Literature DB >> 21626165

New ophthalmic features in a family with triple A syndrome.

Marilita M Moschos1, Ioannis Margetis, Katrin Koehler, Zisis Gatzioufas, Angela Huebner.   

Abstract

We report three subjects of a Greek family affected by triple A syndrome (AAAS). All patients underwent complete ophthalmic examination, full-field electroretinogram (ERG), visual evoked responses (VER), optical coherence tomography (OCT) and molecular analysis of the AAA gene. All patients had alacrima. In two of them, the proband and her brother, bilateral optic atrophy was assessed and the VER were pathological. In contrast, the ERG was normal. OCT showed a decrease of the retinal nerve fiber layer. The third case had only alacrima and the optic nerves were normal. The molecular genetic study of the AAAS gene revealed a homozygous missense mutation p.Ala167Val. To our knowledge this is the first time a family with AAAS has been investigated using OCT, VER and ERG. Our findings illustrate that the retina is not involved. There is also an interfamilial variability concerning the involvement of the optic nerves.

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Year:  2011        PMID: 21626165     DOI: 10.1007/s10792-011-9450-z

Source DB:  PubMed          Journal:  Int Ophthalmol        ISSN: 0165-5701            Impact factor:   2.031


  7 in total

1.  Ophthalmic manifestations of Allgrove syndrome: report of a case.

Authors:  E Tsilou; C A Stratakis; B I Rubin; B N Hay; N Patronas; M I Kaiser-Kupfer
Journal:  Clin Dysmorphol       Date:  2001-07       Impact factor: 0.816

2.  Triple A syndrome with ophthalmic manifestations in two siblings.

Authors:  Kalpana Babu; Krishna R Murthy; Narendra Babu; S Ramesh
Journal:  Indian J Ophthalmol       Date:  2007 Jul-Aug       Impact factor: 1.848

Review 3.  Late-onset triple A syndrome: a risk of overlooked or delayed diagnosis and management.

Authors:  Andrea Salmaggi; Lucia Zirilli; Chiara Pantaleoni; Gabriella De Joanna; Francesca Del Sorbo; Katrin Koehler; Manuela Krumbholz; Angela Huebner; Vincenzo Rochira
Journal:  Horm Res       Date:  2008-10-27

Review 4.  ACTH resistance syndromes.

Authors:  A Huebner; L L Elias; A J Clark
Journal:  J Pediatr Endocrinol Metab       Date:  1999-04       Impact factor: 1.634

5.  Familial achalasia associated with adrenocortical insufficiency, alacrima, and neurological abnormalities.

Authors:  E Ehrich; G Aranoff; W G Johnson
Journal:  Am J Med Genet       Date:  1987-03

6.  Familial glucocorticoid deficiency with achalasia of the cardia and deficient tear production.

Authors:  J Allgrove; G S Clayden; D B Grant; J C Macaulay
Journal:  Lancet       Date:  1978-06-17       Impact factor: 79.321

7.  Triple-A syndrome with prominent ophthalmic features and a novel mutation in the AAAS gene: a case report.

Authors:  Brian P Brooks; Robert Kleta; Rafael C Caruso; Caroline Stuart; Jonathan Ludlow; Constantine A Stratakis
Journal:  BMC Ophthalmol       Date:  2004-06-24       Impact factor: 2.209

  7 in total
  2 in total

1.  Phenotype-genotype spectrum of AAA syndrome from Western India and systematic review of literature.

Authors:  Hiren Patt; Katrin Koehler; Sailesh Lodha; Swati Jadhav; Chaitanya Yerawar; Angela Huebner; Kunal Thakkar; Sneha Arya; Sandhya Nair; Manjunath Goroshi; Hosahithlu Ganesh; Vijaya Sarathi; Anurag Lila; Tushar Bandgar; Nalini Shah
Journal:  Endocr Connect       Date:  2017-11       Impact factor: 3.335

2.  The Retina in Patients With Triple A Syndrome: A Window Into Neurodegeneration?

Authors:  Fiorenza Ulgiati; Sophie Lhoir; Irina Balikova; Sylvie Tenoutasse; Emese Boros; Catheline Vilain; Claudine Heinrichs; Cécile Brachet
Journal:  Front Endocrinol (Lausanne)       Date:  2021-11-12       Impact factor: 5.555

  2 in total

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