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Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease

We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the...

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Autores principales: Kaji, Mariko, Kishi, Takayuki, Miyamae, Takako, Nagata, Satoru, Yamanaka, Hisashi, Fujikawa, Satoshi
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi Publishing Corporation 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4644826/
https://www.ncbi.nlm.nih.gov/pubmed/26609459
http://dx.doi.org/10.1155/2015/716138
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author Kaji, Mariko
Kishi, Takayuki
Miyamae, Takako
Nagata, Satoru
Yamanaka, Hisashi
Fujikawa, Satoshi
author_facet Kaji, Mariko
Kishi, Takayuki
Miyamae, Takako
Nagata, Satoru
Yamanaka, Hisashi
Fujikawa, Satoshi
author_sort Kaji, Mariko
collection PubMed
description We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment.
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spelling pubmed-46448262015-11-25 Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease Kaji, Mariko Kishi, Takayuki Miyamae, Takako Nagata, Satoru Yamanaka, Hisashi Fujikawa, Satoshi Case Rep Rheumatol Case Report We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment. Hindawi Publishing Corporation 2015 2015-11-02 /pmc/articles/PMC4644826/ /pubmed/26609459 http://dx.doi.org/10.1155/2015/716138 Text en Copyright © 2015 Mariko Kaji et al. https://creativecommons.org/licenses/by/3.0/ 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
Kaji, Mariko
Kishi, Takayuki
Miyamae, Takako
Nagata, Satoru
Yamanaka, Hisashi
Fujikawa, Satoshi
Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title_full Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title_fullStr Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title_full_unstemmed Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title_short Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease
title_sort efficacy of adalimumab in a girl with refractory intestinal behcet's disease
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4644826/
https://www.ncbi.nlm.nih.gov/pubmed/26609459
http://dx.doi.org/10.1155/2015/716138
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