Hepatic hemangiomas in children: potential risks and principles of treatment

Authors

  • I. M. Benzar Bogomolets National Medical University, Kyiv, Ukraine, Ukraine https://orcid.org/0000-0001-7637-7769
  • D. V. Zhumick National children’s specialized hospital «OKHMATDYT», Kyiv, Ukraine, Ukraine

DOI:

https://doi.org/10.15574/PS.2019.62.31

Keywords:

hepatic hemangiomas, infantile hemangiomas, hypothyroidism, propranolol

Abstract

Purpose. Clinical manifestations of hepatic hemangiomas (HH) range from asymptomatic lesions to congestive heart failure associated, hepatic failure, hypothyroidism, and death.

The aim of study is to review our experience in the management of HH in children.

Methods. Fifteen patients during period 2011–2019 were diagnosed with HH as follows: 2 focal, 9 multiple and 4 diffuse lesions. All patients were screened for heart failure and hypothyroidism. The median age at diagnosis was 52.3±43.3 days. To clarify the diagnosis, MRI (n=5), CT (n=10), liver biopsy with Glut1 protein (n=1) performed.

Result. Heart failure complicated the course of HH in 3 (20.0%), hepatic immaturity in 5 (33.3%), and hypothyroidism in three (20.0%) patients. Treatment with propranolol was performed in 12 patients 4 with diffuse HH and 8 with multifocal HH), duration of therapy was 6 to 24, on average 14.9±4.3 months. Broncho-obstructive syndrome, with the need to discontinue treatment, occurs in one case (8.3%). One child with multifocal HH died of progressive heart failure, the mortality rate is 6.7%. In 8 children with multifocal HH treated with propranolol, the involution occurred at the age of 6–18 months. In children with diffuse HH there were residual lesions in diameter up to 2 cm (n=2). Children with hypothyroidism were substitution therapy with L-thyroxine. Treatment with focal HHs has not been performed, and their involution has been observed for 9 and 15 months.

Conclusion. Hepatic hemangiomas (HH) represent a diverse spectrum of clinical presentations and high potential of severe complication. Early treatment with beta-blockers of symptomatic HH and careful monitoring of asymptomatic patients can prevent complications and reduce mortality.

References

Benzar IО, Polulyakh OK. (2014). The evacuation of clinical activity and efficiency of treatment the infantile hemangioma with the of ultrasound scanning. Neonatology surgery and perinatal medicine. 3 (13);IV:86–91.. 3 (13); IV:86–91.

Pogoriliy VV, Kotenko OG. Konoplytsky VS, Dmytriiev DV, Kalinchuk AA, Konoplytsky DV. (2015). Own experience of the follow-up of pediatric patients with vascular tumors of the liver. Modern gastroenterology. 1 (81):91–95.

Bosemani T, Puttgen KB, Huisman TA et al. (2012). Multifocal infantile hepatic haemangiomas-imaging strategy and response to treatment after Propranolol and steroids including review of the literature. European journal of pediatrics. 171(7):1023–1028. https://doi.org/10.1007/s00431-011-1671-7; PMid:22234480

Christison-Lagay ER, Burrows PE, Alomari A, Dubois J, Kozakewich HP, Lane TS, Paltiel HJ, Klement G, Mulliken JB, Fishman SJ. (2007). Hepatic hemangiomas: subtype classification and development of a clinical practice algorithm and registry. J Pediatr Surg. 42(1):62–67. https://doi.org/10.1016/j.jpedsurg.2006.09.041; PMid:17208542

Dickie B, Dasgupta R, Nair R, Alonso MH, Ryckman FC, Tiao GM, Adams DM, Azizkhan RG. (2009). Spectrum of hepatic hemangiomas: management and outcome. J Pediatr Surg. 44(1):125–33. https://doi.org/10.1016/j.jpedsurg.2008.10.021; PMid:19159729

Gnarra M, Behr G, Kitajewski A, Wu JK, Anupindi SA, Shawber CJ, Zavras N, Schizas D, Salakos C, Economopoulos KP. (2016). History of the infantile hepatic hemangioma: From imaging to generating a differential diagnosis. World J Clin Pediatr.8;5(3):273–280. https://doi.org/10.5409/wjcp.v5.i3.273; PMid:27610342 PMCid:PMC4978619

Horii KA, Drolet BA, Frieden IJ et al. (2011). Prospective study of the frequency of hepatic haemangiomas in infants with multiple cutaneous infantile haemangiomas. Pediatric dermatology. 28(3):245–253. https://doi.org/10.1111/j.1525-1470.2011.01420.x; PMid:21517952

Huang SA, Tu HM, Harney JW, Venihaki M, Butte AJ, Kozakewich HPW, Fishman SJ, Larsen PR. (2000). Severe hypothyroidism caused by type 3 iodothyronine deiodinase in infantile hemangiomas. N Engl J Med. 343(3):185–189. https://doi.org/10.1056/NEJM200007203430305; PMid:10900278

Iacobas I, Phung TL, Adams DM, Trenor CC 3rd, Blei F, Fishman DS, Hammill A, Masand PM, Fishman SJ. (2018). Guidance Document for Hepatic Hemangioma (Infantile and Congenital) Evaluation and Monitoring. J Pediatr. 203:294–300. https://doi.org/10.1016/j.jpeds.2018.08.012; PMid:30244993

Kassarjian A, Zurakowski D, Dubois J, Paltiel HJ, Fishman SJ, Burrows PE. (2004). Infantile hepatic hemangiomas: clinical and imaging findings and their correlation with therapy. AJR Am J Roentgenol. 182(3):785–95. https://doi.org/10.2214/ajr.182.3.1820785; PMid:14975986

Konrad D, Ellis G, Perlman K. (2003). Spontaneous regression of severe acquired infantile hypothyroidism associated with multiple liver hemangiomas. Pediatrics. 112 6 Pt 1):1424–6. https://doi.org/10.1542/peds.112.6.1424; PMid:14654623

Kulungowskia AM, Alomarib AI, Chawlaa A, Christison-Lagaya ER, Fishman SJ. (2012). Lessons from a liver hemangioma registry: subtype classification. J of Ped Surg. 47(1):165–70. https://doi.org/10.1016/j.jpedsurg.2011.10.037; PMid:22244411

Kuroda T, Kumagai M, Nosaka S, Nakazawa A, Takimoto T, Hoshino K. (2011). Critical infantile hepatic hemangioma: results of a nationwide survey by the Japanese Infantile Hepatic Hemangioma Study Group. Journal of Pediatric Surgery. 46(12): 2239–2243. https://doi.org/10.1016/j.jpedsurg.2011.09.007; PMid:22152857

Leaute-Labreze C, Hoeger P, Mazereeuw-Hautier J et al. (2015). A randomized, controlled trial of oral Propranolol in infantile hemangioma. N Engl J Med. 372(8):735–746.

Li K, Wang Z, Liu Y, Yao W, Gong Y, Xiao X. (2016). Fine clinical differences between patients with multifocal and diffuse hepatic hemangiomas. J Pediatr Surg. 51(12):2086–2090. https://doi.org/10.1016/j.jpedsurg.2016.09.045; PMid:27686480

Meyers RL. (20017). Tumors of the liver in children. Surg Oncol. 16(3):195–203. https://doi.org/10.1016/j.suronc.2007.07.002; PMid:17714939

Mo JQ, Dimashkieh HH, Bove KE. (2004). GLUT1 endothelial reactivity distinguishes hepatic infantile hemangioma from congenital hepatic vascular malformation with associated capillary proliferation. Hum Pathol. 35(2):200–209. https://doi.org/10.1016/j.humpath.2003.09.017; PMid:14991538

North PE, Waner M, Mizeracki A, Mihm MC. (2000). GLUT1: a newly discovered immunohistochemical marker for juvenile hemangiomas. Hum Pathol. 31(1):11–22. https://doi.org/10.1016/S0046-8177(00)80192-6

Rialon KL, Murillo R, Fevurly RD, Kulungowski AM, Christison-Lagay ER, Zurakowski D, Kozakewich HP, Alomari AI, Fishman SJ. (2015). Risk factors for mortality in patients with multifocal and diffuse hepatic hemangiomas. J Pediatr Surg. 50(5):837–841. https://doi.org/10.1016/j.jpedsurg.2014.09.056; PMid:25783331

Vergine G, Marsciani A, Pedini A et al. (2012). Efficacy of Propranolol treatment in thyroid dysfunction associated with severe infantile hepatic hemangioma. Horm Res Paediatr. 78(4):256–260. https://doi.org/10.1159/000337253; PMid:22907522

Vredenborg AD, Janmohamed SR, de Laat PC et al. (2013). Multiple cutaneous infantile haemangiomas and the risk of internal haemangioma. The British journal of dermatology. 169(1):188–191. https://doi.org/10.1111/bjd.12229; PMid:23421718

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Section

Original articles. Thoracic and abdominal surgery