Literature DB >> 9415683

Spherophakia associated with molybdenum cofactor deficiency.

R Parini1, V Briscioli, U Caruso, C Dorche, R Fortuna, G Minniti, A Selicorni, E Vismara, G Mancini.   

Abstract

Molybdenum cofactor deficiency is an autosomal recessive disorder characterized by lack of activity of the enzymes sulfite oxidase, aldehyde oxidase, and xanthine dehydrogenase or oxidase. The clinical manifestations are indistinguishable from those of isolated sulfite oxidase deficiency: craniofacial alterations, intractable neonatal convulsions, very severe mental retardation, lens dislocation, and death in the first decade of life. Lens dislocation is found in nearly all patients after neonatal age. In the present case it developed late (at the age of 8 years) and was preceded by bilateral spherophakia. We hypothesize that an abnormal relaxation of the zonular fibers is the cause of spherophakia in this disease; this causes lens dislocation eventually, after days, months, or years.

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Year:  1997        PMID: 9415683     DOI: 10.1002/(sici)1096-8628(19971219)73:3<272::aid-ajmg8>3.0.co;2-u

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  3 in total

1.  Ocular characteristics of a 6-year-Old boy with molybdenum cofactor deficiency type B.

Authors:  Wenjia Yan; Li Huang; Limei Sun; Xiaoyan Ding
Journal:  Am J Ophthalmol Case Rep       Date:  2022-05-14

2.  Molybdenum cofactor and isolated sulphite oxidase deficiencies: Clinical and molecular spectrum among Egyptian patients.

Authors:  Maha S Zaki; Laila Selim; Hala T El-Bassyouni; Mahmoud Y Issa; Iman Mahmoud; Samira Ismail; Mariane Girgis; Abdelrahim A Sadek; Joseph G Gleeson; Mohamed S Abdel Hamid
Journal:  Eur J Paediatr Neurol       Date:  2016-05-30       Impact factor: 3.140

3.  Mild phenotype in Molybdenum cofactor deficiency: A new patient and review of the literature.

Authors:  Barbara Scelsa; Serena Gasperini; Andrea Righini; Maria Iascone; Valeria G Brazzoduro; Pierangelo Veggiotti
Journal:  Mol Genet Genomic Med       Date:  2019-03-21       Impact factor: 2.183

  3 in total

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