Evidence map›Paper›PMID 33469851›Full record

ArticleJournal of molecular neuroscience : MN2021

Clinico-pathological and Molecular Spectrum of Mitochondrial Polymerase γ Mutations in a Cohort from India.

Sekar Deepha, Periyasamy Govindaraj, Bindu Parayil Sankaran, Shwetha Chiplunkar, Chetan Kashinkunti, Vandana Nunia, Madhu Nagappa, Sanjib Sinha, Tripti Khanna, Kumarasamy Thangaraj and 2 more

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Article in Journal of molecular neuroscience : MN, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 5 papers.

0numbers the graph read from it
0cells of the map it votes in
5citing papers in PubMed
0.8field-weighted citation impact, top 30% of its field
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

5 citing papers in PubMed, 10 citations in OpenAlex.

  1. Trial
  2. Review
  3. Article
  4. Article
  5. Clinico-Genetic Spectrum of POLG1 Mutation Carriers from India.Journal of molecular neuroscience : MN · 2022
    Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

12 authors at 5 institutions in 2 countries.

Sekar DeephaDepartment of Neuropathology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Periyasamy GovindarajDepartment of Neuropathology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Bindu Parayil SankaranNeuromuscular Laboratory, Neurobiology Research Centre (NRC), National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Shwetha ChiplunkarNeuromuscular Laboratory, Neurobiology Research Centre (NRC), National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Chetan KashinkuntiDepartment of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Vandana NuniaCSIR-Centre for Cellular and Molecular Biology, Hyderabad, India.
Madhu NagappaNeuromuscular Laboratory, Neurobiology Research Centre (NRC), National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Sanjib SinhaDepartment of Neurology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Tripti KhannaIndian Council of Medical Research (ICMR), New Delhi, India.
Kumarasamy ThangarajCSIR-Centre for Cellular and Molecular Biology, Hyderabad, India.
Arun B TalyNeuromuscular Laboratory, Neurobiology Research Centre (NRC), National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India.
Narayanappa GayathriDepartment of Neuropathology, National Institute of Mental Health and Neurosciences (NIMHANS), Bangalore, India. gayathri.narayanappa@gmail.com.ORCID http://orcid.org/0000-0003-3903-3363
National Institute of Mental Health and Neurosciences · INCentre for Cellular and Molecular Biology · INChildren's Hospital at Westmead · AUIndian Council of Medical Research · INManipal Academy of Higher Education · IN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Polymerase γ catalytic subunit (POLG), a nuclear gene, encodes the enzyme responsible for mitochondrial DNA (mtDNA) replication. POLG mutations are a major cause of inherited mitochondrial diseases. They present with varied phenotypes, age of onset, and severity. Reports on POLG mutations from India are limited. Hence, this study aimed to describe the clinico-pathological and molecular observations of POLG mutations. A total of 446 patients with clinical diagnosis of mitochondrial disorders were sequenced for all exons and intron-exon boundaries of POLG. Of these, 19 (4.26%) patients (M:F: 10:9) had POLG mutations. The age of onset ranged from 5 to 55 years with an overlapping phenotypic spectrum. Ptosis, peripheral neuropathy, seizures, and ataxia were the common neurological features observed. The most common clinical phenotype was chronic progressive external ophthalmoplegia (CPEO) and CPEO plus (n = 14). Muscle biopsy showed characteristic features of mitochondrial myopathy in fourteen patients (14/19) and respiratory chain enzyme deficiency in eleven patients (11/19). Multiple mtDNA deletions were seen in 47.36% (9/19) patients. Eight pathogenic POLG variations including two novel variations (p.G132R and p.V1106A) were identified. The common pathogenic mutation identified was p.L304R, being present in eight patients (42.1%) predominantly in the younger age group followed by p.W748S in four patients (21%). To the best of our knowledge, this is the first extensive study from India, highlights the clinico-pathological and molecular spectrum of POLG mutations.

Indexed as

MutationPhenotypeAdolescentAdultAtaxiaChildDNA Polymerase gammaFemaleHumansMaleMiddle AgedMitochondrial DiseasesMuscle, SkeletalPeripheral Nervous System DiseasesSeizuresDNA Polymerase gammaPOLG protein, humanCPEOMitochondrial diseasemtDNA deletionsMutationsPOLG

Identifiers

PMID33469851
OpenAlexW3121749206

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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.