Literature DB >> 31149156

POLYGLANDULAR AUTOIMMUNE SYNDROME ASSOCIATED WITH MULTIPLE AUTOIMMUNE CONDITIONS AND ATOPIC DERMATITIS - AN UNUSUAL MANIFESTATION OF A POLYAUTOIMMUNITY PHENOTYPE.

A M Forsea1,2, C Mihai1,3, T Predescu2, I Tudose4, I Margaritescu5, C Giurcaneanu1,2.   

Abstract

INTRODUCTION: The polyglandular autoimmune syndrome (PAS) type III is a rare condition defined as the coexistence of autoimmune thyroid disorder with other endocrine autoimmune diseases, including type 1 diabetes, without adrenal dysfunction. PAS may associate with other non-endocrine autoimmune diseases, overlapping with the multiple autoimmune syndromes (MAS). We present a case of PAS III/ MAS type 3, including autoimmune thyroiditis, autoimmune diabetes, vitiligo, lupus erythematosus, associated with adult-onset atopic dermatitis, a combination not reported previously. CASE REPORT: A 40 years old woman, registered as nurse working in dialysis unit, previously diagnosed with vitiligo, euthyroid autoimmune thyroiditis and disseminated granuloma annulare, with personal and familial history of atopic disorders, presented in our clinic for disseminated eczematous and lichenoid cutaneous rashes. She was tested positive for antinuclear, anti-double stranded DNA and anti-histone antibodies, with inflammatory syndrome and marginal lymphopenia and she was diagnosed with systemic lupus erythematosus (SLE). Subsequently, moderate hyperglycemia, positive anti-glutamic acid decarboxylase antibodies and low C-peptide level prompted the diagnosis of autoimmune diabetes. Recurrent flexural eczematous rashes, with negative epicutaneous tests but positive specific IgE tests for common allergens fulfilled the clinical criteria for the diagnosis of atopic dermatitis. The clinical, immunological and glycemic status were controlled with low doses of oral prednisone (<0.5 mg/kg), methotrexate (10mg/week), antimalarials, metformin, emollients and photoprotection. After changing her workplace, the immunosuppressive treatment could be discontinued, and the patient maintained normal immunological and biochemical profile at 6 months follow-up.This case brings a unique perspective on the evolution, associations spectrum and the management challenges of endocrine polyautoimmunity associated with atopic diathesis.

Entities:  

Keywords:  atopic dermatitis; latent autoimmune diabetes of the adult; multiple autoimmune syndrome; polyglandular autoimmune syndrome

Year:  2017        PMID: 31149156      PMCID: PMC6525744          DOI: 10.4183/aeb.2017.106

Source DB:  PubMed          Journal:  Acta Endocrinol (Buchar)        ISSN: 1841-0987            Impact factor:   0.877


  18 in total

Review 1.  SCHMIDT'S SYNDROME (THYROID AND ADRENAL INSUFFICIENCY). A REVIEW OF THE LITERATURE AND A REPORT OF FIFTEEN NEW CASES INCLUDING TEN INSTANCES OF COEXISTENT DIABETES MELLITUS.

Authors:  C C CARPENTER; N SOLOMON; S G SILVERBERG; T BLEDSOE; R C NORTHCUTT; J R KLINENBERG; I L BENNETT; A M HARVEY
Journal:  Medicine (Baltimore)       Date:  1964-03       Impact factor: 1.889

Review 2.  Autoimmune polyendocrine syndromes.

Authors:  George S Eisenbarth; Peter A Gottlieb
Journal:  N Engl J Med       Date:  2004-05-13       Impact factor: 91.245

3.  The multiple autoimmune syndromes. A clue for the autoimmune tautology.

Authors:  Juan-Manuel Anaya; John Castiblanco; Adriana Rojas-Villarraga; Ricardo Pineda-Tamayo; Roger A Levy; José Gómez-Puerta; Carlos Dias; Ruben D Mantilla; Juan Esteban Gallo; Ricard Cervera; Yehuda Shoenfeld; Mauricio Arcos-Burgos
Journal:  Clin Rev Allergy Immunol       Date:  2012-12       Impact factor: 8.667

4.  Relationship between type 1 diabetes and atopic diseases in a twin population.

Authors:  S F Thomsen; D L Duffy; K O Kyvik; A Skytthe; V Backer
Journal:  Allergy       Date:  2010-12-01       Impact factor: 13.146

5.  Atopic dermatitis in adults.

Authors:  Licia Zeppa; Veronica Bellini; Paolo Lisi
Journal:  Dermatitis       Date:  2011 Jan-Feb       Impact factor: 4.845

6.  Pre- vs. post-pubertal onset of vitiligo: multivariate analysis indicates atopic diathesis association in pre-pubertal onset vitiligo.

Authors:  K Ezzedine; A Diallo; C Léauté-Labrèze; J Seneschal; K Boniface; M Cario-André; S Prey; F Ballanger; F Boralevi; T Jouary; D Mossalayi; A Taieb
Journal:  Br J Dermatol       Date:  2012-09       Impact factor: 9.302

7.  Atopy as a risk factor for thyroid autoimmunity in children.

Authors:  M Pedullà; M Miraglia Del Giudice; V Fierro; T Arrigo; E Gitto; A Salpietro; E Lionetti; V Salpietro; S Leonardi; F Santaniello; L Perrone
Journal:  J Biol Regul Homeost Agents       Date:  2012 Jan-Mar       Impact factor: 1.711

8.  Autoimmune disease comorbidities in patients with atopic dermatitis: a nationwide case-control study in Taiwan.

Authors:  Lung-Chi Wu; Chian-Yaw Hwang; Pei-I Chung; Tuan-Chun Hua; Yen-Da Chen; Szu-Yin Chu; Din-Dar Lee; Yun-Ting Chang; Wen-Jen Wang; Han-Nan Liu; Chih-Chiang Chen
Journal:  Pediatr Allergy Immunol       Date:  2014-10       Impact factor: 6.377

9.  Erythrodermic atopic dermatitis with late onset--case presentation.

Authors:  Cristina Lancrajan; Roxana Bumbacea; Calin Giurcaneanu
Journal:  J Med Life       Date:  2010 Jan-Mar

10.  Atopic diseases and systemic lupus erythematosus: an epidemiological study of the risks and correlations.

Authors:  Yu-Ping Hsiao; Jeng-Dau Tsai; Chih-Hsin Muo; Chung-Hung Tsai; Fung-Chang Sung; Ya-Tang Liao; Yen-Jung Chang; Jen-Hung Yang
Journal:  Int J Environ Res Public Health       Date:  2014-08-08       Impact factor: 3.390

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