ArticleThe American journal of case reports2026
Anesthesia Management in a Patient With a History of Fulminant Malignant Hyperthermia and a Homozygous RYR1 p.Arg530His Variant Undergoing Laparoscopic Surgery: A Case Report.
Article in The American journal of case reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
BACKGROUND Malignant hyperthermia (MH) is a rare but potentially fatal pharmacogenetic disorder triggered by volatile anesthetics or suxamethonium, most commonly associated with variants in the ryanodine receptor type 1 (RYR1) gene. Patients with confirmed or suspected MH susceptibility are advised to avoid triggering agents and undergo regional anesthesia or total intravenous anesthesia (TIVA). Remimazolam, an ultra-short-acting benzodiazepine approved in Japan for general anesthesia, has been shown in vitro not to increase intracellular calcium concentrations in cells expressing MH-associated RYR1 variants. However, clinical evidence regarding its safety in genetically confirmed MH remains limited. CASE REPORT An 80-year-old man with a history of fulminant MH at age 39, successfully treated with dantrolene, was scheduled for laparoscopic inguinal hernia repair. Previous calcium-induced calcium release testing confirmed MH susceptibility. Genetic analysis revealed homozygosity for the RYR1 c.1589G>A (p.Arg530His) variant, classified as likely pathogenic. Given his history and cardiovascular comorbidities, remimazolam-based TIVA was selected. Anesthesia was induced and maintained with remimazolam, remifentanil, and rocuronium under strict MH precautions, including immediate availability of dantrolene. Intraoperative end-tidal carbon dioxide, core temperature, and hemodynamics remained stable, with no signs of MH. Neuromuscular blockade was reversed with sugammadex, and recovery was uneventful without postoperative complications. CONCLUSIONS Remimazolam-based TIVA was successfully administered in a patient with genetically confirmed MH susceptibility carrying a homozygous likely pathogenic RYR1 variant. The perioperative course was uneventful, suggesting that remimazolam-based TIVA may be a feasible anesthetic approach in selected high-risk MH-susceptible patients, including those with severe prior episodes and significant cardiovascular comorbidities.
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