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Journal of Medical Genetics logoLink to Journal of Medical Genetics
. 2005 Apr 14;42(12):903–906. doi: 10.1136/jmg.2005.031302

Methodological issues in longitudinal studies: vestibular schwannoma growth rates in neurofibromatosis 2

M Baser 1, V Mautner 1, D Parry 1, D Evans 1
PMCID: PMC1735973  PMID: 15831594

Abstract

Four longitudinal studies of vestibular schwannoma (VS) growth rates in neurofibromatosis 2 (NF2) have yielded very different results on the relationship of VS growth rates to age. The studies had different patient eligibility criteria, indices of VS growth rates, VS volumetric methods, and sample sizes. We reanalysed data from two of the longitudinal studies and used data from the population based United Kingdom NF2 Registry to determine the most likely reason for the different results and the actual relationship of VS growth rates to age. We found that the eligibility criterion in one study caused selection bias for slower growing VS. The proper interpretation of the results from the four studies is that VS growth rates in NF2 are highly variable but tend to decrease with increasing age. Clinical trials for VS in NF2 should focus on younger patients because VS growth rates tend to decrease with increasing age, and because faster growing VS are more likely to be excised with increasing age than slower growing VS.

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Selected References

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  1. Abaza M. M., Makariou E., Armstrong M., Lalwani A. K. Growth rate characteristics of acoustic neuromas associated with neurofibromatosis type 2. Laryngoscope. 1996 Jun;106(6):694–699. doi: 10.1097/00005537-199606000-00007. [DOI] [PubMed] [Google Scholar]
  2. Antinheimo J., Haapasalo H., Seppälä M., Sainio M., Carpen O., Jäskeläinen J. Proliferative potential of sporadic and neurofibromatosis 2-associated schwannomas as studied by MIB-1 (Ki-67) and PCNA labeling. J Neuropathol Exp Neurol. 1995 Nov;54(6):776–782. doi: 10.1097/00005072-199511000-00004. [DOI] [PubMed] [Google Scholar]
  3. Baser Michael E., Friedman J. M., Aeschliman Dana, Joe Harry, Wallace Andrew J., Ramsden Richard T., Evans D. Gareth R. Predictors of the risk of mortality in neurofibromatosis 2. Am J Hum Genet. 2002 Aug 22;71(4):715–723. doi: 10.1086/342716. [DOI] [PMC free article] [PubMed] [Google Scholar]
  4. Baser Michael E., Kuramoto Lisa, Joe Harry, Friedman J. M., Wallace Andrew J., Gillespie James E., Ramsden Richard T., Evans D. Gareth R. Genotype-phenotype correlations for nervous system tumors in neurofibromatosis 2: a population-based study. Am J Hum Genet. 2004 Jun 9;75(2):231–239. doi: 10.1086/422700. [DOI] [PMC free article] [PubMed] [Google Scholar]
  5. Baser Michael E., Makariou Erini V., Parry Dilys M. Predictors of vestibular schwannoma growth in patients with neurofibromatosis Type 2. J Neurosurg. 2002 Feb;96(2):217–222. doi: 10.3171/jns.2002.96.2.0217. [DOI] [PubMed] [Google Scholar]
  6. Evans D. G., Huson S. M., Donnai D., Neary W., Blair V., Newton V., Harris R. A clinical study of type 2 neurofibromatosis. Q J Med. 1992 Aug;84(304):603–618. [PubMed] [Google Scholar]
  7. Mautner V. F., Lindenau M., Baser M. E., Hazim W., Tatagiba M., Haase W., Samii M., Wais R., Pulst S. M. The neuroimaging and clinical spectrum of neurofibromatosis 2. Neurosurgery. 1996 May;38(5):880–886. doi: 10.1097/00006123-199605000-00004. [DOI] [PubMed] [Google Scholar]
  8. Mautner Victor-Felix, Baser Michael E., Thakkar Sarang D., Feigen Urs M., Friedman J. M., Kluwe Lan. Vestibular schwannoma growth in patients with neurofibromatosis Type 2: a longitudinal study. J Neurosurg. 2002 Feb;96(2):223–228. doi: 10.3171/jns.2002.96.2.0223. [DOI] [PubMed] [Google Scholar]
  9. Nutik S. L., Babb M. J. Determinants of tumor size and growth in vestibular schwannomas. J Neurosurg. 2001 Jun;94(6):922–926. doi: 10.3171/jns.2001.94.6.0922. [DOI] [PubMed] [Google Scholar]
  10. Ogawa K., Kanzaki J., Ogawa S., Yamamoto M., Ikeda S., Shiobara R. The growth rate of acoustic neuromas. Acta Otolaryngol Suppl. 1991;487:157–163. doi: 10.3109/00016489109130462. [DOI] [PubMed] [Google Scholar]
  11. Parry D. M., Eldridge R., Kaiser-Kupfer M. I., Bouzas E. A., Pikus A., Patronas N. Neurofibromatosis 2 (NF2): clinical characteristics of 63 affected individuals and clinical evidence for heterogeneity. Am J Med Genet. 1994 Oct 1;52(4):450–461. doi: 10.1002/ajmg.1320520411. [DOI] [PubMed] [Google Scholar]
  12. Rosenberg S. I., Silverstein H., Gordon M. A., Flanzer J. M., Willcox T. O., Silverstein J. A comparison of growth rates of acoustic neuromas: nonsurgical patients vs. subtotal resection. Otolaryngol Head Neck Surg. 1993 Sep;109(3 Pt 1):482–487. doi: 10.1177/019459989310900316. [DOI] [PubMed] [Google Scholar]
  13. Slattery William H., 3rd, Fisher Laurel M., Iqbal Zarina, Oppenhiemer Mark. Vestibular schwannoma growth rates in neurofibromatosis type 2 natural history consortium subjects. Otol Neurotol. 2004 Sep;25(5):811–817. doi: 10.1097/00129492-200409000-00027. [DOI] [PubMed] [Google Scholar]
  14. Sobel R. A. Vestibular (acoustic) schwannomas: histologic features in neurofibromatosis 2 and in unilateral cases. J Neuropathol Exp Neurol. 1993 Mar;52(2):106–113. doi: 10.1097/00005072-199303000-00002. [DOI] [PubMed] [Google Scholar]

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