Síndrome de Kallmann: uma revisão histórica, clínica e molecular

Kallmann syndrome (KS), the association of hypogonadotropic hypogonadism and anosmia, was described by Maestre de San Juan in 1856 and characterized as a hereditary condition by Franz Josef Kallmann in 1944. Many aspects such as pathogeny, phenotype and genotype in KS were described in the last fift...

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Detalles Bibliográficos
Autores: Ribeiro, Rogério Silicani [UNIFESP], Abucham, Julio [UNIFESP]
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2008
País:Brasil
Institución:Universidade Federal de São Paulo (UNIFESP)
Repositorio:Repositório Institucional da UNIFESP
Idioma:portugués
OAI Identifier:oai:repositorio.unifesp.br:11600/4237
Acceso en línea:http://dx.doi.org/10.1590/S0004-27302008000100004
http://repositorio.unifesp.br/handle/11600/4237
Access Level:acceso abierto
Palabra clave:Hipogonadismo hipogonadotrófico
Kallmann
KAL-1
FGFR1
PROKR2
PROK2
Descripción
Sumario:Kallmann syndrome (KS), the association of hypogonadotropic hypogonadism and anosmia, was described by Maestre de San Juan in 1856 and characterized as a hereditary condition by Franz Josef Kallmann in 1944. Many aspects such as pathogeny, phenotype and genotype in KS were described in the last fifteen years. The knowledge of this condition has grown fast, making it difficult to update. Here we review historical aspects of this condition and its discoverers and describe new findings regarding the embryogenesis of the olfactory bulb and GnRH secreting neuronal tracts that are important for understanding the association of hypogonadism and anosmia. Additionally, we describe the phenotypic and genotypic heterogeneity of KS, including five related genes (KAL-1, FGFR1, PROKR2, PROK2 e NELF), and discuss the function of each codified protein in migration and maturation of the olfactory and GnRH neurons, with data from in vitro and in vivo studies. Finally we describe the clinical phenotype of patients carrying these mutations.