Pathophysiology Nodes

5
5 shared nodes are defined in this module.

Cell Types

2
corticospinal upper motor neuron CL:0000598 Cell Ontology (CL) Relation: this mechanism module involves this cell type This mechanism module involves corticospinal upper motor neuron (CL:0000598). CL:0000598 is a cell type from the Cell Ontology. spinal motor neuron CL:0000100 Cell Ontology (CL) Relation: this mechanism module involves this cell type This mechanism module involves spinal motor neuron (CL:0000100). CL:0000100 is a cell type from the Cell Ontology.

Biological Processes

6
microtubule severing GO:0051013 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves decreased microtubule severing (GO:0051013). GO:0051013 is a biological process from the Gene Ontology. DECREASED endoplasmic reticulum organization GO:0007029 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves dysregulated endoplasmic reticulum organization (GO:0007029). GO:0007029 is a biological process from the Gene Ontology. DYSREGULATED mitochondrion organization GO:0007005 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves dysregulated mitochondrion organization (GO:0007005). GO:0007005 is a biological process from the Gene Ontology. DYSREGULATED anterograde axonal transport GO:0008089 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves decreased anterograde axonal transport (GO:0008089). GO:0008089 is a biological process from the Gene Ontology. DECREASED axonal transport GO:0098930 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves decreased axonal transport (GO:0098930). GO:0098930 is a biological process from the Gene Ontology. DECREASED neuron projection maintenance GO:1990535 Gene Ontology (GO) Relation: this mechanism module involves this biological process This mechanism module involves decreased neuron projection maintenance (GO:1990535). GO:1990535 is a biological process from the Gene Ontology. DECREASED
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Notes

This is a mechanism module, not a specific disease. Disorder entries reference individual nodes via conforms_to (e.g., "corticospinal_tract_axonopathy#Distal Length-Dependent Degeneration of Long CNS Axons"). THIS IS THE CNS COUNTERPART OF peripheral_axonal_degeneration, NOT A DUPLICATE OF IT. Both modules model length-dependent dying-back axonopathy, and the distinction is the compartment and the glial partner. peripheral_axonal_degeneration models the peripheral nerve - a lower motor or sensory neuron whose axon is myelinated by Schwann cells, producing distal weakness, sensory loss, and areflexia. This module models the central compartment - an upper motor neuron whose axon runs in the corticospinal tract and is myelinated by oligodendrocytes, producing spasticity, hyperreflexia, and extensor plantar responses. The signs are opposite at the bedside for the same underlying cell-biological failure. A disorder with both a central and a peripheral axonopathy (as many complicated HSPs have) conforms to both modules at different nodes; do not collapse the two arms onto one node. Key disorder-specific substitutions for the trigger node follow the functional categories of the SPG proteins: microtubule severing and axonal transport (SPG4/spastin, SPG10/KIF5A, SPG30/KIF1A); endoplasmic reticulum morphology (SPG3A/atlastin-1, SPG31/REEP1, SPG12/reticulon 2); mitochondrial function (SPG7/paraplegin, SPG13/HSPD1); endosomal membrane traffic and vesicle formation (the AP-4 complex disorders SPG47/SPG50/SPG51/SPG52, and SPG48/AP5Z1 of the AP-5 complex); protein folding and the ER stress response (SPG6/NIPA1, SPG8/strumpellin, SPG17/BSCL2); and fatty acid and phospholipid metabolism (SPG28/DDHD1, SPG35/FA2H, SPG54/DDHD2, SPG56/CYP2U1). Two further SPG categories are deliberately NOT modelled here because their primary lesion is elsewhere: myelin formation (SPG2/PLP1, SPG42/GJC2), which belongs to cns_myelin_failure, and corticospinal tract neurodevelopment (SPG1/L1CAM, SPG22/MCT8), which is a failure to build the tract rather than a failure to maintain it. A PLP1 or L1CAM disorder that also shows corticospinal degeneration may still conform at the degeneration node, but not at the trigger. THE MODULE IS ABOUT MAINTENANCE, NOT DEVELOPMENT. The chain presupposes an anatomically formed corticospinal tract that subsequently degenerates. A disorder in which the tract never decussates or never forms does not conform, however severe its spasticity.

Used By Disorder Entries

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Pathograph

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Pathograph: causal mechanism network for Corticospinal Tract Axonopathy Module Interactive directed graph showing how this shared module's pathophysiology nodes connect.

Pathophysiology

5
Long-Axon Maintenance Machinery Defect
trigger
A heritable lesion disables one of the cell-biological systems a very long axon depends on to stay supplied: microtubule severing and organisation, the tubular endoplasmic reticulum network that extends the length of the axon, mitochondrial quality control, endolysosomal and vesicular membrane traffic, or the lipid metabolism that maintains axonal membranes. None of these systems is specific to the corticospinal tract, and none is dispensable in shorter neurons; what makes the lesion selective is the downstream length-dependence, not the gene's expression pattern.
corticospinal upper motor neuron CL:0000598 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves corticospinal upper motor neuron, annotated with pyramidal neuron (CL:0000598). CL:0000598 is a cell type from the Cell Ontology.
microtubule severing GO:0051013 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves decreased microtubule severing (GO:0051013). GO:0051013 is a biological process from the Gene Ontology. DECREASED endoplasmic reticulum organization GO:0007029 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves dysregulated endoplasmic reticulum organization (GO:0007029). GO:0007029 is a biological process from the Gene Ontology. DYSREGULATED mitochondrion organization GO:0007005 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves dysregulated mitochondrion organization (GO:0007005). GO:0007005 is a biological process from the Gene Ontology. DYSREGULATED
Impaired Axonal Transport and Organelle Distribution
amplifier
Cargo delivery along the axon fails. Because microtubules are the track on which axonal transport runs, and because the tubular ER and the mitochondrial population are themselves cargo that must be distributed along the axon, all of the trigger lesions converge here. The measurable consequence is reduced anterograde movement of mitochondria and membrane-bound organelles, and the morphological consequence is focal axonal swellings packed with cytoskeletal proteins, mitochondria, and amyloid precursor protein - seen both in mutant mice and in human HSP post-mortem material. The swellings are themselves amplifying: transport distal to a swelling is far worse than transport through an unaffected segment, so the lesion propagates down the axon.
corticospinal upper motor neuron CL:0000598 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves corticospinal upper motor neuron, annotated with pyramidal neuron (CL:0000598). CL:0000598 is a cell type from the Cell Ontology.
anterograde axonal transport GO:0008089 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves decreased anterograde axonal transport (GO:0008089). GO:0008089 is a biological process from the Gene Ontology. DECREASED axonal transport GO:0098930 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves decreased axonal transport (GO:0098930). GO:0098930 is a biological process from the Gene Ontology. DECREASED
axon GO:0030424 Gene Ontology (GO) Relation: this pathophysiological event involves this cellular component This pathophysiological event involves axon (GO:0030424). GO:0030424 is a cellular component from the Gene Ontology.
Distal Length-Dependent Degeneration of Long CNS Axons
central effector
The rate-limiting, disorder-agnostic convergence point of the module and its key conformance target. The distal ends of the longest central axons degenerate in a dying-back pattern. Post-mortem studies of HSP consistently find degeneration of corticospinal tract axons maximal in the thoracic spinal cord - the furthest point from the motor cortical cell bodies - together with degeneration of fasciculus gracilis fibres maximal in the cervico-medullary region, which is the furthest point from the dorsal root ganglion cell bodies of the ascending sensory arm. The two tracts are affected at opposite ends of the cord for the same reason: distance from the soma. Conforming entries should preserve this length-dependence explicitly rather than describing undifferentiated "corticospinal degeneration".
corticospinal upper motor neuron CL:0000598 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves corticospinal upper motor neuron, annotated with pyramidal neuron (CL:0000598). CL:0000598 is a cell type from the Cell Ontology.
neuron projection maintenance GO:1990535 Gene Ontology (GO) Relation: this pathophysiological event involves this biological process This pathophysiological event involves decreased neuron projection maintenance (GO:1990535). GO:1990535 is a biological process from the Gene Ontology. DECREASED
Loss of Supraspinal Inhibitory Control of the Stretch Reflex
effector
Degeneration of the descending corticospinal axons disrupts the balance of supraspinal inhibitory and excitatory inputs converging on the spinal cord, leaving the stretch reflex disinhibited and producing the velocity-dependent hypertonia that defines spasticity. The release is not the whole story: plastic changes within the spinal cord - notably progressive reduction of post-activation depression with limb immobilization - and secondary soft tissue changes in the paretic limbs both add non-reflex hypertonia on top of the reflex component. This distinction between reflex-mediated and intrinsic hypertonia is why physiotherapy and limb mobilization are curated as treatments in conforming entries alongside antispastic pharmacotherapy.
spinal motor neuron CL:0000100 Cell Ontology (CL) Relation: this pathophysiological event involves this cell type This pathophysiological event involves spinal motor neuron, annotated with motor neuron (CL:0000100). CL:0000100 is a cell type from the Cell Ontology.
Progressive Lower-Limb Spasticity and Weakness
consequence
The clinical endpoint of the module: a spastic paraparesis in which lower extremity weakness and spasticity predominate. Symptoms may begin at any age, and tempo depends on age at onset - gait impairment beginning after childhood usually worsens very slowly over many years, whereas impairment beginning in infancy and early childhood may not worsen significantly. In pure HSP this is the whole phenotype; in complicated HSP it is accompanied by the additional features the disorder-specific SPG protein produces outside the corticospinal tract, which conforming entries curate on their own nodes rather than here.