Maturity-onset diabetes of the young (MODY) in Brazil: Establishment of a national registry and appraisal of available genetic and clinical data

Aims: Maturity-Onset Diabetes of the Young (MODY) comprises a heterogeneous group of monogenic forms of diabetes caused by mutations in at least 14 genes, but mostly by mutations in Glucokinase (GCK) and hepatocyte nuclear factor-1 homeobox A (HNF1A). This study aims to establish a national registry...

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Detalles Bibliográficos
Autores: Giuffrida, Fernando de Mello Almada [UNIFESP], Moises, Regina Celia Mello Santiago [UNIFESP], Weinert, Leticia S., Calliari, Luis E., Della Manna, Thais, Dotto, Renata Pires [UNIFESP], Franco, Luciana Ferreira [UNIFESP], Caetano, Lilian A., Teles, Milena G., Lima, Renata Andrade, Alves, Cresio, Dib, Sergio Atala [UNIFESP], Silveiro, Sandra P., Dias-da-Silva, Magnus Régios [UNIFESP], Reis, André Fernandes [UNIFESP]
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2017
País:Brasil
Institución:Universidade Federal de São Paulo (UNIFESP)
Repositorio:Repositório Institucional da UNIFESP
Idioma:inglés
OAI Identifier:oai:repositorio.unifesp.br:11600/56349
Acceso en línea:http://dx.doi.org/10.1016/j.diabres.2016.10.017
https://repositorio.unifesp.br/handle/11600/56349
Access Level:acceso abierto
Palabra clave:MODY
Diabetes mellitus
HNF1A
Glucokinase
Monogenic diabetes
Descripción
Sumario:Aims: Maturity-Onset Diabetes of the Young (MODY) comprises a heterogeneous group of monogenic forms of diabetes caused by mutations in at least 14 genes, but mostly by mutations in Glucokinase (GCK) and hepatocyte nuclear factor-1 homeobox A (HNF1A). This study aims to establish a national registry of MODY cases in Brazilian patients, assessing published and unpublished data. Methods: 311 patients with clinical characteristics of MODY were analyzed, with unpublished data on 298 individuals described in 12 previous publications and 13 newly described cases in this report. Results: 72 individuals had GCK mutations, 9 described in Brazilian individuals for the first time. One previously unpublished novel GCK mutation, Gly178Ala, was found in one family. 31 individuals had HNF1A mutations, 2 described for the first time in Brazilian individuals. Comparisons of GCK probands vs HNF1A: age 16 +/- 11 vs 35 +/- 20 years