Erosive oral lichen planus as a sign of paraneoplastic pemphigus
Erosive oral lichen planus as a sign of paraneoplastic pemphigus
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DOI：
10.1111/1346-8138.13329
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发表时间：
2016-08
期刊：
影响因子：
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通讯作者：
Mingyue Wang;Xue Wang;Tiancheng Chen;Junyu Zhao;Yang Peng;Xixue Chen;Xuejun Zhu
中科院分区：
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作者：
Mingyue Wang;Xue Wang;Tiancheng Chen;Junyu Zhao;Yang Peng;Xixue Chen;Xuejun Zhu
尊敬的编辑， 我们饶有兴趣地阅读了 Motegi 等人的文章。出现在最近一期的《皮肤病学杂志》上。作者描述了他们科室 50 名诊断为 LP 的患者中的 3 名患有扁平苔藓 (LP) 并伴有胸腺瘤的患者，并回顾了已发表的作品中的 29 例此类并发症。该文章促使人们考虑对这两种疾病的患者进行进一步调查和适当治疗的必要性。然而，在上述患者中，至少有一些患者需要排除副肿瘤性天疱疮（PNP）或所谓的副肿瘤性自身免疫性多器官综合征，然后才能在 LP 和胸腺瘤之间建立简单的联系。 Anhalt 等提出的 PNP 诊断标准。包括： (i) 疼痛性、进行性口腔炎，优先累及舌头； (ii) 组织学上的棘层松解症，或苔藓样皮炎或界面性皮炎； (iii) 存在循环抗斑蛋白自身抗体，主要针对 envoplakin 和 periplakin； (iv) 存在潜在的淋巴增殖性肿瘤，例如淋巴瘤、白血病、卡斯尔曼病和胸腺瘤。根据这些标准，我们在 2005 年介绍了两名 PNP 患者，一名患有胸腺瘤，另一名患有滤泡树突细胞肉瘤。此外，在这项研究中，在这些肿瘤的培养基中检测到了针对这些斑蛋白共享的同源片段的自身抗体，表明这两种免疫性疾病之间可能存在联系。此外，在我们按照相同标准诊断的 101 名 PNP 患者中，20 名（19.8%）伴有胸腺瘤。这些患者的显着特征是舌头和颊粘膜糜烂以及手脚角化过度红斑。据我们所知，PNP 可能会出现 LP 样、多形红斑样、天疱疮样或类天疱疮样病变，但这些具有 LP 样皮疹的 PNP 与经典 LP 不同，因为作为自身免疫综合征的一部分，它们通常表现出多形性皮疹（如病例 1 中描述的淡红色红斑和水疱）和非典型重叠组织学特征。在我们的 20 名胸腺瘤 PNP 患者中，8 名（44.4%）出现呼吸困难，主要由闭塞性细支气管炎（与病例 16 的情况类似，也可能是作者总结的已发表作品中病例 29 中支气管扩张的原因）或重症肌无力引起。考虑到大鼠膀胱的间接免疫荧光仅在 52.6% (10/19) 的胸腺瘤 PNP 病例中呈阳性（Castleman 病 PNP 中为 96.2% [51/53]），如果作者进行免疫印迹或免疫沉淀来检测抗斑蛋白自身抗体，以确认或排除这些患者中 PNP 的可能性，那么将提供丰富的信息。此外，我们建议对所有顽固性糜烂性口腔 LP 患者进行这些免疫学研究，这可能有助于发现潜在的淋巴增殖性肿瘤，包括但不限于胸腺瘤。
Dear Editor, We read with great interest the article by Motegi et al. that appeared in the recent issue of the Journal of Dermatology. The authors described three patients with lichen planus (LP) accompanied by thymoma among 50 patients diagnosed with LP in their department, and reviewed 29 such complications in the published work. The article prompted consideration of the necessities of further investigations and proper treatment of patients with these two diseases. Nevertheless, in the mentioned patients, at least some of them were in need of excluding paraneoplastic pemphigus (PNP) or the so-called paraneoplastic autoimmune multiorgan syndrome before drawing a simple linkage between LP and thymoma. The diagnostic criteria of PNP suggested by Anhalt et al. includes: (i) painful and progressive stomatitis with preferential tongue involvement; (ii) acantholysis, or lichenoid or interface dermatitis, in histology; (iii) presence of circulating anti-plakin autoantibodies, mostly against envoplakin and periplakin; and (iv) presence of underlying lymphoproliferative neoplasm such as lymphoma, leukemia, Castleman’s disease and thymoma. Following these criteria, we presented two PNP patients in 2005, one with thymoma and the other with follicular dendritic cell sarcoma. Also in this study, autoantibodies against homologous fragments shared by these plakin proteins were detected in the culture media of these neoplasms, suggesting a possible connection between these two immunological disorders. Furthermore, in 101 PNP patients we diagnosed following the same criteria, 20 (19.8%) were accompanied by thymoma. These patients were remarkably characterized by erosive tongue and buccal mucosa and predominant hyperkeratotic erythema of the hands and feet. To our knowledge, PNP may develop LP-like, erythema multiforme-like, pemphigus-like or pemphigoid-like lesions, but these PNP with LP-like eruptions were distinct from the classical LP in that, as a part of the autoimmune syndrome, they usually show polymorphic eruptions (as both pale-reddish erythema and vesicles described in their case 1) and atypical overlapping histological features. In our 20 thymoma PNP patients, eight (44.4%) experienced dyspnea, mainly caused by bronchiolitis obliterans (similar situation as in case 16 and possibly the cause of bronchiectasis in case 29 in the published work the authors summarized) or myasthenia gravis. Considering that indirect immunofluorescence on rat bladders was only positive in 52.6% (10/19) of thymoma PNP cases (96.2% [51/53] in Castleman’s disease PNP), it would be informative if the authors performed immunoblotting or immunoprecipitation to detect anti-plakin autoantibodies to confirm or exclude the possibilities of PNP in these patients. Additionally, we suggest these immunological investigations on all recalcitrant erosive oral LP patients, which may help finding latent lymphoproliferative neoplasms including, but not limited to, thymoma.