Background

Immune checkpoint blockade therapy may lead to thyroid dysfunction in 3–7% of treated patients. Alemtuzumab is a CD52 inhibitor leading to thyroid dysfunction in approximately 40% of patients. A female patient was affected by multiple sclerosis (MS) and subclinical hyperthyroidism due to an autonomously functioning thyroid nodule (AFTN). After alemtuzumab treatment, she developed aggressive clinical hyperthyroidism consistent with Marine-Lenhart syndrome.

Case presentation

A 36-year-old woman presented in July 2019 with symptoms of hyperthyroidism and eye complaints. Three years earlier, she was diagnosed with MS. Subclinical hyperthyroidism was diagnosed in April 2017. Thyroid scintigraphy showed an intranodular distribution of ^99mTc-pertechnatate consisting of an AFTN in the right lobe of the thyroid. In June 2018, because of the MS, she was treated with alemtuzumab. In November 2018, she was started on methimazole treatment because of the symptoms of hyperthyroidism. In December 2018, thyroid function was normal under methimazole treatment. In June 2019, the patient received a second round of alemtuzumab administration. One month later, she developed symptoms of hyperthyroidism. These symptoms were accompanied by diplopia. Blood tests showed severe hyperthyroidism. Thyroid scintigraphy showed a diffuse distribution of ^99mTc-pertechnatate and the presence of a “cool” area in the right lobe of the thyroid, confirmed by ultrasonography. The nodule was diagnosed as a low-risk indeterminate lesion.

Conclusion

We present a case of Graves’ disease with active, moderate-to-severe Graves’ ophthalmopathy in a patient with pre-existing AFTN presenting with a coexisting, rare case of Marine-Lenhart syndrome associated with immune reconstitution after alemtuzumab treatment.

中文翻译：

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一名接受阿仑单抗治疗多发性硬化症的女性​​患者出现 Marine-Lenhart 综合征和 Graves 眼病。

背景

免疫检查点阻断疗法可能会导致 3-7% 接受治疗的患者出现甲状腺功能障碍。阿仑单抗是一种 CD52 抑制剂，可导致约 40% 的患者出现甲状腺功能障碍。一名女性患者因自主功能性甲状腺结节 (AFTN) 而患有多发性硬化症 (MS) 和亚临床甲状腺功能亢进症。在阿仑单抗治疗后，她出现了与 Marine-Lenhart 综合征一致的侵袭性临床甲状腺功能亢进症。

案例展示

一名 36 岁女性于 2019 年 7 月出现甲状腺功能亢进和眼部不适症状。三年前，她被诊断出患有 MS。2017年4月确诊亚临床甲亢，甲状腺闪烁扫描显示结节内分布^99mTc-pertechnatate 由甲状腺右叶的 AFTN 组成。2018 年 6 月，因为 MS，她接受了阿仑单抗治疗。2018 年 11 月，她因甲亢症状开始接受甲巯咪唑治疗。2018年12月甲巯咪唑治疗甲状腺功能正常。2019 年 6 月，患者接受了第二轮阿仑单抗给药。一个月后，她出现了甲亢的症状。这些症状伴随着复视。血液检查显示严重的甲亢。甲状腺闪烁扫描显示^99m Tc-pertechnatate弥散分布，并且在甲状腺右叶存在一个“冷”区域，超声检查证实。结节被诊断为低风险的不确定病变。

结论

我们介绍了一例 Graves 病伴活动性、中度至重度 Graves 眼病的病例，该患者患有预先存在的 AFTN，同时存在与阿仑单抗治疗后免疫重建相关的罕见的 Marine-Lenhart 综合征病例。