Top

Published in:

01-02-2010 | Original Paper

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

Authors: Kazuhiko Bessho, Yuri Etani, Hiroaki Ichimori, Yoko Miyoshi, Noriyuki Namba, Akihiro Yoneda, Takaharu Ooue, Tsuyoshi Chihara, Eiichi Morii, Tomoyuki Aoki, Masami Murakami, Sotaro Mushiake, Keiichi Ozono

Published in: European Journal of Pediatrics | Issue 2/2010

Abstract

Introduction

Infantile hepatic hemangioma with consumptive hypothyroidism is a rare condition.

Case report

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-α administration. At 20 months of age, 7 months after discontinuing prednisolone and interferon-α, 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.

Discussion

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.

Conclusion

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.

Achilleos OA, Buist LJ, Kelly DA et al (1996) Unresectable hepatic tumors in childhood and the role of liver transplantation. J Pediatr Surg 31:1563–1567CrossRefPubMed

Ayling RM, Davenport M, Hadzic N et al (2001) Hepatic hemangioendothelioma associated with production of humoral thyrotropin-like factor. J Pediatr 138:932–935CrossRefPubMed

Barnés CM, Huang S, Kaipainen A et al (2005) Evidence by molecular profiling for a placental origin of infantile hemangioma. Proc Natl Acad Sci U S A 102:19097–19102CrossRefPubMed

Cho YH, Taplin C, Mansour A et al (2008) Case report: consumptive hypothyroidism consequent to multiple infantile hepatic hemangiomas. Curr Opin Pediatr 20:213–215CrossRefPubMed

Christison-Lagay ER, Burrows PE, Alomari A et al (2007) Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry. J Pediatr Surg 42:62–68CrossRefPubMed

Corbella F, Arico M, Podesta AF et al (1983) Infantile hepatic hemangioendothelioma treated by radiotherapy. Pediatr Radiol 13:297–300CrossRefPubMed

Daller JA, Bueno J, Gutierrez J et al (1999) Hepatic hemangioendothelioma: clinical experience and management strategy. J Pediatr Surg 34:98–106CrossRefPubMed

Fergusson J, Tait N (2007) Resection for hepatic hemangiomas. J Gastroenterol Hepatol 22:1705CrossRefPubMed

Güven A, Aygun C, Ince H et al (2005) Severe hypothyroidism caused by hepatic hemangioendothelioma in an infant of a diabetic mother. Horm Res 63:86–89CrossRefPubMed

10.

Hasan Q, Tan ST, Gush J et al (2000) Steroid therapy of a proliferating hemangioma: histochemical and molecular changes. Pediatrics 105:117–120CrossRefPubMed

11.

Hashimoto M, Sugawara M, Ishiyama K et al (2008) Reduction in the size of a hepatic haemangioma after chemotherapy. Liver Int 28:1043–1044PubMed

12.

Herman TE, Siegel MJ (2001) Solitary hepatic hemangioendothelioma with extensive cystic necrosis and markedly elevated alpha-fetoprotein. J Perinatal 21:568–570CrossRef

13.

Huang SA, Tu HM, Harney JW et al (2000) Severe hypothyroidism caused by type 3 iodothyronine deiodinase in infantile hemangiomas. N Engl J Med 343:185–189CrossRefPubMed

14.

Kalpatthi R, Germak J, Mizelle K et al (2007) Thyroid abnormalities in infantile hepatic hemangioendothelioma. Pediatr Blood Cancer 49:1021–1024CrossRefPubMed

15.

Kassarjian A, Zurakowski D, Dubois J et al (2004) Infantile hepatic hemangiomas: clinical and imaging findings and their correlation with therapy. Am J Roentgenol 182:785–795

16.

Kellie S (2004) Serum markers in tumour diagnosis and treatment. In: Pinkerton R, Plauman P, Pieters R (eds) Paediatric oncology, 3rd edn. Arnold, London, pp 169–188

17.

Kester MH, Martinez de Mena R, Obregon MJ et al (2004) Iodothyronine levels in the human developing brain: major regulatory roles of iodothyronine deiodinases in different areas. J Clin Endocrinol Metab 89:3117–3128CrossRefPubMed

18.

Konrad D, Ellis G, Perlman K (2003) Spontaneous regression of severe acquired infantile hypothyroidism with multiple liver hemangiomas. Pediatrics 112:1424–1426CrossRefPubMed

19.

Lee TC, Barshes NR, Agee EE et al (2006) Resolution of medically resistant hypothyroidism after liver transplantation for hepatic hemangioendothelioma. J Pediatr Surg 41:1783–1785CrossRefPubMed

20.

Lehrnbecher T, Frauendienst-Egger G, Schrod L et al (1996) Hemangioendothelioma in a preterm infant associated with highly elevated alpha-fetoprotein. Eur J Pediatr 155:423–424CrossRefPubMed

21.

Mason KP, Koka BV, Eldredge EA et al (2001) Perioperative considerations in a hypothyroid infant with hepatic haemangioma. Pediatr Anaesth 11:228–232CrossRef

22.

Mo JQ, Dimashkieh HH, Bove KE (2004) GLUT1 endothelial reactivity distinguishes hepatic infantile hemangioma from congenital hepatic vascular malformation with associated capillary proliferation. Hum Pathol 35:200–209CrossRefPubMed

23.

Mouat F, Evans HM, Cutfield WS et al (2008) Massive hepatic hemangioendothelioma and consumptive hypothyroidism. J Pediatr Endocrinol Metab 21:701–703PubMed

24.

North PE, Waner M, Mizeracki A et al (2001) A unique microvascular phenotype shared by juvenile hemangiomas and human placenta. Arch Dermatol 137:559–570PubMed

25.

Sarı N, Yalçin B, Akyüz C et al (2006) Infantile hepatic hamangioendothelioma with elevated serum alpha-fetoprotein. Pediatr Hematol Oncol 23:639–647CrossRefPubMed

26.

Seo IS, Min KW, Mirkin LD (1988) Hepatic hemangioendothelioma of infancy associated with elevated alpha fetoprotein and catecholamine by-products. Pediatr Pathol 8:625–631CrossRefPubMed

27.

Touloukian RJ (1970) Hepatic hemangioendothelioma during infancy: pathology, diagnosis and treatment with prednisone. Pediatrics 45:1–6

28.

Tsuchida Y, Endo Y, Saito S et al (1978) Evaluation of alpha-fetoprotein in early infancy. J Pediatr Surg 13:155–162CrossRefPubMed

29.

Van der Geyten S, Buys N, Sanders JP et al (1999) Acute pretranslational regulation of type iii iodothyronine deiodinase by growth hormone and dexamethasone in chicken embryos. Mol Cell Endocrinol 147:49–56CrossRefPubMed

30.

Woltering MC, Robben S, Egeler RM (1997) Hepatic hemangioendothelioma of infancy: treatment with interferon alpha. J Pediatr Gastroenterol Nutr 24:348–351CrossRefPubMed

Title: Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism
Authors: Kazuhiko Bessho
Yuri Etani
Hiroaki Ichimori
Yoko Miyoshi
Noriyuki Namba
Akihiro Yoneda
Takaharu Ooue
Tsuyoshi Chihara
Eiichi Morii
Tomoyuki Aoki
Masami Murakami
Sotaro Mushiake
Keiichi Ozono
Publication date: 01-02-2010
Publisher: Springer-Verlag
Published in: European Journal of Pediatrics / Issue 2/2010
Print ISSN: 0340-6199
Electronic ISSN: 1432-1076
DOI: https://doi.org/10.1007/s00431-009-1009-x

At a glance: The STEP trials

Springer Medicine

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

Abstract

Introduction

Case report

Discussion

Conclusion

At a glance: The STEP trials

Springer Medicine

Abstract

Introduction

Case report

Discussion

Conclusion

Please log in to get access to this content

Other articles of this Issue 2/2010

Clinical practice

Programmed death-1 (PD-1) gene polymorphisms lodged in the genetic predispositions of Kawasaki Disease

Favourable long-term outcome after immediate treatment of neonatal hyperammonemia due to N-acetylglutamate synthase deficiency

A novel missense mutation in SUCLG1 associated with mitochondrial DNA depletion, encephalomyopathic form, with methylmalonic aciduria

Characteristics associated with maltreatment types in children referred to a hospital protection team

Surveillance study of apparent life-threatening events (ALTE) in the Netherlands