Novel Hypotheses on Neurophatic Pain in Isaacs Sydrome and Drug Therapy

Author(s): Lourdes de Fatima Ibanez Valdes and Humberto Foyaca Sibat*

Abstract

Introduction: In 2001, we reported two patients with Isaacs' syndrome who responded remarkably well clinically and electromyographically to low oral doses of phenytoin. Isaacs Syndrome (IS) is characterized by recurrent spontaneous muscle activity of the peripheral nerve source that can be triggered by induced muscle contraction or voluntary muscular activity. Some authors have proposed an autoimmune aetiology based on clinical events suggesting a possible autoimmune aetiology, including the presence of oligoclonal bands in the cerebrospinal fluid and a good response to plasmapheresis. The primary aim of this study is to answer the following research questions: What is the pathophysiology of neuritic pain in patients presenting to IS? What is the best drug therapy choice?

Methods: We searched the literature, following the guidelines outlined in the PRISMA statement. From January 2023 to January 2025, the authors searched the scientific databases, Scopus, Embassy, Medline, and PubMed Central using the following searches: "Isaac syndrome" OR "neuropathic pain" OR "therapy of neuropathic pain" OR "treatment of Issac syndrome" OR "therapy of NP and IS", OR "mTOR in NP" OR "mTOR in IS".

Results: After screening the full-text articles for relevance, xx were included for final review. However, no article was selected when we searched for association/NP/IS/Drug therapy.

Conclusions: Based on the information reported by other investigators, we have developed some hypotheses regarding the pathogenesis of this condition and novel drug therapy.

Share this article