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Validation of questionnaire-reported hearing with medical records: A report from the Swiss Childhood Cancer Survivor Study

Authors:
  • Bayerisches Landesamt für Pflege
  • University Children's Hospital Bern Inselspital

Abstract and Figures

Background Hearing loss is a potential late effect after childhood cancer. Questionnaires are often used to assess hearing in large cohorts of childhood cancer survivors and it is important to know if they can provide valid measures of hearing loss. We therefore assessed agreement and validity of questionnaire-reported hearing in childhood cancer survivors using medical records as reference. Procedure In this validation study, we studied 361 survivors of childhood cancer from the Swiss Childhood Cancer Survivor Study (SCCSS) who had been diagnosed after 1989 and had been exposed to ototoxic cancer treatment. Questionnaire-reported hearing was compared to the information in medical records. Hearing loss was defined as ≥ grade 1 according to the SIOP Boston Ototoxicity Scale. We assessed agreement and validity of questionnaire-reported hearing overall and stratified by questionnaire respondents (survivor or parent), sociodemographic characteristics, time between follow-up and questionnaire and severity of hearing loss. Results Questionnaire reports agreed with medical records in 85% of respondents (kappa 0.62), normal hearing was correctly assessed in 92% of those with normal hearing (n = 249), and hearing loss was correctly assessed in 69% of those with hearing loss (n = 112). Sensitivity of the questionnaires was 92%, 74%, and 39% for assessment of severe, moderate and mild bilateral hearing loss; and 50%, 33% and 10% for severe, moderate and mild unilateral hearing loss, respectively. Results did not differ by sociodemographic characteristics of the respondents, and survivor- and parent-reports were equally valid. Conclusions Questionnaires are a useful tool to assess hearing in large cohorts of childhood cancer survivors, but underestimate mild and unilateral hearing loss. Further research should investigate whether the addition of questions with higher sensitivity for mild degrees of hearing loss could improve the results.
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RESEARCH ARTICLE
Validation of questionnaire-reported hearing
with medical records: A report from the Swiss
Childhood Cancer Survivor Study
Annette Weiss
1
, Grit Sommer
1
, Rahel Kuonen
1
, Katrin Scheinemann
2
, Michael Grotzer
3
,
Martin Kompis
4
, Claudia E. Kuehni
1,5
*, on behalf of the Swiss Paediatric Oncology Group
(SPOG)
1Swiss Childhood Cancer Registry, Institute of Social and Preventive Medicine, University of Bern, Bern,
Switzerland, 2Division of Pediatric Hematology/ Oncology, University Children‘s Hospital Basel, Basel,
Switzerland, 3Department of Pediatric Oncology, University Children‘s Hospital Zurich, University of Zurich,
Zurich, Switzerland, 4Department of ENT, Head and Neck Surgery, University Hospital Bern, University of
Bern, Bern, Switzerland, 5Children’s University Hospital of Bern, University of Bern, Bern, Switzerland
These authors contributed equally to this work.
¶ Membership of the Swiss Paediatric Oncology Group (SPOG) Scientific Committee is provided in the
Acknowledgments.
*claudia.kuehni@ispm.unibe.ch
Abstract
Background
Hearing loss is a potential late effect after childhood cancer. Questionnaires are often used
to assess hearing in large cohorts of childhood cancer survivors and it is important to know if
they can provide valid measures of hearing loss. We therefore assessed agreement and
validity of questionnaire-reported hearing in childhood cancer survivors using medical rec-
ords as reference.
Procedure
In this validation study, we studied 361 survivors of childhood cancer from the Swiss Childhood
Cancer Survivor Study (SCCSS) who had been diagnosed after 1989 and had been exposed
to ototoxic cancer treatment. Questionnaire-reported hearing was compared to the information
in medical records. Hearing loss was defined as grade 1 according to the SIOP Boston Oto-
toxicity Scale. We assessed agreement and validity of questionnaire-reported hearing overall
and stratified by questionnaire respondents (survivor or parent), sociodemographic character-
istics, time between follow-up and questionnaire and severity of hearing loss.
Results
Questionnaire reports agreed with medical records in 85% of respondents (kappa 0.62),
normal hearing was correctly assessed in 92% of those with normal hearing (n = 249), and
hearing loss was correctly assessed in 69% of those with hearing loss (n = 112). Sensitivity
of the questionnaires was 92%, 74%, and 39% for assessment of severe, moderate and
mild bilateral hearing loss; and 50%, 33% and 10% for severe, moderate and mild unilateral
PLOS ONE | https://doi.org/10.1371/journal.pone.0174479 March 23, 2017 1 / 15
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OPEN ACCESS
Citation: Weiss A, Sommer G, Kuonen R,
Scheinemann K, Grotzer M, Kompis M, et al.
(2017) Validation of questionnaire-reported hearing
with medical records: A report from the Swiss
Childhood Cancer Survivor Study. PLoS ONE 12
(3): e0174479. https://doi.org/10.1371/journal.
pone.0174479
Editor: Marta M. Alonso, Universidad de Navarra,
SPAIN
Received: December 15, 2016
Accepted: March 9, 2017
Published: March 23, 2017
Copyright: ©2017 Weiss et al. This is an open
access article distributed under the terms of the
Creative Commons Attribution License, which
permits unrestricted use, distribution, and
reproduction in any medium, provided the original
author and source are credited.
Data Availability Statement: All relevant data are
within the paper and its Supporting Information
files.
Funding: This study was supported by the Swiss
Cancer Research (grant no: 02783-02-2011),
Swiss Cancer League (grant no: 3412-02-2014),
and received funding from the European Union’s
Seventh Framework Programme for research,
technological development and demonstration
under grant agreement no 602030. The work of the
hearing loss, respectively. Results did not differ by sociodemographic characteristics of the
respondents, and survivor- and parent-reports were equally valid.
Conclusions
Questionnaires are a useful tool to assess hearing in large cohorts of childhood cancer sur-
vivors, but underestimate mild and unilateral hearing loss. Further research should investi-
gate whether the addition of questions with higher sensitivity for mild degrees of hearing
loss could improve the results.
Introduction
Hearing loss is a late effect of childhood cancer that is especially common after ototoxic treat-
ments such as platinum chemotherapy, cranial radiation, and brain surgery [13]. Audiomet-
ric testing is the best way to assess hearing loss, but particularly for the purpose of research
comprehensive audiometric testing in large cohorts of childhood cancer survivors is not
always feasible. Questionnaires are often used instead to ask about survivors’ hearing [2,46].
Previous studies of the validity of questionnaire-reported hearing have been conducted
mainly in older populations [710]. Childhood cancer survivors are different. First, they may
have had several hearing tests during cancer treatment and long-term follow-up. After comple-
tion of cranial radiation therapy, a yearly audiological evaluation for five years is recom-
mended for survivors. After completion of platinum chemotherapy, a baseline evaluation at
entry into follow-up is recommended, followed by yearly testing if hearing loss is detected
[11]. Second, survivors can develop hearing loss very early in life and questionnaires for young
survivors are often directed to their parents [5,12]. It is important to know if validity depends
on the person who answered the questionnaire (survivor or parent), and if sociodemographic
and clinical characteristics of survivors also play a role. However, no study has yet examined
the validity of questionnaire-reported hearing in childhood cancer survivors.
We therefore assessed validity and agreement of questionnaire-reported hearing in child-
hood cancer survivors using medical records as reference, and determined factors associated
with agreement including the person answering the questionnaire (survivor or parent), sociode-
mographic characteristics, time between follow-up and questionnaire, and levels of hearing loss.
Methods
Study population
The Swiss Childhood Cancer Survivor Study (SCCSS) is a population-based, long-term cohort
study of all patients registered in the Swiss Childhood Cancer Registry (SCCR) who were diag-
nosed between 1976 and 2005 at age 20 years, and survived 5 years after initial diagnosis
of cancer [12]. The SCCR is a population-based registry that includes all children and adoles-
cents in Switzerland diagnosed with leukemia, lymphoma, central nervous system (CNS)
tumors, malignant solid tumors, or Langerhans cell histiocytosis before the age of 21 [13]. Eth-
ics approval was granted through the Ethics Committee of the Canton of Bern to the SCCR
and SCCSS (KEK-BE: 166/2014).
Inclusion criteria
We included 2175 survivors in this validation study who were diagnosed in a Swiss specialized
pediatric oncology (SPOG) clinic after 1989 because medical records of patients diagnosed
Validation of questionnaire-reported hearing with medical records
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Swiss Childhood Cancer Registry is supported by
the Swiss Paediatric Oncology Group (www.spog.
ch), Schweizerische Konferenz der kantonalen
Gesundheitsdirektorinnen und –direktoren (www.
gdk-cds.ch), Swiss Cancer Research (www.
krebsforschung.ch), Kinderkrebshilfe Schweiz
(www.kinderkrebshilfe.ch), the Federal Office of
Health (FOH) and the Institute of Cancer
Epidemiology and Registration (www.nicer.ch).
The funders had no role in study design, data
collection and analysis, decision to publish, or
preparation of the manuscript.
Competing interests: The authors have declared
that no competing interests exist.
earlier were rarely available (Fig 1). To determine whether they had been exposed to ototoxic
cancer treatment, we obtained treatment-related information from the SCCR that included
type and year of cancer diagnosis, and age at cancer diagnosis, chemotherapy, radiotherapy,
surgery, and bone marrow transplantation (BMT). Cancer diagnoses were classified according
to the International Classification of Childhood Cancer, third edition (ICCC-3) [14].
Assessment of hearing
The SCCSS questionnaire survey. Between 2007 and 2013, we traced all addresses of sur-
vivors and sent them a long questionnaire. Parents of survivors 15 years old completed the
questionnaire for their children. Survivors who were 16 completed questionnaires them-
selves. Nonresponders received a second copy of the questionnaire. If they again did not
respond, we contacted them by phone. The main topics covered by the questionnaire were
quality of life, somatic health, use of current medication and health services, psychological
distress, health behaviors, and socioeconomic status. The detailed study design has been
Fig 1. Flow chart of study population. Abbreviations: SPOG, Swiss Pediatric Oncology Group; SCCSS,
Swiss Childhood Cancer Survivor Study.
a
SPOG clinics including the following clinics with paediatric oncology
units: Kantonsspital Aarau AG, Universita
¨ts-Kinderspital Basel, Ospedale S. Giovanni Bellinzona, Universita
¨ts-
Kinderklinik Bern, Hospital des Enfants Geneve, CHUV Lausanne, Kantonsspital Luzern, Ostschweizer
Kinderspital St. Gallen, Universita
¨ts-Kinderspital Zu¨rich.
b
Includes survivors who have received platinum
compounds or cranial radiation.
c
Includes survivors who have not received platinum compounds or cranial
radiation.
https://doi.org/10.1371/journal.pone.0174479.g001
Validation of questionnaire-reported hearing with medical records
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published elsewhere [12]. The SCCSS questionnaire included questions on hearing (S1 Fig).
We asked the participant (or participant’s parent) whether she/he had ever been told by a doc-
tor that she/he had problems with hearing. If the answer was yes, the participant was asked to
indicate the laterality (unilateral/bilateral) of the problem, and to identify its severity by select-
ing one of the three ratings: mild problems, not requiring hearing aids or completely corrected
with hearing aid; moderate problems, not completely corrected by hearing aid; or severe prob-
lems, not correctable by hearing aid (deaf). We categorized severity of questionnaire-reported
hearing loss as mild, moderate, or severe and created also a binary variable (yes/no) for ques-
tionnaire-reported hearing loss that combined those with mild, moderate or severe hearing
loss in one category and those with normal hearing to another category.
Medical records. In 2015 we collected hearing tests, the corresponding audiologists´
reports, and oncological follow-up reports from the original medical records in the clinic
archives of ear-nose-throat (ENT) departments and pediatric oncology clinics. When we
found multiple reports or tests with hearing results that differed, we used the most recent
result. We only considered reports or tests that were dated before the completion of the SCCSS
questionnaire. The first author (A.W.) graded all hearing tests from these records for each ear
separately and for frequencies up to 8 kHz according to the SIOP Boston Ototoxicity Scale (Fig
2) [15]. A.W. was blinded to the hearing outcome measured by the questionnaire. Unclear
cases were discussed with a senior audiologist (M.K.). When hearing tests were accompanied
by the reports of audiologists, A.W. used the audiologist results in a second step to validate her
grading. When we found only oncological follow-up reports, we looked for information on
hearing in medical histories and examinations. If no information on hearing was available in
the oncological follow-up reports, we assumed normal hearing and assigned grade 0 (20 dB)
of the SIOP Boston Ototoxicity Scale. To justify the use of oncological follow-up reports only
Fig 2. Flow chart on data collection via medical records. Abbreviations: SIOP, International Society of
Pediatric Oncology; ENT, Ear-nose-throat.
a
We collected information from medical records of the paediatric
oncological departments and ENT departments.
b
When we found more than one report or hearing test with
differing results, we used the most recent report/test.
https://doi.org/10.1371/journal.pone.0174479.g002
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for validation purposes, we checked the reports of 45 randomly selected persons with hearing
loss according to hearing tests to see if the results of the hearing test were mentioned in the cor-
responding oncological follow-up report. In 41 out of 45 (91%) cases, the pathological result of
the hearing test was mentioned in the oncological report. This indicates that hearing test results
were forwarded to the pediatric oncology departments and recorded in oncological follow-up
reports as part of clinical standard procedures. We defined hearing loss from medical records
as grade 1 (>20 dB above 4kHz) according to the SIOP Boston Ototoxicity scale in the most
affected ear. We graded severity of hearing loss from medical records from 0 to 4 [15]. We also
noted the date of the follow-up used for grading of hearing and categorized the interval between
that follow-up and questionnaire into four categories: 0–2, 3–4, 5–9, and 10–17 years.
Statistical analysis
First, we determined prevalence, severity, and laterality of hearing loss assessed by question-
naire and by medical records, and compared differences in prevalence using McNemar
statistics.
Second, we assessed agreement between questionnaires and medical records. We calculated
percent agreement (proportion of those with agreement in questionnaire and medical records)
and Cohen´s kappa statistics [16]. Kappa value >0.75 was considered as an excellent agree-
ment beyond chance, a kappa of 0.40–0.75 as intermediate to good agreement, and a kappa
below 0.40 as poor agreement [17]. To investigate whether agreement varies between sub-
groups, we stratified measures by questionnaire respondent, sociodemographic characteristics,
and time between follow-up and questionnaire. We used chi-square statistics to compare per-
cent agreement between subgroups.
Third, we evaluated validity of questionnaire-reported hearing. Information from medical
records was treated as reference. We calculated sensitivity, specificity, positive predictive value
(PPV), and negative predictive value (NPV). Sensitivity was the proportion of those with hear-
ing loss as defined by the medical records whom the questionnaire classified correctly. Speci-
ficity was the proportion of those with normal hearing whom the questionnaire classified
correctly. PPV was the proportion of those with questionnaire-reported hearing loss, in whom
hearing loss was confirmed by medical records. NPV was the proportion of those with ques-
tionnaire-reported normal hearing, in whom normal hearing was confirmed by medical rec-
ords. To investigate how validity might vary between subgroups, we also stratified measures of
validity.
Last, we assessed whether the questionnaire assessed hearing loss equally well for different
severity levels of hearing loss. We calculated sensitivity and PPV stratified by severity level and
laterality of hearing loss.
We also performed a sensitivity analysis to compare results from all respondents (n = 361)
to results from respondents for whom we found original hearing tests in medical records
(n = 270) by excluding those for whom we did not find hearing tests in medical records and
only used information from oncological follow-up (n = 91).
We used Stata (Version 13, Stata Corporation, Austin, Texas) for all analyses.
Results
Characteristics of study population
Of the 2175 survivors diagnosed after 1989 in a SPOG clinic, 611 (28%) had received ototoxic
cancer treatment. Among these, 222 did not reply to the questionnaire and 28 had missing
medical records (Fig 1). This resulted in 361 persons (59% of those who received otoxic treat-
ment) who were available for analyses. There were no significant differences between those
Validation of questionnaire-reported hearing with medical records
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who were included in the analysis and those who were not, except for a slight difference in
type of cancer diagnosis (p = 0.028, Table 1). Fifty-four percent of the study participants were
male. Median (interquartile range) age at survey was 18 (6–30) years and median age at diag-
nosis 5 (0–15) years. The most common diagnosis was CNS tumor (27%), followed by leuke-
mia (24%), and bone tumor (10%). Of those who had received chemotherapy, 22% had been
treated with carboplatin, 26% with cisplatin, 20% with both carboplatin and cisplatin, and 32%
with neither agent. Of those who had received radiation, 91% had received cranial radiation.
Of those who had undergone surgery, 38% had brain surgery. Eleven percent had BMT.
Median time between last follow-up and questionnaire was 5 (2–7) years.
Assessment of hearing by questionnaire and medical records
For 118 of the 361 respondents (33%) parents answered the questionnaire because they were
younger than 16 (parent-reported). The 243 (67%) older respondents answered the question-
naire themselves (survivor-reported, Fig 1). Hearing tests were found in medical records of
270 respondents (75%) (Fig 2). Among those, 215 (60%) also had an audiological report. The
report confirmed hearing loss at or above grade 1 in 207 of these 215 cases (96%). The remain-
ing 8 (4%) had high-frequency hearing loss according to hearing tests (grade 1, >20 dB HL
above 4kHz) that was not defined as hearing loss by audiologist/clinician. For 91 of the respon-
dents (25%), we used oncological follow-up reports to assess hearing because no hearing test
was available in medical records.
Prevalence and severity of hearing loss
Prevalence of hearing loss was nearly the same in the questionnaire reports and medical rec-
ords (27% vs. 31%, p = 0.040; Table 2). Most respondents reported mild (16%) or moderate
hearing loss (9%), and only few had severe hearing loss (2%). In the medical records, severity
grades of hearing loss according to SIOP Boston Ototoxicity Scale were 6% with grade 1, 9%
with grade 2, 7% with grade 3, and 8% with grade 4. Seven percent of respondents reported
unilateral hearing loss, 17% bilateral hearing loss, and 3% did not specify. Laterality of hearing
loss assessed by medical records resulted in 6% with unilateral and 25% with bilateral hearing
loss.
Agreement and validity
Overall, agreement between questionnaires and medical records was good (85%, 95% CI 81–
89%; kappa 0.64, 95% CI 0.55–0.72; Table 3). Results differed neither by type of respondent
(survivor- or parent-reported) nor by sociodemographic characteristics (gender, education,
migration) (Table 3). Agreement was more better when the time interval between follow-up
and questionnaire was shorter: up to 87% for intervals up to 9 years, and 71% for intervals of
10–17 years (p = 0.051). Questionnaire-reported hearing loss had a sensitivity of 69% (95% CI
59–77%), and a specificity of 92% (95% CI 88–95%) compared to medical records (Table 3).
Among those who reported hearing loss, medical records confirmed hearing loss in 80% (95%
CI 71–88%). Among those who reported normal hearing, medical records confirmed normal
hearing in 87% (95% CI 82–91%).
Severity and laterality of hearing loss. Sensitivity of questionnaire reports varied with
different levels of hearing loss (Table 4,S2 and S3 Figs). Sensitivity was highest in respondents
with moderate (grade 2) and severe (grade 3–4) hearing loss according to the SIOP Boston
Ototoxicity Scale: 71%, 95% CI 53–85%; and 86%, 95% CI 73–94% respectively. Sensitivity was
lowest in those with mild (grade 1) hearing loss: 26%, 95% CI 10–48%. After stratification for
laterality of hearing loss, we found that at all levels of severity those with unilateral hearing loss
Validation of questionnaire-reported hearing with medical records
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Table 1. Characteristics of study population.
Eligible population included in the
study
N = 361
Eligible population not included in
the study N = 250
Sociodemographic characteristics n (%) n (%) P
a
Gender 0.650
Female 167 (46) 111 (44)
Male 194 (54) 139 (56)
Age at survey 0.296
5–15 years 118 (33) 67 (27)
16–20 years 109 (30) 83 (33)
21–40 years 134 (37) 100 (40)
Migration background
b
No 288 (80) n.a.
d
Yes 73 (20)
Education
c
Primary education 37 (12) n.a.
d
Secondary education 216 (60)
Tertiary education 100 (28)
Clinical characteristics
Diagnosis (ICCC3) 0.028
I Leukemia 88 (24) 65 (26)
II Lymphoma 13 (4) 15 (6)
III CNS tumor 99 (27) 59 (24)
IV Neuroblastoma 31 (9) 21 (8)
V Retinoblastoma 28 (8) 10 (4)
VI Renal tumor 3 (1) 12 (5)
VII Hepatic tumor 12 (3) 5 (2)
VIII Bone tumor 35 (10) 17 (7)
IX Soft tissue sarcoma 29 (8) 22 (9)
X Germ cell tumor 22 (6) 21 (8)
XI &XII Other rare tumor 1 (0) 3 (1)
Treatment
Chemotherapy 341 (94) 230 (92) 0.227
No platinum chemotherapy 108 (32) 92 (40)
Carboplatin 76 (22) 48 (21)
Cisplatin 89 (26) 45 (20)
Cisplatin and Carboplatin 68 (20) 45 (20)
Radiotherapy 228 (63) 169 (68) 0.314
No cranial radiation 21 (9) 21 (12)
Cranial radiation <30 Gy 87 (38) 70 (41)
Cranial radiation 30–49 Gy 25 (11) 15 (9)
Cranial radiation 50 Gy 87 (38) 57 (34)
Unknown 8 (4) 6 (4)
Surgery 252 (70) 169 (70) 0.905
Brain surgery 95 (38) 60 (36) .
Bone marrow transplantation 40 (11) 28 (11) 0.284
Time between last follow-up and questionnaire
0–2 years 135 (37) n.a.
d
3–4 years 57 (16)
(Continued)
Validation of questionnaire-reported hearing with medical records
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Table 1. (Continued)
Eligible population included in the
study
N = 361
Eligible population not included in
the study N = 250
Sociodemographic characteristics n (%) n (%) P
a
5–9 years 125 (35)
10–17 years 41 (11)
Unknown 3 (1)
Abbreviations: N, number; n.a., not applicable; ICCC3, International Classification of Childhood Cancer—Third edition; CNS, central nervous system; Gy,
Gray.
a
p-values calculated from chi-square statistics comparing included to excluded survivors.
b
Survivors were coded as having a migration background if they were not born in Switzerland, had no Swiss citizenship at birth or had at least one parent
without Swiss citizenship.
c
Survivor´s and parent´s education was classified into three categories: primary education (compulsory schooling only [9 years]), secondary education
(vocational training [10–13 years], higher vocational training or college), or tertiary education (university or technical college education). If the father and
mother had achieved different levels of education, we selected the parent with the highest education. Education includes parental education when parents
answered the questionnaire, and survivors´ education when survivors answered the questionnaire.
d
Migration background, education, and time between follow-up and questionnaire are not available for nonresponding parents and survivors.
https://doi.org/10.1371/journal.pone.0174479.t001
Table 2. Prevalence, severity, and laterality of hearing loss assessed by questionnaire or medical
records.
N (%)
Hearing measured by questionnaire
Normal hearing 265 (73)
Hearing loss 96 (27)
Severity
Mild hearing loss 59 (16)
Moderate hearing loss 31 (9)
Severe hearing loss (deafness) 6 (2)
Laterality
Unilateral 24 (7)
Bilateral 63 (17)
Unknown 9 (3)
Hearing measured by medical records
Normal hearing
a
249 (69)
Hearing loss 112 (31)
Severity
b
Grade 1 (>20 dB HL above 4kHz) 23 (6)
Grade 2 (>20 dB HL at 4kHz and above) 34 (9)
Grade 3 (>20 dB HL at 2kHz and above) 25 (7)
Grade 4 (>40 dB HL at 2kHz and above) 30 (8)
Laterality
Unilateral 21 (6)
Bilateral 91 (25)
Abbreviations: N, number; SIOP, International Society of Pediatric Oncology.
a
Includes SIOP Boston Ototoxicity Scale Grade 0.
b
According to SIOP Boston Ototoxicity Scale
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Validation of questionnaire-reported hearing with medical records
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more often under-report their impairment than those with bilateral hearing loss: grade 1, 10%
vs. 39%; grade 2, 33% vs. 74%; grades 3–4, 50% vs. 92%.
Sensitivity analysis
We found no important differences in the sensitivity analyses that compared results of all data
to results of data excluding those for whom we did not find hearing tests in medical records
Table 3. Measures of agreement and validity for questionnaire-reported hearing.
Hearing loss by medical record/
questionnaire
Agreement Validity
Y/Y
a
N/Y
b
Y/N
c
N/N
d
Percent agreement,
%
Kappa p
e
Sensitivity
f
, % Specificity
f
, % PPV
f
, % NPV
f
, %
Overall 77 19 35 230 85 [81–89] 0.64 [0.55–
0.72]
69 [59–77] 92 [88–95] 80 [71–88] 87 [82–
91]
By type of questionnaire report
Parent-reported 25 5 9 79 88 [81–93] 0.70 [0.56–
0.85]
0.251 74 [57–87] 94 [87–98] 83 [65–94] 90 [82–
95]
Survivor-reported 52 14 26 151 84 [78–88] 0.60 [0.50–
0.72]
67 [55–77] 92 [86–95] 79 [67–88] 85 [79–
90]
By gender
Female 35 9 11 112 88 [82–93] 0.70 [0.57–
0.82]
0.140 76 [61–87] 93 [86–97] 80 [65–90] 91 [85–
96]
Male 42 10 24 118 82 [76–88] 0.59 [0.47–
0.71]
64 [51–75] 92 [86–96] 81 [68–90] 83 [76–
89]
By education
Primary education 11 3 6 23 79 [64–90] 0.55 [0.29–
0.81]
0.472 65 [38–85] 89 [70–98] 79 [49–95] 79 [60–
92]
Secondary
education
45 11 21 139 85 [80–90] 0.64 [0.52–
0.75]
68 [56–79] 93 [87–96] 80 [68–90] 87 [81–
92]
Tertiary education 20 5 8 67 87 [79–93] 0.67 [0.50–
0.83]
71 [51–87] 93 [85–98] 80 [59–93] 89 [80–
95]
By migration background
No 64 17 28 179 84 [80–88] 0.63 [0.53–
0.73]
0.481 70 [59–79] 91 [87–95] 79 [69–87] 87 [81–
91]
Yes 13 2 7 51 88 [78–94] 0.66 [0.46–
0.86]
65 [41–85] 96 [87–100] 87 [60–98] 88 [77–
95]
By time between follow-up and questionnaire
0–2 years 33 9 8 85 87 [81–92] 0.70 [0.57–
0.84]
0.051 81 [65–91] 90 [83–96] 79 [63–90] 91 [84–
96]
3–4 years 12 3 5 37 86 [74–94] 0.65 [0.43–
0.87]
71 [44–90] 93 [80–98] 80 [52–96] 88 [74–
96]
5–9 years 23 5 11 86 87 [80–93] 0.66 [0.50–
0.81]
68 [50–83] 95 [88–98] 82 [63–94] 89 [81–
94]
10–17 years 9 1 11 20 71 [54–84] 0.41 [0.16–
0.65]
45 [23–69] 95 [76–100] 90 [56–
100]
65 [45–
81]
Abbreviations: n.a., not applicable.
a
Y/Y, hearing loss in medical record and hearing loss in questionnaire.
b
N/Y, normal hearing in medical record and hearing loss in questionnaire.
c
Y/N, hearing loss in medical record and normal hearing in questionnaire.
d
N/N, normal hearing in medical record and normal hearing in questionnaire.
e
p-values calculated from chi-square statistic comparing percent agreement by strata.
f
Data from medical records were considered as reference.
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and only used information from oncological follow-up (n = 91) (S1 and S2 Tables). In this
analysis, agreement between questionnaires and medical records was 83% (95% CI 78–87%)
and kappa 0.63 (95% CI 0.54–0.73). Questionnaires had a sensitivity of 69% (95% CI 59–77%),
a specificity of 92% (95% CI 87–96%), a PPV of 86% (95% CI 78–93%) and NPV of 81% (95%
CI 74–86%) compared to medical records (S1 Table).”
Discussion
Principal findings
In this study, the first to comprehensively investigate agreement and validity of questionnaire-
reported hearing in childhood cancer survivors, we found that questionnaires assessed hearing
in childhood cancer survivors with acceptable validity. Medical records agreed with 85% of
respondents’ questionnaires, which correctly assessed normal hearing in 92% of those with
normal hearing and hearing loss in 69% of those with hearing loss. Results did not differ by
questionnaire respondent (survivor- or parent-report) or sociodemographic characteristics.
Questionnaires did underestimate mild or unilateral hearing loss.
Limitations and strengths
We assumed that medical records were complete and correct. However, we would have missed
tests of survivors who had their hearing tested after oncological long-term follow-up at a pri-
vate practice. We also used information on hearing from the oncological follow-up reports,
when we found no hearing assessment in the medical records and assumed normal hearing
when we found no information on hearing in those reports. This could have contribute to
discrepancies in hearing loss between questionnaires and medical records, and may have
increased the number of cases with false positive results. However, the sensitivity analysis sug-
gested that this has not led to a relevant distortion of the results. Respondents with a long
period between last follow-up and questionnaire might recall the doctor´s diagnosis less well,
but validity decreased only in those with more than 10 years between follow-up and question-
naire. Only a minority of the respondents (11%) had such a long period. Finally, our results
cannot be extrapolated to children who have not received ototoxic cancer treatment.
Our study is strengthened by a study population that is large compared to those of previous
validation studies of childhood cancer survivors [1820]. Because respondents received in
Table 4. Measures of validity for questionnaire-reported hearing for different degrees of hearing loss.
Hearing loss according to medical records Sensitivity
a
, % PPV
a
, %
Mild including grade 1
b
26 [10–48] 24 [9–45]
Unilateral 10 [1–45] 5 [1–25]
Bilateral 39 [14–68] 21 [7–42]
Moderate including grade 2
b
71 [53–85] 56 [40–71]
Unilateral 33 [1–91] 5 [1–25]
Bilateral 74 [55–88] 55 [39–70]
Severe including grade 3–4
b
86 [73–94] 71 [59–82]
Unilateral 50 [16–84] 17 [5–39]
Bilateral 92 [80–98] 69 [56–80]
Abbreviation: PPV, Positive predictive value.
a
Data from medical records were considered as reference.
b
Severity grades according to SIOP Boston Ototoxicity Scale.
https://doi.org/10.1371/journal.pone.0174479.t004
Validation of questionnaire-reported hearing with medical records
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many cases comprehensive audiological follow-up, we could ask them about the results of clin-
ical assessment of their hearing status (S1 Fig) rather than only being able to rely upon subjec-
tive perception of their own hearing—as is often the case in validation studies in the general
population [710].
Comparison with other studies
This is the first study to compare the questionnaire-reported prevalence of hearing loss with medi-
cal records in one group of childhood cancer survivors. The prevalence of hearing loss was slightly
lower assessed by questionnaire than by medical records (27% vs. 31%). The prevalence of hearing
loss is hard to compare in different study populations because of differences in diagnostic groups,
cancer treatment, and type of auditory grading. In previous studies the prevalence of question-
naire-reported hearing loss in groups at risk of ototoxicity ranged from 16 to 31% [2,4,21], and
was even higher when prevalence was assessed by retrospective data collection in medical records
(12–90%) [3,2224]. However, previous studies that are similar to ours may have underestimated
the prevalence of hearing loss just as our study did for mild and unilateral hearing loss.
No other study has investigated validity and agreement of questionnaire-reported hearing
with medical records in childhood cancer survivors. Most publications on validity of question-
naire-reported hearing studied populations of older age using questions on subjective percep-
tion of hearing such as “Do you feel you have hearing loss?” [8,9,2529]. In an Australian
study that pooled seven data sets with 23,001 respondents asked for self-assessment of hearing
(age 45–103 years), sensitivity ranged from 62–78% and specificity from 41–85% [29]. Our
question “Have you ever been told by a doctor that you have, or have had hearing loss?” per-
formed similarly in those with hearing loss (sensitivity 69%), but better in those with normal
hearing (specificity 92%).
We found very similar results for agreement and validity when either survivors or parents
reported on hearing. Both survivors and parents recall hearing equally well. There is no other
study investigating the difference between survivor- and parent-reported hearing.
Validation studies on hearing in mainly elderly people reported that younger, male, and
more highly educated respondents report hearing more accurately [8,9,26,29]. Our findings
and those of other validation studies of childhood cancer survivors that also asked survivors
for health problems reported by a clinician suggested no effect of sociodemographic factors
[18,20]. The different types of questions might be the cause for the discrepancy between find-
ings from the general population and childhood cancer survivors as self-perception might be
more affected by age, gender, or education level.
Not surprisingly, we found that survivors with mild and unilateral hearing loss are more
likely to under-report hearing loss than survivors with severe hearing loss. This may have two
causes. First, survivors may not have been aware of their mild hearing loss because their audi-
ologist/clinician did not tell them about it. In 4% of cases, which we validated with the audiolo-
gist´s results of hearing tests, audiologists did not report on hearing loss though the audiogram
showed limited results in the high frequencies. Second, unlike severe hearing loss unilateral or
mild hearing loss may affect daily life little or not at all and fewer survivors may recall such a
diagnosis, especially when it was some years ago. Several studies have also reported that the
accuracy of questionnaire reports of mild hearing loss is low among the elderly [7,9,27]. They
have shown that single questions such as “Do you have a problem with your hearing?” are less
able to detect mild hearing loss than more comprehensive instruments like the Hearing Handi-
cap Inventory for the Elderly—Screening Version (HHIE-S) [30]. A recent systematic review
that described patient-reported measures for hearing loss identified only two instruments, the
Health Utilities Index (HUI) [31] and the Hearing Measurement Scale (HMS) [32] that are
Validation of questionnaire-reported hearing with medical records
PLOS ONE | https://doi.org/10.1371/journal.pone.0174479 March 23, 2017 11 / 15
used with childhood cancer patients [33]. The HUI includes six questions, the HMS 44 ques-
tions, and both return hearing-specific summary scores. Researchers reported that none of the
questionnaires appeared ideal based upon the number of questions, and wording for children
or adolescents, and psychometric properties were never tested in this population [33].
Interpretation of results
Studies of hearing loss after treatment of childhood cancer ideally should collect data from
medical records or, to obtain the most valid results, prospectively perform audiometry assess-
ments. However, these approaches are often not feasible because they are expensive, and access
to full medical records or additional audiometry assessments may be complicated. Many stud-
ies therefore assessed hearing loss by questionnaires, which inquire about doctor-diagnosed
hearing problems. Reports of survivors and their parents appear equally valid.
Questionnaires assess moderate or severe hearing loss well, but underestimate mild and
unilateral hearing loss. Our questionnaire missed 90% of cases with mild unilateral and 67% of
cases with moderate unilateral hearing loss. Particularly in the absence of well established
questionnaires we suggest that researchers validate questionnaire-reported data in at least a
subsample to avoid misclassification and reduce bias. Since level of hearing loss differentially
affects the accuracy of hearing reports, the results observed for effects of ototoxic risk factors
on hearing or the impact of hearing loss on an outcome could be biased. Instruments currently
used in clinical assessment of hearing loss in childhood cancer patients such as the HUI and
the HMS have questions that focus on difficulties following group conversations with or with-
out a noisy environment. In a future project, we will investigate asking about this type of hear-
ing difficulty to assess mild or unilateral hearing loss independent of clinicians who may not
measure or report mild hearing loss.
Conclusion
Questionnaires are a useful tool to assess hearing in childhood cancer survivors. However,
they underestimate mild and unilateral hearing loss, so researchers should investigate whether
inquiring about difficulty following conversation in a noisy environment can improve the
assessment of this type of hearing loss in childhood cancer survivors.
Supporting information
S1 Fig. Questions on hearing from the questionnaire for adults and adolescents of the
Swiss Childhood Cancer Survivor Study, translated to English.
(TIF)
S2 Fig. Sensitivity of questionnaire-reports by laterality and severity of hearing loss. Abbre-
viation: HL, hearing loss.
according to medical records.
(TIF)
S3 Fig. Questionnaire-reported severity of hearing loss stratified by severity of hearing loss
assessed by medical records. Abbreviation: HL, hearing loss.
according to SIOP Boston Ototoxicity Scale.
(TIF)
S1 Table. Measures of agreement and validity for questionnaire-reported hearing–Sensi-
tivity analysis including survivors with hearing test (n = 270).
(PDF)
Validation of questionnaire-reported hearing with medical records
PLOS ONE | https://doi.org/10.1371/journal.pone.0174479 March 23, 2017 12 / 15
S2 Table. Measures of validity for questionnaire-reported hearing for different degrees of
hearing loss–Sensitivity analysis including survivors with hearing test (n = 270).
(PDF)
Acknowledgments
The authors express their gratitude to all childhood cancer survivors and their siblings in Swit-
zerland for filling in the questionnaire and supporting this study. We thank the study team of
the Swiss Childhood Cancer Survivor Study (Rahel Kasteler, Erika Brantschen Berclaz, Laura
Wengenroth, Corina Rueegg, Cornelia Rebholz), the data managers of the Swiss Paediatric
Oncology Group (Claudia Anderegg, Pamela Balestra, Nadine Beusch, Rosa-Emma Garcia,
Franziska Hochreutener, Friedgard Julmy, Nadia Lanz, Rodolfo Lo Piccolo, Heike Markiewicz,
Annette Reinberg, Renate Siegenthaler, Verena Stahel), and the team of the Swiss Childhood
Cancer Registry (Verena Pfeiffer, Katharina Flandera, Shelagh Redmond, Meltem Altun, Par-
vinder Singh, Vera Mitter, Elisabeth Kiraly, Marlen Spring, Christina Krenger, Priska Wo¨lfli).
Further, we thank the teams of the ENT-departments (Prof. Dr. med. Pascal Senn, Marie-Mad-
eleine Knuchel, PD Dr. med. Hanna Brockmeier, Dr. med. Jochen Rosenfeld, PD Dr. med.
Christof Stieger, Dr. med. Claudia Candreia, Dr. med. Raphael Maire, Cecile Moreno, Dr. med
Dorothe Veraguth, Julia Bernath, Sandra Schenk) and Gierin Thomi, Marie Bulliard, and
Annette Schneeberger for their help in collecting audiological data from medical records. We
thank Christopher Ritter for his editorial suggestions.
The members of the Swiss Paediatric Oncology Group Scientific Committee are: Prof.
Dr. med. R. Ammann (Bern); Dr. med. R. Angst (Aarau); Prof. Dr. med. M. Ansari (Geneva);
Prof. Dr. med. M. Beck Popovic Lausanne); Dr. med. E. Bergstraesser (Zurich); Dr. med. P.
Brazzola (Bellinzona); Dr. med. J. Greiner (St. Gallen); Prof. Dr. med. T. Kuehni (Basel); Prof.
Dr. med. M. Grotzer (Zurich); Prof. Dr. med. K. Leibundgut (Bern); Dr. med. H. Hengartner
(St. Gallen); Prof. Dr. med. F. Niggli (Zurich); PD Dr. med. J. Rischewski (Lucerne); and Prof.
Dr. med. N. von der Weid (Basel); contact: R. Ammann; email: roland.ammann@insel.ch.
Author Contributions
Conceptualization: AW CK.
Data curation: AW.
Formal analysis: AW.
Funding acquisition: CK.
Investigation: AW.
Methodology: AW CK.
Project administration: AW RK.
Visualization: AW.
Writing – original draft: AW.
Writing – review & editing: GS RK KS MG MK CK.
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Validation of questionnaire-reported hearing with medical records
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Lo screening dei disturbi dell’udito nel bambino ha lo scopo di isolare in una determinata popolazione, con esami semplici, rapidi e poco costosi, un sottogruppo di bambini ai quali proporre esami diagnostici della sordità, più specifici, più precisi, ma più costosi e più dispendiosi in termini di tempo. Il test di screening ideale è quello che non manca un bambino che abbia un’ipoacusia, pur avendo il numero minimo di bambini che, alla fine, risultano avere un udito normale. Nel periodo neonatale, lo screening fa ricorso a metodi obiettivi: otoemissioni provocate, prodotti di distorsione o potenziali evocati automatizzati. Nella prima infanzia, utilizza giocattoli sonori. In Francia, l’udito è controllato dalla medicina scolastica prima del passaggio in corso di preparazione e prima dell’ingresso nella scuola secondaria con un test audiometrico tonale con cuffia semplificato. Al di fuori di questi periodi chiave, qualsiasi sospetto di ipoacusia da parte dei genitori o delle persone che si occupano del bambino deve portare a esami di screening dei disturbi uditivi. Se gli esami di screening sono positivi, il bambino deve essere sottoposto a un esame oto-rino-laringoiatrico (ORL) e a esami audiometrici adeguati alla sua età per negare o confermare l’ipoacusia e, in quest’ultimo caso, per precisarne il meccanismo e il grado.
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La detección precoz de los trastornos de la audición en los niños tiene como objetivo identificar, en una población determinada y mediante exploraciones sencillas, rápidas y económicas, un subgrupo de niños candidatos a otras exploraciones diagnósticas de sordera más específicas y más precisas, aunque más costosas y que consumen más tiempo. La prueba de detección precoz ideal es aquélla que no pasa por alto un niño con sordera y que selecciona erróneamente un número mínimo de niños con una audición normal. En el período neonatal, la detección precoz utiliza métodos objetivos: otoemisiones inducidas, productos de distorsión o potenciales provocados automatizados. En la primera infancia, recurre a juguetes sonoros. En Francia, la audición se evalúa en los exámenes de salud escolar antes de pasar a la enseñanza primaria (sobre los 5-6 años) y antes de la entrada en la enseñanza secundaria (sobre los 12 años), mediante una prueba audiométrica tonal simplificada con auriculares. Fuera de estos períodos clave, toda sospecha de hipoacusia por parte de los padres o los cuidadores del niño debe llevar a practicar exploraciones de detección precoz de las alteraciones auditivas. Si las pruebas de detección precoz son positivas, el niño debe someterse a una exploración otorrinolaringológica (ORL) y a pruebas de audiometría adecuadas a su edad, con el fin de descartar o confirmar la hipoacusia y, en este último caso, precisar su mecanismo y grado.
Article
Background: Auditory complications are an adverse event of childhood cancer treatment, especially common in children treated with platinum chemotherapy or cranial radiation. Variation between diagnostic childhood cancer groups has rarely been studied, and we do not know if the burden of auditory complications has changed over the last decades. Procedure: Within the Swiss Childhood Cancer Survivor Study, we sent a questionnaire to all survivors who were diagnosed at age 16 years or less between 1976 and 2005. We compared prevalence of self-reported hearing loss and tinnitus between all diagnostic childhood cancer groups and siblings, used multivariable logistic regression to analyze the effect of treatment-related factors on hearing loss, and compared the cumulative incidence of hearing loss between different periods of cancer diagnosis. Results: Prevalence of self-reported hearing loss was higher in survivors (10%) than in siblings (3%, P < 0.001), and highest in survivors of central nervous system tumors (25%). Significant risk factors were treatment with platinum compounds (carboplatin: odds ratio [OR] 2.4; cisplatin: OR 9.4), cranial radiation (>29 Gy: OR >1.7), or brain surgery (OR 2.2). Children diagnosed in 1986-1995, when platinum compounds came into widespread use, had a significantly higher cumulative incidence of hearing loss than those diagnosed in 1976-1985. In the most recent period, 1996-2005, the risk decreased again, both for patients treated with platinum compounds and with cranial radiation. Conclusions: Our data show that the burden of hearing loss has stabilized in recently treated survivors, suggesting that survivors have benefited from new treatment regimens that use less ototoxic radiation and more carefully dosed platinum compounds.
Article
Purpose: We identified studies that described use of any patient-reported outcome scale for hearing loss or tinnitus among children and adolescents and young adults (AYAs) with cancer or hematopoietic stem cell transplantation (HSCT) recipients. Method: In this systematic review, we performed electronic searches of OvidSP MEDLINE, EMBASE, and PsycINFO to August 2015. We included studies if they used any patient-reported scale of hearing loss or tinnitus among children and AYAs with cancer or HSCT recipients. Only English language publications were included. Two reviewers identified studies and abstracted data. Results: There were 953 studies screened; 6 met eligibility criteria. All studies administered hearing patient-reported outcomes only once, after therapy completion. None of the studies described the psychometric properties of the hearing-specific component. Three instruments (among 6 studies) were used: Health Utilities Index (Barr et al., 2000; Fu et al., 2006; Kennedy et al., 2014), Hearing Measurement Scales (Einar-Jon et al., 2011; Einarsson et al., 2011), and the Tinnitus Questionnaire for Auditory Brainstem Implant (Soussi & Otto, 1994). All had limitations, precluding routine use for hearing assessment in this population. Conclusions: We identified few studies that included hearing patient-reported measures for children and AYA cancer and HSCT patients. None are ideal to take forward into future studies. Future work should focus on the creation of a new psychometrically sound instrument for hearing outcomes in this population.
Article
Ototoxicity is a well-established toxicity associated with a subgroup of antineoplastic therapies that includes platinum chemotherapy, radiation or surgery involving the ear and auditory nerve, and supportive care agents such as aminoglycoside antibiotics and loop diuretics. The reported prevalence of ototoxicity in patients who have received potentially ototoxic therapy ranges from 4% to 90% depending on factors such as age of the patient population, agent(s) used, cumulative dose, and administration techniques. The impact of ototoxicity on subsequent health-related and psychosocial outcomes in these patients can be substantial, and the burden of morbidity related to ototoxic agents is particularly high in very young children. Considerable interindividual variability in the prevalence and severity of ototoxicity has been observed among patients receiving similar treatment, suggesting genetic susceptibility as a risk factor. The development and testing of otoprotective agents is ongoing; however, to the author's knowledge, no US Food and Drug Administration-approved otoprotectants are currently available. Prospective monitoring for ototoxicity allows for comparison of auditory outcomes across clinical trials, as well as for early detection, potential alterations in therapy, and auditory intervention and rehabilitation to ameliorate the adverse consequences of hearing loss. Cancer 2016. © 2016 American Cancer Society.
Article
Objectives: The purpose of this study was to evaluate the severity of hearing loss after cisplatin and/or carboplatin treatment in young children and to analyze its evolution and its relation to different therapy schedules. Methods: One hundred twenty patients treated in the Pediatrics Department at the Institut Gustave-Roussy from 1987 to 1997 for neuroblastoma, osteosarcoma, hepatoblastoma, or germ cell tumors were analyzed. Median age at diagnosis was 2.6 (range 0-17) years. Median follow-up was 7 (1-13) years. Chemotherapy regimens contained cisplatin and/or carboplatin. Three patients also received high-dose carboplatin. Cisplatin was administered at a dose of 200 mg/m/course in 72% of cases. The median cumulative dose was 400 mg/m for cisplatin and 1,600 mg/m for carboplatin. Hearing loss of grade 2 or above, according to Brock's grading scale, was revealed with pure tone audiometry and behavioral techniques. Results: Carboplatin alone was not ototoxic. Deterioration of hearing of grade 2 or above was observed in 37% of patients treated with cisplatin and 43% of patients treated with cisplatin plus carboplatin (P = NS). Fifteen percent of patients experienced grade 3 or 4 ototoxicity. Ototoxicity was most often observed after a total cisplatin dose of at least 400 mg/m. No improvement was observed with time; on the contrary, worsening or progression of hearing loss at lower frequencies was detected during follow-up. Only 5% of audiograms showed toxicity of at least grade 2 before the end of therapy; in contrast, this level was observed in 11% of early post-therapy evaluations and in 44% after more than 2 years of follow-up. Conclusions: Children treated with cisplatin at cumulative doses approaching 400 mg/m require long-term surveillance to avoid overlooking hearing deficits. Carboplatin, at a standard dose, does not appear to be a significant risk factor for ototoxicity even in patients who have already been treated with cisplatin.
Article
This chapter presents analytic methods for matched studies with multiple risk factors of interest. We consider matched sample designs of two types, prospective (cohort or randomized) and retrospective (case-control) studies. We discuss direct and indirect parametric modeling of matched sample data and then focus on conditional logistic regression in matched case-control studies. Next, we describe the general case for matched samples including polytomous outcomes. An illustration of matched sample case-control analysis is presented. A problem solving section appears at the end of the chapter.
Article
Objectives: Self-reported hearing impairment is often used to gauge objective hearing loss in both clinical settings and research studies. The aim of this study was to examine whether demographic factors affect the accuracy of subjective, self-reported hearing in older adults. Design: We examined 3557 participants aged 50 and older in the National Health and Nutrition Examination Survey cycles 1999-2006 and 2009-2010. We examined the relationship between objective and subjective hearing impairment using percent correct classification and misclassification bias in analyses stratified by gender, age group, race/ethnicity, and education. Results: We found that younger participants tended to overestimate and older participants underestimate their hearing impairment. Older women, blacks, and Hispanics were less accurate in self-reporting than their respective younger age groups. Conclusions: The association between subjective and objective hearing differs across gender, age, race/ethnicity, and education, and this observation should be considered by clinicians and researchers employing self-reported hearing.
Article
Background The antineoplastic agents cisplatin and carboplatin are widely-used and highly-effective against a variety of pediatric cancers. Unfortunately, ototoxicity is a frequently encountered side effect of platinum-based chemotherapy. There is currently no treatment or prevention for platinum-induced ototoxicity and development of hearing loss may lead to devastating consequences on the quality of life of pediatric cancer survivors. The objective of this study is to determine the incidence of platinum-induced ototoxicity in a large series of pediatric patients and to evaluate the incidence of progression of ototoxicity after completion of treatment. ProceduresA retrospective chart review of pediatric patients treated with cisplatin or carboplatin between 2000 and 2012 was conducted. The incidence of ototoxicity was determined based on the American-Speech-Language-Hearing Association (ASHA) criteria and severity was based on the Chang classification. ResultsFour hundred and sixty-six patients received platinum-based chemotherapy. Patients were excluded due to congenital hearing loss (n=1) and insufficient data for calculating the platinum dose (n=24) or for assessing ototoxicity (n=135). Three hundred and six patients were included in the analysis. Post-chemotherapy ototoxicity was detected in 148 (48%) patients, and clinically-significant ototoxicity was present in 91 (30%). In addition, based on the ASHA criteria, 48% of patients (97/204) with long-term follow-up had further deterioration of their hearing after completion of treatment. Conclusions Ototoxicity following chemotherapy with cisplatin or carboplatin is common and can frequently progress after the completion of treatment. Long-term follow-up is strongly recommended. Pediatr Blood Cancer 2014;61:2012-2017. (c) 2014 Wiley Periodicals, Inc.