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Pathogenic variant in NFIX gene affecting three sisters due to paternal mosaicism.
Sihombing, Nydia Rena Benita; Winarni, Tri Indah; van Bokhoven, Hans; van der Burgt, Ineke; de Leeuw, Nicole; Faradz, Sultana M H.
Afiliación
  • Sihombing NRB; Doctoral Program of Medical and Health Sciences, Faculty of Medicine, Diponegoro University, Semarang, Indonesia.
  • Winarni TI; Division of Human Genetics, Center for Biomedical Research (CEBIOR), Faculty of Medicine, Diponegoro University, Semarang, Indonesia.
  • van Bokhoven H; Department of Human Genetics, Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.
  • van der Burgt I; Department of Human Genetics, Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.
  • de Leeuw N; Department of Human Genetics, Donders Institute for Brain, Cognition, and Behaviour, Radboud University Medical Center, Nijmegen, the Netherlands.
  • Faradz SMH; Division of Human Genetics, Center for Biomedical Research (CEBIOR), Faculty of Medicine, Diponegoro University, Semarang, Indonesia.
Am J Med Genet A ; 182(11): 2731-2736, 2020 11.
Article en En | MEDLINE | ID: mdl-32945093
We present a family with three girls presenting similar dysmorphic features, including overgrowth, intellectual disability, macrocephaly, prominent forehead, midface retrusion, strabismus, and scoliosis. Both parents were unaffected, suggesting the presence of an autosomal recessive syndrome. Following exome sequencing, a heterozygous nonsense variant was identified in the NFIX gene in all three siblings. The father appeared to have a low-grade (7%) mosaicism for this variant in his blood. Previously, de novo pathogenic variants in NFIX have been identified in Marshall-Smith syndrome and Malan syndrome, which share distinctive phenotypic features shared with the patients of the present family. This case emphasizes the importance of further molecular analysis especially in familial cases, to exclude the possibility of parental mosaicism.
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Texto completo: 1 Bases de datos: MEDLINE Asunto principal: Fenotipo / Discapacidades del Desarrollo / Factores de Transcripción NFI / Trastornos del Crecimiento / Discapacidad Intelectual / Mosaicismo / Mutación Tipo de estudio: Prognostic_studies Límite: Adult / Female / Humans / Male Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2020 Tipo del documento: Article País de afiliación: Indonesia

Texto completo: 1 Bases de datos: MEDLINE Asunto principal: Fenotipo / Discapacidades del Desarrollo / Factores de Transcripción NFI / Trastornos del Crecimiento / Discapacidad Intelectual / Mosaicismo / Mutación Tipo de estudio: Prognostic_studies Límite: Adult / Female / Humans / Male Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2020 Tipo del documento: Article País de afiliación: Indonesia