Literature DB >> 22069215

Detection of a novel FH whole gene deletion in the propositus leading to subsequent prenatal diagnosis in a sibship with fumarase deficiency.

Amelia R Mroch1, Mark Laudenschlager, Jason D Flanagan.   

Abstract

Fumarase deficiency is a rare autosomal recessive metabolic condition. We report on a sibship with molecularly confirmed fumarase deficiency. Prenatal findings included agenesis of the corpus callosum, ventriculomegaly, and ventriculoseptal defect. The postnatal course was significant for metabolic acidosis ultimately leading to death around 3 weeks of age. Postmortem findings were noted including swollen mitochondria with abnormal cristae on electron microscopy within the liver. Molecular testing revealed a novel whole gene deletion in conjunction with a point mutation. While the point mutation has been previously reported, the detection of a whole gene deletion has not been described to date in an individual with fumarase deficiency.
Copyright © 2011 Wiley Periodicals, Inc.

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Year:  2011        PMID: 22069215     DOI: 10.1002/ajmg.a.34344

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

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Review 2.  Clinical, genetic and imaging findings identify new causes for corpus callosum development syndromes.

Authors:  Timothy J Edwards; Elliott H Sherr; A James Barkovich; Linda J Richards
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Review 4.  Advances in hereditary leiomyomatosis and renal cell carcinoma (HLRCC) research.

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6.  Fumarase Deficiency: A Safe and Potentially Disease Modifying Effect of High Fat/Low Carbohydrate Diet.

Authors:  B Ryder; F Moore; A Mitchell; S Thompson; J Christodoulou; S Balasubramaniam
Journal:  JIMD Rep       Date:  2017-10-21

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8.  Uterine smooth muscle tumors with features suggesting fumarate hydratase aberration: detailed morphologic analysis and correlation with S-(2-succino)-cysteine immunohistochemistry.

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Authors:  Samya Chakravorty; Rachel Logan; Molly J Elson; Rebecca R Luke; Sumit Verma
Journal:  Sci Rep       Date:  2020-09-30       Impact factor: 4.379

  9 in total

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