Literature DB >> 6846278

Septo-optic dysplasia and median cleft face syndrome in a patient with isolated growth hormone deficiency and hyperprolactinemia.

C Stewart, M Castro-Magana, J Sherman, M Angulo, P J Collipp.   

Abstract

A pituitary evaluation was carried out in a 12-year-old girl who had early puberty, short stature, optic nerve hypoplasia, and agenesis of the corpus callosum. Her growth hormone (GH) response to insulin-induced hypoglycemia and arginine infusion was blunted. Basal and stimulated levels of prolactin were elevated. The association of GH deficiency and hyperprolactinemia suggests a disruption of the dopaminergic modulation of these hormones. The facial features seen in our patient, such as hypertelorism, V-shaped frontal hairline, and cleft nose and upper lip, are enough to justify the diagnosis of median cleft face (MCF) syndrome. However, the optic nerve hypoplasia and the GH deficiency are characteristics of septo-optic dysplasia, and, to our knowledge, they have never been described in patients with MCF syndrome. Our case fulfills the diagnostic criteria of both, representing a link between both ends of this spectrum.

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Year:  1983        PMID: 6846278     DOI: 10.1001/archpedi.1983.02140310062018

Source DB:  PubMed          Journal:  Am J Dis Child        ISSN: 0002-922X


  4 in total

Review 1.  Optic nerve hypoplasia in children.

Authors:  S M Zeki; G N Dutton
Journal:  Br J Ophthalmol       Date:  1990-05       Impact factor: 4.638

2.  Endocrine disorders in septo-optic dysplasia (De Morsier syndrome)--evaluation and follow up of 18 patients.

Authors:  S Willnow; W Kiess; O Butenandt; H G Dorr; A Enders; B Strasser-Vogel; J Egger; H P Schwarz
Journal:  Eur J Pediatr       Date:  1996-03       Impact factor: 3.183

3.  Endocrine status of patients with septo-optic dysplasia: fourteen Japanese cases.

Authors:  Mikiko Koizumi; Shinobu Ida; Yasuko Shoji; Yuri Etani; Yoshikazu Hatsukawa; Nobuhiko Okamoto
Journal:  Clin Pediatr Endocrinol       Date:  2017-04-22

4.  Acceleration of puberty during growth hormone therapy in a child with septo-optic dysplasia.

Authors:  Gönül Catlı; Ayça Altıncık; Ahmet Anık; Korcan Demir; Handan Güleryüz; Ayhan Abacı; Ece Böber
Journal:  J Clin Res Pediatr Endocrinol       Date:  2014
  4 in total

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