Efficacy of rituximab treatment for thyroid-associated ophthalmopathy as a result of intraorbital B-cell depletion in one patient unresponsive to steroid immunosuppression

Efficacy of rituximab treatment for thyroid-associated ophthalmopathy as a result of intraorbital B-cell depletion in one patient unresponsive to steroid immunosuppression
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DOI:
10.1530/eje.1.02119
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发表时间:
2006-04-01
影响因子:
5.8
通讯作者:
Beck-Peccoz, P
Beck-Peccoz, P
中科院分区:
医学1区
文献类型:
--
作者:
Salvi, M;Vannucchi, G;Beck-Peccoz, P

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1例Graves甲亢合并眼病处于活跃期,对类固醇无反应,采用抗cd20单克隆抗体利妥昔单抗(RTX)治疗。作为公开研究的一部分。研究了RTX对甲状腺和眼眶组织的影响。该眼病对WIN治疗有反应,眼部体征得到改善,临床活动评分在3个月内从5分降至2分,同时患者有外周b细胞衰竭。在b细胞耗竭的6个月期间,甲状腺功能亢进没有改善,RTX治疗后血清tsh受体抗体(TRAb)水平没有显著改变。因此,患者行甲状腺全切除术,甲状腺组织标本中发现少量b细胞。在患者眼病保持稳定(临床活动评分= 2)的情况下,我们进行了眼眶矫正减压,发现眼眶组织标本中淋巴细胞缺失。我们认为RTX治疗Graves病可能通过减少眼眶淋巴细胞总量而改善眼病。Graves甲亢的持续存在表明,单周期RTX不会导致甲状腺组织中淋巴细胞的完全消耗,因此没有观察到血清TRAb的下降。
One patient with Graves' hyperthyroidism and ophthalmopathy in its active phase and unresponsive to steroid, was treated with the anti-CD20 monoclonal antibody, rituximab (RTX). as part of an open study. The effect of RTX in the thyroid and the orbital tissues was Studied. The ophthalmopathy responded to WIN therapy by ameliorating the eye signs with a decrease in the clinical activity score from 5 to 2 in 3 months, while the patient had peripheral B-cell depletion. Hyperthyroidism did not improve during the 6 months of B-cell depletion and serum TSH-receptor antibodies (TRAb) levels did not significantly change after RTX therapy. Therefore, the patient underwent total thyroidectomy and few B-cells were found in the thyroid tissue specimens. While the patient eye disease remained stable (clinical activity score = 2), we performed corrective orbital decompression and we found absence of lymphocytes in the orbital tissue specimens. We believe that RTX treatment in Graves' disease may cause amelioration of ophthalmopathy by depleting total lymphocytes Population in the orbit. The persistence of Graves' hyperthyroidism suggests that a single cycle of RTX does not result in complete lymphocyte depletion in thyroid tissue and thus no decline in serum TRAb was observed.