Literature DB >> 16029350

Cutaneous granulomas associated with primary immunodeficiency disorders.

A Mitra1, B Pollock, J Gooi, J C Darling, A Boon, J A Newton-Bishop.   

Abstract

Cutaneous granulomas are uncommon in primary immunodeficiency disorders. We report cutaneous granulomas in a child with ataxia telangiectasia (AT) and compare the clinical course with similar lesions in an adult with common variable immunodeficiency (CVI). A 4-year-old female with AT developed cutaneous granulomas as erythematous plaques. The largest lesion appeared on her left cheek and continued to progress despite treatment with topical and intralesional steroids. Disease control was obtained initially with oral antibiotics and low-dose oral steroids. On cessation of oral steroids, significant relapse of the facial granuloma occurred. Pulsed and then oral steroids were required to stop the disease process leaving significant scarring. The second case is of cutaneous granulomas in a 66-year-old man, with CVI, who presented with an erythematous reticulate rash on the legs. We consider it useful to report this patient here as disease control was obtained in a similar way with systemic immunosuppression. In this patient a combination of oral steroids and azathioprine was used. These cutaneous granulomas are thought to be a manifestation of immune dysregulation. No infectious cause has been found so far. We recommend the use of broad-spectrum antibiotics in conjunction with systemic steroids for progressive granulomas, as these patients are immunosuppressed and infection with an unidentified organism cannot be excluded.

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Year:  2005        PMID: 16029350     DOI: 10.1111/j.1365-2133.2005.06619.x

Source DB:  PubMed          Journal:  Br J Dermatol        ISSN: 0007-0963            Impact factor:   9.302


  10 in total

1.  Ataxia telangiectasia in Turkey: multisystem involvement of 91 patients.

Authors:  Hacer Akturk; Murat Sutcu; Ayper Somer; Sanem Piskin; Manolya Acar; Meral Ozmen; Umut Altinoglu; Burak Tatli; Nuran Salman
Journal:  World J Pediatr       Date:  2017-01-25       Impact factor: 2.764

2.  Cutaneous granulomatosis and combined immunodeficiency revealing Ataxia-Telangiectasia: a case report.

Authors:  Laura Folgori; Alessia Scarselli; Giulia Angelino; Francesca Ferrari; Antonio Antoccia; Luciana Chessa; Andrea Finocchi
Journal:  Ital J Pediatr       Date:  2010-04-11       Impact factor: 2.638

Review 3.  Granulomatous disease in CVID: retrospective analysis of clinical characteristics and treatment efficacy in a cohort of 59 patients.

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Journal:  J Clin Immunol       Date:  2012-09-18       Impact factor: 8.317

Review 4.  RAG Deficiency: Two Genes, Many Diseases.

Authors:  Ottavia M Delmonte; Catharina Schuetz; Luigi D Notarangelo
Journal:  J Clin Immunol       Date:  2018-07-25       Impact factor: 8.317

5.  Prevalence of Granulomas in Patients With Primary Immunodeficiency Disorders, United States: Data From National Health Care Claims and the US Immunodeficiency Network Registry.

Authors:  Jessica Leung; Kathleen E Sullivan; Ludmila Perelygina; Joseph P Icenogle; Ramsay L Fuleihan; Tatiana M Lanzieri
Journal:  J Clin Immunol       Date:  2018-07-24       Impact factor: 8.317

Review 6.  Immune dysregulation in patients with RAG deficiency and other forms of combined immune deficiency.

Authors:  Ottavia M Delmonte; Anna Villa; Luigi D Notarangelo
Journal:  Blood       Date:  2020-02-27       Impact factor: 22.113

7.  Multi-drugs resistant acne rosacea in a child affected by Ataxia-Telangiectasia: successful treatment with Isotretinoin.

Authors:  Nicoletta Cantarutti; Alessia Claps; Giulia Angelino; Luciana Chessa; Francesco Callea; May El Hachem; Andrea Diociaiuti; Andrea Finocchi
Journal:  Ital J Pediatr       Date:  2015-03-28       Impact factor: 2.638

8.  What Works When Treating Granulomatous Disease in Genetically Undefined CVID? A Systematic Review.

Authors:  Astrid C van Stigt; Willem A Dik; Lieke S J Kamphuis; Bas M Smits; Joris M van Montfrans; P Martin van Hagen; Virgil A S H Dalm; Hanna IJspeert
Journal:  Front Immunol       Date:  2020-12-17       Impact factor: 7.561

9.  The natural history of ataxia-telangiectasia (A-T): A systematic review.

Authors:  Emily Petley; Alexander Yule; Shaun Alexander; Shalini Ojha; William P Whitehouse
Journal:  PLoS One       Date:  2022-03-15       Impact factor: 3.752

10.  Cutaneous sarcoid-like granulomas in a child known with nijmegen breakage syndrome.

Authors:  Rachisan Andreea Liana; Gheban Dan; Miu Nicolae
Journal:  Iran J Pediatr       Date:  2013-02       Impact factor: 0.364

  10 in total

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