塞扎里综合征患者出现嗜酸性毛囊炎和脱发,且对莫甘利珠单抗持续完全应答:病例报告

IF 3.4 3区 医学 Q2 HEMATOLOGY Therapeutic Advances in Hematology Pub Date : 2024-03-06 DOI:10.1177/20406207241235777
Jean-Matthieu L’Orphelin
{"title":"塞扎里综合征患者出现嗜酸性毛囊炎和脱发,且对莫甘利珠单抗持续完全应答:病例报告","authors":"Jean-Matthieu L’Orphelin","doi":"10.1177/20406207241235777","DOIUrl":null,"url":null,"abstract":"Mogamulizumab is a monoclonal antibody that binds to C–C chemokine receptor 4 (CCR4), initiating antibody-dependent cellular cytotoxicity. CCR4 is highly expressed in the cutaneous T-cell lymphoma subtypes mycosis fungoides and Sézary syndrome (SS), and mogamulizumab has been shown to be effective in patients with these conditions who were refractory to at least one prior systemic treatment. One of the more common adverse events encountered with mogamulizumab is rash, which may mimic disease progression and lead to premature discontinuation. Moreover, there has been some evidence to suggest that mogamulizumab-associated rash (MAR) is associated with improved outcomes in some patients, particularly those with SS. This report presents the case of a 72-year-old woman with SS, which manifested with macular and papular lesions and abnormal blood cytometry, who was treated with mogamulizumab after failure of bexarotene and photopheresis combination therapy. She achieved a complete response (CR), but experienced lymphopenia associated with histologically proven eosinophilic folliculitis (EF) of the scalp and alopecia. The EF responded well to initial topical corticosteroids, defined by regression of erythema and pustular involvement and reduction in pruritus-like symptoms, but without hair regrowth. Mogamulizumab was withdrawn after 32 cycles, but CR was maintained. To date, EF persists in the form of diffuse erythema without pustules or pruritus. A link between cluster of differentiation 4 lymphopenia and EF has previously been established; therefore, EF should be considered in patients who develop rash and lymphopenia while receiving treatment with mogamulizumab. MAR has been associated with clinical response to mogamulizumab, and this case report adds to the evidence that EF may also be associated with sustained clinical response following treatment cessation. However, regular monitoring is required to prevent a relapse of SS. Prospective studies are needed to confirm whether such an association between EF and CR following mogamulizumab exists.","PeriodicalId":23048,"journal":{"name":"Therapeutic Advances in Hematology","volume":null,"pages":null},"PeriodicalIF":3.4000,"publicationDate":"2024-03-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"An occurrence of eosinophilic folliculitis and alopecia associated with a sustained complete response to mogamulizumab in Sézary syndrome: a case report\",\"authors\":\"Jean-Matthieu L’Orphelin\",\"doi\":\"10.1177/20406207241235777\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"Mogamulizumab is a monoclonal antibody that binds to C–C chemokine receptor 4 (CCR4), initiating antibody-dependent cellular cytotoxicity. CCR4 is highly expressed in the cutaneous T-cell lymphoma subtypes mycosis fungoides and Sézary syndrome (SS), and mogamulizumab has been shown to be effective in patients with these conditions who were refractory to at least one prior systemic treatment. One of the more common adverse events encountered with mogamulizumab is rash, which may mimic disease progression and lead to premature discontinuation. Moreover, there has been some evidence to suggest that mogamulizumab-associated rash (MAR) is associated with improved outcomes in some patients, particularly those with SS. This report presents the case of a 72-year-old woman with SS, which manifested with macular and papular lesions and abnormal blood cytometry, who was treated with mogamulizumab after failure of bexarotene and photopheresis combination therapy. She achieved a complete response (CR), but experienced lymphopenia associated with histologically proven eosinophilic folliculitis (EF) of the scalp and alopecia. The EF responded well to initial topical corticosteroids, defined by regression of erythema and pustular involvement and reduction in pruritus-like symptoms, but without hair regrowth. Mogamulizumab was withdrawn after 32 cycles, but CR was maintained. To date, EF persists in the form of diffuse erythema without pustules or pruritus. A link between cluster of differentiation 4 lymphopenia and EF has previously been established; therefore, EF should be considered in patients who develop rash and lymphopenia while receiving treatment with mogamulizumab. MAR has been associated with clinical response to mogamulizumab, and this case report adds to the evidence that EF may also be associated with sustained clinical response following treatment cessation. However, regular monitoring is required to prevent a relapse of SS. Prospective studies are needed to confirm whether such an association between EF and CR following mogamulizumab exists.\",\"PeriodicalId\":23048,\"journal\":{\"name\":\"Therapeutic Advances in Hematology\",\"volume\":null,\"pages\":null},\"PeriodicalIF\":3.4000,\"publicationDate\":\"2024-03-06\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Therapeutic Advances in Hematology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/20406207241235777\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"HEMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Therapeutic Advances in Hematology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/20406207241235777","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"HEMATOLOGY","Score":null,"Total":0}
引用次数: 0

摘要

莫加莫珠单抗是一种单克隆抗体,能与C-C趋化因子受体4(CCR4)结合,启动抗体依赖性细胞毒性。CCR4在皮肤T细胞淋巴瘤亚型真菌病(mycosis fungoides)和塞扎里综合征(Sézary syndrome,SS)中高度表达,mogamulizumab已被证明对至少一种既往系统治疗无效的此类患者有效。莫加莫珠单抗较常见的不良反应之一是皮疹,这可能是疾病进展的假象,导致患者过早停药。此外,有证据表明,莫干珠单抗相关皮疹(MAR)与某些患者(尤其是 SS 患者)预后的改善有关。本报告介绍了一名 72 岁女性 SS 患者的病例,该患者表现为黄斑和丘疹病变以及异常血细胞计数,在贝沙罗汀和光动力疗法联合治疗失败后接受了莫干单抗治疗。她获得了完全应答(CR),但出现了淋巴细胞减少症,并伴有组织学证实的头皮嗜酸性毛囊炎(EF)和脱发。嗜酸性毛囊炎对最初的局部皮质类固醇反应良好,表现为红斑和脓疱累及消退,瘙痒症状减轻,但毛发没有再生。莫干单抗在 32 个周期后停药,但 CR 仍得以维持。迄今为止,EF仍以弥漫性红斑的形式存在,没有脓疱或瘙痒症状。此前已证实第4分化群淋巴细胞减少症与EF之间存在联系;因此,在接受莫干珠单抗治疗期间出现皮疹和淋巴细胞减少症的患者应考虑EF。MAR与莫干珠单抗的临床应答有关,本病例报告进一步证明,EF也可能与治疗停止后的持续临床应答有关。然而,需要定期监测以防止 SS 复发。需要进行前瞻性研究,以确认 EF 与莫干珠单抗治疗后的持续临床应答之间是否存在这种关联。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
An occurrence of eosinophilic folliculitis and alopecia associated with a sustained complete response to mogamulizumab in Sézary syndrome: a case report
Mogamulizumab is a monoclonal antibody that binds to C–C chemokine receptor 4 (CCR4), initiating antibody-dependent cellular cytotoxicity. CCR4 is highly expressed in the cutaneous T-cell lymphoma subtypes mycosis fungoides and Sézary syndrome (SS), and mogamulizumab has been shown to be effective in patients with these conditions who were refractory to at least one prior systemic treatment. One of the more common adverse events encountered with mogamulizumab is rash, which may mimic disease progression and lead to premature discontinuation. Moreover, there has been some evidence to suggest that mogamulizumab-associated rash (MAR) is associated with improved outcomes in some patients, particularly those with SS. This report presents the case of a 72-year-old woman with SS, which manifested with macular and papular lesions and abnormal blood cytometry, who was treated with mogamulizumab after failure of bexarotene and photopheresis combination therapy. She achieved a complete response (CR), but experienced lymphopenia associated with histologically proven eosinophilic folliculitis (EF) of the scalp and alopecia. The EF responded well to initial topical corticosteroids, defined by regression of erythema and pustular involvement and reduction in pruritus-like symptoms, but without hair regrowth. Mogamulizumab was withdrawn after 32 cycles, but CR was maintained. To date, EF persists in the form of diffuse erythema without pustules or pruritus. A link between cluster of differentiation 4 lymphopenia and EF has previously been established; therefore, EF should be considered in patients who develop rash and lymphopenia while receiving treatment with mogamulizumab. MAR has been associated with clinical response to mogamulizumab, and this case report adds to the evidence that EF may also be associated with sustained clinical response following treatment cessation. However, regular monitoring is required to prevent a relapse of SS. Prospective studies are needed to confirm whether such an association between EF and CR following mogamulizumab exists.
求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
4.30
自引率
0.00%
发文量
54
审稿时长
7 weeks
期刊介绍: Therapeutic Advances in Hematology delivers the highest quality peer-reviewed articles, reviews, and scholarly comment on pioneering efforts and innovative studies across all areas of hematology. The journal has a strong clinical and pharmacological focus and is aimed at clinicians and researchers in hematology, providing a forum in print and online for publishing the highest quality articles in this area.
期刊最新文献
Pharmacokinetics, pharmacodynamics, safety, and efficacy of crizanlizumab in patients with sickle cell disease: final results from the phase II SOLACE-adults study. Long-term risks of cardiovascular-specific mortality among myeloproliferative neoplasms patients. Clinical features and treatment of newly diagnosed multiple myeloma with secondary myelofibrosis: a retrospective study. Real-world efficacy of chidamide plus R-CHOP in newly diagnosed double-expressor diffuse large B-cell lymphoma. Iron deficiency anemia: an early clinical presentation of cytomegalovirus-induced hemorrhagic colitis in chronic myeloid leukemia patients under dasatinib treatment.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1