Suppr超能文献

副肿瘤性天疱疮:未确诊淋巴瘤的一种显著并发症。

Paraneoplastic Pemphigus: A Striking Complication of Undiagnosed Lymphoma.

作者信息

Adnani Blake O, O' Brien Kathleen, Myint Zaw W, Adler Brandon L

机构信息

Department of Internal Medicine, Keck School of Medicine, University of Southern California, Los Angeles, USA.

Department of Dermatology, Keck School of Medicine, University of Southern California, Los Angeles, USA.

出版信息

Case Rep Hematol. 2022 Oct 25;2022:3641474. doi: 10.1155/2022/3641474. eCollection 2022.

Abstract

A 49-year-old male with no past medical history presented with acute-onset painful mucosal erosions along with flaccid bullae on his trunk, scalp, and intertriginous areas. The patient initially underwent a skin biopsy which demonstrated suprabasilar acantholysis and lichenoid interface dermatitis. This was followed by a computed tomography scan which identified a large abdominal lymph node. Core needle biopsy of this node demonstrated follicular lymphoma. Lastly, indirect immunofluorescence (IIF) in rat bladder was positive (titer 1 : 10,240). This finding confirmed the diagnosis of paraneoplastic pemphigus (PNP) in the setting of follicular lymphoma. The patient's cutaneous disease was treated with a combination of intravenous immunoglobulin and methylprednisolone, along with intravenous rituximab, with a resolution of his cutaneous symptoms. His lymphoma was treated with six cycles of rituximab, cyclophosphamide, doxorubicin, vincristine, and prednisone (R-CHOP), with an interval decrease in his tumor burden. PNP is an autoimmune-mediated mucocutaneous disease associated with underlying neoplasm, most commonly non-Hodgkin lymphoma or chronic lymphocytic leukemia. Affected patients develop variable autoantibodies to antigens on keratinocytes and the basement membrane zone. Severe intractable stomatitis is characteristic, in addition to polymorphous cutaneous eruptions including bullae and erosions. Mortality rates can reach up to 90% due to malignancy, sepsis, or bronchiolitis obliterans, an irreversible and often lethal cause of pulmonary insufficiency. We highlight PNP manifesting in a patient with lymphoma, who responded well to the skin- and malignancy-directed treatments. PNP is an exceedingly rare diagnosis that should be considered in a patient with intractable stomatitis.

摘要

一名49岁男性,无既往病史,出现急性发作的疼痛性黏膜糜烂,同时在其躯干、头皮和褶皱部位出现松弛性大疱。患者最初接受了皮肤活检,结果显示基底细胞上层棘层松解和苔藓样界面性皮炎。随后进行的计算机断层扫描发现一个大的腹部淋巴结。对该淋巴结进行粗针活检显示为滤泡性淋巴瘤。最后,大鼠膀胱间接免疫荧光(IIF)呈阳性(滴度1∶10240)。这一发现证实了该患者在滤泡性淋巴瘤背景下诊断为副肿瘤性天疱疮(PNP)。患者的皮肤疾病采用静脉注射免疫球蛋白和甲泼尼龙联合静脉注射利妥昔单抗进行治疗,皮肤症状得到缓解。他的淋巴瘤采用六个周期的利妥昔单抗、环磷酰胺、阿霉素、长春新碱和泼尼松(R-CHOP)方案进行治疗,肿瘤负荷逐渐减轻。PNP是一种与潜在肿瘤相关的自身免疫介导的黏膜皮肤疾病,最常见于非霍奇金淋巴瘤或慢性淋巴细胞白血病。受影响的患者会产生针对角质形成细胞和基底膜区抗原的多种自身抗体。除了包括大疱和糜烂在内的多形性皮肤疹外,严重的顽固性口腔炎是其特征。由于恶性肿瘤、败血症或闭塞性细支气管炎(一种不可逆且通常致命的肺功能不全病因),死亡率可达90%。我们重点介绍了一名淋巴瘤患者表现出的PNP,该患者对针对皮肤和恶性肿瘤的治疗反应良好。PNP是一种极其罕见的诊断,对于患有顽固性口腔炎的患者应予以考虑。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7270/9626227/9e5d31f592bb/CRIHEM2022-3641474.001.jpg

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验