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Myotonic Dystrophy type 1 (DM1) is caused by a CTG repeat expansion in the 3' untranslated region of the DMPK gene. This expansion leads to the production of toxic RNA transcripts, which accumulate in the nucleus and interfere with normal RNA processing. DM1 affects a broad range of tissues and systems such as the skeletal muscle, the central nervous system, cardiac, visual, reproductive, and gastrointestinal (GI) system. GI dysfunction is a significant but poorly understood aspect of DM1. Particularly, it is unknown if there are alterations in the intestinal microbiome in DM1. Here, we used a transgenic humanized mouse model (DMSXL) to explore how the gut microbiome may be linked to GI issues in DM1. For this purpose, 68 stool samples from Homozygous, Heterozygous, and Wild-Type (WT) mice were collected. These samples were sequenced by MiSeq and analyzed with DADA2 to generate taxonomic profiles. Our analysis indicated that the overexpression of CTG repeats significantly influences the bacterial structure of the gut microbiome in Homozygous mice samples, especially in terms of the relative abundance of the Patescibacteria and Defferibacterota Phyla. These results provide valuable information about the gut microbiota structure thus improving the understanding of the role of these changes in the pathogenicity as well as GI problems of DM1 patients.
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http://dx.doi.org/10.1016/j.csbj.2025.02.016 | DOI Listing |
Parkinsonism Relat Disord
September 2025
Federal University of São Paulo, Department of Neurology and Neurosurgery, São Paulo, SP, Brazil.
Background: Huntington disease-like 2 (HDL2) is an autosomal dominant disorder caused by an abnormal CAG/CTG repeat in exon 2A of junctophilin-3. This is the most common Huntington's Disease phenocopy and is characterized by psychiatric, cognitive, and movement disorders. This study aimed to describe the clinical phenotype of HDL2 patients in Brazil and compare the findings with those in the literature.
View Article and Find Full Text PDFMol Ther
September 2025
Genomic Medicine Unit, Sanofi; Waltham, MA, 02451. Electronic address:
Myotonic dystrophy type 1 (DM1), characterized by life-threatening muscle weakness, compromised respiration, and often cardiac conduction abnormalities, is the most common form of adult muscular dystrophy it is. DM1 is caused by a CTG repeat expansion in the 3' untranslated region of the dystrophia myotonica protein kinase (DMPK) gene resulting in aggregation of DMPK mRNA into insoluble ribonuclear foci which sequester RNA-binding proteins. Redistribution of essential splicing factors causes mis-splicing of factors responsible for muscle differentiation.
View Article and Find Full Text PDFMol Ther Nucleic Acids
September 2025
Molecular Cardiology Laboratory, IRCCS Policlinico San Donato, San Donato Milanese, Milan 20097, Italy.
Myotonic dystrophy type 1 (DM1) is a multisystemic disorder caused by expanded CTG repeats in the 3'-UTR of the gene that lead to nuclear foci accumulation and splicing defects. Circular RNAs (circRNAs) are emerging regulators of muscular disorders, but their role in DM1 remains largely unknown. By analyzing available RNA-sequencing datasets from DM1 patients, followed by validation in patients and matching control muscle biopsies, we identified seven circRNAs that were significantly increased in DM1 muscles and displayed high circular-to-linear isoform ratios.
View Article and Find Full Text PDFBMC Neurol
September 2025
Department of Neurology, Asahi General Hospital, 1326 I, Asahi, Chiba 289-2511, Japan.
Background: Spinocerebellar ataxia type 8 (SCA8) is a rare neurodegenerative disease that is caused by CAG/CTG repeat expansion in the overlapping ATXN8 and ATXN8OS genes and basically entails slowly progressive cerebellar dysfunction with resultant dysarthria, limb incoordination, and gait instability. Moreover, patients with SCA8 may also exhibit pyramidal and extrapyramidal signs, cognitive decline, and involuntary movements. Although SCA8 is an autosomal dominant inheritance disorder, it sometimes seems to be sporadic because of reduced penetrance.
View Article and Find Full Text PDFAnn Neurol
August 2025
Raymond G. Perelman Center for Cellular and Molecular Therapeutics, The Children's Hospital of Philadelphia, Philadelphia, PA.
Objective: Myotonic dystrophy type 1 (DM1) is a highly variable, multisystemic genetic disorder caused by a CTG repeat expansion in the 3' untranslated region of DMPK. Toxicity is exerted by repeat-containing DMPK transcripts that sequester muscleblind-like (MBNL) proteins and lead to deleterious yet predictable changes in alternative splicing. To contend with high phenotypic and molecular variability that complicate application of viral-based therapies, we develop and test a DM1-responsive genetic element to control viral-based therapeutic output.
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