Treatment of Lymphatic Malformations with OK-432: the First Experience of a Single Hospital

PROBLEMS OF PEDIATRICS
Iryna Benzar

Department of Pediatric Surgery, Bogomolets National Medical University; Kyiv, Ukraine

*Corresponding author: Iryna Benzar. Department of Pediatric Surgery, Bogomolets National Medical University, Kyiv, Ukraine. E-mail: ira_benzar@yahoo.com

Published: December 23, 2014. 

Abstract: 

Purpose: This study aims to evaluate the effectiveness of OK-432 immunotherapy in the treatment of lymphatic malformation (LM) in children and to determine its complications and contraindications.

Methods: Twenty-eight patients with LM of head and neck were enrolled. Twenty-three (82.2%) patients were treated with OK-432 immunotherapy, and surgery was performed in 5 (17.8%). LMs were classified as macrocystic, microcystic, or combined, and according to de Serres clinical stages. OK-432 immunotherapy consisted of a puncture, aspiration and intralesional injection of OK-432.

Results: Three patients with contraindication to OK-432 were successfully treated surgically. In patients with parotid involvement recurrence of LM and facial nerve branch, paresis occurred. After OK-432 treatment and a single session of 1 KE of OK-432, favorable results were obtained in patients with stage I and macrocystic lesions. In patients with stage III, the result was excellent or good in 7 (70%) and fair in 3 (30%). We didn’t receive satisfactory results in groups IV and V (good – 1, fair – 4, poor – 1).

Conclusion: OK-432 immunotherapy is safe, effective, and simple to use. The result of treatment depends on cystic size, extent of lesions, and previous interventions, and was most successful in unilateral macrocystic LM. 

References: 
  1. Acevedo JL, Shah RK, Brietzke SE. Nonsurgical therapies for lymphangiomas: a systematic review. Otolaryngol Head Neck Surg  2008; 138(4):418-424. doi: 10.1016/j.otohns.2007.11.018
  2. Gilony D, Schwartz M, Shpitzer T, Feinmesser R, Kornreich L, Raveh E. Treatment of lymphatic malformations: a more conservative approach. J Pediatric Surgery 2012; 47(10):1837–1842. doi:10.1016/j.jpedsurg.2012.06.005
  3. Perkins JA, Manning SC, Tempero RM, Cunningham MJ, Edmonds JL Jr, Hoffer FA, et al. Lymphatic malformations: review of current treatment. Otolaryngol Head Neck Surg 2010; 142(6):795-803, 803.e1. doi: 10.1016/j.otohns.2010.02.026
  4. Churchill P, Otal D, Pemberton J, Ali A, Flageole H, Walton JM. Sclerotherapy for lymphatic malformations in children: a scoping review. J Pediatr Surg 2011; 46(5):912-22. doi: 10.1016/j.jpedsurg.2011.02.027
  5. Ogita S, Tsuto T, Tokiwa K, Takahashi T. Intracystic injection of OK-432: a new sclerosing therapy for cystic hygroma in children. Br J Surg  1987; 74(8):690-1.
  6. Smith MC, Zimmerman MB, Burke DK, Bauman NM, Sato Yu, Smith RJ. Efficacy and Safety of OK-432 Immunotherapy of Lymphatic Malformations. Laryngoscope 2009; 119(1):107-15. doi: 10.1002/lary.20041
  7. Claesson G, Kuylenstierna R. OK-432 therapy for lymphatic malformation in 32 patients (28 children). Int J Pediatr Otorhinolaryngol 2002; 65(1):1-6.
  8. De Serres LM, Sie KCY, Richardson MA. Lymphatic malformations of the head and neck. A proposal for staging. Arch Otolaryngol Head Neck Surg 1995; 121(5):577-582.
  9. Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: a classification based on endothelial characteristics. Plast Reconstr Surg 1982; 69(3):412-22.
  10. Al-Adnani M, Williams S, Rampling D, Ashworth M, Malone M, Sebire NJ. Histopathological reporting of paediatric cutaneous vascular anomalies in relation to proposed multidisciplinary classification system. J Clin Pathol 2006; 59(12):1278–82.
  11. Legiehn GM, Heran MK. A Step-by-Step Practical Approach to Imaging Diagnosis and Interventional Radiologic Therapy in Vascular Malformations. Semin Intervent Radiol 2010; 27(2):209–231. doi: 10.1055/s-0030-1253521
  12. Rautio R, Keski-Nisula L, Laranne J, Laasonen E. Treatment of lymphangiomas with OK-432 (Picibanil). Cardiovasc Intervent Radiol 2003; 26(1):31-6. doi: 10.1007/s00270-002-1980-3
  13. Richter GT, Friedman AB. Hemangiomas and vascular malformations: current theory and management. Int J Pediatr 2012; 2012:645678. doi: 10.1155/2012/645678
  14. Giguère CM, Bauman NM, Sato Y, Burke DK, Greinwald JH, Pransky S, et al. Treatment of lymphangiomas with OK-432 (Picibanil) sclerotherapy: a prospective multi-institutional trial. Arch Otolaryngol Head Neck Surg 2012; 128(10):1137-44. doi:10.1001/archotol.128.10.1137
  15. Ogita S, Tsuto T, Nakamura K, Deguchi E, Tokiwa K, Iwai N. OK-432 therapy for lymphangioma in children: why and how does it work? J Pediatr Surg 1996; 31(4):477-80. doi: 0022-3468/96/3104-0003s03.00
  16. Wiegand S, Eivazi B, Sel S, Renz H, Werner JA Folz BJ. Analysis of Cytokine Levels in Human Lymphangiomas. In Vivo 2008; 22: 253-6.
  17. Yoo JC, Ahn Y, Lim YS, Hah JH, Kwon TK, Sung MW, et al. OK-432 sclerotherapy in head and neck lymphangiomas: long-term follow-up result. Otolaryngol Head Neck Surg 2009; 140(1):120-3. doi: 10.1016/j.otohns.2008.10.026
  18. Boardman SJ, Cochrane LA, Roebuck D, Elliott MJ, Hartley BE. Multimodality treatment of pediatric lymphatic malformations of the head and neck using surgery and sclerotherapy. Arch Otolaryngol Head Neck Surg 2010; 136(3):270-6. doi: 10.1001/archoto.2010.6

The fully formatted PDF version is available.       

Download Article

Int J Biomed. 2014; 4(4):237-241. © 2014 International Medical Research and Development Corporation. All rights reserved.