EDUCATIONAL ACHIEVEMENTS OF SURVIVORS OF CHILDHOOD CANCER
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
A. Allen, BA, J.S. Malpas, FRCP, FRCR, FFPM, and J. E. Kingston, MRCP n Imperial Cancer Research Fund Department of Paediatric Oncology, St. Bartholomew’s Hospital, West Smithfield, London EClA 7BE, England
The educational achievement o j 37 lonf-term survivors of childhood cancer were matched with the sibling closest in age. Analysis of the sibling pairs revealed no sipzificant difference in educational achievement between the study group and controls. A subset, who were treated with cranial prophylaxis, also showed no difference, but their numbers were small. K E Y WORDS: childhood cancer, educa!ional achievemen!, late effccts, siblings.
INTRODUCTION With the improvement in outlook for children with cancer, it is important to examine the needs and aspirations of those surviving into adolescence and young adulthood. Relatively little is known about the educational achievement of English children, although studies from the United States of survivors of childhood Hodgkin’s disease’ and of cancer in general‘ have been reassuring. Following a pilot study in children treated in the Paediatric Oncology Unit at St. Bartholomew’s Hospital, it was decided to investigate the effects of cancer on educational achievement.
MATERIAL AND METHODS A questionnaire was sent to 100 adolescents and young adults who were over the age of 16, had had histologically proven cancer, been treated in the Unit between 1973 and 1987, and were in continuous complete remission. Children who had had cerebral tumors treated either by surgery or radiotherapy were excluded, but children who had received cranial prophylaxis with radiotherapy and intrathecal methotrexate were included. Nearly 90 76 of subjects completed the questionnaire. From this population 37 patients, who had siblings of similar age and had completed their education, were sent a second questionnaire. Nearly all of the children and the siblings We are grateful to Douglas Altman for his statistical advice.
Pediatric Hmtology and Oncology, 7:339-345, 1990 CopJn&ht 0 1990 by Hemisphere Publishing Corporation
339
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
340
A. ALLEN ET AL.
were interviewed by one of the authors (A. A.) following completion of this second questionnaire. The diagnosis, age at diagnosis, and age at interview of the group studied is shown in Tables 1 and 2. The questionnaire asked about age; the last college or school attended; achievement in the national examinations of Certificate of Secondary Education (CSE), General Certificate of Education ordinary level (GCE “0” level, ages 15-16), GSE advanced level (“A” level, ages 17-19), and the more recently introduced General Certificate of Secondary Education (GCSE); and the results of first degrees and diplomas. The educational achievement of these patients and their siblings are given in Table 3. The number of those completing each level of secondary and tertiary education is given, arranged in increasing order of achievement. No attempt was made to detail achievement with regard to number or grade of pass, except where siblings had achieved a similar level and there was a major discrepancy in both the number of certificates and grades. This occurred, however, in only 2 sibling pairs. The achievements of 9 children treated with 2400 cGy cranial irradiation together with 5 doses of intrathecal methotrexate are compared with their siblings in Table 4.
ANALYSIS OF DATA The percentages of survivors and controls reaching each level of education was calculated, along with the exact 95% confidence intervals appropriate for the difference between paired populations“:
RESULTS The largest number of survivors had been treated for either Hodgkin’s disease or non-Hodgkin’s lymphoma (Table 1). Over half the survivors were between 10 and 15 years old at diagnosis. At the time of interview, their ages ranged between 16 and 25 years with a mean age of 20.5 years, whereas the sibling controls were between 17 to 29 years old with a mean age of 20.3 years. Of the 37 survivors, only 9 achieved no educational standards, compared with 7 of the control siblings. The close similarity of achievement at each stage is shown in Table 3 . When analyzed for achievement at various levels, the survivors did not vary significantly at any level. When separately analyzed, those children having cranial prophylaxis showed no significant difference from controls. Three survivors reached the same level of education; 4 survivors got further than their sibling pair; and 2 siblings got further than their sibling survivor.
EDUCATIONAL ACHIEVEMENTS OF CANCER SURVIVORS
341
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
TABLE 1. Diagnosis of Patients in Study Diagnosis
No.
ALL HD NHL Rhabdomyosarcoma Synovial sarcoma Osteosarcoma Neuroblastomn Undifferentiated carcinoma
5 11 10 6
TOTAL
37
1
2 1 1
ALL: acute lymphoblastic leukemia; HD: Hodgkin's disease; NHL: non-Hodgkin's lymphoma.
DISCUSSION The consequences of treatment for cancer on intellectual function and psychological state have given rise to great concern in recent years. In children with acute leukemia, many studies have attempted to identify the effect of cranial prophylaxis on cerebral function.'." Some studies have convincingly demonstrated a fall in measures of intelligence, memory, and other central nervous functions, but it has not been clear whether this translates into failure to achieve. Criticisms of many studies have centered on the fact that some studies included children whose intellectual impairment was present before treatment, children who had central nervous system tumors that had required surgery and intensive radiotherapy, other children who had relapsed and required further central nervous system irradiation for remission, or control groups that had not been properly matched. One of the most important single factors affecting outcome is the family's social background,' so that studies that do not include sibling controls could be considered deficient. This study shows that in a selected group of children over the age of 16, diagnosed between 1973 and 1987, there is no significant difference in educaTABLE 2. Age Range of Patients and Controls and Age at Diagnosis of Study Group Age of' patients at interview Age of controls at interview Age at diagnosis
16-25 years 17-29 years 0-5 years 5-lo years 10-15 years
--
mean 20.5 years mean 20.3 years no. 5 no. 12 no. 20
--
Total
37
H
e
W
-
~
76 62 38 16
(n = 37) %
~~
30 23 13 5
Sibling no.
81 62 35 14
(n
37) %
-15 to + 9 - 17 to 17 -18 to + 2 2 - 12 to + 15
-5 0 +3 +3
+
95% Confidence interval
Difference in %
Certificate of Secondary Education; GCSE = General Certificate of Secondary Education.
28 23 14 6
CSE “0”level/GCSE “A” level Degree/diploma
CSE
Survivor no.
Examination passed
TABLE 3. Achievement of Various Levels of Education
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
t
W
-
6 3 1
“0”level/GCSE “A” level Degreeldiploma
CSE
89 67
8
CSE
9) %
Certificate of Secondary Education; GCSE
33 11
(n
Survivor no.
=
33 11
3 1
0 0
+11 + 22
Difference in 70
General Certificate of Secondary Education.
78 44
7
(n = 9) B
4
Sibling no.
-
- 1 1 to + 1 1 -27 to 4-44 -38 to +38
95% Confidence interval
of Various Levels of Education Among Those Survivors Having Cranial Irradiation and Their Siblings
Examination passed
TABLE 4. Achievement
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
344
A. ALLEN ET AL.
tional achievement between them and an older sibling. The choice of comparison with an older sibling does, of course, produce bias, in that these controls will tend to have a greater chance of obtaining a higher qualification. That this does not result in a trend toward lower achievement in the survivors is impressive. It is also possible that both these age groups were achieving less well than the comparable population in general. This was tested in a pilot trial,"' where the level of educational achievement of children in the pilot study was compared with that of children in London and southeast England, where the number achieving educational qualifications in 1985 in the general population was 8376, but in the pilot study population of survivors was 90%. This difference was not statistically significant, but showed that the survivors of cancer were not disadvantaged with regard to their peer group. Although only 9 children had cranial prophylaxis, there was no evidence of any difference in educational achievement compared with those who had not received cranial prophylaxis. It must, however, be emphasized that these children had 2400 cGy on one occasion only. It is also noteworthy that 2 children who were under 4 years of age at the time of central nervous system irradiation both achieved good CSE results. However much their education has been disrupted, these children achieve normal educational standards, and despite reports of adverse effects on cerebral function, children who have had prophylactic central nervous system irradiation appear to succeed in school, at a university, and in later life. Parents, pediatricians, and teachers should, therefore, encourage these children to achieve their full potential in their educational pursuits.
REFERENCES 1. Wasserman AL, Thompson EI, Wilimas JA, Fairclough DL: The psychological status of survivors of childhood/adolescent Hodgkin's disease. Am J Dis Child 141:626-631, 1987. 2. Kelaghan J, Myers M H , Mulvihill JJ, et al.: Educational achievement of long term survivors of childhood and adolescent cancer. Med Ped Oncol 16:320-326, 1988. 3. Gardner MJ, Altman DG: Calculating confidence intervals for proportions and their differences. In: Statistics With Confidence, edited by M . J . Gardner and D. G . Altman, pp. 28-33, London: Br Med J , 1989. 4. Fabair P, Hoppe RT, Bloom J. et al.: Psychosocial problems among survivors of Hodgkin's disease. J Clin Oncol 4:805-814, 1986. 5. Soni S, Marten G, Pitner S, Duenas DA, Powazek M : Effects of central nervous system irradiation on the neuropsychological functioning of children with acute lymphoblastic leukaemia. New Eng J Med 293:113-118, 1975. 6. Eiser C: Intellectual abilities among survivors of childhood leukaemia as a function of CNS irradiation. Arch Dis Child 53:391-395, 1978. 7. Meadows A, Massari D, Fergusson J: Decline in IQscores and cognitive dysfunction in children with acute lymphoblastic leukaemia treated with cranial irradiation. Lancet i: 1015-1018, 1981. 8. Moss H , Nannis E, Poplack DG: The effect of prophylactic treatment of the central nervous system on the intellectual functioning of children with acute lymphoblastic leukaemia. Am J Med 71 :47-52, 1981.
EDUCATIONAL ACHIEVEMENTS OF CANCER SURVIVORS
345
9. Poplack DG, Brouwers P: Adverse sequelae of CNS therapy. In: Clinics in Oncology, vol. 4, edited by M. E. Nesbit, pp. 263-285, Philadelphia, London: Saunders, 1985. 10. Malpas JS: Cancer: The consequences olcure. Clin Radio1 39:166-172, 1988.
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
Receiued May 16, I990 Accepted June 12, 1990 Request reprintsfrom J . S.Malpas.
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
A. Allen, BA, J.S. Malpas, FRCP, FRCR, FFPM, and J. E. Kingston, MRCP n Imperial Cancer Research Fund Department of Paediatric Oncology, St. Bartholomew’s Hospital, West Smithfield, London EClA 7BE, England
The educational achievement o j 37 lonf-term survivors of childhood cancer were matched with the sibling closest in age. Analysis of the sibling pairs revealed no sipzificant difference in educational achievement between the study group and controls. A subset, who were treated with cranial prophylaxis, also showed no difference, but their numbers were small. K E Y WORDS: childhood cancer, educa!ional achievemen!, late effccts, siblings.
INTRODUCTION With the improvement in outlook for children with cancer, it is important to examine the needs and aspirations of those surviving into adolescence and young adulthood. Relatively little is known about the educational achievement of English children, although studies from the United States of survivors of childhood Hodgkin’s disease’ and of cancer in general‘ have been reassuring. Following a pilot study in children treated in the Paediatric Oncology Unit at St. Bartholomew’s Hospital, it was decided to investigate the effects of cancer on educational achievement.
MATERIAL AND METHODS A questionnaire was sent to 100 adolescents and young adults who were over the age of 16, had had histologically proven cancer, been treated in the Unit between 1973 and 1987, and were in continuous complete remission. Children who had had cerebral tumors treated either by surgery or radiotherapy were excluded, but children who had received cranial prophylaxis with radiotherapy and intrathecal methotrexate were included. Nearly 90 76 of subjects completed the questionnaire. From this population 37 patients, who had siblings of similar age and had completed their education, were sent a second questionnaire. Nearly all of the children and the siblings We are grateful to Douglas Altman for his statistical advice.
Pediatric Hmtology and Oncology, 7:339-345, 1990 CopJn&ht 0 1990 by Hemisphere Publishing Corporation
339
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
340
A. ALLEN ET AL.
were interviewed by one of the authors (A. A.) following completion of this second questionnaire. The diagnosis, age at diagnosis, and age at interview of the group studied is shown in Tables 1 and 2. The questionnaire asked about age; the last college or school attended; achievement in the national examinations of Certificate of Secondary Education (CSE), General Certificate of Education ordinary level (GCE “0” level, ages 15-16), GSE advanced level (“A” level, ages 17-19), and the more recently introduced General Certificate of Secondary Education (GCSE); and the results of first degrees and diplomas. The educational achievement of these patients and their siblings are given in Table 3. The number of those completing each level of secondary and tertiary education is given, arranged in increasing order of achievement. No attempt was made to detail achievement with regard to number or grade of pass, except where siblings had achieved a similar level and there was a major discrepancy in both the number of certificates and grades. This occurred, however, in only 2 sibling pairs. The achievements of 9 children treated with 2400 cGy cranial irradiation together with 5 doses of intrathecal methotrexate are compared with their siblings in Table 4.
ANALYSIS OF DATA The percentages of survivors and controls reaching each level of education was calculated, along with the exact 95% confidence intervals appropriate for the difference between paired populations“:
RESULTS The largest number of survivors had been treated for either Hodgkin’s disease or non-Hodgkin’s lymphoma (Table 1). Over half the survivors were between 10 and 15 years old at diagnosis. At the time of interview, their ages ranged between 16 and 25 years with a mean age of 20.5 years, whereas the sibling controls were between 17 to 29 years old with a mean age of 20.3 years. Of the 37 survivors, only 9 achieved no educational standards, compared with 7 of the control siblings. The close similarity of achievement at each stage is shown in Table 3 . When analyzed for achievement at various levels, the survivors did not vary significantly at any level. When separately analyzed, those children having cranial prophylaxis showed no significant difference from controls. Three survivors reached the same level of education; 4 survivors got further than their sibling pair; and 2 siblings got further than their sibling survivor.
EDUCATIONAL ACHIEVEMENTS OF CANCER SURVIVORS
341
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
TABLE 1. Diagnosis of Patients in Study Diagnosis
No.
ALL HD NHL Rhabdomyosarcoma Synovial sarcoma Osteosarcoma Neuroblastomn Undifferentiated carcinoma
5 11 10 6
TOTAL
37
1
2 1 1
ALL: acute lymphoblastic leukemia; HD: Hodgkin's disease; NHL: non-Hodgkin's lymphoma.
DISCUSSION The consequences of treatment for cancer on intellectual function and psychological state have given rise to great concern in recent years. In children with acute leukemia, many studies have attempted to identify the effect of cranial prophylaxis on cerebral function.'." Some studies have convincingly demonstrated a fall in measures of intelligence, memory, and other central nervous functions, but it has not been clear whether this translates into failure to achieve. Criticisms of many studies have centered on the fact that some studies included children whose intellectual impairment was present before treatment, children who had central nervous system tumors that had required surgery and intensive radiotherapy, other children who had relapsed and required further central nervous system irradiation for remission, or control groups that had not been properly matched. One of the most important single factors affecting outcome is the family's social background,' so that studies that do not include sibling controls could be considered deficient. This study shows that in a selected group of children over the age of 16, diagnosed between 1973 and 1987, there is no significant difference in educaTABLE 2. Age Range of Patients and Controls and Age at Diagnosis of Study Group Age of' patients at interview Age of controls at interview Age at diagnosis
16-25 years 17-29 years 0-5 years 5-lo years 10-15 years
--
mean 20.5 years mean 20.3 years no. 5 no. 12 no. 20
--
Total
37
H
e
W
-
~
76 62 38 16
(n = 37) %
~~
30 23 13 5
Sibling no.
81 62 35 14
(n
37) %
-15 to + 9 - 17 to 17 -18 to + 2 2 - 12 to + 15
-5 0 +3 +3
+
95% Confidence interval
Difference in %
Certificate of Secondary Education; GCSE = General Certificate of Secondary Education.
28 23 14 6
CSE “0”level/GCSE “A” level Degree/diploma
CSE
Survivor no.
Examination passed
TABLE 3. Achievement of Various Levels of Education
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
t
W
-
6 3 1
“0”level/GCSE “A” level Degreeldiploma
CSE
89 67
8
CSE
9) %
Certificate of Secondary Education; GCSE
33 11
(n
Survivor no.
=
33 11
3 1
0 0
+11 + 22
Difference in 70
General Certificate of Secondary Education.
78 44
7
(n = 9) B
4
Sibling no.
-
- 1 1 to + 1 1 -27 to 4-44 -38 to +38
95% Confidence interval
of Various Levels of Education Among Those Survivors Having Cranial Irradiation and Their Siblings
Examination passed
TABLE 4. Achievement
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
344
A. ALLEN ET AL.
tional achievement between them and an older sibling. The choice of comparison with an older sibling does, of course, produce bias, in that these controls will tend to have a greater chance of obtaining a higher qualification. That this does not result in a trend toward lower achievement in the survivors is impressive. It is also possible that both these age groups were achieving less well than the comparable population in general. This was tested in a pilot trial,"' where the level of educational achievement of children in the pilot study was compared with that of children in London and southeast England, where the number achieving educational qualifications in 1985 in the general population was 8376, but in the pilot study population of survivors was 90%. This difference was not statistically significant, but showed that the survivors of cancer were not disadvantaged with regard to their peer group. Although only 9 children had cranial prophylaxis, there was no evidence of any difference in educational achievement compared with those who had not received cranial prophylaxis. It must, however, be emphasized that these children had 2400 cGy on one occasion only. It is also noteworthy that 2 children who were under 4 years of age at the time of central nervous system irradiation both achieved good CSE results. However much their education has been disrupted, these children achieve normal educational standards, and despite reports of adverse effects on cerebral function, children who have had prophylactic central nervous system irradiation appear to succeed in school, at a university, and in later life. Parents, pediatricians, and teachers should, therefore, encourage these children to achieve their full potential in their educational pursuits.
REFERENCES 1. Wasserman AL, Thompson EI, Wilimas JA, Fairclough DL: The psychological status of survivors of childhood/adolescent Hodgkin's disease. Am J Dis Child 141:626-631, 1987. 2. Kelaghan J, Myers M H , Mulvihill JJ, et al.: Educational achievement of long term survivors of childhood and adolescent cancer. Med Ped Oncol 16:320-326, 1988. 3. Gardner MJ, Altman DG: Calculating confidence intervals for proportions and their differences. In: Statistics With Confidence, edited by M . J . Gardner and D. G . Altman, pp. 28-33, London: Br Med J , 1989. 4. Fabair P, Hoppe RT, Bloom J. et al.: Psychosocial problems among survivors of Hodgkin's disease. J Clin Oncol 4:805-814, 1986. 5. Soni S, Marten G, Pitner S, Duenas DA, Powazek M : Effects of central nervous system irradiation on the neuropsychological functioning of children with acute lymphoblastic leukaemia. New Eng J Med 293:113-118, 1975. 6. Eiser C: Intellectual abilities among survivors of childhood leukaemia as a function of CNS irradiation. Arch Dis Child 53:391-395, 1978. 7. Meadows A, Massari D, Fergusson J: Decline in IQscores and cognitive dysfunction in children with acute lymphoblastic leukaemia treated with cranial irradiation. Lancet i: 1015-1018, 1981. 8. Moss H , Nannis E, Poplack DG: The effect of prophylactic treatment of the central nervous system on the intellectual functioning of children with acute lymphoblastic leukaemia. Am J Med 71 :47-52, 1981.
EDUCATIONAL ACHIEVEMENTS OF CANCER SURVIVORS
345
9. Poplack DG, Brouwers P: Adverse sequelae of CNS therapy. In: Clinics in Oncology, vol. 4, edited by M. E. Nesbit, pp. 263-285, Philadelphia, London: Saunders, 1985. 10. Malpas JS: Cancer: The consequences olcure. Clin Radio1 39:166-172, 1988.
Pediatr Hematol Oncol Downloaded from informahealthcare.com by University of Newcastle Upon Tyne on 12/21/14 For personal use only.
Receiued May 16, I990 Accepted June 12, 1990 Request reprintsfrom J . S.Malpas.
