Bibliometric analysis is a widely used method to identify and evaluate the trends and characteristics of the scientific publications in a specific research field. A large volume of literature has been published in the field of malignant hyperthermia (MH). However, no bibliometric studies have been conducted to describe the characteristics of highly cited articles on MH. Therefore, this study aims to identify the 100 most frequently cited articles about malignant hyperthermia, describe their characteristics, and investigate research trends. Searches were performed in Web of Science Core Collection, Google Scholar, and Scopus from January 1900 to March 2023, with no language limitation. The 100 most frequently cited articles were selected and analyzed. Characteristics including publication year, journal, impact factor, authors, authors countries, citation number, journal citation indicator, study design, and topics were analyzed. For the 100 most cited articles, the number of times each article was cited ranged from 84 to 1254, with a median of 133. The number of articles published peaked in the decade of 1990s (n = 30). The articles were published in 39 journals, with Anesthesiology leading with 13 publications and then the British Journal of Anaesthesia with 12 publications. The authors were from 21 countries, led by the United States (n = 37). Forty articles focused on genetic susceptibility screening, 27 articles were about MH etiology, and 11 articles were about diagnosis and treatment. The mortality rate of MH and dantrolene availability are known to be uneven worldwide. This may partly be explained by the fact that scientific publications primarily report by authors from developed countries. This bibliometric analysis highlights the characteristics of the most influential research related to malignant hyperthermia, providing a reference for conducting future research.