Clinical Genetics · 1997 · 22 citations · 23 references
Life SciencesGeneticsMolecular GeneticsCytogeneticsHuman GeneticsMedical GeneticsChromosomal AbnormalitiesChromosomal DisordersClinical CytogeneticsGenetic DisordersGenetic SyndromesStructural Chromosome AbnormalitiesAutosomal Dominant InheritanceChromosomal DeletionsChromosome DeletionsChromosome MicrodeletionsCytogenetic AbnormalitiesMicrodeletion Syndrome DetectionMicrodeletion SyndromesZinc Finger Proteins
A male had several features of Greig cephalopolysyndactyly syndrome (GCPS) and significant developmental delay. He was found to have a de novo chromosomal deletion of chromosome no. 7 involving p13; this resulted in loss of the zinc finger gene, GLI3, which is the candidate gene in this syndrome. Modification of the CGPS phenotype in a sporadic case emphasizes the importance of searching for a chromosomal origin of this autosomal dominant disorder. Detection of a chromosomal deletion in these patients may be associated with a poor prognosis from the standpoint of cognitive development, and the potential for other structural abnormalities not normally associated with GCPS.
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GLI3 frameshift mutations cause autosomal dominant Pallister-Hall syndrome
Seongman Kang, John M. Graham, Ann Haskins Olney et al. · Nature Genetics · 1997 · 510 citations
Judith G. Hall, Philip D. Pallister, Sterling K. Clarren et al. · American Journal of Medical Genetics · 1980 · 346 citations