Andrew El Alam, Mohamad Fleifel, Dana El Masri, Bertha Maria Nassani, Jessica Abou Chaaya, Mahamadou Minkailou, Mariana Barbat, Arnaud Monier
{"title":"A Case of Myxedema Coma and Adrenal Insufficiency Post Pembrolizumab.","authors":"Andrew El Alam, Mohamad Fleifel, Dana El Masri, Bertha Maria Nassani, Jessica Abou Chaaya, Mahamadou Minkailou, Mariana Barbat, Arnaud Monier","doi":"10.1155/2024/5444975","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>Despite their important clinical benefits, immune checkpoint inhibitors (ICIs) are associated with a spectrum of side effects known as immune-related adverse events (irAEs). These can be of various organ system backgrounds, including dermatologic, pulmonary, gastrointestinal, and endocrine. Polyglandular endocrinopathies (PLEs) post-ICIs therapy has been reported in the literature; however, to our knowledge, only a few have been documented with pembrolizumab. <i>Case Report</i>. We present a case of a female patient who developed myxedema coma (MC) and adrenal insufficiency (AI) after 4 months of stopping pembrolizumab, a programed-cell death-1 inhibitor. The patient was clinically symptomatic and was subsequently treated with levothyroxine and hydrocortisone. <i>Discussion</i>. It is very important to be vigilant and alert in detecting MC and AI to avoid any mortality. Pembrolizumab's effect on inducing antitumor responses leads to a wide variety of multiorgan alterations. Its role in raising the risk of all-grade endocrine disorders has been previously highlighted along with thyroidal dysfunctions. Our patient's presentation falls within the literature-based median time for hypothyroidism and AI with respect to the period from the initiation of pembrolizumab. The patient's predisposition to hypothyroidism and the likelihood of intertwined manifestations of AI and hypothyroidism should always be considered in the setting of critical illness.</p><p><strong>Conclusion: </strong>It is of high significance to explore the mechanism of action of ICIs and their side effects. PLEs can house some endocrinologic emergencies that are life threatening.</p>","PeriodicalId":9621,"journal":{"name":"Case Reports in Endocrinology","volume":null,"pages":null},"PeriodicalIF":0.9000,"publicationDate":"2024-07-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11254456/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Case Reports in Endocrinology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1155/2024/5444975","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/1/1 0:00:00","PubModel":"eCollection","JCR":"Q4","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
引用次数: 0
Abstract
Background: Despite their important clinical benefits, immune checkpoint inhibitors (ICIs) are associated with a spectrum of side effects known as immune-related adverse events (irAEs). These can be of various organ system backgrounds, including dermatologic, pulmonary, gastrointestinal, and endocrine. Polyglandular endocrinopathies (PLEs) post-ICIs therapy has been reported in the literature; however, to our knowledge, only a few have been documented with pembrolizumab. Case Report. We present a case of a female patient who developed myxedema coma (MC) and adrenal insufficiency (AI) after 4 months of stopping pembrolizumab, a programed-cell death-1 inhibitor. The patient was clinically symptomatic and was subsequently treated with levothyroxine and hydrocortisone. Discussion. It is very important to be vigilant and alert in detecting MC and AI to avoid any mortality. Pembrolizumab's effect on inducing antitumor responses leads to a wide variety of multiorgan alterations. Its role in raising the risk of all-grade endocrine disorders has been previously highlighted along with thyroidal dysfunctions. Our patient's presentation falls within the literature-based median time for hypothyroidism and AI with respect to the period from the initiation of pembrolizumab. The patient's predisposition to hypothyroidism and the likelihood of intertwined manifestations of AI and hypothyroidism should always be considered in the setting of critical illness.
Conclusion: It is of high significance to explore the mechanism of action of ICIs and their side effects. PLEs can house some endocrinologic emergencies that are life threatening.
背景:尽管免疫检查点抑制剂(ICIs)具有重要的临床疗效,但也会产生一系列副作用,即免疫相关不良事件(irAEs)。这些副作用可能来自不同的器官系统,包括皮肤、肺部、胃肠道和内分泌系统。有文献报道了ICIs治疗后的多腺体内分泌病(PLEs);然而,据我们所知,只有少数病例记录了使用pembrolizumab治疗后的多腺体内分泌病。病例报告。我们报告了一例女性患者在停用程序性细胞死亡-1抑制剂pembrolizumab 4个月后出现肌水肿昏迷(MC)和肾上腺功能不全(AI)的病例。患者临床症状明显,随后接受了左甲状腺素和氢化可的松治疗。讨论。在发现 MC 和 AI 时保持警惕和警觉以避免任何死亡是非常重要的。Pembrolizumab 诱导抗肿瘤反应的作用会导致多种多器官改变。以前曾强调过它与甲状腺功能障碍一样,会增加所有级别内分泌失调的风险。我们这位患者的病例与文献报道的甲状腺功能减退症和 AI 的中位时间相符,与开始使用 pembrolizumab 的时间相符。在病情危重的情况下,应始终考虑患者的甲状腺功能减退症易感性以及甲状腺功能减退症和甲状腺功能减退症交织表现的可能性:探索ICIs的作用机制及其副作用具有重要意义。PLE可引发一些危及生命的内分泌急症。