Literature DB >> 18301319

A case report of monozygotic twins with Smith-Magenis syndrome.

Matthew Hicks1, Susan Ferguson, Francois Bernier, Jean-François Lemay.   

Abstract

Monozygotic 3.5-year-old twin boys presented for developmental assessment with a history of global developmental delay, behavioral issues including self-harm, and severe receptive and expressive language delays. Chromosome testing confirmed for both a 17p11.2 interstitial microdeletion commonly seen in Smith-Magenis syndrome (SMS), which is characterized by developmental delay, cognitive impairment, and facial and behavioral phenotype. To our knowledge, this is the first description in the literature of monozygotic twins with SMS. Despite their zygosity, the twins had marked differences in presentation including cardiac and renal anomalies, language development, and behavioral phenotype. Both twins displayed disordered speech development, impairments in social interaction, and stereotyped behaviors consistent with autism spectrum disorder, common in the vast majority of cases of SMS. Examining the differences in behavioral and clinical phenotype in monozygotic twins may lead to a better understanding of the cause of the clinical variability seen in SMS, as well as the natural history of this syndrome.

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Year:  2008        PMID: 18301319     DOI: 10.1097/DBP.0b013e318146903d

Source DB:  PubMed          Journal:  J Dev Behav Pediatr        ISSN: 0196-206X            Impact factor:   2.225


  2 in total

1.  Neurodevelopment of children under 3 years of age with Smith-Magenis syndrome.

Authors:  Pamela L Wolters; Andrea L Gropman; Staci C Martin; Michaele R Smith; Hanna L Hildenbrand; Carmen C Brewer; Ann C M Smith
Journal:  Pediatr Neurol       Date:  2009-10       Impact factor: 3.372

2.  Behavioral disturbance and treatment strategies in Smith-Magenis syndrome.

Authors:  Alice Poisson; Alain Nicolas; Pierre Cochat; Damien Sanlaville; Caroline Rigard; Hélène de Leersnyder; Patricia Franco; Vincent Des Portes; Patrick Edery; Caroline Demily
Journal:  Orphanet J Rare Dis       Date:  2015-09-04       Impact factor: 4.123

  2 in total

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