Successful treatment of multicentric reticulohistiocytosis with leflunomide
Successful treatment of multicentric reticulohistiocytosis with leflunomide
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来氟米特成功治疗多中心网状组织细胞增多症
DOI：
10.1111/j.1365-2133.2009.09227.x
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发表时间：
2009
影响因子：
10.3
通讯作者：
S. Young
中科院分区：
文献类型：
--
作者：
A. Lonsdale;A. Haworth;F. McCrae;S. Young
SIR，我们描述了一位患有多中心网状组织细胞增多症 (MRH) 的患者，该患者对甲氨蝶呤、泼尼松龙、羟氯喹、柳氮磺吡啶和阿仑膦酸的组合治疗无效。在使用来氟米特后不久，他的病情就得到了改善。据我们所知，这是文献中第一篇成功使用来氟米特治疗 MRH 的报道。一名 74 岁男性，有 2 个月的手指肿胀病史，并伴有沿着手指边缘出现的非压痛性甲周丘疹和结节（图 1a、b）。一个月后，他出现关节痛，前胸和肩部出现非瘙痒性光分布性红斑皮疹。他的肘部（图 1c）和口腔内也出现了更多结节。既往病史包括高血压、高胆固醇血症、心房颤动和高级别滤泡性非霍奇金淋巴瘤，11 年前接受根治性放疗成功治疗。从出疹的丘疹和黄斑区域以及颊粘膜进行切开活检。所有样本均显示真皮浸润物由组织细胞样细胞片组成，具有丰富的“玻璃状”亮粉色细胞质。偶见多核细胞；特殊染色显示弥漫性 CD68 阳性，但 AE1/AE2、S100 和 CD34 阴性。细胞质膜图案中有一些阳性 CD31 染色。这些发现与 MRH 的诊断一致（图 2）。为了排除复发性淋巴瘤，患者接受了淋巴结活检：显示滤泡反应性增生。胃镜检查排除胃淋巴瘤，发现幽门处有异常皱襞；该区域的活检显示组织学变化与 MRH 一致。进一步的恶性肿瘤筛查包括胸部、腹部和骨盆的正常计算机断层扫描。肿瘤标志物正常（乳酸脱氢酶、前列腺特异性抗原、Ca 19-9、癌胚抗原和 CA125）。自身免疫概况
SIR, We describe a patient with multicentric reticulohistiocytosis (MRH) who failed to respond to a combination of methotrexate, prednisolone, hydroxychloroquine, sulfasalazine and alendronic acid. Improvement in his condition followed shortly after the introduction of leflunomide. To our knowledge, this is the first report in the literature of the successful use of leflunomide to treat MRH. A 74-year-old man presented with a 2-month history of swollen fingers with nontender periungual papules and nodules arising along the edge of his fingers (Fig. 1a,b). One month later he developed arthralgia and a nonpruritic, photodistributed, erythematous rash on his anterior chest and shoulders. He also developed further nodules on his elbows (Fig. 1c) and inside his mouth. Past medical history included hypertension, hypercholesterolaemia, atrial fibrillation and high-grade follicular non-Hodgkin lymphoma, successfully treated with radical radiotherapy 11 years previously. Incisional biopsies were taken from papular and macular areas of the eruption, as well as from the buccal mucosa. All samples showed a dermal infiltrate consisting of sheets of histiocyte-like cells with abundant ‘glassy’ bright pink cytoplasm. There were occasional multinucleate cells; special stains showed diffuse CD68 positivity but negative AE1 ⁄AE2, S100 and CD34. There was some positive CD31 staining in a cytoplasmic membrane pattern. These findings were consistent with a diagnosis of MRH (Fig. 2). To rule out recurrent lymphoma, the patient underwent lymph node biopsy: this showed follicular reactive hyperplasia. Gastroscopy to exclude gastric lymphoma revealed abnormal folds at the pylorus; biopsy from this area showed histological changes consistent with MRH. Further malignancy screen included a normal computed tomographic scan of the chest, abdomen and pelvis. Tumour markers were normal (lactate dehydrogenase, prostate-specific antigen, Ca 19-9, carcinoembryonic antigen and CA125). Autoimmune profile