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Peribiliary Gland Hyperplasia That Required Differentiation from Extrahepatic Bile Duct Cancer: A Case Report. 需要与肝外胆管癌鉴别的胆管周围腺体增生:病例报告。
IF 0.5 Q4 GASTROENTEROLOGY & HEPATOLOGY Pub Date : 2024-05-23 eCollection Date: 2024-01-01 DOI: 10.1159/000538491
Kazuhiro Takami, Kuniharu Yamamoto, Hiroto Sakurai, Yoshihiro Sato, Noriko Kondo, Toru Nakano, Shingo Tsujinaka, Akinobu Koiwai, Morihisa Hirota, Keigo Murakami, Kazuhiro Murakami, Chikashi Shibata, Yu Katayose

Introduction: The peribiliary gland is an accessory bile duct gland. Hyperplasia of these tissues may lead to elevation of the mucosa in the bile ducts and bile duct stenosis. We herein report a case of peribiliary gland hyperplasia that required preoperative differentiation from bile duct cancer, with a discussion of the literature.

Case presentation: The patient had an adenomatous lesion in the ascending colon that was difficult to treat endoscopically; therefore, surgery was planned. Preoperative abdominal ultrasonography revealed a bile duct tumor, and endoscopic ultrasonography revealed a mass lesion around the confluence of the cystic duct. Computed tomography revealed localized wall thickening in the middle bile duct, and the upstream bile ducts were slightly dilated. In addition, continuous thickening of the bile duct wall from the gallbladder to the confluence of the cystic duct was observed. No distant metastases, such as liver metastases, or nearby enlarged lymph nodes were observed. Endoscopic retrograde cholangiopancreatography and magnetic resonance cholangiopancreatography revealed a papillary-like elevated lesion in the bile duct near the confluence of the cystic duct, and a biopsy and bile cytology from the same area showed no malignant findings. As a result, the possibility that the patient had gallbladder or bile duct cancer could not be ruled out; therefore, a policy of surgery together with the ascending colon tumor was decided after receiving sufficient informed consent. During surgery, the patient underwent extrahepatic bile duct resection, reconstruction of the biliary tract using the Roux-en-Y method, and right hemicolectomy. Both duodenal and hilar bile duct transects were subjected to a fast frozen section analysis during surgery, and the results were negative for cancer. A histopathological examination of the resected specimen revealed no malignant findings in the bile duct lesions, and a diagnosis of peribiliary gland hyperplasia with chronic inflammatory cell infiltration and fibrosis of the extrahepatic bile duct wall was made.

Conclusions: We encountered a case of peribiliary gland hyperplasia that was difficult to distinguish from bile duct cancer.

导言:胆管周围腺体是胆管的附属腺体。这些组织的增生可能导致胆管粘膜隆起和胆管狭窄。我们在此报告一例胆管周围腺增生病例,该病例需要在术前与胆管癌进行鉴别,并对文献进行了讨论:患者升结肠有一处腺瘤性病变,难以通过内镜治疗,因此计划进行手术。术前腹部超声波检查发现了胆管肿瘤,内镜超声波检查发现了胆囊管汇合处的肿块病变。计算机断层扫描显示中胆管局部管壁增厚,上游胆管轻度扩张。此外,还观察到从胆囊到胆囊管汇合处的胆管壁持续增厚。未发现远处转移(如肝转移)或附近淋巴结肿大。内镜逆行胰胆管造影和磁共振胰胆管造影显示,胆囊管汇合处附近的胆管内有乳头状隆起病变,同一部位的活检和胆汁细胞学检查未发现恶性病变。因此,不能排除患者患有胆囊癌或胆管癌的可能性,所以在征得患者充分知情同意后,决定对其进行升结肠肿瘤一并手术治疗。手术期间,患者接受了肝外胆管切除术,采用 Roux-en-Y 方法重建胆道,并进行了右半结肠切除术。手术期间对十二指肠和肝门胆管横断面进行了快速冰冻切片分析,结果显示癌症阴性。切除标本的组织病理学检查显示胆管病变无恶性发现,诊断为胆管周围腺体增生伴慢性炎症细胞浸润和肝外胆管壁纤维化:结论:我们遇到了一例胆管周围腺体增生症与胆管癌难以区分的病例。
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引用次数: 0
Side Effects of Stem Cell Transplant Mimicking Symptoms of Known Amyloidosis. 模仿已知淀粉样变性症状的干细胞移植副作用。
IF 0.6 Q3 Medicine Pub Date : 2024-05-21 eCollection Date: 2024-01-01 DOI: 10.1159/000538947
Gabriel Heering, Zilan Lin, Michael Rosman, Nao Hara, Fouzia Shakil, Dimitrios Georgostathis

Introduction: AL amyloidosis can involve the gastrointestinal (GI) tract in a sporadic manner, affecting certain anatomical areas while sparing others.

Case presentation: Our patient with AL amyloidosis and confirmed colonic involvement was found to have new odynophagia, GI bleeding, and imaging findings that might suggest AL amyloidosis. However, negative pathology results from esophageal biopsies suggested the patient's new ulcerations were more likely a side effect of her autologous stem cell transplant (SCT) and chemotherapy meant to target amyloidosis, as opposed to an effect of amyloid infiltration itself.

Conclusion: GI involvement of amyloidosis requires a high degree of clinical suspicion and should be considered in patients with systemic diseases affecting the kidney, heart, and GI tract; however, when satisfactory biopsies obtained from endoscopy results are negative, other causes should be considered.

导言:AL淀粉样变性可能以散发性方式累及胃肠道(GI),影响某些解剖区域,而其他区域则不受影响:我们的患者患有 AL 淀粉样变性并确诊为结肠受累,但发现新的吞咽困难、消化道出血和影像学检查结果可能提示 AL 淀粉样变性。然而,食管活检的阴性病理结果表明,患者的新溃疡更可能是自体干细胞移植(SCT)和针对淀粉样变性的化疗的副作用,而非淀粉样变性浸润本身的影响:结论:临床上需要高度怀疑淀粉样变性的消化道受累,如果患者患有影响肾脏、心脏和消化道的全身性疾病,则应考虑淀粉样变性的消化道受累。
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引用次数: 0
Oesophageal Lichen Planus Successfully Treated with Budesonide Orodispersible Tablets: A Case Report. 用布地奈德口腔分散片成功治疗食道扁平苔藓:病例报告。
IF 0.6 Q3 Medicine Pub Date : 2024-05-15 eCollection Date: 2024-01-01 DOI: 10.1159/000538894
Harald Bagger-Jörgensen, Mohammed Abdulrasak, Kevin Sandeman, Mohammed Binsalman, Klas Sjöberg

Introduction: Lichen planus is a relatively common inflammatory condition of the nails, skin, and mucosal surfaces. Oesophageal involvement of lichen planus is thought to be very rare, mainly described in case reports, but is associated with a high risk of oesophageal stenosis as well as squamous cell carcinoma. No evidence-based treatment recommendations exist, with the majority of described treatment regimens involving systemic immunosuppression.

Case report: In this case report, we describe a novel approach in treating oesophageal lichen planus in a patient with budesonide orodispersible tablets, a treatment normally reserved for eosinophilic oesophagitis. The patient achieved complete relief of dysphagia, with a follow-up oesophagogastroduodenoscopy 2 months after treatment commencement being macroscopically and microscopically free of inflammatory activity. This case report is to our knowledge the first to report this treatment regimen in oesophageal lichen planus.

Conclusion: We consider a trial of budesonide orodispersible tablets a reasonable initial management as it's a local therapy specific to the oesophagus with a more benign side effect profile than systemic immunosuppression, but further studies need to be undertaken to corroborate our findings. Also, based on the severity and malignant potential of oesophageal lichen planus, we suggest that physicians be liberal in ordering oesophagogastroduodenoscopy with biopsy taking as part of the workup of dysphagia in a patient with known lichen planus.

简介扁平苔藓是一种比较常见的指甲、皮肤和粘膜表面炎症。扁平苔藓累及食道被认为非常罕见,主要见于病例报告,但与食道狭窄和鳞状细胞癌的高风险相关。目前尚无循证治疗建议,大多数描述的治疗方案都涉及全身免疫抑制:在本病例报告中,我们介绍了一种使用布地奈德口崩片剂治疗食管扁平苔藓患者的新方法,布地奈德口崩片剂通常用于治疗嗜酸性粒细胞性食管炎。该患者的吞咽困难得到了完全缓解,治疗开始 2 个月后进行的食道胃十二指肠镜随访检查显示,其宏观和微观均无炎症活动。据我们所知,本病例报告是首次报道食道扁平苔藓的这种治疗方案:我们认为试用布地奈德口服分散片是一种合理的初始治疗方法,因为它是一种针对食道的局部治疗方法,与全身性免疫抑制相比副作用更小。此外,鉴于食道扁平苔藓的严重性和恶变可能性,我们建议医生在对已知扁平苔藓患者进行吞咽困难检查时,应谨慎要求患者接受食道胃十二指肠镜检查并进行活检。
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引用次数: 0
Biliary Cast Syndrome and Secondary Sclerosing Cholangitis in Critically Ill Patient after Long-Term Treatment in the Intensive Care Unit. 重症监护室长期治疗后重症患者的胆道铸型综合征和继发性硬化性胆管炎
IF 0.6 Q3 Medicine Pub Date : 2024-05-10 eCollection Date: 2024-01-01 DOI: 10.1159/000537957
Adnan Alkurdi, Johannes Herrmann, Damir Bikmukhametov, Rebecca Tschöpe

Introduction: Secondary sclerosing cholangitis in critically ill patients (SSC-CIP) is a rare but underdiagnosed entity that occurs after life-threatening events and treatment in the intensive care unit (ICU). The etiology of SSC-CIP is not fully understood but may be caused by ischemic bile duct injury. SSC-CIP is a cholestatic liver disease that rapidly progresses to liver cirrhosis, with a high mortality rate in the first year of 50%. Endoscopic retrograde cholangiopancreatography (ERCP), which is the gold standard for diagnosing SSC-CIP, shows primary SC-like changes, usually in the intrahepatic bile ducts. Biliary cast formation is pathognomonic for SSC-CIP. No proven effective conservative treatment is available for SSC-CIP, and liver transplantation is the only curative therapy when liver cirrhosis or recurrent cholangitis occurs.

Case presentation: We report the case of a 47-year-old male patient who developed cholestasis after a long treatment in the ICU for severe pneumonia. ERCP showed characteristic findings with rarefication and multiple segmental stenosis in the intrahepatic bile ducts. We removed multiple biliary casts from the bile ducts.

Conclusion: SSC-CIP should be considered for ICU patients with unclear cholestasis, especially when the cholestasis persists after recovery from the underlying disease. Early diagnosis is important to achieve better outcomes; without liver transplantation, the prognosis is generally poor.

简介:重症患者继发性硬化性胆管炎(SSC-CIP)是一种罕见但诊断不足的疾病,多发生在危及生命的事件和重症监护室(ICU)的治疗之后。SSC-CIP 的病因尚不完全清楚,但可能是由缺血性胆管损伤引起的。SSC-CIP 是一种胆汁淤积性肝病,会迅速发展为肝硬化,第一年的死亡率高达 50%。内镜逆行胰胆管造影术(ERCP)是诊断 SSC-CIP 的金标准,可显示原发性 SC 样改变,通常发生在肝内胆管。胆管铸型形成是 SSC-CIP 的病理标志。SSC-CIP 目前尚无行之有效的保守治疗方法,当出现肝硬化或复发性胆管炎时,肝移植是唯一可治愈的治疗方法:我们报告了一例 47 岁男性患者的病例,他因重症肺炎在重症监护室接受长期治疗后出现胆汁淤积。ERCP 显示肝内胆管稀疏化和多节段狭窄。我们从胆管中取出了多个胆汁栓:结论:对于胆汁淤积不清的 ICU 患者,尤其是胆汁淤积在基础疾病康复后仍持续存在时,应考虑使用 SSC-CIP。早期诊断对取得更好的治疗效果非常重要;如果不进行肝移植,预后一般较差。
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引用次数: 0
A Fatal Outcome after Cessation of Nucleotide Analogue Therapy in a Patient with Chronic Hepatitis B: A Case Report. 一名慢性乙型肝炎患者在停止核苷酸类似物治疗后死亡:病例报告。
IF 0.6 Q3 Medicine Pub Date : 2024-04-26 eCollection Date: 2024-01-01 DOI: 10.1159/000538342
Sylvia M Brakenhoff, Heng Chi, Pieter Friederich, Michail Doukas, Caroline den Hoed, Hajo J Flink, Robert J de Knegt, Robert A de Man

Introduction: Emerging evidence suggests that long-term nucleos(t)ide analogue (NA) therapy can be ceased in a selective group of chronic hepatitis B (CHB). This is being gradually implemented in clinical practice.

Case presentation: A 68-year-old man known with a chronic hepatitis B e antigen-positive hepatitis B infection without signs of advanced liver fibrosis or cirrhosis was admitted with acute liver failure. Two months prior to his admission, he ceased his NA therapy. During the admission, NA therapy was restarted, but the liver function worsened. The patient was put on the high-urgency liver transplantation waiting list, and the next day, he was successfully transplanted. However, the patient died 17 days later due to hemorrhagic shock that resulted from intra-abdominal bleeding and acute pancreatitis.

Conclusion: Current guidelines suggest that NA therapy can be discontinued in a selective group of CHB patients. However, these guidelines suggest different stopping and follow-up criteria. This case illustrates that NA withdrawal is not without risks and that these differences in recommendations may lead to inadequate management and eventually a fatal outcome.

导言:新的证据表明,对于部分慢性乙型肝炎(CHB)患者,可以停止长期核苷酸类似物(NA)治疗。病例介绍:一名已知患有慢性乙型肝炎 e 抗原阳性、无晚期肝纤维化或肝硬化症状的 68 岁男性因急性肝衰竭入院。入院前两个月,他停止了 NA 治疗。入院期间,他重新开始接受 NA 治疗,但肝功能恶化。患者被列入肝移植高危候诊名单,并于第二天成功接受了肝移植。然而,17天后,患者因腹腔内出血和急性胰腺炎导致失血性休克而死亡:结论:现行指南建议,有选择性地对部分慢性阻塞性肺病患者停止NA治疗。然而,这些指南提出了不同的停药和随访标准。本病例说明,停用NA并非没有风险,这些不同的建议可能会导致处理不当,最终造成致命后果。
{"title":"A Fatal Outcome after Cessation of Nucleotide Analogue Therapy in a Patient with Chronic Hepatitis B: A Case Report.","authors":"Sylvia M Brakenhoff, Heng Chi, Pieter Friederich, Michail Doukas, Caroline den Hoed, Hajo J Flink, Robert J de Knegt, Robert A de Man","doi":"10.1159/000538342","DOIUrl":"https://doi.org/10.1159/000538342","url":null,"abstract":"<p><strong>Introduction: </strong>Emerging evidence suggests that long-term nucleos(t)ide analogue (NA) therapy can be ceased in a selective group of chronic hepatitis B (CHB). This is being gradually implemented in clinical practice.</p><p><strong>Case presentation: </strong>A 68-year-old man known with a chronic hepatitis B e antigen-positive hepatitis B infection without signs of advanced liver fibrosis or cirrhosis was admitted with acute liver failure. Two months prior to his admission, he ceased his NA therapy. During the admission, NA therapy was restarted, but the liver function worsened. The patient was put on the high-urgency liver transplantation waiting list, and the next day, he was successfully transplanted. However, the patient died 17 days later due to hemorrhagic shock that resulted from intra-abdominal bleeding and acute pancreatitis.</p><p><strong>Conclusion: </strong>Current guidelines suggest that NA therapy can be discontinued in a selective group of CHB patients. However, these guidelines suggest different stopping and follow-up criteria. This case illustrates that NA withdrawal is not without risks and that these differences in recommendations may lead to inadequate management and eventually a fatal outcome.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-04-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11052559/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140849401","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
Damage to the Descending Colon after Endoscopic Balloon Dilatation following a Minor Traffic Accident in a Patient with Crohn’s Disease: A Case Report 一名克罗恩病患者在轻微交通事故后接受内窥镜球囊扩张术,导致降结肠受损:病例报告
IF 0.6 Q3 Medicine Pub Date : 2024-04-25 DOI: 10.1159/000537973
Takaomi Seki, K. Osone, Hiroomi Ogawa, Takuhisa Okada, Takuya Shiraishi, M. Sohda, Ken Shirabe, Hiroshi Saeki
Abstract Introduction Crohn’s disease (CD) is complicated by intestinal strictures and fistula formation; however, intestinal perforation is relatively rare. Case Presentation Following a traffic accident in the evening, a 39-year-old woman experienced abdominal pain that worsened the following morning and was taken to the emergency department. She had a 17-year history of CD and eight endoscopic balloon dilations for descending colonic strictures. She presented with a high fever of 40.0°C, along with tenderness and rebound pain throughout her abdomen, with the most substantial point being in the lower left abdomen. Computed tomography showed thickening of the descending colon wall, increased fat concentration around the wall, and a slight presence of air in the mesentery near the intestinal wall. We diagnosed the patient with generalized peritonitis due to traumatic penetration of the mesentery of the descending colon and performed emergency surgery. Intraoperative observation of the abdominal cavity with a laparoscope revealed purulent ascites but no apparent perforation or edematous mesentery, with white moss and redness in the descending colon. This prompted the decision to perform peritoneal lavage drainage and a transverse colonic double colostomy. The postoperative course was favorable, and the patient was discharged from the hospital on the postoperative day 14. Four months after discharge, colostomy closure was performed. Conclusion Relatively minor trauma in patients with CD can result in colon injury. An injured bowel is usually accompanied by active lesions due to CD; however, caution is required, as endoscopic balloon dilatation without accompaniment may be a background factor.
摘要 引言 克罗恩病(CD)可并发肠狭窄和瘘管形成,但肠穿孔相对罕见。病例介绍 一名 39 岁的女性在傍晚发生交通事故后出现腹痛,次日早晨疼痛加剧,被送往急诊科。她有 17 年的 CD 病史,曾因降结肠狭窄接受过八次内窥镜球囊扩张术。她出现了 40.0°C 的高烧,整个腹部有触痛和反跳痛,最明显的部位在左下腹。计算机断层扫描显示,降结肠壁增厚,肠壁周围脂肪浓度增加,肠系膜靠近肠壁处有轻微空气。我们诊断患者因降结肠系膜外伤穿透而导致全身腹膜炎,并为其实施了急诊手术。术中用腹腔镜观察腹腔,发现有化脓性腹水,但没有明显的穿孔或水肿肠系膜,降结肠有白色苔藓和发红。因此决定进行腹腔灌洗引流和横结肠双结肠造口术。术后情况良好,患者于术后第 14 天出院。出院四个月后,进行了结肠造口缝合术。结论 CD 患者相对轻微的外伤也可能导致结肠损伤。损伤的肠道通常伴有 CD 引起的活动性病变;但需要谨慎,因为无伴有的内窥镜球囊扩张可能是一个背景因素。
{"title":"Damage to the Descending Colon after Endoscopic Balloon Dilatation following a Minor Traffic Accident in a Patient with Crohn’s Disease: A Case Report","authors":"Takaomi Seki, K. Osone, Hiroomi Ogawa, Takuhisa Okada, Takuya Shiraishi, M. Sohda, Ken Shirabe, Hiroshi Saeki","doi":"10.1159/000537973","DOIUrl":"https://doi.org/10.1159/000537973","url":null,"abstract":"Abstract Introduction Crohn’s disease (CD) is complicated by intestinal strictures and fistula formation; however, intestinal perforation is relatively rare. Case Presentation Following a traffic accident in the evening, a 39-year-old woman experienced abdominal pain that worsened the following morning and was taken to the emergency department. She had a 17-year history of CD and eight endoscopic balloon dilations for descending colonic strictures. She presented with a high fever of 40.0°C, along with tenderness and rebound pain throughout her abdomen, with the most substantial point being in the lower left abdomen. Computed tomography showed thickening of the descending colon wall, increased fat concentration around the wall, and a slight presence of air in the mesentery near the intestinal wall. We diagnosed the patient with generalized peritonitis due to traumatic penetration of the mesentery of the descending colon and performed emergency surgery. Intraoperative observation of the abdominal cavity with a laparoscope revealed purulent ascites but no apparent perforation or edematous mesentery, with white moss and redness in the descending colon. This prompted the decision to perform peritoneal lavage drainage and a transverse colonic double colostomy. The postoperative course was favorable, and the patient was discharged from the hospital on the postoperative day 14. Four months after discharge, colostomy closure was performed. Conclusion Relatively minor trauma in patients with CD can result in colon injury. An injured bowel is usually accompanied by active lesions due to CD; however, caution is required, as endoscopic balloon dilatation without accompaniment may be a background factor.","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-04-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140656681","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Severe Hepatitis Complicating Olmesartan Enteropathy: A Case Report 严重肝炎并发奥美沙坦肠病:病例报告
IF 0.6 Q3 Medicine Pub Date : 2024-04-25 DOI: 10.1159/000538552
Ruyi Tan, J. Abasszade, Hardesh Dhillon, Chih Chien Kuan, Thomas Worland, Shireen Tabatabai
Abstract Introduction Olmesartan, an angiotensin II receptor antagonist, is associated with an uncommon complication of enteropathy that presents insidiously, usually months to years after initial commencement of anti-hypertensive therapy which can be dose-dependent. It has a variable spectrum of clinical presentation but commonly presents as a moderate to severe malabsorptive process with potential severe complications related to poor end-organ perfusion. Lymphocytic gastritis and microscopic colitis are often noted in patients presenting with olmesartan-induced enteropathy; however, hepatic involvement has been less frequently observed. Case Presentation We illustrate a case of a 43-year-old female presenting with 2 weeks of profuse non-bloody diarrhea in the context of olmesartan enteropathy which was complicated by an acute severe ischemic and enteropathic hepatopathy. Conclusion Our case prompts clinicians to maintain a high index of suspicion in cases presenting with a seronegative enteropathy and concurrent acute liver injury while on olmesartan therapy. Cessation of olmesartan therapy resulted in prompt resolution of diarrheal symptoms and normalization of the acute transaminitis on subsequent three-week follow-up.
摘要 引言 奥美沙坦是一种血管紧张素 II 受体拮抗剂,与一种不常见的肠病并发症有关。这种并发症的临床表现多种多样,但通常表现为中度至重度吸收不良,并可能出现与末梢器官灌注不良有关的严重并发症。淋巴细胞性胃炎和显微镜下结肠炎经常出现在奥美沙坦诱发的肠病患者身上,但肝脏受累的情况较少见。病例介绍 我们展示了一例 43 岁女性患者的病例,她在奥美沙坦肠病的背景下出现了为期 2 周的大量非血性腹泻,随后并发了急性严重缺血性和肠病性肝炎。结论 我们的病例提示临床医生,在使用奥美沙坦治疗期间出现血清阴性肠病并同时伴有急性肝损伤的病例时,应保持高度怀疑。停止奥美沙坦治疗后,腹泻症状迅速缓解,在随后三周的随访中,急性转氨酶也恢复正常。
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引用次数: 0
Distal Duodenal Stricture Secondary to Mesenteric Fibromatosis (Intra-Abdominal Desmoid Tumor) of the Jejunum 十二指肠远端狭窄继发于肠系膜纤维瘤病(腹腔内蝶形瘤
IF 0.6 Q3 Medicine Pub Date : 2024-04-20 DOI: 10.1159/000538489
Sarah Huang, Jamil Shah, Eduardo Quintero, Philip Q. Xiao, Armand P. Asarian, Madhavi Reddy
Abstract Introduction Mesenteric fibromatosis (intra-abdominal desmoid tumor) is rare, with only a few cases reported in the literature. Clinical symptoms range from asymptomatic, nausea, early satiety, abdominal pain, and gastrointestinal bleeding. Although histologically benign, such a tumor may become locally invasive, and aggressive forms contribute to significant morbidity and mortality. Case Presentation We report the case of a 52-year-old West African male with a 1-year history of intermittent hematochezia and intermittent bloating. Colonoscopy revealed a 4-mm rectal polyp and internal hemorrhoids. Esophagogastroduodenoscopy revealed a severe duodenal stricture 4–5 cm distal to the ampulla. Further work-up with contrast-enhanced computed tomography of the abdomen and pelvis revealed a 5.0 × 3.7 × 4.3-cm mass within the mesentery, encasing the distal portion of the duodenum. Exploratory laparotomy was performed, and the mass was excised from the jejunum. Histopathology findings and immunohistochemical analysis revealed the diagnosis to be mesenteric fibromatosis (desmoid tumor), positive for nuclear β-catenin and SMA, and negative expression of STAT6, desmin, caldesmon, pan-cytokeratin, or c-KIT. The Ki67 index is <1%. Conclusion This case report highlights the diagnostic challenges of mesenteric fibromatosis due to its nonspecific clinical presentation. Recognizing uncommon presentations of mesenteric fibromatosis and risk factors aids in early diagnosis, management, and treatment. Importantly, this also aids in the prevention of complications such as intestinal obstruction, bowel ischemia, and fistula formation.
摘要 引言 肠系膜纤维瘤病(腹腔内类固醇瘤)十分罕见,文献中仅有几例报道。临床症状包括无症状、恶心、早饱、腹痛和消化道出血。此类肿瘤虽然在组织学上是良性的,但可能会发生局部浸润,侵袭性肿瘤会导致严重的发病率和死亡率。病例介绍 我们报告了一名 52 岁西非男性的病例,他有 1 年的间歇性便血和间歇性腹胀病史。结肠镜检查发现了一个 4 毫米的直肠息肉和内痔。食管胃十二指肠镜检查发现,十二指肠严重狭窄,位于安瓿远端 4-5 厘米处。腹部和盆腔造影剂增强计算机断层扫描进一步检查发现,肠系膜内有一个 5.0 × 3.7 × 4.3 厘米的肿块,包裹着十二指肠的远端部分。患者接受了探查性开腹手术,从空肠中切除了肿块。组织病理学检查结果和免疫组化分析显示,诊断结果为肠系膜纤维瘤病(类肉瘤),核β-catenin和SMA阳性,STAT6、desmin、caldesmon、pan-cytokeratin或c-KIT阴性。Ki67 指数小于 1%。结论 本病例报告强调了肠系膜纤维瘤病因其非特异性临床表现而给诊断带来的挑战。认识肠系膜纤维瘤病的不常见表现和风险因素有助于早期诊断、管理和治疗。重要的是,这还有助于预防肠梗阻、肠缺血和瘘管形成等并发症。
{"title":"Distal Duodenal Stricture Secondary to Mesenteric Fibromatosis (Intra-Abdominal Desmoid Tumor) of the Jejunum","authors":"Sarah Huang, Jamil Shah, Eduardo Quintero, Philip Q. Xiao, Armand P. Asarian, Madhavi Reddy","doi":"10.1159/000538489","DOIUrl":"https://doi.org/10.1159/000538489","url":null,"abstract":"Abstract Introduction Mesenteric fibromatosis (intra-abdominal desmoid tumor) is rare, with only a few cases reported in the literature. Clinical symptoms range from asymptomatic, nausea, early satiety, abdominal pain, and gastrointestinal bleeding. Although histologically benign, such a tumor may become locally invasive, and aggressive forms contribute to significant morbidity and mortality. Case Presentation We report the case of a 52-year-old West African male with a 1-year history of intermittent hematochezia and intermittent bloating. Colonoscopy revealed a 4-mm rectal polyp and internal hemorrhoids. Esophagogastroduodenoscopy revealed a severe duodenal stricture 4–5 cm distal to the ampulla. Further work-up with contrast-enhanced computed tomography of the abdomen and pelvis revealed a 5.0 × 3.7 × 4.3-cm mass within the mesentery, encasing the distal portion of the duodenum. Exploratory laparotomy was performed, and the mass was excised from the jejunum. Histopathology findings and immunohistochemical analysis revealed the diagnosis to be mesenteric fibromatosis (desmoid tumor), positive for nuclear β-catenin and SMA, and negative expression of STAT6, desmin, caldesmon, pan-cytokeratin, or c-KIT. The Ki67 index is <1%. Conclusion This case report highlights the diagnostic challenges of mesenteric fibromatosis due to its nonspecific clinical presentation. Recognizing uncommon presentations of mesenteric fibromatosis and risk factors aids in early diagnosis, management, and treatment. Importantly, this also aids in the prevention of complications such as intestinal obstruction, bowel ischemia, and fistula formation.","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-04-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140679100","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Fever and Increased Gastrointestinal Uptake on Positron Emission Tomography after Anti-Tumour Necrosis Factor Therapy: A Case Report of Whipple’s Disease 抗肿瘤坏死因子治疗后发热和正电子发射断层扫描胃肠道摄取增加:Whipple病病例报告
IF 0.6 Q3 Medicine Pub Date : 2024-04-20 DOI: 10.1159/000538462
Arteen Arzivian, Brett Jones, Fredrick Joshua, Miriam Paul, Thomas Lynch, Martin Brown, Robin Gasiorowski
Abstract Introduction Whipple’s disease is a rare condition that can present with atypical and non-specific features requiring a high index of suspicion for diagnosis. Case Presentation We present a case of a man in his 40s with peripheral arthritis and bilateral sacro-ileitis for 4–5 years that was treated with an anti-tumour necrosis factor therapy, which led to worsening of his symptoms, elevation of the inflammatory markers, and the development of fever, night sweats, anorexia, and a significant weight loss. The patient had no abdominal pain, diarrhoea, or other gastrointestinal symptoms. An FDG-PET scan showed increased uptake in the stomach and caecum. Endoscopic examination showed inflammatory changes in the stomach and normal mucosa of the duodenum, jejunum, terminal ileum, caecum, and colon. Histopathology was inconclusive, but the diagnosis was confirmed with Tropheryma whipplei PCR testing. He had no neurological symptoms, but cerebrospinal fluid Tropheryma whipplei PCR was positive. He was treated with intravenous ceftriaxone 2 g daily for 4 weeks, followed by trimethoprim/sulfamethoxazole 160/800 mg twice daily for 1 year with close monitoring and follow-up. Conclusion This case presents an atypical and challenging presentation of Whipple’s disease and the importance of proactive testing for neurological involvement.
摘要 引言 惠普尔病是一种罕见疾病,可表现为非典型和非特异性特征,需要高度怀疑才能确诊。病例介绍 我们接诊了一例 40 多岁的男性患者,他患有外周关节炎和双侧骶尾部炎症长达 4-5 年之久,在接受抗肿瘤坏死因子治疗后,症状加重,炎症标志物升高,并出现发热、盗汗、厌食和体重明显下降。患者没有腹痛、腹泻或其他胃肠道症状。FDG-PET 扫描显示胃和盲肠的摄取量增加。内窥镜检查显示胃部有炎症变化,十二指肠、空肠、回肠末端、盲肠和结肠粘膜正常。组织病理学检查没有得出结论,但通过 Tropheryma whipplei PCR 检测确诊。他没有神经系统症状,但脑脊液 Tropheryma whipplei PCR 呈阳性。他接受了静脉注射头孢曲松治疗,每天 2 克,连续 4 周,随后使用三甲双胍/磺胺甲噁唑 160/800 毫克,每天两次,连续 1 年,并进行了密切监测和随访。结论 本病例是威普尔氏病的一个非典型和具有挑战性的表现,以及对神经系统受累进行前瞻性检测的重要性。
{"title":"Fever and Increased Gastrointestinal Uptake on Positron Emission Tomography after Anti-Tumour Necrosis Factor Therapy: A Case Report of Whipple’s Disease","authors":"Arteen Arzivian, Brett Jones, Fredrick Joshua, Miriam Paul, Thomas Lynch, Martin Brown, Robin Gasiorowski","doi":"10.1159/000538462","DOIUrl":"https://doi.org/10.1159/000538462","url":null,"abstract":"Abstract Introduction Whipple’s disease is a rare condition that can present with atypical and non-specific features requiring a high index of suspicion for diagnosis. Case Presentation We present a case of a man in his 40s with peripheral arthritis and bilateral sacro-ileitis for 4–5 years that was treated with an anti-tumour necrosis factor therapy, which led to worsening of his symptoms, elevation of the inflammatory markers, and the development of fever, night sweats, anorexia, and a significant weight loss. The patient had no abdominal pain, diarrhoea, or other gastrointestinal symptoms. An FDG-PET scan showed increased uptake in the stomach and caecum. Endoscopic examination showed inflammatory changes in the stomach and normal mucosa of the duodenum, jejunum, terminal ileum, caecum, and colon. Histopathology was inconclusive, but the diagnosis was confirmed with Tropheryma whipplei PCR testing. He had no neurological symptoms, but cerebrospinal fluid Tropheryma whipplei PCR was positive. He was treated with intravenous ceftriaxone 2 g daily for 4 weeks, followed by trimethoprim/sulfamethoxazole 160/800 mg twice daily for 1 year with close monitoring and follow-up. Conclusion This case presents an atypical and challenging presentation of Whipple’s disease and the importance of proactive testing for neurological involvement.","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-04-20","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140679958","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Two Cases of a Portal Annular Pancreas in Patients Undergoing Pancreaticoduodenectomy 接受胰十二指肠切除术的两例门静脉环状胰腺患者
IF 0.6 Q3 Medicine Pub Date : 2024-04-16 DOI: 10.1159/000538370
Mineto Ohta, Keisuke Fukushima, Hiroyasu Nishimaki, Takayuki Morita, Yuta Ito, Kenji Namiki
Abstract Introduction Portal annular pancreas (PAP) is a congenital anomaly resulting from aberrant fusion of the ventral and dorsal pancreatic buds around the portal vein (PV). PAP was classified into three types by Joseph et al., based on the location of the main pancreatic duct around the PV. The presence of PAP is important for the surgical procedure because it is associated with the postoperative pancreatic fistula. There are no standardized surgical procedures of resection and reconstruction for PAP. Case Presentation We report 2 cases of subtotal stomach-preserving pancreatoduodenectomy in patients with PAP. One case of PAP was discovered coincidentally intraoperatively, and the other case was diagnosed before surgery. The first case was an 84-year-old male patient who underwent surgery for distal bile duct cancer. PAP was noticed intraoperatively when the uncinate process of the pancreas was detached from behind the PV. The second case was an 84-year-old female patient who also underwent surgery for distal bile duct cancer. We recognized PAP from preoperative computed tomography images. In both cases, the ductal anatomy was consistent with type IIIA PAP, and the dorsal pancreas was resected using a stapling device. During the postoperative period, there was no clinically relevant postoperative pancreatic fistula. Conclusion PAP is rarely encountered intraoperatively; however, it is important to recognize it before surgery and take it into consideration when deciding upon the procedures for resection and reconstruction.
摘要 引言 门环状胰腺(PAP)是门静脉(PV)周围腹侧和背侧胰腺芽异常融合导致的先天性畸形。Joseph 等人根据主胰管在门静脉周围的位置将 PAP 分为三种类型。PAP 的存在对手术过程非常重要,因为它与术后胰瘘有关。目前还没有针对 PAP 进行切除和重建的标准化手术程序。病例介绍 我们报告了两例为 PAP 患者实施的保胃胰十二指肠次全切除术。其中一例在术中偶然发现 PAP,另一例在术前确诊。第一例患者是一名 84 岁的男性,因远端胆管癌接受手术。术中,当胰腺钩突从PV后方分离时,发现了PAP。第二个病例是一名 84 岁的女性患者,她也因远端胆管癌接受了手术。我们从术前计算机断层扫描图像中识别出了 PAP。两例患者的胰管解剖结构均符合 IIIA 型 PAP,我们使用订书机切除了胰背。术后未出现与临床相关的胰瘘。结论 PAP 在术中极少出现,但术前识别 PAP 并在决定切除和重建手术时将其考虑在内非常重要。
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Case Reports in Gastroenterology
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