Pancreaticopleural fistula in children with chronic pancreatitis: a case report and literature review

BackgroundPancreaticopleural fistula (PPF) is a very rare and critical complication of pancreatitis in children. The majority of publications relevant to PPF are case reports. No pooled analyses of PPF cases are available. Little is known about the pathogenesis and optimal therapeutic schedule. The...

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Veröffentlicht in:BMC pediatrics 2020-06, Vol.20 (1), p.274-274, Article 274
Hauptverfasser: Zhang, Jia-yu, Deng, Zhao-hui, Gong, Biao
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description BackgroundPancreaticopleural fistula (PPF) is a very rare and critical complication of pancreatitis in children. The majority of publications relevant to PPF are case reports. No pooled analyses of PPF cases are available. Little is known about the pathogenesis and optimal therapeutic schedule. The purpose of this study was to identify the pathogenesis and optimal therapeutic schedule of PPF in children.Case presentationThe patient was a 13-year-old girl who suffered from intermittent chest tightness and dyspnea for more than 3months; she was found to have chronic pancreatitis complicated by PPF. The genetic screening revealed SPINK1 mutation. She was treated with endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic retrograde pancreatic drainage (ERPD); her symptoms improved dramatically after the procedures.ConclusionsPPF is a rare pancreatic complication in children and causes significant pulmonary symptoms that can be misdiagnosed frequently. PPF in children is mainly associated with chronic pancreatitis (CP); therefore, we highlight the importance of genetic testing. Endoscopic treatment is recommended when conservative treatment is ineffective.
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The majority of publications relevant to PPF are case reports. No pooled analyses of PPF cases are available. Little is known about the pathogenesis and optimal therapeutic schedule. The purpose of this study was to identify the pathogenesis and optimal therapeutic schedule of PPF in children.Case presentationThe patient was a 13-year-old girl who suffered from intermittent chest tightness and dyspnea for more than 3months; she was found to have chronic pancreatitis complicated by PPF. The genetic screening revealed SPINK1 mutation. She was treated with endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic retrograde pancreatic drainage (ERPD); her symptoms improved dramatically after the procedures.ConclusionsPPF is a rare pancreatic complication in children and causes significant pulmonary symptoms that can be misdiagnosed frequently. PPF in children is mainly associated with chronic pancreatitis (CP); therefore, we highlight the importance of genetic testing. Endoscopic treatment is recommended when conservative treatment is ineffective.</description><identifier>ISSN: 1471-2431</identifier><identifier>EISSN: 1471-2431</identifier><identifier>DOI: 10.1186/s12887-020-02174-x</identifier><identifier>PMID: 32493299</identifier><language>eng</language><publisher>LONDON: Springer Nature</publisher><subject>Abdomen ; Adolescent ; Case Report ; Case reports ; Child ; Cholangiopancreatography, Endoscopic Retrograde ; Chronic pancreatitis ; Cysts ; Dyspnea ; Endoscopy ; Female ; Fistula ; Fistulas ; Genetic aspects ; Genetic testing ; Health aspects ; Hospitalization ; Humans ; Life Sciences &amp; Biomedicine ; Medical imaging ; Mutation ; Pain ; Pancreas ; Pancreatic Fistula - diagnosis ; Pancreatic Fistula - etiology ; Pancreaticopleural fistula ; Pancreatitis ; Pancreatitis, Chronic - complications ; Pancreatitis, Chronic - diagnosis ; Pancreatitis, Chronic - therapy ; Pathogenesis ; Pediatrics ; Pleural Diseases - diagnosis ; Pleural Diseases - etiology ; Pleural effusion ; Science &amp; Technology ; Surgery ; Teenage girls ; Trypsin Inhibitor, Kazal Pancreatic</subject><ispartof>BMC pediatrics, 2020-06, Vol.20 (1), p.274-274, Article 274</ispartof><rights>COPYRIGHT 2020 BioMed Central Ltd.</rights><rights>2020. 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The majority of publications relevant to PPF are case reports. No pooled analyses of PPF cases are available. Little is known about the pathogenesis and optimal therapeutic schedule. The purpose of this study was to identify the pathogenesis and optimal therapeutic schedule of PPF in children.Case presentationThe patient was a 13-year-old girl who suffered from intermittent chest tightness and dyspnea for more than 3months; she was found to have chronic pancreatitis complicated by PPF. The genetic screening revealed SPINK1 mutation. She was treated with endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic retrograde pancreatic drainage (ERPD); her symptoms improved dramatically after the procedures.ConclusionsPPF is a rare pancreatic complication in children and causes significant pulmonary symptoms that can be misdiagnosed frequently. PPF in children is mainly associated with chronic pancreatitis (CP); therefore, we highlight the importance of genetic testing. 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Technology</topic><topic>Surgery</topic><topic>Teenage girls</topic><topic>Trypsin Inhibitor, Kazal Pancreatic</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Zhang, Jia-yu</creatorcontrib><creatorcontrib>Deng, Zhao-hui</creatorcontrib><creatorcontrib>Gong, Biao</creatorcontrib><collection>Web of Science - Science Citation Index Expanded - 2020</collection><collection>Web of Science Core Collection</collection><collection>Science Citation Index Expanded</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health &amp; Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health &amp; Medical Complete (Alumni)</collection><collection>Health &amp; Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Access via ProQuest (Open Access)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>BMC pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Zhang, Jia-yu</au><au>Deng, Zhao-hui</au><au>Gong, Biao</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Pancreaticopleural fistula in children with chronic pancreatitis: a case report and literature review</atitle><jtitle>BMC pediatrics</jtitle><stitle>BMC PEDIATR</stitle><addtitle>BMC Pediatr</addtitle><date>2020-06-03</date><risdate>2020</risdate><volume>20</volume><issue>1</issue><spage>274</spage><epage>274</epage><pages>274-274</pages><artnum>274</artnum><issn>1471-2431</issn><eissn>1471-2431</eissn><abstract>BackgroundPancreaticopleural fistula (PPF) is a very rare and critical complication of pancreatitis in children. The majority of publications relevant to PPF are case reports. No pooled analyses of PPF cases are available. Little is known about the pathogenesis and optimal therapeutic schedule. The purpose of this study was to identify the pathogenesis and optimal therapeutic schedule of PPF in children.Case presentationThe patient was a 13-year-old girl who suffered from intermittent chest tightness and dyspnea for more than 3months; she was found to have chronic pancreatitis complicated by PPF. The genetic screening revealed SPINK1 mutation. She was treated with endoscopic retrograde cholangiopancreatography (ERCP) and endoscopic retrograde pancreatic drainage (ERPD); her symptoms improved dramatically after the procedures.ConclusionsPPF is a rare pancreatic complication in children and causes significant pulmonary symptoms that can be misdiagnosed frequently. PPF in children is mainly associated with chronic pancreatitis (CP); therefore, we highlight the importance of genetic testing. Endoscopic treatment is recommended when conservative treatment is ineffective.</abstract><cop>LONDON</cop><pub>Springer Nature</pub><pmid>32493299</pmid><doi>10.1186/s12887-020-02174-x</doi><tpages>8</tpages><oa>free_for_read</oa></addata></record>
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subjects Abdomen
Adolescent
Case Report
Case reports
Child
Cholangiopancreatography, Endoscopic Retrograde
Chronic pancreatitis
Cysts
Dyspnea
Endoscopy
Female
Fistula
Fistulas
Genetic aspects
Genetic testing
Health aspects
Hospitalization
Humans
Life Sciences & Biomedicine
Medical imaging
Mutation
Pain
Pancreas
Pancreatic Fistula - diagnosis
Pancreatic Fistula - etiology
Pancreaticopleural fistula
Pancreatitis
Pancreatitis, Chronic - complications
Pancreatitis, Chronic - diagnosis
Pancreatitis, Chronic - therapy
Pathogenesis
Pediatrics
Pleural Diseases - diagnosis
Pleural Diseases - etiology
Pleural effusion
Science & Technology
Surgery
Teenage girls
Trypsin Inhibitor, Kazal Pancreatic
title Pancreaticopleural fistula in children with chronic pancreatitis: a case report and literature review
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