Heterozygous HESX1 mutations associated with isolated congenital pituitary hypoplasia and septo-optic dysplasia

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Heterozygous HESX1 mutations associated with isolated congenital pituitary hypoplasia and septo-optic dysplasia.

We have previously shown that familial septo-optic dysplasia (SOD), a syndromic form of congenital hypopituitarism involving optic nerve hypoplasia and agenesis of midline brain structures, is associated with homozygosity for an inactivating mutation in the homeobox gene HESX1/Hesx1 in man and mouse. However, as most SOD/congenital hypopituitarism occurs sporadically, the possible contribution ...

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Septo-optic dysplasia with olfactory tract hypoplasia

Copyright © Journal of Pediatric Neurosciences Septo-optic dysplasia with olfactory tract hypoplasia Manuel Ribeiro, Álvaro Machado, and João Soares-Fernandes Department of Neuradiology, Hospital de São Marcos, Braga, Portugal. Department of Neurology, Hospital de São Marcos, Braga, Portugal. Address for Correspondence: Dr. Alvaro Machado, Hospital de São Marcos Largo Carlos Amarante Ap 4200 Br...

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Molecular analysis of the PROP1 and HESX1 genes in patients with septo-optic dysplasia and/or pituitary hormone deficiency.

OBJECTIVE The present study aimed at evaluating the PROP1 and HESX1 genes in a group of patients with septo-optic dysplasia (SOD) and pituitary hormone deficiency (combined - CPHD; isolated GH deficiency - GHD). Eleven patients with a clinical and biochemical presentation consistent with CPHD, GHD or SOD were evaluated. SUBJECTS AND METHODS In all patients, the HESX1 gene was analyzed by dire...

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Ectopic posterior pituitary lobe and periventricular heterotopia: cerebral malformations with the same underlying mechanism?

BACKGROUND AND PURPOSE Ectopic posterior pituitary lobe often occurs in children with growth hormone deficiency and is part of the spectrum associated with septo-optic dysplasia. Some cases of septo-optic dysplasia are caused by homozygous mutations in the homeobox gene HESX1, whereas heterozygous mutations are associated with milder phenotypes. To date, HESX1 is the only gene associated with e...

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Monocular nystagmus with sectoral optic nerve hypoplasia in a patient with septo-optic dysplasia.

This case depicts an unusual presentation of septo-optic dysplasia. A four-year-old female presented with monocular nystagmus and temporal optic disc pallor in her left eye. Despite a normal sized optic nerve head, magnetic resonance imaging (MRI) showed a hypoplastic intraorbital and intracranial left optic nerve in the absence of a septum pellucidum. She was subsequently diagnosed with septo-...

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ژورنال

عنوان ژورنال: Human Molecular Genetics

سال: 2001

ISSN: 1460-2083

DOI: 10.1093/hmg/10.1.39