sábado, 25 de febrero de 2017

Improving diagnosis of inherited peripheral neuropathies through gene panel analysis | Orphanet Journal of Rare Diseases | Full Text

Improving diagnosis of inherited peripheral neuropathies through gene panel analysis | Orphanet Journal of Rare Diseases | Full Text

Biomed Central

Orphanet Journal of Rare Diseases - IMPACT FACTOR 3.29



Improving diagnosis of inherited peripheral neuropathies through gene panel analysis

  • Petra LaššuthováEmail author,
  • Dana Šafka Brožková,
  • Marcela Krůtová,
  • Jana Neupauerová,
  • Jana Haberlová,
  • Radim Mazanec,
  • Pavel Dřímal and
  • Pavel Seeman
Orphanet Journal of Rare Diseases201611:118
DOI: 10.1186/s13023-016-0500-5
Received: 28 April 2016
Accepted: 9 August 2016
Published: 22 August 2016

Abstract

Background

Inherited peripheral neuropathies (IPN) are the most common inherited neurological condition. It represents a highly heterogeneous group, both clinically and genetically.
Targeted disease specific gene panel massively parallel sequencing (MPS) seems to be a useful tool in diagnosis of disorders with high genetic heterogeneity.

Methods

In our study, we have designed, validated and updated our own custom gene panel of all known genes associated with IPN. One hundred and ninety-eight patients have been tested so far. Only patients in whom mutations in more common causes or relevant genes have already been excluded were enrolled. Five consecutive panel designs were prepared according to recent literature search, the last one covering ninety-three genes. Each patient was tested only once. All data were evaluated with at least two different pipelines.

Results

In summary, causative mutation has been found in fifty-one patients (26 %). The results were inconclusive in thirty-one (16 %) patients. No variants of likely significance to IPN were found in one hundred and sixteen (58 %) patients.

Conclusion

MPS gene panel enables testing of all known IPN causes at once with high coverage and at an affordable cost making it truly a method of choice also in IPN. Gene panel testing results in several interesting results and findings.

Keywords

Inherited peripheral neuropathies Charcot-Marie-Tooth Targeted gene panel testing Mutation Phenotype

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