Literature DB >> 9295069

Acute lymphoblastic leukemia in a child with the CHIME neuroectodermal dysplasia syndrome.

R E Schnur1, B H Greenbaum, W R Heymann, K Christensen, A S Buck, C S Reid.   

Abstract

The "CHIME" syndrome (MIM#280000) is a rare neuroectodermal disorder comprised of Colobomas of the eye, Heart defects, Ichthyosiform dermatosis, Mental retardation, and Ear defects. We report on the sixth child with this syndrome and the first of these to develop acute lymphoblastic leukemia at age 4 1/2 years. Her major problems included a migratory ichthyosiform dermatosis, multiple skin infections and infestations, bilateral retinal coloboma, developmental delay, seizures, infantile macrosomia, facial anomalies, a duplicated renal collecting system, and conductive hearing loss. Histologic examination of the skin demonstrated findings of an epidermal nevus with deep rete pegs, hyperkeratosis, and a markedly increased granular layer. The cause of the CHIME syndrome is unknown, but the disorder is easily recognized because of the striking phenotype. The diagnosis is important to make because of the potential for associated congenital heart disease, neurologic compromise, possible autosomal recessive inheritance, and possible association with malignancy.

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Year:  1997        PMID: 9295069

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  3 in total

1.  Mutations in the glycosylphosphatidylinositol gene PIGL cause CHIME syndrome.

Authors:  Bobby G Ng; Karl Hackmann; Melanie A Jones; Alexey M Eroshkin; Ping He; Roy Wiliams; Shruti Bhide; Vincent Cantagrel; Joseph G Gleeson; Amy S Paller; Rhonda E Schnur; Sigrid Tinschert; Janice Zunich; Madhuri R Hegde; Hudson H Freeze
Journal:  Am J Hum Genet       Date:  2012-03-22       Impact factor: 11.025

2.  Hyperphosphatasia with mental retardation syndrome, expanded phenotype of PIGL related disorders.

Authors:  Ruqaiah Altassan; Stephanie Fox; Chantal Poulin; Daniela Buhas
Journal:  Mol Genet Metab Rep       Date:  2018-02-06

3.  Large deletion in PIGL: a common mutational mechanism in CHIME syndrome?

Authors:  José Rm Ceroni; Guilherme L Yamamoto; Rachel S Honjo; Chong A Kim; Maria R Passos-Bueno; Débora R Bertola
Journal:  Genet Mol Biol       Date:  2018-02-19       Impact factor: 1.771

  3 in total

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