Your browser doesn't support javascript.
loading
Biallelic ZNF407 mutations in a neurodevelopmental disorder with ID, short stature and variable microcephaly, hypotonia, ocular anomalies and facial dysmorphism.
Zahra, Qandeel; Çakmak, Çagla; Koprulu, Mine; Shuaib, Muhammad; Sobreira, Nara; Kalsner, Louisa; Sobreira, Joselito; Guillen Sacoto, Maria J; Malik, Sajid; Tolun, Aslihan.
Afiliação
  • Zahra Q; Human Genetics Program, Department of Zoology, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan.
  • Çakmak Ç; Department of Molecular Biology and Genetics, Bogaziçi University, Istanbul, Turkey.
  • Koprulu M; Department of Molecular Biology and Genetics, Bogaziçi University, Istanbul, Turkey.
  • Shuaib M; Department of Molecular Biology and Genetics, Istanbul Technical University, Istanbul, Turkey.
  • Sobreira N; Human Genetics Program, Department of Zoology, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan.
  • Kalsner L; McKusick-Nathans Department of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, MD, 21205, USA.
  • Sobreira J; Departments of Neurology and Pediatrics, University of Connecticut School of Medicine and Connecticut Children's Medical Center, Farmington, CT, USA.
  • Guillen Sacoto MJ; Division of Genetics, Department of Morphology and Genetics, Universidade Federal de Sao Paulo, Sao Paulo, Brazil.
  • Malik S; GeneDx, Gaithersburg, MD, USA.
  • Tolun A; Human Genetics Program, Department of Zoology, Faculty of Biological Sciences, Quaid-i-Azam University, Islamabad, Pakistan. malik@qau.edu.pk.
J Hum Genet ; 65(12): 1115-1123, 2020 Dec.
Article em En | MEDLINE | ID: mdl-32737394
We describe five members of a consanguineous Pakistani family (Family I) plus two affected children from families of different ethnic origins presenting with neurodevelopmental disorders with overlapping features. All affected individuals from families have intellectual disability (ID), ranging from mild to profound, and reduced motor and cognitive skills plus variable features including short stature, microcephaly, developmental delay, hypotonia, dysarthria, deafness, visual problems, enuresis, encopresis, behavioural anomalies, delayed pubertal onset and facial dysmorphism. We first mapped the disease locus in the large family (Family I), and by exome sequencing identified homozygous ZNF407 c.2814_2816dup (p.Val939dup) in four affected members where DNA samples were available. By exome sequencing we detected homozygous c.2405G>T (p.Gly802Val) in the affected member of Family II and compound heterozygous variants c.2884C>G (p.Arg962Gly) and c.3642G>C (p.Lys1214Asn) in the affected member of Family III. Homozygous c.5054C>G (p.Ser1685Trp) has been reported in two brothers with an ID syndrome. Affected individuals we present did not exhibit synophrys, midface hypoplasia, kyphosis, 5th finger camptodactyly, short 4th metatarsals or limited knee mobility observed in the reported family.
Assuntos

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Fatores de Transcrição / Proteínas de Ligação a DNA / Nanismo / Transtornos do Neurodesenvolvimento / Deficiência Intelectual / Microcefalia Tipo de estudo: Prognostic_studies Limite: Child / Child, preschool / Female / Humans / Male Idioma: En Ano de publicação: 2020 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Fatores de Transcrição / Proteínas de Ligação a DNA / Nanismo / Transtornos do Neurodesenvolvimento / Deficiência Intelectual / Microcefalia Tipo de estudo: Prognostic_studies Limite: Child / Child, preschool / Female / Humans / Male Idioma: En Ano de publicação: 2020 Tipo de documento: Article