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Pathophysiology Nodes

5
5 shared nodes are defined in this module.
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Cell Types

0
No cell types are annotated for this module.
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Biological Processes

2
intraciliary transport GO:0042073 ABNORMAL protein localization to cilium GO:0061512 ABNORMAL
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Notes

This is a mechanism module, not a specific disease. Disorder entries reference individual nodes via conforms_to (e.g., "bbsome_trafficking#BBSome-Dependent Ciliary Cargo Trafficking Failure"). Scope discipline: this module covers the BBSome machine plus its dedicated operators (ARL6, LZTFL1, and the BBSome-recycling GTPase IFT27). General IFT-B particle subunits (IFT74, IFT172), transition-zone proteins (CEP290, MKS1, SDCCAG8, NPHP1) and basal-body/other ciliary proteins are shared infrastructure and are modeled in the ciliopathy_dysfunction module, even though variants in those genes also cause the Bardet-Biedl syndrome phenotype. A disease may conform to both modules: its BBSome-core nodes conform here while its transition-zone node conforms to ciliopathy_dysfunction.
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Used By Disorder Entries

3
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Pathograph

Use the checkboxes to hide or show graph categories. Hover nodes for evidence-backed metadata.
Pathograph: causal mechanism network for BBSome Trafficking Module Interactive directed graph showing how this shared module's pathophysiology nodes connect.
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Pathophysiology

5
BBSome Subunit Deficiency
trigger
The initiating lesion in this arm is a biallelic loss-of-function variant in a core BBSome subunit gene (BBS1, BBS2, BBS4, BBS5, BBS7, TTC8/BBS8, BBS9, or BBIP1/BBS18). Because the assembled complex is obligate and highly interconnected, loss of any one subunit destabilizes the whole BBSome. Conforming entries substitute the disorder-specific subunit gene.
BBSome GO:0034464
BBS Chaperonin Assembly Defect
trigger
The initiating lesion in this arm is a biallelic variant in one of the chaperonin-like BBS genes (MKKS/BBS6, BBS10, BBS12). These three proteins form a CCT/TRiC-associated BBS-chaperonin complex required to fold and assemble the BBSome from its subunits; their loss prevents the complex from being built even when subunit genes are intact. Conforming entries substitute the disorder-specific chaperonin gene.
Defective BBSome Assembly
Convergence node: loss of an obligate subunit (subunit arm) or of the chaperonin assembly machinery (chaperonin arm) prevents formation of a stable, functional BBSome. The native complex is highly interconnected and obligate, so partial complexes are non-functional.
BBSome GO:0034464 primary cilium GO:0005929
BBSome Membrane Recruitment and Retrograde IFT Coupling
trigger
Independent trigger arm acting on an intact BBSome. The ARF-like GTPase ARL6/BBS3 recruits the autoinhibited BBSome to the ciliary membrane, where the regulator LZTFL1/BBS17 tunes its trafficking and the BBSome couples to retrograde intraflagellar transport (IFT) trains for ciliary exit of cargo. The general IFT-B machinery is shared infrastructure (modeled in ciliopathy_dysfunction); here it is the train the BBSome boards. Loss of ARL6, LZTFL1, or the BBSome-recycling GTPase IFT27 blocks BBSome-dependent cargo movement even when the BBSome is fully assembled.
intraciliary transport GO:0042073 ABNORMAL
primary cilium GO:0005929
BBSome-Dependent Ciliary Cargo Trafficking Failure
convergent phenotype
Shared output node and the conformance anchor for the BBSome-opathies. Whether the BBSome fails to assemble or fails to be recruited and coupled to IFT, the common consequence is loss of BBSome-dependent trafficking of signaling receptors (e.g. GPCRs) and other cargo into and out of the ciliary compartment. Downstream, the disorder-specific organ branches are determined by which cilia-dependent cell types depend most on this cargo flux.
protein localization to cilium GO:0061512 ABNORMAL intraciliary transport GO:0042073 ABNORMAL
primary cilium GO:0005929