Literature DB >> 7735502

Trisomy 17p11-pter: unbalanced pericentric inversion, inv(17)(p11q25) in two patients, unbalanced translocations t(4;17)(q27;p11) in a newborn and t(4;17) (p16;p11.2) in a fetus.

I W Lurie1, D B Gurevich, F Binkert, A Schinzel.   

Abstract

Three patients and one fetus with almost complete trisomy 17p due to familial rearrangements are described. Two patients followed unbalanced transmission of a familial pericentric inversion, and one patient and one fetus were due to unbalanced segregation of familial translocations. In the inversion family, another two patients with multiple malformations had died before chromosome examination could be performed. The pattern of congenital anomalies as revealed from eleven cases of trisomy 17p11-pter include as the most prominent features: prenatal growth retardation, microcephaly, downslanting palpebral fissures, small mouth, small mandible, poorly shaped ears, short and webbed neck, genital hypoplasia, clinodactyly of fingers, crowding of toes, a high incidence of congenital heart defects and hernias. Postnatal survival is short mainly in patients with congenital heart defects. From the age of about 6 years onward, clinical findings become more distinct, with some signs of Charcot-Marie-Tooth neuropathy (pes cavus, adducted thumbs, dorsiflexed hallux, camptodactyly and limitation of movements in different joints), and the nose gets narrow and sharp, with hypoplastic alae.

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Year:  1995        PMID: 7735502

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  4 in total

1.  Evidence for involvement of TRE-2 (USP6) oncogene, low-copy repeat and acrocentric heterochromatin in two families with chromosomal translocations.

Authors:  Zhishuo Ou; Małgorzata Jarmuz; Steven P Sparagana; Jacques Michaud; Jean-Claude Décarie; Svetlana A Yatsenko; Beata Nowakowska; Patti Furman; Chad A Shaw; Lisa G Shaffer; James R Lupski; A Craig Chinault; Sau W Cheung; Paweł Stankiewicz
Journal:  Hum Genet       Date:  2006-06-22       Impact factor: 4.132

2.  De novo inv(17)(p11.2q21.3) in an intellectually disabled girl: appraisal of 21 inv(17) constitutional instances.

Authors:  Miriam Partida-Pérez; María G Domínguez; Vivian Alejandra Neira; Luis E Figuera; Horacio Rivera
Journal:  J Genet       Date:  2012-08       Impact factor: 1.166

3.  A molecular, cytogenetic, and clinical evaluation of mosaic tandem duplication 17p and Charcot-Marie-Tooth type 1A neuropathy.

Authors:  K S Reddy; M B Larsen
Journal:  J Med Genet       Date:  1998-02       Impact factor: 6.318

4.  Road to a rare diagnosis: Description of novel unbalanced translocation causing partial trisomy 17p.

Authors:  Melab Musabi; Ayman Saker; Jessi Baer; Peter Wang; Anahita Mohseni Meybodi; Chitra Prasad; Soume Bhattacharya
Journal:  Clin Case Rep       Date:  2022-10-06
  4 in total

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