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PMID: 15901625 Published · ppublish English Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't Research Support, U.S. Gov't, P.H.S.

Cancer risk in persons with oral cleft--a population-based study of 8,093 cases.

American journal of epidemiology ·Vol. 161 ·No. 11 ·2005-06-01 ·Pages 1047-55

Bille C, Winther JF, Bautz A, Murray JC, Olsen J, Christensen K

Abstract

The authors conducted a nationwide study of the occurrence of cancer among 8,093 Danish oral cleft cases born in 1936 through 1998 and followed in the Danish Cancer Registry from 1968 through 1998, a total of 175,863 person-years, to assess a possible association between cancer and oral clefts. Observed and expected numbers of cancers among oral cleft cases were summarized as the overall and as 52 site-specific standardized incidence ratios. The expected overall number of all cancers was 131, but 140 incident cancers were found, corresponding to a standardized incidence ratio of 1.07 (95% confidence interval (CI): 0.90, 1.26). Analyses of the 52 sites for all oral cleft cases and analyses stratified for three cleft subgroups and the two sexes revealed only a few significant associations: an increased occurrence of breast cancer among females born with cleft lip and/or cleft palate (standardized incidence ratio (SIR) = 1.52, 95% CI: 1.05, 2.14), primary brain cancer among females born with cleft palate (SIR = 3.11, 95% CI: 1.14, 6.78), and primary lung cancer among males born with both cleft lip and cleft palate (SIR = 2.49, 95% CI: 1.00, 5.14). The results do not provide evidence for an increased overall cancer risk for individuals born with oral clefts.

MeSH Terms
Adolescent Adult Aged Child Cleft Lip/complications Cleft Palate/complications Cohort Studies Denmark/epidemiology Female Humans Incidence Male Middle Aged Neoplasms/epidemiology,etiology Registries/statistics & numerical data Risk Factors
Authors & Affiliations
6 authors, click to expand affiliations / ORCID
Bille Camilla
Center for the Prevention of Congenital Malformations, Institute of Public Health, University of Southern Denmark, Odense, Denmark. cbille@health.sdu.dk
Winther Jeanette Falck
Bautz Andrea
Murray Jeffrey C
Olsen Jørn
Christensen Kaare
References (57)
57 references, click to expand
  1. Heterozygous germline mutations in the p53 homolog p63 are the cause of EEC syndrome.
    Cell. 1999 Oct 15;99(2):143-53 PMID: 10535733
  2. Nonsyndromic cleft lip and palate is not associated with cancer or other birth defects.
    Am J Med Genet. 2000 Jan 3;90(1):17-24 PMID: 10602112
  3. Congenital abnormalities in children with acute leukemia: a report from the Children's Cancer Group.
    J Pediatr. 1998 Nov;133(5):617-23 PMID: 9821417
  4. Oral clefts, transforming growth factor alpha gene variants, and maternal smoking: a population-based case-control study in Denmark, 1991-1994.
    Am J Epidemiol. 1999 Feb 1;149(3):248-55 PMID: 9927220
  5. A population-based study of survival and childbearing among female subjects with birth defects and the risk of recurrence in their children.
    N Engl J Med. 1999 Apr 8;340(14):1057-62 PMID: 10194234
  6. Homeobox genes and cancer.
    Exp Cell Res. 1999 Apr 10;248(1):1-9 PMID: 10094807
  7. Prenatal exposures and childhood cancer.
    Pediatr Dev Pathol. 1999 May-Jun;2(3):208-14 PMID: 10191343
  8. The 20th century Danish facial cleft population--epidemiological and genetic-epidemiological studies.
    Cleft Palate Craniofac J. 1999 Mar;36(2):96-104 PMID: 10213053
  9. Changing lifestyles and oral clefts occurrence in Denmark.
    Cleft Palate Craniofac J. 2005 May;42(3):255-9 PMID: 15865458
  10. A novel loss-of-function mutation in TTF-2 is associated with congenital hypothyroidism, thyroid agenesis and cleft palate.
    Hum Mol Genet. 2002 Aug 15;11(17):2051-9 PMID: 12165566
  11. Do parents of children with congenital malformations have a higher cancer risk? A nationwide study in Denmark.
    Br J Cancer. 2002 Aug 27;87(5):524-8 PMID: 12189550
  12. Epidemiology of primary brain tumors: current concepts and review of the literature.
    Neuro Oncol. 2002 Oct;4(4):278-99 PMID: 12356358
  13. Breast health and disease over a lifetime.
    Clin Obstet Gynecol. 2002 Dec;45(4):1140-61 PMID: 12438891
  14. Mortality and cancer incidence among individuals with Down syndrome.
    Arch Intern Med. 2003 Mar 24;163(6):705-11 PMID: 12639204
  15. Somatic mutations in PTPN11 in juvenile myelomonocytic leukemia, myelodysplastic syndromes and acute myeloid leukemia.
    Nat Genet. 2003 Jun;34(2):148-50 PMID: 12717436
  16. Clinical manifestations of genetic instability overlap one another.
    Pathol Oncol Res. 2004;10(1):12-6 PMID: 15029255
  17. Brain tumours: incidence, survival, and aetiology.
    J Neurol Neurosurg Psychiatry. 2004 Jun;75 Suppl 2:ii12-7 PMID: 15146034
  18. Long term follow up study of survival associated with cleft lip and palate at birth.
    BMJ. 2004 Jun 12;328(7453):1405 PMID: 15145797
  19. Genetic evidence for lineage-related and differentiation stage-related contribution of somatic PTPN11 mutations to leukemogenesis in childhood acute leukemia.
    Blood. 2004 Jul 15;104(2):307-13 PMID: 14982869
  20. Congenital anomalies in children with malignancy.
    Paediatr Univ Tokyo. 1968 Jul;16:31-7 PMID: 4305810
  21. Childhood cancer and congenital defects. A study of U.S. death certificates during the period 1960-1966.
    Pediatr Res. 1969 Sep;3(5):389-97 PMID: 4310035
  22. Associated congenital malformations in retinoblastoma.
    Clin Genet. 1975 Jan;7(1):37-9 PMID: 1116308
  23. The fetal hydantoin syndrome.
    J Pediatr. 1975 Aug;87(2):285-90 PMID: 50428
  24. Oral clefts and childhood cancer.
    Lancet. 1980 Mar 29;1(8170):722 PMID: 6103151
  25. Fetal hydantoin syndrome, neuroblastoma, and hemorrhagic disease in a neonate.
    JAMA. 1980 Sep 26;244(13):1464-5 PMID: 7420637
  26. Aicardi syndrome, papilloma of the choroid plexus, cleft lip, and cleft of the posterior palate.
    J Pediatr. 1984 Mar;104(3):404-5 PMID: 6707796
  27. A population-based study of cancer incidence in twins and in children with congenital malformations or low birth weight, Norway, 1967-1980.
    Am J Epidemiol. 1985 Jan;121(1):49-56 PMID: 3155484
  28. Increased prevalence of minor anomalies in childhood malignancy.
    Eur J Pediatr. 1985 Sep;144(3):243-54 PMID: 4054163
  29. Median cleft of the upper lip associated with lipomas of the central nervous system and cutaneous polyps.
    Am J Med Genet. 1987 Apr;26(4):921-4 PMID: 3591835
  30. Maternal and perinatal risk factors for childhood leukemia.
    Cancer Res. 1991 Jul 15;51(14):3696-701 PMID: 2065325
  31. Selection bias in genetic-epidemiological studies of cleft lip and palate.
    Am J Hum Genet. 1992 Sep;51(3):654-9 PMID: 1496993
  32. Risk of childhood cancer for infants with birth defects. I. A record-linkage study, Atlanta, Georgia, 1968-1988.
    Am J Epidemiol. 1993 Mar 15;137(6):629-38 PMID: 8470664
  33. Risk of childhood cancer for infants with birth defects. II. A record-linkage study, Iowa, 1983-1989.
    Am J Epidemiol. 1993 Mar 15;137(6):639-44 PMID: 8470665
  34. Congenital abnormalities in children with cancer and their relatives: results from a case-control study (IRESCC).
    Br J Cancer. 1993 Aug;68(2):357-63 PMID: 8347491
  35. Etiological subgroups in non-syndromic isolated cleft palate. A genetic-epidemiological study of 52 Danish birth cohorts.
    Clin Genet. 1994 Nov;46(5):329-35 PMID: 7889640
  36. Psychosocial adjustment in Norwegian adults who had undergone standardised treatment of complete cleft lip and palate. I. Education, employment and marriage.
    Scand J Plast Reconstr Surg Hand Surg. 1995 Sep;29(3):251-7 PMID: 8539569
  37. Occurrence of cancer in women with Turner syndrome.
    Br J Cancer. 1996 May;73(9):1156-9 PMID: 8624281
  38. Psychosocial adjustment in Norwegian adults who had undergone standardised treatment of complete cleft lip and palate. II. Self-reported problems and concerns with appearance.
    Scand J Plast Reconstr Surg Hand Surg. 1995 Dec;29(4):329-36 PMID: 8771260
  39. Epidemiology of primary CNS neoplasms.
    Neurol Clin. 1996 May;14(2):273-90 PMID: 8827171
  40. Genetics and epidemiology, congenital anomalies and cancer.
    Am J Hum Genet. 1997 Mar;60(3):469-73 PMID: 9042905
  41. Risks of leukaemia and solid tumours in individuals with Down's syndrome.
    Lancet. 2000 Jan 15;355(9199):165-9 PMID: 10675114
  42. Congenital malformations and childhood cancer.
    Med Pediatr Oncol. 2000 Apr;34(4):250-4 PMID: 10742060
  43. What causes childhood brain tumors? Limited knowledge, many clues.
    Pediatr Neurosurg. 2000 Jun;32(6):321-6 PMID: 10971194
  44. Age at any birth is associated with breast cancer risk.
    Epidemiology. 2001 Jan;12(1):68-73 PMID: 11138822
  45. Increased incidence of a midline brain anomaly in patients with nonsyndromic clefts of the lip and/or palate.
    J Neuroimaging. 2001 Oct;11(4):418-24 PMID: 11677883
  46. Mutations in PTPN11, encoding the protein tyrosine phosphatase SHP-2, cause Noonan syndrome.
    Nat Genet. 2001 Dec;29(4):465-8 PMID: 11704759
  47. Guidelines for the design and analysis of studies on nonsyndromic cleft lip and cleft palate in humans: summary report from a Workshop of the International Consortium for Oral Clefts Genetics.
    Cleft Palate Craniofac J. 2002 Jan;39(1):93-100 PMID: 11772175
  48. Increased prevalence of minor anomalies in children with hematologic malignancies.
    Med Pediatr Oncol. 2002 Feb;38(2):128-30 PMID: 11813182
  49. Hereditary cleft lip/palate and Wilms tumor: a rare association.
    Cleft Palate Craniofac J. 2002 May;39(3):376-9 PMID: 12019017
  50. Facial clefting and psychiatric diseases: a follow-up of the Danish 1936-1987 Facial Cleft cohort.
    Cleft Palate Craniofac J. 2002 Jul;39(4):392-6 PMID: 12071787
  51. [Prevention of cancer--what do we do in Denmark and what can we achieve?].
    Ugeskr Laeger. 2002 May 27;164(22):2876-81 PMID: 12082813
  52. Congenital anomalies and childhood cancer in Great Britain.
    Am J Hum Genet. 1997 Mar;60(3):474-85 PMID: 9042906
  53. Clinical manifestations in 105 persons with nevoid basal cell carcinoma syndrome.
    Am J Med Genet. 1997 Mar 31;69(3):299-308 PMID: 9096761
  54. Homeobox genes: molecular link between congenital anomalies and cancer.
    Eur J Cancer. 1997 Apr;33(4):635-7 PMID: 9274447
  55. Excess of mild errors of morphogenesis in childhood lymphoblastic leukemia.
    Am J Med Genet. 1998 Jan 6;75(1):22-7 PMID: 9450852
  56. Avoidable cancers in the Nordic countries. Tobacco smoking.
    APMIS Suppl. 1997;76:9-47 PMID: 9462818
  57. Mutation of the gene encoding human TTF-2 associated with thyroid agenesis, cleft palate and choanal atresia.
    Nat Genet. 1998 Aug;19(4):399-401 PMID: 9697705
Article Info
Journal
American journal of epidemiology
Abbr.
Am J Epidemiol
ISSN
0002-9262
DOI
10.1093/aje/kwi132
Email
cbille@health.sdu.dk
Published
2005-06-01
Pages
1047-55
Language
English
Region
United States
NLM ID
7910653
PMCID
PMC2839121
Subset
IM
Grants
NIDCR NIH HHS · R01 DE011948 · United States
NIDCR NIH HHS · R01 DE011948-05 · United States
NIDCR NIH HHS · R01 DE 11948 · United States
External Links
PubMed source DOI: 10.1093/aje/kwi132 PMC Full Text View journal details
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