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The unusual association of Graves' disease, chronic spontaneous urticaria, and premature ovarian failure: report of a case and HLA haplotype characterization / A associação incomum entre doença de Graves, urticária crônica idiopática e insuficiência ovariana prematura: relato de caso clínico e tipagem HLA
Ruggeri, Rosaria Maddalena; Vita, Giuseppe; D'Angelo, Anna Grazia; Quattrocchi, Paolina; Certo, Rosaria; Benvenga, Salvatore; Cannavò, Salvatore; Gangemi, Sebastiano.
  • Ruggeri, Rosaria Maddalena; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Vita, Giuseppe; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • D'Angelo, Anna Grazia; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Quattrocchi, Paolina; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Certo, Rosaria; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Benvenga, Salvatore; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Cannavò, Salvatore; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
  • Gangemi, Sebastiano; Department of Clinical and Experimental Medicine. Unit of Endocrinology. IT
Arq. bras. endocrinol. metab ; 57(9): 748-752, Dec. 2013. tab
Article in English | LILACS | ID: lil-696923
ABSTRACT
Chronic spontaneous urticaria (CSU), defined as the occurrence of spontaneous wheals for more than six weeks, has been associated with autoimmune diseases. Herein, we report the unusual association of CSU, Graves' disease, and premature ovarian failure. Human leukocyte antigen (HLA) studies were performed. A 36-year-old woman presented symptoms and signs of hyperthyroidism for three months. In the same period, the patient complained of widespread urticarial wheals, intensely itchy, and poorly responsive to therapy with antihistaminic agents. Hyperthyroidism was confirmed biochemically, and treatment with methimazole was started. As hyperthyroidism improved, a marked improvement in her urticaria was also observed. However, the patient continued to complain of amenorrhea. Endocrine evaluation, at the age 38, was consistent with premature ovarian failure. This is the first report of coexistence of GD, CSU, and POF. The genetic background of such unusual association is a specific combination of HLA.
RESUMO
A urticária crônica idiopática, caracterizada pelo aparecimento de pápulas espontâneas e persistentes por pelo menos seis semanas, tem sido associada a doenças autoimunes. Apresentamos aqui o caso da associação incomum entre urticária crônica idiopática, doença de Graves e falência ovariana prematura. Foram conduzidos estudos de tipagem HLA. Uma mulher de 36 anos apresentou sinais e sintomas de hipertireoidismo por três meses. No mesmo período, a paciente queixou-se do aparecimento de pápulas urticariformes generalizadas que coçavam intensamente e não eram responsivas ao tratamento com anti-histamínicos. O hipertireoidismo foi confirmado bioquimicamente, e o tratamento com metimazol foi iniciado. Assim que os valores hormonais se normalizaram, observou-se uma melhoria significativa do quadro de urticária. No entanto, a paciente continuou a apresentar amenorreia. A avaliação endocrinológica, com a idade de 38 anos, mostrou falência ovariana prematura. Este é o primeiro caso de associação entre doença de Graves, urticária idiopática crônica e falência ovariana prematura. A base genética dessa associação incomum é representada por combinações específicas de haplótipos HLA.
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Full text: Available Index: LILACS (Americas) Main subject: Urticaria / Haplotypes / Graves Disease / Primary Ovarian Insufficiency / HLA Antigens Type of study: Risk factors Limits: Adult / Female / Humans Language: English Journal: Arq. bras. endocrinol. metab Journal subject: Endocrinology / Metabolism Year: 2013 Type: Article Affiliation country: Italy Institution/Affiliation country: Department of Clinical and Experimental Medicine/IT

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Full text: Available Index: LILACS (Americas) Main subject: Urticaria / Haplotypes / Graves Disease / Primary Ovarian Insufficiency / HLA Antigens Type of study: Risk factors Limits: Adult / Female / Humans Language: English Journal: Arq. bras. endocrinol. metab Journal subject: Endocrinology / Metabolism Year: 2013 Type: Article Affiliation country: Italy Institution/Affiliation country: Department of Clinical and Experimental Medicine/IT