Mother and daughter with Kenny-Caffey syndrome: the adult phenotype

Kenny-Caffey Syndrome (KCS) is a genetic syndrome characterized by growth retardation with short stature, cortical thickening and medullary stenosis of long bones, and hypoparathyroidism with hypocalcemia. KCS and the related but more severe condition osteocraniostenosis are determined by monoalleli...

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Veröffentlicht in:European journal of medical genetics 2024-06, Vol.69, p.104943-104943, Article 104943
Hauptverfasser: Tonelli, L., Sanchini, M., Margutti, A., Buldrini, B., Superti-Furga, A., Ferlini, A., Selvatici, R., Bigoni, S.
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Sprache:eng
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Zusammenfassung:Kenny-Caffey Syndrome (KCS) is a genetic syndrome characterized by growth retardation with short stature, cortical thickening and medullary stenosis of long bones, and hypoparathyroidism with hypocalcemia. KCS and the related but more severe condition osteocraniostenosis are determined by monoallelic variants in the FAM111A gene. Here we describe the KCS phenotype resulting from the monoallelic FAM111A variant p.Y511H in a 31-year-old woman and in her 56-year-old mother, who is one of the oldest affected individuals known so far. To our knowledge, it is also one of the few molecularly confirmed cases of a mother-to-child transmission of KCS.
ISSN:1769-7212
1878-0849
DOI:10.1016/j.ejmg.2024.104943