Literature DB >> 29453517

Probable Diagnosis of a Patient with Niemann-Pick Disease Type C: Managing Pitfalls of Exome Sequencing.

William A Zeiger1,2, Nasheed I Jamal3,4, Maren T Scheuner5,6, Patricia Pittman4, Kimiyo M Raymond7, Massimo Morra8, Shri K Mishra3,4,9,10.   

Abstract

Here, we present a case of a 31-year-old man with progressive cognitive decline, ataxia, and dystonia. Extensive laboratory, radiographic, and targeted genetic studies over the course of several years failed to yield a diagnosis. Initial whole exome sequencing through a commercial laboratory identified several variants of uncertain significance; however, follow-up clinical examination and testing ruled each of these out. Eventually, repeat whole exome sequencing identified a known pathogenic intronic variant in the NPC1 gene (NM_000271.4, c.1554-1009G>A) and an additional heterozygous exonic variant of uncertain significance in the NPC1 gene (NM_000271.4, c.2524T>C). Follow-up biochemical testing was consistent with a diagnosis of probable Niemann-Pick disease Type C (NP-C). This case illustrates the potential of whole exome sequencing for diagnosing rare complex neurologic diseases. It also identifies several potential common pitfalls that must be navigated by clinicians when interpreting commercial whole exome sequencing results.

Entities:  

Keywords:  Ataxia; Dementia; Dystonia; Metabolic disease; Neurodegeneration; Niemann–Pick type C; Whole exome sequencing

Year:  2018        PMID: 29453517      PMCID: PMC6122056          DOI: 10.1007/8904_2018_90

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  10 in total

1.  Laboratory diagnosis of Niemann-Pick disease type C: the filipin staining test.

Authors:  Marie T Vanier; Philippe Latour
Journal:  Methods Cell Biol       Date:  2015-01-14       Impact factor: 1.441

2.  Exome sequencing in the clinical diagnosis of sporadic or familial cerebellar ataxia.

Authors:  Brent L Fogel; Hane Lee; Joshua L Deignan; Samuel P Strom; Sibel Kantarci; Xizhe Wang; Fabiola Quintero-Rivera; Eric Vilain; Wayne W Grody; Susan Perlman; Daniel H Geschwind; Stanley F Nelson
Journal:  JAMA Neurol       Date:  2014-10       Impact factor: 18.302

3.  Rare Loss-of-Function Variants in NPC1 Predispose to Human Obesity.

Authors:  Ruixin Liu; Yaoyu Zou; Jie Hong; Min Cao; Bin Cui; Huiwen Zhang; Maopei Chen; Juan Shi; Tinglu Ning; Shaoqian Zhao; Wen Liu; Hui Xiong; Cuijie Wei; Zhengqing Qiu; Weiqiong Gu; Yifei Zhang; Wanyu Li; Lin Miao; Yingkai Sun; Minglan Yang; Rui Wang; Qinyun Ma; Min Xu; Yu Xu; Tiange Wang; Kei-Hang Katie Chan; Xianbo Zuo; Haoyan Chen; Lu Qi; Shenghan Lai; Shumin Duan; Baoliang Song; Yufang Bi; Simin Liu; Weiqing Wang; Guang Ning; Jiqiu Wang
Journal:  Diabetes       Date:  2017-01-27       Impact factor: 9.461

4.  Development of a suspicion index to aid diagnosis of Niemann-Pick disease type C.

Authors:  F A Wijburg; F Sedel; M Pineda; C J Hendriksz; M Fahey; M Walterfang; M C Patterson; J E Wraith; S A Kolb
Journal:  Neurology       Date:  2012-04-18       Impact factor: 9.910

5.  Antisense oligonucleotide treatment for a pseudoexon-generating mutation in the NPC1 gene causing Niemann-Pick type C disease.

Authors:  Laura Rodríguez-Pascau; Maria Josep Coll; Lluïsa Vilageliu; Daniel Grinberg
Journal:  Hum Mutat       Date:  2009-11       Impact factor: 4.878

Review 6.  Diagnostic tests for Niemann-Pick disease type C (NP-C): A critical review.

Authors:  Marie T Vanier; Paul Gissen; Peter Bauer; Maria J Coll; Alberto Burlina; Christian J Hendriksz; Philippe Latour; Cyril Goizet; Richard W D Welford; Thorsten Marquardt; Stefan A Kolb
Journal:  Mol Genet Metab       Date:  2016-06-07       Impact factor: 4.797

7.  The National Niemann-Pick Type C1 Disease Database: correlation of lipid profiles, mutations, and biochemical phenotypes.

Authors:  William S Garver; David Jelinek; F John Meaney; James Flynn; Kathleen M Pettit; Glen Shepherd; Randall A Heidenreich; Cate M Walsh Vockley; Graciela Castro; Gordon A Francis
Journal:  J Lipid Res       Date:  2009-09-09       Impact factor: 5.922

8.  Early co-occurrence of a neurologic-psychiatric disease pattern in Niemann-Pick type C disease: a retrospective Swiss cohort study.

Authors:  Lucia Abela; Barbara Plecko; Antonella Palla; Patricie Burda; Jean-Marc Nuoffer; Diana Ballhausen; Marianne Rohrbach
Journal:  Orphanet J Rare Dis       Date:  2014-11-26       Impact factor: 4.123

Review 9.  Recommendations for the detection and diagnosis of Niemann-Pick disease type C: An update.

Authors:  Marc C Patterson; Peter Clayton; Paul Gissen; Mathieu Anheim; Peter Bauer; Olivier Bonnot; Andrea Dardis; Carlo Dionisi-Vici; Hans-Hermann Klünemann; Philippe Latour; Charles M Lourenço; Daniel S Ory; Alasdair Parker; Miguel Pocoví; Michael Strupp; Marie T Vanier; Mark Walterfang; Thorsten Marquardt
Journal:  Neurol Clin Pract       Date:  2017-12

Review 10.  Niemann-Pick disease type C symptomatology: an expert-based clinical description.

Authors:  Eugen Mengel; Hans-Hermann Klünemann; Charles M Lourenço; Christian J Hendriksz; Frédéric Sedel; Mark Walterfang; Stefan A Kolb
Journal:  Orphanet J Rare Dis       Date:  2013-10-17       Impact factor: 4.123

  10 in total

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