Rearrangement of chromosome 15 in the region q11.2----q12 in an individual with obesity syndrome and her normal mother.

Rearrangement of the proximal long arm of chromosome 15 have been found in most patients with the Prader-Willi syndrome (PWS) and in some with Angelman syndrome. We present an individual with syndromic obesity and her normal mother, who both have an abnormal chromosome 15. The proposita is a 26-year-old women with marked obesity, acanthosis, nigricans, short fingers, and severe cone degeneration of the retina. She has high plasma insulin levels, hypothyroidism, and an empty sella on CT scan. High-resolution chromosome banding demonstrated an increase in band 15q12. Further analysis showed the same abnormal 15 in her normal mother but not in her normal sister. This case and recent reports in the literature indicate that duplication of chromosome 15q in the PWS region may be associated with a syndrome of obesity, acanthosis nigricans, empty sella, and rodcore dystrophy as well as with a normal phenotype. Whether normal individuals with such a duplication carry increased risk of having offspring with an obesity syndrome is yet to be determined.

[1]  D. Ledbetter,et al.  Second Annual Prader‐Willi Syndrome Scientific Conference , 1987 .

[2]  D. Ledbetter,et al.  Duplication of proximal 15q as a cause of Prader-Willi syndrome. , 1987, American journal of medical genetics.

[3]  A. Veleba,et al.  Proximal 15q variant with normal phenotype in three unrelated individuals , 1987, Clinical genetics.

[4]  E. Smith,et al.  Obesity, acanthosis nigricans, insulin resistance, and hyperandrogenemia: pediatric perspective and natural history. , 1985, The Journal of pediatrics.

[5]  S. Schwartz,et al.  Deletions of proximal 15q and non-classical Prader-Willi syndrome phenotypes. , 1985, American journal of medical genetics.

[6]  F. Beemer,et al.  Duplication in chromosome 15q in a boy with the Prader‐Willi syndrome; further cytogenetic confusion , 1984, Clinical genetics.

[7]  A. Köhler,et al.  Duplication or insertion in 15q11–13 associated with mental retardation ‐short stature and obesity ‐ Prader‐Willi or Cohen syndrome? , 1984, Clinical genetics.

[8]  S. Cassidy,et al.  Deletion of chromosome 15 (q11-13) in a Prader-Labhart-Willi syndrome clinic population. , 1984, American journal of medical genetics.

[9]  B. Kousseff The cytogenetic controversy in the Prader-Labhart-Willi syndrome. , 1982, American journal of medical genetics.

[10]  D. Ledbetter,et al.  Chromosome 15 abnormalities and the Prader-Willi syndrome: a follow-up report of 40 cases. , 1982, American journal of human genetics.

[11]  J. Heckenlively,et al.  Telangiectasia and optic atrophy in cone-rod degenerations. , 1981, Archives of ophthalmology.

[12]  D. Ledbetter,et al.  Deletions of chromosome 15 as a cause of the Prader-Willi syndrome. , 1981, The New England journal of medicine.

[13]  J. Yunis High resolution of human chromosomes. , 1976, Science.