Genoa syndrome and central diabetes insipidus: A case report


Hacihamdioǧlu B., ŞIKLAR Z., Erdeve Ş. S., BERBEROĞLU M., Deda G., Tiraş S., ...More

JCRPE Journal of Clinical Research in Pediatric Endocrinology, vol.2, no.2, pp.89-91, 2010 (SCI-Expanded, Scopus)

  • Publication Type: Article / Article
  • Volume: 2 Issue: 2
  • Publication Date: 2010
  • Doi Number: 10.4274/jcrpe.v2i2.89
  • Journal Name: JCRPE Journal of Clinical Research in Pediatric Endocrinology
  • Journal Indexes: Science Citation Index Expanded (SCI-EXPANDED), Scopus
  • Page Numbers: pp.89-91
  • Keywords: Central diabetes insipidus, Cleft lip/palate, Craniosynostosis, Genoa syndrome, Holoprosencephaly
  • Ankara University Affiliated: Yes

Abstract

Genoa syndrome was first described by Camera et al in 1993 in two patients with semilobar holoprosencephaly (HPE), craniosynostosis and abnormal small hands with cone-shaped epiphyses and hypoplastic terminal phalanges of fingers (OMIM: 601370). In 2001, Lapunzina et al reported a case of craniosynostosis and HPE associated with several other malformations and suggested that these findings could be attributed to a severe form of Genoa syndrome or to a newly recognized syndrome. Endocrinopathies in association with HPE are frequently reported in the literature. Diabetes insipidus, hypothyroidism, hypocortisolism, and growth hormone deficiency are frequently associated with HPE. We here report a case of semilobar HPE, craniosynostosis and cleft lip/palate, possibly a case of Genoa syndrome, associated with central diabetes insipidus. © Journal of Clinical Research in Pediatric Endocrinology.