Pediatric Gastroenterology, Hepatology & Nutrition

The Korean Society of Pediatric Gastroenterology, Hepatology and Nutrition (대한소아소화기영양학회)
권호 표지

A Case of a Retroperitoneal Cystic Lymphangioma Treated by Percutaneous Catheter Drainage and Sclerotherapy

경피적 도관 배액술과 경화요법으로 치료한 후복막 낭성 림프관종 1예

  • Kang, Hyun-Sik (Department of Pediatrics, Jeju National University School of Medicine) ;
  • Kim, Seung-Hyung (Department of Radiology, Jeju National University School of Medicine) ;
  • Kim, Bong-Soo (Department of Radiology, Jeju National University School of Medicine) ;
  • Kang, Ki-Soo (Department of Pediatrics, Jeju National University School of Medicine)
  • 강현식 (제주대학교 의과대학 소아과학교실) ;
  • 김승형 (제주대학교 의과대학 영상의학과학교실) ;
  • 김봉수 (제주대학교 의과대학 영상의학과학교실) ;
  • 강기수 (제주대학교 의과대학 소아과학교실)
  • Received : 2010.01.31
  • Accepted : 2010.03.07
  • Published : 2010.03.30
Download PDF
⟨ Previous Next ⟩

Abstract

Retroperitoneal cystic lymphangiomas are benign, extremely rare tumors. Although surgical resection is the treatment of choice, sclerotherapy should be considered initially. A 9-year-old boy was admitted due do worsening abdominal pain of 4 days duration. Serial complete blood counts revealed a hemoglobin level of 12.8 g/dL on admission to 10.6 g/dL on hospital day 3. An abdominal computed tomography (CT) scan showed a large, lobulated, septated, retroperitoneal cystic mass ($10{\times}9.5{\times}5cm$) in the left anterior pararenal space with intracystic hemorrhage surrounding the inferior mesenteric vein (IMV). Because of the high operative risk, we performed a tubogram of the cystic mass, percutaneous catheter drainage (PCD), and ethanol sclerotherapy. The follow-up abdominal CT scan showed that the cystic mass had decreased in size. He is well without relapse of the retroperitoneal cystic mass for 13 months after discharge. Sclerotherapy with PCD should be considered as initial therapy for patients with retroperitoneal cystic lymphangiomas at high surgical risk.

후복막 낭성 림프관종은 양성종양으로 매우 드문 질환이다. 수술적 절제가 원칙이나, 경화요법을 우선적으로 고려할 수도 있다. 9세 남자가 병원 방문 4일 전부터점점 심해지는 복통을 주소로 입원하였다. 혈색소 농도가 입원 당일 12.8 g/dL에서, 입원 3병일째 10.6 g/dL로 감소한 소견이 있었다. 복부 전산화 단층촬영에서 크고 종양내부에 격막이 있는 분엽상의 후복막 낭종($10{\times}9.5{\times}5cm$) 한 개가 좌측 신장 주위 공간에서 관찰되었다. 종양 내부에는 출혈이 동반되어 있었으며, 하장간막정맥이 종양에 의해 둘러싸여져 있었다. 수술적 치료의 위험도가 매우 높을 것으로 판단되어 낭종조영술을 시행하였고, 경피적 도관 배액술과 에탄올 경화요법을 시행하였다. 추적 검사로 시행한 복부 전산화 단층촬영에서 종양의 크기가 현저히 감소된 소견을 보였다. 환자는 퇴원 후 13개월 째 현재까지 낭종의 재발없이 건강하게 지내고 있다. 결론적으로, 수술적 치료의 위험이 높은 후복막 낭성 림프관종이 있는 경우 경피적 도관 배액술과 경화요법을 우선적으로 고려해야 할 것이다.

Keywords

References

  1. Okazaki T, Iwatani S, Yanai T, Kobayashi H, Kato Y, Marusasa T, et al. Treatment of lymphangioma in children: our experience of 128 cases. J Pediatr Surg 2007;42:386-9. https://doi.org/10.1016/j.jpedsurg.2006.10.012
  2. de Perrot M, Rostan O, Morel P, Le Coultre C. Abdominal lymphangioma in adults and children. Br J Surg 1998;85:395-7. https://doi.org/10.1046/j.1365-2168.1998.00628.x
  3. Hancock BJ, St-Vil D, Luks FI, Di Lorenzo M, Blanchard H. Complications of lymphangiomas in children. J Pediatr Surg 1992;27:220-4; discussion 4-6. https://doi.org/10.1016/0022-3468(92)90316-Y
  4. Steyaert H, Guitard J, Moscovici J, Juricic M, Vaysse P, Juskiewenski S. Abdominal cystic lymphangioma in children: benign lesions that can have a proliferative course. J Pediatr Surg 1996;31:677-80. https://doi.org/10.1016/S0022-3468(96)90673-9
  5. Konen O, Rathaus V, Dlugy E, Freud E, Kessler A, Shapiro M, et al. Childhood abdominal cystic lymphangioma. Pediatr Radiol 2002;32:88-94. https://doi.org/10.1007/s00247-001-0612-4
  6. Davidson AJ, Hartman DS. Lymphangioma of the retroperitoneum: CT and sonographic characteristic. Radiology 1990;175:507-10. https://doi.org/10.1148/radiology.175.2.2183287
  7. Roisman I, Manny J, Fields S, Shiloni E. Intra-abdominal lymphangioma. Br J Surg 1989;76:485-9. https://doi.org/10.1002/bjs.1800760519
  8. Yang DH, Goo HW. Generalized lymphangiomatosis: radiologic findings in three pediatric patients. Korean J Radiol 2006;7:287-91. https://doi.org/10.3348/kjr.2006.7.4.287
  9. Shankar KR, Roche CJ, Carty HM, Turnock RR. Cystic retroperitoneal lymphangioma: treatment by image-guided percutaneous catheter drainage and sclerotherapy. Eur Radiol 2001;11:1021-3. https://doi.org/10.1007/s003300000669
  10. Kosumi T, Kubota A, Yonekura T, Yamauchi K. Cystic lymphangioma of Retzius space manifested as acute abdomen. Eur J Pediatr Surg 2006;16:120-2. https://doi.org/10.1055/s-2006-923999
  11. Kosumi T, Kubota A, Yonekura T, Yamauchi K. Cystic lymphangioma of Retzius space manifested as acute abdomen. Eur J Pediatr Surg 2006;16:120-2. https://doi.org/10.1055/s-2006-923999
  12. Yang DM, Jung DH, Kim H, Kang JH, Kim SH, Kim JH, et al. Retroperitoneal cystic masses: CT, clinical, and pathologic findings and literature review. Radiographics 2004;24:1353-65. https://doi.org/10.1148/rg.245045017
  13. Iyer R, Eftekhari F, Varma D, Jaffe N. Cystic retroperitoneal lymphangioma: CT, ultrasound and MR findings. Pediatr Radiol 1993;23:305-6. https://doi.org/10.1007/BF02010922
  14. de Lagausie P, Bonnard A, Berrebi D, Lepretre O, Statopoulos L, Delarue A, et al. Abdominal lymphangiomas in children: interest of the laparoscopic approach. Surg Endosc 2007;21:1153-7. https://doi.org/10.1007/s00464-006-9091-x
  15. Tran Ngoc N, Tran Xuan N. Cystic hygroma in children: a report of 126 cases. J Pediatr Surg 1974;9:191-5. https://doi.org/10.1016/S0022-3468(74)80120-X
  16. Luzzatto C, Midrio P, Tchaprassian Z, Guglielmi M. Sclerosing treatment of lymphangiomas with OK-432. Arch Dis Child 2000;82:316-8. https://doi.org/10.1136/adc.82.4.316
  17. Park SW, Cha IH, Kim KA, Hong SJ, Park CM, Chung HH. Percutaneous sclerotherapy using acetic acid after failure of alcohol ablation in an intra-abdominal lymphangioma. Cardiovasc Intervent Radiol 2004;27:285-7.
  18. Ozdemir H, Kocakoc E, Bozgeyik Z, Cobanoglu B. Recurrent retroperitoneal cystic lymphangioma. Yonsei Med J 2005;46:715-8. https://doi.org/10.3349/ymj.2005.46.5.715
  19. Fujino A, Moriya Y, Morikawa Y, Hoshino K, Watanabe T, Shimojima N, et al. A role of cytokines in OK-432 injection therapy for cystic lymphangioma: an approach to the mechanism. J Pediatr Surg 2003;38:1806-9. https://doi.org/10.1016/j.jpedsurg.2003.08.041
  20. Dubois J, Garel L, Abela A, Laberge L, Yazbeck S. Lymphangiomas in children: percutaneous sclerotherapy with an alcoholic solution of zein. Radiology 1997;204:651-4. https://doi.org/10.1148/radiology.204.3.9280239
  21. Tanaka K, Inomata Y, Utsunomiya H, Uemoto S, Asonuma K, Katayama T, et al. Sclerosing therapy with bleomycin emulsion for lymphangioma in children. Pediatr Surg Int 1990;5:270-3.
  22. Molitch HI, Unger EC, Witte CL, vanSonnenberg E. Percutaneous sclerotherapy of lymphangiomas. Radiology 1995;194:343-7. https://doi.org/10.1148/radiology.194.2.7529933