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A Case Report of Acinar Cell Cystadenoma with Pancreatic Intraepithelial Neoplasia: Is It Always Benign? 腺泡细胞囊腺瘤合并胰腺上皮内瘤变1例:是否总是良性的?
IF 0.6 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-08-06 eCollection Date: 2025-01-01 DOI: 10.1159/000546668
Thai Hau Koo, Venkata Sunkesula, Salah Abdel Jalil, Richard Wong, Ala Abdel-Jalil, Elham Abdel Jalil

Introduction: The exact etiology of acinar cell cystadenoma (ACC) has been debated, primarily whether it originates from or carries the risk of an underlying neoplasia. Pancreatic intraepithelial neoplasia (PanIN) is presumed to be a noninvasive precursor of pancreatic ductal adenocarcinoma. This report presents a rare case of ACC with low-grade PanIN that required surgical resection.

Case presentation: A 60-year-old female with an unremarkable medical history presented with epigastric pain for 2 weeks. Her initial laboratory workup was notable for mild isolated elevation of alkaline phosphatase. Abdominal computed tomography revealed a 5.0 × 4.0 cm cystic lesion in the pancreatic head with thick internal septations. Magnetic resonance cholangiopancreatography showed a 5.2 × 4.5 × 6.8 cm lobulated cystic lesion in the pancreatic head with a microcystic configuration, multiple internal septations, and a hypointense central scar. Endosonographic examination showed a large multicystic lesion in the pancreatic head region. Fine-needle aspiration showed a carcinoembryonic antigen level of 555 ng/mL and an amylase level of 13,593 U/L. No KRAS or GNAS mutations or loss of heterozygosity was detected. Subsequently, the patient underwent a Whipple procedure. Pathologic examination revealed a complex cystic lesion with well-differentiated acinar cells and patches of ductal epithelium compatible with ACC. Histological examination confirmed the presence of low-grade PanIN without invasive carcinoma. The patient recovered well from surgery, and repeat imaging 2 months later was unremarkable.

Conclusion: ACC is a rare benign pancreatic lesion. Low-grade PanIN is typically found in benign pancreatic lesions. Resection is recommended for symptomatic patients.

腺泡细胞囊腺瘤(ACC)的确切病因一直存在争议,主要是它是否起源于潜在的肿瘤,还是有潜在的肿瘤风险。胰腺上皮内瘤变(PanIN)被认为是胰腺导管腺癌的非侵袭性前体。本文报告一例罕见的ACC合并低级别PanIN,需要手术切除。病例介绍:60岁女性,无明显病史,腹痛2周。她最初的实验室检查显示轻度孤立性碱性磷酸酶升高。腹部计算机断层扫描显示胰腺头部一5.0 × 4.0 cm囊性病变,内隔较厚。磁共振胰胆管造影示胰腺头部一5.2 × 4.5 × 6.8 cm分叶状囊性病变,呈微囊状结构,多发内隔,中心处低信号瘢痕。超声检查显示胰头区有一个大的多囊性病变。细针穿刺癌胚抗原水平为555 ng/mL,淀粉酶水平为13593 U/L。未发现KRAS或GNAS突变或杂合性缺失。随后,患者接受了惠普尔手术。病理检查显示一个复杂的囊性病变,有分化良好的腺泡细胞和与ACC相容的导管上皮斑块。组织学检查证实存在低级别PanIN,无浸润性癌。患者术后恢复良好,2个月后复查无明显异常。结论:ACC是一种罕见的胰腺良性病变。低级别PanIN通常见于良性胰腺病变。有症状的患者建议切除。
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引用次数: 0
A Case Report of Mixed Acinar-Neuroendocrine Pancreatic Carcinoma: A Rare Subtype of Pancreatic Cancer. 腺泡-神经内分泌混合型胰腺癌1例:罕见的胰腺癌亚型。
IF 0.6 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-08-06 eCollection Date: 2025-01-01 DOI: 10.1159/000546722
Thai Hau Koo, Venkata Sunkesula, Melissa Daou, Salah Abdel Jalil, Richard Wong, Ala Abdel-Jalil, Elham Abdel Jalil

Introduction: Mixed acinar-neuroendocrine carcinoma (MANEC) is a rare variant of pancreatic carcinoma, the morphology of which shows features of both acinar cell carcinoma and neuroendocrine carcinomas. To date, only approximately 70 cases of MANEC have been reported.

Case presentation: We report a rare case of a 63-year-old male cirrhotic patient who presented with a pancreatic body/tail mass, which was later confirmed to be an MANEC. He was initiated on neoadjuvant chemotherapy with FOLFIRINOX, but unfortunately died before surgery.

Conclusion: MANEC is a rare sporadic tumor of the pancreas. Further studies on the clinicopathological behavior of MANEC are needed to help better understand the disease and establish standardized management.

摘要:腺泡-神经内分泌混合性癌(MANEC)是一种罕见的胰腺肿瘤,其形态具有腺泡细胞癌和神经内分泌癌的双重特征。迄今为止,仅报告了大约70例MANEC病例。病例介绍:我们报告一例罕见的63岁男性肝硬化患者,他表现为胰腺体/尾部肿块,后来被证实为MANEC。患者开始接受FOLFIRINOX新辅助化疗,但不幸在手术前死亡。结论:MANEC是一种罕见的胰腺散发性肿瘤。需要进一步研究MANEC的临床病理行为,以帮助更好地了解疾病并建立规范的管理。
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引用次数: 0
Gastrointestinal Presentation of Eosinophilic Granulomatosis with Polyangiitis, Formerly Churg-Strauss Syndrome: A Case Report. 嗜酸性肉芽肿病合并多血管炎,原Churg-Strauss综合征的胃肠道表现:1例报告。
IF 0.6 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-08-01 eCollection Date: 2025-01-01 DOI: 10.1159/000546038
Muhammad Umair Tayyub, Arsh Gupta, Rabia Ashraf, Meenal Gupta, Priyan Tantrige, Howard Curtis, Vinod Audimoolam

Introduction: Some of the most common presentations of gastrointestinal diseases are nausea, vomiting, and generalized abdominal pain. These symptoms could also be associated with other diseases and require a structured approach to the specific diagnosis. Eosinophilic granulomatosis with polyangiitis (EGPA), also known as Churg-Strauss syndrome, is a small and medium-sized vessel vasculitis that can affect any body system and present with a wide range of symptoms and atypical presentation. Therefore, diagnosis of this condition may not always be straightforward.

Case presentation: This case report presents a challenging case of a young man presenting with symptoms of abdominal pain, nausea, vomiting and non-specific symptoms of lethargy and fatigue. The case describes overlapping features with granulomatosis with polyangiitis (GPA) and anti-neutrophil cytoplasmic antibodies (ANCA)-positive and -negative vasculitis. The patient needed extensive workup and investigations to be eventually diagnosed with EGPA. In this case, myeloperoxidase (MPO) ANCA was negative, which is positive in the majority of EGPA cases, and this poses an additional diagnostic dilemma.

Conclusion: The case highlights that non-specific complaints of vomiting and abdominal pain should be addressed methodically and not just be treated symptomatically.

胃肠道疾病的一些最常见的表现是恶心、呕吐和全身腹痛。这些症状也可能与其他疾病有关,需要有条理的方法进行具体诊断。嗜酸性肉芽肿病合并多血管炎(EGPA),也称为Churg-Strauss综合征,是一种中小型血管炎,可影响身体的任何系统,表现为广泛的症状和不典型表现。因此,这种情况的诊断可能并不总是直截了当的。病例报告:本病例报告提出了一个具有挑战性的病例,一名年轻男子表现为腹痛、恶心、呕吐和非特异性嗜睡和疲劳症状。该病例描述了与肉芽肿病合并多血管炎(GPA)和抗中性粒细胞细胞质抗体(ANCA)-阳性和阴性血管炎的重叠特征。患者需要广泛的检查和调查才能最终诊断为EGPA。在本例中,髓过氧化物酶(MPO) ANCA为阴性,而在大多数EGPA病例中为阳性,这造成了额外的诊断困境。结论:该病例强调非特异性呕吐和腹痛的主诉应有条不紊地处理,而不仅仅是对症治疗。
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引用次数: 0
COVID-19 Induced Cholangiopathy: A Case Report. COVID-19致胆管病1例报告
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-07-16 eCollection Date: 2025-01-01 DOI: 10.1159/000546723
Bassem Al Hariri, Muhammad Sharif, Lujain Al-Emadi, Richard Shamoon, Memon Noor Illahi, Nabil S Mahmood, Muayad Kasim Khalid

Introduction: COVID-19, although primarily a respiratory illness, has been linked to complications in multiple organ systems, including the liver. Proposed mechanisms for liver injury include direct viral cytopathic effects, systemic inflammation, hypoxia, and drug-induced liver injury (DILI). Moreover, post-COVID cholangiopathy is an emerging entity with features that may overlap with autoimmune phenomena.

Case presentation: A 60-year-old male patient with multiple comorbidities presented with fever, chills, and cough for 1 day. In the emergency department, he tested positive for COVID-19 by PCR and his chest X-ray revealed features suggestive of pulmonary edema. The patient was intubated and admitted to the Medical Intensive Care Unit (MICU) for management of COVID-19 pneumonia with pulmonary edema. During hospitalization, he developed cardiac complications that required targeted management. Approximately 1 week after admission, his liver enzymes began to rise. Although drug-DILI was initially suspected and hepatotoxic medications were discontinued with the initiation of ursodeoxycholic acid (UDCA), the liver function tests (LFTs) remained elevated. Subsequent magnetic resonance cholangiopancreatography revealed periportal inflammation with intrahepatic biliary dilatation and stricturing, findings consistent with COVID-19 induced cholangiopathy. The UDCA dosage was doubled, resulting in gradual biochemical improvement; however, the patient ultimately discharged against medical advice.

Conclusion: COVID-19-induced cholangiopathy is a rare but serious liver complication. Effective management requires a multidisciplinary team. Ongoing research is needed to better understand long-term liver effects and improve care strategies.

导言:COVID-19虽然主要是一种呼吸道疾病,但与包括肝脏在内的多个器官系统的并发症有关。提出的肝损伤机制包括直接的病毒细胞病变作用、全身性炎症、缺氧和药物性肝损伤(DILI)。此外,后covid胆管病是一种新兴的实体,其特征可能与自身免疫性现象重叠。病例介绍:60岁男性患者,有多种合并症,表现为发热、发冷、咳嗽1天。在急诊室,他的PCR检测结果为COVID-19阳性,胸部x光片显示肺水肿的特征。患者插管后住进医疗重症监护室(MICU)治疗COVID-19肺炎合并肺水肿。在住院期间,他出现了心脏并发症,需要有针对性的治疗。入院后约1周,他的肝酶开始升高。虽然最初怀疑是药物- dili,并且在开始使用熊去氧胆酸(UDCA)后停止了肝毒性药物,但肝功能测试(LFTs)仍然升高。随后的磁共振胆管造影显示门静脉周围炎症伴肝内胆道扩张和狭窄,与COVID-19诱导的胆管病一致。UDCA用量加倍,生化逐渐改善;然而,病人最终不顾医嘱出院了。结论:新型冠状病毒感染的胆管病是一种罕见但严重的肝脏并发症。有效的管理需要一个多学科的团队。为了更好地了解对肝脏的长期影响和改善护理策略,需要进行持续的研究。
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引用次数: 0
Case Report of Hepatic Sinusoidal Obstruction Syndrome Complicated with Myeloproliferative Neoplasm and Focal Segmental Glomerulosclerosis. 肝窦阻塞综合征并发骨髓增生性肿瘤及局灶节段性肾小球硬化1例。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-07-10 eCollection Date: 2025-01-01 DOI: 10.1159/000546801
Yuxuan Song, Yajing Zhen, Hui Ma, Bo Feng, Yandi Xie

Introduction: A 19-year-old male presented with a 6-month history of recurrent ascites and lower limb edema, prompting a detailed diagnostic evaluation at our hospital.

Case presentation: The patient displayed a ruddy complexion, deepening pigmentation in the limbs and abdomen, visible reticular skin pattern changes, and pronounced abdominal striae. Diagnostic investigations included a renal biopsy, which confirmed focal segmental glomerulosclerosis, and an abdominal enhanced CT scan, suggesting hepatic sinusoidal obstruction syndrome. Hematological tests revealed elevated white blood cell count (19.73 × 109/L), hemoglobin level (183 g/L), and platelet count (395 × 109/L). Bone marrow morphology indicated proliferation of red blood cells, white blood cells, and platelets, suspicious for myeloproliferative neoplasm. PCR testing confirmed the presence of the JAK2 V617F mutation, leading to a diagnosis of polycythemia vera. The patient was administered a comprehensive treatment regimen consisting of methylprednisolone, telmisartan, rivaroxaban, furosemide, and spironolactone. This therapeutic approach led to a decrease in the patient's weight and 24-h urinary protein, along with a significant reduction in pleural and abdominal effusions.

Conclusion: This case underscores the significance of a meticulous diagnostic process in uncovering multiple concurrent severe pathologies presenting with nonspecific symptoms. It also highlights the importance of a targeted treatment strategy to achieve clinical improvement. The successful management of this patient's complex case illustrates the value of a multidisciplinary approach in addressing polycythemia vera, hepatic sinusoidal obstruction syndrome, and focal segmental glomerulosclerosis.

简介:一名19岁男性,因6个月的复发性腹水和下肢水肿病史,在我院进行了详细的诊断评估。病例表现:患者面色红润,四肢及腹部色素沉着加深,可见网状皮肤改变,腹部纹明显。诊断检查包括肾活检,证实局灶节段性肾小球硬化,腹部增强CT扫描,提示肝窦梗阻综合征。血液学检查显示白细胞计数(19.73 × 109/L)、血红蛋白水平(183 g/L)和血小板计数(395 × 109/L)升高。骨髓形态提示红细胞、白细胞、血小板增生,疑为骨髓增生性肿瘤。PCR检测证实了JAK2 V617F突变的存在,导致真性红细胞增多症的诊断。给予患者综合治疗方案,包括甲泼尼龙、替米沙坦、利伐沙班、呋塞米和螺内酯。这种治疗方法导致患者体重和24小时尿蛋白下降,同时胸膜和腹腔积液显著减少。结论:本病例强调了细致的诊断过程在发现以非特异性症状为表现的多重并发严重病理中的重要性。它还强调了有针对性的治疗策略对实现临床改善的重要性。该患者复杂病例的成功治疗说明了多学科方法在解决真性红细胞增多症、肝窦阻塞综合征和局灶节段性肾小球硬化方面的价值。
{"title":"Case Report of Hepatic Sinusoidal Obstruction Syndrome Complicated with Myeloproliferative Neoplasm and Focal Segmental Glomerulosclerosis.","authors":"Yuxuan Song, Yajing Zhen, Hui Ma, Bo Feng, Yandi Xie","doi":"10.1159/000546801","DOIUrl":"10.1159/000546801","url":null,"abstract":"<p><strong>Introduction: </strong>A 19-year-old male presented with a 6-month history of recurrent ascites and lower limb edema, prompting a detailed diagnostic evaluation at our hospital.</p><p><strong>Case presentation: </strong>The patient displayed a ruddy complexion, deepening pigmentation in the limbs and abdomen, visible reticular skin pattern changes, and pronounced abdominal striae. Diagnostic investigations included a renal biopsy, which confirmed focal segmental glomerulosclerosis, and an abdominal enhanced CT scan, suggesting hepatic sinusoidal obstruction syndrome. Hematological tests revealed elevated white blood cell count (19.73 × 10<sup>9</sup>/L), hemoglobin level (183 g/L), and platelet count (395 × 10<sup>9</sup>/L). Bone marrow morphology indicated proliferation of red blood cells, white blood cells, and platelets, suspicious for myeloproliferative neoplasm. PCR testing confirmed the presence of the JAK2 V617F mutation, leading to a diagnosis of polycythemia vera. The patient was administered a comprehensive treatment regimen consisting of methylprednisolone, telmisartan, rivaroxaban, furosemide, and spironolactone. This therapeutic approach led to a decrease in the patient's weight and 24-h urinary protein, along with a significant reduction in pleural and abdominal effusions.</p><p><strong>Conclusion: </strong>This case underscores the significance of a meticulous diagnostic process in uncovering multiple concurrent severe pathologies presenting with nonspecific symptoms. It also highlights the importance of a targeted treatment strategy to achieve clinical improvement. The successful management of this patient's complex case illustrates the value of a multidisciplinary approach in addressing polycythemia vera, hepatic sinusoidal obstruction syndrome, and focal segmental glomerulosclerosis.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"502-508"},"PeriodicalIF":0.5,"publicationDate":"2025-07-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12245151/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144607501","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Acute Intestinal Obstruction from Small Bowel Stricture following Superior Mesenteric and Portal Vein Thrombosis: A Case Report. 肠系膜上腔及门静脉血栓形成后小肠狭窄致急性肠梗阻1例。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-07-10 eCollection Date: 2025-01-01 DOI: 10.1159/000546789
Saurabh Raj, Rudrakshi Mahaldar, Bishal Saha, Subhra Banerjee

Introduction: Portal vein thrombosis (PVT) is a critical factor in portal hypertension, often linked to liver disease but also occurring independently. Superior mesenteric vein thrombosis and PVT can lead to mesenteric ischemia, even without predisposing hepatic or abdominal conditions. While acute or chronic PVT may present with variable occlusion, the development of intestinal strictures is rare. Persistent symptoms necessitate close follow-up for early detection and timely intervention.

Case presentation: A male patient in his early 40s presented with 15 days of progressive upper abdominal pain and one day of vomiting. He had a history of diabetes and hypertension. Examination revealed tachycardia and epigastric tenderness. Laboratory tests showed neutrophilic leukocytosis and positive occult blood in the stool. Contrast-enhanced CT (CECT) revealed PVT extending into its right branch, jejunal wall thickening, and mesenteric engorgement suggestive of ischemia. Upper GI endoscopy showed esophageal varices and portal hypertensive gastropathy. He was managed conservatively with anticoagulation and discharged. One month later, he returned with recurrent vomiting, worsened by solid food. Repeat CECT showed jejunal obstruction with a 3-cm stricture. Exploratory laparotomy revealed dense adhesions; adhesiolysis, bowel resection, anastomosis, and feeding jejunostomy were performed. Histopathological examination showed a sealed-off perforation with acute suppurative inflammation and fibrotic changes consistent with ischemic stricture.

Conclusion: While mesenteric venous thrombosis is primarily managed with anticoagulation, progressive bowel strictures may develop over time, necessitating surgical intervention. Long-term follow-up is crucial, as delayed complications can arise despite initial success in preventing infarction.

门静脉血栓形成(PVT)是门静脉高压的一个关键因素,通常与肝脏疾病有关,但也独立发生。肠系膜上静脉血栓形成和PVT可导致肠系膜缺血,即使没有易感的肝脏或腹部疾病。虽然急性或慢性PVT可能表现为不同的闭塞,但肠狭窄的发展是罕见的。持续症状需要密切随访,以便及早发现和及时干预。病例介绍:男性患者,40岁出头,表现为15天进行性上腹部疼痛和1天呕吐。他有糖尿病和高血压病史。检查发现心动过速和上腹压痛。实验室检查显示嗜中性粒细胞增多,粪便隐血阳性。增强CT (CECT)显示PVT延伸至其右支,空肠壁增厚,肠系膜充血提示缺血。上消化道内窥镜检查显示食道静脉曲张及门脉高压性胃病。患者接受了保守的抗凝治疗并出院。一个月后,他复发性呕吐,并因食用固体食物而恶化。重复CECT显示空肠梗阻伴3厘米狭窄。剖腹探查发现粘连致密;行粘连松解、肠切除、吻合、空肠喂养造口术。组织病理学检查显示封闭穿孔伴急性化脓性炎症和纤维化改变,符合缺血性狭窄。结论:虽然肠系膜静脉血栓形成主要通过抗凝治疗,但随着时间的推移,进行性肠狭窄可能会发展,需要手术干预。长期随访是至关重要的,因为尽管最初成功地预防了梗死,但延迟并发症可能会出现。
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引用次数: 0
A Case Report of Gastric Squamous Cell Carcinoma Associated with Pancreatic Adenocarcinoma and Literature Review. 胃鳞癌合并胰腺癌1例报告并文献复习。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-07-09 eCollection Date: 2025-01-01 DOI: 10.1159/000546802
Shiwei Yao, Xueyong Deng

Introduction: It is common for advanced pancreatic adenocarcinoma to infiltrate the gastric wall, and the tumor cells invading the gastric wall are usually consistent with pancreatic adenocarcinoma. However, it is very rare for the tumor cells infiltrating the gastric wall to be squamous cell carcinoma.

Case presentation: In this case, we report a 57-year-old man who presented with weight loss for 2 months. CT revealed a pancreatic mass. Endoscopic ultrasound biopsy of the pancreatic mass confirmed pancreatic adenocarcinoma. He refused treatment. Eight months later, he developed melena. CT revealed that the pancreatic mass had invaded the gastric wall. Gastric squamous cell carcinoma was confirmed by biopsy of gastric tissue under gastroscopy.

Conclusion: By observing the evolution process of this case, we found that the occurrence of gastric squamous cell carcinoma in this case was consistent with one of its pathogenesis: the theory of adenocarcinoma differentiating into squamous cell carcinoma. It provides certain clinical significance for the study of the mechanism of gastric squamous cell carcinoma.

导读:晚期胰腺腺癌浸润胃壁是常见的,肿瘤细胞浸润胃壁通常与胰腺腺癌相一致。然而,浸润胃壁的肿瘤细胞为鳞状细胞癌是非常罕见的。病例介绍:在这个病例中,我们报告了一位57岁的男性,体重下降了2个月。CT显示胰腺肿块。胰腺肿块的内镜超声活检证实为胰腺腺癌。他拒绝接受治疗。八个月后,他患上了黑肠病。CT显示胰腺肿块侵入胃壁。胃镜下行胃组织活检证实为胃鳞癌。结论:通过观察本病例的演变过程,我们发现本病例胃鳞状细胞癌的发生符合其发病机制之一:腺癌向鳞状细胞癌分化的理论。为研究胃鳞状细胞癌的发生机制提供了一定的临床意义。
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引用次数: 0
Hepatic Angiosarcoma Presenting as Ascites: Case Report of a Rare yet Lethal Disease. 以腹水表现的肝血管肉瘤:罕见但致命的病例报告。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-07-02 eCollection Date: 2025-01-01 DOI: 10.1159/000546375
Archit Garg, Mehak Bassi, Capecomorin Pitchumoni, Arkady Broder

Introduction: Angiosarcomas, constituting less than 1% of all sarcomas, are rare soft tissue tumors originating from the endothelial cells. Hepatic angiosarcoma (HAS) is a rare and aggressive primary hepatic malignancy accounting for only 0.5%-2% of all liver tumors. The patients often endorse nonspecific symptoms like vague abdominal pain, nausea, vomiting, and jaundice making the diagnosis challenging. Most patients succumb to death within 6 months of diagnosis due to liver failure or hemorrhage from spontaneous rupture of HAS. Therapeutic guidelines remain undefined, and management often involves a multidisciplinary approach. Surgical resection is the only potentially curative option, which has been shown to be most beneficial when HAS is limited to one lobe. Hepatic artery embolization is used in the case of rupture of HAS. Chemotherapy can be used for palliative care in cases of advanced tumors. We present a fatal case of metastatic HAS to underscore diagnostic pitfalls and therapeutic challenges.

Case description: A 56-year-old male presented with 2 months of abdominal pain, distension, fatigue, and weight loss. Imaging revealed multifocal hypodense liver and splenic lesions. Laboratory findings included severe anemia (Hb 6.1 g/dL), thrombocytopenia (63 × 103/mm3), and elevated liver enzymes. Ascitic fluid analysis demonstrated exudative, bloody ascites (SAAG <1.1) without malignant cytology. Liver biopsy confirmed HAS, showing atypical spindle cells infiltrating vascular channels, positive for CD34 and factor VIII. Despite transfusions, paracentesis, and palliative care, the patient developed disseminated intravascular coagulation and died 2 weeks post-diagnosis.

Conclusion: HAS is a rapidly fatal malignancy often diagnosed at advanced stages due to nonspecific symptoms and lack of definitive risk factors in most cases. Multidisciplinary collaboration is essential for symptom management, though treatment options remain limited, and prognosis is poor. Therefore, it becomes imperative for clinicians to keep in mind the common presentation of a rare but lethal disease.

血管肉瘤是一种罕见的软组织肿瘤,起源于内皮细胞,占所有肉瘤的不到1%。肝血管肉瘤(HAS)是一种罕见的侵袭性原发性肝脏恶性肿瘤,仅占所有肝脏肿瘤的0.5%-2%。患者通常表现出非特异性症状,如模糊腹痛、恶心、呕吐和黄疸,使诊断具有挑战性。大多数患者在诊断后6个月内死于肝衰竭或自发性肝破裂出血。治疗指南仍然不明确,管理通常涉及多学科方法。手术切除是唯一潜在的治疗选择,当肿瘤局限于一个肺叶时,手术切除是最有益的。肝动脉栓塞用于肝动脉破裂的病例。化疗可用于晚期肿瘤的姑息治疗。我们提出一个致命的病例转移性HAS强调诊断陷阱和治疗的挑战。病例描述:56岁男性,腹痛、腹胀、乏力、体重下降2个月。影像学显示肝脏及脾脏多灶性低密度病变。实验室结果包括严重贫血(血红蛋白6.1 g/dL)、血小板减少(63 × 103/mm3)和肝酶升高。结论:HAS是一种快速致命的恶性肿瘤,由于大多数病例的非特异性症状和缺乏明确的危险因素,通常在晚期诊断出来。多学科合作对症状管理至关重要,尽管治疗选择仍然有限,预后较差。因此,临床医生必须牢记这种罕见但致命的疾病的常见表现。
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引用次数: 0
Beyond Inflammatory Bowel Disease: Calprotectin as a Marker for Trichobezoar? A Case Report. 炎性肠病之外:钙保护蛋白作为毛癣的标志物?一个病例报告。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-06-24 eCollection Date: 2025-01-01 DOI: 10.1159/000546623
Ammar Khayat

Introduction: Trichobezoar is an aggregation of swallowed hair strands that entangle in the stomach. It presents with abdominal pain, vomiting, and diarrhea. Diagnosis is usually made by imaging and endoscopy. Surgical removal is typically required, but endoscopic retrieval has been successful. Fecal calprotectin is a biomarker of inflammation that is typically elevated in inflammatory bowel disease (IBD) but is also elevated in other gastrointestinal (GI) disorders such as infections. Trichobezoar is not known to precipitate GI inflammation and has never been reported among the etiologies of elevated fecal calprotectin. Herein is a case presenting with vague abdominal pain and elevated fecal calprotectin that was attributed solely to a trichobezoar.

Case presentation: A 7-year-old girl presented with abdominal pain for 6 months associated with occasional vomiting and diarrhea. Family history was positive for ulcerative colitis. Physical examination was unremarkable including normal growth. Diagnostic workup revealed elevated fecal calprotectin at 433.9 mg/kg. Upper and lower endoscopies were performed. A trichobezoar with Rapunzel syndrome was incidentally found in the stomach extending into the duodenum without frank obstruction. Histology showed reactive gastropathy in a few areas of mechanical rubbing by hair strands; otherwise, it was completely unremarkable in both upper and lower GI biopsies. Endoscopic retrieval of piecemeal hair locks was done. Surgical removal was subsequently done due to the generous size of the mass.

Conclusion: Fecal calprotectin is an important biomarker for IBD, as well as other unusual GI pathologies, and clinical context is crucial when interpreting its value.

简介:毛粪是一种被吞下的头发在胃里缠绕的聚集物。表现为腹痛、呕吐和腹泻。诊断通常通过影像学和内窥镜检查。手术切除通常是必需的,但内窥镜切除是成功的。粪钙保护蛋白是炎症的生物标志物,通常在炎症性肠病(IBD)中升高,但在其他胃肠道(GI)疾病(如感染)中也升高。目前尚不知道毛粪石会引起胃肠道炎症,也从未报道过粪钙保护蛋白升高的病因。这里是一个病例,表现为腹痛不清和粪钙保护蛋白升高,这完全归因于毛粪。病例介绍:一名7岁女孩,腹痛6个月,偶有呕吐和腹泻。溃疡性结肠炎家族史呈阳性。体格检查无异常,生长正常。诊断检查显示粪便钙保护蛋白升高至433.9 mg/kg。进行上、下腔镜检查。偶然在胃内发现一伴有莴苣综合征的毛滴虫,并延伸至十二指肠,无明显梗阻。组织学表现为机械性发束摩擦的少数部位出现反应性胃病;除此之外,在上消化道和下消化道活检中完全不明显。内镜下取下零散的头发。由于肿块很大,随后进行了手术切除。结论:粪便钙保护蛋白是IBD以及其他异常胃肠道病变的重要生物标志物,在解释其价值时,临床背景至关重要。
{"title":"Beyond Inflammatory Bowel Disease: Calprotectin as a Marker for Trichobezoar? A Case Report.","authors":"Ammar Khayat","doi":"10.1159/000546623","DOIUrl":"10.1159/000546623","url":null,"abstract":"<p><strong>Introduction: </strong>Trichobezoar is an aggregation of swallowed hair strands that entangle in the stomach. It presents with abdominal pain, vomiting, and diarrhea. Diagnosis is usually made by imaging and endoscopy. Surgical removal is typically required, but endoscopic retrieval has been successful. Fecal calprotectin is a biomarker of inflammation that is typically elevated in inflammatory bowel disease (IBD) but is also elevated in other gastrointestinal (GI) disorders such as infections. Trichobezoar is not known to precipitate GI inflammation and has never been reported among the etiologies of elevated fecal calprotectin. Herein is a case presenting with vague abdominal pain and elevated fecal calprotectin that was attributed solely to a trichobezoar.</p><p><strong>Case presentation: </strong>A 7-year-old girl presented with abdominal pain for 6 months associated with occasional vomiting and diarrhea. Family history was positive for ulcerative colitis. Physical examination was unremarkable including normal growth. Diagnostic workup revealed elevated fecal calprotectin at 433.9 mg/kg. Upper and lower endoscopies were performed. A trichobezoar with Rapunzel syndrome was incidentally found in the stomach extending into the duodenum without frank obstruction. Histology showed reactive gastropathy in a few areas of mechanical rubbing by hair strands; otherwise, it was completely unremarkable in both upper and lower GI biopsies. Endoscopic retrieval of piecemeal hair locks was done. Surgical removal was subsequently done due to the generous size of the mass.</p><p><strong>Conclusion: </strong>Fecal calprotectin is an important biomarker for IBD, as well as other unusual GI pathologies, and clinical context is crucial when interpreting its value.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":"19 1","pages":"482-487"},"PeriodicalIF":0.5,"publicationDate":"2025-06-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12187109/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144483259","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Gastroduodenal Strongyloidiasis Diagnosed during Iron Deficiency Anemia Workup. 缺铁性贫血检查中诊断胃十二指肠圆线虫病。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2025-06-20 eCollection Date: 2025-01-01 DOI: 10.1159/000539769
Malek Kreidieh, Neville Mobarakai, Sherif Andrawes, Robert Colef, Lara Kreidieh, Hassan Al Moussawi, Jean M Chalhoub

Introduction: The Strongyloidae family includes at least fifty species of intestinal nematode worms, and Strongyloides stercoralis is the most prevalent one among humans. As a result of this parasite's complex lifecycle, it can either remain localized in the intestines or disseminate to different organs throughout the body, culminating in hyperinfection in case of immunosuppression. Strongyloidiasis is rarely encountered in the USA, and an associated gastric involvement is uncommon.

Case presentation: In this case report, we describe the case of an asymptomatic and immunocompetent Liberian patient with gastroduodenal strongyloidiasis whose diagnosis was based on histologic findings during endoscopic evaluation for iron deficiency anemia.

Conclusion: This case sheds light on the importance of suspecting the diagnosis of disseminated strongyloidiasis in high-risk individuals with iron deficiency anemia in combination with peripheral eosinophilia.

导读:蛔虫科包括至少50种肠道线虫,其中粪形蛔虫是人类中最常见的一种。由于这种寄生虫复杂的生命周期,它既可以局限于肠道,也可以传播到全身的不同器官,在免疫抑制的情况下最终导致过度感染。类圆线虫病在美国是罕见的,并伴有胃累及是罕见的。病例介绍:在这个病例报告中,我们描述了一个无症状和免疫能力的利比里亚胃十二指肠圆线虫病患者的病例,其诊断是基于内镜评估缺铁性贫血的组织学结果。结论:本病例提示在缺铁性贫血合并外周血嗜酸性粒细胞增多的高危人群中怀疑弥散性圆线虫病诊断的重要性。
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引用次数: 0
期刊
Case Reports in Gastroenterology
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