Primary brain and other central nervous system tumors in Appalachia: regional differences in incidence, mortality, and survival
The Appalachian region is a large geographic and economic area, representing 7.69% of the United States (US). This region is more rural, whiter, older, and has a higher level of poverty as compared to the rest of the US. Limited research has been done on primary brain and other central nervous system tumors (PBT) epidemiology in this region. In this analysis we characterize incidence, mortality, and survival patterns.
Data from 2006 to 2015 were obtained from the central brain tumor registry of the US (provided by CDC and NCI). Appalachian counties were categorized using the Appalachia Regional Council scheme. Overall and histology-specific age-adjusted incidence and mortality rates per 100,000 population were generated. 1-, 5-, and 10-year relative survival (RS) was estimated using CDC national program of cancer registry data from 2001 to 2014.
Overall PBT incidence within Appalachia was 22.62 per 100,000, which is not significantly different from the non-Appalachian US (22.77/100,000, p = 0.1189). Malignant incidence was 5% higher in Appalachia (7.55/100,000 vs. 7.23/100,000, p < 0.0001), while non-malignant incidence was 3% lower (15.07/100,000 vs. 15.54/100,000, p < 0.0001). 5-year RS for malignant PBT was lower (31.4% vs. 36.0%), and mortality due to malignant PBT was higher in Appalachia (4.86/100,000 vs. 4.34/100,000, p < 0.0001).
Appalachia has increased malignant and decreased non-malignant PBT incidence, and poorer survival outcomes for malignant PBT compared to the non-Appalachian US.
KeywordsBrain and CNS tumors Incidence Relative survival Mortality Appalachia
This work was previously presented at the 2018 annual meeting of the Society for Neuro-Oncology.
Conceptualization: QTO. Formal analysis: QTO. Data curation: QTO, HG. Writing—original draft: QTO. Writing - review and editing: QTO, HG, CK, JSB. Funding acquisition: CK, JSB.
QTO is supported by a Research Training Grant from the Cancer Prevention and Research Institute of Texas (CPRIT; RP160097T). Funding for CBTRUS was provided by the Centers for Disease Control and Prevention (CDC) under Contract No. 2016-M-9030, the American Brain Tumor Association, The Sontag Foundation, Novocure, Abbvie, the Musella Foundation, and the National Cancer Institute (NCI) under contract No. HHSN261201800176P, as well as private and in kind donations. Contents are solely the responsibility of the authors and do not necessarily reflect the official views of the CDC or the NCI.
Compliance with ethical standards
Conflict of interest
The authors declare that they have no conflict of interest.
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. This study was approved by the Institutional Review Board at University Hospitals Cleveland Medical Center.
This was a retrospective study using de-identified national cancer registries. Thus, formal consent was not required.
- 3.Ostrom QT, Bauchet L, Davis FG, Deltour I, Fisher JL, Langer CE, Pekmezci M, Schwartzbaum JA, Turner MC, Walsh KM, Wrensch MR, Barnholtz-Sloan JS (2014) The epidemiology of glioma in adults: a “state of the science” review. Neuro Oncol 16:896–913. https://doi.org/10.1093/neuonc/nou087 CrossRefGoogle Scholar
- 4.Amirian ES, Ostrom QT, Liu Y, Barnholtz-Sloan J, Bondy ML (2017) Nervous System. In: Thun M, Linet MS, Cerhan JR, Haiman CA, Schottenfeld D (eds) Cancer epidemiology and prevention, 4 edn. Oxford University Press, New YorkGoogle Scholar
- 5.Appalachian Regional Commission Counties in Appalachia. https://www.arc.gov/appalachian_region/CountiesinAppalachia.asp
- 6.Marshall JL, Thomas L, Lane NM, Arcury TA, Randolph R, Silberman P, Holding W, Villamil L, Sharita T, Lane M, Latus J, Rodgers J, Ivey K (2017) Health disparities in appalachia. Creating a culture of health in appalachia. Appalachian Regional Commission. The University of North Carolina at Chapel Hill, Chapel HillGoogle Scholar
- 7.Pollard K, Jacobsen LA, Population Reference Bureau (2017) The Appalachian Region: A Data Overview from the 2011–2015 American Community Survey. Population Reference BureauGoogle Scholar
- 10.Meit M, Heffernan M, Tanenbaum E, Hoffmann T, The Walsh Center for Rural Health Analysis (2007) Final report: appalachian diseases of despair. https://www.arc.gov/assets/research_reports/AppalachianDiseasesofDespairAugust2017.pdf
- 14.Paskett ED, Fisher JL, Lengerich EJ, Schoenberg NE, Kennedy SK, Conn ME, Roberto KA, Dwyer SK, Fickle D, Dignan M (2011) Disparities in underserved white populations: the case of cancer-related disparities in Appalachia. Oncologist 16:1072–1081. https://doi.org/10.1634/theoncologist.2011-0145 CrossRefGoogle Scholar
- 15.Fisher JL, Engelhardt HL, Stephens JA, Smith BR, Haydu GG, Indian RW, Paskett ED (2008) Cancer-related disparities among residents of Appalachia Ohio. J Health Disparities Res Pract 2:61–74Google Scholar
- 17.Aldrich TE, Freitas SJ, Ling L, McKinney P (2008) Brain cancer survival in Kentucky: 1996–2000. J Ky Med Assoc 106:489–493Google Scholar
- 18.U.S. Cancer Statistics Working Group (2018) United States Cancer Statistics: 1999–2015 Incidence and Mortality Web-based Report. U.S. Department of Health and Human Services, Centers for Disease Control and Prevention and National Cancer Institute,. http://www.cdc.gov/uscs
- 19.Appalachian Regional Commission Subregions in Appalachia. https://www.arc.gov/research/MapsofAppalachia.asp?MAP_ID=31
- 20.Surveillance Epidemiology and End Results (SEER) Program (2016) SEER*Stat software version 8.3.2. National Cancer Institute, DCCPS, Surveillance Research Program. http://www.seer.cancer.gov/seerstat
- 22.Surveillance Epidemiology and End Results (SEER) Program SEER*Stat Database: Mortality - All COD, Aggregated With State, Total U.S. (1969–2015) < Katrina/Rita Population Adjustment>, National Cancer Institute, DCCPS, Surveillance Research Program, Surveillance Systems Branch, released April 2018. Underlying mortality data provided by NCHS (http://www.cdc.gov/nchs).
- 23.Surveillance Epidemiology and End Results (SEER) Program overview of the SEER program. Natl Cancer Inst http://seer.cancer.gov/about/overview.html
- 24.Surveillance Epidemiology and End Results (SEER) Program (2015) Number of Persons by Race and Hispanic Ethnicity for SEER Participants (2010 Census Data). http://seer.cancer.gov/registries/data.html
- 25.Surveillance Epidemiology and End Results (SEER) Program (2017) SEER*Stat Database: Incidence - SEER 18 Regs Custom Data (with additional treatment fields), Nov 2016 Sub (2000–2014) < Katrina/Rita Population Adjustment> - Linked To County Attributes - Total U.S., 1969–2015 Counties, National Cancer Institute, DCCPS, Surveillance Research Program, released April 2017, based on the November 2016 submission.Google Scholar
- 27.R Core Team (2018) R: A language and environment for statistical computing. R Foundation for Statistical Computing. http://www.R-project.org/
- 28.Program JR, Version 220.127.116.11 - June 2015; Statistical methodology and applications branch, surveillance research program, National Cancer InstituteGoogle Scholar
- 35.Nilsson J, Holgersson G, Jaras J, Bergstrom S, Bergqvist M (2018) The role of income in brain tumor patients: a descriptive register-based study: no correlation between patients’ income and development of brain cancer. Med Oncol 35:52. https://doi.org/10.1007/s12032-018-1108-5 CrossRefGoogle Scholar
- 42.Shiels MS, Engels EA, Linet MS, Clarke CA, Li J, Hall HI, Hartge P, Morton LM (2013) The epidemic of non-Hodgkin lymphoma in the United States: disentangling the effect of HIV, 1992–2009. Cancer Epidemiol Biomark Prev 22:1069–1078. https://doi.org/10.1158/1055-9965.epi-13-0040 CrossRefGoogle Scholar
- 46.Bos D, Poels MM, Adams HH, Akoudad S, Cremers LG, Zonneveld HI, Hoogendam YY, Verhaaren BF, Verlinden VJ, Verbruggen JG, Peymani A, Hofman A, Krestin GP, Vincent AJ, Feelders RA, Koudstaal PJ, van der Lugt A, Ikram MA, Vernooij MW (2016) Prevalence, clinical management, and natural course of incidental findings on brain mr images: the population-based rotterdam scan study. Radiology 281:507–515. https://doi.org/10.1148/radiol.2016160218 CrossRefGoogle Scholar
- 48.Morris Z, Whiteley WN, Longstreth WT Jr, Weber F, Lee YC, Tsushima Y, Alphs H, Ladd SC, Warlow C, Wardlaw JM, Al-Shahi Salman R (2009) Incidental findings on brain magnetic resonance imaging: systematic review and meta-analysis. BMJ 339:b3016. https://doi.org/10.1136/bmj.b3016 CrossRefGoogle Scholar
- 49.Bruner JM, Louis DN, McLendon R, Rosenblum MK, Archambault WT, Most S, Tihan T (2017) The utility of expert diagnosis in surgical neuropathology: analysis of consultations reviewed at 5 national comprehensive cancer network institutions. J Neuropathol Exp Neurol 76:189–194. https://doi.org/10.1093/jnen/nlw122 Google Scholar
- 50.Louis DN, Perry A, Reifenberger G, von Deimling A, Figarella-Branger D, Cavenee WK, Ohgaki H, Wiestler OD, Kleihues P, Ellison DW (2016) The 2016 World Health Organization classification of tumors of the central nervous system: a summary. Acta Neuropathol 131:803–820. https://doi.org/10.1007/s00401-016-1545-1 CrossRefGoogle Scholar