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R177C

Category 3/4 — Most DruggableConflictingCytoplasmic · predictedEditorial
ArginineCysteine at position 177 · N-terminal cytoplasmic domain (87-313) · WFS1 (Wolframin)

Arginine → Cysteine at position 177 in N-terminal cytoplasmic domain. ClinVar Conflicting including Wolfram syndrome 1. AlphaMissense 0.902, ΔΔG -0.88. Charge loss + thiol introduction.

Interactive 3D Structure

Wild-type reference
Wild-type R177 — ionic bond to E169
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DynaMut2 mutant · R177C
Mutant C177 — ionic bond to E169 lost (11 contacts lost)
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Bond changes · DynaMut2 interaction analysis

11 lost2 gained12 preserved
Interaction typeWild-type partnerMutant partnerStatus
Ionic bondE169Lost
Hydrogen bondA134Lost
Hydrogen bondK135Lost
Hydrogen bondE173E173Preserved
Hydrogen bondR174R174Preserved
Hydrogen bondA180A180Preserved
Hydrogen bondL181L181Preserved
Hydrogen bondK252Gained
Polar contactA134Lost
Polar contactK135Lost
Polar contactE169Lost
Polar contactE173E173Preserved
Polar contactR174R174Preserved
Polar contactA175A175Preserved
Polar contactA179Lost
Polar contactA180A180Preserved
Polar contactL181L181Preserved
Van der WaalsL166Lost
Van der WaalsE169Lost
Van der WaalsA175A175Preserved
Van der WaalsA179Lost
Van der WaalsL181L181Preserved
Van der WaalsK252Gained
HydrophobicL181Lost
HydrophobicV248V248Preserved

Lost / gained / preserved interatomic contacts at the variant residue, from the DynaMut2 (Arpeggio) interaction analysis of the wild-type and energy-minimized mutant structures.

Computational Predictions

DynaMut2 ΔΔG
-0.88kcal/mol
Destabilising — mild
AlphaMissense
0.902
LPath
AlphaFold pLDDT
91
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationConflicting classifications of pathogenicity
Review statuscriteria provided, conflicting classifications
Associated conditionsWolfram syndrome 1
InheritanceWolfram syndrome 1 documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00082%
cDNA changec.529C>T
ClinVar accessionVCV000809614
Last evaluated2025/01/23 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 177 sits in the N-terminal cytoplasmic domain. Neighbors: LYS178 (2.5 Å — adjacent existing lysine), VAL176 (2.5 Å), GLU173 (3.7 Å — likely wild-type salt-bridge partner with R177), ARG174 (3.8 Å — second nearby arginine).

The wild-type R177 contributes to a positively-charged surface patch (with K178, R174) and likely salt-bridges with E173. Replacing R177 with cysteine eliminates the positive charge contribution and introduces a free thiol in the cytosol (less prone to aberrant disulfide than ER lumen but still a misfolding consideration).

|ΔΔG| 0.88 + AlphaMissense 0.902 + Wolfram 1 confirm severe consequence.

Amino-acid chemistry
Arginine (R) → Cysteine (C) — long positively-charged guanidinium replaced by short thiol.
Position in the protein
N-terminal cytoplasmic domain · position 177 (pLDDT 91).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.88 — fold survives. AlphaMissense 0.902 + Wolfram 1 confirm severe consequence.

Mechanism: loss of R177-E173 salt bridge plus charge loss from cytoplasmic surface patch. Therapeutic: site-directed at the R174-R177-E173 cluster.

Why this matters

R177C is part of a growing R→C class (with R703C, R708C, R732C, R685C, R653C). Drug discovery for R→C variants attends to both charge loss and aberrant thiol chemistry.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the R177C PDF below and upload it to Wolfram Intelligence to generate therapeutic-strategy proposals — guanidinium mimetics, sigma-1 agonist docking, NAC thiol-capping. NAC is already on the bench-testing list.

Download R177C PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin
Region1321 · Interaction with ATP6V1A