Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab

Butsabong Lerkvaleekul,1 Suporn Treepongkaruna,2 Nichanan Ruangwattanapaisarn,3 Tharintorn Treesit,3 Soamarat Vilaiyuk11Division of Rheumatology, Department of Paediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 2Division of Gastroenterology, Department of P...

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Autores principales:	Lerkvaleekul B, Treepongkaruna S, Ruangwattanapaisarn N, Treesit T, Vilaiyuk S
Formato:	article
Lenguaje:	EN
Publicado:	Dove Medical Press 2019
Materias:	Vasculitis Gastrointestinal bleeding Tumor necrosis factor Infliximab Biologic therapy Medicine (General) R5-920
Acceso en línea:	https://doaj.org/article/db952d31e76f414a939f6076427d4299
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spelling	oai:doaj.org-article:db952d31e76f414a939f6076427d42992021-12-02T03:06:44ZRecurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab1177-5491https://doaj.org/article/db952d31e76f414a939f6076427d42992019-06-01T00:00:00Zhttps://www.dovepress.com/recurrent-ruptured-abdominal-aneurysms-in-polyarteritis-nodosa-success-peer-reviewed-article-BTThttps://doaj.org/toc/1177-5491Butsabong Lerkvaleekul,1 Suporn Treepongkaruna,2 Nichanan Ruangwattanapaisarn,3 Tharintorn Treesit,3 Soamarat Vilaiyuk11Division of Rheumatology, Department of Paediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 2Division of Gastroenterology, Department of Paediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 3Department of Diagnostic and Therapeutic Radiology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, ThailandAbstract: Systemic polyarteritis nodosa (PAN) is a rare form of necrotizing vasculitis in children. Recurrent episodes of abdominal aneurysm ruptures are uncommon and life-threatening condition in children. Failures of response to immunosuppressive medications and radiological intervention also lead to high mortality. Some reports suggested that tumor necrosis factor (TNF) might have role in the inflammation of this disease. After an English-language literature review, this is the first case report in children of recurrent abdominal-ruptured aneurysms with a failure of conventional therapy but successfully treated with anti-TNF-α monoclonal antibody. We herein describe a 9-year-old girl who presented with chronic abdominal pain, hypertension, and massive lower gastrointestinal bleeding. The disease was refractory to conventional treatment, including administration of a corticosteroid, cyclophosphamide, and intravenous immunoglobulin, and recurrent-ruptured aneurysms developed in the gastrointestinal tract. Arterial embolization during angiography resulted in temporary improvement of the gastrointestinal bleeding. Infliximab, a chimeric anti-tumor necrosis factor-α monoclonal antibody, was initiated and resulted in disease remission with resolution of the gastrointestinal bleeding and abdominal pain. Anti-TNF therapy might be another treatment option for refractory disease to prevent ongoing inflammation that could lead to aneurysmal dilatation or even rupture. However, early recognition of refractory disease and aggressive treatment in the early course of the disease are crucial to reduce morbidity and mortality.Keywords: vasculitis, gastrointestinal bleeding, tumor necrosis factor, infliximab, biologic therapy  Lerkvaleekul BTreepongkaruna SRuangwattanapaisarn NTreesit TVilaiyuk SDove Medical PressarticleVasculitisGastrointestinal bleedingTumor necrosis factorInfliximabBiologic therapyMedicine (General)R5-920ENBiologics: Targets & Therapy, Vol Volume 13, Pp 111-116 (2019)
institution	DOAJ
collection	DOAJ
language	EN
topic	Vasculitis Gastrointestinal bleeding Tumor necrosis factor Infliximab Biologic therapy Medicine (General) R5-920
spellingShingle	Vasculitis Gastrointestinal bleeding Tumor necrosis factor Infliximab Biologic therapy Medicine (General) R5-920 Lerkvaleekul B Treepongkaruna S Ruangwattanapaisarn N Treesit T Vilaiyuk S Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
description	Butsabong Lerkvaleekul,1 Suporn Treepongkaruna,2 Nichanan Ruangwattanapaisarn,3 Tharintorn Treesit,3 Soamarat Vilaiyuk11Division of Rheumatology, Department of Paediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 2Division of Gastroenterology, Department of Paediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand; 3Department of Diagnostic and Therapeutic Radiology, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, ThailandAbstract: Systemic polyarteritis nodosa (PAN) is a rare form of necrotizing vasculitis in children. Recurrent episodes of abdominal aneurysm ruptures are uncommon and life-threatening condition in children. Failures of response to immunosuppressive medications and radiological intervention also lead to high mortality. Some reports suggested that tumor necrosis factor (TNF) might have role in the inflammation of this disease. After an English-language literature review, this is the first case report in children of recurrent abdominal-ruptured aneurysms with a failure of conventional therapy but successfully treated with anti-TNF-α monoclonal antibody. We herein describe a 9-year-old girl who presented with chronic abdominal pain, hypertension, and massive lower gastrointestinal bleeding. The disease was refractory to conventional treatment, including administration of a corticosteroid, cyclophosphamide, and intravenous immunoglobulin, and recurrent-ruptured aneurysms developed in the gastrointestinal tract. Arterial embolization during angiography resulted in temporary improvement of the gastrointestinal bleeding. Infliximab, a chimeric anti-tumor necrosis factor-α monoclonal antibody, was initiated and resulted in disease remission with resolution of the gastrointestinal bleeding and abdominal pain. Anti-TNF therapy might be another treatment option for refractory disease to prevent ongoing inflammation that could lead to aneurysmal dilatation or even rupture. However, early recognition of refractory disease and aggressive treatment in the early course of the disease are crucial to reduce morbidity and mortality.Keywords: vasculitis, gastrointestinal bleeding, tumor necrosis factor, infliximab, biologic therapy
format	article
author	Lerkvaleekul B Treepongkaruna S Ruangwattanapaisarn N Treesit T Vilaiyuk S
author_facet	Lerkvaleekul B Treepongkaruna S Ruangwattanapaisarn N Treesit T Vilaiyuk S
author_sort	Lerkvaleekul B
title	Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
title_short	Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
title_full	Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
title_fullStr	Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
title_full_unstemmed	Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
title_sort	recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab
publisher	Dove Medical Press
publishDate	2019
url	https://doaj.org/article/db952d31e76f414a939f6076427d4299
work_keys_str_mv	AT lerkvaleekulb recurrentrupturedabdominalaneurysmsinpolyarteritisnodosasuccessfullytreatedwithinfliximab AT treepongkarunas recurrentrupturedabdominalaneurysmsinpolyarteritisnodosasuccessfullytreatedwithinfliximab AT ruangwattanapaisarnn recurrentrupturedabdominalaneurysmsinpolyarteritisnodosasuccessfullytreatedwithinfliximab AT treesitt recurrentrupturedabdominalaneurysmsinpolyarteritisnodosasuccessfullytreatedwithinfliximab AT vilaiyuks recurrentrupturedabdominalaneurysmsinpolyarteritisnodosasuccessfullytreatedwithinfliximab
_version_	1718401949442768896

Recurrent ruptured abdominal aneurysms in polyarteritis nodosa successfully treated with infliximab

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