Evidence map›Paper›PMID 30094186›Full record

ArticleMolecular genetics and metabolism reports2018

Morquio B patient/caregiver survey: First insight into the natural course of a rare GLB1 related condition.

Maria Bleier, Nataliya Yuskiv, Tina Priest, Marioara Angela Moisa Popurs, Sylvia Stockler-Ipsiroglu, BC Children's Hospital, University of British Columbia

Open access · goldAbstract read
In one paragraph

Article in Molecular genetics and metabolism reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 7 papers.

0numbers the graph read from it
0cells of the map it votes in
7citing papers in PubMed
1.5field-weighted citation impact, top 18% 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

7 citing papers in PubMed, 14 citations in OpenAlex.

  1. Article
  2. Morquio B disease: a case report.Frontiers in pediatrics · 2024
    Article
  3. Article
  4. Review
  5. Review
  6. Article
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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

7 authors at 2 institutions in 1 country.

Maria BleierDivision of Biochemical Diseases, BC Children's Hospital Vancouver, Department of Pediatrics, University of British Columbia, Vancouver, BC, Canada.
Nataliya YuskivDivision of Biochemical Diseases, BC Children's Hospital Vancouver, Department of Pediatrics, University of British Columbia, Vancouver, BC, Canada.
Tina PriestThe Priest Family Fund for Morquio B, Vancouver, BC, Canada.
Marioara Angela Moisa PopursDivision of Biochemical Diseases, BC Children's Hospital Vancouver, Department of Pediatrics, University of British Columbia, Vancouver, BC, Canada.
Sylvia Stockler-IpsirogluDivision of Biochemical Diseases, BC Children's Hospital Vancouver, Department of Pediatrics, University of British Columbia, Vancouver, BC, Canada.
BC Children's Hospital
University of British Columbia
University of British Columbia · CABC Children's Hospital · CA

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Morquio B disease (MBD) or Mucopolysaccharidosis type IV B (MPS IV B) is caused by particular GLB1 mutations specifically affecting the affinity of beta-galactosidase to keratan sulphate, resulting in dysostosis multiplex resembling Morquio A (MPS IV A) disease (GALNS deficiency). Additional neuronopathic features of GM1 II/III (juvenile/adult) gangliosidosis have been reported in some patients. Our patient/caregiver online survey was aimed at elucidating the clinical manifestations of this ultra-rare condition. Comparing to previously published data on MPS IV A, the 30 respondents in our MBD group presented with greater growth chart values (weight and height) and with lesser effects of odontoid hypoplasia. The most common concerns are: (1) mobility issues - 84% having difficulty walking; (2) chronic pain - 96%; (3) surgeries - average 3 per person, 80% for hip problems; (4) hip dysplasia, knee/ankle concerns, and scoliosis. Approximately 50% of MBD participants live independently and actively contributing to society. Evidence from our survey results supports the notion that skeletal manifestations in MBD are milder than in the majority of patients with MPS IV A. The data collected will help with the establishment of clinically meaningful outcomes for future therapeutic trials, and with the counseling of newly diagnosed patients about their health expectations.

Indexed as

GM1 gangliosidosisLysosomeNatural historySkeletal dysplasia

Identifiers

PMID30094186
PMCPMC6072644
OpenAlexW2883880312

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
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.