Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine

Background: Azathioprine is an immunosuppressive agent used in the treatment of immune-mediated diseases. Azathioprine hypersensitivity syndrome is a rare adverse reaction occurring a few days to weeks after the administration of azathioprine. Case 1: A 36-year-old male with ulcerative colitis prese...

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Main Authors: Majed Aleissa, Perrine Nicol, Marion Godeau, Emilie Tournier, Frederic de Bellissen, Marie-Angèle Robic, Cristina Bulai Livideanu, Juliette Mazereeuw-Hautier, Carle Paul
Format: Article
Language:English
Published: Karger Publishers 2017-01-01
Series:Case Reports in Dermatology
Subjects:
Online Access:http://www.karger.com/Article/FullText/454876
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author Majed Aleissa
Perrine Nicol
Marion Godeau
Emilie Tournier
Frederic de Bellissen
Marie-Angèle Robic
Cristina Bulai Livideanu
Juliette Mazereeuw-Hautier
Carle Paul
author_facet Majed Aleissa
Perrine Nicol
Marion Godeau
Emilie Tournier
Frederic de Bellissen
Marie-Angèle Robic
Cristina Bulai Livideanu
Juliette Mazereeuw-Hautier
Carle Paul
author_sort Majed Aleissa
collection DOAJ
description Background: Azathioprine is an immunosuppressive agent used in the treatment of immune-mediated diseases. Azathioprine hypersensitivity syndrome is a rare adverse reaction occurring a few days to weeks after the administration of azathioprine. Case 1: A 36-year-old male with ulcerative colitis presented with erythematous plaques, pustules and erosions on the lower back, buttocks and thighs associated with high fever (39°C) 2 weeks after the initiation of azathioprine 100 mg/day. Additional findings included leukocytosis (18.6 g/L) with neutrophilia (11.1 g/L) and elevated C-reactive protein (128 mg/L). Histopathology showed a dense infiltrate of neutrophils in the hair follicles. We increased the dose of prednisone to 1 mg/kg/day (60 mg/day) and azathioprine was discontinued. He had marked improvement within 3 weeks and did not have any relapse with a 1-year follow-up. Case 2: A 57-year-old male with ulcerative colitis presented with erythematous plaques and pustules on the lower limbs associated with high fever (40°C) 1 week after the initiation of azathioprine 75 mg/day. Leukocytosis with neutrophilia (13.6 g/L) and elevated C-reactive protein (344 mg/L) were among the laboratory findings. Histopathology showed a dense infiltrate of neutrophils in the hair follicles. The dose of prednisone was increased to 20 mg/day and azathioprine was discontinued, which led to complete remission within 7 days. He did not have any relapse with a 6-month follow-up. Conclusion: The development of acute neutrophilic dermatitis 2 weeks after the initiation of azathioprine and the complete resolution after its withdrawal were in favor of azathioprine hypersensitivity syndrome. It should not be confused with Sweet syndrome associated with inflammatory bowel disease, as maintenance of azathioprine treatment may lead to life-threatening reactions.
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spelling doaj.art-2cd9cbe53aaf487886b5b50f05f6a7172022-12-21T18:43:50ZengKarger PublishersCase Reports in Dermatology1662-65672017-01-019161110.1159/000454876454876Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by AzathioprineMajed AleissaPerrine NicolMarion GodeauEmilie TournierFrederic de BellissenMarie-Angèle RobicCristina Bulai LivideanuJuliette Mazereeuw-HautierCarle PaulBackground: Azathioprine is an immunosuppressive agent used in the treatment of immune-mediated diseases. Azathioprine hypersensitivity syndrome is a rare adverse reaction occurring a few days to weeks after the administration of azathioprine. Case 1: A 36-year-old male with ulcerative colitis presented with erythematous plaques, pustules and erosions on the lower back, buttocks and thighs associated with high fever (39°C) 2 weeks after the initiation of azathioprine 100 mg/day. Additional findings included leukocytosis (18.6 g/L) with neutrophilia (11.1 g/L) and elevated C-reactive protein (128 mg/L). Histopathology showed a dense infiltrate of neutrophils in the hair follicles. We increased the dose of prednisone to 1 mg/kg/day (60 mg/day) and azathioprine was discontinued. He had marked improvement within 3 weeks and did not have any relapse with a 1-year follow-up. Case 2: A 57-year-old male with ulcerative colitis presented with erythematous plaques and pustules on the lower limbs associated with high fever (40°C) 1 week after the initiation of azathioprine 75 mg/day. Leukocytosis with neutrophilia (13.6 g/L) and elevated C-reactive protein (344 mg/L) were among the laboratory findings. Histopathology showed a dense infiltrate of neutrophils in the hair follicles. The dose of prednisone was increased to 20 mg/day and azathioprine was discontinued, which led to complete remission within 7 days. He did not have any relapse with a 6-month follow-up. Conclusion: The development of acute neutrophilic dermatitis 2 weeks after the initiation of azathioprine and the complete resolution after its withdrawal were in favor of azathioprine hypersensitivity syndrome. It should not be confused with Sweet syndrome associated with inflammatory bowel disease, as maintenance of azathioprine treatment may lead to life-threatening reactions.http://www.karger.com/Article/FullText/454876AzathioprineAzathioprine hypersensitivity syndromeDrug reactionNeutrophilic dermatosisSweet syndromeInflammatory bowel disease
spellingShingle Majed Aleissa
Perrine Nicol
Marion Godeau
Emilie Tournier
Frederic de Bellissen
Marie-Angèle Robic
Cristina Bulai Livideanu
Juliette Mazereeuw-Hautier
Carle Paul
Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
Case Reports in Dermatology
Azathioprine
Azathioprine hypersensitivity syndrome
Drug reaction
Neutrophilic dermatosis
Sweet syndrome
Inflammatory bowel disease
title Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
title_full Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
title_fullStr Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
title_full_unstemmed Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
title_short Azathioprine Hypersensitivity Syndrome: Two Cases of Febrile Neutrophilic Dermatosis Induced by Azathioprine
title_sort azathioprine hypersensitivity syndrome two cases of febrile neutrophilic dermatosis induced by azathioprine
topic Azathioprine
Azathioprine hypersensitivity syndrome
Drug reaction
Neutrophilic dermatosis
Sweet syndrome
Inflammatory bowel disease
url http://www.karger.com/Article/FullText/454876
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