Introduction
Materials and methods
Patients
Methods
Data analysis
Results
Male (%) | 51 (42.9) | |
Median age at diagnosis of neuroblastoma in years (range) | 0.8 (0.0–10.5) | |
Tumour stage (INSS) | I–III (%) | 74 (62.2) |
IV (%) | 45 (37.8) | |
Tumour MYCN amplification | Yes (%) | 4 (3.4) |
Unknown (%) | 49 (41.2) | |
Median age at echocardiography in years (range) | 15.7 (0.8–41.4) |
Systematic review of literature, methods and results
Study | Miller et al. [21] | Miller et al. [20] | Berry et al. [2] | De la Monte et al. [6] | Nakissa et al. [23] | Neglia et al. [25] | Mann et al. [16] | Mili et al. [19] | Foulkes et al. [9] | Narod et al. [24] | Friedman et al. [10] | Nishi et al. [26] | George et al. [11] | Menegaux et al. [18] | Yanai et al. [29] | Chow et al. [5] | Munzer et al. [22] | Current study |
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Country and time period | USA, different time periods within 1941–1964 | USA, 1960–1966 | UK, time period nm | USA, 1889–1982 | USA, 1965–1980 | USA, 1969–nm | England, time period nm | USA, 1983–1988 | Canada, 1977–1993 | England, Scotland, Wales, 1971–1986 | USA, in parts 1965–1994 | Japan, 1969–1996 | USA, 1990–2000 | USA/Canada, 1992–1994 | Japan, 1990–2002 | USA, 1980–2004 | France, 2003–2004 | Netherlands, 1966–2006 |
Design and setting | Multicentre retrospective patient series | Multicentre (all USA) retrospective cohort study | Single centre retrospective patient series | Single centre retrospective cohort study | Single centre retrospective patient series | Multicentre retrospective patient series | Multicentre retrospective patient series | Multicentre (cancer registry) retrospective cohort study | Single centre retrospective cohort study | Multicentre (national cancer registry) retrospective cohort study | Single centre retrospective patient series | Multicentre (national cancer registry) retrospective cohort study | Single centre retrospective cohort study | Multicentre (139 hospitals) retrospective cohort study | Two-centre retrospective patient series | Multicentre (national cancer registry) retrospective cohort study | Multi-centre (National Registry of childhood solid tumours) retrospective cohort study | Single centre retrospective/prospective cohort study |
Patients and methods | ||||||||||||||||||
NB patients | Patients (<15 years) with NB listed in the diagnostic files of the hospitals and National Cooperative Leukaemia Survey (n = nm) | Patients (<15 years) who died of NB (n = 2,093) | Patients with NB (n = nm) | Patients with NB listed in autopsy files with tumour present at autopsy (n = 63)a
| Patients (0–12 years) with NB visiting the centre within time period (n = nm) | Patients with NB newly diagnosed or seen in survey region and born in survey region (n = 97) | Patients newly diagnosed with NB (n = nm) | Incident cases of NB registered in cancer registry and born in Iowa (n = 34) | Patients newly diagnosed with NB (n = 141) | Patients (<15 years) diagnosed with NB in registry (n = 1208) | Patients with NB listed in autopsy files (n = 58) | Patients (<=14 years) with NB in registry (n = nm) | Patients newly diagnosed with NB (n = 158)b
| Patients (<19 years) newly diagnosed with NB (n = 741) | Patients newly diagnosed with NB (n = nm)c
| Patients (<20 yrs) newly diagnosed with NB and born in Washington (n = 240) | Patients (<15 years) newly diagnosed with NB, surviving and not terminally ill (n = 235) | Patients newly diagnosed with NB, alive at time of study (n = 133) |
N of patients analysed (%) | 504 (% unclear) | 2,093 (100) | 144 (% unclear) | 63 (100) | 32 (% unclear) | 97 (100) | 35 (% unclear) | 34 (100) | 141 (100) | 1208 (100) | 58 (100) | 323 (>95) | 70 (44) | 538 (73) | 156 (% unclear) | 240 (100) | 191 (75) | 119 (89.5) |
NB stage | nm | nm | nm | nm | nm | Stage 1, 8%; 2, 13%; 3, 18%; 4, 50%; 4s, 11% | nm | nm | Stage 1, 23.4%; 2, 9.2%; 3, 24.1%; 4, 31.9%; 4s, 11.3% | nm | nm | nm | INSS stage 1–3, 31%; stage 4, 69% | nm | nm | Localised, 12.9%; regional, 20%; distant metastatic, 40.8%; unspecified, 26.3% | nm | INSS stage I–3, 62.2%; stage 4, 37.8% |
Control patients | – | – | – | A. Patients from remaining autopsy population minus peripheral neuroblastic tumours (n = 43,149) | – | Birth certificates of age matched controls randomly selected from all live births in the same state (n = 388) | Age- and sex-matched designated controls from general practitioner lists (n = 555) | – | Live births in British Columbia Health Surveillance Registry 1979–1988 (n = 419,646) | – | – | – | Consecutive patients with acute lymphoblastic leukaemia from the same centre, same period (n = 192) | Age matched controls selected through a random digit dialling method (n = 504) | – | Birth certificates of age matched controls from the same area (n = 2,400) | Age and sex matched controls selected through a random digit dialing method (n = 1,681) | Consecutive patients with acute lymphoblastic leukaemia ontrol group presented by George et al. [11] (n = 192) |
B. Patients with malignant melanoma or central neuroblastic tumour in same autopsy population (n = 135) | ||||||||||||||||||
Method of review of cardiac status | Review of medical charts | Review of death certificates | Review of medical records | Review of autopsy files (in addition all available fixed heart specimens and post-mortem coronary angiograms of CHD were studied) | Review of radiographs and re-examination of patients | Review of birth certificates and supplemental information forms | Parental interview, verification of information in obstetric records and general practitioners’ records | Review of records from birth defects registry after linkage with cancer registry | Patients: review of charts | Information from hospitals and family doctors, postal questionnaire to family doctors (in part of patients) | Review of data in computerised autopsy databank | Cancer registry data | Retrospective review of echocardiographic reports | Standardised telephone interview with mother | Review of charts | Review of birth certificates and hospital discharge records database after linkage with cancer registry | Standardised telephone interview with mother | Prospective echocardiography in 102 patients; review of echocardiography images in 17 patients |
Controls: data in registry | ||||||||||||||||||
Results | ||||||||||||||||||
N of patients with CHD (%) | 7 (1.4) | 6 (0.3) | 2 (1.4) | 7 (11.1) | 0 (0) | 0 (0) | 1 (2.9) | 1 (2.9) | 6 (4.3) | 7 (0.6) | 2 (3.5) | 2 (0.6) | 14 (20.0) | 15 (2.0) | 4 (2.6) | 5 (2.1) | 0 (0) | 2 (1.68) |
N of controls with CHD (%) | – | – | – | A. 2,081 (4.8) | – | 0 (0) | 0 (0) | – | Expected in patient cohort: 1.75 | – | – | – | 7 (3.6) | 3 (0.6) | – | 9 (0.4) | 5 (0.3) | 7 (3.6) |
B. 0 (0) | ||||||||||||||||||
Significance | – | – | – | nm | – | nm | nm | – |
P < 0.01 | – | – | – |
P = 0.0001 | Odds ratio 4.27 (95% CI 1.22–15.0)d
| – | Odds ratio 5.84 (95% CI 1.93–17.66)d
| Odds ratio 0d
|
P = 0.49 |