American Journal of Case Reports最新文献

BACKGROUND Immune-checkpoint inhibitors (ICIs) targeting programmed cell death-1 (PD-1), its ligand PD-L1, and cytotoxic T lymphocyte antigen-4 (CTLA-4) have revolutionized the treatment landscape of non-small cell lung cancer (NSCLC). These agents restore antitumor immunity by reactivating suppressed T cells. Although PD-L1 expression is widely used as a predictive biomarker, responses to ICIs can occur even in tumors lacking PD-L1 expression, underscoring the complexity of the tumor immune microenvironment. Ongoing research on the tumor microenvironment aims to achieve a better understanding of cancer progression mechanisms and to improve the assessment of therapeutic efficacy. CASE REPORT We present an 80-year-old man with advanced NSCLC, without any remarkable past medical history, clinically staged as IVB (cT4N3M1c), and demonstrating a PD-L1 tumor proportion score (TPS) of less than 1%. Despite this, he exhibited an excellent response to combination therapy with anti-PD-1, anti-CTLA-4 monoclonal antibodies, and cytotoxic chemotherapy during hospitalization, with manageable adverse events. Notably, pathological analysis revealed marked infiltration of CD3-positive tumor-infiltrating lymphocytes (TILs), averaging 1100/mm². CD4- and CD8-positive TILs were present in equal numbers, suggesting a balanced population of helper and cytotoxic T cells. The patient received a total of 24 cycles of immunotherapy before disease progression was confirmed. CONCLUSIONS This case highlights a striking dissociation between TIL density and PD-L1 expression, suggesting that CD3-positive TILs may reflect underlying immune activity not captured by PD-L1 status alone. Our findings emphasize the need to further explore TIL profiling as a complementary biomarker, particularly in patients treated with anti-PD-1/anti-CTLA-4-containing regimens.

BACKGROUND Occult foreign body residue after penetrating buttock injury is rare. When it progresses to a suprasphincteric high anal fistula, diagnosis and treatment are challenging because early imaging findings may be subtle and symptoms are often non-specific. This report shows the importance of obtaining a detailed trauma history, careful review of serial imaging, and sphincter-sparing surgery in such cases. CASE REPORT A 34-year-old man sustained a penetrating buttock injury from woody brambles after falling from a height. Initial X-ray results were normal, and he underwent simple debridement and suturing. Over the next 18 months, he developed recurrent perianal abscesses that were repeatedly drained at 2 centers, with persistently elevated inflammatory markers and a non-healing wound, but the occult wooden foreign body was missed on early computed tomography (CT) and magnetic resonance imaging (MRI). On presentation to our center, he had perianal pain, purulent discharge, and liquid fecal incontinence. Pelvic MRI showed a left ischiorectal fossa abscess with a central low-signal focus suggestive of a retained foreign body, and a high anal fistula tract. Trans-sphincteric surgery was performed, 5 bramble fragments were removed, and the fistula tract was adequately drained while preserving the sphincter. The wound healed completely within 2 months and no recurrence or incontinence was observed at 6-month follow-up. CONCLUSIONS In patients with a history of penetrating buttock trauma and recurrent perianal infection or non-healing wounds, the possibility of an occult retained foreign body causing a high anal fistula should be considered. Multi-modal imaging, particularly MRI, and a sphincter-sparing trans-sphincteric approach are essential to achieve complete foreign body removal, control infection, and preserve anal function.

BACKGROUND Device embolization is a rare complication of percutaneous closure of paravalvular leaks after transcatheter, surgical aortic, or mitral valve replacement. Management typically involves surgical or endovascular intervention. However, the optimal management of asymptomatic patients with a delayed diagnosis of device embolization remains unclear, as evidence for watchful waiting and/or delayed intervention is limited. CASE REPORT We present a case of 73-year-old man who had undergone endovascular closure of a paravalvular leak following transcatheter aortic valve replacement. The patient experienced device failure and recurrence of aortic stenosis symptoms, and during the redo surgical aortic valve replacement, the occluded device was discovered to have embolized to the left external iliac artery. He initially declined immediate intervention and has remained asymptomatic 1 year after diagnosis. CONCLUSIONS This case highlights the need for individualized management strategies of device embolization. While endovascular or surgical intervention remains the standard approach, watchful waiting may be appropriate in selected asymptomatic patients with delayed diagnosis of peripheral device embolization.

BACKGROUND Gastrointestinal stromal tumors (GISTs) are the most common mesenchymal neoplasms of the gastrointestinal tract. Their presentation varies from bleeding to perforation, but they rarely manifest as gastroduodenal intussusception or obstructive jaundice. We report a rare case of a gastric GIST that caused intussusception leading to gastric outlet obstruction with biliary obstruction and acute pancreatitis. CASE REPORT A 71-year-old woman presented with melena, vomiting, and generalized weakness. Evaluation revealed severe anemia and elevated liver and pancreatic enzymes. Imaging and endoscopy identified a large polypoid mass in the gastric antrum prolapsing into the duodenum. Magnetic resonance cholangiopancreatography showed a dilated common bile duct but no choledocholithiasis. She was advised to undergo surgery, during which a gastroduodenal intussusception compressing the ampulla was identified. The mass was excised via gastrotomy with wide margins. Postoperative recovery was uneventful. Histopathology confirmed a low-grade (G1) gastric GIST (pT3, CD117/Discovered On GIST-1 [DOG1]-positive, Ki-67 ~3%) with clear margins. CONCLUSIONS Gastroduodenal intussusception is a rare condition in adults. Fewer than 50 cases have been reported, and more than half were attributed to GISTs. Obstructive jaundice or pancreatitis due to external ampullary compression is uncommon. Imaging modalities such as computed tomography and magnetic resonance imaging are essential for diagnosis. Surgical excision remains the mainstay of management. This case highlights a rare, complex presentation of gastroduodenal intussusception with biliary obstruction due to a gastric GIST.

BACKGROUND Histiocytic sarcoma is a rare and aggressive hematopoietic malignancy, characterized by tumor cells exhibiting features of histiocytes or dendritic cells. Extranodal involvement, particularly in the gastrointestinal tract, skin, and soft tissues, is common and often associated with aggressive clinical behavior and poor outcomes. Due to its rarity, no standardized treatment has been established. Current therapeutic approaches are generally adapted from aggressive non-Hodgkin lymphoma protocols, typically involving multi-agent chemotherapy. However, therapeutic responses are often suboptimal, with frequent relapses. In selected cases, hematopoietic stem cell transplantation has been employed following intensive chemotherapy for disease control. CASE REPORT A 44-year-old woman presented with pancytopenia and was initially diagnosed with immune thrombocytopenia. After splenectomy for refractory disease, histopathlogic examination of spleen confirmed histiocytic sarcoma. She received 6 cycles of chemotherapy with cyclophosphamide, doxorubicin (hydroxydaunorubicin), vincristine (Oncovin), etoposide, and prednisone. Despite an initial response, the disease progressed. She underwent high-dose chemotherapy followed by autologous hematopoietic stem cell transplantation, but ultimately died due to disease relapse. CONCLUSIONS This case demonstrates the highly aggressive course and poor prognosis of histiocytic sarcoma, even after intensive chemotherapy and autologous stem cell transplanatation. Given the highly aggressive nature of histiocytic sarcoma and its generally poor prognosis, allogeneic stem cell transplantation may offer a more effective strategy for achieving long-term remission, particularly in patients with relapsed or refractory disease. Large-scale studies are needed to establish treatment guidelines for histiocytic sarcoma.

BACKGROUND Wellens syndrome is considered as equivalent to ST elevation myocardial infarction and is highly specific for critical stenosis of the proximal left anterior descending (LAD) coronary artery. Prolonged QT interval is a risk factor for life threating arrhythmia characterized by a prolongation of the QT interval (QTc) longer than 440 ms corrected for heart rhythm on the ECG. It might be seen in patients suffering from acute myocardial infarction and constitutes a negative prognostic factor. These 2 ECG pathologies are rarely seen together. CASE REPORT A 75-year-old female patient presented with a non-ST elevation myocardial infarction. On admission, a normal sinus rhythm, with negative T waves in leads I, aVL, and V6, and QTc interval of 431 ms were observed. Coronary angiography revealed critical stenosis of the distal left main (LM) and proximal LAD coronary artery. Deep inverted T waves in the V2-V4 leads and QTc prolongation to 828 ms were observed in her ECG 2 days after coronary angiography. An intravascular ultrasound-guided percutaneous coronary intervention with orbital atherectomy and drug-eluting stent was performed. During the 14 days of hospital stay, a gradual reduction of the QT interval was observed. CONCLUSIONS These ECG changes may precede the clinical symptoms of the threatening LAD occlusion. Wellens syndrome and QT prolongation occurrence are associated with a higher risk of myocardial infarction complications, so emergency percutaneous coronary intervention should be performed as soon as possible.

BACKGROUND Fungal endocarditis constitutes up to 3% of diagnosed infective endocarditis cases, and Histoplasma spp. cause approximately 10% of fungal endocarditis cases. Histoplasma infection occurs through inhalation of spores typically found in contaminated soil. The clinical forms are variable, ranging from asymptomatic to fatal presentations. Asymptomatic individuals may develop reactivated disease years after the primary exposure. Most cases of histoplasmosis are confined to the pulmonary system. In rare instances, fungal elements disseminate to the heart valves, where destructive injury occurs. CASE REPORT A 59-year-old woman presented with vascular bypass occlusion, 2 months after aortobifemoral bypass for aortoiliac occlusive disease. She reported severe, unrelenting nausea and vomiting; a large aortic valve mass was detected on transthoracic echocardiography. Bacterial endocarditis was initially suspected, but cultures were unrevealing. Follow-up microbial cell-free DNA testing identified Histoplasma capsulatum and low-level Streptococcus mitis/oralis, prompting further evaluation for disseminated histoplasmosis. Bone marrow biopsy showed Histoplasma negativity according to polymerase chain reaction. The patient subsequently underwent aortic valve replacement, and pathology confirmed fungal elements consistent with H. capsulatum. Gram and acid-fast bacilli staining of the aortic valve yielded negative results. CONCLUSIONS Fungal endocarditis carries substantial morbidity and is challenging to diagnose due to its nonspecific presentation over time and the absence of standardized blood culture protocols. Treatment options include surgical valve replacement and targeted antifungal therapy. This case underscores the diagnostic difficulties associated with fungal endocarditis, particularly in the context of negative cultures and a history of travel to endemic areas, when microbial coinfection cannot be definitively excluded.

BACKGROUND Acupuncture-related infection can result from use of unsterilized or used needles, inadequate skin disinfection, or contact between needles and contaminated surfaces. Immunocompromised patients are at increased risk for opportunistic infection and further complications. We present the case of a Staphylococcus epidermidis infection related to acupuncture in a 69-year-old man on immunosuppressive therapy for multiple myeloma. CASE REPORT A 69-year-old man with acute-on-chronic left shoulder pain was evaluated at our Emergency Department after recent acupuncture treatments. He was undergoing chemotherapy for relapsed multiple myeloma, which developed 14 years after a stem cell transplant. Upon admission, a large collection of fluid was identified in his left shoulder bursa, prompting consultation with an orthopedic surgeon and the aspiration of fluid, which contained a total white blood cell count of 91 560 cells/µL and absolute neutrophil count of 85 150 cells/µL. Treatment with intravenous ceftazidime and vancomycin was initiated, followed by surgical irrigation and debridement. Infectious Disease specialists added metronidazole. Cultures of excess fluid from the surgical site grew S. epidermidis, which was linked to his acupuncture treatments. The patient was discharged home to complete antibiotic therapy but was readmitted due to vancomycin-related acute kidney injury, fluid overload, and heart failure exacerbation, requiring further care. CONCLUSIONS Our patient's septic arthritis, presumed to be secondary to acupuncture therapy, underscores the risks associated with alternative medicine practices as potential sources of serious infections in immunocompromised patients. It also highlights the importance of using a multidisciplinary approach to successfully manage complex cases involving infectious and noninfectious complications.

BACKGROUND Hungry bone syndrome (HBS) is a severe, rapid, profound, prolonged, and sometimes fatal condition characterized by hypocalcemia, exacerbated by suppressed parathyroid hormone levels following parathyroidectomy in severe primary hyperparathyroidism (PHPT). PHPT is reported to affect 1% of the general population and its prevalence is increasing with age, with a prevalence rate of 2% among people over age 55 years. More than 80% of all cases of primary hyperparathyroidism are asymptomatic for most of the disease duration. CASE REPORT We described a rare case of primary hyperparathyroidism with brown tumors complicated by severe HBS after combined parathyroid and thyroid surgery. To the best of our knowledge, this is one of the first case reports ever published that presented with such a low level of calcium. Our patient required unprecedented amounts of calcium in the postoperative treatment during her hospital stay. The cumulative dose of intravenous calcium was 17 112 mg of elemental calcium. In this review we present signs and symptoms of HBS and discuss treatment and prevention methods of this syndrome. CONCLUSIONS When HBS occurs, hospitalization and aggressive intravenous calcium infusion should be continued until hypocalcemia resolves to prevent neuromuscular and cardiac complications. It is also essential to achieve normal magnesium levels. Currently, there are no specific treatment guidelines for HBS. This and similar case reports demonstrate appropriate diagnostic and therapeutic approaches and provides valuable guidance for clinicians. HBS can be prevented by early identification of risk factors and the use of calcitriol and bisphosphonates.

BACKGROUND Mesenteric hemolymphangioma is an exceedingly rare benign vascular malformation, with lesions at the mesenteric root being particularly uncommon. Preoperative diagnosis is challenging due to nonspecific clinical and radiological features. This study presents the first pediatric case of hemolymphangioma located at the mesenteric root and reviews the literature to elucidate its clinical characteristics and management. CASE REPORT A 15-year-old boy presented with a 2-year history of recurrent abdominal pain. The patient had no history of abdominal trauma or prior surgery. Preoperative computed tomography (CT) revealed a large, spindle-shaped, hypodense retroperitoneal mass (10.3×3.5×9.3 cm) near the duodenum and pancreatic head, with localized vascular enhancement. Intraoperatively, the tumor was located at the mesenteric root, adherent to the duodenum and pancreas, and caused localized small intestinal lymphatic dilation with chylous leakage. Complete surgical resection was achieved. Histopathological examination confirmed the diagnosis of hemolymphangioma, with immunohistochemical staining positive for D2-40, CD31, and CD34. The postoperative course was uneventful, and a follow-up CT scan at 2 weeks showed no evidence of recurrence. CONCLUSIONS This is the first reported case of pediatric hemolymphangioma at the mesenteric root. Complete surgical excision remains the cornerstone of treatment to prevent recurrence, especially given the high recurrence rates associated with partial resection. Diagnosis relies on histopathology and immunohistochemistry, as clinical and imaging findings are often nonspecific. This report highlights the importance of considering this rare entity in the differential diagnosis of mesenteric masses in children and adolescents.