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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 ped...

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Autores principales: De Guerra, Viveka Clare, Hashmi, Humaira, Kramer, Bree, Balluz, Rula, Son, Mary Beth, Stein, Deborah, Lieberman, Alicia, Zahra, Mahmoud, Abdul-Aziz, Rabheh
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6594285/
https://www.ncbi.nlm.nih.gov/pubmed/31285932
http://dx.doi.org/10.1155/2019/8157969
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author De Guerra, Viveka Clare
Hashmi, Humaira
Kramer, Bree
Balluz, Rula
Son, Mary Beth
Stein, Deborah
Lieberman, Alicia
Zahra, Mahmoud
Abdul-Aziz, Rabheh
author_facet De Guerra, Viveka Clare
Hashmi, Humaira
Kramer, Bree
Balluz, Rula
Son, Mary Beth
Stein, Deborah
Lieberman, Alicia
Zahra, Mahmoud
Abdul-Aziz, Rabheh
author_sort De Guerra, Viveka Clare
collection PubMed
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 pubmed-65942852019-07-08 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 De Guerra, Viveka Clare Hashmi, Humaira Kramer, Bree Balluz, Rula Son, Mary Beth Stein, Deborah Lieberman, Alicia Zahra, Mahmoud Abdul-Aziz, Rabheh Case Rep Rheumatol Case Report 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. Hindawi 2019-06-11 /pmc/articles/PMC6594285/ /pubmed/31285932 http://dx.doi.org/10.1155/2019/8157969 Text en Copyright © 2019 Viveka Clare De Guerra et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
De Guerra, Viveka Clare
Hashmi, Humaira
Kramer, Bree
Balluz, Rula
Son, Mary Beth
Stein, Deborah
Lieberman, Alicia
Zahra, Mahmoud
Abdul-Aziz, Rabheh
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 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
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6594285/
https://www.ncbi.nlm.nih.gov/pubmed/31285932
http://dx.doi.org/10.1155/2019/8157969
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