Fox Katriona, Fitzsimons Aisling, Sharif Farhana, Lee Graham Robert, O'Grady Michael Joseph
Department of Paediatrics, Regional Hospital Mullingar, Co. Westmeath, Ireland.
Department of Paediatrics, Royal College of Surgeons in Ireland, Dublin, Ireland.
Endocrinol Diabetes Metab Case Rep. 2023 Aug 16;2023(3). doi: 10.1530/EDM-23-0064. Print 2023 Aug 1.
Rare patients who have both thyroid-stimulating hormone (TSH) receptor-stimulating and -blocking antibodies can develop 'pendulum swinging' thyroid dysfunction. A 9-year-old girl with Down syndrome was treated with carbimazole for Graves' disease. After 2 years of treatment, she became profoundly biochemically hypothyroid, and this persisted after carbimazole was discontinued. Low-dose L-thyroxine was commenced. This was subsequently also discontinued as biochemical hyperthyroidism developed. TSH receptor antibody bioassay identified both TSH receptor-stimulating and -blocking antibodies. Mild hyperthyroidism persisted and while consultations regarding definitive treatment were ongoing, medication was not recommenced. Thyroid function normalised spontaneously and she has remained euthyroid for the past 3 years. Previous reports have advised definitive treatment; however, our patient developed spontaneous remission which has been prolonged and definitive therapies have been avoided. It is not yet known how commonly this particular phenomenon occurs.
Rare patients who have both TSH receptor-stimulating and -blocking antibodies can switch between hyperthyroidism and hypothyroidism or vice versa during treatment with antithyroid drugs or thyroxine. Metamorphic thyroid autoimmunity is more common in Down syndrome. Switching between hyperthyroidism and hypothyroidism and back again is less commonly reported. Definitive treatment such as radioactive iodine or thyroidectomy are usually recommended. Prolonged remission was achieved off all medication, without recourse to definitive treatments.
罕见的同时具有促甲状腺激素（TSH）受体刺激抗体和阻断抗体的患者可出现“钟摆样”甲状腺功能障碍。一名9岁唐氏综合征女孩因格雷夫斯病接受卡比马唑治疗。治疗2年后，她出现严重的生化性甲状腺功能减退，停用卡比马唑后仍持续存在。开始使用低剂量左甲状腺素。随后，由于出现生化性甲状腺功能亢进，左甲状腺素也停用。TSH受体抗体生物测定发现了TSH受体刺激抗体和阻断抗体。轻度甲状腺功能亢进持续存在，在进行关于确定性治疗的会诊期间，未重新开始用药。甲状腺功能自发恢复正常，在过去3年中她一直保持甲状腺功能正常。既往报告建议进行确定性治疗；然而，我们的患者出现了自发缓解，且缓解期延长，避免了进行确定性治疗。目前尚不清楚这种特殊现象的常见程度。
罕见的同时具有TSH受体刺激抗体和阻断抗体的患者在使用抗甲状腺药物或甲状腺素治疗期间可在甲状腺功能亢进和甲状腺功能减退之间转换，反之亦然。变形性甲状腺自身免疫在唐氏综合征中更常见。甲状腺功能亢进和甲状腺功能减退之间的转换较少被报道。通常建议进行放射性碘或甲状腺切除术等确定性治疗。在未使用所有药物的情况下实现了长期缓解，无需进行确定性治疗。
