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一名青少年患者以持续性呃逆和严重食管炎为表现的视神经脊髓炎谱系障碍

Neuromyelitis Optica Spectrum Disorder Manifested by Persistent Hiccups and Severe Esophagitis in an Adolescent Patient.

作者信息

Singh Priya, Garcia Reinaldo, Y Phadke Madhura

机构信息

From the Northeast Ohio Medical University, Rootstown, OH.

Division of Pediatric Gastroenterology, Hepatology and Nutrition, Akron Children's Hospital, Akron, OH.

出版信息

JPGN Rep. 2021 Aug 26;2(4):e117. doi: 10.1097/PG9.0000000000000117. eCollection 2021 Nov.

DOI:10.1097/PG9.0000000000000117
PMID:37206459
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10191573/
Abstract

Patients with persistent hiccups are often referred to gastrointestinal services. Hiccups lasting greater than 48 hours are classified as persistent, while those lasting longer than 2 months are termed intractable. The etiology of hiccups is broad and can include many organ systems. Here, we present the case of an 18-year-old male patient who presented to the emergency department with an 8-day history of nausea, emesis, and intermittent hiccups. The patient was admitted to the gastroenterology service and underwent workup including esophagogastroduodenoscopy and imaging. Esophagogastroduodenoscopy revealed severe distal esophagitis. He was started on high-dose proton pump inhibitor and sucralfate. During the course of hospital admission, the patient's intermittent hiccups worsened to become constant. Several medical therapies led to little improvement. We initially felt that the esophagitis was the etiology of the patient's symptoms, however, due to ongoing symptoms unresponsive to typical therapy, we obtained a neurological consult. Brain MRI revealed a dorsal medullary lesion. Subsequent work up confirmed the diagnosis of neuromyelitis optica spectrum disorder. Neuromyelitis optica spectrum disorder is a rare, serious, sometimes fatal autoimmune condition of the central nervous system. Early diagnosis and treatment are important predictors of future relapses. Our patient was started on high-dose intravenous steroids with rapid improvement in symptoms. He was discharged home on steroids, proton pump inhibitor and close neurology and gastroenterology follow up. This case outlines the importance of consideration of neurological causes of hiccups or other centrally mediated conditions with gastrointestinal manifestations, even when a concomitant gastrointestinal condition is present.

摘要

持续性呃逆的患者常被转诊至胃肠科。持续时间超过48小时的呃逆被归类为持续性呃逆,而持续时间超过2个月的则称为顽固性呃逆。呃逆的病因广泛,可涉及多个器官系统。在此,我们报告一例18岁男性患者,他因恶心、呕吐和间歇性呃逆8天就诊于急诊科。该患者被收入胃肠病科,并接受了包括食管胃十二指肠镜检查和影像学检查在内的评估。食管胃十二指肠镜检查显示严重的远端食管炎。患者开始接受高剂量质子泵抑制剂和硫糖铝治疗。在住院期间,患者的间歇性呃逆加重,变为持续性。多种药物治疗效果甚微。我们最初认为食管炎是患者症状的病因,然而,由于持续存在对典型治疗无反应的症状,我们请了神经科会诊。脑部磁共振成像显示延髓背侧病变。随后的检查确诊为视神经脊髓炎谱系障碍。视神经脊髓炎谱系障碍是一种罕见、严重、有时甚至致命的中枢神经系统自身免疫性疾病。早期诊断和治疗是未来复发的重要预测指标。我们的患者开始接受高剂量静脉注射类固醇治疗,症状迅速改善。他出院时带药包括类固醇、质子泵抑制剂,并需接受神经科和胃肠病科的密切随访。该病例强调了即使存在并发的胃肠疾病,也需考虑呃逆或其他具有胃肠道表现的中枢介导疾病的神经学病因的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/deae/10191573/5327a4703ced/pg9-2-e117-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/deae/10191573/9fc7ff1fee09/pg9-2-e117-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/deae/10191573/5327a4703ced/pg9-2-e117-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/deae/10191573/9fc7ff1fee09/pg9-2-e117-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/deae/10191573/5327a4703ced/pg9-2-e117-g002.jpg

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本文引用的文献

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Area postrema syndrome: a neurological presentation of nausea, vomiting and hiccups.最后区综合征:一种以恶心、呕吐和呃逆为表现的神经症状。
BMJ Case Rep. 2020 Nov 3;13(11):e238588. doi: 10.1136/bcr-2020-238588.
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Update on neuromyelitis optica spectrum disorder.视神经脊髓炎谱系疾病的最新进展。
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Isolated Area Postrema Syndrome Presenting as Intractable Nausea and Vomiting.以顽固性恶心和呕吐为表现的孤立性最后区综合征
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