Human T-cell lymphotropic virus type 1-associated myelopathy, Sjögren syndrome, and lymphocytic pneumonitis.
Human T-cell lymphotropic virus type 1-associated myelopathy, Sjögren syndrome, and lymphocytic pneumonitis.
复制标题
人类 T 细胞嗜淋巴细胞病毒 1 型相关脊髓病、干燥综合征和淋巴细胞性肺炎。
作者：
Aikaterini Kompoliti;B. Gage;Leena Sharma;James C. Daniels
客观化
描述非流行区首例人类T细胞嗜淋巴病毒1型(HTLV-1)相关性脊髓病、Sjögren综合征和淋巴细胞性肺炎。
背景
逆转录病毒与自身免疫性疾病的发病机制有关，包括Sjögren综合征。无症状淋巴细胞性肺炎在HTLV-1相关性脊髓病中很常见。有7例合并HTLV-1相关性脊髓病、Sjögren综合征和淋巴细胞性肺炎的病例报告，所有病例都在HTLV-1流行区。
设计
病例报告和文献复习。
结果
路易斯安那州新奥尔良一名40岁的克里奥尔妇女，表现为进行性痉挛瘫痪和劳力性呼吸困难。对系统的审查发现，慢性主诉与干燥综合征一致。通过脑脊液中的聚合酶链式反应发现她患有HTLV-1相关性脊髓病。SSA水平升高，Schirmer试验阳性，小唾液腺活检结果与Sjögren综合征一致。肺活检标本显示有明显的淋巴细胞浸润。
结论
本病例对HTLV-1在自身免疫发展中的作用提出了疑问。这在非流行地区也是独一无二的。
OBJECTIVE
To describe the first case of human T-cell lymphotropic virus type 1 (HTLV-1)-associated myelopathy, Sjögren syndrome, and lymphocytic pneumonitis in a nonendemic area.
BACKGROUND
Retroviruses are implicated in the pathogenesis of autoimmune diseases, including Sjögren syndrome. Asymptomatic lymphocytic pneumonitis is prevalent in HTLV-1-associated myelopathy. There are 7 case reports with the combination of HTLV-1-associated myelopathy, Sjögren syndrome, and lymphocytic pneumonitis, all of them in endemic areas for HTLV-1.
DESIGN
Case report and literature review.
RESULTS
A 40-year-old Creole woman from New Orleans, La, presented with progressive spastic paraparesis and exertional dyspnea. Review of systems revealed chronic complaints consistent with sicca syndrome. She was found to have HTLV-1-associated myelopathy by polymerase chain reaction in the cerebrospinal fluid. Increased levels of SSA, positive results on a Schirmer test, and the findings of biopsy of the minor salivary gland were consistent with Sjögren syndrome. A lung biopsy specimen showed marked lymphocytic infiltration.
CONCLUSIONS
The present case raises questions about the role of HTLV-1 in the development of autoimmunity. It also happens to be a unique occurrence in a nonendemic area.