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K363T

Category 3/4 — Most DruggableLikely pathogenicTransmembrane · predictedEditorial
LysineThreonine at position 363 · Connecting loop · WFS1 (Wolframin)

Lysine → Threonine at position 363 in a connecting loop. ClinVar Likely pathogenic for Wolfram syndrome 1. AlphaMissense 0.874, DynaMut2 ΔΔG -0.43 kcal/mol (destabilising). Charge-loss variant adjacent to T361 (T361I Atlas card).

Interactive 3D Structure

Wild-type reference
Wild-type K363 — hydrogen bond to D367
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DynaMut2 mutant · K363T
Mutant T363 — hydrogen bond to C360 lost (5 contacts lost)
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Bond changes · DynaMut2 interaction analysis

5 lost3 gained11 preserved
Interaction typeWild-type partnerMutant partnerStatus
Hydrogen bondI359I359Preserved
Hydrogen bondC360C360Preserved
Hydrogen bondQ366Q366Preserved
Hydrogen bondD367D367Preserved
Hydrogen bondP404Gained
Hydrogen bondS630Lost
Polar contactI359I359Preserved
Polar contactC360C360Preserved
Polar contactT361T361Preserved
Polar contactF365F365Preserved
Polar contactQ366Q366Preserved
Polar contactD367D367Preserved
Polar contactS630Lost
Van der WaalsI359Gained
Van der WaalsT361Gained
Van der WaalsF365F365Preserved
Van der WaalsD367Lost
Van der WaalsS630Lost
HydrophobicP404Lost

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
-0.43kcal/mol
Destabilising — mild
AlphaMissense
0.874
LPath
AlphaFold pLDDT
86
model confidence
Schema
Cat 3/4
Category 3/4 — Most Druggable

Clinical Evidence

ClinVar classificationLikely pathogenic
Review statusno assertion criteria provided
Associated conditionsWolfram syndrome 1
InheritanceWolfram syndrome 1 (AR) documented.
Population frequency (gnomAD v4)Ultra-rare · AF 0.00066%
cDNA changec.1088A>C
ClinVar accessionVCV001327580
Last evaluated2021/11/25 00:00

Observed at very low frequency in gnomAD.

Structural Context

Position 363 sits in a connecting loop, two residues from T361 (T361I Atlas card). The AlphaFold model places K363 within 5 Å of LEU362 (2.5 Å), VAL364 (2.5 Å), CYS360 (3.6 Å), ILE359 (3.8 Å), and GLN366 (4.2 Å).

The wild-type lysine at 363 is the partner residue identified in the T361I Atlas card — T361's hydroxyl was hypothesized to H-bond with K363's amine. K363T replaces lysine with threonine, which is precisely the residue that T361I introduced at the wild-type T position. The K363 amine is replaced by a hydroxyl.

This means K363T and T361I together replace the wild-type T361-K363 H-bond pair (hydroxyl-amine) with a T361-T363 pair (hydroxyl-hydroxyl in the T361 variant; or the wild-type T plus T363 in the K363T variant). The geometry changes but H-bonding may persist.

The |ΔΔG| of 0.43 reflects modest fold cost. AlphaMissense's 0.874 + Wolfram 1 confirm severe functional consequence — the partner-recognition geometry depends on the precise wild-type chemistry, not just the H-bonding capacity.

Amino-acid chemistry
Lysine (K) → Threonine (T) — large positively-charged amine replaced by small polar hydroxyl. Loss of charge and side-chain length.
Position in the protein
Connecting loop · position 363 in a loop region adjacent to T361 (pLDDT 86).

Druggability Assessment

Category 3/4 — Most Druggable. |ΔΔG| = 0.43 — fold survives. AlphaMissense 0.874 + Wolfram 1 confirm severe functional consequence.

Mechanism is disruption of the T361-K363 H-bond pair. Therapeutic strategy: same microregion as T361I.

Why this matters

K363T and T361I are sister variants at the same H-bond partner pair. Drug discovery in the T361-K363 microregion has two convergent variant targets.
Therapeutic Strategy Handoff · prediction

Feed this card to Wolfram Intelligence

Download the K363T 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 K363T PDF card ↓Strategies are AI-generated predictions, not validated therapeutics.

UniProt Domain Annotation

Chain1890 · Wolframin