Journal of Neuro-Oncology

, Volume 116, Issue 1, pp 179–185

Gross total resection improves overall survival in children with choroid plexus carcinoma

  • Matthew Z. Sun
  • Michael E. Ivan
  • Aaron J. Clark
  • Michael C. Oh
  • Arthur R. Delance
  • Taemin Oh
  • Michael Safaee
  • Gurvinder Kaur
  • Orin Bloch
  • Annette Molinaro
  • Nalin Gupta
  • Andrew T. Parsa
Clinical Study

Abstract

Choroid plexus carcinoma (CPC) is a rare, malignant, primary brain tumor with a poor prognosis. While previous reports have shown benefits of aggressive surgery, very few large-scale studies have focused exclusively on the pediatric population, for whom the risks of aggressive surgery must be weighed carefully against the benefits. We performed a comprehensive systematic review of pediatric CPCs to test the effects of gross total resection (GTR) on overall survival (OS) and progression-free survival (PFS). A Pubmed search was performed to identify children with CPC who underwent surgical resection. Only disaggregated clinical cases in which extent of resection was confirmed by CT or MRI were included for analysis. Kaplan–Meier and multivariate Cox regression survival analyses were performed to determine the effects of extent of resection on OS and PFS. Disaggregated clinical data from a total of 102 pediatric CPC patients (age ≤18 years) with known extent of resection and overall survival were analyzed. GTR was significantly associated with better OS by Kaplan–Meier analysis (logrank p < 0.001). Multivariate Cox regression analysis adjusting for age, gender, tumor location (supratentorial vs. infratentorial), and type of adjuvant therapy (chemotherapy, radiation, and combined therapy), showed that GTR independently increased OS (p = 0.006). While GTR also improved PFS on Kaplan–Meier analysis (p = 0.027), the effect did not meet our criteria for significance in our multivariate Cox model (p = 0.120). GTR improved OS of pediatric CPC and is recommended if it can be safely performed.

Keywords

Choroid plexus carcinoma Extent of resection Gross total resection Overall survival Progression free survival Pediatric 

Abbreviations

GTR

Gross total resection

STR

Subtotal resection

OS

Overall survival

PFS

Progression free survival

EOR

Extent of resection

CI

Confidence interval

Supplementary material

11060_2013_1281_MOESM1_ESM.docx (43 kb)
Supplementary material 1 (DOCX 43 kb)

References

  1. 1.
    Berger C, Thiesse P, Lellouch-Tubiana A, Kalifa C, Pierre-Kahn A, Bouffet E (1998) Choroid plexus carcinomas in childhood: clinical features and prognostic factors. Neurosurgery 42:470–475PubMedCrossRefGoogle Scholar
  2. 2.
    Louis DN, Ohgaki H, Wiestler OD, Cavenee WK (eds) (2007) WHO classification of tumours of the central nervous system. International Agency for Research on Cancer, LyonGoogle Scholar
  3. 3.
    Fitzpatrick LK, Aronson LJ, Cohen KJ (2002) Is there a requirement for adjuvant therapy for choroid plexus carcinoma that has been completely resected? J Neurooncol 57:123–126PubMedCrossRefGoogle Scholar
  4. 4.
    Ogiwara H, Dipatri AJ, Alden TD, Bowman RM, Tomita T (2011) Choroid plexus tumors in pediatric patients. Br J Neurosurg 116(5):793–808Google Scholar
  5. 5.
    Packer RJ, Perilongo G, Johnson D, Sutton LN, Vezina G, Zimmerman RA, Ryan J, Reaman G, Schut L (1992) Choroid plexus carcinoma of childhood. Cancer 69:580–585PubMedCrossRefGoogle Scholar
  6. 6.
    Menon G, Nair SN, Baldawa SS, Rao RB, Krishnakumar KP, Gopalakrishnan CV (2010) Choroid plexus tumors: an institutional series of 25 patients. Neurol India 58:429–435PubMedCrossRefGoogle Scholar
  7. 7.
    Ellenbogen RG, Winston KR, Kupsky WJ (1989) Tumors of the choroid plexus in children. Neurosurgery 25:327–335PubMedCrossRefGoogle Scholar
  8. 8.
    Johnson DL (1989) Management of choroid plexus tumors in children. Pediatr Neurosci 15:195–206PubMedCrossRefGoogle Scholar
  9. 9.
    Pierga JY, Kalifa C, Terrier-Lacombe MJ, Habrand JL, Lemerle J (1993) Carcinoma of the choroid plexus: a pediatric experience. Med Pediatr Oncol 21:480–487PubMedCrossRefGoogle Scholar
  10. 10.
    Paulus W, Janisch W (1990) Clinicopathologic correlations in epithelial choroid plexus neoplasms: a study of 52 cases. Acta Neuropathol 80:635–641PubMedCrossRefGoogle Scholar
  11. 11.
    Rickert CH, Paulus W (2001) Tumors of the choroid plexus. Microsc Res Tech 52:104–111PubMedCrossRefGoogle Scholar
  12. 12.
    Gopal P, Parker JR, Debski R, Joseph C, Parker J (2008) Choroid plexus carcinoma. Arch Pathol Lab Med 192(8):1350–1354Google Scholar
  13. 13.
    Lafay-Cousin L, Keene D, Carret AS, Fryer C, Brossard J, Crooks B, Eisenstat D, Johnston D, Larouche V, Silva M, Wilson B, Zelcer S, Bartels U, Bouffet E (2011) Choroid plexus tumors in children less than 36 months: the Canadian Pediatric Brain Tumor Consortium (CPBTC) experience. Childs Nerv Syst 27:259–264PubMedCrossRefGoogle Scholar
  14. 14.
    Bettegowda C, Adogwa O, Mehta V, Chaichana KL, Weingart J, Carson BS, Jallo GI, Ahn ES (2012) Treatment of choroid plexus tumors: a 20-year single institutional experience. J Neurosurg Pediatr 10:398–405PubMedCrossRefGoogle Scholar
  15. 15.
    Tena-Suck ML, Salinas-Lara C, Rembao-Bojorquez D, Castillejos M (2010) Clinicopathologic and immunohistochemical study of choroid plexus tumors: single-institution experience in Mexican population. J Neurooncol 98:357–365PubMedCrossRefGoogle Scholar
  16. 16.
    Wolff JE, Sajedi M, Brant R, Coppes MJ, Egeler RM (2002) Choroid plexus tumours. Br J Cancer 87:1086–1091PubMedCentralPubMedCrossRefGoogle Scholar
  17. 17.
    Wrede B, Hasselblatt M, Peters O, Thall PF, Kutluk T, Moghrabi A, Mahajan A, Rutkowski S, Diez B, Wang X, Pietsch T, Kortmann RD, Paulus W, Jeibmann A, Wolff JE (2009) Atypical choroid plexus papilloma: clinical experience in the CPT-SIOP-2000 study. J Neurooncol 95:383–392PubMedCrossRefGoogle Scholar
  18. 18.
    Wrede B, Liu P, Wolff JE (2007) Chemotherapy improves the survival of patients with choroid plexus carcinoma: a meta-analysis of individual cases with choroid plexus tumors. J Neurooncol 85:345–351PubMedCrossRefGoogle Scholar

Copyright information

© Springer Science+Business Media New York 2013

Authors and Affiliations

  • Matthew Z. Sun
    • 1
  • Michael E. Ivan
    • 1
  • Aaron J. Clark
    • 1
  • Michael C. Oh
    • 1
  • Arthur R. Delance
    • 1
  • Taemin Oh
    • 3
  • Michael Safaee
    • 1
  • Gurvinder Kaur
    • 3
  • Orin Bloch
    • 3
  • Annette Molinaro
    • 1
  • Nalin Gupta
    • 1
    • 2
  • Andrew T. Parsa
    • 3
  1. 1.Department of Neurological SurgeryUniversity of California San FranciscoSan FranciscoUSA
  2. 2.Department of PediatricsUniversity of California San FranciscoSan FranciscoUSA
  3. 3.Department of Neurological Surgery, Feinberg School of MedicineNorthwestern UniversityChicagoUSA

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