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Preservation of endocrine function after Ommaya reservoir insertion in children with cystic craniopharyngioma

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Abstract

Introduction

Children with craniopharyngiomas (CP) can experience significant morbidities caused by extensive surgery and/or radiation. Ommaya reservoir insertion (ORI) into cystic CP represents a minimally invasive approach allowing immediate decompression and aims to avoid additional injuries. The purpose of this study was to determine the surgical outcome and relevance of upfront ORI (± intracystic treatment) for preservation of endocrine function.

Methods

We performed a retrospective chart review of children with CP treated at the Hospital for Sick Children between 01/01/2000 and 15/01/2020. Endocrine function was reviewed at the time of initial surgery and throughout follow-up. New endocrinological deficits related to the index procedure were defined as immediate failure (IF), whereas postoperative duration of endocrinological stability (ES) was analyzed using the Kaplan-Meier method. The rate of IF and ES was compared between the treatment groups.

Results

Seventy-nine patients were included and had a median age of 8.3 years (range 2.1–18.0 years); 31 were males. Fifty-three patients with upfront surgical treatment, including 29 ORI and 24 gross total or partial resections had sufficient endocrinological follow-up data. Endocrine dysfunction occurring immediately after the index procedure (IF) was observed in 15 patients (62.5%) in the resection group compared to two patients (6.8%) in the ORI group, odds ratio: 0.05 (CI: 0.01–0.26, p < 0.0001). Excluding those with immediate endocrinological deficits, mean ES after ORI was 19.4 months (CI: 11.6–34.2), compared to 13.4 months (CI:10.6-NA) after surgical resection.

Conclusions

Endocrine function was preserved in patients with upfront ORI (± intracystic treatment), which was confirmed as a minimally invasive procedure with an overall low morbidity profile.

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Data availability

The datasets generated during and/or analysed during the current study are available from the corresponding author on reasonable request.

Abbreviations

ACTH:

Adrenocorticotropic hormone

BX:

Biopsy

CT:

Computerized tomography

CCT:

Cranial computerized tomography

cMRI:

Cranial magnet resonance imaging

DI:

Diabetes insipidus

ES:

Endocrinological stability

FSH:

Follicle-stimulating hormone

GH:

Growth hormone

GTR:

Gross total resection

HSC:

Hospital for Sick Children

IF:

Immediate failure

IFN-α:

Interferon-alpha

LH:

Luteinizing hormone

MRI:

Magnet Resonance Imaging

PFS:

Progression-free survival

QoL:

Quality of Life

STR:

Subtotal resection

TSH:

Thyroid-stimulating hormone

References

  1. Bunin GR, Surawicz TS, Witman PA, Preston-Martin S, Davis F, Bruner JM (1998) The descriptive epidemiology of craniopharyngioma. J Neurosurg 89(4):547–551. https://doi.org/10.3171/jns.1998.89.4.0547

    Article  CAS  PubMed  Google Scholar 

  2. Louis DN, Perry A, Wesseling P et al (2021) The 2021 WHO classification of tumors of the central nervous system: a summary. Neuro Oncol 23(8):1231–1251. https://doi.org/10.1093/neuonc/noab106

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  3. Bartels U, Laperriere N, Bouffet E, Drake J (2012) Intracystic therapies for cystic craniopharyngioma in childhood. Front Endocrinol 3:39. https://doi.org/10.3389/fendo.2012.00039

    Article  Google Scholar 

  4. Puget S (2012) Treatment strategies in childhood craniopharyngioma. Front Endocrinol 3:64. https://doi.org/10.3389/fendo.2012.00064

    Article  Google Scholar 

  5. Srinivasan S, Ogle GD, Garnett SP, Briody JN, Lee JW, Cowell CT (2004) Features of the metabolic syndrome after childhood craniopharyngioma. J Clin Endocrinol Metab 89(1):81–86. https://doi.org/10.1210/jc.2003-030442

    Article  CAS  PubMed  Google Scholar 

  6. Villani RM, Tomei G, Bello L et al (1997) Long-term results of treatment for craniopharyngioma in children. Child’s Nervous Syst 13(7):397–405. https://doi.org/10.1007/s003810050108

    Article  CAS  Google Scholar 

  7. Kalapurakal JA, Goldman S, Hsieh YC, Tomita T, Marymont MH (2003) Clinical outcome in children with craniopharyngioma treated with primary surgery and radiotherapy deferred until relapse. Med Pediatr Oncol 40(4):214–218. https://doi.org/10.1002/mpo.10247

    Article  PubMed  Google Scholar 

  8. Müller HL (2011) Consequences of craniopharyngioma surgery in children. J Clin Endocrinol Metab 96(7):1981–1991. https://doi.org/10.1210/jc.2011-0174

    Article  CAS  PubMed  Google Scholar 

  9. Müller HL (2010) Childhood craniopharyngioma: current controversies on management in diagnostics, treatment and follow-up. Expert Rev Neurother 10(4):515–524. https://doi.org/10.1586/ern.10.15

    Article  PubMed  Google Scholar 

  10. Puget S, Garnett M, Wray A et al (2007) Pediatric craniopharyngiomas: classification and treatment according to the degree of hypothalamic involvement. J Neurosurg 106(1 Suppl):3–12. https://doi.org/10.3171/ped.2007.106.1.3

    Article  PubMed  Google Scholar 

  11. Pierre-Kahn A, Recassens C, Pinto G et al (2005) Social and psycho-intellectual outcome following radical removal of craniopharyngiomas in childhood: a prospective series. Child’s Nervous Syst 21(8–9):817–24. https://doi.org/10.1007/s00381-005-1205-6

    Article  CAS  Google Scholar 

  12. Hargrave DR (2006) Does chemotherapy have a role in the management of craniopharyngioma? J Pediatric Endocrinol Metabol 19(Suppl 1):407–412

    Google Scholar 

  13. Jakacki RI, Cohen BH, Jamison C et al (2000) Phase II evaluation of interferon-alpha-2a for progressive or recurrent craniopharyngiomas. J Neurosurg 92(2):255–260. https://doi.org/10.3171/jns.2000.92.2.0255

    Article  CAS  PubMed  Google Scholar 

  14. Puget S, Grill J, Habrand JL, Sainte-Rose C (2006) Multimodal treatment of craniopharyngioma: defining a risk-adapted strategy. J Pediatric Endocrinol 19(Suppl 1):367–370

    Google Scholar 

  15. Flitsch J, Aberle J, Burkhardt T (2015) Surgery for pediatric craniopharyngiomas: is less more? J Pediatric Endocrinol 28(1–2):27–33. https://doi.org/10.1515/jpem-2014-0417

    Article  Google Scholar 

  16. Frio F, Solari D, Cavallo LM, Cappabianca P, Raverot G, Jouanneau E (2019) Ommaya reservoir system for the treatment of cystic craniopharyngiomas: surgical results in a series of 11 adult patients and review of the literature. World Neurosurg. https://doi.org/10.1016/j.wneu.2019.07.217

    Article  PubMed  Google Scholar 

  17. Gutin PH, Klemme WM, Lagger RL, MacKay AR, Pitts LH, Hosobuchi Y (1980) Management of the unresectable cystic craniopharyngioma by aspiration through an Ommaya reservoir drainage system. J Neurosurg 52(1):36–40. https://doi.org/10.3171/jns.1980.52.1.0036

    Article  CAS  PubMed  Google Scholar 

  18. Rogers LR, Barnett G (1991) Percutaneous aspiration of brain tumor cysts via the Ommaya reservoir system. Neurology 41(2):279–82. https://doi.org/10.1212/wnl.41.2_part_1.279

    Article  CAS  PubMed  Google Scholar 

  19. Hukin J, Steinbok P, Lafay-Cousin L et al (2007) Intracystic bleomycin therapy for craniopharyngioma in children: the Canadian experience. Cancer 109(10):2124–2131. https://doi.org/10.1002/cncr.22633

    Article  CAS  PubMed  Google Scholar 

  20. Mottolese C, Stan H, Hermier M et al (2001) Intracystic chemotherapy with bleomycin in the treatment of craniopharyngiomas. Child’s Nervous Syst 17(12):724–730. https://doi.org/10.1007/s00381-001-0524-5

    Article  CAS  Google Scholar 

  21. Zhang S, Fang Y, Cai BW, Xu JG, You C (2016) Intracystic bleomycin for cystic craniopharyngiomas in children. Cochrane Database System Rev 7:CD008890. https://doi.org/10.1002/14651858.CD008890.pub4

    Article  Google Scholar 

  22. Steinbok P, Hukin J (2010) Intracystic treatments for craniopharyngioma. Neurosurg Focus 28(4):E13. https://doi.org/10.3171/2010.1.focus09315

    Article  PubMed  Google Scholar 

  23. Kilday JP, Caldarelli M, Massimi L et al (2017) Intracystic interferon-alpha in pediatric craniopharyngioma patients: an international multicenter assessment on behalf of SIOPE and ISPN. Neuro Oncol 19(10):1398–1407. https://doi.org/10.1093/neuonc/nox056

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  24. Ierardi DF, Fernandes MJ, Silva IR et al (2007) Apoptosis in alpha interferon (IFN-alpha) intratumoral chemotherapy for cystic craniopharyngiomas. Child’s Nervous Syst 23(9):1041–1046. https://doi.org/10.1007/s00381-007-0409-3

    Article  CAS  Google Scholar 

  25. Cavalheiro S (2017) Intracystic interferon-alpha in pediatric craniopharyngioma patients. Neuro Oncol 19(10):1419. https://doi.org/10.1093/neuonc/nox123

    Article  PubMed  PubMed Central  Google Scholar 

  26. Dastoli PA, Nicacio JM, Silva NS et al (2011) Cystic craniopharyngioma: intratumoral chemotherapy with alpha interferon. Arq Neuropsiquiatr 69(1):50–55

    Article  Google Scholar 

  27. Choux MLG, Genitori L., Children. Ci, Neurochirurgie 37(Suppl. 1), (1991).

  28. Moussa AH, Kerasha AA, Mahmoud ME (2013) Surprising outcome of ommaya reservoir in treating cystic craniopharyngioma: a retrospective study. Br J Neurosurg 27(3):370–373. https://doi.org/10.3109/02688697.2012.741732

    Article  CAS  PubMed  Google Scholar 

  29. Rahmathulla G, Barnett GH (2013) Minimally invasive management of adult craniopharyngiomas: an analysis of our series and review of literature. Surg Neurol Int 4(Suppl 6):S411–S421. https://doi.org/10.4103/2152-7806.121612

    Article  PubMed  PubMed Central  Google Scholar 

  30. Reda WA, Hay AA, Ganz JC (2002) A planned combined stereotactic approach for cystic intracranial tumors: report of two cases. J Neurosurg 97(5 Suppl):610–2. https://doi.org/10.3171/jns.2002.97.supplement

    Article  PubMed  Google Scholar 

  31. Jarebi M, Coutte A, Bartier F, Khormi Y, Peltier J, Lefranc M (2019) A novel, hybrid, stereotactic approach (radiosurgery and dual Ommaya reservoirs) for the treatment of mixed (polycystic and solid) craniopharyngioma. Stereotact Funct Neurosurg 97(4):266–271. https://doi.org/10.1159/000503690

    Article  PubMed  Google Scholar 

  32. Schubert T, Trippel M, Tacke U et al (2009) Neurosurgical treatment strategies in childhood craniopharyngiomas: is less more? Child’s Nervous Syst 25(11):1419–1427. https://doi.org/10.1007/s00381-009-0978-4

    Article  Google Scholar 

  33. Cavalheiro S, Di Rocco C, Valenzuela S et al (2010) Craniopharyngiomas: intratumoral chemotherapy with interferon-alpha: a multicenter preliminary study with 60 cases. Neurosurg Focus 28(4):E12. https://doi.org/10.3171/2010.1.focus09310

    Article  PubMed  Google Scholar 

  34. Mrowczynski OD, Langan ST, Rizk EB (2018) Craniopharyngiomas: a systematic review and evaluation of the current intratumoral treatment landscape. Clin Neurol Neurosurg 166:124–130. https://doi.org/10.1016/j.clineuro.2018.01.039

    Article  PubMed  Google Scholar 

  35. Takahashi H, Yamaguchi F, Teramoto A (2005) Long-term outcome and reconsideration of intracystic chemotherapy with bleomycin for craniopharyngioma in children. Child’s Nervous Syst 21(8–9):701–704. https://doi.org/10.1007/s00381-005-1208-3

    Article  Google Scholar 

  36. Hukin J, Visser J, Sargent M, Goddard K, Fryer C, Steinbok P (2005) Childhood craniopharyngioma: Vancouver experience. Child’s Nervous Syst 21(8–9):758–765. https://doi.org/10.1007/s00381-005-1220-7

    Article  Google Scholar 

  37. Rachinger W, Oehlschlaegel F, Kunz M et al (2017) Cystic craniopharyngiomas: microsurgical or stereotactic treatment? Neurosurgery 80(5):733–743. https://doi.org/10.1227/neu.0000000000001408

    Article  PubMed  Google Scholar 

  38. Olsson DS, Andersson E, Bryngelsson IL, Nilsson AG, Johannsson G (2015) Excess mortality and morbidity in patients with craniopharyngioma, especially in patients with childhood onset: a population-based study in Sweden. J Clin Endocrinol Metab 100(2):467–474. https://doi.org/10.1210/jc.2014-3525

    Article  CAS  PubMed  Google Scholar 

  39. Lucas C, Benderitter T, Choux M (1982) Pre- and postoperative endocrine function in children with craniopharyngioma. Archives francaises de pediatrie 39(5):303–307

    CAS  PubMed  Google Scholar 

  40. Clark AJ, Cage TA, Aranda D, Parsa AT, Auguste KI, Gupta N (2012) Treatment-related morbidity and the management of pediatric craniopharyngioma: a systematic review. J Neurosurg Pediatr 10(4):293–301. https://doi.org/10.3171/2012.7.Peds11436

    Article  PubMed  Google Scholar 

  41. Müller HLSN (2001) K, Multizentrische PB, mit vKuJ. Verlag KMlHE, Isensee O, Germany

    Google Scholar 

  42. Merchant TE, Kiehna EN, Sanford RA et al (2002) Craniopharyngioma: the St Jude Children’s Research Hospital experience 1984–2001. Int J Radiat Oncol Biol Phys 53(3):533–42. https://doi.org/10.1016/s0360-3016(02)02799-2

    Article  PubMed  Google Scholar 

  43. Zuccarelli B, Aalbers B, Grabb P (2016) Hemiballismus as a complication of an intratumoral chemotherapy catheter. J Clin Neurosci 30:129–131. https://doi.org/10.1016/j.jocn.2016.01.031

    Article  PubMed  Google Scholar 

  44. Pettorini BL, Tamburrini G, Massimi L, Caldarelli M, Di Rocco C (2009) Endoscopic transventricular positioning of intracystic catheter for treatment of craniopharyngioma. Technical note. J Neurosurg Pediatr 4(3):245–248. https://doi.org/10.3171/2009.4.peds0978

    Article  PubMed  Google Scholar 

  45. Peyrl A, Chocholous M, Azizi AA et al (2014) Safety of Ommaya reservoirs in children with brain tumors: a 20-year experience with 5472 intraventricular drug administrations in 98 patients. J Neurooncol 120(1):139–145. https://doi.org/10.1007/s11060-014-1531-1

    Article  CAS  PubMed  Google Scholar 

  46. Kramer K, Smith M, Souweidane MM (2014) Safety profile of long-term intraventricular access devices in pediatric patients receiving radioimmunotherapy for central nervous system malignancies. Pediatr Blood Cancer 61(9):1590–1592. https://doi.org/10.1002/pbc.25080

    Article  CAS  PubMed  Google Scholar 

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Acknowledgements

We thank the entire interdisciplinary team, including our nurses, nurse practitioners and social workers, who were involved in the care of these patients.

Funding

The authors declare that no funds, grants, or other support were received for this study nor during the preparation of this manuscript.

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Authors and Affiliations

  1. Division of Neurosurgery, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada

    Laura-Nanna Lohkamp, Abhaya V. Kulkarni, James M. Drake, James T. Rutka, Peter B. Dirks, Michael Taylor & George M. Ibrahim

  2. Division of Haematology/Oncology, The Hospital for Sick Children, 555 University Ave, Toronto, ON, M5G2J9, Canada

    Ute K. Bartels

  3. Division of Endocrinology, The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada

    Jill Hamilton

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  1. Laura-Nanna Lohkamp
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by LNL. The first draft of the manuscript was written by LNL and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.

Corresponding author

Correspondence to Ute K. Bartels.

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Conflict of interest

The authors have no relevant financial or non-financial interests to disclose.

Ethical approval

The study was approved by the Research Ethics Board at Sickkids and conducted in accordance with their ethics guidelines and those of the University of Toronto.

Consent to participate

Due to the retrospective design of the study, the requirement for informed consent was waived by the REB.

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Lohkamp, LN., Kulkarni, A.V., Drake, J.M. et al. Preservation of endocrine function after Ommaya reservoir insertion in children with cystic craniopharyngioma. J Neurooncol 159, 597–607 (2022). https://doi.org/10.1007/s11060-022-04099-0

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