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Titolo:
Hepatic autoantigens in patients with autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy
Autore:
Obermayer-Straub, P; Perheentupa, J; Braun, S; Kayser, A; Barut, A; Loges, S; Harms, A; Dalekos, G; Strassburg, CP; Manns, MP;
Indirizzi:
Hannover Med Sch, Dept Gastroenterol & Hepatol, D-30625 Hannover, Germany Hannover Med Sch Hannover Germany D-30625 tol, D-30625 Hannover, Germany Univ Helsinki, Hosp Children & Adolescents, Helsinki, Finland Univ Helsinki Helsinki Finland hildren & Adolescents, Helsinki, Finland
Titolo Testata:
GASTROENTEROLOGY
fascicolo: 3, volume: 121, anno: 2001,
pagine: 668 - 677
SICI:
0016-5085(200109)121:3<668:HAIPWA>2.0.ZU;2-S
Fonte:
ISI
Lingua:
ENG
Soggetto:
CHRONIC ACTIVE HEPATITIS; POLYGLANDULAR SYNDROME TYPE-1; SYNDROME TYPE-I; AMINO-ACID DECARBOXYLASE; DISEASE TYPE-I; ADDISONS-DISEASE; OVARIAN FAILURE; AUTOANTIBODIES; IDENTIFICATION; ANTIBODIES;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Clinical Medicine
Life Sciences
Citazioni:
37
Recensione:
Indirizzi per estratti:
Indirizzo: Manns, MP Hannover Med Sch, Dept Gastroenterol & Hepatol, Carl Neuberg Str1, D-30625 Hannover, Germany Hannover Med Sch Carl Neuberg Str 1 HannoverGermany D-30625 ny
Citazione:
P. Obermayer-Straub et al., "Hepatic autoantigens in patients with autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy", GASTROENTY, 121(3), 2001, pp. 668-677

Abstract

Background & Aims: Autoimmune polyendocrinopathycandidlasis-ectodermal dystrophy (APECED) is caused by mutations of both copies of the autoimmune regulator (AIRE) gene. It is characterized by susceptibility to mucocutaneous candidiasis and multiple autoimmune lesions. A serious disease component ishepatitis. To identify diagnostic autoantibodies for APECED hepatitis, sera from 64 patients with APECED were screened for autoantibodies establishedin the diagnosis of idiopathic autoimmune hepatitis, and for autoantibodies against 10 cytochrome P450s. Methods: Screening methods were indirect immunofluorescence, Western blot, Ouchterlony gel diffusion, enzyme-linked immunosorbent assay, and immunoprecipitation. Results: Anti-liver microsomal antibodies were detected in 50% of the patients with APECED hepatitis and 11% of those without hepatitis. Prevalences of antinuclear, smooth muscle, anti-liver cytosol, anti-soluble liver protein/liver pancreas, and anti-CYP2D6 autoantibodies were 9%, 6%, 3%, 0%, and 0%, respectively. CYP1A1, CYP2B6,CYP1A2, and CYP2A6 were identified as autoantigens. Thirty percent of patients with anti-CYP2A6 and :100% of patients with anti-CYP1A2 were affected by hepatitis. Despite the high specificity of anti-CYP1A2 for APECED hepatitis, its sensitivity was low (50%). Anti-CYP2A6 and anti-CYP1A2 were not detected in patients with autoimmune hepatitis (N = 68) or nonhepatitic controls (N = 81). Conclusions: Anti-CYP1A2 is a highly specific but insensitivemarker for APECED hepatitis. No clinical correlation was observed for anti-CYP2A6. Autoimmune hepatitis and APECED hepatitis are characterized by different molecular targets of autoantibodies with no overlap.

ASDD Area Sistemi Dipartimentali e Documentali, Università di Bologna, Catalogo delle riviste ed altri periodici
Documento generato il 05/12/20 alle ore 13:50:53