Literature DB >> 16801186

Partial cerebellar hypoplasia in a patient with Prader-Willi syndrome.

Luigi Titomanlio1, Daniele De Brasi, Alfonso Romano, Rita Genesio, Alvaro A Diano, Ennio Del Giudice.   

Abstract

UNLABELLED: We report a 3-y-old male infant with Prader-Willi syndrome (PWS) caused by a de novo interstitial deletion of 15q11-q13. Additional features included a right cerebellar hemisphere hypoplasia. The extent of deletion was determined by FISH analysis using an SNRPN PW/AS probe that maps in the PWS/AS critical region (CR) and with specific 15q BACs. We unravelled an interstitial 15q11.2-q13.1 deletion spanning about 3 Mb.
CONCLUSION: To date only a few other PWS patients--including autopsy cases--with CNS structural anomalies have been described. Our case report adds knowledge to the issue of brain involvement in Prader-Willi syndrome. Further MRI studies of PWS patients will be helpful to clarify a correlation between PWS and brain abnormalities.

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Year:  2006        PMID: 16801186     DOI: 10.1080/08035250500527307

Source DB:  PubMed          Journal:  Acta Paediatr        ISSN: 0803-5253            Impact factor:   2.299


  6 in total

1.  The neuroanatomy of genetic subtype differences in Prader-Willi syndrome.

Authors:  Robyn A Honea; Laura M Holsen; Rebecca J Lepping; Rodrigo Perea; Merlin G Butler; William M Brooks; Cary R Savage
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2012-03       Impact factor: 3.568

2.  Structural malformations of the brain, eye, and pituitary gland in PHACE syndrome.

Authors:  Jack E Steiner; Garrett N McCoy; Christopher P Hess; William B Dobyns; Denise W Metry; Beth A Drolet; Mohit Maheshwari; Dawn H Siegel
Journal:  Am J Med Genet A       Date:  2017-11-24       Impact factor: 2.802

3.  Unilateral cerebellar hypoplasia with different clinical features.

Authors:  Gulcin Benbir; Simay Kara; Beyza Citci Yalcinkaya; Geysu Karhkaya; Beyhan Tuysuz; Naci Kocer; Cengiz Yalcinkaya
Journal:  Cerebellum       Date:  2011-03       Impact factor: 3.847

4.  Small gray matter volume in orbitofrontal cortex in Prader-Willi syndrome: a voxel-based MRI study.

Authors:  Kaeko Ogura; Toshikatsu Fujii; Nobuhito Abe; Yoshiyuki Hosokai; Mayumi Shinohara; Shoki Takahashi; Etsuro Mori
Journal:  Hum Brain Mapp       Date:  2010-07-28       Impact factor: 5.038

5.  Early childhood obesity is associated with compromised cerebellar development.

Authors:  Jennifer L Miller; Jessica Couch; Krista Schwenk; Michelle Long; Stephen Towler; Douglas W Theriaque; Guojun He; Yijun Liu; Daniel J Driscoll; Christiana M Leonard
Journal:  Dev Neuropsychol       Date:  2009       Impact factor: 2.253

6.  One Year of Recombinant Human Growth Hormone Treatment in Adults with Prader-Willi Syndrome Improves Body Composition, Motor Skills and Brain Functional Activity in the Cerebellum.

Authors:  Laia Casamitjana; Laura Blanco-Hinojo; Olga Giménez-Palop; Jesús Pujol; Gerard Martínez-Vilavella; Susanna Esteba-Castillo; Rocío Pareja; Valentín Freijo; Laura Vigil; Joan Deus; Assumpta Caixàs
Journal:  J Clin Med       Date:  2022-03-25       Impact factor: 4.241

  6 in total

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