Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism

被引：35

作者：

Bessho, Kazuhiko ^{[1
]}

Etani, Yuri ^{[1
]}

Ichimori, Hiroaki ^{[1
]}

Miyoshi, Yoko ^{[1
]}

Namba, Noriyuki ^{[1
]}

Yoneda, Akihiro ^{[2
]}

Ooue, Takaharu ^{[2
]}

Chihara, Tsuyoshi ^{[3
]}

Morii, Eiichi ^{[3
]}

Aoki, Tomoyuki ^{[4
]}

Murakami, Masami ^{[4
]}

Mushiake, Sotaro ^{[1
]}

Ozono, Keiichi ^{[1
]}

机构：

[1] Osaka Univ, Grad Sch Med, Dept Pediat, Suita, Osaka 5650871, Japan

[2] Osaka Univ, Grad Sch Med, Dept Pediat Surg, Suita, Osaka 5650871, Japan

[3] Osaka Univ, Grad Sch Med, Dept Pathol, Suita, Osaka 5650871, Japan

[4] Gunma Univ, Grad Sch Med, Dept Clin Lab Med, Maebashi, Gunma 371, Japan

来源：

EUROPEAN JOURNAL OF PEDIATRICS | 2010年 / 169卷 / 02期

关键词：

Diffuse hepatic hemangioma; Consumptive hypothyroidism; Type 3 iodothyronine deiodinase; Regrow; alpha-fetoprotein; HEMANGIOENDOTHELIOMA; INFANT;

D O I：

10.1007/s00431-009-1009-x

中图分类号：

R72 [儿科学];

学科分类号：

100202 ;

摘要：

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.

引用

页码：215 / 221

页数：7

共 30 条

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