Evidence map›Paper›PMID 25557462›Full record

ArticleNeuromuscular disorders : NMD2015

Delayed diagnosis of congenital myasthenia due to associated mitochondrial enzyme defect.

Yiran Guo, Minal J Menezes, Manoj P Menezes, Jinlong Liang, Dong Li, Lisa G Riley, Nigel F Clarke, P Ian Andrews, Lifeng Tian, Richard Webster and 9 more

Abstract read
In one paragraph

Article in Neuromuscular disorders : NMD, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed, 2 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

12 citing papers in PubMed, 2 syntheses or guidelines pooled it.

  1. Pooled it
  2. Congenital myasthenic syndromes.Orphanet journal of rare diseases · 2019
    Pooled it
  3. Review
  4. Article
  5. Expression assay of theClinical case reports · 2023
    Article
  6. Review
  7. Article
  8. Review
  9. Expanding the Allelic Heterogeneity ofNeurology. Genetics · 2023
    Article
  10. Article
  11. Article
  12. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

19 authors.

Yiran GuoThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Minal J MenezesGenetic Metabolic Disorders Research Unit, Western Sydney Genetics Program, The Children's Hospital at Westmead, Sydney, NSW, Australia; Discipline of Paediatrics & Child Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia.
Manoj P MenezesDiscipline of Paediatrics & Child Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia; Institute for Neuroscience and Muscle Research, The Children's Hospital at Westmead, Sydney, NSW, Australia.
Jinlong LiangBGI-Shenzhen, Shenzhen 518083, China.
Dong LiThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Lisa G RileyGenetic Metabolic Disorders Research Unit, Western Sydney Genetics Program, The Children's Hospital at Westmead, Sydney, NSW, Australia.
Nigel F ClarkeDiscipline of Paediatrics & Child Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia; Institute for Neuroscience and Muscle Research, The Children's Hospital at Westmead, Sydney, NSW, Australia.
P Ian AndrewsDepartment of Neurology, Sydney Children's Hospital, Randwick, NSW, Australia.
Lifeng TianThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Richard WebsterInstitute for Neuroscience and Muscle Research, The Children's Hospital at Westmead, Sydney, NSW, Australia.
Fengxiang WangThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Xuanzhu LiuBGI-Shenzhen, Shenzhen 518083, China.
Yulan ShenBGI-Shenzhen, Shenzhen 518083, China.
David R ThorburnMurdoch Childrens Research Institute and Victorian Clinical Genetics Services, Royal Children's Hospital, Melbourne, VIC, Australia; Department of Paediatrics, University of Melbourne, Melbourne, VIC, Australia.
Brendan J KeatingThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States; Department of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United States; Department of Human Genetics Graduate School of Public Health, University of Pittsburgh, Pittsburgh, PA, United States.
Andrew EngelDepartment of Neurology, Mayo Clinic, Rochester, MN, United States.
Hakon HakonarsonThe Center for Applied Genomics, The Children's Hospital of Philadelphia, Philadelphia, PA, United States; Department of Pediatrics, The Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, United States; Department of Human Genetics Graduate School of Public Health, University of Pittsburgh, Pittsburgh, PA, United States.
John ChristodoulouGenetic Metabolic Disorders Research Unit, Western Sydney Genetics Program, The Children's Hospital at Westmead, Sydney, NSW, Australia; Discipline of Paediatrics & Child Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia; Discipline of Genetic Medicine, Sydney Medical School, University of Sydney, Sydney, NSW, Australia. Electronic address: john.christodoulou@health.nsw.gov.au.
Xun XuBGI-Shenzhen, Shenzhen 518083, China; The Guangdong Enterprise Key Laboratory of Human Disease Genomics, BGI-Shenzhen, Shenzhen, China.

Funding

ELECTRON MICROSCOPY OF MYOPATHIESR01NS006277 · NINDS · MAYO CLINIC ROCHESTER · PI ENGEL, ANDREW GEORGE · 1985 to 2015
$7.8M
The Intellectual and Developmental Disabilities Research Center at CHOP/PennU54HD086984 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI PUTT, MARY E. · 2016 to 2020
$6.3M
Congenital Myasthenic SyndromesR56NS006277 · NINDS · MAYO CLINIC ROCHESTER · PI ENGEL, ANDREW GEORGE, SELCEN, DUYGU · 2017 to 2017
$539k
NICHD NIH HHS U54 HD086984NINDS NIH HHS NS6277NINDS NIH HHS R01 NS006277NINDS NIH HHS R56 NS006277
6 · The paper itself

Abstract

Clinical phenotypes of congenital myasthenic syndromes and primary mitochondrial disorders share significant overlap in their clinical presentations, leading to challenges in making the correct diagnosis. Next generation sequencing is transforming molecular diagnosis of inherited neuromuscular disorders by identifying novel disease genes and by identifying previously known genes in undiagnosed patients. This is evident in two patients who were initially suspected to have a mitochondrial myopathy, but in whom a clear diagnosis of congenital myasthenic syndromes was made through whole exome sequencing. In patient 1, whole exome sequencing revealed compound heterozygous mutations c.1228C > T (p.Arg410Trp) and c.679C > T (p.Arg227*) in collagen-like tail subunit (single strand of homotrimer) of asymmetric acetylcholinesterase (COLQ). In patient 2, in whom a deletion of exon 52 in Dystrophin gene was previously detected by multiplex ligation-dependent probe amplification, Sanger sequencing revealed an additional homozygous mutation c.1511_1513delCTT (p.Pro504Argfs*183) in docking protein7 (DOK7). These case reports highlight the need for careful diagnosis of clinically heterogeneous syndromes like congenital myasthenic syndromes, which are treatable, and for which delayed diagnosis is likely to have implications for patient health. The report also demonstrates that whole exome sequencing is an effective diagnostic tool in providing molecular diagnosis in patients with complex phenotypes.

Indexed as

Delayed DiagnosisAcetylcholinesteraseChild, PreschoolCollagenDystrophinExomeHumansInfantMaleMitochondriaMitochondrial MyopathiesMuscle ProteinsMutationMyasthenic Syndromes, CongenitalPedigreeSequence Analysis, DNAAcetylcholinesteraseCollagenCOLQ protein, humanDOK7 protein, humanDystrophinMuscle ProteinsCOLQCongenital myastheniaDOK7Duchenne muscular dystrophyMitochondrial respiratory chainMutationWhole exome sequencing

Identifiers

PMID25557462
PMCPMC5642298

What OpenQuestion holds

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LicenceTDM
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.