Osada Asami, Araki Eriko, Yamashita Yukio, Ishii Tomohiro
Department of Pediatrics, Yokohama Municipal Citizen's Hospital, Yokohama, Japan.
Department of Pediatrics, Keio University School of Medicine, Tokyo, Japan.
Clin Pediatr Endocrinol. 2019;28(1):9-14. doi: 10.1297/cpe.28.9. Epub 2019 Jan 31.
Infantile hepatic hemangioma (IHH) can be accompanied by consumptive hypothyroidism. We report the case of a 4-mo-old boy who showed massive hepatomegaly, peripheral coldness, lethargy, and failure to thrive. An enhanced computed tomography scans demonstrated multiple hemangiomas in both lobes of the liver, and a thyroid function tests showed severe hypothyroidism: TSH 561.5 µIU/mL, free triiodothyronine (fT) 1.0 pg/mL, and free thyroxine (fT) < 0.7 ng/dL. IHH gradually regressed following propranolol treatment and fT increased to a low normal level (1.0 ng/dL) by high dose replacement of levothyroxine, while fT remained very low (< 1.0 pg/mL), even following high doses of levothyroxine; fT eventually normalized following the administration of liothyronine. We suggest that treatment strategies should be individualized based on thyroid function, and that the combination therapy of propranolol for anti-tumor treatment and levothyroxine and liothyronine for respective thyroid hormone replacement is effective, particularly in cases of severe consumptive hypothyroidism due to multiple IHHs.
婴儿肝血管瘤（IHH）可伴有消耗性甲状腺功能减退。我们报告一例4个月大男婴，表现为肝脏肿大、外周发冷、嗜睡及发育不良。增强计算机断层扫描显示肝脏两叶有多个血管瘤，甲状腺功能检查显示严重甲状腺功能减退：促甲状腺激素（TSH）561.5 μIU/mL，游离三碘甲状腺原氨酸（fT3）1.0 pg/mL，游离甲状腺素（fT4）<0.7 ng/dL。普萘洛尔治疗后IHH逐渐消退，高剂量左甲状腺素替代治疗使fT4升至低正常水平（1.0 ng/dL），但即使高剂量左甲状腺素治疗后fT3仍很低（<1.0 pg/mL）；使用碘塞罗宁后fT3最终恢复正常。我们建议治疗策略应根据甲状腺功能个体化制定，普萘洛尔抗肿瘤治疗联合左甲状腺素和碘塞罗宁分别进行甲状腺激素替代治疗是有效的，尤其适用于因多发性IHH导致的严重消耗性甲状腺功能减退病例。
