Literature DB >> 10051020

Absent pituitary gland and hypoplasia of the cerebellar vermis associated with partial ophthalmoplegia and postaxial polydactyly: a variant of orofaciodigital syndrome VI or a new syndrome?

L I Al-Gazali1, L Sztriha, J Punnose, W Shather, M Nork.   

Abstract

We report two sibs with features overlapping those of orofaciodigital syndrome type VI (Varadi syndrome). Both presented at birth with oculomotor abnormalities, dysmorphic facial features, and dysgenesis of the cerebellar vermis. There were minimal oral manifestations (high arched palate) in both of them and one had postaxial polydactyly of both hands and one foot. In addition, there was evidence of aplasia of the pituitary gland on MRI scan in both of them with evidence of hypopituitarism. Both responded well to hormone replacement therapy with improvement in their linear growth and mental ability. These cases may represent a new autosomal recessive midline defect syndrome with features overlapping OFDS VI. Alternatively the features in these children could represent variability within OFDS VI.

Entities:  

Mesh:

Year:  1999        PMID: 10051020      PMCID: PMC1734312     

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  5 in total

Review 1.  Joubert Syndrome and related disorders.

Authors:  Francesco Brancati; Bruno Dallapiccola; Enza Maria Valente
Journal:  Orphanet J Rare Dis       Date:  2010-07-08       Impact factor: 4.123

2.  Joubert syndrome with multiple pituitary hormone deficiency.

Authors:  Nese Akcan; Firdevs Bas; Sukran Poyrazoglu; Ruveyde Bundak
Journal:  BMJ Case Rep       Date:  2019-04-23

3.  Congenital adenohypophysis aplasia: clinical features and analysis of the transcriptional factors for embryonic pituitary development.

Authors:  T Arrigo; M Wasniewska; F De Luca; M Valenzise; F Lombardo; D Vivenza; T Vaccaro; E Coradi; A Biason-Lauber
Journal:  J Endocrinol Invest       Date:  2006-03       Impact factor: 4.256

4.  Delineation and diagnostic criteria of Oral-Facial-Digital Syndrome type VI.

Authors:  Andrea Poretti; Giuseppina Vitiello; Raoul C M Hennekam; Filippo Arrigoni; Enrico Bertini; Renato Borgatti; Francesco Brancati; Stefano D'Arrigo; Francesca Faravelli; Lucio Giordano; Thierry A G M Huisman; Miriam Iannicelli; Gerhard Kluger; Marten Kyllerman; Magnus Landgren; Melissa M Lees; Lorenzo Pinelli; Romina Romaniello; Ianina Scheer; Christoph E Schwarz; Ronen Spiegel; Daniel Tibussek; Enza Maria Valente; Eugen Boltshauser
Journal:  Orphanet J Rare Dis       Date:  2012-01-11       Impact factor: 4.123

Review 5.  Fifteen years of research on oral-facial-digital syndromes: from 1 to 16 causal genes.

Authors:  Ange-Line Bruel; Brunella Franco; Yannis Duffourd; Julien Thevenon; Laurence Jego; Estelle Lopez; Jean-François Deleuze; Diane Doummar; Rachel H Giles; Colin A Johnson; Martijn A Huynen; Véronique Chevrier; Lydie Burglen; Manuela Morleo; Isabelle Desguerres; Geneviève Pierquin; Bérénice Doray; Brigitte Gilbert-Dussardier; Bruno Reversade; Elisabeth Steichen-Gersdorf; Clarisse Baumann; Inusha Panigrahi; Anne Fargeot-Espaliat; Anne Dieux; Albert David; Alice Goldenberg; Ernie Bongers; Dominique Gaillard; Jesús Argente; Bernard Aral; Nadège Gigot; Judith St-Onge; Daniel Birnbaum; Shubha R Phadke; Valérie Cormier-Daire; Thibaut Eguether; Gregory J Pazour; Vicente Herranz-Pérez; Jaclyn S Goldstein; Laurent Pasquier; Philippe Loget; Sophie Saunier; André Mégarbané; Olivier Rosnet; Michel R Leroux; John B Wallingford; Oliver E Blacque; Maxence V Nachury; Tania Attie-Bitach; Jean-Baptiste Rivière; Laurence Faivre; Christel Thauvin-Robinet
Journal:  J Med Genet       Date:  2017-03-13       Impact factor: 6.318

  5 in total

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