Literature DB >> 16222663

Further delineation of the phenotype maps for partial trisomy 16q24 and Jacobsen syndrome by a subtle familial translocation t(11;16)(q24.2;q24.1).

Susanne Zahn1, Antje Ehrbrecht, Kristin Bosse, Vera Kalscheuer, Peter Propping, Gesa Schwanitz, Beate Albrecht, Hartmut Engels.   

Abstract

We report on two cases of distal monosomy 11q and partial trisomy 16q due to a familial subtle translocation detected by FISH subtelomere screening. Exact breakpoint analyses by FISH with panels of BAC probes demonstrated a 9.3-9.5 megabase partial monosomy of 11q24.2-qter and a 4.9-5.4 megabase partial trisomy of 16q24.1-qter. The index patient displayed craniofacial dysmorphisms, mild mental retardation and postnatal growth retardation, muscular hypotonia, mild periventricular leukodystrophy, patent ductus arteriosus, thrombocytopenia, recurrent infections, inguinal hernia, cryptorchidism, pes equinovarus, and hearing deficiencies. In his mother's cousin who bears the identical unbalanced translocation, mild mental retardation, patent ductus arteriosus, hypogammaglobulinemia, recurrent infections, unilateral kidney hypoplasia, pes equinovarus, and hearing deficiencies were reported. Since only four descriptions of cryptic or subtle partial trisomies 16q have been published to date, our patients contribute greatly to the delineation of the phenotype of this genomic imbalance. In contrast to this, terminal deletions of the long arm of chromosome 11 cause a haploinsufficiency disorder (Jacobsen syndrome) in which karyotype-phenotype correlations are already being established. Here, our findings contribute to the refinement of a phenotype map for several Jacobsen syndrome features including abnormal brain imaging, renal malformations, thrombocytopenia/pancytopenia, inguinal hernia, testicular ectopy, pes equinovarus, and hearing deficiency.

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Year:  2005        PMID: 16222663     DOI: 10.1002/ajmg.a.30995

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

1.  Immune Deficiency in Jacobsen Syndrome: Molecular and Phenotypic Characterization.

Authors:  Raquel Rodríguez-López; Fátima Gimeno-Ferrer; Elena Montesinos; Irene Ferrer-Bolufer; Carola Guzmán Luján; David Albuquerque; Carolina Monzó Cataluña; Virginia Ballesteros; Monserrat Aleu Pérez-Gramunt
Journal:  Genes (Basel)       Date:  2021-07-31       Impact factor: 4.096

Review 2.  Jacobsen syndrome.

Authors:  Teresa Mattina; Concetta Simona Perrotta; Paul Grossfeld
Journal:  Orphanet J Rare Dis       Date:  2009-03-07       Impact factor: 4.123

Review 3.  Partial monosomy 8p and trisomy 16q in two children with developmental delay detected by array comparative genomic hybridization.

Authors:  Zoe Papadopoulou; Ioannis Papoulidis; Stavros Sifakis; Georgios Markopoulos; Annalisa Vetro; Angeliki-Maria Vlaikou; Monica Ziegler; Thomas Liehr; Loretta Thomaidis; Orsetta Zuffardi; Maria Syrrou; Kitsos George; Emmanouil Manolakos
Journal:  Mol Med Rep       Date:  2017-10-10       Impact factor: 2.952

4.  A rare description of pure partial trisomy of 16q12.2q24.3 and review of the literature.

Authors:  Joshua Manor; Daniela Dinu; Mahshid S Azamian; Weimin Bi; Sandra Darilek; Seema R Lalani
Journal:  Am J Med Genet A       Date:  2021-06-01       Impact factor: 2.578

5.  Submicroscopic deletions of 11q24-25 in individuals without Jacobsen syndrome: re-examination of the critical region by high-resolution array-CGH.

Authors:  Christine Tyson; Ying Qiao; Chansonette Harvard; Xudong Liu; Francois P Bernier; Barbara McGillivray; Sandra A Farrell; Laura Arbour; Albert E Chudley; Lorne Clarke; William Gibson; Sarah Dyack; Ross McLeod; Teresa Costa; Margot I Vanallen; Siu-Li Yong; Gail E Graham; Patrick Macleod; Millan S Patel; Jane Hurlburt; Jeanette Ja Holden; Suzanne Me Lewis; Evica Rajcan-Separovic
Journal:  Mol Cytogenet       Date:  2008-11-11       Impact factor: 2.009

  5 in total

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