An Individual Patient Data Meta-Analysis on Characteristics and Outcome of Patients with Papillary Glioneuronal Tumor, Rosette Glioneuronal Tumor with Neuropil-Like Islands and Rosette Forming Glioneuronal Tumor of the Fourth Ventricle
Background and Purpose In 2007, the WHO classification of brain tumors was extended by three new entities of glioneuronal tumors: papillary glioneuronal tumor (PGNT), rosette-forming glioneuronal tumor of the fourth ventricle (RGNT) and glioneuronal tumor with neuropil-like islands (GNTNI). Focusing on clinical characteristics and outcome, the authors performed a comprehensive individual patient data (IPD) meta-analysis of the cases reported in literature until December 2012. Methods PubMed, Embase and Web of Science were searched for peer-reviewed articles reporting on PGNT, RGNT, and GNTNI using predefined keywords. Results 95 publications reported on 182 patients (PGNT, 71; GNTNI, 26; RGNT, 85). Median age at diagnosis was 23 years (range 4–75) for PGNT, 27 years (range 6–79) for RGNT, and 40 years (range 2–65) for GNTNI. Ninety-seven percent of PGNT and 69% of GNTNI were located in the supratentorial region, 23% of GNTNI were in the spinal cord, and 80% of RGNT were localized in the posterior fossa. Complete resection was reported in 52 PGNT (73%), 36 RGNT (42%), and 7 GNTNI (27%) patients. Eight PGNT, 3 RGNT, and 12 GNTNI patients were treated with chemo- and/or radiotherapy as the primary postoperative treatment. Follow-up data were available for 132 cases. After a median follow-up time of 1.5 years (range 0.2–25) across all patients, 1.5-year progression-free survival rates were 52±12% for GNTNI, 86±5% for PGNT, and 100% for RGNT. The 1.5-year overall-survival were 95±5%, 98±2%, and 100%, respectively. Conclusions The clinical understanding of the three new entities of glioneuronal tumors, PGNT, RGNT and GNTNI, is currently emerging. The present meta-analysis will hopefully contribute to a delineation of their diagnostic, therapeutic, and prognostic profiles. However, the available data do not provide a solid basis to define the optimum treatment approach. Hence, a central register should be established.
References
[1]
Adachi J, Nishikawa R, Hirose T, Matsutani M (2005) Mixed neuronal-glial tumor of the fourth ventricle and successful treatment of postoperative mutism with bromocriptine: case report. Surg Neurol 63: 375–379. doi: 10.1016/j.surneu.2004.05.039
[2]
Agarwal S, Sharma MC, Singh G, Suri V, Sarkar C, et al. (2012) Papillary glioneuronal tumor - a rare entity: report of four cases and brief review of literature. Childs Nerv Syst 28: 1897–1904. doi: 10.1007/s00381-012-1860-3
[3]
Allende DS, Prayson RA (2009) The expanding family of glioneuronal tumors. Adv Anat Pathol 16: 33–39. doi: 10.1097/pap.0b013e3181915e3b
[4]
Amemiya S, Shibahara J, Aoki S, Takao H, Ohtomo K (2008) Recently established entities of central nervous system tumors: review of radiological findings. J Comput Assist Tomogr 32: 279–285. doi: 10.1097/rct.0b013e31814ce981
[5]
Anan M, Inoue R, Ishii K, Abe T, Fujiki M, et al. (2009) A rosette-forming glioneuronal tumor of the spinal cord: the first case of a rosette-forming glioneuronal tumor originating from the spinal cord. Hum Pathol 40: 898–901. doi: 10.1016/j.humpath.2008.11.010
[6]
Atri S, Sharma MC, Sarkar C, Garg A, Suri A (2007) Papillary glioneuronal tumour: a report of a rare case and review of literature. Childs Nerv Syst 23: 349–353. doi: 10.1007/s00381-006-0196-2
Benesch M, Eder HG, Sovinz P, Raith J, Lackner H, et al. (2006) Residual or recurrent cerebellar low-grade glioma in children after tumor resection: is re-treatment needed? A single center experience from 1983 to 2003. Pediatr Neurosurg 42: 159–164. doi: 10.1159/000091859
[9]
Bisson EF, Pendlebury WW, Horgan MA (2005) Glioneuronal tumor with unique imaging and histologic features. J Neurooncol 72: 89–90. doi: 10.1007/s11060-004-2277-y
[10]
Bouvier-Labit C, Daniel L, Dufour H, Grisoli F, Figarella-Branger D (2000) Papillary glioneuronal tumour: clinicopathological and biochemical study of one case with 7-year follow up. Acta Neuropathol 99: 321–326. doi: 10.1007/pl00007445
[11]
Broholm H, Madsen FF, Wagner AA, Laursen H (2002) Papillary glioneuronal tumor - a new tumor entity. Clin Neuropathol 21: 1–4.
[12]
Buccoliero AM, Castiglione F, Degl’innocenti DR, Moncini D, Paglierani M, et al. (2012) Glioneuronal tumor with neuropil-like islands: clinical, morphologic, immunohistochemical, and molecular features of three pediatric cases. Ped Dev Pathol 15: 352–360. doi: 10.2350/12-01-1147-oa.1
[13]
Celli P, Caroli E, Giangaspero F, Ferrante L (2006) Papillary glioneuronal tumor. Case report and literature review. J Neurooncol 80: 185–189. doi: 10.1007/s11060-006-9170-9
[14]
Collett D (2003) Modelling survival data in medical research. 2nd edition ed. London: Chapman and Hall.
Epelbaum S, Kujas M, Van Effenterre R, Poirier J (2006) Two cases of papillary glioneuronal tumours. Br J Neurosurg 20: 90–93. doi: 10.1080/02688690600682465
[17]
Fraum TJ, Barak S, Pack S, Lonser RR, Fine HA, et al. (2012) Spinal cord glioneuronal tumor with neuropil-like islands with 1p/19q deletion in an adult with low-grade cerebral oligodendroglioma. J Neurooncol 107: 421–426. doi: 10.1007/s11060-011-0760-9
[18]
Friedrich C, Müller K, von Hoff K, Kwiecien R, Rutkowski S, et al. (2014) Adults with CNS primitive neuroectodermal tumors/pineoblastomas: results of multimodal treatment according to the pediatric HIT 2000 protocol. J Neurooncol 116: 567–575. doi: 10.1007/s11060-013-1327-8
[19]
Frydenberg E, Laherty R, Rodriguez M, Ow-Yang M, Steel T (2010) A rosette-forming glioneuronal tumour of the pineal gland. J Clin Neurosci 17: 1326–1328. doi: 10.1016/j.jocn.2010.03.003
[20]
Gelpi E, Preusser M, Czech T, Slavc I, Prayer D, et al. (2007) Papillary glioneuronal tumor. Neuropathology 27: 468–473. doi: 10.1111/j.1440-1789.2007.00802.x
[21]
Harris BT, Horoupian DS (2000) Spinal cord glioneuronal tumor with “rosette” neuropil islands and meningeal dissemination: a case report. Acta neuropathol 100: 575–579. doi: 10.1007/s004010000217
[22]
Ishizawa T, Komori T, Shibahara J, Ishizawa K, Adachi J, et al. (2006) Papillary glioneuronal tumor with minigemistocytic components and increased proliferative activity. Hum Pathol 37: 627–630. doi: 10.1016/j.humpath.2005.12.014
[23]
Javahery RJ, Davidson L, Fangusaro J, Finlay JL, Gonzalez-Gomez I, et al. (2009) Aggressive variant of a papillary glioneuronal tumor. Report of 2 cases. J Neurosurg Pediatr 3: 46–52. doi: 10.3171/2008.10.peds08242
[24]
Komori T, Scheithauer BW, Anthony DC, Rosenblum MK, McLendon RE, et al. (1998) Papillary glioneuronal tumor: a new variant of mixed neuronal-glial neoplasm. Am J Surg Pathol 22: 1171–1183. doi: 10.1097/00000478-199810000-00002
[25]
Komori T, Scheithauer BW, Hirose T (2002) A rosette-forming glioneuronal tumor of the fourth ventricle: infratentorial form of dysembryoplastic neuroepithelial tumor? Am J Surg Pathol 26: 582–591. doi: 10.1097/00000478-200205000-00004
[26]
Kuchelmeister K, Demirel T, Schlorer E, Bergmann M, Gullotta F (1995) Dysembryoplastic neuroepithelial tumour of the cerebellum. Acta neuropathol 89: 385–390. doi: 10.1007/bf00309634
[27]
Lamszus K, Makrigeorgi-Butera M, Laas R, Westphal M, Stavrou D (2003) September 2002: 24-year-old female with a 6-month history of seizures. Brain Pathol 13: 115–117.
[28]
Lavrnic S, Macvanski M, Ristic-Balos D, Gavrilov M, Damjanovic D, et al. (2012) Papillary glioneuronal tumor: unexplored entity. J Neurol Surg A Cent Eur Neurosurg 73: 224–229. doi: 10.1055/s-0032-1313636
[29]
Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC, et al. (2007) The 2007 WHO classification of tumours of the central nervous system. Acta neuropathol 114: 97–109. doi: 10.1007/s00401-007-0243-4
[30]
Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC, et al. (2007) WHO Classification of tumours of the central nervous system. Lyon, IARC Press.
[31]
Lu JQ, Scheithauer BW, Sharma P, Scott JN, Parney IF, et al. (2009) Multifocal complex glioneuronal tumor in an elderly man: an autopsy study: case report. Neurosurgery 64: E1193–1195. doi: 10.1227/01.neu.0000345640.40566.48
[32]
Malats N, Bustos A, Nascimento CM, Fernandez F, Rivas M, et al. (2005) P53 as a prognostic marker for bladder cancer: a meta-analysis and review. Lancet Oncol 6: 678–686. doi: 10.1016/s1470-2045(05)70315-6
[33]
Mazloom A, Hodges JC, Teh BS, Chintagumpala M, Paulino AC (2012) Outcome of patients with pilocytic astrocytoma and leptomeningeal dissemination. Int J Radiat Oncol Biol Phys 84: 350–354. doi: 10.1016/j.ijrobp.2011.12.044
[34]
Moher D, Liberati A, Tetzlaff J, Altman DG (2009) Group P (2009) Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. J Clin Epidemiol 62: 1006–1012. doi: 10.1016/j.jclinepi.2009.06.005
[35]
Newton HB, Dalton J, Ray-Chaudhury A, Gahbauer R, McGregor J (2008) Aggressive papillary glioneuronal tumor: case report and literature review. Clin Neuropathol 27: 317–324. doi: 10.5414/npp27317
[36]
Peduzzi P, Concato J, Feinstein AR, Holford TR (1995) Importance of events per independent variable in proportional hazards regression analysis. II. Accuracy and precision of regression estimates. J Clin Epidemiol 48: 1503–1510. doi: 10.1016/0895-4356(95)00048-8
[37]
Poliani PL, Sperli D, Valentini S, Armentano A, Bercich L, et al. (2009) Spinal glioneuronal tumor with neuropil-like islands and meningeal dissemination: histopathological and radiological study of a pediatric case. Neuropathology 29: 574–578. doi: 10.1111/j.1440-1789.2008.00988.x
[38]
Prayson RA (2000) Papillary glioneuronal tumor. Arch Path Lab Med 124: 1820–1823.
[39]
Rainov NG, Wagner T, Heidecke V (2010) Rosette-forming glioneuronal tumor of the fourth ventricle. Cent Eur Neurosurg 71: 219–221. doi: 10.1055/s-0029-1242760
[40]
Rosenblum MK (2007) The 2007 WHO Classification of Nervous System Tumors: newly recognized members of the mixed glioneuronal group. Brain Pathol 17: 308–313. doi: 10.1111/j.1750-3639.2007.00079.x
[41]
Ruppert B, Welsh CT, Hannah J, Giglio P, Rumboldt Z, et al. (2011) Glioneuronal tumor with neuropil-like islands of the spinal cord with diffuse leptomeningeal neuraxis dissemination. J Neurooncol 104: 529–533. doi: 10.1007/s11060-010-0505-1
[42]
Scheithauer BW, Silva AI, Ketterling RP, Pula JH, Lininger JF, et al. (2009) Rosette-forming glioneuronal tumor: report of a chiasmal-optic nerve example in neurofibromatosis type 1: special pathology report. Neurosurgery 64: E771–772. doi: 10.1227/01.neu.0000340979.81362.f3
[43]
Shah MN, Leonard JR, Perry A (2010) Rosette-forming glioneuronal tumors of the posterior fossa. J Neurosurg Pediatr 5: 98–103. doi: 10.3171/2009.7.peds09113
[44]
Simmonds MC, Higgins JP, Stewart LA, Tierney JF, Clarke MJ, et al. (2005) Meta-analysis of individual patient data from randomized trials: a review of methods used in practice. Clin Trials 2: 209–217. doi: 10.1191/1740774505cn087oa
[45]
Solis OE, Mehta RI, Lai A, Mehta RI, Farchoukh LO, et al. (2011) Rosette-forming glioneuronal tumor: a pineal region case with IDH1 and IDH2 mutation analyses and literature review of 43 cases. J Neurooncol 102: 477–484. doi: 10.1007/s11060-010-0335-1
[46]
Teo JG, Gultekin SH, Bilsky M, Gutin P, Rosenblum MK (1999) A distinctive glioneuronal tumor of the adult cerebrum with neuropil-like (including “rosette”) islands: report of 4 cases. Am J Surg Pathol 23: 502–510. doi: 10.1097/00000478-199905000-00002
Wang Y, Xiong J, Chu SG, Liu Y, Cheng HX, et al. (2009) Rosette-forming glioneuronal tumor: report of an unusual case with intraventricular dissemination. Acta neuropathol 118: 813–819. doi: 10.1007/s00401-009-0569-1
[49]
Williams SR, Joos BW, Parker JC, Parker JR (2008) Papillary glioneuronal tumor: a case report and review of the literature. Ann Clin Lab Sci 38: 287–292.
[50]
Xiao H, Ma L, Lou X, Gui Q (2011) Papillary glioneuronal tumor: radiological evidence of a newly established tumor entity. J Neuroimaging 21: 297–302. doi: 10.1111/j.1552-6569.2010.00478.x
[51]
Xiong J, Liu Y, Chu SG, Chen H, Chen HX, et al. (2012) Rosette-forming glioneuronal tumor of the septum pellucidum with extension to the supratentorial ventricles: rare case with genetic analysis. Neuropathology 32: 301–305. doi: 10.1111/j.1440-1789.2011.01261.x
[52]
Zhang J, Babu R, McLendon RE, Friedman AH, Adamson C (2013) A comprehensive analysis of 41 patients with rosette-forming glioneuronal tumors of the fourth ventricle. J Clin Neurosci 20: 335–341. doi: 10.1016/j.jocn.2012.09.003
[53]
Zwiener I, Blettner M, Hommel G (2011) Survival analysis: part 15 of a series on evaluation of scientific publications. Dtsch Arztebl Int 108: 163–169.
