A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation

Background. Takayasu’s arteritis with comorbid chronic recurrent multifocal osteomyelitis and ulcerative colitis is rare in the pediatric population. Treatment with anti-TNF alpha agents such as infliximab has been a successful treatment strategy in adults and can be used effectively in the pediatri...

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Main Authors: Viveka Clare De Guerra, Humaira Hashmi, Bree Kramer, Rula Balluz, Mary Beth Son, Deborah Stein, Alicia Lieberman, Mahmoud Zahra, Rabheh Abdul-Aziz
Format: Article
Language:English
Published: Wiley 2019-01-01
Series:Case Reports in Rheumatology
Online Access:http://dx.doi.org/10.1155/2019/8157969
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author Viveka Clare De Guerra
Humaira Hashmi
Bree Kramer
Rula Balluz
Mary Beth Son
Deborah Stein
Alicia Lieberman
Mahmoud Zahra
Rabheh Abdul-Aziz
author_facet Viveka Clare De Guerra
Humaira Hashmi
Bree Kramer
Rula Balluz
Mary Beth Son
Deborah Stein
Alicia Lieberman
Mahmoud Zahra
Rabheh Abdul-Aziz
author_sort Viveka Clare De Guerra
collection DOAJ
description Background. Takayasu’s arteritis with comorbid chronic recurrent multifocal osteomyelitis and ulcerative colitis is rare in the pediatric population. Treatment with anti-TNF alpha agents such as infliximab has been a successful treatment strategy in adults and can be used effectively in the pediatric population. Case Presentation. We present the case of a 15-year-old Caucasian girl with a history of chronic recurrent multifocal osteomyelitis and ulcerative colitis presenting with hypertensive emergency secondary to Takayasu’s arteritis with middle aortic syndrome. She was treated with corticosteroids and methotrexate and ultimately required infliximab infusions of 15 mg/kg every 4 weeks to successfully control her symptoms and normalize her inflammatory markers. Conclusions. In this case, we discuss the use of infliximab in an adolescent patient with chronic recurrent multifocal osteomyelitis, ulcerative colitis, and Takayasu’s arteritis. The significance of this case is determined by the unique occurrence of all three conditions in a pediatric patient, the important consideration of vasculitis in the differential of a pediatric patient presenting with hypertensive emergency, the need for vigilance for detecting diagnostic clues, signs, and symptoms, knowledge of disease associations when evaluating a patient with a predisposition for autoinflammatory conditions, and the use of increasing doses of infliximab to control symptoms.
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spelling doaj-art-e48a95959b8048fcaafc244599e097012025-08-20T03:26:26ZengWileyCase Reports in Rheumatology2090-68892090-68972019-01-01201910.1155/2019/81579698157969A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune DysregulationViveka Clare De Guerra0Humaira Hashmi1Bree Kramer2Rula Balluz3Mary Beth Son4Deborah Stein5Alicia Lieberman6Mahmoud Zahra7Rabheh Abdul-Aziz8Department of Pediatrics, University at Buffalo, Oishei Children’s Hospital, 1001 Main Street, Buffalo, NY 14203, USAWestern New York Pediatric Gastroenterology, 166 Washington Avenue, Batavia, NY 14020, USADepartment of Pediatrics, University at Buffalo, Oishei Children’s Hospital, 1001 Main Street, Buffalo, NY 14203, USADepartment of Pediatrics, University at Buffalo, Oishei Children’s Hospital, 1001 Main Street, Buffalo, NY 14203, USADepartment of Pediatric Rheumatology, Boston Children’s Hospital, 300 Longwood Ave, Boston, MA 02115, USADepartment of Pediatric Nephrology, Center for Midaortic Syndrome and Renovascular Hypertension, Boston Children’s Hospital, 300 Longwood Ave, Boston, MA 02115, USADepartment of Pediatric Rheumatology, Strong Memorial Hospital at the University of Rochester, 601 Elmwood Ave, Rochester, NY 14642, USADepartment of Pediatrics, University at Buffalo, Oishei Children’s Hospital, 1001 Main Street, Buffalo, NY 14203, USADepartment of Pediatrics, University at Buffalo, Oishei Children’s Hospital, 1001 Main Street, Buffalo, NY 14203, USABackground. Takayasu’s arteritis with comorbid chronic recurrent multifocal osteomyelitis and ulcerative colitis is rare in the pediatric population. Treatment with anti-TNF alpha agents such as infliximab has been a successful treatment strategy in adults and can be used effectively in the pediatric population. Case Presentation. We present the case of a 15-year-old Caucasian girl with a history of chronic recurrent multifocal osteomyelitis and ulcerative colitis presenting with hypertensive emergency secondary to Takayasu’s arteritis with middle aortic syndrome. She was treated with corticosteroids and methotrexate and ultimately required infliximab infusions of 15 mg/kg every 4 weeks to successfully control her symptoms and normalize her inflammatory markers. Conclusions. In this case, we discuss the use of infliximab in an adolescent patient with chronic recurrent multifocal osteomyelitis, ulcerative colitis, and Takayasu’s arteritis. The significance of this case is determined by the unique occurrence of all three conditions in a pediatric patient, the important consideration of vasculitis in the differential of a pediatric patient presenting with hypertensive emergency, the need for vigilance for detecting diagnostic clues, signs, and symptoms, knowledge of disease associations when evaluating a patient with a predisposition for autoinflammatory conditions, and the use of increasing doses of infliximab to control symptoms.http://dx.doi.org/10.1155/2019/8157969
spellingShingle Viveka Clare De Guerra
Humaira Hashmi
Bree Kramer
Rula Balluz
Mary Beth Son
Deborah Stein
Alicia Lieberman
Mahmoud Zahra
Rabheh Abdul-Aziz
A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
Case Reports in Rheumatology
title A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
title_full A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
title_fullStr A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
title_full_unstemmed A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
title_short A Case Report of Takayasu’s Arteritis and Ulcerative Colitis in a Pediatric Patient with Chronic Recurrent Multifocal Osteomyelitis Successfully Treated with Infliximab: Diagnostic Clues in Disease Associations and Immune Dysregulation
title_sort case report of takayasu s arteritis and ulcerative colitis in a pediatric patient with chronic recurrent multifocal osteomyelitis successfully treated with infliximab diagnostic clues in disease associations and immune dysregulation
url http://dx.doi.org/10.1155/2019/8157969
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