Victoria CAMPBELL, Rachel BECKETT, Noina ABİD, Susannah HOEY

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

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.

Tam Metin

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Kilcline C, Frieden IJ. Infantile hemangiomas: how common are they? A systematic review of the medical literature. Pediatr Dermatol 2008;25:168-173.

Drolet BA, Esterly NB, Frieden IJ. Hemangiomas in children. New Engl J Med 1999;341:173-181.

Chen XD, Ma G, Chen H, Ye XX, Jin YB, Lin XX. Maternal and perinatal risk factors for infantile hemangioma: A case-control study. Pediatr Dermatol 2013;30:457-461. Epub 2012 Dec 26

Christison-Lagay ER, Burrows PE, Alomari A, Dubois J, Kozakewich HP, Lane TS, Paltiel HJ, Klement G, Mulliken JB, Fishman SJ. Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry. J Pediatr Surg 2007;42:62-68.

Horii KA, Drolet BA, Frieden IJ, Baselga E, Chamlin SL, Haggstrom AN, Holland KE, Mancini AJ, McCuaig CC, Metry DW, Morel KD, Newell BD, Nopper AJ, Powell J, Garzon MC; Hemangioma Investigator Group. Prospective study of the frequency of hepatic hemangiomas in infants with multiple cutaneous infantile hemangiomas. Pediatr Dermatol 2011;28:245-253. Epub 2011 Apr 26

Yeh I, Bruckner A, Sanchez R, Jeng MR, Newell BD, Frieden IJ. Diffuse infantile hepatic hemangiomas: a report of four cases successfully managed with medical therapy. Pediatr Dermatol 2011;28:267-275. Epub 2011 Apr 26

Huang SA, Tu HM, Harney JW, Venihaki M, Butte AJ, Kozakewich HP, Fishman SJ, Larsen PR. Severe hypothyroidism caused by type 3 iodothyronine deiodinase in infantile hemangiomas. New Eng J Med 2000;343:185-189.

Cho YH, Taplin C, Mansour A, Howman-Giles R, Hardwick R, Lord D, Howard NJ. Case report: consumptive hypothyroidism consequent to multiple infantile hepatic hemangiomas. Curr Opin in Pediatr 2008;20:213-215.

Mouat F, Evans HM, Cutfield WS, Hofman PL, Jefferies C. Massive hepatic hemangioendothelioma and consumptive hypothyroidism. J Pediatr Endocrinol Metab 2008;21:701-703.

Bessho K, Etani Y, Ichimori H, Miyoshi Y, Namba N, Yoneda A, Ooue T, Chihara T, Morii E, Aoki T, Murakami M, Mushiake S, Ozono K. Increased type 3 iodothyronine deiodinase activity in a regrown hepatic hemangioma with consumptive hypothyroidism. Eur J Pediatr 2010;169:215-221. Epub 2009 Jun 23

Fisher DA. Clinical review 19: Management of congenital hypothyroidism. J Clin Endocr Metab 1991;72:523-529.

Tan ST, Itinteang T, Leadbitter P. Low-dose propranolol for multiple hepatic and cutaneous hemangiomas with deranged liver function. Pediatrics 2011;127:772-776. Epub 2011 Feb 28

Marsciani A, Pericoli R, Alaggio R, Brisigotti M, Vergine G. Letter to the editor: massive response of severe infantile hepatic hemangioma to propranolol. Pediatric Blood & Cancer 2010;54:176.

Mazereeuw-Hautier J, Hoeger P, Benlahrech S, Ammour A, Broue P, Vial J, Ohanessian G, Leaute-Labreze C, Labenne M, Vabres P, Rössler J, Bodemer C. Efficacy of propranolol in hepatic infantile hemangiomas with diffuse neonatal hemangiomatosis. J Paediatr 2010;157:340-342. Epub 2010 May 20

Mhanna A, Franklin WH, Mancini AJ. Hepatic infantile hemangiomas treated with oral propranolol -- a case series. Pediatr Dermatol 2011;28:39-45. Epub 2011 Jan 25.

Dickie B, Dasgupta R, Nair R, Alonso MH, Ryckman FC, Tiao GM, Adams DM, Azizkhan RG. Spectrum of hepatic hemangiomas: management and outcome. J Pediatr Surg 2009;44:125-133.

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

North PE, Waner M, Mizeracki A, Mrak RE, Nicholas R, Kincannon J, Suen JY, Mihm MC Jr. A unique microvascular phenotype shared by juvenile hemangiomas and human placenta. Arch Dermatol 2001;137: 559-570.

Emir S, Ekici F, Ikiz MA, Vidinlisan S. The association of consumptive hypothyroidism secondary to hepatic hemangioma and severe heart failure in infancy. Turk Pediatri Ars 2016;51:52-56.

Al Tasseh F, El-Khansa M, Abd O, Khalek AA, El-Rifai N. Diffuse hepatic hemangioma with single cutaneous hemangioma: an alerting occurrence. Clin Case Rep 2017;5:887-890.

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

Leaute-Labreze C, Dumas de la Roque E, Hubiche T, Boralevi F, Thambo JB, Taieb A. Propranolol for severe hemangiomas of infancy. New Eng J Med 2008;358:2649-2651.