A case of an infant with extremely low birth weight and hypothyroidism associated with massive cutaneous infantile hemangioma
-
Aiko Igarashi
,
Ikue Hata
Abstract
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.
Author contributions: All authors have accepted responsibility for the entire contents of this manuscript and have approved its submission.
Research funding: None declared.
Employment or leadership: None declared.
Honorarium: None declared.
Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; the writing of the report; or the decision to submit the report for publication.
References
1. Huang SA, Tu HM, Harney JW, Venihaki M, Butte AJ, et al. Severe hypothyroidism caused by type 3 iodothyronine deiodinase in infantile hemangiomas. N Engl J Med 2000;343:185–9.10.1056/NEJM200007203430305Search in Google Scholar PubMed
2. Kulungowski AM, Alomari AI, Chawla A, Christinson-Lagay ER, Fishman SJ. Lessons from a liver hemangioma registry: subtype classification. J Pediatr Surg 2012;47:165–70.10.1016/j.jpedsurg.2011.10.037Search in Google Scholar PubMed
3. Vigone MC, Cortinovis F, Rabbiosi S, Di Frenna M, Passoni A, et al. Difficult treatment of consumptive hypothyroidism in a child with massive parotid hemangioma. J Pediatr Endocrinol Metab 2012;25:153–5.10.1515/jpem.2011.438Search in Google Scholar PubMed
4. De Corti F, Crivellaro C, Zanon GF, Luzzatto C. Consumptive hypothyroidism associated with parotid infantile hemangioma. J Pediatr Endocrinol Metab 2015;28:467–9.10.1515/jpem-2013-0420Search in Google Scholar PubMed
5. Metwalley KA, Farghaly HS. Consumptive hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis: a case report. J Med Case Rep 2013;7:48.10.1186/1752-1947-7-48Search in Google Scholar PubMed PubMed Central
6. Chakraborty PP, Bera M, Patra S, Roy S. Consumptive hypothyroidism in solitary cutaneous haemangioma. BMJ Case Rep 2017;2017. pii: bcr-2017-221366.10.1136/bcr-2017-221366Search in Google Scholar PubMed PubMed Central
7. Pasa MW, Scheffel RS, Zanella AB, Maia AL, Dora JM. Consumptive hypothyroidism: case report of hepatic hemangioendotheliomas successfully treated with vincristine and systematic review of the syndrome. Eur Thyroid J 2017;6:321–7.10.1159/000481253Search in Google Scholar PubMed PubMed Central
8. Bessho K, Etani Y, Ichimori H, Miyoshi Y, Namba M, et al. Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism. Eur J Pediatr 2010;169:215–21.10.1007/s00431-009-1009-xSearch in Google Scholar PubMed
9. Lombard F, Dalla-Vale F, Veyrac C, Plan O, Cambonie G, et al. Severe hypothyroidism after contrast enema in premature infants. Eur J Pediatr 2009;168:499–500.10.1007/s00431-008-0782-2Search in Google Scholar PubMed
10. Pinsker JE, McBayne K, Edwards M, Jensen K, Crudo DF, et al. Transient hypothyroidism in premature infants after short-term topical iodine exposure: an avoidable risk? Pediatr Neonatol 2013;54:128–31.10.1016/j.pedneo.2012.10.005Search in Google Scholar PubMed
11. Higuchi S, Takagi M, Hasegawa Y. Use of liothyronine without levothyroxine in the treatment of mild consumptive hypothyroidism caused by hepatic hemangiomas. Endocr J 2017;64:639–43.10.1507/endocrj.EJ16-0559Search in Google Scholar PubMed
12. Polites SF, Rodrigue BB, Chute C, Hammill A, Dasgupta R. Propranolol versus steroids for the treatment of ulcerated infantile hemangiomas. Pediatr Blood Cancer 2018;65:e27280.10.1002/pbc.27280Search in Google Scholar PubMed
©2018 Walter de Gruyter GmbH, Berlin/Boston
Articles in the same Issue
- Frontmatter
- Original Articles
- Targeted next generation sequencing in patients with maturity-onset diabetes of the young (MODY)
- Assessment of dental caries and gingival status among a group of type 1 diabetes mellitus and healthy children of South India – a comparative study
- Early adiposity rebound in patients with Prader-Willi syndrome
- Arterial stiffness as a measure of cardiovascular risk in obese adolescents and adolescents with diabetes type 1
- Dysregulated glucose homeostasis in congenital central hypoventilation syndrome
- Do parents of children with metabolic diseases benefit from the Triple P – Positive Parenting Program? A pilot study
- Early higher dosage of alglucosidase alpha in classic Pompe disease
- Insulin resistance in children with familial hyperlipidemia
- Insulin resistance linked to subtle myocardial dysfunction in normotensive Turner syndrome young patients without structural heart diseases
- Acetylcholinesterase activity and bone biochemical markers in premature and full-term neonates
- Baseline characteristics of gender dysphoric youth
- Case Reports
- Multi-disciplinary evaluation of a 5-month-old with hypertrophic cardiomyopathy related to a functional adrenocortical tumor
- A case of an infant with extremely low birth weight and hypothyroidism associated with massive cutaneous infantile hemangioma
- Possible hints and pitfalls in diagnosing Peutz-Jeghers syndrome
Articles in the same Issue
- Frontmatter
- Original Articles
- Targeted next generation sequencing in patients with maturity-onset diabetes of the young (MODY)
- Assessment of dental caries and gingival status among a group of type 1 diabetes mellitus and healthy children of South India – a comparative study
- Early adiposity rebound in patients with Prader-Willi syndrome
- Arterial stiffness as a measure of cardiovascular risk in obese adolescents and adolescents with diabetes type 1
- Dysregulated glucose homeostasis in congenital central hypoventilation syndrome
- Do parents of children with metabolic diseases benefit from the Triple P – Positive Parenting Program? A pilot study
- Early higher dosage of alglucosidase alpha in classic Pompe disease
- Insulin resistance in children with familial hyperlipidemia
- Insulin resistance linked to subtle myocardial dysfunction in normotensive Turner syndrome young patients without structural heart diseases
- Acetylcholinesterase activity and bone biochemical markers in premature and full-term neonates
- Baseline characteristics of gender dysphoric youth
- Case Reports
- Multi-disciplinary evaluation of a 5-month-old with hypertrophic cardiomyopathy related to a functional adrenocortical tumor
- A case of an infant with extremely low birth weight and hypothyroidism associated with massive cutaneous infantile hemangioma
- Possible hints and pitfalls in diagnosing Peutz-Jeghers syndrome
