Literature DB >> 22605691

Morbid obesity in a child with monosomy 1p36 syndrome.

Ana Zagalo1, Patricia Dias, Carla Pereira, Maria de Lurdes Sampaio.   

Abstract

The monosomy 1p36 syndrome is a cause of syndromic obesity. It is characterised by psychomotor delay, hypotonia and typical craniofacial dysmorphism. Other features commonly associated are behavioural anomalies including hyperphagia and self-injuring, seizures, congenital heart disease and hypothyroidism. The authors report the case of a 9-year and 5-month-boy referred to the paediatric endocrinology clinics for morbid obesity. Clinical findings were generalised obesity with a body mass index >95th centile, acanthosis nigricans of the neck, arms with self inflicted lesions, deep-set eyes, straight eyebrows, broad nasal bridge and pointed chin. He was unable to walk and had no expressive language. Cytogenetic analysis identified 1p36.33-pter deletion (~139 Mb terminal deletion in chromosome 1 short arm) and Y chromosome duplication. The blood analysis showed insulin resistance and dyslipidaemia. The authors emphasise the need to consider monosomy 1p36 as a cause of severe psychomotor delay and obesity.

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Year:  2012        PMID: 22605691      PMCID: PMC3316789          DOI: 10.1136/bcr.01.2012.5503

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  6 in total

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3.  Refinement of causative genes in monosomy 1p36 through clinical and molecular cytogenetic characterization of small interstitial deletions.

Authors:  Jill A Rosenfeld; John A Crolla; Susan Tomkins; Patricia Bader; Bernice Morrow; Jerome Gorski; Robin Troxell; Cynthia Forster-Gibson; Deirdre Cilliers; R Gordon Hislop; Allen Lamb; Beth Torchia; Blake C Ballif; Lisa G Shaffer
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4.  Diagnosis and mortality in 47,XYY persons: a registry study.

Authors:  Kirstine Stochholm; Svend Juul; Claus H Gravholt
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5.  Further delineation of deletion 1p36 syndrome in 60 patients: a recognizable phenotype and common cause of developmental delay and mental retardation.

Authors:  Agatino Battaglia; H Eugene Hoyme; Bruno Dallapiccola; Elaine Zackai; Louanne Hudgins; Donna McDonald-McGinn; Nadia Bahi-Buisson; Corrado Romano; Charles A Williams; Lisa L Brailey; Lisa L Braley; Sameer M Zuberi; John C Carey
Journal:  Pediatrics       Date:  2008-02       Impact factor: 7.124

6.  Extending the phenotype of monosomy 1p36 syndrome and mapping of a critical region for obesity and hyperphagia.

Authors:  Carla S D'Angelo; Ilana Kohl; Monica Castro Varela; Cláudia I E de Castro; Chong A Kim; Débora R Bertola; Charles M Lourenço; Célia P Koiffmann
Journal:  Am J Med Genet A       Date:  2010-01       Impact factor: 2.802

  6 in total
  3 in total

1.  Partial deficiency of CTRP12 alters hepatic lipid metabolism.

Authors:  Stefanie Y Tan; Hannah C Little; Xia Lei; Shuoyang Li; Susana Rodriguez; G William Wong
Journal:  Physiol Genomics       Date:  2016-11-04       Impact factor: 3.107

2.  CTRP12 inhibits triglyceride synthesis and export in hepatocytes by suppressing HNF-4α and DGAT2 expression.

Authors:  Stefanie Y Tan; Hannah C Little; Dylan C Sarver; Paul A Watkins; G William Wong
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3.  Identification of 1p36 deletion syndrome in patients with facial dysmorphism and developmental delay.

Authors:  Go Hun Seo; Ja Hye Kim; Ja Hyang Cho; Gu-Hwan Kim; Eul-Ju Seo; Beom Hee Lee; Jin-Ho Choi; Han-Wook Yoo
Journal:  Korean J Pediatr       Date:  2016-01-22
  3 in total

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