Evidence map›Paper›PMID 38053937›Full record

ArticleMolecular genetics and metabolism reports2023

The diagnostic journey for patients with late-onset GM2 Gangliosidoses.

Mariah C Lopshire, Cynthia Tifft, John Burns, Rebecca Gould, Riliang Zheng, Isabela Batsu

Open access · goldAbstract read
In one paragraph

Article in Molecular genetics and metabolism reports, 2023. 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
1.2field-weighted citation impact, top 20% 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, 7 citations in OpenAlex.

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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

6 authors at 2 institutions in 1 country.

Mariah C LopshireSanofi, 450 Water Street, Cambridge, MA, USA.
Cynthia TifftNational Human Genome Research Institute, National Institutes of Health, 1010 Center Drive, Bethesda, MD, USA.
John BurnsFulcrum Research Group, 200 West Street, Waltham, MA, USA.
Rebecca GouldFulcrum Research Group, 200 West Street, Waltham, MA, USA.
Riliang ZhengSanofi, 450 Water Street, Cambridge, MA, USA.
Isabela BatsuSanofi, Corporate Drive, Bridgewater, NJ, USA.
Sanofi (United States) · USNational Human Genome Research Institute · US

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Late-onset forms of GM2 gangliosidosis-mainly, Tay-Sachs disease and Sandhoff disease-are under-recognized in clinical practice. In these rare lysosomal storage disorders, deficiency of β-hexosaminidase A results in excessive accumulation of GM2 ganglioside primarily within neurons, leading to cell death and progressive neurodegenerative symptoms, including ataxia, dysarthria, muscle weakness, tremors, atrophy, and psychosis. Presentation is variable and often mimics more common neurodegenerative disorders. We conducted semi-structured interviews on GM2 gangliosidoses diagnosis and treatment with five experts, 30 neurologists, and 28 patients and caregivers. Symptom onset occurred during adolescence/early adulthood in 92% of patients (median age: 14 years). Patients first visited a healthcare provider at a median age of 20 years and received a GM2 diagnosis at a median age of 26 years. Nearly all patients reported problems with their legs and balance starting from symptom onset. Problems with memory, attention span, speech and fatigue were reported more after diagnosis. Patients visited an average of eight healthcare providers before receiving a diagnosis; 64% were diagnosed by a neurologist. Four neurologists (13%) in our sample were aware that there are late-onset forms of GM2 gangliosidosis. The path to diagnosis is long for this late-onset form of a classically fatal infantile disease.

Indexed as

DiagnosisLate-onset GM2 gangliosidosesPatient perspectiveSandhoff diseaseTay-Sachs diseaseβ-hexosaminidase A

Identifiers

PMID38053937
PMCPMC10694732
OpenAlexW4387762597

What OpenQuestion holds

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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.