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Hemostasis with Metallic Stent for Multiple Metastatic Pancreatic Tumors Complicated with Hemobilia. 用金属支架为并发血友病的多发性转移性胰腺肿瘤止血。
IF 0.6 Q3 Medicine Pub Date : 2024-02-06 eCollection Date: 2024-01-01 DOI: 10.1159/000536221
Hiroyuki Ito, Yuji Omura, Tomonori Makuuchi, Tsubomi Chou, Ayano Ito, Ryutaro Fujimoto, Masashi Yokota, Shingo Tsuda, Junko Nagata, Shunji Hirose, Hisamichi Yoshii, Hideki Izumi, Tomoko Sugiyama, Takuma Tajiri, Takayoshi Suzuki

Introduction: Hemobilia, which refers to bleeding from the bile duct, is rare and difficult to treat. We report a case of successful hemostasis of a pancreatic tumor complicated by hemobilia.

Case presentation: A 76-year-old man was referred to our hospital with a pancreatic head tumor. Endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic ultrasonography-FNA were performed, and the patient was diagnosed with pancreatic metastasis of renal cell carcinoma. After discharge, the patient noted worsening jaundice and progressive anemia and was readmitted. ERCP reveals active bleeding from the duodenal papillae. The patient was placed on a fully covered metallic stent and discharged after confirming hemostasis.

Conclusion: Renal cell carcinoma is a tumor with abundant blood flow. If hemobilia occurs, bleeding from pancreatic metastatic tumors should be considered. Additionally, hemostasis using a fully covered metallic stent is useful for treating hemobilia in tumors.

导言:胆管出血是一种罕见且难以治疗的疾病。我们报告了一例胰腺肿瘤并发血友病的成功止血病例:一名 76 岁的男性因胰腺头部肿瘤转诊至我院。患者被诊断为肾细胞癌的胰腺转移。出院后,患者出现黄疸加重和进行性贫血,再次入院。ERCP 显示十二指肠乳头有活动性出血。患者被放置了一个全覆盖的金属支架,确认止血后出院:结论:肾细胞癌是一种血流丰富的肿瘤。结论:肾细胞癌是一种血流丰富的肿瘤,如果出现血肿,应考虑胰腺转移性肿瘤出血。此外,使用全覆盖金属支架止血也有助于治疗肿瘤出血。
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引用次数: 0
Chronic Liver Disease in Patients with Prolidase Deficiency: A Case Series. Prolidase 缺乏症患者的慢性肝病:病例系列
IF 0.6 Q3 Medicine Pub Date : 2024-02-01 eCollection Date: 2024-01-01 DOI: 10.1159/000536117
Harish Gopalakrishna, Bilal Asif, Anjali Rai, Hari S Conjeevaram, Maria Mironova, David E Kleiner, Alexandra F Freeman, Theo Heller

Introduction: Prolidase deficiency is a rare autosomal recessive disorder caused by variants in the PEPD gene. Patients usually have multi-organ involvement and a wide range of clinical features including recurrent skin ulcers, dysmorphic facial features, recurrent infections, intellectual disability, and splenomegaly. Studies have shown that patients with prolidase deficiency may have hepatic manifestations including hepatomegaly and abnormal liver enzymes. However, there is no detailed description of liver disease in this patient population.

Case presentation: Here, we present 3 patients with prolidase deficiency with varying extents of hepatic involvement.

Conclusion: Prolidase deficiency patients with liver disease should be followed up long term to understand more about the pathophysiology and the impact of liver disease on long-term outcomes.

简介Prolidase 缺乏症是一种罕见的常染色体隐性遗传疾病,由 PEPD 基因变异引起。患者通常有多器官受累和多种临床特征,包括反复皮肤溃疡、面部畸形、反复感染、智力障碍和脾肿大。研究显示,普罗利酶缺乏症患者可能有肝脏表现,包括肝肿大和肝酶异常。然而,目前还没有关于这类患者肝脏疾病的详细描述:在此,我们介绍了 3 例不同程度肝脏受累的普罗利酶缺乏症患者:结论:对患有肝病的普罗利酶缺乏症患者应进行长期随访,以进一步了解其病理生理学以及肝病对长期预后的影响。
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引用次数: 0
5-Aminosalicylic Acid-Induced Liver Injury in a Patient with Ulcerative Colitis: A Case Report. 溃疡性结肠炎患者的 5-氨基水杨酸肝损伤:病例报告。
IF 0.6 Q3 Medicine Pub Date : 2024-01-29 eCollection Date: 2024-01-01 DOI: 10.1159/000536097
Asuka Watanabe, Tsutomu Nishida, Naoto Osugi, Takao Kitanaka, Yutaro Minoura, Satoru Okabe, Naohiro Sakamoto, Yoshifumi Fujii, Aya Sugimoto, Dai Nakamatsu, Kengo Matsumoto, Masashi Yamamoto, Shiro Adachi, Koji Fukui

Introduction: Drug-induced liver injury (DILI) associated with 5-aminosalicylic acid (5-ASA) is a rare but potentially life-threatening adverse event.

Case presentation: We report the case of a 58-year-old woman with ulcerative colitis who developed DILI after initiating maintenance therapy with the multimatrix system 5-ASA. The patient presented with grade 4 liver enzyme elevation on day 98 after initiating 5-ASA and was admitted to the hospital. Blood tests revealed the mixed liver injury, and imaging studies showed no abnormalities except for mild lymph node enlargement. Liver biopsy revealed acute lobular hepatitis with interfacial activity. The patient's score on the International Autoimmune Hepatitis Group 1999 revised scoring system was a total score of 10, causing a suspicion for the diagnosis of autoimmune hepatitis. The DDW-J 2004 scale calculated a total score of six, indicating a high probability of DILI. We suspected DILI due to 5-ASA, and the 5-ASA formulations were discontinued. The patient was treated with ursodeoxycholic acid and neominophagen C, and her liver function gradually improved without steroid treatment. Finally, we definitively diagnosed DILI based on the pathological findings and clinical course after discontinuation of 5-ASA.

Conclusion: This case highlights the importance of monitoring liver function in patients receiving 5-ASA therapy.

导言:5-氨基水杨酸(5-ASA)引起的药物性肝损伤(DILI)是一种罕见但可能危及生命的不良事件:我们报告了一例患有溃疡性结肠炎的 58 岁女性患者,她在开始接受多矩阵系统 5-ASA 的维持治疗后出现了 DILI。患者在开始使用 5-ASA 后的第 98 天出现肝酶 4 级升高并入院治疗。血液检查显示为混合性肝损伤,影像学检查显示除轻度淋巴结肿大外无其他异常。肝活检显示患者患有急性小叶性肝炎,并伴有界面活性。患者在国际自身免疫性肝炎小组 1999 年修订的评分系统中的总分为 10 分,因此怀疑诊断为自身免疫性肝炎。DDW-J 2004评分标准计算出的总分为6分,表明DILI的可能性很高。我们怀疑 DILI 是由 5-ASA 引起的,因此停用了 5-ASA 制剂。患者接受熊去氧胆酸和新诺明 C 治疗后,肝功能逐渐改善,无需类固醇治疗。最后,根据病理结果和停用 5-ASA 后的临床表现,我们最终确诊为 DILI:本病例强调了对接受 5-ASA 治疗的患者进行肝功能监测的重要性。
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引用次数: 0
Incarcerated Small Bowel Herniation in a Stoma Mimicking Sigmoid End Colostomy Prolapse. 模仿乙状结肠造口术末端脱垂的造口嵌顿小肠疝。
IF 0.6 Q3 Medicine Pub Date : 2024-01-19 eCollection Date: 2024-01-01 DOI: 10.1159/000535988
Kaoru Abe, Daisuke Yamai, Chihiro Katsumi, Manabu Oyamatsu, Kenji Sato

Introduction: A stoma prolapse is easy to diagnose by visual examination, and it rarely incarcerates. Therefore, manual reduction is usually performed as soon as the diagnosis is made. In this report, we describe a case of stoma prolapse that could not be reduced manually and ruptured because an incarcerated parastomal hernia occurred in the stoma, mimicking stoma prolapse.

Case presentation: A 66-year-old woman underwent total hysterectomy, bilateral salpingo-oophorectomy, pelvic and para-aortic lymphadenectomy, omentectomy, resection of dissemination, and low anterior resection with formation of a sigmoid end colostomy for endometrial cancer with infiltration of the rectum. Fourteen months after the initial operation, she presented with stoma prolapse and multiple episodes of vomiting. The prolapsed stoma was 20 cm in length, appeared swollen and edematous, and was somewhat firm. Although it looked viable, some of the mucosa was darkish red, indicating congestion. Therefore, the diagnosis was sigmoid end colostomy prolapse with an ischemic component. An attempt at manual reduction resulted in rupture, so an emergency laparotomy was performed. Intraoperatively, we found that the ileum was incarcerated in the aperture created where the colostomy had been formed. When the incarcerated ileum was released, the stoma prolapse could be reduced easily. The end colostomy was refashioned in the left upper quadrant of the abdomen.

Conclusion: An incarcerated parastomal hernia can mimic stoma prolapse. If the findings differ from those of typical stoma prolapse, imaging should be performed to confirm whether another clinical entity is involved in the stoma prolapse.

介绍:造口脱垂很容易通过肉眼检查确诊,而且很少发生嵌顿。因此,一旦确诊,通常会立即进行人工减张术。在本报告中,我们描述了一例造口脱垂病例,由于造口内发生了嵌顿性旁疝,模仿造口脱垂而无法进行人工减张并导致破裂:一位 66 岁的妇女因子宫内膜癌伴有直肠浸润而接受了全子宫切除术、双侧输卵管切除术、盆腔和主动脉旁淋巴结切除术、卵巢切除术、播散切除术和低位前切除术,并形成乙状结肠造口。初次手术后 14 个月,她出现造口脱垂和多次呕吐。脱垂的造口长 20 厘米,看起来肿胀、水肿,而且有些坚硬。虽然看起来还活着,但部分粘膜呈暗红色,表明有充血现象。因此,诊断结果是乙状结肠末端造口脱垂,并伴有缺血成分。尝试用手将结肠切除后发现结肠破裂,于是进行了紧急开腹手术。术中,我们发现回肠嵌顿在结肠造口形成的孔道中。松开嵌顿的回肠后,造口脱垂就很容易缩小了。结肠造口末端在左上腹重新成形:结论:腹腔旁疝嵌顿可模拟造口脱垂。如果检查结果与典型的造口脱垂不同,则应进行造影检查,以确认造口脱垂是否涉及其他临床实体。
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引用次数: 0
Trichobezoar Found Accidently while Diagnosing Resistance to Thyroid Hormone. 在诊断甲状腺激素抗药性时意外发现三叶虫
IF 0.6 Q3 Medicine Pub Date : 2024-01-19 eCollection Date: 2024-01-01 DOI: 10.1159/000534548
Kinga Szopińska, Karolina Tracz, Żaneta Malczyk, Anna Jarzumbek, Andrzej Grabowski, Katarzyna Bąk-Drabik

Introduction: Bezoars are masses of indigestible foreign material in the gastrointestinal tract, usually in the stomach. These materials could be indigestible fruits, hair, milk products, or tablets. In children, the most common type of bezoar is trichobezoar (formed from hair).

Case presentation: We describe a female patient who has been complaining about deterioration of mood, collapse without losing consciousness, scotomas, and cardiac arrhythmia for 2 years. Based on the results of thyroid hormone, resistance to thyroid hormone (RTH) was suspected. Physical examination during hospitalization revealed a palpable upper abdominal mass. Several diagnostic examinations were performed. The abdominal ultrasound showed acoustic shadowing caused by a pathological structure in the upper abdomen. Therefore, the contrast X-ray of the digestive tract revealed a deficit of contrast with an irregular shape in the stomach body and the pylorus region. Due to these results, a gastroscopy was performed, which revealed a large trichobezoar of the stomach. The trichobezoar was surgically removed without complications.

Conclusion: The case presented shows that these nonspecific symptoms and laboratory test suggesting RTH require multi-path diagnostics and the cooperation of many specialists, ultimately giving a surprising diagnosis. It is crucial to interpret diagnostic examinations with regard to the patient's physical condition. Diagnosis of trichobezoar requires a detailed search of causes to avoid another incident.

简介牛粪是胃肠道中难以消化的异物块,通常位于胃中。这些物质可能是难以消化的水果、头发、奶制品或药片。在儿童中,最常见的是三叶虫(由头发形成):病例介绍:我们描述了一名女性患者,她主诉情绪恶化、昏倒但不失去知觉、视力模糊和心律失常已有两年。根据甲状腺激素检测结果,怀疑患者存在甲状腺激素抵抗(RTH)。住院期间的体格检查发现了一个可触及的上腹部肿块。对其进行了多项诊断性检查。腹部超声波检查显示,上腹部的病理结构引起了声影。因此,消化道造影剂 X 光检查显示胃体和幽门区域造影剂不足,形状不规则。根据上述结果,医生进行了胃镜检查,发现胃部有一个巨大的三叶虫。手术切除了该三叶虫,未出现并发症:本病例表明,这些非特异性症状和实验室检查提示的 RTH 需要多途径诊断和多位专家的合作,最终才能得出令人惊讶的诊断结果。根据患者的身体状况解释诊断检查至关重要。诊断三叶虫病需要详细查找病因,以避免再次发生。
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引用次数: 0
Gastric Lipomas: A Case Series and Review of the Literature. 胃脂肪瘤:病例系列和文献综述。
IF 0.6 Q3 Medicine Pub Date : 2024-01-09 eCollection Date: 2024-01-01 DOI: 10.1159/000534973
Simone A Jarrett, Sahana Tito, Matthew Chan, Dominic E Jarrett, Kevin B Lo, Richard DePalma

Introduction: The purpose of this case series was to review a rare subset of tumors known as gastric lipomas, which are typically found incidentally. The motivation for this study arose from the identification of 2 cases within our institution in a short period.

Case presentation: The study involved a review of the diagnosis and management of 2 patients presenting with gastric lipomas at our institution after symptoms of gastrointestinal bleeding. With the advent of new radiologic investigations such as computed tomography and magnetic resonance imaging and advances in endoscopy, there are new approaches to identifying and managing these tumors. On further evaluation of the literature, we found that despite the availability of minimally invasive endoscopic techniques such as mucosal resection and submucosal dissection in the setting of large tumors, most patients tend to have to resort to surgical management.

Conclusion: This case series underscores the rarity of gastric lipomas and their often-incidental discovery. Further investigation into endoscopic approaches for managing these tumors is needed, and additionally, there is a need to explore a potential association between gastric lipomas and malignancy, as chronic inflammation of the overlying mucosa may play a significant role.

导言本病例系列旨在回顾胃脂肪瘤这一罕见的肿瘤亚群,这种肿瘤通常是偶然发现的。本研究的动机源于我院在短期内发现的两例病例:病例介绍:本研究回顾了我院 2 例因消化道出血症状而就诊的胃脂肪瘤患者的诊断和治疗情况。随着计算机断层扫描和磁共振成像等新型放射检查手段的出现以及内窥镜检查的进步,识别和处理这些肿瘤有了新的方法。在对文献进行进一步评估后,我们发现,尽管有微创内镜技术,如粘膜切除术和粘膜下剥离术,但对于大肿瘤,大多数患者还是不得不采用手术治疗:本系列病例强调了胃脂肪瘤的罕见性,以及发现时的偶然性。此外,还需要探讨胃脂肪瘤与恶性肿瘤之间的潜在联系,因为上覆黏膜的慢性炎症可能起着重要作用。
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引用次数: 0
A Case of Immune-Related Aseptic Meningitis during Atezolizumab plus Bevacizumab for Hepatocellular Carcinoma. 阿特珠单抗联合贝伐单抗治疗肝细胞癌期间的一例免疫相关性无菌性脑膜炎病例。
IF 0.6 Q3 Medicine Pub Date : 2024-01-04 eCollection Date: 2024-01-01 DOI: 10.1159/000535476
Hiroki Kawanaka, Kazuto Tajiri, Nozomu Muraishi, Aiko Murayama, Takamasa Nukui, Ichiro Yasuda

Introduction: Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first case of atezolizumab-induced aseptic meningitis, which occurred during treatment for advanced hepatocellular carcinoma (HCC).

Case presentation: A 74-year-old woman diagnosed with advanced HCC and treated with first-line atezolizumab plus bevacizumab developed anorexia, fatigue, and fever, after three treatment cycles. Cerebrospinal fluid examination showed slightly increased cell count and protein level but no infection or malignancy. Contrast enhancement along the cerebral sulcus was evident in contrast-enhanced magnetic resonance imaging, and the patient was diagnosed with aseptic meningitis associated with atezolizumab. Steroid therapy soon improved her clinical symptoms, and the contrast enhancement along the cerebral sulcus disappeared.

Conclusion: Clinicians should monitor to avoid serious immune-related adverse events, such as aseptic meningitis, in patients during treatment of HCC with immune checkpoint inhibitors and make the diagnosis as soon as possible.

导言:免疫检查点抑制剂在治疗期间或治疗后有时会出现与免疫相关的不良事件。其中,无菌性脑膜炎是一种罕见的严重并发症。我们报告了首例阿特珠单抗诱发无菌性脑膜炎的病例,该病例发生在晚期肝细胞癌(HCC)的治疗过程中:一名被诊断为晚期肝细胞癌并接受一线阿特珠单抗加贝伐单抗治疗的74岁女性,在三个治疗周期后出现厌食、乏力和发热。脑脊液检查显示细胞计数和蛋白水平略有升高,但未发现感染或恶性肿瘤。对比增强磁共振成像显示沿脑沟的对比增强明显,患者被诊断为与阿特珠单抗相关的无菌性脑膜炎。类固醇治疗很快改善了她的临床症状,沿脑沟的造影剂强化也消失了:临床医生在使用免疫检查点抑制剂治疗 HCC 期间应进行监测,避免患者出现严重的免疫相关不良事件,如无菌性脑膜炎,并尽快做出诊断。
{"title":"A Case of Immune-Related Aseptic Meningitis during Atezolizumab plus Bevacizumab for Hepatocellular Carcinoma.","authors":"Hiroki Kawanaka, Kazuto Tajiri, Nozomu Muraishi, Aiko Murayama, Takamasa Nukui, Ichiro Yasuda","doi":"10.1159/000535476","DOIUrl":"10.1159/000535476","url":null,"abstract":"<p><strong>Introduction: </strong>Immune checkpoint inhibitors are sometimes associated with immune-related adverse events during or after treatment. Among these, aseptic meningitis is a rare and serious complication. We report the first case of atezolizumab-induced aseptic meningitis, which occurred during treatment for advanced hepatocellular carcinoma (HCC).</p><p><strong>Case presentation: </strong>A 74-year-old woman diagnosed with advanced HCC and treated with first-line atezolizumab plus bevacizumab developed anorexia, fatigue, and fever, after three treatment cycles. Cerebrospinal fluid examination showed slightly increased cell count and protein level but no infection or malignancy. Contrast enhancement along the cerebral sulcus was evident in contrast-enhanced magnetic resonance imaging, and the patient was diagnosed with aseptic meningitis associated with atezolizumab. Steroid therapy soon improved her clinical symptoms, and the contrast enhancement along the cerebral sulcus disappeared.</p><p><strong>Conclusion: </strong>Clinicians should monitor to avoid serious immune-related adverse events, such as aseptic meningitis, in patients during treatment of HCC with immune checkpoint inhibitors and make the diagnosis as soon as possible.</p>","PeriodicalId":9614,"journal":{"name":"Case Reports in Gastroenterology","volume":null,"pages":null},"PeriodicalIF":0.6,"publicationDate":"2024-01-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10766420/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"139377233","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
Bladder-Adherent Primary Appendiceal Carcinoma Masquerading as a Carpeting Rectal Lesion Detected by a Fecal Immunochemical Test: A Case Report. 粪便免疫化学检验发现的膀胱粘连性原发性阑尾癌伪装成地毯状直肠病变:病例报告。
IF 0.6 Q3 Medicine Pub Date : 2024-01-04 eCollection Date: 2024-01-01 DOI: 10.1159/000535273
Nishigandha Burute, Olexiy Aseyev, Fauzia Hasan, Erika Allard-Ihala, Walid Shahrour, Ahmed Kotb, William Harris, Radu Rozenberg

Introduction: Primary appendiceal carcinoma is rare and comprises up to 1% of all colorectal malignancies. Its invasion into adjacent organs, such as the bladder and rectum, especially as a presenting characteristic, is even less common.

Case presentation: A 75-year-old asymptomatic male tested positive on a screening fecal immunochemical test (FIT). Colonoscopy showed a rectosigmoid tumor and normal appendiceal orifice. Staging MRI surprisingly showed that the cancer was, in fact, of appendiceal origin, coursed posteriorly to invade the rectosigmoid and form adhesions with the urinary bladder. Staging CT did not show metastatic disease. Low anterior resection, en bloc appendectomy, and right hemicolectomy were performed along with cystectomy and ileal conduit. Hematoxylin and eosin stains showed appendiceal adenocarcinoma invading through the appendiceal wall into the rectal muscularis and submucosa. Features of neuroendocrine carcinoma were not identified on immunohistochemistry. This was a colonic type of adenocarcinoma of the appendix.

Conclusion: This is a rare case of appendiceal carcinoma invading the rectum and presenting as a positive screening fecal immunochemical test in an asymptomatic individual. We effectively demonstrate the use of preoperative MRI to identify the appendiceal origin of the tumor, as well as to demonstrate the extent of tumor spread, which assisted with operative management and treatment planning.

简介:原发性阑尾癌非常罕见,在所有结直肠恶性肿瘤中占 1%。其侵犯邻近器官,如膀胱和直肠,尤其是作为一种表现特征,更是少见:一名 75 岁的无症状男性在粪便免疫化学检验(FIT)筛查中呈阳性。结肠镜检查显示直肠乙状结肠肿瘤,阑尾口正常。分期核磁共振成像(MRI)令人惊讶地显示,癌症实际上源自阑尾,向后方发展,侵犯了直乙状结肠,并与膀胱形成粘连。分期 CT 未显示转移性疾病。患者接受了低位前切除术、阑尾全切术、右半结肠切除术以及膀胱切除术和回肠导管术。苏木精和伊红染色显示,阑尾腺癌通过阑尾壁侵入直肠肌层和粘膜下层。免疫组化未发现神经内分泌癌的特征。这是一种结肠型阑尾腺癌:这是一例罕见的阑尾癌侵犯直肠并在无症状者的粪便免疫化学检查中呈阳性的病例。我们有效地展示了术前磁共振成像在确定肿瘤来源于阑尾以及显示肿瘤扩散范围方面的应用,这有助于手术管理和治疗计划的制定。
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引用次数: 0
Down Syndrome Patients with Congenital Portosystemic Shunts: A Case Report and Review. 患有先天性门静脉分流的唐氏综合征患者:病例报告与回顾
IF 0.6 Q3 Medicine Pub Date : 2023-12-18 eCollection Date: 2023-01-01 DOI: 10.1159/000535477
Brian Nohomovich, Minh H N Nguyen, Joseph Fakhoury, Russell C Cameron, Theotonius Gomes

Introduction: Down syndrome is due to trisomy 21 and is characterized by intellectual disability, dysmorphic facial features, congenital malformations, and gastrointestinal abnormalities. There is an increased appreciation of congenital portosystemic shunts in Down syndrome patients. Congenital portosystemic shunts have been associated with many defects in body systems, including cardiac, metabolic, and neurological.

Case presentation: Herein, we describe a portosystemic shunt in a Down syndrome patient that resulted in hyperammonemia with altered mental status and choreiform movements. Computed tomography angiography of the abdomen and pelvis identified a connection between the right portal vein and inferior vena cava. An 18 mm Amplatzer PFO closure device was placed within the congenital shunt, significantly improving symptoms. The patient has no sequelae from the related shunt or the device at the 2-year follow-up. We extensively reviewed the literature and identified cases of portosystemic shunts in Down syndrome patients. Shunts can either be extrahepatic or intrahepatic and are classified by vasculature connections.

Conclusion: From our literature review and case presentation, we identify other conditions in patients, including cardiac and gastrointestinal defects. We then review the available treatment options, whether observation or surgical, depending on the patient's clinical picture.

简介唐氏综合征是由 21 三体综合征引起的,以智力障碍、面部畸形、先天性畸形和胃肠道异常为特征。唐氏综合征患者先天性门静脉分流的发生率越来越高。先天性门静脉分流与许多身体系统缺陷有关,包括心脏、代谢和神经系统:在此,我们描述了一名唐氏综合征患者的门静脉分流术,该分流术导致了高氨血症,并伴有精神状态改变和舞蹈样运动。腹部和盆腔的计算机断层扫描血管造影确定了右门静脉和下腔静脉之间的连接。在先天性分流处放置了一个18毫米的Amplatzer PFO闭合装置,明显改善了症状。在两年的随访中,患者没有因相关分流或装置而留下后遗症。我们广泛查阅了相关文献,发现了唐氏综合征患者门静脉分流的病例。分流可以是肝外分流,也可以是肝内分流,并根据血管连接情况进行分类:通过文献回顾和病例介绍,我们发现了患者的其他病症,包括心脏和胃肠道缺陷。然后,我们根据患者的临床表现,回顾了现有的治疗方案,无论是观察治疗还是手术治疗。
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引用次数: 0
Dapsone-Induced Granulomatous Cholestatic Hepatitis Unmasked by Steroid Taper: A Case Report 类固醇减量掩盖了多潘松诱发的肉芽肿性胆汁淤积性肝炎:病例报告
IF 0.6 Q3 Medicine Pub Date : 2023-12-11 DOI: 10.1159/000534818
Shahana Prakash, Matthew Gosse, Tomohiro Tanaka
Abstract Introduction Dapsone is known to cause drug-induced liver injury (DILI) but can rarely induce the formation of hepatic granulomas. We describe a patient with jaundice who demonstrated granulomas on liver biopsy in response to dapsone. Her symptoms were only evident once steroids, used to also treat her pyoderma gangrenosum, had been tapered. Case Presentation In this case, a 67-year-old female was hospitalized due to 1 day of jaundice. She had started dapsone and prednisone concurrently 7 weeks ago to treat her pyoderma gangrenosum. Steroids were discontinued 4 days prior to symptoms. Her laboratories were notable for newly elevated alkaline phosphatase (756 U/L), aspartate transaminase (199 U/L), alanine transaminase (273 U/L), and total bilirubin (12.6 mg/dL). Dapsone was held due to suspicion for DILI. A liver biopsy was performed and disclosed non-necrotizing hepatic granulomas. After infectious and autoimmune causes were excluded, dapsone was determined to be the cause of her hepatic granulomas. Her bilirubin and liver enzymes steadily normalized over the next 4 weeks following discontinuation of dapsone. Conclusion Thus, dapsone-related liver injury may present following a steroid taper if dapsone and steroids had been initially started together. Hepatic granulomas, though rare, can be seen when dapsone causes DILI.
氨苯砜可引起药物性肝损伤(DILI),但很少引起肝肉芽肿的形成。我们描述了一位黄疸患者,他在肝活检中表现出肉芽肿,对氨苯砜的反应。她的症状只有在类固醇(也用于治疗坏疽性脓皮病)逐渐减少后才明显。本例患者为67岁女性,因黄疸1天住院。7周前,她开始同时使用氨苯砜和强的松治疗坏疽性脓皮病。在出现症状前4天停用类固醇。她的实验室新发现碱性磷酸酶(756 U/L)、天冬氨酸转氨酶(199 U/L)、丙氨酸转氨酶(273 U/L)和总胆红素(12.6 mg/dL)升高。Dapsone因涉嫌DILI而被拘留。肝活检显示非坏死性肝肉芽肿。在排除感染和自身免疫性原因后,确定氨苯砜是她肝肉芽肿的原因。她的胆红素和肝酶在停药后的4周内稳定恢复正常。因此,如果最初同时使用氨苯砜和类固醇,那么在类固醇逐渐减少后可能出现氨苯砜相关的肝损伤。肝肉芽肿,虽然罕见,可以看到当氨苯砜引起DILI。
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引用次数: 0
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