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Wolf-Hirschhorn Syndrome: Clinical and Genetic Study of 7 New Cases, and Mini Review.
Gavril, Eva-Cristiana; Luca, Alina Costina; Curpan, Alexandrina-Stefania; Popescu, Roxana; Resmerita, Irina; Panzaru, Monica Cristina; Butnariu, Lacramioara Ionela; Gorduza, Eusebiu Vlad; Gramescu, Mihaela; Rusu, Cristina.
Afiliación
  • Gavril EC; Department of Medical Genetics, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
  • Luca AC; Department of Pediatric Cardiology, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
  • Curpan AS; "St. Mary" Children's Hospital, Vasile Lupu Street, No 62-64, 700309 Iasi, Romania.
  • Popescu R; Department of Biology, Faculty of Biology, "Alexandru Ioan Cuza" University of Iasi, Bd. Carol I, 20A, 700505 Iasi, Romania.
  • Resmerita I; Department of Medical Genetics, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
  • Panzaru MC; "St. Mary" Children's Hospital, Vasile Lupu Street, No 62-64, 700309 Iasi, Romania.
  • Butnariu LI; Department of Medical Genetics, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
  • Gorduza EV; Department of Medical Genetics, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
  • Gramescu M; "St. Mary" Children's Hospital, Vasile Lupu Street, No 62-64, 700309 Iasi, Romania.
  • Rusu C; Department of Medical Genetics, Faculty of Medicine, "Grigore T. Popa" University of Medicine and Pharmacy, University Street, No 16, 700115 Iasi, Romania.
Children (Basel) ; 8(9)2021 Aug 30.
Article en En | MEDLINE | ID: mdl-34572183
Wolf-Hirschhorn syndrome (WHS), a rare disorder determined by distal 4p deletion, is characterized by a pre and postnatal growth retardation, hypotonia, intellectual disability, epilepsy, craniofacial dysmorphism, and congenital fusion anomalies. The clinical aspects are dependent on the deletion' size. Our aim was to identify rare specific characteristics in a cohort of seven cases with 4p deletion and to assess the utility of Multiplex ligation-dependent probe amplification (MLPA) (cheap and sensitive test)-combined kits-as a diagnostic test and selection tool for cases that require other investigations (chromosomal microarray analysis-CMA, karyotype). For all cases we conducted a clinical examination with the main features identified: facial dysmorphism, intellectual disability, postnatal development delay, cardiac defects and hypotonia. In some cases, we observed seizures, structural brain abnormalities, immunodeficiencies, and renal anomalies. Prenatal growth retardation was detected in a relatively small number of cases, but postnatal growth failure was a constant feature. In all cases, the clinical diagnosis was confirmed by genetic analyses: karyotype and/or MLPA. In conclusion, renal and brain defects, as well as immunodeficiency are rare manifestations and should be looked for. Although CMA is the standard test, in our experience, MLPA is also a reliable screening method as the identified cases were either confirmed by MLPA or selected for further investigations.
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Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Tipo de estudio: Prognostic_studies Idioma: En Revista: Children (Basel) Año: 2021 Tipo del documento: Article País de afiliación: Rumanía

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Tipo de estudio: Prognostic_studies Idioma: En Revista: Children (Basel) Año: 2021 Tipo del documento: Article País de afiliación: Rumanía