Literature DB >> 22156782

A child with night blindness: preventing serious symptoms of Refsum disease.

Alfried Kohlschütter1, René Santer, Zoltan Lukacs, Christiane Altenburg, Markus J Kemper, Klaus Rüther.   

Abstract

Refsum disease is a genetic progressive neurological disorder caused by neurotoxic phytanic acid, a nutritional component patients are unable to metabolize. Symptoms include retinopathy, polyneuropathy, ataxia, and deafness. They are variable and rarely recognized before adulthood. The authors report the case of a 14-year-old girl diagnosed because of night blindness. They treated her with a phytanic acid-poor diet and extracorporeal lipid apheresis. They used different methods over a 30-month period. Thereafter, the patient was treated with diet only. Membrane filtration and heparin-induced extracorporeal low-density lipoprotein precipitation apheresis were well tolerated. Withdrawal of phytanic acid was studied quantitatively. During a 5-year period, blood phytanic acid levels decreased to a noncritical range. The patient remained free of ophthalmological and neurological progression for a total observation of 12 years. Early diagnosis and effective measures to keep the phytanic acid load low can probably prevent the serious sequelae of Refsum disease. Developing a method for newborn screening is desirable.

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Year:  2011        PMID: 22156782     DOI: 10.1177/0883073811424799

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  5 in total

1.  The Challenges of a Successful Pregnancy in a Patient with Adult Refsum's Disease due to Phytanoyl-CoA Hydroxylase Deficiency.

Authors:  Karolina M Stepien; Anthony S Wierzbicki; Bwee T Poll-The; Hans R Waterham; Christian J Hendriksz
Journal:  JIMD Rep       Date:  2016-08-13

2.  A deep intronic CLRN1 (USH3A) founder mutation generates an aberrant exon and underlies severe Usher syndrome on the Arabian Peninsula.

Authors:  Arif O Khan; Elvir Becirovic; Christian Betz; Christine Neuhaus; Janine Altmüller; Lisa Maria Riedmayr; Susanne Motameny; Gudrun Nürnberg; Peter Nürnberg; Hanno J Bolz
Journal:  Sci Rep       Date:  2017-05-03       Impact factor: 4.379

3.  Next-generation sequencing reveals the mutational landscape of clinically diagnosed Usher syndrome: copy number variations, phenocopies, a predominant target for translational read-through, and PEX26 mutated in Heimler syndrome.

Authors:  Christine Neuhaus; Tobias Eisenberger; Christian Decker; Sandra Nagl; Cornelia Blank; Markus Pfister; Ingo Kennerknecht; Cornelie Müller-Hofstede; Peter Charbel Issa; Raoul Heller; Bodo Beck; Klaus Rüther; Diana Mitter; Klaus Rohrschneider; Ute Steinhauer; Heike M Korbmacher; Dagmar Huhle; Solaf M Elsayed; Hesham M Taha; Shahid M Baig; Heidi Stöhr; Markus Preising; Susanne Markus; Fabian Moeller; Birgit Lorenz; Kerstin Nagel-Wolfrum; Arif O Khan; Hanno J Bolz
Journal:  Mol Genet Genomic Med       Date:  2017-07-06       Impact factor: 2.183

Review 4.  Roles of HIF and 2-Oxoglutarate-Dependent Dioxygenases in Controlling Gene Expression in Hypoxia.

Authors:  Julianty Frost; Mark Frost; Michael Batie; Hao Jiang; Sonia Rocha
Journal:  Cancers (Basel)       Date:  2021-01-19       Impact factor: 6.639

5.  Targeted Therapies for Hereditary Peripheral Neuropathies: Systematic Review and Steps Towards a 'treatabolome'.

Authors:  Matthew J Jennings; Angela Lochmüller; Antonio Atalaia; Rita Horvath
Journal:  J Neuromuscul Dis       Date:  2021
  5 in total

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