Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism
Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism
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DOI：
10.1007/s00431-009-1009-x
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发表时间：
2010-02-01
影响因子：
3.6
通讯作者：
Ozono, Keiichi
中科院分区：
文献类型：
--
作者：
Bessho, Kazuhiko;Etani, Yuri;Ozono, Keiichi
摘要一位四个月大的女婴，在接受先天性甲状腺功能减退症的治疗时，出现弥漫性的肝血管瘤。血清反三碘甲状腺原氨酸升高，她的甲状腺功能减退症改善伴随着消退的血管瘤后，泼尼松龙和干扰素α管理。在20个月大时，停用强的松龙和干扰素-α 7个月后，一个局灶性血管瘤从以前的病变之一重新生长，并手术切除。3型碘甲腺原氨酸脱碘酶（D3）的表达和活性升高切除肿瘤组织相比，胎盘。在这里，我们描述了一个病人的消耗性甲状腺功能减退症和弥漫性婴儿肝血管瘤，其中一个复发后复旧后，药物治疗。升高的D3活性的病因也discussed.It是重要的，以确定婴儿肝血管瘤的甲状腺功能减退症难治性激素替代治疗，谁有低游离三碘甲状腺原氨酸，尽管高促甲状腺激素和正常的游离甲状腺素水平，这些患者将需要长期随访。
Infantile hepatic hemangioma with consumptive hypothyroidism is a rare condition.A 4-month-old girl presented with diffuse hepatic hemangiomas during treatment of congenital hypothyroidism. Serum reverse triiodothyronine was elevated, and her hypothyroidism improved concomitant with involution of the hemangioma following prednisolone and interferon-alpha administration. At 20 months of age, 7 months after discontinuing prednisolone and interferon-alpha, a focal hemangioma regrew from one of the previous lesions and was surgically resected. The expression and activity of type 3 iodothyronine deiodinase (D3) were elevated in the resected tumor tissue compared with placenta.Here, we describe a patient with consumptive hypothyroidism and diffuse infantile hepatic hemangiomas, one of which regrew after involution following pharmacotherapy. The etiology of elevated D3 activity is also discussed.It is important to identify infantile hepatic hemangioma in patients with hypothyroidism refractory to hormone replacement therapy, who have low free triiodothyronine despite high thyrotropin and normal free thyroxine levels, and long-term follow-up will be needed for these patients.