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March 1, 20260 citations

Expanding the Phenotype of Biallelic PIGG Variants: Motor Neuropathy With Peripheral Nerve Hyperexcitability.

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PSPedro Henrique Marte de Arruda SampaioCMCristiane Araújo Martins MorenoFPFilipe Tupinamba Di Pace

Key Points

  • This report aims to describe the clinical features and underlying mechanisms associated with biallelic PIGG variants in a patient.
  • Case report of a 27-year-old woman with a homozygous PIGG variant
  • Electrophysiological evaluation using electromyography to assess nerve function
  • Analysis of clinical symptoms including myokymia, gait ataxia, and tremor.
  • Identified lower limb myokymia and gait ataxia in the patient
  • Electromyography revealed myokymic discharges and pure motor polyneuropathy
  • Expanded understanding of PIGG-related disorders by linking PIGG variants to peripheral nerve hyperexcitability.

Abstract

Pathogenic variants in PIGG (phosphatidylinositol glycan anchor biosynthesis, class G) disrupt glycosylphosphatidylinositol (GPI) anchoring of cell-surface proteins. Recently, biallelic PIGG variants have been linked to motor neuropathy with conduction block and temporal dispersion, suggesting a role for defective GPI anchoring in peripheral nerve function. We describe a 27-year-old woman carrying a homozygous nonsense variant in PIGG, c.1515G>A (p.Trp505*), presenting with continuous lower limb myokymia, gait ataxia, tremor and distal weakness since early adolescence. Electrophysiological evaluation revealed widespread myokymic discharges on electromyography, consistent with peripheral nerve hyperexcitability, and a pure motor polyneuropathy with temporal dispersion. This case report expands the clinical spectrum of PIGG-related disorders by identifying peripheral nerve hyperexcitability as a defining feature. The potential mechanistic link between defective GPI anchoring and neuronal hyperexcitability mediated through impaired function of GPI-anchored proteins such as contactin-1 and contactin-2 offers a compelling hypothesis connecting peripheral neuropathy, hyperexcitability, and cerebellar dysfunction.

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Cite This Study

Sampaio et al. (2026) studied this question.

synapsesocial.com/papers/69a3d867ec16d51705d2f341https://doi.org/10.1002/ajmg.a.70106
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