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Neuroblastoma Screening Data: An Epidemiologic Analysis

Neuroblastoma Screening Data: An Epidemiologic Analysis Abstract • Neuroblastoma is one of the most common malignant neoplasms in children under age 5 years. Little progress has been made in the prognosis of advanced stage disease in the past three decades. Since the development in the 1960s of a simple urine test to detect neuroblastoma metabolites, there has been hope that mortality from this disease could be reduced by early detection via mass screening of young infants. Encouraging reports from Japan on mass screening programs instituted in the 1970s have been appearing in the medical literature since 1982, resulting in widespread interest in screening. This article applies standard epidemiologic criteria for screening evaluations to the Japanese reports. We find that the data needed to definitively assess the value of screening were not a part of those reports and that the benefits claimed from the reported data could be due to overdiagnosis. In addition, the most recent Japanese data, combined with recent advances in biologic understanding of neuroblastoma, show that screening at age 6 months may not detect tumors with poor prognoses. Even if it could, it is uncertain whether those outcomes could be substantially altered by earlier diagnosis. Although a final verdict on the value of neuroblastoma screening is not yet possible, these neuroblastoma studies are an excellent example of how screening results must be viewed with extreme caution in the absence of age-specific, population-based incidence and mortality rates. (AJDC. 1991;145:1415-1422) References 1. Young J, Reis L, Silverberg E, Horm J, Miller R. Cancer incidence, survival, and mortality for children younger than age 15 years . Cancer . 1986;58:598-602.Crossref 2. Woods W, Tuchman M. Neuroblastoma: the case for screening infants in North America . Pediatrics . 1987;79:869-873. 3. Carlsen N, Schroeder H, Bro P, et al. Neuroblastoma treated at the four major child oncologic clinics in Denmark 1943-80: an evaluation of 180 cases . Med Pediatr Oncol . 1985;13:180-186.Crossref 4. Kinnier Wilson L, Draper G. Neuroblastoma, its natural history and prognosis: a study of 487 cases . BMJ . 1974;3:301-307.Crossref 5. LaBrosse EH. Biochemical diagnosis of neuroblastoma: use of a urine spot test . Proc Am Assoc Cancer Res . 1968;9:39. 6. Sawada T, Todo S, Fujita K, lino S, Imashuku S, Kusunoki T. Mass screening of neuroblastoma in infancy . AJDC . 1982;136:710-712. 7. Tuchman M, Lemieux B, Auray-Blais C, et al. Screening for neuroblastoma in North America: preliminary results from the Quebec project . Pediatrics . 1990;86:765-773. 8. Craft A, Dale C, McGill A, Seviour J, Spence E. Biochemical screening for neuroblastoma in infants: a feasibility study . Med Pediatr Oncol . 1989;17:373-378.Crossref 9. Gellis S. Isn't it time for mass screening of infants for neuroblastoma? Pediatr Notes . 1990;14:39. 10. Mauer A. Screening for neuroblastoma . J Pediatr . 1988;112:576-577.Crossref 11. Tuchman M, Lemieux B, Woods WB. Screening for neuroblastoma in infants: investigate or implement? Pediatrics . 1990;86:791-793. 12. Murphy S, Cohn S, Craft A, et al. Do children benefit from mass screening for neuroblastoma? consensus statement for the American Cancer Society Workshop on Neuroblastoma Screening . Lancet . 1991;337:344-346.Crossref 13. Davis S, Rogers M, Prendergrass T. The incidence and epidemiologic characteristics of neuroblastoma in the United States . Am J Epidemiol . 1987;126:1063-1074. 14. Breslow N, McCann B. Statistical estimation of prognosis for children with neuroblastoma . Cancer Res . 1971;31: 2098-2103. 15. Coldman A, Fryer C, Elwood J, Sonley M. Influence of age at diagnosis, stage, tumor site, and sex on prognosis . Cancer . 1980;46:1896-1901.Crossref 16. Evans A, Chatten J, D'Angio G, Gerson J, Robinson J, Schnauffer L. A review of 17 IV-S neuroblastoma patients at the children's hospital of Philadelphia . Cancer . 1989;45:833-839.Crossref 17. Haas D, Ablin A, Miller C, et al. Complete pathologic maturation and regression of stage IV-S neuroblastoma without treatment . Cancer . 1988;62:818-825.Crossref 18. Beckwith J, Perrin E. In situ neuroblastomas: a contribution to the natural history of neural crest tumors . Am J Pathol . 1963;43:1089-1104. 19. Brodeur G, Seeger R, Schwab M, Varmus HE, Bishop JM. Amplification of n-myc in untreated human neuroblastomas correlates with advanced disease stage . Science . 1984;224:1121-1124.Crossref 20. Look A, Hayes F, Noitschke R, McWilliams N, Green A. Cellular DNA content as a predictor of response to chemotherapy in infants with unresectable neuroblastoma . N Engl J Med . 1984;311:231-235.Crossref 21. Hayashi Y, Inaba T, Hanada R, Yamamoto K. Chromosome findings and prognosis in 15 patients with neuroblastoma found by VMA mass screening . J Pediatr . 1988;112:567-571.Crossref 22. Kaneko Y, Kanda N, Maseki N, et al. Different karyotypic patterns in early and advanced stage neuroblastomas . Cancer Res . 1987;47:311-318. 23. Oppedal B, Storm-Mathisen I, Lie S, Brandtzaeb P. Prognostic factors in neuroblastoma: clinical, histopathologic, and immunohistochemical features and DNA ploidy in relation to prognosis . Cancer . 1988;62:772-780.Crossref 24. Nakagawara A. Genomic aspects of neuroblastomas in children under one year of age . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:47. 25. Nakagawara A, Ikeda K, Tsuda T, Higashi K. Biological characteristics of n-myc amplified neuroblastoma in patients over one year of age . Adv Neuroblastoma Res . 1988;2:31-39. 26. Nakagawara A, Ikeda K, Tsuda T, Higashi K. N-myc oncogene amplification and prognostic factors of neuroblastoma in children . J Pediatr Surg . 1987;22:895-898.Crossref 27. Tsuda T, Obara M, Hirano H, et al. Analysis of n-myc amplification in relation to disease stage and histologic types in human neuroblastomas . Cancer . 1987;60:820-826.Crossref 28. Bourhis J, Benard J, Wilson G, et al. Prognostic value of ploidic index and N-myc DNA amplification in 59 neuroblastomas . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:10. 29. Brodeur G. Clinical significance of genetic rearrangements in human neuroblastomas . Clin Chem . 1989;35/7B:B38-B42. 30. Brodeur G. The involvement of oncogenes and suppressor genes in human neoplasia . Adv Pediatr . 1987;34:1-44. 31. Auray-Blais C, Giguere R, Lemieux B. Thin-layer chromatography of urinary homovanillic acid and vanillylmandelic acid for large-scale neuroblastoma mass screening . Med Pediatr Oncol . 1989;17:364-367.Crossref 32. Matsumoto M, Anazawa A, Suzuki K, et al. Urine mass screening for neuroblastoma by high performance liquid chromatography (HPLC) . Pediatr Res . 1985;19:625.Crossref 33. Sato Y, Hanai J, Takasugi N, Takeda T. Determination of urinary vanillylmandelic acid and homovanillic acid in urine on filter paper for mass screening of neuroblastoma in infants . Tohoku J Exp Med . 1986;150:169-174.Crossref 34. Sawada T, Kidowaki T, Sakamoto I, et al. Neuroblastoma: mass screening for early detection and its prognosis . Cancer . 1984;53:2731-2735.Crossref 35. Sawada T, Kidowaki T, Sugimoto T, Kusunoki T. Incidence of neuroblastoma in infancy in Japan . Med Pediatr Oncol . 1984;12:101-103.Crossref 36. Sawada T, Nakaza T, Takasugi N. Mass screening for neuroblastoma in infants in Japan . Lancet . 1984;2:271-273.Crossref 37. Takeda T. History and current status of neuroblastoma screening in Japan . Med Pediatr Oncol . 1989;17:361-363.Crossref 38. Sawada T. Outcome of 25 neuroblastomas revealed by mass screening in Japan . Lancet . 1986;1:377.Crossref 39. Nishi M, Miyake H, Takeda T, et al. Effects of the mass screening of neuroblastoma in Sapporo City . Cancer . 1987;60:433-436.Crossref 40. Sawada T, Kawakatu H, Sugimoto T. Screening for neuroblastoma . Lancet . 1987;2:1204.Crossref 41. Sawada T, Sugimoto T, Matsumura T, Tunoda A, Takeda T, et al. Mass screening for neuroblastoma in infancy . In: Advances in Neuroblastoma Research 2 . New York, NY: Alan R Liss Inc; 1988:525-534. 42. Nishi M, Hirotsugo M, Takeda T, et al. Cases of neuroblastoma missed by the mass screening programs . Pediatr Res . 1989;26:603-607.Crossref 43. Naito H, Sasaki M, Yamashiro K, et al. Improvement in prognosis of neuroblastoma through mass population screening . J Pediatr Surg . 1990;25:245-248.Crossref 44. Cole P, Morrison AS. Basic issues in population screening for Cancer . J Natl Cancer Inst . 1980;64:1263-1272. 45. Miller A. Screening for cancer: issues and future directions . J Chronic Dis . 1986;39:1067-1077.Crossref 46. Sawada T, Kawakatus H, Sugimoto T, et al. Neuroblastoma mass screening in infancy in Kyoto, Japan . Indian J Pediatr. 1987;54:874-882.Crossref 47. Kramer S, Meadows A, Jarnett P, Evans A. Incidence of childhood cancer: experience of a decade in a population-based registry . J Natl Cancer Inst . 1983;70:49-55. 48. Evans A, D'Angio G, Propert K, Anderson J, Hahn H-W. Prognostic factors in neuroblastoma . Cancer . 1987;59:1853-1859.Crossref 49. Kaneko Y, Maseki N, Sakurai M, et al. Chromosomes and screening for neuroblastoma . Lancet . 1988;1:174-175.Crossref 50. Ishimoto K, Kiyokawa N, Ohya T, Miyano T, Shinohara T, Sera Y. Biological analysis of neuroblastoma in mass screened negative cases . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:117. 51. Kaneko K, Kanda N, Maseki N, et al. Current urinary mass screening for catecholamine metabolites at 6 months of age may be detecting only a small portion of high-risk neuroblastomas: a chromosome and N-myc amplification study . J Clin Oncol . 1990;8:2005-2013. 52. Brodeur G, Hayes A, Green A, et al. Consistent n-myc copy number in simultaneous or consecutive neuroblastoma samples from sixty individual patients . Cancer Res . 1987; 47:4248-4253. 53. Holtzman NA. What drives neonatal screening programs? N Engl J Med . 1991;325:802-804.Crossref http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png American Journal of Diseases of Children American Medical Association

Neuroblastoma Screening Data: An Epidemiologic Analysis

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American Medical Association
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Copyright © 1991 American Medical Association. All Rights Reserved.
ISSN
0002-922X
DOI
10.1001/archpedi.1991.02160120083024
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Abstract

Abstract • Neuroblastoma is one of the most common malignant neoplasms in children under age 5 years. Little progress has been made in the prognosis of advanced stage disease in the past three decades. Since the development in the 1960s of a simple urine test to detect neuroblastoma metabolites, there has been hope that mortality from this disease could be reduced by early detection via mass screening of young infants. Encouraging reports from Japan on mass screening programs instituted in the 1970s have been appearing in the medical literature since 1982, resulting in widespread interest in screening. This article applies standard epidemiologic criteria for screening evaluations to the Japanese reports. We find that the data needed to definitively assess the value of screening were not a part of those reports and that the benefits claimed from the reported data could be due to overdiagnosis. In addition, the most recent Japanese data, combined with recent advances in biologic understanding of neuroblastoma, show that screening at age 6 months may not detect tumors with poor prognoses. Even if it could, it is uncertain whether those outcomes could be substantially altered by earlier diagnosis. Although a final verdict on the value of neuroblastoma screening is not yet possible, these neuroblastoma studies are an excellent example of how screening results must be viewed with extreme caution in the absence of age-specific, population-based incidence and mortality rates. (AJDC. 1991;145:1415-1422) References 1. Young J, Reis L, Silverberg E, Horm J, Miller R. Cancer incidence, survival, and mortality for children younger than age 15 years . Cancer . 1986;58:598-602.Crossref 2. Woods W, Tuchman M. Neuroblastoma: the case for screening infants in North America . Pediatrics . 1987;79:869-873. 3. Carlsen N, Schroeder H, Bro P, et al. Neuroblastoma treated at the four major child oncologic clinics in Denmark 1943-80: an evaluation of 180 cases . Med Pediatr Oncol . 1985;13:180-186.Crossref 4. Kinnier Wilson L, Draper G. Neuroblastoma, its natural history and prognosis: a study of 487 cases . BMJ . 1974;3:301-307.Crossref 5. LaBrosse EH. Biochemical diagnosis of neuroblastoma: use of a urine spot test . Proc Am Assoc Cancer Res . 1968;9:39. 6. Sawada T, Todo S, Fujita K, lino S, Imashuku S, Kusunoki T. Mass screening of neuroblastoma in infancy . AJDC . 1982;136:710-712. 7. Tuchman M, Lemieux B, Auray-Blais C, et al. Screening for neuroblastoma in North America: preliminary results from the Quebec project . Pediatrics . 1990;86:765-773. 8. Craft A, Dale C, McGill A, Seviour J, Spence E. Biochemical screening for neuroblastoma in infants: a feasibility study . Med Pediatr Oncol . 1989;17:373-378.Crossref 9. Gellis S. Isn't it time for mass screening of infants for neuroblastoma? Pediatr Notes . 1990;14:39. 10. Mauer A. Screening for neuroblastoma . J Pediatr . 1988;112:576-577.Crossref 11. Tuchman M, Lemieux B, Woods WB. Screening for neuroblastoma in infants: investigate or implement? Pediatrics . 1990;86:791-793. 12. Murphy S, Cohn S, Craft A, et al. Do children benefit from mass screening for neuroblastoma? consensus statement for the American Cancer Society Workshop on Neuroblastoma Screening . Lancet . 1991;337:344-346.Crossref 13. Davis S, Rogers M, Prendergrass T. The incidence and epidemiologic characteristics of neuroblastoma in the United States . Am J Epidemiol . 1987;126:1063-1074. 14. Breslow N, McCann B. Statistical estimation of prognosis for children with neuroblastoma . Cancer Res . 1971;31: 2098-2103. 15. Coldman A, Fryer C, Elwood J, Sonley M. Influence of age at diagnosis, stage, tumor site, and sex on prognosis . Cancer . 1980;46:1896-1901.Crossref 16. Evans A, Chatten J, D'Angio G, Gerson J, Robinson J, Schnauffer L. A review of 17 IV-S neuroblastoma patients at the children's hospital of Philadelphia . Cancer . 1989;45:833-839.Crossref 17. Haas D, Ablin A, Miller C, et al. Complete pathologic maturation and regression of stage IV-S neuroblastoma without treatment . Cancer . 1988;62:818-825.Crossref 18. Beckwith J, Perrin E. In situ neuroblastomas: a contribution to the natural history of neural crest tumors . Am J Pathol . 1963;43:1089-1104. 19. Brodeur G, Seeger R, Schwab M, Varmus HE, Bishop JM. Amplification of n-myc in untreated human neuroblastomas correlates with advanced disease stage . Science . 1984;224:1121-1124.Crossref 20. Look A, Hayes F, Noitschke R, McWilliams N, Green A. Cellular DNA content as a predictor of response to chemotherapy in infants with unresectable neuroblastoma . N Engl J Med . 1984;311:231-235.Crossref 21. Hayashi Y, Inaba T, Hanada R, Yamamoto K. Chromosome findings and prognosis in 15 patients with neuroblastoma found by VMA mass screening . J Pediatr . 1988;112:567-571.Crossref 22. Kaneko Y, Kanda N, Maseki N, et al. Different karyotypic patterns in early and advanced stage neuroblastomas . Cancer Res . 1987;47:311-318. 23. Oppedal B, Storm-Mathisen I, Lie S, Brandtzaeb P. Prognostic factors in neuroblastoma: clinical, histopathologic, and immunohistochemical features and DNA ploidy in relation to prognosis . Cancer . 1988;62:772-780.Crossref 24. Nakagawara A. Genomic aspects of neuroblastomas in children under one year of age . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:47. 25. Nakagawara A, Ikeda K, Tsuda T, Higashi K. Biological characteristics of n-myc amplified neuroblastoma in patients over one year of age . Adv Neuroblastoma Res . 1988;2:31-39. 26. Nakagawara A, Ikeda K, Tsuda T, Higashi K. N-myc oncogene amplification and prognostic factors of neuroblastoma in children . J Pediatr Surg . 1987;22:895-898.Crossref 27. Tsuda T, Obara M, Hirano H, et al. Analysis of n-myc amplification in relation to disease stage and histologic types in human neuroblastomas . Cancer . 1987;60:820-826.Crossref 28. Bourhis J, Benard J, Wilson G, et al. Prognostic value of ploidic index and N-myc DNA amplification in 59 neuroblastomas . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:10. 29. Brodeur G. Clinical significance of genetic rearrangements in human neuroblastomas . Clin Chem . 1989;35/7B:B38-B42. 30. Brodeur G. The involvement of oncogenes and suppressor genes in human neoplasia . Adv Pediatr . 1987;34:1-44. 31. Auray-Blais C, Giguere R, Lemieux B. Thin-layer chromatography of urinary homovanillic acid and vanillylmandelic acid for large-scale neuroblastoma mass screening . Med Pediatr Oncol . 1989;17:364-367.Crossref 32. Matsumoto M, Anazawa A, Suzuki K, et al. Urine mass screening for neuroblastoma by high performance liquid chromatography (HPLC) . Pediatr Res . 1985;19:625.Crossref 33. Sato Y, Hanai J, Takasugi N, Takeda T. Determination of urinary vanillylmandelic acid and homovanillic acid in urine on filter paper for mass screening of neuroblastoma in infants . Tohoku J Exp Med . 1986;150:169-174.Crossref 34. Sawada T, Kidowaki T, Sakamoto I, et al. Neuroblastoma: mass screening for early detection and its prognosis . Cancer . 1984;53:2731-2735.Crossref 35. Sawada T, Kidowaki T, Sugimoto T, Kusunoki T. Incidence of neuroblastoma in infancy in Japan . Med Pediatr Oncol . 1984;12:101-103.Crossref 36. Sawada T, Nakaza T, Takasugi N. Mass screening for neuroblastoma in infants in Japan . Lancet . 1984;2:271-273.Crossref 37. Takeda T. History and current status of neuroblastoma screening in Japan . Med Pediatr Oncol . 1989;17:361-363.Crossref 38. Sawada T. Outcome of 25 neuroblastomas revealed by mass screening in Japan . Lancet . 1986;1:377.Crossref 39. Nishi M, Miyake H, Takeda T, et al. Effects of the mass screening of neuroblastoma in Sapporo City . Cancer . 1987;60:433-436.Crossref 40. Sawada T, Kawakatu H, Sugimoto T. Screening for neuroblastoma . Lancet . 1987;2:1204.Crossref 41. Sawada T, Sugimoto T, Matsumura T, Tunoda A, Takeda T, et al. Mass screening for neuroblastoma in infancy . In: Advances in Neuroblastoma Research 2 . New York, NY: Alan R Liss Inc; 1988:525-534. 42. Nishi M, Hirotsugo M, Takeda T, et al. Cases of neuroblastoma missed by the mass screening programs . Pediatr Res . 1989;26:603-607.Crossref 43. Naito H, Sasaki M, Yamashiro K, et al. Improvement in prognosis of neuroblastoma through mass population screening . J Pediatr Surg . 1990;25:245-248.Crossref 44. Cole P, Morrison AS. Basic issues in population screening for Cancer . J Natl Cancer Inst . 1980;64:1263-1272. 45. Miller A. Screening for cancer: issues and future directions . J Chronic Dis . 1986;39:1067-1077.Crossref 46. Sawada T, Kawakatus H, Sugimoto T, et al. Neuroblastoma mass screening in infancy in Kyoto, Japan . Indian J Pediatr. 1987;54:874-882.Crossref 47. Kramer S, Meadows A, Jarnett P, Evans A. Incidence of childhood cancer: experience of a decade in a population-based registry . J Natl Cancer Inst . 1983;70:49-55. 48. Evans A, D'Angio G, Propert K, Anderson J, Hahn H-W. Prognostic factors in neuroblastoma . Cancer . 1987;59:1853-1859.Crossref 49. Kaneko Y, Maseki N, Sakurai M, et al. Chromosomes and screening for neuroblastoma . Lancet . 1988;1:174-175.Crossref 50. Ishimoto K, Kiyokawa N, Ohya T, Miyano T, Shinohara T, Sera Y. Biological analysis of neuroblastoma in mass screened negative cases . In: D'Angio G, Evans A, Knudson A, Seeger R, eds. The Fifth Symposium on Neuroblastoma Research . Philadelphia, Pa: The Children's Hospital of Pennsylvania; 1990:117. 51. Kaneko K, Kanda N, Maseki N, et al. Current urinary mass screening for catecholamine metabolites at 6 months of age may be detecting only a small portion of high-risk neuroblastomas: a chromosome and N-myc amplification study . J Clin Oncol . 1990;8:2005-2013. 52. Brodeur G, Hayes A, Green A, et al. Consistent n-myc copy number in simultaneous or consecutive neuroblastoma samples from sixty individual patients . Cancer Res . 1987; 47:4248-4253. 53. Holtzman NA. What drives neonatal screening programs? N Engl J Med . 1991;325:802-804.Crossref

Journal

American Journal of Diseases of ChildrenAmerican Medical Association

Published: Dec 1, 1991

References