Femi Williams Adeoye, Nida Jaffar, Sanggeeta Surandran, Gulshad Begum, Mohammad Rafiqul Islam
{"title":"杜伐单抗诱发的三M综合征","authors":"Femi Williams Adeoye, Nida Jaffar, Sanggeeta Surandran, Gulshad Begum, Mohammad Rafiqul Islam","doi":"10.12890/2024_004729","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>While the use of immunotherapy has revolutionised the treatment of various cancers, it is often associated with a myriad of immune-related adverse effects.</p><p><strong>Case presentation: </strong>In this article, we report a rare case of durvalumab-induced triple-M syndrome in a 69-year-old woman with stage III lung adenocarcinoma. She was admitted with profound generalised muscle weakness, myalgia, and exertional breathlessness, about a week into her second cycle of durvalumab, an immune checkpoint inhibitor. She had clinicopathological features of myositis, myasthenia and myocarditis with acute onset symptomatic tri-fascicular block on electrocardiogram, requiring urgent cardiology intervention. Durvalumab was discontinued and she was treated with a combination of high-dose steroids and intravenous immunoglobulin after which she had clinical and biochemical improvement, albeit with residual muscle weakness.</p><p><strong>Conclusion: </strong>Myocarditis-myositis-myasthenia complex is a rare side effect of immunotherapy which has been reported in other immune checkpoint inhibitors, but less so with durvalumab. We report this clinical case to raise awareness of this rare and potentially life-threatening adverse effect of this agent.</p><p><strong>Learning points: </strong>Triple-M syndrome is a rare immune-related adverse effect, which has been noted in other immune checkpoint inhibitors, but less so with durvalumab specifically.Immunotherapy-induced myositis, myocarditis and myasthenia can occur in isolation or, rarely, in association as a syndrome.This case demonstrates the potentially life-threatening nature of this entity, the need for early recognition, and multi-specialist teamwork to ensure good outcome.</p>","PeriodicalId":11908,"journal":{"name":"European journal of case reports in internal medicine","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2024-07-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11313117/pdf/","citationCount":"0","resultStr":"{\"title\":\"Durvalumab-Induced Triple-M Syndrome.\",\"authors\":\"Femi Williams Adeoye, Nida Jaffar, Sanggeeta Surandran, Gulshad Begum, Mohammad Rafiqul Islam\",\"doi\":\"10.12890/2024_004729\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Background: </strong>While the use of immunotherapy has revolutionised the treatment of various cancers, it is often associated with a myriad of immune-related adverse effects.</p><p><strong>Case presentation: </strong>In this article, we report a rare case of durvalumab-induced triple-M syndrome in a 69-year-old woman with stage III lung adenocarcinoma. She was admitted with profound generalised muscle weakness, myalgia, and exertional breathlessness, about a week into her second cycle of durvalumab, an immune checkpoint inhibitor. She had clinicopathological features of myositis, myasthenia and myocarditis with acute onset symptomatic tri-fascicular block on electrocardiogram, requiring urgent cardiology intervention. Durvalumab was discontinued and she was treated with a combination of high-dose steroids and intravenous immunoglobulin after which she had clinical and biochemical improvement, albeit with residual muscle weakness.</p><p><strong>Conclusion: </strong>Myocarditis-myositis-myasthenia complex is a rare side effect of immunotherapy which has been reported in other immune checkpoint inhibitors, but less so with durvalumab. We report this clinical case to raise awareness of this rare and potentially life-threatening adverse effect of this agent.</p><p><strong>Learning points: </strong>Triple-M syndrome is a rare immune-related adverse effect, which has been noted in other immune checkpoint inhibitors, but less so with durvalumab specifically.Immunotherapy-induced myositis, myocarditis and myasthenia can occur in isolation or, rarely, in association as a syndrome.This case demonstrates the potentially life-threatening nature of this entity, the need for early recognition, and multi-specialist teamwork to ensure good outcome.</p>\",\"PeriodicalId\":11908,\"journal\":{\"name\":\"European journal of case reports in internal medicine\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-07-30\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11313117/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"European journal of case reports in internal medicine\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.12890/2024_004729\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"Q3\",\"JCRName\":\"Medicine\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"European journal of case reports in internal medicine","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.12890/2024_004729","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
Background: While the use of immunotherapy has revolutionised the treatment of various cancers, it is often associated with a myriad of immune-related adverse effects.
Case presentation: In this article, we report a rare case of durvalumab-induced triple-M syndrome in a 69-year-old woman with stage III lung adenocarcinoma. She was admitted with profound generalised muscle weakness, myalgia, and exertional breathlessness, about a week into her second cycle of durvalumab, an immune checkpoint inhibitor. She had clinicopathological features of myositis, myasthenia and myocarditis with acute onset symptomatic tri-fascicular block on electrocardiogram, requiring urgent cardiology intervention. Durvalumab was discontinued and she was treated with a combination of high-dose steroids and intravenous immunoglobulin after which she had clinical and biochemical improvement, albeit with residual muscle weakness.
Conclusion: Myocarditis-myositis-myasthenia complex is a rare side effect of immunotherapy which has been reported in other immune checkpoint inhibitors, but less so with durvalumab. We report this clinical case to raise awareness of this rare and potentially life-threatening adverse effect of this agent.
Learning points: Triple-M syndrome is a rare immune-related adverse effect, which has been noted in other immune checkpoint inhibitors, but less so with durvalumab specifically.Immunotherapy-induced myositis, myocarditis and myasthenia can occur in isolation or, rarely, in association as a syndrome.This case demonstrates the potentially life-threatening nature of this entity, the need for early recognition, and multi-specialist teamwork to ensure good outcome.
期刊介绍:
The European Journal of Case Reports in Internal Medicine is an official journal of the European Federation of Internal Medicine (EFIM), representing 35 national societies from 33 European countries. The Journal''s mission is to promote the best medical practice and innovation in the field of acute and general medicine. It also provides a forum for internal medicine doctors where they can share new approaches with the aim of improving diagnostic and clinical skills in this field. EJCRIM welcomes high-quality case reports describing unusual or complex cases that an internist may encounter in everyday practice. The cases should either demonstrate the appropriateness of a diagnostic/therapeutic approach, describe a new procedure or maneuver, or show unusual manifestations of a disease or unexpected reactions. The Journal only accepts and publishes those case reports whose learning points provide new insight and/or contribute to advancing medical knowledge both in terms of diagnostics and therapeutic approaches. Case reports of medical errors, therefore, are also welcome as long as they provide innovative measures on how to prevent them in the current practice (Instructive Errors). The Journal may also consider brief and reasoned reports on issues relevant to the practice of Internal Medicine, as well as Abstracts submitted to the scientific meetings of acknowledged medical societies.