Consumptive hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis: a case report

Abstract

IntroductionBenign neonatal hemangiomatosis is a condition in which multiple cutaneous hemangiomas appear at birth or shortly thereafter; visceral complications are absent. Here, we report a case of a consumption hypothyroidism in an Egyptian baby with benign neonatal hemangiomatosis.Case presentationAn 8-month-old Egyptian boy with benign neonatal hemangiomatosis was referred to our institution for evaluation of developmental delay. Initial examination revealed a quiet baby who was able to sit only with support. He had hypotonia, a large anterior fontanelle, puffy eyes, cold extremities, hypothermia, bradycardia, and abdominal distension. An examination of his skin revealed more than 100 dome-shaped red-purple cutaneous hemangiomas that varied in size from 5 to 10mm on the back, the abdomen and the extremities without mucus membrane involvement. He had low serum free thyroxine concentration and triiodothyronine levels and high thyroid-stimulating hormone and reverse-triiodothyronine levels. A work-up that involved appropriate imaging ruled out visceral involvement. Based on the above mentioned data, a diagnosis of consumptive hypothyroidism due to benign neonatal hemangiomatosis was made. He was started on oral thyroid medication which was gradually increased to 90μg L-thyroxine daily (15μg/kg/day). After three months of treatment, he was able to sit alone without support and he had normal levels of thyroid-stimulating hormone and serum free thyroxine.ConclusionThyroid function should be assessed periodically in babies with benign neonatal hemangiomatosis, especially if symptoms of hypothyroidism appear or the size and number of hemangiomatosis increase rapidly. Moreover, high doses of L-thyroxine may be needed to achieve euthyroidism during the infancy.

Consumptive Hypothyroidism: Case Report of Hepatic Hemangioendotheliomas Successfully Treated with Vincristine and Systematic Review of the Syndrome

“…The diagnosis of CHS requires a high suspicion index in patients with positive neonatal screening for hypothyroidism [6]. Of note, in our review, neonatal screening identified 32% (7/22) of CHS cases in children as congenital hypothyroidism.…”

“…Of note, in our review, neonatal screening identified 32% (7/22) of CHS cases in children as congenital hypothyroidism. Some characteristics can help to differentiate CHS from other forms of congenital hypothyroidism, namely, normal thyroid morphology and iodine incorporation, high levels of rT 3 and the requirement of high doses of LT 4 /LT 3 to restore serum TSH normal levels [6]. Notwithstanding, since hemangiomas are the most common vascular tumors in children, occurring in approximately 5–10% of 1-year-old children [7], it is reasonable to speculate that the actual prevalence of CHS may be underestimated.…”

Type 3 Deiodinase and Consumptive Hypothyroidism: A Common Mechanism for a Rare Disease

“…In fact, two cases of consumptive hypothyroidism associated with cutaneous hemangioma have been reported in infants (40). Moreover, a case of neonatal hemangioma in the skin, associated with consumptive hypothyroidism, has been reported in an 8-month-old infant (46). The first case of consumptive hypothyroidism secondary to a large parotid hemangioma was reported in 2012 in a child with congenital hypothyroidism and hypoplastic thyroid gland (47).…”