副肿瘤抗srp抗体阳性免疫介导的坏死性肌病与淋巴瘤相关的年轻女性。

IF 3.4 4区 医学 Q2 CLINICAL NEUROLOGY
Journal of neuromuscular diseases Pub Date : 2025-07-01 Epub Date: 2025-04-23 DOI:10.1177/22143602251327002
Johanna Heugenhauser, Franziska Di Pauli, Günther Stockhammer, Andreas Chott, Clemens Feistritzer, Daniel Egle, Benjamin Henninger, Julia Wanschitz
{"title":"副肿瘤抗srp抗体阳性免疫介导的坏死性肌病与淋巴瘤相关的年轻女性。","authors":"Johanna Heugenhauser, Franziska Di Pauli, Günther Stockhammer, Andreas Chott, Clemens Feistritzer, Daniel Egle, Benjamin Henninger, Julia Wanschitz","doi":"10.1177/22143602251327002","DOIUrl":null,"url":null,"abstract":"<p><p>We report an exceptional case of immune-mediated necrotizing myopathy (IMNM) associated with anaplastic large cell lymphoma (ALCL). A 26-year-old female patient presented with subacute bilateral proximal muscle weakness and myalgia, highly elevated creatin kinase (CK), and seropositivity for anti-SRP antibodies. Tumor screening by FDG-PET/CT detected an enlarged axillary lymph node with high FDG uptake. Histology of the excised lymph node revealed ALCL, positive for ALK and CD30. Therapy with brentuximab, cyclophosphamide and doxorubicin resulted in complete remission of the lymphoma, additional treatment with oral steroids and repeated cycles of intravenous immunoglobulins led to improvement of muscle weakness and normalization of CK. 18 months after diagnosis the patient remains tumor free with mild to moderate residual axial weakness. A literature review of paraneoplastic anti-SRP antibody positive IMNM identified eleven cases of whom five had a tumor diagnosis within a ±3 years window. The majority of patients had different solid tumors except one with a hematological malignancy. Despite the rare association of anti-SRP antibody positive IMNM and malignancy, early extensive tumor screening was crucial for the management of our patient.</p>","PeriodicalId":16536,"journal":{"name":"Journal of neuromuscular diseases","volume":" ","pages":"567-574"},"PeriodicalIF":3.4000,"publicationDate":"2025-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Paraneoplastic anti-SRP antibody positive immune-mediated necrotizing myopathy in a young female associated with lymphoma.\",\"authors\":\"Johanna Heugenhauser, Franziska Di Pauli, Günther Stockhammer, Andreas Chott, Clemens Feistritzer, Daniel Egle, Benjamin Henninger, Julia Wanschitz\",\"doi\":\"10.1177/22143602251327002\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>We report an exceptional case of immune-mediated necrotizing myopathy (IMNM) associated with anaplastic large cell lymphoma (ALCL). A 26-year-old female patient presented with subacute bilateral proximal muscle weakness and myalgia, highly elevated creatin kinase (CK), and seropositivity for anti-SRP antibodies. Tumor screening by FDG-PET/CT detected an enlarged axillary lymph node with high FDG uptake. Histology of the excised lymph node revealed ALCL, positive for ALK and CD30. Therapy with brentuximab, cyclophosphamide and doxorubicin resulted in complete remission of the lymphoma, additional treatment with oral steroids and repeated cycles of intravenous immunoglobulins led to improvement of muscle weakness and normalization of CK. 18 months after diagnosis the patient remains tumor free with mild to moderate residual axial weakness. A literature review of paraneoplastic anti-SRP antibody positive IMNM identified eleven cases of whom five had a tumor diagnosis within a ±3 years window. The majority of patients had different solid tumors except one with a hematological malignancy. Despite the rare association of anti-SRP antibody positive IMNM and malignancy, early extensive tumor screening was crucial for the management of our patient.</p>\",\"PeriodicalId\":16536,\"journal\":{\"name\":\"Journal of neuromuscular diseases\",\"volume\":\" \",\"pages\":\"567-574\"},\"PeriodicalIF\":3.4000,\"publicationDate\":\"2025-07-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of neuromuscular diseases\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1177/22143602251327002\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2025/4/23 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q2\",\"JCRName\":\"CLINICAL NEUROLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of neuromuscular diseases","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1177/22143602251327002","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/4/23 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0

摘要

我们报告一个例外的病例免疫介导坏死性肌病(IMNM)与间变性大细胞淋巴瘤(ALCL)。一位26岁的女性患者表现为亚急性双侧近端肌无力和肌痛,产生激酶(CK)高升高,抗srp抗体血清阳性。FDG- pet /CT肿瘤筛查发现腋窝淋巴结肿大,FDG摄取高。切除淋巴结的组织学显示ALCL, ALK和CD30阳性。布伦妥昔单抗、环磷酰胺和阿霉素治疗导致淋巴瘤完全缓解,口服类固醇和反复静脉注射免疫球蛋白的额外治疗导致肌肉无力改善和CK正常化。诊断后18个月,患者仍无肿瘤,伴有轻度至中度残余轴向无力。文献回顾了11例副肿瘤抗srp抗体阳性的IMNM,其中5例在±3年内被诊断为肿瘤。除一例合并血液学恶性肿瘤外,大多数患者均为不同类型的实体瘤。尽管抗srp抗体阳性的IMNM与恶性肿瘤很少有关联,但早期广泛的肿瘤筛查对我们患者的治疗至关重要。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Paraneoplastic anti-SRP antibody positive immune-mediated necrotizing myopathy in a young female associated with lymphoma.

We report an exceptional case of immune-mediated necrotizing myopathy (IMNM) associated with anaplastic large cell lymphoma (ALCL). A 26-year-old female patient presented with subacute bilateral proximal muscle weakness and myalgia, highly elevated creatin kinase (CK), and seropositivity for anti-SRP antibodies. Tumor screening by FDG-PET/CT detected an enlarged axillary lymph node with high FDG uptake. Histology of the excised lymph node revealed ALCL, positive for ALK and CD30. Therapy with brentuximab, cyclophosphamide and doxorubicin resulted in complete remission of the lymphoma, additional treatment with oral steroids and repeated cycles of intravenous immunoglobulins led to improvement of muscle weakness and normalization of CK. 18 months after diagnosis the patient remains tumor free with mild to moderate residual axial weakness. A literature review of paraneoplastic anti-SRP antibody positive IMNM identified eleven cases of whom five had a tumor diagnosis within a ±3 years window. The majority of patients had different solid tumors except one with a hematological malignancy. Despite the rare association of anti-SRP antibody positive IMNM and malignancy, early extensive tumor screening was crucial for the management of our patient.

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
Journal of neuromuscular diseases
Journal of neuromuscular diseases Medicine-Neurology (clinical)
CiteScore
5.10
自引率
6.10%
发文量
102
期刊介绍: The Journal of Neuromuscular Diseases aims to facilitate progress in understanding the molecular genetics/correlates, pathogenesis, pharmacology, diagnosis and treatment of acquired and genetic neuromuscular diseases (including muscular dystrophy, myasthenia gravis, spinal muscular atrophy, neuropathies, myopathies, myotonias and myositis). The journal publishes research reports, reviews, short communications, letters-to-the-editor, and will consider research that has negative findings. The journal is dedicated to providing an open forum for original research in basic science, translational and clinical research that will improve our fundamental understanding and lead to effective treatments of neuromuscular diseases.
×
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
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
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 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信