Clinical and Experimental Pediatrics

대한소아청소년과학회 (The Korean Pediatric Society)
권호 표지
DOI QR코드

DOI QR Code

Clinical features of infantile hepatic hemangioendothelioma

  • Kim, Eun-Hee (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Koh, Kyung-Nam (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Park, Mee-Rim (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Kim, Bo-Eun (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Im, Ho-Joon (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine) ;
  • Seo, Jong-Jin (Department of Pediatrics, Asan Medical Center, University of Ulsan College of Medicine)
  • 투고 : 2010.12.13
  • 심사 : 2011.04.05
  • 발행 : 2011.06.15
PDF 다운로드
⟨ 이전 논문 다음 논문 ⟩

초록

Purpose: Infantile hepatic hemangioendothelioma (IHHE) is the most common type of hepatic vascular tumor in infancy. We conducted this study to review our clinical experience of patients with IHHE and to suggest management strategies. Methods: We retrospectively analyzed the medical records of 23 IHHE patients (10 males, 13 females) treated at the Asan Medical Center between 1996 and 2009. Results: Median age at diagnosis was 38 days (range, 1 to 381 days). Seven patients (30%) were diagnosed with IHHE based on sonographically detected fetal liver masses, 5 (22%) were diagnosed incidentally in the absence of symptoms, 5 (22%) had congestive heart failure, 3 (13%) had skin hemangiomas, 2 (9%) had abnormal liver function tests, and 1 (4%) had hepatomegaly. All diagnoses were based on imaging results, and were confirmed in three patients by histopathology analysis. Six patients were observed without receiving any treatment, whereas 12 received corticosteroids and/or interferonalpha. One patient with congestive heart failure and a resectable unilobar tumor underwent surgical resection. Three patients with congestive heart failure and unresectable tumors were managed by hepatic artery embolization with/without medical treatment. At a median follow-up of 29 months (range, 1 to 156 months), 21 (91%) patients showed complete tumor disappearance or >50% decrease in tumor size. One patient died due to tumor-related causes. Conclusion: IHHE generally has a benign clinical course with low morbidity and mortality rates. Clinical course and treatment outcome did not differ significantly between medically treated and non-treated groups. Surgically unresectable patients with significant symptoms may be treated medically or with hepatic artery embolization.

키워드

참고문헌

  1. Emre S, McKenna GJ. Liver tumors in children. Pediatr Transplant 2004;8:632-8. https://doi.org/10.1111/j.1399-3046.2004.00268.x
  2. Shafford EA, Pritchard J. Liver tumors. In: Pinkerton CR, Plowman PN, Pieters R, editors. Paediatric oncology. 3rd ed. London: Arnold Publishers, 2004:448-68.
  3. Daller JA, Bueno J, Gutierrez J, Dvorchik I, Towbin RB, Dickman PS, et al. Hepatic hemangioendothelioma: clinical experience and management strategy. J Pediatr Surg 1999;34:98-105. https://doi.org/10.1016/S0022-3468(99)90237-3
  4. Chen CC, Kong MS, Yang CP, Hung IJ. Hepatic hemangioendothelioma in children: analysis of thirteen cases. Acta Paediatr Taiwan 2003;44:8- 13.
  5. Moon SB, Kwon HJ, Park KW, Yun WJ, Jung SE. Clinical experience with infantile hepatic hemangioendothelioma. World J Surg 2009;33:597- 602. https://doi.org/10.1007/s00268-008-9882-4
  6. Ganguly R, Mukherjee A. Infantile hemangioendothelioma: a case report and discussion. Pathol Res Pract 2010;206:53-8. https://doi.org/10.1016/j.prp.2009.01.017
  7. Sevinir B, Ozkan TB. Infantile hepatic hemangioendothelioma: clinical presentation and treatment. Turk J Gastroenterol 2007;18:182-7.
  8. Davenport M, Hansen L, Heaton ND, Howard ER. Hemangioendothelioma of the liver in infants. J Pediatr Surg 1995;30:44-8. https://doi.org/10.1016/0022-3468(95)90607-X
  9. Amonkar P, Desai S, Deb R, Kane S, Kurkure P, Deshpande RK, et al. Infantile hemangioendothelioma of the liver. Med Pediatr Oncol 1999;32:392-4. https://doi.org/10.1002/(SICI)1096-911X(199905)32:5<392::AID-MPO17>3.0.CO;2-I
  10. Lu CC, Ko SF, Liang CD, Kuo HW, Tiao MM. Infantile hepatic hemangioendothelioma presenting as early heart failure: report of two cases. Chang Gung Med J 2002;25:405-10.
  11. Riley MR, Garcia MG, Cox KL, Berquist WE, Kerner JA Jr. Hepatic infantile hemangioendothelioma with unusual manifestations. J Pediatr Gastroenterol Nutr 2006;42:109-13. https://doi.org/10.1097/01.mpg.0000174330.51595.1b
  12. Selby DM, Stocker JT, Waclawiw MA, Hitchcock CL, Ishak KG. Infantile hemangioendothelioma of the liver. Hepatology 1994;20(1 Pt 1):39-45.
  13. Robbins RC, Chin C, Yun KL, Berry GJ, Bernstein D, Reitz BA. Arterial switch and resection of hepatic hemangioendothelioma. Ann Thorac Surg 1995;59:1575-7. https://doi.org/10.1016/0003-4975(94)00971-9
  14. Ito H, Yamasaki T, Okamoto O, Tahara E. Infantile hemangioendothelioma of the liver in patient with interstitial deletion of chromosome 6q: report of an autopsy case. Am J Med Genet 1989;34:325-9. https://doi.org/10.1002/ajmg.1320340306
  15. Shah KD, Beck AR, Jhaveri MK, Keohane M, Weinberg B, Gerber MA. Infantile hemangioendothelioma of heterotopic intrathoracic liver associated with diaphragmatic hernia. Hum Pathol 1987;18:754-6. https://doi.org/10.1016/S0046-8177(87)80250-2
  16. Kalpatthi R, Germak J, Mizelle K, Yeager N. Thyroid abnormalities in infantile hepatic hemangioendothelioma. Pediatr Blood Cancer 2007;49:1021-4. https://doi.org/10.1002/pbc.20801
  17. Ng WH, Ching AS, Chan KF, Fung WT. Clinics in diagnostic imaging (89). Infantile hepatosplenic haemangioendotheliomas. Singapore Med J 2003;44:491-5.
  18. Lucaya J, Enriquez G, Amat L, Gonzalez-Rivero MA. Computed tomography of infantile hepatic hemangioendothelioma. AJR Am J Roentgenol 1985;144:821-6. https://doi.org/10.2214/ajr.144.4.821
  19. Halefoğlu AM. Magnetic resonance imaging of infantile hemangioendothelioma. Turk J Pediatr 2007;49:77-81.
  20. Konez O, Burrows PE. An appropriate diagnostic workup for suspected vascular birthmarks. Cleve Clin J Med 2004;71:505-10. https://doi.org/10.3949/ccjm.71.6.505
  21. Sari N, Yalçin B, Akyüz C, Haliloglu M, Büyükpamukçu M. Infantile hepatic hemangioendothelioma with elevated serum alpha-fetoprotein. Pediatr Hematol Oncol 2006;23:639-47. https://doi.org/10.1080/08880010600954397
  22. Seo IS, Min KW, Mirkin LD. Hepatic hemangioendothelioma of infancy associated with elevated alpha fetoprotein and catecholamine by-products. Pediatr Pathol 1988;8:625-31. https://doi.org/10.3109/15513818809022319
  23. Kim TJ, Lee YS, Song YS, Park CK, Shim SI, Kang CS, et al. Infantile hemangioendothelioma with elevated serum alpha fetoprotein: report of 2 cases with immunohistochemical analysis. Hum Pathol 2010;41:763-7. https://doi.org/10.1016/j.humpath.2009.05.019
  24. Bachmann R, Genin B, Bugmann P, Belli D, Hanquinet S, Liniger P, et al. Selective hepatic artery ligation for hepatic haemangioendothelioma:case report and review of the literature. Eur J Pediatr Surg 2003;13:280-4. https://doi.org/10.1055/s-2003-42245
  25. Park EA, Seo JW, Lee SW, Choi HY, Lee SJ. Infantile hemangioendothelioma treated with high dose methylprednisolone pulse therapy. J Korean Med Sci 2001;16:127-9. https://doi.org/10.3346/jkms.2001.16.1.127
  26. Samuel M, Spitz L. Infantile hepatic hemangioendothelioma: the role of surgery. J Pediatr Surg 1995;30:1425-9. https://doi.org/10.1016/0022-3468(95)90397-6
  27. Shamaly H, Abu-Nassar Z, Groisman GM, Shamir R. Hepatic hemangloendothelioma: the need for early diagnosis and resection. Isr Med Assoc J 2006;8:585-6.
  28. Woltering MC, Robben S, Egeler RM. Hepatic hemangioendothelioma of infancy: treatment with interferon alpha. J Pediatr Gastroenterol Nutr 1997;24:348-51. https://doi.org/10.1097/00005176-199703000-00019

피인용 문헌

  1. Infantile Hepatic Hemangioendothelioma in Comparison with Hepatoblastoma in Children: Clinical and Ultrasound Features vol.13, pp.8, 2013, https://doi.org/10.5812/hepatmon.11103
  2. Spontaneous Regression of Infantile Hepatic Hemangioendothelioma vol.21, pp.2, 2013, https://doi.org/10.1016/j.jmu.2013.04.003
  3. Angiosarcoma successfully treated with liver transplantation and sirolimus vol.18, pp.4, 2011, https://doi.org/10.1111/petr.12245
  4. Primary mesenchymal liver tumors: radiological spectrum, differential diagnosis, and pathologic correlation vol.40, pp.5, 2011, https://doi.org/10.1007/s00261-014-0268-9
  5. Infantile Hepatic Hemangioendothelioma Associated With Congestive Heart Failure : Two Case Reports With Different Outcomes vol.94, pp.52, 2011, https://doi.org/10.1097/md.0000000000002344
  6. Elevated Serum Levels of Alpha-Fetoprotein in Patients with Infantile Hemangioma Are Not Derived from within the Tumor vol.3, pp.None, 2011, https://doi.org/10.3389/fsurg.2016.00005
  7. Medication Repurposing in Pediatric Patients: Teaching Old Drugs New Tricks vol.21, pp.1, 2016, https://doi.org/10.5863/1551-6776-21.1.36
  8. Infantile Hepatic Hemangioendothelioma: An Uncommon Cause of Persistent Pulmonary Hypertension in a Newborn Infant vol.6, pp.3, 2011, https://doi.org/10.1055/s-0036-1585578
  9. Beta blocker and steroid therapy in the treatment of infantile hepatic hemangioendothelioma vol.11, pp.3, 2011, https://doi.org/10.5582/ddt.2017.01025
  10. A Case of Consumptive Hypothyroidism in a 1-Month-Old Boy with Diffuse Infantile Hepatic Hemangiomas vol.35, pp.22, 2011, https://doi.org/10.3346/jkms.2020.35.e180
  11. Contrast-Enhanced Ultrasound for the Characterization of Infantile Hepatic Hemangioma in Premature Neonate vol.12, pp.8, 2020, https://doi.org/10.7759/cureus.9580
  12. Propranolol for infantile hepatic hemangioendothelioma: Clinical evaluation of drug efficacy and safety using a single-center patient cohort vol.19, pp.5, 2011, https://doi.org/10.1016/j.aohep.2020.04.008
  13. Use of Propranolol for Infantile Hepatic Hemangioma vol.32, pp.1, 2011, https://doi.org/10.14734/pn.2021.32.1.36
  14. Giant hepatic hemangioma in the fetus: case reports and updated review of the literature vol.34, pp.15, 2011, https://doi.org/10.1080/14767058.2019.1669555