Pathophysiology Nodes

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5 shared nodes are defined in this module.

Cell Types

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neuron CL:0000540 Cell Ontology (CL) Relation: this mechanism module involves this cell type This mechanism module involves neuron (CL:0000540). CL:0000540 is a cell type from the Cell Ontology.

Biological Processes

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mismatch repair GO:0006298 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves dysregulated mismatch repair (GO:0006298). GO:0006298 is a biological process from the Gene Ontology. DYSREGULATED
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Notes

This is a mechanism module, not a specific disease. Disorder entries reference individual nodes via conforms_to (for example, "mismatch_repair_driven_repeat_instability#Somatic Repeat Expansion in Post-Mitotic Cells", the key conformance target — the disorder-agnostic, rate-limiting central_effector node). The module is deliberately upstream of and complementary to the downstream toxicity modules: a CAG/polyQ disorder conforms to BOTH this module (the instability engine) and polyglutamine_expansion_proteotoxicity (the protein-level consequence), with the seam at the "Cell-Type-Selective Toxicity Threshold Crossing" effector node handing off to the toxicity module's trigger. The same compositional logic extends to CTG/DMPK (repeat-RNA toxicity) and CGG/FMR1 or GAA/FXN (gene silencing) disorders, which substitute their repeat and their downstream route while reusing this instability chain. This is NOT a duplicate of genome_instability_mutation (the cancer mutator / chromosomal-instability hallmark): there the loss of MMR causes a general mutator phenotype; here the MMR machinery is intact and paradoxically DRIVES a specific pathogenic tandem-repeat expansion — a gain of a toxic repair outcome rather than a loss of repair. Evidence architecture: the molecular MMR-driven expansion mechanism (trigger and amplifier nodes) is most cleanly demonstrated in knock-in mouse models (MODEL_ORGANISM evidence across Fragile X CGG and Huntington CAG models), while the disease-level somatic-expansion, selective-vulnerability, and onset/ progression consequences are supported by human clinical/GWAS evidence (HUMAN_CLINICAL). The pan-repeat scope is grounded in ≥2 repeat classes here (CAG: HD and the SCAs; CGG: Fragile X) plus the explicit generalization that the mechanism "is likely applicable to other trinucleotide repeat diseases". GAA/FXN (Friedreich ataxia) remains a named extensible conformer to be wired once its own MMR-instability evidence is curated. STRchive locus tags somatic_instability, anticipation, and length_affects_onset name the phenomena this module models mechanistically. Worked conformers span three repeat classes: Huntington disease (striatal medium spiny neurons, CAG) and Machado-Joseph disease / SCA3 (cerebellar and brainstem neurons, CAG); myotonic dystrophy type 1 (skeletal muscle and other tissues, CTG/DMPK, feeding CUG-RNA toxicity); and Fragile X syndrome (CGG/FMR1, feeding full-mutation methylation silencing). The CTG and CGG conformers demonstrate the module's intended composition — the shared instability engine feeding a different downstream toxicity route per repeat class (RNA gain-of-toxicity for CTG; transcriptional silencing for CGG).

Used By Disorder Entries

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Pathograph

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Pathograph: causal mechanism network for Mismatch-Repair-Driven Repeat Instability Module Interactive directed graph showing how this shared module's pathophysiology nodes connect.

Pathophysiology

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Expandable Tandem Repeat Above the Instability Threshold
trigger
The shared substrate is a germline tandem repeat expanded (or in the premutation range) above a length/instability threshold, at which the repeat forms slipped-strand and hairpin secondary structures that become substrates for aberrant repair. This applies across the repeat classes of the STR expansion diseases — CAG (Huntington disease, the polyglutamine SCAs, DRPLA, SBMA), CGG (FMR1), CTG (DMPK), and GAA (FXN) — with the specific expandable locus substituted by the conforming disorder.
Aberrant Mismatch-Repair Processing of the Repeat
amplifier
The MutSβ complex (MSH2-MSH3) recognizes the slipped/hairpin repeat structure and, instead of faithfully repairing it, licenses incremental expansion; the MutLγ complex (MLH1-MLH3) acts downstream of MutSβ to effect the expansion, while FAN1 (an interstrand-cross-link-repair nuclease) and EXO1 oppose it and are protective. This aberrant, gain-of-toxic-outcome use of a normally protective repair pathway is the disorder-agnostic molecular driver of the expansion; human genetic-modifier studies converge on these same MMR factors.
mismatch repair GO:0006298 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves dysregulated mismatch repair (GO:0006298). GO:0006298 is a biological process from the Gene Ontology. DYSREGULATED
Somatic Repeat Expansion in Post-Mitotic Cells
central effector
Because the aberrant MMR-driven expansion continues throughout life and is not limited to replication, the repeat keeps expanding in specific post-mitotic somatic cells — most consequentially neurons — generating a mosaic of progressively larger alleles far exceeding the inherited germline length. This ongoing, cell-type-biased somatic expansion (not the static germline allele) is the rate-limiting driver of pathology and is the key conformance target: conforming disorder entries substitute their own expanding repeat and vulnerable cell population (striatal medium spiny neurons in Huntington disease; cerebellar and brainstem neurons in SCA3).
neuron CL:0000540 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves neuron (CL:0000540). CL:0000540 is a cell type from the Cell Ontology.
mismatch repair GO:0006298 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves dysregulated mismatch repair (GO:0006298). GO:0006298 is a biological process from the Gene Ontology. DYSREGULATED
Cell-Type-Selective Toxicity Threshold Crossing
effector
When cumulative somatic expansion in a vulnerable cell pushes the repeat past that cell's toxicity threshold, the cell tips into the disorder-specific downstream consequence — translated polyglutamine proteotoxicity for CAG, repeat-RNA gain-of-toxicity for CTG, or transcriptional silencing for CGG and GAA. Because the expansion rate is cell-type-specific, this threshold crossing is the event that explains the selective regional vulnerability of a ubiquitously expressed gene and is the seam handing off to the disorder's toxicity module.
neuron CL:0000540 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves neuron (CL:0000540). CL:0000540 is a cell type from the Cell Ontology.
Repeat-Length-Dependent Onset, Progression, and Anticipation
consequence
The organism-scale readout of the molecular engine: somatic-expansion burden sets the age of onset — earlier than the inherited germline allele alone predicts — and the rate of progression, while germline instability with parent-of-origin bias produces intergenerational anticipation. These are the defining clinical hallmarks of the repeat expansion diseases (mirrored by the STRchive locus tags somatic_instability, anticipation, and length_affects_onset), here derived mechanistically from MMR-driven instability rather than merely correlated with germline repeat length.