Your browser doesn't support javascript.
loading
Biallelic Mutations in MITF Cause Coloboma, Osteopetrosis, Microphthalmia, Macrocephaly, Albinism, and Deafness.
George, Aman; Zand, Dina J; Hufnagel, Robert B; Sharma, Ruchi; Sergeev, Yuri V; Legare, Janet M; Rice, Gregory M; Scott Schwoerer, Jessica A; Rius, Mariana; Tetri, Laura; Gamm, David M; Bharti, Kapil; Brooks, Brian P.
Afiliação
  • George A; Ophthalmic Genetics and Visual Function Branch, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Zand DJ; Children's National Medical Center, Washington, DC 20010, USA.
  • Hufnagel RB; Ophthalmic Genetics and Visual Function Branch, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Sharma R; Unit on Ocular and Stem Cell Translational Research, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Sergeev YV; Ophthalmic Genetics and Visual Function Branch, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Legare JM; Department of Pediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA.
  • Rice GM; Department of Pediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA.
  • Scott Schwoerer JA; McPherson Eye Research Institute and Department of Ophthalmology and Visual Sciences, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA.
  • Rius M; Ophthalmic Genetics and Visual Function Branch, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Tetri L; Department of Pediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA.
  • Gamm DM; Department of Pediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA; McPherson Eye Research Institute and Department of Ophthalmology and Visual Sciences, University of Wisconsin School of Medicine and Public Health, Madison, WI 53726, USA.
  • Bharti K; Unit on Ocular and Stem Cell Translational Research, National Eye Institute, NIH, Bethesda, MD 20892, USA.
  • Brooks BP; Ophthalmic Genetics and Visual Function Branch, National Eye Institute, NIH, Bethesda, MD 20892, USA; Children's National Medical Center, Washington, DC 20010, USA. Electronic address: brian.brooks1@nih.gov.
Am J Hum Genet ; 99(6): 1388-1394, 2016 Dec 01.
Article em En | MEDLINE | ID: mdl-27889061
Human MITF is, by convention, called the "microphthalmia-associated transcription factor" because of previously published seminal mouse genetic studies; however, mutations in MITF have never been associated with microphthalmia in humans. Here, we describe a syndrome that we term COMMAD, characterized by coloboma, osteopetrosis, microphthalmia, macrocephaly, albinism, and deafness. COMMAD is associated with biallelic MITF mutant alleles and hence suggests a role for MITF in regulating processes such as optic-fissure closure and bone development or homeostasis, which go beyond what is usually seen in individuals carrying monoallelic MITF mutations.
Assuntos

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Osteopetrose / Albinismo / Coloboma / Microftalmia / Surdez / Alelos / Fator de Transcrição Associado à Microftalmia / Megalencefalia Limite: Animals / Child, preschool / Female / Humans / Infant / Male Idioma: En Revista: Am J Hum Genet Ano de publicação: 2016 Tipo de documento: Article País de afiliação: Estados Unidos

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Osteopetrose / Albinismo / Coloboma / Microftalmia / Surdez / Alelos / Fator de Transcrição Associado à Microftalmia / Megalencefalia Limite: Animals / Child, preschool / Female / Humans / Infant / Male Idioma: En Revista: Am J Hum Genet Ano de publicação: 2016 Tipo de documento: Article País de afiliação: Estados Unidos