Pancreaticopleural fistula in a Thai boy with SPINK1 c.101A>G substitution variant-related chronic pancreatitis: a case report and literature review.

IF 0.4 4区医学 Q4 MEDICINE, RESEARCH & EXPERIMENTAL Asian Biomedicine Pub Date : 2022-04-01 DOI:10.2478/abm-2022-0012

Chomanad Chittchang, Nisa Netinatsunton, Supika Kritsaneepaiboon

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Abstract

Background: Chronic pancreatitis is the most common etiology of pancreaticopleural fistula (PPF) in children, and underlying genetic variations are now widely known, accounting for most chronic pediatric pancreatitis.

Case report: We describe a case of previously undetected chronic pancreatitis and PPF with a SPINK1 variation in a 10-year-old Thai boy who presented with massive left pleural effusion. Magnetic resonance cholangiopancreatography (MRCP) revealed disruption of the pancreatic duct, which was communicating with a large pancreatic pseudocyst with mediastinal extension. The patient subsequently underwent endoscopic intervention with improved clinical symptoms. We also reviewed the imaging findings of 12 other reported cases of pediatric PPF.

Conclusions: Massive pleural effusion due to PPF can be an atypical manifestation in children with chronic pancreatitis. MRCP is the preferable imaging study for PPF due to the production of highly detailed images of pancreatic duct disruptions and anatomy, and the imaging is helpful to guide for appropriate treatment. Tests for genetic variation are also recommended in a child with chronic pancreatitis.

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1例泰国男孩伴有SPINK1 c.101A>G代换变异相关慢性胰腺炎的胰胸膜瘘:1例报告并文献复习

背景:慢性胰腺炎是儿童胰胸膜瘘(PPF)最常见的病因，潜在的遗传变异现已广为人知，占大多数慢性儿童胰腺炎的病因。病例报告:我们描述了一例以前未被发现的慢性胰腺炎和PPF与SPINK1变异在一个10岁的泰国男孩谁提出了大量的左胸腔积液。磁共振胰胆管造影(MRCP)显示胰管破裂，胰管与纵隔延伸的大胰腺假性囊肿相通。患者随后接受内镜介入治疗，临床症状有所改善。我们也回顾了其他12例儿科PPF的影像学表现。结论:PPF引起的大量胸腔积液可能是儿童慢性胰腺炎的不典型表现。MRCP是PPF的首选成像研究，因为它可以产生非常详细的胰管破坏和解剖图像，并且成像有助于指导适当的治疗。对于患有慢性胰腺炎的儿童，也建议进行遗传变异检测。

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来源期刊

Asian Biomedicine 医学-医学：研究与实验

CiteScore

1.20

自引率

0.00%

发文量

审稿时长

6-12 weeks

期刊介绍： Asian Biomedicine: Research, Reviews and News (ISSN 1905-7415 print; 1875-855X online) is published in one volume (of 6 bimonthly issues) a year since 2007. [...]Asian Biomedicine is an international, general medical and biomedical journal that aims to publish original peer-reviewed contributions dealing with various topics in the biomedical and health sciences from basic experimental to clinical aspects. The work and authorship must be strongly affiliated with a country in Asia, or with specific importance and relevance to the Asian region. The Journal will publish reviews, original experimental studies, observational studies, technical and clinical (case) reports, practice guidelines, historical perspectives of Asian biomedicine, clinicopathological conferences, and commentaries Asian biomedicine is intended for a broad and international audience, primarily those in the health professions including researchers, physician practitioners, basic medical scientists, dentists, educators, administrators, those in the assistive professions, such as nurses, and the many types of allied health professionals in research and health care delivery systems including those in training.