Journal of Medical Case Reports最新文献

Introduction: Double pylorus, or acquired gastroduodenal fistula, is a rare endoscopic finding, reported in only 0.001-0.4% of upper gastrointestinal examinations. We describe a case of acute gastrointestinal bleeding associated with a double pylorus in an elderly patient.

Case presentation: A 78-year-old white woman recovering from a pelvic ring fracture was admitted to a rehabilitation unit and received prophylactic dalteparin. She had no prior history of peptic ulcer disease or gastritis. After 2 weeks, she developed acute weakness, abdominal discomfort, nausea, and vomiting, accompanied by a 2.9 g/dL drop in hemoglobin. Urgent upper endoscopy revealed a double pylorus, and biopsy confirmed Helicobacter pylori infection. We reviewed published clinical reports of double pylorus. Most patients were older adults, and abdominal pain or gastrointestinal bleeding were frequent presenting symptoms. Use of nonsteroidal anti-inflammatory drugs or corticosteroids and the presence of Helicobacter pylori infection were commonly reported among described cases, although the quality and completeness of available data varied.

Conclusion: Double pylorus is a rare but clinically relevant condition that may first be detected when complications such as gastrointestinal bleeding occur. Reported associations with H. pylori infection and nonsteroidal anti-inflammatory drug exposure represent observational trends rather than established causal relationships, as the evidence remains heterogeneous and limited. The condition likely develops in the setting of multifactorial impairment of gastroduodenal mucosal integrity, particularly in older or medically vulnerable individuals.

Background: Mammary metaplastic squamous cell carcinoma is an exceptionally rare and aggressive triple-negative breast cancer. Its propensity to mimic benign breast abscesses poses a significant diagnostic challenge, often leading to treatment delays.

Case presentation: A 41-year-old Asian woman presented with a rapidly enlarging right breast mass, initially misdiagnosed and treated as an abscess at a local hospital. Definitive biopsy revealed triple-negative mammary metaplastic squamous cell carcinoma. The patient received neoadjuvant chemotherapy with a nab-paclitaxel, epirubicin, cyclophosphamide regimen. After an initial partial response observed in the first five cycles, the tumor exhibited progressive disease following the sixth cycle, a phenomenon potentially indicative of acquired chemoresistance. The patient subsequently underwent a modified radical mastectomy. Given the high-risk features and inspired by the SYSUCC-001 trial, adjuvant dose-dense capecitabine was administered postoperatively. No recurrence was detected during follow-up.

Conclusion: This case highlights a classic diagnostic pitfall of mammary metaplastic squamous cell carcinoma masquerading as an abscess and illustrates a unique pattern of acquired resistance to a standard taxane-anthracycline neoadjuvant chemotherapy regimen. It underscores the need for heightened clinical vigilance. The potential benefit of adjuvant capecitabine for patients with high-risk mammary metaplastic squamous cell carcinoma observed here warrants further investigation.

Background: Spontaneous rupture of the uterus during the early weeks of gestation is rare. When it occurs, it is a life-threatening obstetric emergency. Most uterine ruptures happen in scarred uteri and during the late third trimester. This case is unique because it involves a spontaneous rupture in an unscarred uterus during the second trimester. Identified risk factors include antepartum hemorrhage in a previous pregnancy, history of retained placenta, prior manual vacuum aspiration for incomplete abortion, and use of herbal medicine. This case adds to the limited global literature indicating that spontaneous rupture of an unscarred uterus in the second trimester can occur owing to the combined effect of uncommon risk factors.

Case presentation: A 30-year-old ethnically Amhara, gravida IV, para II, and abortion I mother presented at Debre Tabor Comprehensive and Specialized Hospital with severe abdominal pain lasting 2 weeks. The patient reported using unspecified herbal medicine, a history of manual vacuum aspiration for incomplete abortion, retained placenta, and antepartum hemorrhage in a previous pregnancy. On physical examination, she appeared acutely ill, pale, and tachycardic. Her abdomen was distended and tender. Ultrasound results revealed two dead fetuses, one outside and one partially inside a ruptured uterus. A large intraabdominal fluid collection was also observed. An exploratory laparotomy confirmed a 10-cm transverse rupture of the uterine fundus with necrotic margins. No gross uterine congenital anomalies were detected during exploration. After stabilizing the patient, a hysterectomy with bilateral salpingectomy was performed. She received broad-spectrum antibiotics and recovered gradually with supportive care. Following psychological counseling, she was discharged after 1 week with a smooth postoperative course.

Conclusion: This case report demonstrates that spontaneous rupture of the uterus in the second trimester can occur even in an unscarred uterus. Unusual risk factors such as a history of antepartum hemorrhage, retained placenta, manual vacuum aspiration for incomplete abortion, and use of herbal medicine might have acted together to contribute to uterine rupture in an already overdistended uterus. Clinicians should have a high index of suspicion for uterine rupture in pregnant women presenting with unusual abdominal pain during pregnancy, even in the absence of classical risk factors.

Background: Primary peritoneal rhabdomyosarcoma is an exceedingly rare soft tissue malignancy in the pediatric population. Little is known about iatrogenic tumor cell peritoneal seeding after primary surgical resection of peritoneal rhabdomyosarcoma, and there is limited guidance on optimal management of such cases. Its aggressive nature, coupled with widespread peritoneal tumor seeding, contributes to the poor outcome of such cases.

Case presentation: A 6-year-old Tigrayan girl from Northern Ethiopia presented with lower abdominal swelling of 1 month duration. Abdominopelvic ultrasound and computed tomography scan revealed a well-defined pelvic solid mass located in front of the uterus and bladder. Exploratory laparotomy demonstrated an 8-cm mass attached to the lower abdominal wall and anterior pelvic peritoneum with no other mass lesion. Complete resection of the mass was done, and a subsequent histopathologic examination along with immunohistochemistry for desmin and myogenin confirmed the diagnosis of embryonal rhabdomyosarcoma. Five weeks postoperatively, she returned with multiple peritoneal malignant depositions. She was given systemic chemotherapy but died before receiving the second cycle.

Conclusion: The aggressive nature of primary peritoneal rhabdomyosarcoma and the possible influence of surgical handling on recurrence are both highlighted in this case. To achieve an optimal outcome, early multimodal therapy and preventive surgical techniques are essential.

Background: Gallbladder duplication is a rare congenital anomaly resulting from abnormal bifurcation of the caudal bud during embryogenesis. It may cause diagnostic and surgical challenges. Classification systems of gallbladder duplication are based on cystic duct anatomy. Though often asymptomatic, it can be associated with complications such as cholelithiasis.

Case presentation: We report the case of a 22-year-old North African male presenting to the emergency department with a left renal colic episode. He complained of left lumbar pain with typical radiation and no other associated symptoms. The patient has a medical history of myasthenia gravis and major depressive disorder. Physical examination revealed left lumbar and iliac tenderness without any other notable findings. The patient was initially referred to the radiology department for a computed tomography scan to investigate potential renal colic and rule out a surgical emergency. The computed tomography scan identified a nonobstructive left renal calculus and a duplicated gallbladder. An abdominal ultrasound was performed for further evaluation, confirming the gallbladder duplication and ruling out gallstones or cholecystitis. To obtain better visualization of the biliary tree and assess for any additional anatomical anomalies, magnetic resonance imaging with magnetic resonance cholangiopancreatography was conducted. Magnetic resonance cholangiopancreatography confirmed a type H gallbladder duplication.

Conclusion: Diagnostic methods, including ultrasound, computed tomography scan, and magnetic resonance imaging, help identify gallbladder duplication. Magnetic resonance cholangiopancreatography is considered the gold standard for detailed biliary tract imaging.

Background: The prevalence of inflammatory bowel diseases is increasing among the elderly. Older patients with inflammatory bowel disease represent a vulnerable population whose treatment strategies are significantly impacted by comorbidities and frailty.

Case presentation: We present the case of a 69-year-old, Hungarian, nonsmoker female patient with a history of elderly-onset inflammatory bowel disease. The patient has longstanding type 2 diabetes (on insulin since 2011) and is being treated for hypertension and hyperlipidemia. In 2011, a diagnosis of left-sided ulcerative colitis was made, presenting with symptoms of fever and bloody diarrhea. Following the ineffectiveness of several conservative treatment modalities, the patient initially underwent adalimumab therapy, exhibiting primary nonresponse. Subsequently, the treatment was switched to vedolizumab, but she later developed secondary loss of response. She was diagnosed with end-stage renal disease and was started on hemodialysis in January 2022. In May 2023, because of severe endoscopic and symptomatic disease activity (endoscopic Mayo score 3, partial Mayo score 6, C-reactive protein 26.0 mg/L, hemoglobin 111 g/L, albumin 39 g/L), infliximab therapy was initiated. We measured serial serum drug and anti-drug antibody concentrations to monitor the efficacy of the therapy and the effect of hemodialysis on drug concentrations. The patient responded to infliximab therapy, leading to clinical remission. Infliximab serum drug concentrations remained unaffected by hemodialysis.

Conclusion: This case clearly illustrates the impact of comorbidities and the importance of a multidisciplinary approach in the management of elderly patients with inflammatory bowel disease. We demonstrated that infliximab therapy is effective and safe in the presence of concomitant severe inflammatory bowel disease and end-stage renal disease requiring hemodialysis.

Background: Gastrointestinal basidiobolomycosis is a rare fungal infection caused by Basidiobolus ranarum, increasingly recognized as an intestinal pathogen in children. Its clinical features closely mimic malignancy or inflammatory bowel disease, leading to frequent diagnostic delays. Although endemic in regions such as Saudi Arabia, Iran, and Oman, sporadic cases occur worldwide. Amphotericin B-liposomal remains a cornerstone therapy for invasive fungal disease, but no standardized treatment protocol for pediatric gastrointestinal basidiobolomycosis has been established.

Case presentation: We report a 6-year-old Asian girl, born preterm with very low birth weight, who presented with persistent abdominal pain without organomegaly, alternating diarrhea and constipation, fever, and anorexia. Initial ultrasonography revealed colonic wall thickening with aneurysmal dilation at the splenic flexure. Colon biopsy confirmed Basidiobolus ranarum. The patient received intravenous liposomal amphotericin B (90 mg daily), with partial radiologic improvement but persistent symptoms, prompting exploratory laparotomy. Adhesiolysis, left hemicolectomy, and resection of jejunal and colonic masses were performed. Histopathology demonstrated dense eosinophil-rich granulomatous inflammation with multinucleated giant cells and a solitary fungal hypha, without classic Splendore-Hoeppli phenomenon, likely due to prior antifungal therapy. Postoperatively, the patient was discharged on oral itraconazole for 28 days with adjunctive cotrimoxazole. At 1-year follow-up, she remained asymptomatic with no evidence of recurrence.

Conclusion: This case highlights the diagnostic challenges of pediatric gastrointestinal basidiobolomycosis and demonstrates successful management with liposomal amphotericin B followed by itraconazole. It underscores the ongoing gap in establishing standardized treatment and supports further prospective studies to define optimal antifungal and surgical strategies for this rare entity.

Background: Various neurological diseases can cause rapidly progressive dementia, which is a clinical syndrome characterized by a rapid decline in cognitive function over a short period, typically less than 1 or 2 years. It can be caused by various neurological diseases, including neurodegenerative, inflammatory, vascular, metabolic, and neoplastic central nervous system diseases. Rapidly progressive dementia is particularly associated with Creutzfeldt-Jakob disease, but other conditions such as immune-mediated encephalitis, rapidly progressive subtypes of Alzheimer's disease, and various other mimics of prion diseases must also be considered. Multiple cerebral microbleeds are typical imaging features of cerebral small vessel diseases, but can also appear in other rare conditions.

Case presentation: We describe the case of a 45-year-old Asian female patient with lung adenocarcinoma who exhibited rapidly progressive dementia and multiple cerebral microbleeds. The patient was a 45-year-old woman who experienced rapid cognitive decline without obvious triggers, accompanied by disorganized speech, difficulty in expression, and short-term memory loss. Brain magnetic resonance imaging revealed widely distributed microhemorrhages, while computed tomography and pathological examination further confirmed the diagnosis of lung adenocarcinoma. The patient did not undergo a brain biopsy because of the rapid deterioration of her illness. Her condition deteriorated rapidly, leading to death in the fourth month.

Conclusion: Herein, we discuss the presence of the apolipoprotein ε4 allele risk gene and the role of the tumor in causing multiple nodular lesions in the patient's brain, as well as multiple microbleeds. The role of the apolipoprotein ε4 allele risk gene in multiple nodular lesions of the brain and CMB requires further study, as it may be responsible for the rapid cognitive decline and imaging findings observed in patients. The role of the tumor in causing these brain lesions and cerebral microbleeds is also of interest as it may help to provide insight into the pathophysiological mechanisms of rapidly progressive dementia in the context of cancer. This case highlights the need for a comprehensive diagnosis, including magnetic resonance imaging, blood and cerebrospinal fluid analyses, and brain biopsy, to identify treatable causes of rapidly progressive dementia.

Background: Wilms tumor, or nephroblastoma, arises from embryonal metanephric blastemal cells and is the most common renal malignancy in the pediatric population. However, its occurrence in adults is extremely rare, with only a limited number of cases documented in the literature. In adults, renal masses most commonly turn out to be renal cell carcinomas, which can closely mimic Wilms tumor on imaging, posing a significant diagnostic challenge. Particularly in resource-limited settings, definitive diagnosis is often established only after histopathological evaluation of nephrectomy specimens.

Case presentation: We present the case of a 24-year-old Indian woman who presented with a progressively enlarging lump in the right flank. The subsequent investigations revealed a right renal mass, suspected to be malignant, for which the patient underwent a right radical nephrectomy. Histopathological examination confirmed the diagnosis of adult Wilms tumor (nephroblastoma).

Conclusion: This case highlights the rare occurrence of Wilms tumor in an adult female and underscores the diagnostic challenges it poses. We aim to contribute to the limited body of literature on adult nephroblastoma and promote awareness of this rare entity among clinicians and pathologists.