Igarashi Aiko, Hata Ikue, Yuasa Miori, Okuno Takashi, Ohshima Yusei
Department of Pediatrics, Faculty of Medical Sciences, University of Fukui, Matsuoka, Eiheiji-cho, Yoshida-gun, Fukui, Japan.
J Pediatr Endocrinol Metab. 2018 Dec 19;31(12):1377-1380. doi: 10.1515/jpem-2018-0369.
Background Although hepatic infantile hemangioma (IH) may correlate with consumptive hypothyroidism consequent to the overexpression of thyroid hormone inactivating enzyme by hemangioma cells, hypothyroidism has been rarely recognized in infants with cutaneous hemangioma. Case presentation A male infant born at 28 weeks of gestational age with an extremely low birth weight (775 g) developed a massive cutaneous hemangioma on his neck and severe abdominal distension. Imaging examinations detected a small mass lesion in the brain but no hepatic hemangioma. Laboratory findings at the age of 26 days revealed hypothyroidism. Although high-dose levothyroxine therapy failed to normalize the thyroid function, hypothyroidism improved and cutaneous hemangioma regressed after initiating propranolol therapy. Conclusions Our findings suggest that consumptive hypothyroidism should be considered as a critical comorbidity in patients with massive cutaneous IH. Propranolol therapy can effectively normalize thyroid function and cause hemangioma regression.
尽管肝脏婴儿血管瘤（IH）可能与血管瘤细胞中甲状腺激素失活酶过度表达导致的消耗性甲状腺功能减退相关，但皮肤血管瘤婴儿中甲状腺功能减退很少被认识到。病例报告：一名孕28周出生、极低出生体重（775克）的男婴颈部出现巨大皮肤血管瘤并伴有严重腹胀。影像学检查在脑部发现一个小肿块病变，但未发现肝脏血管瘤。26日龄时的实验室检查结果显示甲状腺功能减退。尽管高剂量左甲状腺素治疗未能使甲状腺功能正常化，但在开始普萘洛尔治疗后，甲状腺功能减退得到改善且皮肤血管瘤消退。结论：我们的研究结果表明，消耗性甲状腺功能减退应被视为巨大皮肤IH患者的一种关键合并症。普萘洛尔治疗可有效使甲状腺功能正常化并导致血管瘤消退。
