American Journal of Case Reports最新文献

BACKGROUND Eosinophilic pancreatitis (EP), representing <1% of pancreatitis cases, poses significant diagnostic challenges due to its heterogeneous manifestations ranging from mild to life-threatening. The overlapping clinical symptoms and imaging features with other types of pancreatitis often make EP particularly difficult to differentiate. Although tissue demonstration of eosinophilic infiltration (>10/high-power field) after excluding secondary causes is diagnostic, the invasiveness of pancreatic biopsy and disease rarity lead to high misdiagnosis rates in clinical practice. The present case report includes detailed imaging (CT, MRCP, EUS) and histopathology findings, providing a visual guide for clinicians encountering similar cases. CASE REPORT We present the case of a 68-year-old Chinese man diagnosed with EP manifesting as recurrent pancreatitis episodes. His chief concern was recurrent severe abdominal pain of unknown etiology. During his first admission, an upper endoscopy was performed despite the absence of gastrointestinal symptoms and revealed eosinophilic gastroenteritis (EGE). One year later, upon recurrence of pancreatitis, endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA) of the pancreas was performed, confirming EP with approximately 15 eosinophils/high-power field. The patient's symptoms and pancreatic duct dilation resolved rapidly following corticosteroid therapy. The diagnosis was definitively established based on a composite framework comprising recurrent pancreatitis, persistent peripheral eosinophilia, the rigorous exclusion of secondary causes, and histological confirmation of eosinophilic infiltration via EUS-FNA. CONCLUSIONS Eosinophilic pancreatitis (EP) is a rare yet potentially severe acute pancreatitis subtype requiring early diagnosis. It can also be associated with subclinical eosinophilic gastroenteritis (EGE). Immunomodulatory therapy, especially glucocorticoids, is highly effective. Our findings support using endoscopy in cases of unexplained pancreatitis for a definitive histological diagnosis. Prompt diagnosis is crucial for initiating effective corticosteroid therapy, which can resolve symptoms and prevent disease progression.

BACKGROUND Marfan syndrome is a multisystemic, autosomal dominant connective tissue disorder caused by mutations in the fibrillin-1 gene. It is associated with life-threatening complications, especially aortic aneurysms and dissections, and has a global prevalence of 1 in 3000-5000 individuals. Although kidney transplantation is the standard treatment for end-stage renal disease (ESRD), deceased kidney donors with Marfan syndrome remain uncommon due to concerns about connective tissue fragility. Emerging reports, however, indicate that kidneys from such donors may be safely used. This report describes 2 successful kidney transplantations from a 39-year-old male donor with confirmed Marfan syndrome. CASE REPORT Case 1. The donor died from subarachnoid hemorrhage and had a history of aortic arch replacement for aortic dissection. Both kidneys were retrieved from the retroperitoneal space, prepared according to standard protocols, and stored in University of Wisconsin Store Protect Plus organ preservation solution under static cold storage at 4°C. Case 2. A 59-year-old man with ESRD secondary to diabetic nephropathy underwent his first kidney transplantation. He was hemodialyzed with residual diuresis. The right kidney was transplanted without complications, and the recipient achieved immediate and stable graft function. Case 3. The second recipient was a 34-year-old man with ESRD due to vesicoureteral reflux and obstructive nephropathy, also on hemodialysis with preserved diuresis. Left kidney transplantation proceeded uneventfully. He experienced delayed graft function, which resolved with appropriate management, resulting in satisfactory renal recovery. CONCLUSIONS These cases support the growing evidence that kidneys from donors with Marfan syndrome can function effectively after transplantation. Such donors may represent a valuable and underutilized resource, with the potential to expand the kidney donor pool safely.

BACKGROUND Hydatid disease, a parasitic zoonotic infection caused by Echinococcus granulosus, is characterized by the formation of cysts in various organs. Transmission occurs via the fecal-oral route. The disease is endemic in regions where livestock farming is common, including rural areas of the Mediterranean, Africa, the Middle East, and South America. The liver and lungs are the most commonly affected organs, with reported involvement rates of 50% to 70% and 20% to 30%, respectively. In rare cases, cardiac involvement occurs (0.5%-2%), most frequently affecting the left ventricle due to its rich blood supply. Cysts may occur in any cardiac location; however, pericardial involvement is particularly uncommon, representing approximately 7% of cardiac cases. CASE REPORT A 42-year-old man from an endemic region developed sudden chest discomfort, severe dyspnea, hypotension, and orthopnea. Clinical examination revealed findings consistent with cardiac tamponade and pulmonary edema. Imaging studies demonstrated a ruptured pericardial hydatid cyst; multiple floating daughter cysts were evident within the pericardial sac. The patient underwent cystectomy followed by mitral valve replacement and received postoperative albendazole therapy. Recovery was uneventful, and the patient remained asymptomatic at the 12-month follow-up. CONCLUSIONS Rupture of a pericardial hydatid cyst, although rare, should be considered in the differential diagnosis of cardiac tamponade and pulmonary edema, particularly in endemic regions. Early diagnosis and combined surgical and medical management are essential to achieve favorable outcomes.

BACKGROUND Hemangioma is the most common benign primary tumor of the spleen and is often detected incidentally. Laparoscopic-assisted microwave ablation (LMWA) has recently been introduced as a novel approach for managing splenic hemangioma. This report describes the case of a 67-year-old woman with an asymptomatic, progressively enlarging splenic hemangioma successfully treated with LMWA. CASE REPORT A 67-year-old woman was admitted for evaluation of a gradually enlarging splenic mass found during routine examination. She was asymptomatic, with normal tumor markers and a mildly elevated D-dimer level. Contrast-enhanced CT revealed a 6.0 cm hypervascular lesion in the upper pole of the spleen. After multidisciplinary discussion, LMWA was performed under direct laparoscopic visualization. The lesion was completely ablated, with a total operative time of 80 minutes, including 25 minutes of ablation. Estimated blood loss was minimal (20 mL). Postoperative pathology examination confirmed a splenic cavernous hemangioma. The postoperative course was uneventful, and she was discharged on day 5. Follow-up imaging showed reduction of the ablation zone to 5.4 cm, and laboratory results returned to normal. CONCLUSIONS This is the first report on the feasibility and safety of LMWA for treating splenic hemangioma. This approach effectively reduces intraoperative bleeding and postoperative hemolysis while preserving splenic function, offering a novel treatment option for splenic hemangioma.

BACKGROUND Cryoglobulinemia is a rare disease with a prevalence of <5 cases per 10 000. In many cases there is significant diagnostic delay, leading to years of morbidity. There are no randomized trials on non-infectious type II cryoglobulinemia, and choice of therapy is based on clinical expertise and observational data. Therapy based on the monoclonal anti-CD20 antibody rituximab in combination with glucocorticosteroids is the preferred choice in many centers. This strategy induces clinical remission in two-thirds of patients. However, one-third of the initially-responding patients experience relapse within the first year. As such, many patients will not achieve sustained remission. CASE REPORT We describe the clinical course of 2 patients with non-infectious type II cryoglobulinemia. They were initially treated according to standard clinical practice, without lasting symptom relief. A novel treatment strategy was attempted, targeting the underlying B-cell clone responsible for the production of disease-inducing monoclonal immunoglobulins. These protocols, initially developed for treatment of chronic cold agglutinin disease, contain rituximab in combination with fludarabine or bendamustine. Sustained clinical, immunological, and hematological remissions were achieved in both patients, and the treatment was well tolerated with no need for hospitalization or other supportive measures. CONCLUSIONS Treating patients with type II cryoglobulinemia using chemo-immune therapy in the same regimens as used in cold agglutinin disease could be considered as a therapeutic option. Based on our observations in the 2 patients described in this case series, this approach seems feasible and well tolerated. Deep sustained remissions may be possible.

BACKGROUND Gluteal augmentation has gained significant popularity worldwide, with a parallel increase in complications related to unregulated procedures. The use of permanent fillers by non-medical personnel poses serious risks and can complicate future surgical interventions. CASE REPORT We report the case of a 29-year-old woman seeking revision of a prior gluteal augmentation performed abroad with subcutaneous silicone implants. The patient desired greater upper-pole projection and improved contour. During liposuction of the lower back, an unexpected gel-like material was encountered throughout the subcutaneous tissue of the lower back, hips, and infra-gluteal fold, consistent with previously injected permanent filler. The procedure was immediately adapted: fat grafting was abandoned due to the risk of infection and poor graft viability, and thorough manual extraction and irrigation were performed. Implant exchange was completed successfully, with new biconvex silicone implants placed in intramuscular pockets. The postoperative course was uneventful, and at 3-month follow-up, the patient demonstrated improved contour and was satisfied with the outcome. CONCLUSIONS This case illustrates the need for accurate preoperative evaluation and the ability to adapt intraoperatively in response to unsafe prior procedures. It also highlights the growing concern over illegal filler use and reinforces the importance of regulation, patient education, and adherence to evidence-based surgical practices to ensure safety and satisfactory outcomes.

BACKGROUND Sarcomatoid squamous cell carcinoma is a rare and highly aggressive subtype of squamous cell carcinoma. Due to its highly invasive biological characteristics, the prognosis of sarcomatoid squamous cell carcinoma in the skin area is usually poor. CASE REPORT This study reports a rare case of a skin tumor involving a 48-year-old male patient with a diagnosis of sarcomatoid squamous cell carcinoma. The patient presented with multiple primary squamous cell carcinomas on both hands and a sarcomatoid tumor on the third finger of the right hand over a 2-year period. The patient exhibited chronic erythema, exudation, ulcers, and nail destruction. Imaging studies and pathological biopsies confirmed a diagnosis of moderately differentiated squamous cell carcinoma involving multiple digits. Despite undergoing surgical excision and photodynamic therapy, local recurrence was observed 3 months later, and the patient refused further treatment. He was subsequently lost to follow-up. CONCLUSIONS This case underscores the aggressiveness of multifocal digital sarcomatoid squamous cell carcinoma and the challenges in clinical diagnosis, highlighting the importance of early intervention to improve patient outcomes. This case also emphasizes that sarcomatoid squamous cell carcinoma of the digits is highly aggressive, prone to early recurrence despite initial surgery, and necessitates extensive resection and close follow-up from the outset.

BACKGROUND Eales' disease - a rare idiopathic retinal vasculitis typically affecting young adult men - is characterized by periphlebitis, retinal ischemia, and neovascularization. Vitreous hemorrhage and tractional retinal detachment often occur; timely diagnosis and individualized management remain critical. This report describes bilateral Eales' disease with an asymmetrical clinical course and complex treatment strategy incorporating vitrectomy, systemic immunosuppression, and targeted anti-vascular endothelial growth factor (anti-VEGF) therapy. CASE REPORT A 32-year-old White man without systemic illness, infectious exposure, or substance use exhibited left-eye vitreous hemorrhage. Best-corrected visual acuity (BCVA) was hand motion (≈2.3 logarithm of the minimum angle of resolution [logMAR]) in the left eye and 5/6 (≈0.08 logMAR) in the right eye. Ultrasonography and fluorescein angiography demonstrated vitreoretinal traction and peripheral retinal ischemia. After exclusion of infectious etiologies, the diagnosis was bilateral Eales' disease. We performed left-eye vitrectomy with cataract extraction, panretinal photocoagulation (PRP), and systemic immunosuppression with azathioprine, followed by methotrexate. One year later, right-eye disease progression required intravenous methylprednisolone, PRP, and preoperative intravitreal faricimab, followed by vitrectomy. Faricimab-mediated inhibition of vascular endothelial growth factor A (VEGF-A) and angiopoietin-2 (Ang-2) enhanced vascular stabilization. At final follow-up - 18 months after left-eye vitrectomy and 12 months after right-eye vitrectomy - BCVA was 0.00 logMAR (5/5) in both eyes. CONCLUSIONS Early vitrectomy with systemic immunosuppression and adjunctive anti-VEGF therapy may achieve excellent long-term visual outcomes in bilateral Eales' disease. Faricimab application highlights the utility of dual VEGF-A/Ang-2 pathway inhibition in surgical planning. Future studies will clarify anti-VEGF agent efficacies in this rare retinal vasculitis.

BACKGROUND Dalbavancin is a lipoglycopeptide antibiotic largely used to treat serious and complex bacterial infections, particularly those caused by gram-positive bacteria. Its main advantages include a prolonged half-life, which allows for once-weekly dosing, effectiveness against resistant pathogens, most notably, methicillin-resistant Staphylococcus aureus, and a good profile of tolerability and patient adherence. Herein we report the first documented case of a patient who developed palpable purpura on both lower limbs following treatment with dalbavancin, which was clinically and histologically diagnosed as leukocytoclastic vasculitis (LV). CASE REPORT A 66-year-old woman received dalbavancin due to an acute bacterial skin and skin structure infection (ABSSSI) of her right leg. The patient initially showed a favorable clinical response, with marked improvement of the infection. However, 3 weeks after the administration of dalbavancin, she developed a diffuse erythematous palpable purpura, predominantly affecting the lower limbs, and accompanied by mild arthralgia and generalized asthenia. To further investigate and confirm the underlying cause of these new symptoms, a skin biopsy was performed, revealing histopathological findings consistent with LV. Following the diagnosis, corticosteroid therapy was initiated, resulting in a significant symptomatic improvement. CONCLUSIONS To the best of our knowledge, this is the first published, histologically confirmed case report of LV developing after treatment with dalbavancin. Given the widespread use of dalbavancin for treating ABSSSIs, clinicians should be aware of the potential adverse effects, including LV, that may arise during its administration.

BACKGROUND Internal hernias through the pars flaccida of the lesser omentum are rare variants that pose unique diagnostic and therapeutic challenges, representing less than 1% of all internal hernias. Diagnosis is particularly challenging due to their nonspecific presentation and subtle radiological features. CASE REPORT We present a case of successful laparoscopic repair of a pars flaccida internal hernia in a 52-year-old woman with complex surgical history, including previous colorectal cancer surgery. We detail our systematic surgical approach and conducted a comprehensive literature review of laparoscopically managed pars flaccida hernias using the PubMed database. The patient presented with recurrent episodes of epigastric pain and nausea. Diagnostic laparoscopy revealed herniation of the small bowel through the pars flaccida anterior to the stomach, along with a secondary mesenteric defect. Both defects were successfully repaired laparoscopically using non-absorbable sutures. Key technical elements included strategic 5-port placement, systematic adhesiolysis, and meticulous reduction of the herniated bowel. The patient's symptoms resolved after repair. CONCLUSIONS Laparoscopic repair of pars flaccida hernias is feasible with appropriate technical expertise. Success depends on careful preoperative planning, strategic port placement, and thorough inspection for additional defects. This case highlights the importance of considering internal hernias in patients with intermittent abdominal pain following previous surgery, even when initial imaging is inconclusive.