Resolution of Consumptive Hypothyroidism Secondary to Infantile Hepatic Hemangiomatosis with a Combination of Propranolol and Levothyroxine

Abstract

Infantile hepatic hemangiomas (IHH), particularly of the diffuse subtype can, in severe cases, be associated with hepatic and cardiac failure, compartment syndrome and consumptive hypothyroidism. Early recognition and treatment of these pathologies is paramount in order to minimise the risk of long-term sequelae.We report an interesting case of a female infant who presented with systemic compromise, in the absence of large or obvious cutaneous infantile hemangiomas. Imaging identified innumerable hepatic hemangiomas, consistent with diffuse infantile hepatic hemangiomatosis. Subsequent to this, thyroid function tests confirmed an associated but comparatively rare form of hypothyroidism, known as consumptive hypothyroidism. Following joint consultation with dermatology and endocrinology she was promptly treated with oral propranolol and levothyroxine, with subsequent improvement in her clinical parameters.This case reiterates the importance of aggressive investigation and management of consumptive hypothyroidism in any infant diagnosed with IHH, particularly when there is systemic compromise. We advocate propranolol as a single first line treatment for IHH, supported by thyroid replacement when appropriate.

Multiple infantile hepatic hemangiomas leading to consumptive hypothyroidism successfully treated with propranolol: A case report

“…[ 9 ] Propranolol has recently been introduced as the first line treatment of hemangiomas and its use has now been proven in various published articles at doses of 2-3.5 mg/kg/day. [ 11 12 13 ] Our patient was also treated with propranolol which resulted in resolution of hepatic hemangioma and ultimately corrected the thyroid dysfunction.…”

Adult diffuse hepatic hemangiomatosis

“…In infants, DHH is associated with a challenging form of consumptive hypothyroidism which requires prompt treatment with propranolol and an increased dose of L-thyroxine to prevent longterm sequelae. 24 The association of adult DHH and hypothyroidism in the index case could be fortuitous, although further study is required before concluding.…”

Multifocal Infantile Hepatic Hemangiomas Complicated by Consumptive Hypothyroidism: The Benefits of Early Diagnosis and Treatment

“…12 Currently propranolol is the first line of treatment in IHH with a favorable riskbenefit profile, whereas corticosteroids are reserved for refractory cases. 6,11 The present report describes a case of multifocal IHH associated with subcutaneous and lingual hemangiomas, complicated by consumptive hypothyroidism and successfully managed with oral propranolol and thyroid replacement therapy, without documented adverse effects.…”

“…3 From an historical point of view, systemic corticosteroids were the first line of treatment, despite significant lack of response (as high as 25%) and frequent side effects. 1,11 This perspective changed in 2008 with the discovery of beta-blockers as an effective treatment for infantile hemangiomas. 12 Currently propranolol is the first line of treatment in IHH with a favorable riskbenefit profile, whereas corticosteroids are reserved for refractory cases.…”