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Article

Natural history study and statistical modeling of disease progression in a preclinical model of myotubular myopathy.

Disease models & mechanisms - 1 Jul 2022

Buono Suzie, Monseur Arnaud, Menuet Alexia, Robé Anne, Koch Catherine, Laporte Jocelyn, Thielemans Leen, Depla Marion, Cowling Belinda S

Abstract excerpt

Generating reliable preclinical data in animal models of disease is essential in therapy development. Here, we performed statistical analysis and joint longitudinal-survival modeling of the progressive phenotype observed in Mtm1-/y mice, a reliable model for myotubular myopathy. Analysis of historical data was used to generate a model for phenotype progression, which was then confirmed with phenotypic data from a...

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Methodology Data sources Editorial policy Privacy Delete account & data About Contact Helena Bioinformatics

Share to Muscle, Skeletal

Share to Myopathies, Structural, Congenital

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Share to Protein Tyrosine Phosphatases, Non-Receptor

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Source

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Identifiers

PMID: 35642830
DOI: 10.1242/dmm.049284
PMCID: PMC9346515

Provenance

PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 7 Sept 2026. Manifest publication-graph-cohort-2026-09-08.1.

Source receipt: pubmed, retrieved 7 Sept 2026.

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