Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism.
BACKGROUND
Infantile hepatic hemangioma with consumptive hypothyroidism is a rare condition.


METHODS
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.


CONCLUSIONS
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.


CONCLUSIONS
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.
Bessho
Kazuhiko
K
Department of Pediatrics, Osaka University Graduate School of Medicine, 2-2 Yamadaoka, Suita, Osaka 565-0871, Japan.
Etani
Yuri
Y
Ichimori
Hiroaki
H
Miyoshi
Yoko
Y
Namba
Noriyuki
N
Yoneda
Akihiro
A
Ooue
Takaharu
T
Chihara
Tsuyoshi
T
Morii
Eiichi
E
Aoki
Tomoyuki
T
Murakami
Masami
M
Mushiake
Sotaro
S
Ozono
Keiichi
K
D014408:Biomarkers, Tumor / Q000235:genetics / Q000378:metabolism; D003937:Diagnosis, Differential; D005260:Female; D005500:Follow-Up Studies; D015972:Gene Expression Regulation, Neoplastic; D006391:Hemangioma / Q000150:complications / Q000175:diagnosis / Q000201:enzymology*; D006801:Humans; D007037:Hypothyroidism / Q000150:complications* / Q000201:enzymology; D007223:Infant; D007453:Iodide Peroxidase / Q000235:genetics / Q000378:metabolism*; D008099:Liver / Q000378:metabolism / Q000473:pathology; D008113:Liver Neoplasms / Q000150:complications / Q000175:diagnosis / Q000201:enzymology*; D008279:Magnetic Resonance Imaging; D016133:Polymerase Chain Reaction; D012334:RNA, Neoplasm / Q000235:genetics; D013997:Time Factors
D014408:Biomarkers, Tumor; D012334:RNA, Neoplasm; C117274:iodothyronine deiodinase type III; D007453:Iodide Peroxidase