decortication was performed. Postoperative recovery was uneventful, and the patient was discharged on day 13 without complications.

Conclusions: This case highlights that VATS can be a viable and safe treatment option for acute empyema, even in high-risk older adults with a high RAPID score, provided clinical stability and appropriate disease staging.

Background: Primary pulmonary myxoid sarcoma (PPMS) is an exceedingly rare low-grade malignant sarcoma characterized by the presence of EWSR1 rearrangement and EWSR1::CREB1 fusion, which serve as critical diagnostic markers. Due to its nonspecific symptoms, PPMS is often detected incidentally, complicating its differentiation from other pulmonary neoplasms.

Case description: We report a case of a 52-year-old female patient who was admitted in May 2024 after a nodule was incidentally found in the right middle lobe during a routine health examination. Imaging studies revealed a solid nodule with mildly increased radiotracer uptake. The patient declined a biopsy and subsequently underwent uniportal video-assisted thoracoscopic surgery (UVATS) for right middle lobectomy. Intraoperative findings indicated a spherical nodule with significant adherence to the middle lobe vein. The frozen section analysis suggested a likely benign lesion, leading to the omission of lymph node dissection. Postoperative pathological examination confirmed the diagnosis of PPMS with EWSR1::CREB1 fusion, supported by immunohistochemical and fluorescence in situ hybridization analyses. The patient recovered uneventfully, with no abnormalities observed at the six-month follow-up.

Conclusions: PPMS is an extremely rare low-grade sarcoma with significant diagnostic implications due to its genetic characteristics. Surgical resection remains the primary treatment modality, though the potential for local recurrence or metastasis necessitates ongoing monitoring and further research to establish standardized treatment protocols and understand its biological behavior.

Background: Chronic nonbacterial osteomyelitis (CNO) is a rare inflammatory disorder that commonly causes bone destruction in the metaphysis of long bones, pelvic and vertebral bones, the clavicle, and the mandible. CNO is often misdiagnosed and underreported. This presentation of debilitating back pain caused by CNO showcases diagnostic challenges and potential for novel therapeutic treatment.

Case description: A 14-year-old male with Crohn's disease presented with acute, worsening lower back pain and two months of hematochezia after recently stopping infliximab. Physical examination showed sacral tenderness, diffuse erythematous lesions, normal strength and no focal neurological deficits. A T2 hyperintense mass at S4 on magnetic resonance imaging (MRI) was identified. Skeletal survey was negative; however, repeat MRI indicated phlegmon and epidural abscesses suggestive of osteomyelitis. Initial treatments with intravenous cefazolin and oral minocycline were ineffective, leading to interventional radiology (IR) guided S1 biopsy that revealed fibrous tissue without signs of infection. A left L5-S1 laminotomy and microdiscectomy were performed with surgical pathology of the L5-S1 disc showing focal acute inflammation with no evidence of microorganisms, leading to a diagnosis of CNO. Despite surgery, he experienced worsening pain and gait difficulties. After starting Ustekinumab for Crohn's disease, his back pain and mobility significantly improved, and he returned to school. Follow-up MRI showed improvement in the epidural swelling, decreased enhancement of the lumbar and sacral canal extending from L4-S3, and significantly decreased osseous enhancement at S1.

Conclusions: This case illustrates an unusual manifestation of CNO with an S1 vertebral lesion. The initial MRI findings and failed treatment with antibiotics resulted in a diagnostic challenge leading to an invasive workup involving multiple biopsies to rule out infection and malignancy and a subsequent L5 laminotomy and microdiscectomy. The timing of symptom onset following cessation of infliximab suggests a Crohn's flare likely triggered CNO. The soft tissue abnormalities noted on imaging were likely secondary inflammatory changes associated with the vertebral lesion. The resolution of back pain with ustekinumab is in line with other cases of successful resolution of CNO with biologic agents and supports the theory of shared pathophysiology in CNO and inflammatory bowel disease (IBD).

Background: Idiopathic esophageal perforation complicated by thyroid abscess formation constitutes a rare and severe clinical emergency. The precise pathogenesis of this condition remains elusive, and its potential for recurrence necessitates further investigation to establish evidence-based, personalized therapeutic protocols.

Case description: We present a case of a 41-year-old female who initially presented with a spontaneous cervical esophageal perforation associated with an ipsilateral thyroid abscess. The patient underwent prompt surgical intervention consisting of meticulous debridement and drainage, facilitated by intraoperative neuromonitoring to preserve recurrent laryngeal nerve function and thyroid integrity. The postoperative course was uneventful, and the patient was discharged with preserved thyroid function. However, a recurrence manifested 10 months postoperatively. In light of the well-localized abscess and the patient's hemodynamically stable condition, a conservative management strategy was instituted. This approach, comprising targeted antimicrobial therapy and vigilant clinical surveillance, resulted in complete resolution without the need for further surgical intervention.

Conclusions: This case provides substantive clinical insights for formulating individualized management strategies for idiopathic esophageal perforation. A review of the extant literature suggests that underlying anatomical anomalies, such as congenital weakness or diverticula, may predispose individuals to recurrent episodes. This underscores the imperative of tailoring treatment-ranging from aggressive surgical intervention to meticulous conservative management-based on a comprehensive assessment of infection severity, anatomical involvement, and the patient's overall clinical status. Further multicentric studies are warranted to elucidate the etiopathogenesis and to refine risk-stratified treatment algorithms for this uncommon but potentially life-threatening condition.

Background: Pancreatic pseudocysts (PPCs), common sequelae of pancreatitis, can lead to life-threatening complications. Hemorrhage from a PPC eroding into adjacent structures is a critical emergency with high mortality. However, a case involving simultaneous erosion into the stomach, diaphragm, and splenic artery, causing massive dual-compartment hemorrhage (gastrointestinal and thoracic), is exceptionally rare and presents a profound challenge for diagnosis and management, necessitating urgent, coordinated multidisciplinary intervention.

Case description: We report the case of a 56-year-old male with a history of chronic alcohol abuse and recurrent pancreatitis who presented with hematemesis, abdominal pain, and hemorrhagic shock. An urgent contrast-enhanced computed tomography (CT) scan and subsequent angiography revealed a large, complex PPC in the pancreatic tail. The pseudocyst had eroded through the posterior gastric wall, the left hemidiaphragm, and a branch of the splenic artery, causing active extravasation. This complex erosion resulted in both massive upper gastrointestinal bleeding and a large left-sided hemothorax. The patient was managed with a staged, multidisciplinary approach. Immediate resuscitation was followed by emergency selective coil embolization of the bleeding arterial branch, which successfully achieved initial hemostasis. After stabilization, he underwent a definitive open surgical procedure on hospital day three, which included distal pancreatectomy, splenectomy, complete pseudocyst excision, and primary repair of both the gastric and diaphragmatic perforations. Integrated throughout his care, the case management nursing model (CMNM) was pivotal in coordinating communication, ensuring adherence to perioperative protocols, and facilitating patient education, particularly on alcohol cessation. Despite a postoperative course complicated by a managed pulmonary infection and transient ileus, the patient recovered well without a pancreatic fistula and was discharged on postoperative day (POD) 17. He remained asymptomatic at his 3-month follow-up.

Conclusions: This case highlights the successful management of a catastrophic PPC complication through a strategic hybrid approach combining emergency endovascular embolization and definitive surgery. The integration of the CMNM proved invaluable for navigating the complex perioperative pathway, enhancing multidisciplinary collaboration, and ultimately contributing to a favorable outcome. This integrated model warrants further consideration in the management of complex surgical emergencies.

Background: Intraoperative hyperkalemia from necrotic tumor cells is a rare but serious complication of neoadjuvant chemotherapy (NAC) for liver tumors. Herein, we report a case of acute hyperkalemia that occurred intraoperatively during liver resection in a patient with tumor bleeding induced by preoperative chemotherapy.

Case description: The patient was a 68-year-old man, 170 cm in height and weighing 62 kg, with a history of hepatitis C. Contrast-enhanced computed tomography revealed a massive tumor measuring 16 cm in diameter in the right lobe of the liver. The patient received three courses of preoperative chemotherapy with atezolizumab and bevacizumab. Due to bleeding from the tumor, early surgical resection was scheduled. Preoperative laboratory evaluations showed normal renal function and potassium levels. During intraoperative manipulation, including ligation of feeding vessels and compression of the liver, arterial blood gas analysis showed a sudden rise in serum potassium from 5.0 to 6.3 mmol/L. Glucose-insulin (GI) therapy was administered, and the Pringle maneuver (PM) was avoided to limit further potassium release. By the end of the procedure, potassium level had decreased to 4.6 mmol/L.

Conclusions: The potential for hyperkalemia due to potassium release from necrotic tumor cells should be considered in patients undergoing liver resection after NAC. Careful intraoperative monitoring, along with appropriate surgical and anesthetic strategies, is crucial to prevent fatal arrhythmias.

Background: Sodium-glucose cotransporter-2 inhibitors (SGLT2is) are associated with euglycemic diabetic ketoacidosis (EDKA), particularly in the perioperative setting. This report highlights EDKA complicated by postoperative gastrointestinal (GI) dysfunction following lung surgery.

Case description: A 42-year-old woman with type 2 diabetes managed with metformin and empagliflozin (SGLT2i) underwent video-assisted thoracoscopic right upper lobectomy for suspected early lung cancer. Empagliflozin was continued preoperatively. Postoperative day 2, she developed acute dyspnea, right lower abdominal pain (despite normal bowel movements), and tachycardia. Initial arterial blood gas (ABG) revealed severe metabolic acidosis (pH 7.022, HCO₃ ^- 4.9 mmol/L, base excess -24.4 mmol/L) without significant hyperglycemia (fasting glucose 6.23 mmol/L). Administration of 500 mL sodium bicarbonate yielded minimal improvement (pH 7.153). Abdominal computed tomography (CT) confirmed significant GI distension. Urinalysis on postoperative day 3 showed marked ketonuria (80 mg/dL) and glycosuria (2,000 mg/dL), confirming EDKA (β-hydroxybutyrate 8.11 mmol/L). Treatment involved intravenous insulin infusion (added to 5% glucose-saline with potassium), fluid resuscitation, and nutritional support, leading to resolution of symptoms and acidosis by postoperative day 5.

Conclusions: Postoperative GI distension in SGLT2i users may signal impending EDKA. Sodium bicarbonate monotherapy provided limited benefit. Successful management requires early recognition, cessation of SGLT2i, adequate caloric intake via dextrose-containing fluids, and insulin to suppress ketogenesis. Preoperative discontinuation of SGLT2i 3-4 days prior to surgery is strongly recommended.

Background: Euglycemic diabetic ketoacidosis (eDKA) is associated with sodium-glucose co-transporter 2 inhibitor (SGLT2i) use in patients with type 2 diabetes (T2D) who are ketosis-prone because of factors such as acute illness and surgery. This report presents a case of eDKA that was admitted to the stepdown medical unit at an academic tertiary hospital following laparoscopic sleeve gastrectomy (LSG) and compares it with previous cases in the literature. The aim of this report is to describe the characteristics of eDKA presentation in LSG and identify measures to avoid such a preventable condition.

Case description: A 62-year-old woman with T2D on insulin and severe obesity with an initial body mass index of 55 kg/m² underwent an uneventful LGS and was discharged on empagliflozin and metformin for glycemic control. She returned to hospital with nausea, vomiting, and an inability to tolerate oral intake and was found to have acute kidney injury, high anion gap metabolic acidosis, ketonuria, and glycosuria. However, her glucose level was normal, which prompted a diagnosis of eDKA after excluding surgical complications. She was discharged safely after she was placed back on an insulin-only regimen.

Conclusions: eDKA in patients who have undergone bariatric surgery may present with similar symptoms to post-bariatric surgical complications. This case emphasizes the need for clearer perioperative guidance.

Background: Neuropsychiatric systemic lupus erythematosus (NPSLE) represents a severe and heterogeneous manifestation of systemic lupus erythematosus (SLE), occasionally progressing to catastrophic outcomes. Although rare, fulminant forms with rapid neurological decline and brain death are exceptionally uncommon.

Case description: We report a 21-year-old woman with an 8-year history of SLE (articular, cutaneous, renal, and hematologic involvement) and hypothyroidism, who presented with acute neuropsychiatric symptoms and rapidly deteriorated to coma and brain death in less than 72 hours. Initial computed tomography (CT) was unremarkable. Laboratory results showed leukocytosis, negative anti-double-stranded DNA (dsDNA) antibodies, and normal complement (C3 and C4). Cerebrospinal fluid (CSF) revealed mild pleocytosis without infection. Despite intravenous methylprednisolone and multiple anticonvulsants, she developed refractory status epilepticus. Cranial CT angiography demonstrated severe cerebral and cerebellar edema, absent posterior circulation, and partial anterior circulation opacification; no definite hemorrhage was observed. Electroencephalogram (EEG) showed an isoelectric tracing after 24 hours off sedation, and brain death was confirmed in June 2025.

Conclusions: The clinical course was consistent with fulminant cerebral involvement, possibly related to lupus vasculitis, though histologic confirmation was not possible. This case highlights that NPSLE may progress with extreme rapidity, underlining the importance of early suspicion, aggressive immunosuppression, and advanced imaging techniques, such as vessel-wall magnetic resonance imaging (MRI), which may improve timely diagnosis and outcomes.