Consumptive Hypothyroidism due to Hepatic Hemangiomas: A Case Series and Review of the Literature

Author:

Siano Maria Anna1,Ametrano Orsola2,Barbato Filomena2,Sammarco Elena2,Ranucci Giusy3,Pietrobattista Andrea4,Rossomando Alessia1,Mandato Claudia13

Affiliation:

1. Department of Medicine, Surgery and Dentistry “Scuola Medica Salernitana”, Università di Salerno, Baronissi (Salerno), Italy

2. Department of Pediatric Dermatology, Santobono-Pausilipon Children Hospital, Naples, Italy

3. Department of Pediatrics, Santobono-Pausilipon Children Hospital, Naples, Italy

4. Liver Unit, Liver Transplant Program, Bambino Gesù Children Hospital, Rome, Italy.

Abstract

Introduction: Consumptive hypothyroidism (CH) is a rare and potentially overlooked complication of hepatic hemangiomas (HH) overexpressing the enzyme deiodinase, which converts thyroxine (T4) to reverse triiodothyronine (rT3). Materials and methods: Here, we report a case series of 3 patients and a systematic review of the literature. Results: Hypothyroidism (mean serum TSH 52.03 mIU/L) was detected at a mean age of 4.6 months (range 3–6) in 3 infants with infantile hepatic hemangiomas, treated with thyroxine (mean dose 12 µg/kg/day). All received treatment with propranolol (1–3 mg/kg/day) from the mean age of 4 months. Hormonal treatment was stopped at a mean age of 20 months (range 12–30). Hypothyroidism reoccurred in a patient concurrently with the increase of liver lesions, requiring liver transplantation (LT) at age 39 months. Literature review retrieved 42 studies (48 patients): HH (n = 43) were isolated in 24 infants and associated with cutaneous hemangiomas in 19. Hemangiomas were only cutaneous in 5. In the first 43 patients, hypothyroidism was detected at a mean age of 1 month; 21 of 43 patients were prescribed propranolol alone (n = 8) or associated with other medicaments (n = 13); 2 of 43 patients underwent LT. Hormonal treatment consisted of T4 in 35 of 43 patients and T3 in 10. CH associated with only cutaneous and extrahepatic visceral hemangiomas (n = 5), detected at a mean age of 7 months (TSH mean levels at diagnosis of 150.3 mIU/L). Three of 5 patients received treatment with propranolol ± other medicaments. All 5 patients were treated with T4. Conclusions: Periodical thyroid function assessment is necessary in patients with hepatic hemangiomas, particularly when lesions’ size and number increase rapidly.

Publisher

Wiley

Subject

General Earth and Planetary Sciences,General Environmental Science

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