Surgical removal of cystic craniopharyngiomas in children is associated with significant operative morbidity and recurrence rates. The purpose of this study was to review our experience with a less invasive therapy, namely, intratumoral bleomycin, in the treatment of predominantly cystic craniopharyngiomas. All children with craniopharyngiomas treated at a tertiary care pediatric neurosurgical center since 1994, when bleomycin was first used, were reviewed retrospectively. Seven patients received intratumoral bleomycin therapy. Patients received 2–5 mg bleomycin per dose, 3 times per week, for 3–5 weeks as an initial course. Mean follow-up of these patients was 3 years. In 4 patients, treatment resulted in a significant decrease (>50%) in tumor size, which has remained stable. Two patients’ tumors progressed and underwent resection, and 1 patient had surgical removal because of persistent headaches, although no growth of residual tumor had been noted. One patient developed peritumoral edema as a result of bleomycin therapy. Intratumoral bleomycin is a useful alternative therapy for cystic craniopharyngiomas, and may control tumor growth and delay potentially harmful resection and/or radiotherapy in young children.

Keywords:
Cystic craniopharyngioma, Bleomycin, Intratumoral chemotherapy
1.
Katz EL: Late results of radical excision of craniopharyngiomas in children. J Neurosurg 1975;42:86–90.
2.
Hoffman HJ, De Silva M, Humphreys RP, Drake JM, Smith ML, Blaser SI: Aggressive surgical management of craniopharyngiomas in children. J Neurosurg 1992;76:47–52.
3.
Tomita T, McLone DG: Radical resection of childhood craniopharyngiomas. Pediatr Neurosurg 1993;19:6–14.
4.
Flickinger JC: Megavoltage external beam irradiation of craniopharyngiomas: Analysis of tumor control and morbidity. Int J Radiat Oncol Biol Phys 1990;19:117–122.
5.
Rajan B, Ashley S, Gorman C, Jose CC, Horwich A, Bloom HJG, Marsh H, Brada M: Craniopharyngioma – Long term results following limited surgery and radiotherapy. Radiother Oncol 1993;27:13–21.
6.
Scott RM, Hetelekidis S, Barnes PD, Goumnerova L, Tarbell NJ: Surgery, radiation and combination therapy in the treatment of childhood craniopharyngioma – A 20-year experience. Pediatr Neurosurg 1994;21(suppl 1):75–81.
7.
Lundsford LD, Pollock BE, Kondziolka D, Levine G, Flickinger JC: Stereotactic options in the management of craniopharyngiomas. Pediatr Neurosurg 1994;21(suppl 1):90–97.
8.
Gutin DH, Klemme WM, Lagger RI, Mackay AR, Pitts LH, Hojobuchi Y: Management of unresectable cystic craniopharyngioma by aspiration through an Ommaya reservoir drainage system. J Neurosurg 1980;52:3–40.
9.
Backlund EO, Axelsson B, Bergstrand CG, Eriksson AL, Noren G, Ribbesjo E, Rahn T, Schnell PO, Tallstedt L, Saaf M, Thoren M: Treatment of craniopharyngiomas: The stereotactic approach in a ten to twenty year perspective. I. Surgical, radiological and ophthalmological aspects. Acta Neurochir (Wien) 1989;99:11–19.
10.
Pollock BE, Lundsford LD, Kondziolka D, Levine G, Flickinger JC: Phosphorous-32 intracavitary irradiation of cystic craniopharyngiomas: Current technique and long-term results. Int J Rad Oncol Biol Phys 1995;33:437–446.
11.
Van den Berge JH, Biaauw G, Breeman WAP, Rahm A, Wijangaarde R: Intracavitary brachytherapy of cystic craniopharyngioma. J Neurosurg 1992;77:545–550.
12.
Voges J, Sturm V, Lehrke R, Treuer H, Gauss C, Berthold F: Cystic craniopharyngioma: Long-term results after intracavitary irradiation with stereotactically applied colloidal β-emitting radioactive sources. Neurosurgery 1997;40:263–270.
13.
Broggi G, Giorgi C, Franzini A, Leocata F, Riva D: Therapeutic role of intracavitary bleomycin administration in cystic craniopharyngioma; in Broggi G (ed): Craniopharyngioma. Surgical Treatment. Milan, Springer-Verlag, 1995, pp 113–119.
14.
Cavalheiro S, Veiga DE Castro Sparapani F, Franco JOB, Braga FM: Use of bleomycin in intratumoral chemotherapy for cystic craniopharyngioma. Case report. J Neurosurg 1996;84:124–126.
15.
Frank F, Fabrizi AP, Frank G, Fioravanti A: Sterotactic management of craniopharyngiomas. Stereotact Funct Neurosurg 1995;65:176–183.
16.
Haisa T, Ueki K, Yoshida S: Toxic effects of bleomycin on the hypothalamus following its administration into a cystic craniopharyngioma. BJNS 1994;8:747–750.
17.
Mottolese C, Guyotat J, Bret P, Mirecvski V, Abdulrahman M, Lapras C: Treatment of craniopharyngiomas with local intracystic chemotherapy with bleomycin: Our experience. American Association of Neurological Surgeons Meeting. J Neurosurg 1996;84:343 (abstract).
18.
Ono N, Kohga H, Zama A, Inoue HK, Tamura M: A comparison of children with suprasellar germ cell tumors and craniopharyngiomas: Final height, weight, endocrine and visual sequelae after treatment. Surg Neurol 1996;46:370–377.
19.
Takahashi H, Nakazawa S, Shimura T: Evaluation of postoperative intratumoral injection of bleomycin for craniopharyngioma in children. J Neurosurg 1985;62:120–127.
20.
Sagoh M, Murakami H, Hirose Y, Mayanagi K: Occlusive cerebrovasculopathy after internal radiation and bleomycin therapy for craniopharyngioma – Case report. Neurol Med Chir 1997;37:920–923.
21.
Savas A, Erdem A, Tun K, Kanpolat Y: Fatal toxic effect of bleomycin on brain tissue after intracystic chemotherapy for a craniopharyngioma: Case report. Neurosurgery 2000;46:213–217.
22.
Cavazzuti V, Fischer EG, Welch K, Belli JA, Winston KR: Neurological and psychophysiological sequelae following different treatments of craniopharyngioma in children. J Neurosurg 1983;59:409–417.
23.
De Vile CJ, Grant DB, Kendall BE, Neville BGR, Stanhope R, Watkins KE, Hayward RD: Management of childhood craniopharyngioma: Can the morbidity of radical surgery be predicted? J Neurosurg 1996;85:73–81.
24.
Saaf M, Thoren M, Bergstrand CG, Noren G, Rahn T, Tallstedt L, Backlund EO: Treatment of craniopharyngiomas – The stereotactic approach in a ten to twenty-three years’ perspective. II. Psychosocial situation and pituitary function. Acta Neurochir (Wien) 1989;99:97–103.
25.
Shiminski-Maher T: Patient/family preparation and education for complications and late sequelae of craniopharyngiomas. Pediatr Neurosurg 1994;21 (suppl 1):114–119.
26.
Villani R, Tomei G, Bello L, Sganzerla E, Ambrosi B, Re T: Long-term results of treatment for craniopharyngiomas in children. Childs Nerv Syst 1997;13:397–405.
27.
Umezawa H, Maeda K, Takeuchi T, Okami Y: New antibiotics, bleomycin A and B. J Antibiot 1966;19:200–209.
28.
Bosch DA, Kindmorsch TH, Larsson ST, Backlund EO: Intraneoplastic administration of bleomycin in intracerebral gliomas, a pilot study. Acta Neurochir Suppl 1980;30:441–444.
29.
Morantz RA, Kimler BF, Vats TS, Henderson SD: Bleomycin and brain tumors: A review. J Neurooncol 1983;1:249–255.
30.
Nakazawa S, Ohwaki K, Shimura T, Itoh Y, Yajima K: A new treatment of malignant brain tumor. I. Local injection of bleomycin (in Japanese). No Shinkei Geka 1981;9:1487–1493.
31.
Ellenberg L, McComb JG, Siegel SE, Stowe S: Factors affecting intellectual outcome in pediatric brain tumor patients. Neurosurgery 1987;21:638–644.
32.
Pierre-Kahn A, Sainte-Rose C, Renier D: Surgical approach to children with craniopharyngiomas and severely impaired vision: Special considerations. Pediatr Neurosurg 1994;21(suppl 1):50–56.
33.
Simpson AB, Paul J, Graham J, Kaye SB: Fatal bleomycin pulmonary toxicity in the west of Scotland 1991–1995: A review of patients with germ cell tumors. Br J Cancer 1998;78:1061–1066.
34.
Wisoff JH: Surgical management of recurrent craniopharyngiomas. Pediatr Neurosurg 1994;21(suppl 1):108–113.
© 2000 S. Karger AG, Basel
2000
Copyright / Drug Dosage / Disclaimer
Copyright: All rights reserved. No part of this publication may be translated into other languages, reproduced or utilized in any form or by any means, electronic or mechanical, including photocopying, recording, microcopying, or by any information storage and retrieval system, without permission in writing from the publisher.
Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in government regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug.
Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.
You do not currently have access to this content.