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Pre- and postnatal findings in a patient with a recombinant chromosome rec(8)(qter→q21.11::p23.3→qter) due to a paternal pericentric inversion inv(8)(p23.3q21.11) and review of the literature.
Habhab, Wisam; Mau-Holzmann, Ulrike; Singer, Sylke; Rieß, Angelika; Kagan, Karl-Oliver; Gerbig, Ines; Schäferhoff, Karin; Dufke, Andreas; Kehrer, Martin.
Afiliación
  • Habhab W; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
  • Mau-Holzmann U; Department of Genetic Medicine, Faculty of Medicine, King Abdulaziz University, Jeddah, Saudi Arabia.
  • Singer S; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
  • Rieß A; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
  • Kagan KO; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
  • Gerbig I; Department of Obstetrics and Gynecology, University of Tübingen, Tübingen, Germany.
  • Schäferhoff K; University Children's Hospital, University of Tübingen, Tübingen, Germany.
  • Dufke A; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
  • Kehrer M; Institute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
Am J Med Genet A ; 182(11): 2680-2684, 2020 11.
Article en En | MEDLINE | ID: mdl-32803851
Recombinant chromosome 8 (Rec8) syndrome (San Luis Valley [SLV] syndrome; OMIM #179613) is a rare chromosome disorder associated with intellectual disability, congenital heart defects, variable skeletal and urogenital anomalies, and dysmorphic features. It is characterized by a partial terminal deletion of 8p and a partial terminal duplication of 8q, which is usually due to meiotic recombination of a pericentric inversion of chromosome 8 of a healthy carrier parent. There are only few reports of cases with breakpoints defined at the molecular level by molecular karyotyping. We report on a case of Rec8 syndrome with previously unreported breakpoints in a male fetus with intrauterine growth restriction, hypogenesis of the corpus callosum, bilateral cleft lip/palate, and congenital heart defect. Cytogenetic analysis revealed a recombinant chromosome 8 [46,XY,rec(8)(qter→q21.11::p23.3→qter)] secondary to a paternal pericentric inversion [46,XY,inv(8)(p23.3q21.11)]. Molecular karyotyping correspondingly showed a terminal copy number loss of 1.4 Mb (arr[hg19] 8p23.3(158048_1514749)×1) and a terminal copy number gain of chromosome band 8q21.11q24.3 of 69.8 Mb (arr[hg19] 8q21.11q24.3(76477367_146295771)×3). To our knowledge, this is the fourth reported case diagnosed prenatally. We describe the postnatal clinical course of the male newborn. Furthermore, we review and compare the phenotypic features and breakpoints of 74 reported Rec8/SLV cases.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Anomalías Múltiples / Cromosomas Humanos Par 8 / Trastornos de los Cromosomas / Enfermedades Fetales / Inversión Cromosómica Tipo de estudio: Diagnostic_studies Límite: Adult / Female / Humans / Male / Newborn Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2020 Tipo del documento: Article País de afiliación: Alemania

Texto completo: 1 Colección: 01-internacional Banco de datos: MEDLINE Asunto principal: Anomalías Múltiples / Cromosomas Humanos Par 8 / Trastornos de los Cromosomas / Enfermedades Fetales / Inversión Cromosómica Tipo de estudio: Diagnostic_studies Límite: Adult / Female / Humans / Male / Newborn Idioma: En Revista: Am J Med Genet A Asunto de la revista: GENETICA MEDICA Año: 2020 Tipo del documento: Article País de afiliación: Alemania