1115

Utilization of Special Education Services and

Educational Attainment among Long-Term Survivors
of Childhood Cancer
A Report from the Childhood Cancer Survivor Study

Pauline A. Mitby, M.P.H.1 BACKGROUND. The objective of the current report was to compare the self-reported
Leslie L. Robison, Ph.D.1 rates of special education (SE) and educational attainment among specific groups
John A. Whitton, M.S.2 of childhood cancer survivors and a random sample of sibling controls.
Michael A. Zevon, Ph.D.3 METHODS. The Childhood Cancer Survivor Study is a retrospective cohort of
Iris C. Gibbs, M.D.4 individuals who were diagnosed with a cancer in childhood and survived at least 5
Jean M. Tersak, M.D.5 years postdiagnosis. This analysis includes 12,430 survivors and 3410 full siblings.
Anna T. Meadows, M.D.6 Reported use of SE services and educational attainment were analyzed within
Marilyn Stovall, Ph.D.7 subgroups defined by type of cancer, age at diagnosis, and type of treatment.
Lonnie K. Zeltzer, M.D.8 RESULTS. The use of SE services was reported in 23% of survivors and 8% of
Ann C. Mertens, Ph.D.1 siblings, with the greatest differences observed among survivors who were diag-
nosed before age 6 years, most notably survivors of central nervous system (CNS)
1
Department of Pediatrics, University of Minne- tumors (odds ratio [OR], 18.8; 95% confidence interval [95%CI], 15.01–23.49),
sota Medical School and Cancer Center, Minneap- leukemia (OR, 4.4; 95%CI, 3.75–5.16), and Hodgkin disease (OR, 4.4; 95%CI, 2.64 –
olis, Minnesota. 7.24). It was found that intrathecal methotrexate (IT MTX) and cranial radiation
2
Cancer Prevention Research Program, Fred (CRT), administered alone or in combination, significantly increased the likelihood
Hutchinson Cancer Research Center, Seattle, that a survivor would use SE (IT MTX only: OR, 1.3; 95%CI, 1.09 –1.78; CRT only:
Washington. OR, 7.2; 95%CI, 6.14 – 8.39; IT MTX and CRT combined: OR, 2.6; 95%CI, 2.30 –2.95).
3
Department of Psychosocial Oncology, Roswell A positive dose response was identified between higher doses of CRT and use of SE.
Park Cancer Institute, Buffalo, New York. It was determined that survivors of leukemia (OR, 1.6; 95%CI, 1.23–2.16), CNS
4
Department of Radiation Oncology, Stanford Uni- tumors (OR, 2.7; 95%CI, 1.92–3.81), non-Hodgkin lymphoma (OR, 1.8; 95%CI,
versity, Stanford, California. 1.15–2.78), and neuroblastoma (OR, 1.7; 95%CI, 1.14 –2.61) were significantly less
5 likely to finish high school compared with siblings; however, when survivors
Department of Pediatric Hematology-Oncology,
Children’s Hospital of Pittsburgh, Pittsburgh, Penn- received SE services, risk estimates approximated those of the sibling SE
sylvania. population.
6
Division of Oncology, Children’s Hospital of Phil-
adelphia, Philadelphia, Pennsylvania.
7
Department of Radiation Physics, The University Francisco, CA: Arthur Ablin, M.D. (Institutional Frederick Li, M.D. (member of the CCSS Steering
of Texas M. D. Anderson Cancer Center, Houston, Principal Investigator); University of Alabama, Bir- Committee); Texas Children’s Center, Houston, TX:
Texas. mingham, AL: Roger Berkow, M.D. (Institutional Zoann Dreyer, M.D. (Institutional Principal Investi-
Principal Investigator); International Epidemiology gator); Children’s Hospital and Medical Center,
8
Department of Pediatrics, University of Califor- Institute, Rockville, MD: John Boice, Sc.D. (mem- Seattle, WA: Debra Friedman, M.D., M.P.H. (Insti-
nia-Los Angeles School of Medicine and Jonsson ber of the CCSS Steering Committee); University of tutional Principal Investigator) and Thomas Pen-
Comprehensive Cancer Center, Los Angeles, Cali- Washington, Seattle, WA: Norman Breslow, Ph.D. dergrass, M.D. (former Institutional Principal Inves-
fornia. (member of the CCSS Steering Committee); UT- tigator); Roswell Park Cancer Institute, Buffalo, NY:
Supported by grant CA 55727 from the National Southwestern Medical Center at Dallas, TX: George Daniel M. Green, M.D. (Institutional Principal In-
Cancer Institute, (Bethesda, MD) and by the Chil- R. Buchanan, M.D. (Institutional Principal Investi- vestigator and member of the CCSS Steering Com-
dren’s Cancer Research Fund (Minneapolis, MN). gator) and Kevin Oeffinger, M.D. (member of the mittee); Hospital for Sick Children, Toronto, On-
CCSS Steering Committee); Dana-Farber Cancer tario, Canada: Mark Greenberg, M.B., Ch.B.
The following CCSS Institutions and Investigators Institute, Boston, MA: Lisa Diller, M.D. (Institutional (Institutional Principal Investigator); St. Louis Chil-
participated in the Childhood Cancer Survivor Principal Investigator), Holcombe Grier, M.D. dren’s Hospital, St. Louis, MO: Robert Hayashi,
Study. University of California-San Francisco, San (former Institutional Principal Investigator), and M.D. (Institutional Principal Investigator) and

© 2003 American Cancer Society

 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
1116 CANCER February 15, 2003 / Volume 97 / Number 4

CONCLUSIONS. Children who are diagnosed with cancer should be followed closely
during and after treatment to identify early signs of learning disabilities and to
maximize intervention strategies for the successful completion of scholastic goals.
Cancer 2003;97:1115–26. © 2003 American Cancer Society.
DOI 10.1002/cncr.11117

KEYWORDS: late effects, psychosocial, scholastic achievement, radiation, special

education, education attainment, childhood cancer.

W ith the introduction of multimodal therapy in the

early 1970s, 5-year survival rates for most child-
hood cancers improved dramatically. Data from the
tors, including health-related behaviors and educa-
tional achievement.
Educational achievement and diminished cogni-
Surveillance, Epidemiology, and End Results (SEER) tive functioning are two outcomes that have been
Program show that 5-year survival rates for all child- reported extensively in the literature on survivors of
hood cancers increased from 56% in 1974 to 75% by childhood cancer. To varying degrees, deficits have
1995. The current 5-year survival rates are now as high been noted most often among children diagnosed
as 81% for patients with acute lymphoblastic leukemia with ALL and central nervous system (CNS) tumors
(ALL) and 93% for patients with Hodgkin disease and who received intrathecal methotrexate (IT MTX)
Wilms tumor.1 The need to identify the degree to and/or cranial radiation (CRT).2– 6 The CCSS provides
which late effects of therapy may impact quality of life a unique opportunity to investigate these treatment
becomes critical as increasing numbers of children effects by making it possible to explore their potential
become long-term survivors. The Childhood Cancer impact on survivors’ long-term educational achieve-
Survivor Study (CCSS) was created to serve as a re- ment across all major childhood cancer diagnoses.
source for the investigation of a broad spectrum of The purpose of this analysis was twofold: 1) to de-
outcomes among long-term survivors of childhood scribe the self-reported utilization and factors associ-
cancer, such as the risk of second cancers, cardiovas- ated with the use of special education (SE) services,
cular events, fertility, mortality, and psychosocial fac- and 2) to determine the level of education attained

Teresa Vietti, M.D. (former Institutional Principal Investigator); St. Jude Children’s Research Hospital, Memphis, TN: Melissa Hudson, M.D. (Institutional Principal
Investigator and member of the CCSS Steering Committee); University of Michigan, Ann Arbor, MI: Raymond Hutchinson, M.D. (Institutional Principal Investigator);
Stanford University School of Medicine, Stanford, CA: Michael P. Link, M.D. (Institutional Principal Investigator) and Sarah S. Donaldson, M.D. (member of the CCSS
Steering Committee); Children’s Hospital of Philadelphia, Philadelphia, PA: Anna Meadows, M.D. (Institutional Principal Investigator and member of the CCSS Steering
Committee) and Bobbie Bayton (member of the CCSS Steering Committee); Children’s Hospital, Oklahoma City, OK: John Mulvihill, M.D. (member of the CCSS
Steering Committee); Children’s Hospital, Denver, CO: Brian Greffe, M.D. (Institutional Principal Investigator) and, Lorrie Odom, M.D. (former Institutional Principal
Investigator); Children’s Health Care-Minneapolis, Minneapolis, MN: Maura O’Leary, M.D. (Institutional Principal Investigator); Columbus Children’s Hospital,
Columbus, OH: Amanda Termuhlen, M.D. (Institutional Principal Investigator), Frederick Ruymann, M.D. (former Institutional Principal Investigator) and Stephen
Qualman, M.D. (member of the CCSS Steering Committee); Children’s National Medical Center, Washington, DC: Gregory Reaman, M.D. (Institutional Principal
Investigator) and Roger Packer, M.D. (member of the CCSS Steering Committee); Children’s Hospital of Pittsburgh, Pittsburgh, PA: A. Kim Ritchey, M.D. (Institutional
Principal Investigator) and Julie Blatt, M.D. (former Institutional Principal Investigator); University of Minnesota, Minneapolis, MN: Leslie L. Robison, Ph.D. (Institutional
Principal Investigator and member of the CCSS Steering Committee), Ann Mertens, Ph.D. (member of the CCSS Steering Committee), Joseph Neglia, M.D., M.P.H.
(member of the CCSS Steering Committee), Mark Nesbit, M.D. (member of the CCSS Steering Committee), and Stella Davies, M.D., Ph.D. (member of the CCSS
Steering Committee); Children’s Hospital, Los Angeles, CA: Kathy Ruccione, R.N., M.P.H. (Institutional Principal Investigator); Memorial Sloan-Kettering Cancer
Center, New York, NY: Charles Sklar, M.D. (Institutional Principal Investigator and member of the CCSS Steering Committee); National Cancer Institute, Bethesda,
MD: Malcolm Smith, M.D. (member of the CCSS Steering Committee) and Peter Inskipp, Ph.D. (member of the CCSS Steering Committee); Mayo Clinic, Rochester,
MN: W. Anthony Smithson, M.D. (Institutional Principal Investigator) and Gerald Gilchrist, M.D. (former Institutional Principal Investigator); The University of Texas
M. D. Anderson Cancer Center, Houston, TX: Louise Strong, M.D. (Institutional Principal Investigator and member of the CCSS Steering Committee) and Marilyn
Stovall, Ph.D. (member of the CCSS Steering Committee); Riley Hospital for Children, Indianapolis, IN: Terry A. Vik, M.D. (Institutional Principal Investigator) and Robert
Weetman, M.D. (former Institutional Principal Investigator); Fred Hutchinson Cancer Center, Seattle, WA: Yutaka Yasui, Ph.D. (Institutional Principal Investigator and
member of the CCSS Steering Committee) and John Potter, M.D., Ph.D. (former Institutional Principal Investigator and member of the CCSS Steering Committee);
University of California-Los Angeles, Los Angeles, CA: Lonnie Zeltzer, M.D. (Institutional Principal Investigator and member of the CCSS Steering Committee).

The authors express their appreciation to Catherine Moen for her valuable contribution to this effort.

Address for reprints: Pauline A. Mitby, M.P.H., Division of Epidemiology and Clinical Research, 420 Delaware Street SE, University of Minnesota Medical School and
Cancer Center, MMC 715, Minneapolis, MN, 55455; Fax: (612) 624-7147; E-mail: [email protected]

Received July 22, 2002; revision received September 18, 2002; accepted September 19, 2002.
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Special Education among Cancer Survivors/Mitby et al. 1117

among a large population of long-term survivors of in general, participants and refusals were very similar
childhood cancer. with regard to gender, cancer diagnosis, age at diag-
nosis, age at contact, and type of cancer treatment.
MATERIALS AND METHODS The rate of refusal was significantly higher (P ⫽ 0.001)
The Long-Term Follow-Up Study is a retrospective among the next of kin (34%) of patients who had
cohort comprised of individuals diagnosed with can- survived at least 5 years from diagnosis but died sub-
cer, leukemia, tumors or other serious illnesses of sequently compared with patients who currently were
childhood. Those individuals with a confirmed diag- living (21%). Comparisons between survivors who
nosis of cancer comprise the CCSS. Details of the completed the baseline questionnaire and survivors
study design and CCSS cohort characteristics have who were classified as lost to follow-up did not reveal
been published elsewhere.7 Briefly, study patients any statistically significant differences other than vital
were identified from the 25 CCSS institutions from status.7
across the United States and Canada. Eligibility crite- During the collection of baseline data, informa-
ria for the study included a newly diagnosed cancer tion was obtained from the cancer survivors on their
(leukemia, CNS tumor, Hodgkin disease, non-Hodgkin siblings. To establish a sibling cohort, first, a random
lymphoma, kidney, neuroblastoma, soft tissue sar- sample of 50% of participating survivors was selected.
coma, or bone tumor) between January 1, 1970 and Then, if the survivor who was selected through ran-
December 31, 1986; survival of 5 years from the date of domization reported a living full brother or sister, the
diagnosis; age ⬍ 21 years at the time of diagnosis; sibling closest in age to the survivor was invited to
English-speaking or Spanish-speaking; and living in participate in the study. If the sibling agreed, they or
the United States or Canada at the time of diagnosis. their parents completed an age specific baseline ques-
The CCSS protocol and contact documents were tionnaire similar to that completed by the survivors.
reviewed and approved by the Human Subjects Com- The target sample for the sibling cohort is 5000 par-
mittee at each participating institution. Baseline ques- ticipants. Recruitment for the sibling cohort was ini-
tionnaires were collected from September, 1994 tiated in July, 1996 and is still in progress. At the time
through November, 2000. Age specific questionnaires of the current analysis, 5800 siblings had been con-
were developed for survivors age ⬍ 18 years and sur- tacted to participate; 3528 siblings completed a ques-
vivors age ⱖ 18 years at the time of contact. Survivors tionnaire, 472 siblings chose not to participate, and
age ⱖ 18 years were sent a baseline questionnaire and 1800 siblings were pending. Of the 3528 sibling ques-
consent form directly and were asked to complete and tionnaires received, 110 questionnaires are being re-
return them. For survivors age ⬍ 18 years, the ques- viewed by the coordinating center for possible content
tionnaire and consent form were sent to the parent(s) and logic discrepancies and were not included in the
to complete. The next of kin of survivors who subse- current analysis.
quently died at least 5 years postdiagnosis were asked
to complete a questionnaire for that survivor. After Data Analysis
obtaining consent, survivors’ medical records, includ- Items from the baseline questionnaire relating to ed-
ing radiotherapy records, were abstracted by trained ucation and school history included the highest grade
data managers at each institution according to a stan- or level of schooling completed and the type of high
dardized protocol to obtain information such as type, school diploma received: standard versus general ed-
dates, and schedule of treatment. Copies of the base- ucational development credential (GED). A GED is a
line questionnaire and the medical record abstraction high school equivalency test that is an alternative way
form used in data collection are available for review to complete high school. Also included was whether
online at www.cancer.umn.edu/ccss. the participant received learning-disabled or SE ser-
Among the 20,276 eligible 5-year survivors, 14,054 vices in elementary, junior, or high school. Possible
survivors completed a questionaire at the time of this responses were yes, no, or not sure. For participants
analysis, 2996 survivors were classified as lost to fol- who reported receiving SE services, additional infor-
low-up, 3132 survivors did not participate, and 94 mation was obtained, including reasons for SE and
survivors were pending inclusion into the cohort. At specific grade levels at which the services were ren-
the time of this analysis 12,431 of the 14,054 partici- dered.
pants have signed a medical release allowing for the When evaluating possible associations between
abstraction of medical records. SE placement and exposure to CRT, we examined CRT
Comparisons made to determine whether demo- both as a dichotomous variable and by dose range.
graphic and/or treatment-related characteristics dif- CRT doses were coded as the maximum dose to any
fered between participants and refusals showed that, part of the brain. Assigned CRT dose incorporated all
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
1118 CANCER February 15, 2003 / Volume 97 / Number 4

treatments, including those given for recurrences and TABLE 1

new malignancies, as long as at least one treatment Demographics of the Current Analysis
occurred prior to initiation of SE services. Tumor lo-
No. (%)
cation was not available, and surgical intervention was
not considered in this analysis for survivors of CNS Characteristic Patient Siblings
tumors. Survivors who initiated SE services prior to
their primary cancer diagnosis were not included in Survival
Alive 11425 (91.9) 3410 (100.0)
analysis of treatment effects.
Dead 1005 (8.1) 0
Because the age at each grade level was not pro- Gender
vided by respondents, the expected age at grade level Male 6543 (52.6) 1642 (48.2)
was calculated, accounting for birthdays during the Female 5887 (47.4) 1768 (51.8)
school year and assuming that participants completed Race
White, non-Hispanic 10969 (88.2) 3087 (90.5)
each grade without being held back or skipping
Black, non-Hispanic 478 (3.9) 84 (2.5)
grades. Thus, it was considered that age in preschool Hispanic 607 (4.9) 133 (3.9)
was ⬍ 6 years, age in kindergarten was 6 years, age in Other 346 (2.8) 97 (2.8)
first grade was 7 years, etc. Both survivors and siblings Not specified 30 (0.2) 9 (0.2)
age ⬍ 6 years at the time of questionnaire completion Age at questionnaire completion (yrs)
6–9 154 (1.2) 68 (2.0)
(not of school age) were excluded from this analysis
10–14 1734 (14.0) 337 (9.9)
(one survivor and eight siblings). 15–19 2739 (22.0) 587 (17.2)
Odds ratios (ORs) and 95% confidence intervals 20–24 2622 (21.1) 589 (17.3)
(95%CIs) were calculated using Cochran–Mantel– 25–29 2378 (19.1) 633 (18.6)
Haenszel statistics for disease specific and treatment 30–34 1693 (13.6) 563 (16.5)
35–39 813 (6.5) 378 (11.1)
specific analyses. Because the basic study design of
40–44 276 (2.2) 19.5 (5.7)
the CCSS is such that not every survivor has a sibling 45–59 21 (0.2) 60 (1.7)
control, generalized estimating equation models were Diagnosis
used to calculate estimates of ORs.8 This method per- Leukemia 4213 (33.9) —
forms logistic regression analyses, controlling for the Hodgkin’s disease 1680 (13.5) —
CNS tumor 1637 (13.2) —
nonindependence of members of the same family
Soft tissue sarcoma 1075 (8.6) —
(survivors and their siblings). Kidney 1063 (8.6) —
Bone cancer 1033 (8.3) —
RESULTS Non-Hodgkin lymphoma 908 (7.3) —
The current report includes 12,430 survivors who were Neuroblastoma 821 (6.6) —
Age at diagnosis (yrs)
age ⱖ 6 years at the time of questionnaire completion
0–5 5707 (45.9) —
for whom both baseline questionnaires and medical 6–10 2425 (19.5) —
record abstraction forms were complete at the time of 11–15 2618 (21.1) —
analysis. Sibling data were available for 3410 individ- 16–20 1680 (13.5) —
uals age ⱖ 6 years. Table 1 shows characteristics of the Special education
Yes 2866 (23.1) 282 (8.3)
study participants. Compared with cancer survivors,
No 8150 (65.6) 2747 (80.6)
siblings were more likely to be female and were some- Not sure 81 (0.7) 24 (0.7)
what older. Survivors ranged in age from 6 years to 47 Ambiguous/missing data 1333 (10.7) 357 (10.5)
years, with a median age of 23 years, whereas siblings
were between the ages of 6 years and 56 years, with a CNS: central nervous system.

median age of 26 years. The three major cancer diag-

noses were leukemia, Hodgkin disease, and CNS tu-
mors, accounting for 60.5% of the survivor population. ing for age at questionnaire completion, younger age
Twenty-three percent of the survivors indicated they at diagnosis is associated significantly with a higher
were in SE at least some time during their K–12 need for SE across all cancer types. Recognizing that
schooling, compared with 8% of the sibling cohort (P males typically receive SE services more often than
⬍ 0.01). females, we also stratified utilization of SE by gender
while still controlling for age at questionnaire comple-
SE Utilization tion. This analysis shows that, whereas both male and
Examination of the use of SE services by diagnosis female survivors are more likely to be in SE compared
type within age-at-diagnosis strata are presented in with same-gender siblings, the differences were
Table 2. These findings demonstrate, that after adjust- greater among females. For example, male survivors of
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Special Education among Cancer Survivors/Mitby et al. 1119

TABLE 2
Special Education Services by Age Diagnosis

Age 0–5 yrs Age 6–10 yrs Age 11–15 yrs Age 16–20 yrs

Diagnosis Yes (%)a ORb 95%CI Yes (%)a ORb 95%CI Yes (%)a ORb 95%CI Yes (%)a ORb 95%CI

Total
Leukemia 853 (35.9) 4.4 3.75–5.16 19.6 (26.2) 3.3 2.66–4.00 65 (13.3) 1.7 1.26–2.24 16 (9.5) 1.4 0.80–2.33
Central nervous system
tumor 437 (70.3) 18.8 15.01–23.49 235 (55.8) 11.9 9.48–14.98 109 (32.3) 5.3 4.08–6.92 27 (23.9) 4.1 2.55–6.55
Hodgkin disease 25 (33.8) 4.4 2.64–7.24 33 (14.7) 1.8 1.22–2.61 80 (13.6) 1.9 1.42–2.45 34 (5.7) 0.9 0.61–1.31
Non-Hodgkin lymphoma 46 (26.3) 2.9 2.01–4.14 52 (21.8) 2.6 1.84–3.57 37 (14.3) 1.9 1.32–2.78 8 (6.6) 1.0 0.46–2.11
Kidney 148 (18.6) 1.8 1.44–2.24 15 (12.8) 1.2 0.70–2.21 0 — — 0 — —
Neuroblastoma 169 (24.8) 2.5 2.02–3.13 8 (23.5) 2.5 1.15–5.31 0 — — 0 — —
Soft tissue sarcoma 95 (23.7) 2.6 1.96–3.33 33 (19.6) 2.3 1.52–3.40 20 (9.0) 1.1 0.70–1.84 16 (5.6) 1.4 0.80–2.32
Bone malignancy 10 (20.0) 2.1 1.01–4.16 32 (17.0) 1.9 1.28–2.87 41 (10.6) 1.3 0.94–1.91 23 (8.1) 1.2 0.78–1.94
Siblings 282 (9.2) 1.0 — 282 (9.2) 1.0 — 282 (9.2) 1.0 — 282 (9.2) 1.0 —
Males
Leukemia 428 (34.5) 2.9 2.37–3.58 112 (28.6) 2.7 2.03–3.48 31 (12.7) 1.1 0.76–1.71 13 (12.9) 1.3 0.68–2.35
Central nervous system
tumor 233 (70.4) 13.3 9.91–17.76 140 (60.6) 10.6 7.79–14.39 69 (35.8) 4.4 3.14–6.19 19 (29.2) 3.6 2.03–6.38
Hodgkin disease 22 (38.6) 3.8 2.13–6.65 30 (19.0) 1.7 1.13–2.66 49 (16.7) 1.7 1.17–2.36 26 (9.2) 1.0 0.62–1.53
Non-Hodgkin lymphoma 29 (24.2) 1.9 1.21–2.94 44 (24.4) 2.2 1.51–3.20 31 (17.4) 1.7 1.14–2.66 7 (8.2) 0.8 0.38–1.88
Kidney 84 (22.6) 1.7 1.23–2.21 5 (10.9) 0.8 0.31–2.07 0 — — 0 — —
Neuroblastoma 88 (27.9) 2.1 1.54–2.83 2 (15.4) 1.3 0.29–5.93 0 — — 0 — —
Soft tissue sarcoma 60 (26.7) 2.2 1.55–3.01 20 (20.2) 1.8 1.06–2.98 11 (10.6) 1.0 0.51–1.88 8 (9.0) 0.9 0.44–2.01
Bone malignancy 4 (19.1) 1.4 0.45–4.25 16 (16.8) 1.4 0.83–2.51 17 (9.2) 0.8 0.48–1.39 18 (11.1) 1.2 0.68–1.98
Siblings 184 (12.3) 1.0 — 184 (12.3) 1.0 — 184 (12.3) 1.0 — 184 (12.3) 1.0 —
Females
Leukemia 425 (37.4) 7.6 5.78–9.91 84 (23.6) 4.4 3.17–5.99 34 (13.9) 2.7 1.77–4.13 3 (4.4) 1.1 0.37–3.56
Central nervous system
tumor 204 (70.1) 30.5 21.40–43.50 95 (50.0) 13.7 9.51–19.66 40 (27.8) 6.7 4.36–10.31 8 (16.7) 4.4 2.00–9.63
Hodgkin disease 3 (17.7) 2.9 0.83–10.47 3 (4.55) 0.8 0.24–2.36 31 (10.5) 2.3 1.44–3.51 8 (2.6) 0.7 0.31–1.49
Non-Hodgkin lymphoma 17 (30.9) 5.5 2.94–10.30 8 (13.6) 2.2 0.94–4.92 6 (7.4) 1.4 0.60–3.35 1 (2.7) 0.4 0.01–14.69
Kidney 64 (15.1) 2.2 1.56–3.18 10 (14.1) 2.2 1.14–4.44 0 — — 0 — —
Neuroblastoma 81 (22.1) 3.5 2.47–4.92 6 (28.6) 5.2 1.99–13.36 0 — — 0 — —
Soft tissue sarcoma 35 (19.9) 3.2 2.07–4.96 13 (18.8) 2.9 1.49–5.80 9 (7.6) 1.5 0.73–3.05 8 (8.2) 2.0 0.94–4.19
Bone malignancy 6 (20.7) 3.3 1.29–8.45 16 (17.2) 3.0 1.73–5.34 24 (11.9) 2.4 1.48–3.90 5 (4.1) 1.0 0.42–2.30
Siblings 98 (6.3) 1.0 — 98 (6.3) 1.0 — 98 (6.3) 1.0 — 98 (6.3) 1.0 —

Yes: received special education services; OR: odds ratio; 95% CI: 95% confidence interval.
a
Participants who answered “don’t know” or did not respond to the question are not represented in this table.
b
ORs are adjusted by age at questionnaire completion

CNS tumors who were diagnosed between the ages of ⫽ 0.001) by era, with overall rates of 17%, 22%, and
0 years and 5 years were 13.3 times more likely to be in 25% for patients who were diagnosed during 1970 –
SE compared with male siblings (95%CI, 9.91–17.76), 1975, 1976 –1980, and 1981–1986, respectively.
whereas female survivors of CNS tumors who were Categories of treatment type were assigned to ex-
diagnosed in the same period were 30.5 times more amine the possible effect of CRT alone or combined
likely to be in SE compared with female siblings with IT MTX on the utilization of SE services (Fig. 1A),
(95%CI, 21.40 – 43.50). Additional analyses were per- which shows that SE use was associated significantly
formed restricting the use of SE to the period after with all three treatment categories: IT MTX only (OR,
completion of therapy. Comparison of siblings with 1.3; 95%CI, 1.09 –1.78), CRT only (OR, 7.2; 95%CI,
diagnosis specific subgroups of survivors who re- 6.14 – 8.39), and CRT plus IT MTX (OR, 2.6; 95%CI,
ported using SE after they completed therapy pro- 2.30 –2.95). Although both males and females showed
vided ORs comparable to those obtained when includ- increased risk for utilizing SE, the risk estimates for
ing both the on-therapy and off-therapy periods (data females were markedly higher compared with males in
not shown). Analysis of SE utilization by treatment era the groups that received CRT alone or in combination
revealed that the rates of SE differed significantly (P with IT MTX. Because the majority of survivors who
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
1120 CANCER February 15, 2003 / Volume 97 / Number 4

risk for entering SE, although the risk was somewhat

attenuated for the lower dose. Females accounted for
a significantly larger portion of survivors who received
CRT and used SE, with a clear dose response to in-
creasing levels of CRT.

Reasons for Entry into SE

A unique feature of this cohort is that participants
were asked to give the underlying reasons for being
placed in SE programs. A mark all that apply question
was given with possible responses of no, yes, or not
sure for the following reasons: missing school, low
scores on tests, problems with learning or concentrat-
ing, and emotional or behavioral problems. Data for
reasons given by age at diagnosis and diagnosis type
are provided in Table 3. Survivors who were diagnosed
between the ages of 0 and 15 years reported needing
SE services due to missed school. Low test scores also
were associated significantly with survivors who were
between the ages of 0 and 10 years at the time of
diagnosis. Compared with the sibling cohort, survivors
of all cancer diagnoses used SE services at an in-
creased rate due to missing school, with survivors of
bone cancer (OR, 7.4; 95%CI, 3.95–13.92) and soft tis-
sue sarcoma (OR, 6.2; 95%CI, 3.34 –11.33) showing the
FIGURE 1. Estimated odds ratios (OR) and 95% confidence intervals for strongest associations. Survivors of leukemia, CNS tu-
receiving special education according to groups defined by type of treatment mor, non-Hodgkin lymphoma, kidney, and neuroblas-
(A) and dose of cranial radiation (B) in centigrays (cGy). All analyses were toma showed significant increased use of SE due to
adjusted for age at the time of diagnosis and at the time the questionnaire was low test scores. Ideally, it would have been preferable
completed. Note that an odds ratio of 1.0 indicates no difference in the to show diagnosis specific and gender specific reasons
standard error between the study group and the referent group. When a for entering SE within age groups stratified by diagno-
confidence interval spanned the value of 1.0, we were unable to conclude that sis; however, unfortunately, even with a population
our findings were statistically significant. IT MTX: intrathecal methotrexate; this large, small cell sizes became problematic.
CRT: cranial radiation. Reasons for SE were also examined by treatment
data, adjusting for age at diagnosis and age at ques-
received both IT MTX and CRT were leukemia survi- tionnaire completion (data not shown). Survivors who
vors, a logistic regression analysis using leukemia sur- received CRT alone or in combination with IT MTX
vivors only was performed to identify a possible inter- demonstrated the most difficulty with low test scores
action between the two treatment modalities. (CRT only: OR, 1.4; 95%CI, 1.07–1.71; CRT plus IT
Dichotomous variables for IT MTX and CRT were MTX: OR, 1.4; 95%CI, 1.13–1.73) and problems with
measured against the combined effect of the two learning and concentrating (CRT only: OR, 3.2; 95%CI,
treatments. After adjusting for age at diagnosis and 2.26 – 4.42; CRT plus IT MTX: OR, 2.0; 95%CI, 1.51–
age at questionnaire completion, our data supported 2.67). When analyzing dose ranges, problems with
the appearance of a multiplicative effect between IT learning and concentrating were most evident among
MTX and CRT. The risk of needing SE associated with those who received the highest CRT doses (⬎ 5000
the combined effect of CRT and IT MTX was 2.7 times cGy: OR, 2.7; 95%CI, 1.44 – 4.89). Female gender, again,
the risk relative to what would be expected if the two was associated with the highest risk of utilizing SE
treatments were independent of each other (OR, 2.7; within these treatment groups.
95%CI 1.50 – 4.76; data not shown).
ORs for the use of SE also were calculated accord- Duration of SE Services
ing to CRT dose (Fig. 1B). The risk of using SE services In an effort to determine whether reported utilization
increased significantly as the level of CRT increased. It of SE was either an acute intervention strategy for
was found that dose ranges that included both 1800 survivors of childhood cancer or a long-term compo-
centigrays (cGy) and 2400 cGy increased a survivor’s nent of their education experience, we examined du-
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Special Education among Cancer Survivors/Mitby et al. 1121

TABLE 3
Reasons for Special Education Servicesa

Missed school Low tests Learning/concentrating Emotional/behavioral

Characteristic Yes (%)b ORc 95%CI Yes (%)b ORc 95%CI Yes (%)b ORc 95%CI Yes (%)b ORc 95%CI

Age at diagnosis (yrs)

0–5 306 (18.3) 2.2 1.20–4.05 908 (54.8) 2.4 1.48–3.81 1461 (86.8) 1.5 0.89–2.53 219 (13.4) 1.5 0.69–3.27
6–10 214 (38.8) 5.1 2.84–9.16 272 (50.4) 2.0 1.28–3.22 421 (76.7) 0.9 0.56–1.52 62 (11.7) 1.3 0.62–2.85
11–15 114 (37.5) 4.0 2.24–7.24 94 (31.5) 0.9 0.59–1.52 213 (71.2) 0.8 0.51–1.38 45 (15.2) 1.9 0.87–3.96
16–20 16 (14.8) 1.0 — 35 (32.4) 1.0 – 78 (71.6) 1.0 — 9 (8.6) 1.0 —
Diagnosis
Leukemia 164 (29.3) 5.0 3.08–8.10 558 (53.4) 1.8 1.35–2.38 908 (85.3) 1.3 0.94–1.91 113 (11.0) 0.6 0.43–0.94
Central nervous system tumor 109 (24.0) 3.6 2.21–5.94 376 (50.8) 1.7 1.26–2.26 659 (88.2) 1.9 1.27–2.70 87 (12.0) 0.7 0.47–1.06
Hodgkin disease 49 (37.4) 6.1 3.42–11.00 54 (40.0) 1.3 0.85–2.02 93 (66.0) 0.6 0.39–1.01 18 (13.2) 0.8 0.46–1.51
Non-Hodgkin lymphoma 35 (35.4) 6.1 3.33–11.06 65 (48.5) 1.7 1.09–2.49 94 (72.3) 0.7 0.43–1.17 19 (14.7) 0.9 0.51–1.65
Kidney 16 (26.2) 4.4 2.13–9.06 83 (56.5) 2.0 1.30–2.95 126 (82.9) 1.0 0.61–1.77 35 (23.7) 1.6 0.94–2.63
Neuroblastoma 11 (20.0) 3.1 1.33–6.96 81 (50.6) 1.5 1.00–2.26 135 (83.9) 1.1 0.62–1.82 30 (19.1) 1.2 0.70–2.04
Soft tissue sarcoma 36 (36.0) 6.2 3.34–11.33 59 (38.8) 1.1 0.71–1.62 102 (65.8) 0.5 0.32–0.78 18 (11.8) 0.7 0.39–1.28
Bone cancer 35 (41.2) 7.4 3.95–13.92 33 (36.7) 1.1 0.67–1.81 56 (62.2) 0.5 0.30–0.86 15 (16.7) 1.1 0.56–2.08
Siblings 20 (7.6) 1.0 — 96 (36.8) 1.0 — 205 (78.2) 1.0 — 41 (15.9) 1.0 —

OR: odds ratio; 95%CI: 95% confidence interval.

a
Results were based on 2,866 participants who indicated they were in special education.
b
The Yes (%) column indicates those who answered “yes” within each strata. Participants who answered “don’t know” or who did not indicate specific reasons are not represented in this table.
c
ORs are adjusted by age at questionnaire completion.

ration of SE services relative to grade level at diagno-

sis. Figure 2 represents the overall frequency of SE by
grade level at diagnosis for survivors and siblings.
Children who were diagnosed at younger ages tended
to be placed in SE services and remained in those
programs for a longer time. Duration of SE services
also was examined comparing survivors who received
CRT with survivors who did not receive CRT (data not
shown). The mean number of years in SE was 5.7 years
for the group that received CRT and 4.7 for the group
that did not receive CRT (t ⫽ 6.77; P ⬍ 0.01). Further-
more, a statistically significant difference was found in
the duration of SE services between survivors who
received no CRT, survivors who received low-dose
CRT(1–1999 cGy), and survivors who received high-
dose CRT (ⱖ 2000 cGy) using a one way analysis of
variance (F ⫽ 25.91; 2 degrees of freedom; P ⬍ 0.01).
The means for all three groups were significantly dif-
ferent (P ⫽ 0.05) from each other, with survivors who
received high-dose CRT needing SE services for the
longest time (5.8 years) and survivors who never re-
ceived CRT needing them for the shortest time (4.7
years).

Educational Attainment
Educational attainment of survivors, compared with
members of the sibling cohort, is presented in Table 4.
Logistic regression analysis showed that survivors of FIGURE 2. Overall frequencies of special education by grade at the time of
leukemia, CNS tumors, non-Hodgkin lymphoma, and diagnosis (patients with all cancer diagnoses and siblings).
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
1122 CANCER February 15, 2003 / Volume 97 / Number 4

TABLE 4
Educational Attainment Overall and by Enrollment into Special Educationa

% SE % SE
enrollees enrollees
% Non-high % Non- who did not who did not
school college complete graduate
Characteristic completersb OR 95%CI graduatesa OR 95%CI high school OR 95%CI college OR 95%CI

Diagnosis
Sibling controls 8.7 1.0 — 60.9 1.0 — 20.5 1.0 — 81.3 1.0 —
Leukemia 14.2 1.6 1.23–2.16 76.2 1.3 1.15–1.47 26.6 1.4 0.75–2.57 88.2 1.2 0.71–1.86
Central nervous system tumor 18.1 2.7 1.92–3.81 77.1 1.6 1.38–1.92 29.4 2.0 1.04–3.73 23.4 2.3 1.36–3.91
Hodgkin disease 7.1 1.2 0.71–2.13 60.7 1.1 0.97–1.25 12.0 0.8 0.30–2.19 76.6 0.9 0.49–1.47
Non-Hodgkin lymphoma 13.1 1.8 1.15–2.78 68.6 1.2 0.97–1.37 28.6 1.5 0.64–3.61 84.0 1.0 0.54–1.95
Kidney 12.0 1.3 0.88–1.97 79.7 1.4 1.13–1.75 39.5 2.6 1.13–5.93 90.5 1.3 0.52–3.46
Neuroblastoma 15.3 1.7 1.14–2.61 79.0 1.3 1.00–1.74 32.0 1.9 0.80–4.31 88.5 1.0 0.43–2.50
Soft tissue sarcoma 9.1 1.1 0.71–1.83 64.3 1.0 0.84–1.16 21.7 1.4 0.53–3.49 83.5 1.0 0.55–2.15
Bone cancer 9.0 1.5 0.89–2.52 61.0 0.9 0.81–1.10 16.7 1.2 0.36–3.83 79.8 0.9 0.48–1.77
Treatment
Sibling controls 8.7 1.0 — 60.9 1.0 — 20.5 1.0 — 81.3 1.0 —
Neither CRT nor IT MTX 11.2 1.5 1.10–1.94 65.8 1.1 0.99–1.20 26.3 1.7 0.88–3.09 83.7 1.1 0.68–1.68
CRT only 16.8 2.5 1.76–3.67 78.0 1.7 1.41–2.03 27.5 1.8 0.92–3.43 91.0 1.9 1.08–3.17
IT MTX only 14.2 1.6 1.11–2.18 72.8 1.1 0.97–1.31 34.6 1.9 0.90–3.78 90.2 1.5 0.81–2.90
Both CRT and IT MTX 14.6 1.8 1.33–2.42 77.4 1.4 1.19–1.58 24.8 1.3 0.70–2.43 88.0 1.1 0.68–1.82

OR: odds ratio; 95%CI: 95% confidence interval; SE: special education; CRT: cranial radiation; IT MTX: intrathecal methotrexate.
a
The ORs for this table are adjusted by age at questionnaire completion. They reflect the probability that of survivors are more likely than siblings to be non-completers of high school and college.
b
Calculated using patients age 18–24 years at questionnaire completion for comparison with national data.
c
Calculated using the whole adult cohort.

neuroblastoma were significantly less likely to com- uation for survivors who were in SE programs, and
plete high school compared with siblings. No signifi- only survivors who were in SE and received CRT were
cant difference in high school completion was identi- significantly less likely to complete college compared
fied between siblings and survivors of Hodgkin with siblings.
disease, kidney, soft tissue sarcoma, and bone tumors.
Furthermore, comparison of treatment types (no CRT DISCUSSION
or IT MTX, CRT only, IT MTX only, and CRT plus IT Long-term sequelae of childhood cancer have been
MTX) showed that all survivors, regardless of their the focus of a large amount of research over the past 3
type of treatment, were significantly less likely to com- decades. Unfortunately, issues such as small sample
plete high school compared with their siblings. Col- sizes, single-institution populations, and short length
lege graduates showed a pattern similar to those who of follow-up often have hindered the generalizibility of
completed high school: Typically, the same diagnostic important findings among this group. Most of the
groups were less likely to complete college compared research involving educational difficulties has focused
with siblings. on cognitive testing through various intelligence quo-
A slightly different picture emerged when we ex- tient (IQ) measurements, whereas little attention has
amined high school and college completion for those been paid to actual SE placement. In one of the few
who had ever received SE. The only survivors who published reports that examined SE, Haupt et al. stud-
received SE services and were significantly less likely ied survivors of ALL who were more likely than their
to complete high school compared with siblings were siblings to require SE services.9 Similarly, Mulhern et
patients who were diagnosed with CNS tumors (OR, al. also studied ALL survivors and found they had a
2.0; 95%CI, 1.04 –3.73) and kidney cancer (OR, 2.6; greater frequency of SE intervention compared with
95%CI, 1.13–5.93). Survivors of CNS tumors who re- survivors of Wilms tumor.10 The CCSS is able to ex-
ceived SE also were less likely to finish college com- pand on previous literature not only by determining
pared with siblings, but patients with all other diag- utilization of SE and educational attainment for sur-
noses showed no significant difference with siblings in vivors of ALL but also by describing and comparing
terms of college completion. Treatment type did not these outcomes among a much broader spectrum of
show a significant correlation with high school grad- childhood cancer survivors.
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Special Education among Cancer Survivors/Mitby et al. 1123

The current analysis was conducted to identify 1) residence nor school district information was included
the utilization of SE services by patient and disease in the baseline survey and, thus, was not available for
characteristics and 2) the level of education attained analysis. The rate of SE utilization among survivors did
among survivors and siblings. However, the results of vary by treatment era in this analysis, with rates lower
this analysis must be interpreted within the confines in the earlier periods. It is unclear how much of this
of the study’s limitations. Again, it is important to effect may be attributable to changes in treatment and
mention that the data used for this analysis were self- how much may be attributable to increases in educa-
reported and were not validated through school tional funding during this era. It is a fact that federal
records. Whether or not a participant indicated they support for SE has increased steadily during the pe-
received SE services depended on their perception of riod under study.
what SE is. Furthermore, there is potential for bias The current analysis supports previous research
when examining the reasons for entering SE, in that it showing that survivors of leukemia are at increased
probably is more desirable socially to report the need risk for utilizing SE services. However, an important
for SE due to missing school rather than low test distinction between this study and previous research
scores. It also is possible that some survivors may have is that not only were survivors of leukemia more likely
been placed into SE programs preemptively by school than siblings to utilize SE services, but survivors of all
staff in an effort to provide as much assistance as the major childhood cancer diagnoses utilized these
possible to these children, a benefit probably not of- services more often compared with siblings. This in-
fered equally to siblings. In addition, approximately cludes survivors with diagnoses for which CNS treat-
10% of survivors in the CCSS are from racial minority ment is administered rarely, if ever.
groups, which is slightly lower compared with the Risk factors previously identified to affect cogni-
published SEER data for this same period (16%).11 tion and poorer educational outcomes include early
Because minority students typically receive SE at age of treatment,3,6,10,13,14 female gender,3,13,15,16 and
higher rates compared with white students,12 we may the administration of CRT.2– 4,9 –11,14,17 In the current
be underestimating the utilization of SE within the study, 72% of survivors of leukemia and 58% of survi-
survivor population. Moreover, although it has been vors of CNS tumors were diagnosed before age 11
shown that parental education level and SES affect a years, whereas the majority of survivors of other can-
child’s educational attainment, these variables were cers (excluding kidney and neuroblastoma) were di-
not available for the current analysis. One method to agnosed later in adolescence. In terms of treatment,
control for parental education and SES effects would 94% of survivors of leukemia received IT MTX and/or
be to use a 1:1 match of full siblings. Because this CRT, and 59% of survivors of CNS tumors were treated
analysis included closer to a 4:1 match, we were not with CRT. Although the percentage of survivors of CNS
able to control completely for these effects on the tumors who received CRT may seem high by current
utilization of SE and educational attainment in our treatment standards, it is reflective of the overall pop-
cohort. Finally, because information relating to SE was ulation of patients with CNS tumors who were treated
obtained from two different sources (i.e., directly from during the study period of 1970 –1986.
survivors age ⱖ 18 years and from parents if the sur- Because both diagnosis and treatment are corre-
vivor was age ⬍ 18 years or had died subsequently), lated highly with age at diagnosis, SE use was exam-
there is the potential for introduction of a reporting ined by diagnosis within age at diagnosis strata. This
bias. We investigated the frequency of reported SE analysis showed a significant increase in the utiliza-
utilization by year of cancer diagnosis and found no tion of SE services for survivors who were diagnosed at
consistent pattern suggestive of under-reporting or younger ages, with the strongest association found in
over-reporting according to survivor or parental re- females. We found a ⬎ 4-fold total increased risk of
port. entering an SE program for survivors of leukemia who
The baseline questionnaire for CCSS asked about were diagnosed between the ages of 0 and 5 years (2.9
placement into advanced program classes. Because times higher in males and 7.6 times higher in females).
these types of programs vary greatly in terms of con- Although it is widely accepted that CRT is associ-
tent and accessibility, not only within each state but ated with diminished cognitive functioning, the dose
across the country, we felt that an analysis for ad- level at which these problems occur seems to be in-
vanced programs would not be appropriate. SE ser- conclusive. A large proportion of previous studies
vices, conversely, are at least somewhat consistent found that patients who received at least 2400 cGy
between states, because federal funding is appropri- were at the greatest risk;3,4,9,10,13,14 while a few studies
ated for such classes for all school districts, although also have reported this association in individuals who
additional state funding may vary. Neither state of received a lower dose of radiation: typically, 1800
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
1124 CANCER February 15, 2003 / Volume 97 / Number 4

cGy.2,17 In addition, a possible synergistic effect be- when examining off-therapy relative odds. However,
tween the administration of IT MTX in combination because the question relating to the reasons for SE was
with CRT also has been implicated.18,19 The current not specific for each year in SE, we were unable to
analysis shows an association between the utilization determine whether reasons for continued SE use
of SE services and survivors who were treated with changed over time.
CRT. This association is evident when examining CRT We also identified low test scores as one of the
as a dichotomous variable and by dose range. We reasons associated with utilizing SE services for survi-
found that survivors who received CRT alone were vors who were diagnosed before age 11 years as well as
seven times more likely to utilize SE services com- for survivors of kidney cancer, CNS tumors, and non-
pared with survivors who did not receive either CRT or Hodgkin lymphoma. The questionnaire, however, did
IT MTX. It is important to note that the dose level of not distinguish between low scores on standardized
CRT may be a better predictor of SE compared with tests or individual subject tests. Therefore, some of the
whether a survivor ever received CRT. Nearly 74% of association between low test scores and the need for
survivors who received CRT alone are survivors of CNS SE may be related to missing too much of a particular
tumors. Because these patients typically receive subject area.
higher doses of CRT, this association may be due to a Survivors who received CRT also appear to have
higher radiation dose within the CRT-only group. problems with learning and concentrating. Only sur-
However, when CRT is administered in combination vivors of CNS tumors showed an elevated risk due to
with IT MTX, the deleterious effects of CRT seem to be this cause. CRT, which is the most common form of
compounded. It also is evident that, although a lower treatment for these patients, also was associated sig-
dose of CRT seems to modify the risk of utilizing SE nificantly with problems of learning and concentrat-
services, the risk remains significantly elevated com- ing. When we examined CRT by dose range, we found
pared with the risk in survivors who did not receive that the association primarily held true only for survi-
any CRT. vors who received the highest dose of CRT. It also is
A secondary focus of this analysis was to deter- possible that specific cognitive deficits among the
mine the reasons why survivors of childhood cancer group with CNS tumors are the result at least in part to
are placed into SE programs. The format of this project the location of the tumor and/or the surgical interven-
was such that we were only able to measure the use of tion, two factors that were not included in the current
SE and the self-reported reasons for entering such analysis. It is worth noting that emotional and behav-
programs. It did not allow us to make any conclusions ioral problems were not associated with the need for
about cognitive deficits among survivors of childhood SE for any group of survivors in terms of diagnosis
cancer. Undoubtedly, some survivors of childhood category, age at diagnosis, or treatment type.
cancer are placed into SE because of cognitive impair- The current report indicates that survivors of leu-
ments, but it is also possible that survivors are placed kemia, CNS tumors, non-Hodgkin lymphoma, and
into SE for reasons not necessarily due to cognition. neuroblastoma were significantly less likely to gradu-
Clearly, when school-age children are diagnosed with ate high school compared with siblings. This is in
cancer or other serious childhood illnesses, absentee- contrast to previous findings. One study found that,
ism becomes an important issue during the treatment although survivors of ALL were more likely than sib-
phase of the disease. Standard therapy for patients ling controls to enter an SE or learning-disabled pro-
with childhood cancers often consist initially of inten- gram, most were able to overcome their problems and
sive therapy that may last several months to 1 year. generally had the same probability as their siblings of
This often is followed by a less intensive regimen that finishing high school, entering college, and earning a
may continue for several more years. The length of bachelors degree.5 A similar study that was conducted
treatment can have a serious impact on scholastic among patients who were diagnosed with several dif-
functioning. When a child is receiving treatment on a ferent types of childhood cancer also found that, ex-
regular schedule, it is very likely that the same school cept for patients with CNS tumors, survivors of child-
subject is missed routinely. We found that missing hood cancer had no impairment of educational
school was a significant factor in determining place- attainment compared with their sibling controls.20
ment into SE programs for survivors of all cancer An encouraging aspect of this investigation is that
diagnoses, with survivors of bone cancer, soft tissue when we examined high school graduation rates of
sarcoma, Hodgkin disease, and non-Hodgkin lym- survivors who were enrolled in SE programs, we found
phoma reporting a increased risk of six times the risk that, in general, they were no less likely to complete
for siblings. It was found that this association was high school compared with other children in SE with-
significantly higher for the same groups of survivors out a cancer history. We have no clear explanation for
 10970142, 2003, 4, Downloaded from https://acsjournals.onlinelibrary.wiley.com/doi/10.1002/cncr.11117 by CAPES, Wiley Online Library on [09/03/2024]. See the Terms and Conditions (https://onlinelibrary.wiley.com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
Special Education among Cancer Survivors/Mitby et al. 1125

the apparently high proportion of survivors of kidney be an invaluable resource in better understanding the
cancer who received SE and did not graduate from potential late effects of treating young children and
high school. The therapy they received would not be adolescents for malignant disease. In terms of this
expected to affect their scholastic ability, nor would it investigation, the CCSS is unique in that it is able to
render them more likely to utilize SE at all. However, describe the educational achievements of a much
the absolute numbers are small and are not very dif- broader spectrum of childhood cancer diagnoses than
ferent from the numbers for survivors of neuroblas- any other publication to date. Although treatment fac-
toma, who have a similar age specific distribution. tors, such as CRT and IT MTX clearly play a role in
With the exception of survivors of CNS tumors who poor academic achievement, this study shows that, to
used SE, survivors of all other types of cancer also some degree, other non-CNS treatment variables also
were just as likely to complete college as siblings, can impact scholastic functioning.
suggesting that interventions may be effective among Although the results from the current analysis of
survivors who are less likely to have received treat- the CCSS cohort provide important information, there
ment involving the CNS. is a clear need for researchers to further investigate the
Although it is true that most long-term survivors utilization of SE and educational attainment among
of childhood cancer will complete high school suc- childhood cancer survivors. The CCSS is under-repre-
cessfully, and many will go on to college, some survi- sented in terms of racial minority survivors; thus, the
vors will have significant difficulty doing so. By iden- potential confounding effect of race and SES needs to
tifying the individuals most at risk for problems with be examined in other, more diverse survivor popula-
school performance, clinicians and educators will be tions. Understanding the influence of race and SES
able to foresee potential problem areas better and can may provide important insights into how to maximize
choose to initiate SE services early on in these chil- educational opportunities within racial/ethnically de-
dren’s education. Several long-term follow-up clinics fined subgroups. Moreover, with the increasing num-
have created models for the interaction between phy- ber of immigrant children of non-English speaking
sicians, nurses, and school staff members to better families, it also would be beneficial to focus research
serve the increasing population of cancer patients and on this distinct population. Another area that the cur-
survivors at risk for cognitive impairments.21,22 rent analysis was unable to address adequately related
Recommendations have been made suggesting to the reason surrounding the need for SE over time. It
that baseline IQ and achievement tests and yearly is likely that individuals who are newly diagnosed with
sequential testing should be performed on all children a childhood cancer may be placed into SE due to an
who are treated for CNS cancers because of the high
excessive number of school days missed. However, is
incidence of learning disabilities in this population.23
it still true that children are in SE years later for the
In light of the current research findings, an argument
same reason, potentially due to somatic issues as a
can be made that these tests should be performed in a
result of their treatment, or do low test scores and
much larger segment of children who are diagnosed
behavioral issues tend to develop at a later time? Our
with cancer. Clearly, children who are treated with
analysis showed that rates of SE differed significantly
CRT and IT MTX should be followed much more care-
across the period of eligibility for CCSS. SE went
fully to identify early signs of learning disabilities.
through a considerable amount of reform during the
Children who already are at risk for poorer educa-
1970s, which may or may not have had a significant
tional outcomes, such as racial minorities and patients
impact on our findings. Additional research may be
from lower SES families, also should be monitored
limited to include more recently diagnosed and
closely to determine the extent to which the diagnosis
treated children to evaluate current SE standards and
of cancer further impedes school performance. Be-
criteria more closely. Future research clearly is needed
cause providing at-risk children with the necessary SE
to address these issues and aid physicians, nurses,
services and guidance from teachers and other men-
social workers, and educators to work together to de-
tors early on can be the key to successful completion
velop individualized plans that help survivors accom-
of scholastic goals. We can also suggest all children
diagnosed and treated for cancer should be monitored plish educational goals.
closely and evaluated to allow for early intervention.
The CCSS is comprised of a wide age range of over
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