publication history
Publication Date:24 July 2008 (online)
Summary of the
Background: The therapy of choice for macrocystic lymphangiomas is complete resection. However, due to the growth pattern of this congenital malformation and the main localization in the head and neck area, resection is often only incompletely possible. An alternative is sclerosing with Picibanil. Experience with the use of Picibanil in infants has so far only been available in the form of case reports.
Patient/Method: From 2002 to 2006, retrospectively the data of 8 children, aged from 2 weeks to 12 months, who were treated with Picibanil for macrocystic or mixed cystic lymphangioma as part of a curative trial, were processed. The follow-up period was 3 months to 3 years.
Results: Postoperatively all patients showed a local swelling, in 4 cases accompanied by redness and fever. A tumor regress of > 50% occurred in 7 of the 8 patients.
Conclusion: The injection of Picibanil also offers a safe alternative or supplement to surgical resection in infants, especially if complete removal can only be achieved by injuring important structures. However, multi-centric prospective studies are required to demonstrate superiority over primary surgical resection.
abstract
Background: The therapeutic gold standard of cystic hygroma is its complete resection. Because of its growth pattern and its main location in the head and neck region complete resection is not always possible. An alternative is the local injection of Picibanil, but only few cases have been published about its use in infants.
Patients/Method: We retrospectively analyzed the data of 8 infants (age: 2 weeks – 12months) who got Picibanil therapy because of cystic hygroma in the time period 2002 until 2006. Follow up ranged from 3 months up to 3 years.
Results: During the postoperative period all patients had local swelling, in 4 cases accompanied with local inflammation and fever. Tumorreduction of > 50% was obtained in 7 of 8 patients.
Conclusions: Local injection of Picibanil in infants with cystic hygroma seems to be a safe alternative to surgical therapy, especially when complete tumorresection means damage of important neighbouring structures. Prospective trials are necessary to confirm the better outcome after therapy with Picibanil compare to primary surgery.
key words
Picibanil – Lymphangioma – OK 432 – Infants – Childhood
Key words
Picibanil – lymphangioma – OK 432 – newborns – infants
literature
- 1 Claesson G, Kuylenstierna R. OK-432-therapy for lymphatic malformation in 32 patients (28 children). Int J Ped OtoRhinoLaryngol. 2002; 65 1-6
- 2 Giguere CM, Bauman NM, Smith RJ. New treatment options for lymphangioma in infants and children. Ann Otol Rhinol Laryngol. 2002; 111 1066-1075
- 3 Giguere CM, Bauman NM, Sato Y, Burke DK, Greinwald JH, Pransky S, Kelley P, Georgeson K, Smith RJ. Treatment of lymphangiomas with OK-432 (Picibanil) sclerotherapy: a prospective multi-institutional trial. Arch Otolaryngol Head Neck Surg. 2002; 128 ((10)) 1137-1144
- 4 Heether J, Whalen T, Doolin E. Follow-up of complex unresectable lymphangiomas. Am Surg. 1994; 60 840-841
- 5 Kennedy TL, Whitaker M, Pellitteri P, Wood WE. Cystic hygroma/lymphangioma: a rational approach to management. Laryngoscope. 2001; 111 1929-1937
- 6 Ley P, Scholz H, Müller-Schimpfle M, Schrod L. Complete regression of a large retropharyngeal lymphangioma after three injections of OK-432. Clin Paediatric 2007; 219 110
- 7 Luzzatto C, Lo Piccolo R, Fascetti Leon F, Zanon GF, Toffolutti T, Tregnaghi A. Further experience with Picibanil for lymphangiomas. Ped surg Int. 2005; 21 ((12)) 969-972
- 8 Mitoch HI, Unger EC, Witte CL, Sonnenberg E van. Percutaneous sclerotherapy of lymphangiomas. Radiology. 1995; 194 343-347
- 9 Muir T, Kirsten M, Fourie P, Dippenaar N, Ionescu GO. Intralesional bleomycin injection (IBI) treatment for haemangiomas and congenital vascular malformations. Pediatr Surg Int. 2004; 19 ((12)) 766-773
- 10 Ogita S, Tsuto T, Tokiwa K, Takahashi T. Treatment of cystic hgroma in children with special reference to OK-432 therapy. Z Kinderchir. 1987; 42 279-281
- 11 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 Ped Surg. 1996; 31 477-480
- 12 Ogita S, Tsuto T, Deguchi E, Iwai N. OK-432 therapy in 64 patients with lymphangioma. J Ped Surg. 1994; 29 784-778
- 13 Padwa BL, Hayward PG, Ferraro NF, Mulliken JB. Cervicofacial lymphatic malformation: clinical course, surgical intervention, and pathogenesis of skeletal hypertrophy. Plast Reconstr Surg. 1995; 95 951-960
- 14 Riechelmann H, Muehlfay G, Keck T, Mattfeldt T, Rettinger G. Total, subtotal, and partial surgical removal of cervicofacial lymphangiomas. Arch Otolaryngol Head Neck Surg. 1999; 125 ((6)) 643-648
- 15 Sasaki Y, Chiba Y. Successful intrauterine treatment of cystic hygroma colli using OK-432. A case report. Fetal Diagn Ther. 2003; 18 ((6)) 391-396
correspondence address
Dr. K. Reinshagen
Pediatric Surgery, University Hospital Mannheim