Evidence map›Paper›PMID 31603145›Full record

ArticleMedical science monitor : international medical journal of experimental and clinical research2019

Identification of Five Novel Mutations Causing Rare Lysosomal Storage Diseases.

Chenxi Yang, Jianyan Pan, Siyuan Linpeng, Zhuo Li, Hu Tan, Lingqian Wu

Open access · hybridAbstract readCase Reports
In one paragraph

Article in Medical science monitor : international medical journal of experimental and clinical research, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.4field-weighted citation impact, top 35% 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

3 citing papers in PubMed, 7 citations in OpenAlex.

  1. Epidemiology of Mucopolysaccharidoses Update.Diagnostics (Basel, Switzerland) · 2021
    Review
  2. Review
  3. Review
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

6 authors at 1 institution in 1 country.

Chenxi YangCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Jianyan PanCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Siyuan LinpengCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Zhuo LiCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Hu TanCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Lingqian WuCenter for Medical Genetics, School of Life Sciences, Central South University, Changsha, Hunan, China (mainland).
Central South University · CN

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND Lysosomal storage diseases (LSDs), a group of rare inherited metabolic disorders, result from specific lysosomal proteins deficiencies in the degradation of biomacromolecule, including over 70 different diseases, most of which are autosomal recessive. LSDs are multisystem disorders, and the clinical manifestations are usually broad and severe, involving the skeletal system, central nervous system (CNS), cardiovascular system, etc. Besides, patients with some subtypes of LSD have distinctive facial features. MATERIAL AND METHODS We performed next generation sequencing on 4 suspected mucopolysaccharidosis (MPS) cases to determine the genetic causes of the disease. By in vitro molecular cell assay, such as real-time polymerase chain reaction (RT-PCR) and western blot, we tested the pathogenicity of candidate variants. RESULTS We detected 5 novel mutations in 4 patients. The mutations were: c.211_214del and c.1270C>T in GUSB; c.1284+1C>A and c.2404C>T in GNPTAB; and c.717C>A in FUCA1). We identified a rare mucopolysaccharidosis VII patient, a rare fucosidosis patient, and 2 rare mucolipidosis II patients, one of which was an atypical patient. We also present a new pathogenic conjecture about a small deletion in GUSB. CONCLUSIONS Our study described rare diseases in Chinese patients and our results enrich the phenotype spectrum of related diseases, as well as mutation spectrum of related genes, which might be significant for clinical disease diagnosis and prenatal diagnosis.

Indexed as

Genetic Predisposition to DiseaseBase SequenceChildChild, PreschoolFatal OutcomeFemaleHeterozygoteHomozygoteHumansInfantLysosomal Storage DiseasesMaleMutationPhenotypeRare DiseasesTransferases (Other Substituted Phosphate Groups)GNPTAB protein, humanTransferases (Other Substituted Phosphate Groups)

Identifiers

PMID31603145
PMCPMC6800466
OpenAlexW2979592970

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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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.