10.14639/0392-100X-N2263
Objective. translate and validate the Penn Acoustic Neuroma Quality of Life Scale into Italian language (PANQOL-It).Methods. the instrument was translated and psychometric properties were subsequently assessed by administering the PANQOL-It to 124 outpatients together with the Depression Anxiety Stress Scale (DASS21) and the Understanding and Communicating domain of the World Health Organization Disability Assessment Schedule II (WHODAS II-D1). The internal consistency, test-retest reliability, construct and criterion-related validity were assessed. Results. Cronbach’s alpha coefficient was 0.92 for the total score and ranged from 0.44 to 0.90 in the seven domains. Significant test-retest reliability was observed (intraclasscorrelation = 0.75; p &lt; 0.01). Moderate correlation was reported between facial dysfunction domain and objective facial involvement (p &lt; 0.01). Moderate to strong correlations were observed between anxiety, general health domains and all subscales of the DASS21, and between WHODAS II-D1 and general health and energy domains (p &lt; 0.01). These latter results indicated good construct and criterion-related validity respectively. Conclusions. PANQOL-It presented more than acceptable psychometric properties and its adoption is justified for both clinical and research purposes.
text/xml
1827-675X
2263
en
Riccardo Nocini
http://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
health
Italian validation of the Penn Acoustic Neuroma Quality of Life Scale (PANQOL-It)
Text.Serial.Journal
https://www.actaitalica.it/
43
Audiology
2023-04-26
2022-08-07
2023-04-27
2023-04-27
130-139
1
Riccardo Nocini
2023/04/26
10.14639/0392-100X-N2263
130
Section of Ear, Nose and Throat (ENT), Department of Surgical Sciences, Dentistry, Gynaecology and Paediatrics, University of Verona, Borgo Roma Hospital, Verona, Italy
1827-675X
health
en
139
Italian validation of the Penn Acoustic Neuroma Quality of Life Scale (PANQOL-It)
https://www.actaitalica.it/article/download/2263/962
43
ACTA OTORHINOLARYNGOLOGICA ITALICA 2023;43:130-139; doi: 10.14639/0392-100X-N2263




Audiology


Italian validation of the Penn Acoustic Neuroma
Quality of Life Scale (PANQOL-It)
Validazione italiana del Penn Acoustic Neuroma Quality of Life Scale (PANQOL-It)
Enrico Apa1, Francesco Maccarrone2, Chiara Gherpelli1, Luca Sacchetto3, Daniele Monzani3, Silvia Palma4,
Riccardo Nocini3
1
 Department of Medical and Surgical Sciences for Children and Adults, Otorhinolaryngology Unit, University of Modena and Reggio
Emilia, Azienda Ospedaliero-Universitaria of Modena, Modena, Italy; 2 Otorhinolaryngology Department, Ramazzini Hospital, Carpi
(MO), Italy; 3 Section of Ear, Nose and Throat (ENT), Department of Surgical Sciences, Dentistry, Gynaecology and Paediatrics,
University of Verona, Borgo Roma Hospital, Verona, Italy; 4 Audiology, Primary Care Department; AUSL of Modena, Modena, Italy


SUMMARY
Objective. translate and validate the Penn Acoustic Neuroma Quality of Life Scale into
Italian language (PANQOL-It)
Methods. the instrument was translated and psychometric properties were subsequently
assessed by administering the PANQOL-It to 124 outpatients together with the Depres-
sion Anxiety Stress Scale (DASS21) and the Understanding and Communicating domain
of the World Health Organization Disability Assessment Schedule II (WHODAS II-D1).
The internal consistency, test-retest reliability, construct and criterion-related validity were
assessed.
Results. Cronbach’s alpha coefficient was 0.92 for the total score and ranged from 0.44
to 0.90 in the seven domains. Significant test-retest reliability was observed (intraclass-
correlation = 0.75; p < 0.01). Moderate correlation was reported between facial dysfunction
domain and objective facial involvement (p < 0.01). Moderate to strong correlations were
observed between anxiety, general health domains and all subscales of the DASS21, and
                                                                                                   Received: August 7, 2022
between WHODAS II-D1 and general health and energy domains (p < 0.01). These latter
                                                                                                   Accepted: January 17, 2023
results indicated good construct and criterion-related validity respectively.
Conclusions. PANQOL-It presented more than acceptable psychometric properties and its
                                                                                                   Correspondence
adoption is justified for both clinical and research purposes.
                                                                                                   Enrico Apa
KEY WORDS: vestibular schwannoma, dizziness, hearing loss, tinnitus, health                        Department of Medical and Surgical Sciences for
                                                                                                   Children and Adults, Otorhinolaryngology Unit,
                                                                                                   University of Modena and Reggio Emilia, Azienda
RIASSUNTO                                                                                          Ospedaliero-Universitaria of Modena, via Largo del
Obiettivo. Validare in lingua italiana il Penn Acoustic Neuroma Quality of Life Scale              Pozzo 71, 41125 Modena, Italy
(PANQOL-It).                                                                                       Tel. +39 059 4222806; +39 059 4225660
Metodi. Lo strumento è stato somministrato a 124 pazienti ambulatoriali congiuntamente             E-mail: apa.enrico@gmail.com
alle versioni italiane del Depression Anxiety Stress Scale (DASS21) e della sezione “Un-
derstanding and Communicating” del World Health Organization Disability Assessment                   How to cite this article: Apa E, Maccarrone
Schedule II (WHODAS II-D1). Sono state valutate la consistenza interna, la riproducibilità,          F, Gherpelli C, et al. Italian validation of
la validità di costrutto e di criterio.                                                              the Penn Acoustic Neuroma Quality of Life
Risultati. Il coefficiente alfa di Cronbach è risultato di 0,92 per il punteggio totale e com-       Scale (PANQOL-It). Acta Otorhinolaryn-
preso tra 0,44 e 0,90 nelle sette dimensioni del questionario, mentre la correlazione intra-         gol    Ital   2023;43:130-139.    https://doi.
classe è risultata significativa (ICC = 0,75; p < 0,01). Sono state osservate una moderata           org/10.14639/0392-100X-N2263
correlazione tra la dimensione “facial dysfunction” e il grado di paralisi del nervo fac-
ciale, da moderate a forti correlazioni tra le dimensioni “anxiety” e “general health” e il        © Società Italiana di Otorinolaringoiatria
DASS21, e tra il WHODAS II-D1 e le dimensioni “general health” ed “energy” (p < 0,01).             e Chirurgia Cervico-Facciale
Questi riscontri indicano rispettivamente una buona validità di costrutto e di criterio.
Conclusioni. Il PANQOL-It ha presentato proprietà psicometriche ottime e la sua adozione                          OPEN ACCESS
è giustificata sia per scopi clinici che di ricerca.                                               This is an open access article distributed in accordance with
                                                                                                   the CC-BY-NC-ND (Creative Commons Attribution-Non-
PAROLE CHIAVE: schwannoma vestibolare, instabilità, perdita dell’udito, acufene, salute            Commercial-NoDerivatives 4.0 International) license. The
                                                                                                   article can be used by giving appropriate credit and mentio-
                                                                                                   ning the license, but only for non-commercial purposes and
                                                                                                   only in the original version. For further information: https://
                                                                                                   creativecommons.org/licenses/by-nc-nd/4.0/deed.en




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                                                                 First Italian disease-specific quality-of-life instrument for vestibular schwannoma




Introduction                                                         examination were considered. The following exclusion cri-
                                                                     teria were established: presence of a cerebellopontine angle
Vestibular schwannomas (VS), also referred to as acoustic
                                                                     tumour other than VS, neurofibromatosis type II, cultural
neuromas, are benign tumours that origin from the VIII cra-
                                                                     barriers, major neurological or psychiatric disorders, or any
nial nerve with growth within the cerebellopontine angle in
                                                                     other morbidity that could prevent them from understand-
approximately 90% of cases. Its incidence has increased
                                                                     ing and answering the self-administered questionnaires by
over the past few years from 10.9 to 33.8 per 1,000.000
                                                                     paper and pencil. Tumour characteristics were obtained
per year most probably due to the routine use of magnetic
                                                                     from medical and surgical records; recent history, a careful
resonance imaging (MRI) 1. In most cases, initial clinical
                                                                     bedside examination and un updated audiogram were per-
symptoms (progressive hearing loss, tinnitus, unsteadiness,
                                                                     formed in the same session. The tumour size was measured
facial numbness) are mild with no/low impact on quality of
                                                                     considering the maximum diameter and the extension in
life 2. Despite the routine use of neuroimaging, there may
                                                                     the internal acoustic meatus (IAM) and/or the cerebello-
be a significant delay between the onset of signs and symp-
                                                                     pontine angle (CPA) according to the Koos classification 11.
toms and the definitive diagnosis. On the contrary, the sur-
                                                                     Facial nerve involvement was measured considering the
gical removal of the tumour often causes severe aesthetic
                                                                     House-Brackmann (HB) grading system 12. The mean pure
and functional damages in the cranial-cervical district due
                                                                     tone average (PTA) was calculated over 500, 1000, 2000
to accidental injury to the facial, trigeminal and acoustic
                                                                     and 4000 Hz and for the hearing level the classification of
nerves. Taken together, these lesions account for a dramatic
                                                                     the American Academy of Otolaryngology-Head and Neck
post-operative symptomatology and a significantly worsen-
                                                                     Surgery Foundation guidelines that also considers the word
ing of quality of life. While mortality rates are low, comor-
                                                                     recognition threshold was used, whereas the unsteadiness
bidities are relevant in relation to remarkable psychologi-
                                                                     and tinnitus were considered according to patient reports 13.
cal distress 3. The comparison between different treatments
and consequent results was barely performed across cul-
                                                                     Instruments
tures and countries until 2010, due to the methodological
                                                                     The PANQOL is a 26-item disease-specific question-
weakness of the published studies and the lack of a disease-
specific quality of life inventory. In fact, most of the stud-       naire for VS 5. Each item is rated on a Likert scale from
ies appreciated the complexity of this clinical condition            1 (“strongly disagree”) to 5 (“strongly agree”) and items
through a plethora of generic questionnaires 4. In 2010, the         on a certain factor are grouped together in domains, result-
first disease-specific quality of life instrument for VS, the        ing in seven scores calculated on a normalised scale from
Penn Acoustic Neuroma Quality of Life Scale (PANQOL),                0 to 100, from worst to best quality of life experienced.
was developed with more than acceptable psychometric                 Each domain is composed by a different number of items:
properties 5. Up to now successful validation studies, in            6 items for balance (Ba) and energy subscales (En), 4
most widely spoken languages different from the original             items for hearing loss (HL) and anxiety subscales (Ax), 3
English version, have been carried out 6-9. Although an Ital-        items for facial dysfunction domain (FD), 2 items for gen-
ian version of PANQOL has recently been used, there is no            eral health domain (GH) and 1 item for the pain domain
validation study or assessment of the psychometric proper-           (Pa). The total instrument score is calculated as the aver-
ties of this instrument 10. Hence, the purpose of the present        age of domain scores. The PANQOL was translated and
study is to translate and validate the PANQOL into Italian           adapted into Italian language following the standards es-
in order to provide the scientific community with an Italian         tablished by the International Quality of Life Assessment
version of an appropriate disease-specific questionnaire for         project (IQOLA), for academic use and under licence by
both clinical and research aims.                                     the original authors 14. The first phase was the reproduction
                                                                     of the questionnaire through an iterative translation process
                                                                     conducted by four bilingual audiologists of the Azienda
Methods and materials                                                Ospedaliero-Universitaria of Modena, Italy (Fig. 1). In this
Participants                                                         way, the Italian version of PANQOL (PANQOL-It) was ob-
A cohort of 124 subjects was recruited in this monocentric           tained (Appendix A). Relationships with other scales and
and cross-sectional study from July 2019 to October 2021.            external variables were evaluated. In particular, facial nerve
This sample size was adopted in accordance with the origi-           involvement, hearing level, self-perceived unsteadiness and
nal article 5. Only consecutive outpatients attending the            tinnitus were considered as external variables, whereas oth-
Otorhinolaryngology Unit of the Azienda Ospedaliero-Uni-             er scales used were the Understanding and Communicat-
versitaria of Modena (Italy), aged 18 years or more, with a          ing domain of the Italian version of the World Health Or-
diagnosis of VS or treated for VS at least six months before         ganization Disability Assessment Schedule II (WHODAS



                                                                                                                                              131
E. Apa et al.




                                                                 rates respectively. One-way ANOVA test and Chi-square
                                                                 test were used to compare continuous and qualitative vari-
                                                                 ables across different treatment groups. Given the different
                                                                 indications for the various therapeutic options, a significant
                                                                 difference in neuroma and functional parameters was ex-
                                                                 pected 17. Furthermore, PANQOL-It scores were reported
                                                                 by descriptive statistics in order to compare them with re-
                                                                 sults from other validation studies.
                                                                 As a crucial part of the validation process of PANQOL-
                                                                 It, different psychometric properties were evaluated 18. The
                                                                 reliability is important for discriminative purposes since it
                                                                 concerns the degree to which patients can be distinguished
                                                                 from each other. It can be expressed in terms of internal
                                                                 consistency and reproducibility. The former is the extent
                                                                 to which items in a questionnaire and its domains are ho-
                                                                 mogeneous. In order to assess it, the correlations among
                                                                 different subscales were analysed. Since PANQOL-It is a
                                                                 disease-specific instrument composed of items grouped to-
                                                                 gether in seven different domains, significant but not high
                                                                 correlations across subscales were expected, whereas sig-
Figure 1. Iterative translation process.                         nificant high correlations between each domain and the
                                                                 total score was expected. In order to measure the internal
                                                                 consistency Cronbach’s alpha coefficient was used. It indi-
II-D1) and the Depression Anxiety Stress Scale (DASS21).         cates the correlation among the items and should be calcu-
Hence, in addition to PANQOL-It patients were invited            lated for each domain separately. Despite the use of cut-off
to complete both questionnaires already validated in Ital-       being debated, in accordance with the international com-
ian 15,16. All three questionnaires were delivered to patients   mon interpretation, the minimum acceptable value of al-
and self-administered by paper-and-pencil. The WHODAS            pha is 0.7, an alpha from 0.8 to 0.9 is considered good and
II-D1 is the first domain of an instrument developed by the      an alpha from 0.9 excellent. However, an alpha that is too
World Health Organization (WHO). It consists of 6 items          high may suggest that some items are redundant, such as
aimed at measuring behavioural limitations and restrictions      they are testing the same question but in a different guise.
in participation experienced by an individual in the last 30     Thus, the statistical procedure “Cronbach’s alpha if item
days, independently from diseases, lesions, psychological        is deleted” was performed in order to establish how much
difficulties, or problems related to alcohol and drugs. Each     any single item contributes to the whole Cronbach’s alpha
item is ranked on a 5-point ordinal scale and higher score       coefficient. In addition, the floor and ceiling effects were
indicates higher disability. The DASS21 is a self-reported       calculated by percentage frequency of lowest or highest
scale which measures symptoms of depression (De), anx-           possible score achieved by subjects. A percentage > 15%
iety (An) and stress (St) in both clinical and non-clinical      indicates that the reliability is reduced due to the difficulty
samples of adults. It consists of 21 items, 7 for each do-       in distinguishing patients with the lowest or highest possi-
main, rated on a 4-point severity/frequency scale where          ble score from each other.
higher scores indicate greater psychological impairment.         The reproducibility is the degree to which repeated meas-
All data were collected anonymously in a Microsoft Excel®        urements in stable patients provide a similar answer. Thus,
database.                                                        it measures the stability of the instrument (test-retest reli-
                                                                 ability). PANQOL-It was administered a second time to 52
Psychometric tests and statistical analysis                      randomly selected patients who responded with no change
Statistical analysis and graphical representation were per-      in their quality of life after a six-week interval in a phone-
formed with the Statistical Package for Social Sciences          interview format. Intraclass Correlation Coefficients (ICC)
(IBM SPSS® version 25.0 for Microsoft Windows®). Epi-            with 95% confidence intervals were used for this purpose.
demiological features of the participants were reported by       The construct validity refers to the ability of an instrument
descriptive statistics. Quantitative and qualitative variables   to measure a theoretically derived hypothesis concerning
were expressed as means with standard deviations (SD) and        the concepts that are being measured. Even if no meaning-



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                                                                   First Italian disease-specific quality-of-life instrument for vestibular schwannoma




ful correlation with age, gender, tumour size, or audiometric
data and PANQOL subscales was reported in the original
article, given the presence of seven different domains, the
construct validity was assessed through a correlation study.
In detail, correlations between tumour size and FD, facial
involvement and FD, hearing function and HL, complaints
of tinnitus and HL, self-perceived unsteadiness and Ba were
evaluated. Finally, the criterion-related validity was assessed.
This indicates how well the scores converge with other meas-
ures of the same construct. The PANQOL-It domains were
correlated to De, An and St of the DASS21 and WHODAS
II-D1. In particular, since PANQOL-It scores are calculated            Figure 2. PANQOL-It domains and total score in the study sample.
                                                                       PANQOL-It: Italian version of the Penn Acoustic Neuroma Quality of Life Scale;
from 0 to 100, from worst to best quality of life experienced,         Ba: balance domain; HL: hearing loss domain; FD: facial dysfunction domain;
whereas for WHODAS II-D1 and DASS21 higher scores                      En: energy domain; GH: general health domain; Ax: anxiety domain; Pa: pain
indicate higher disability and greater psychological impair-           domain.
ment respectively, significant negative correlations between
Ax and An, GH and all DASS21 subscales and WHODAS
II-D1, and En and all DASS21 subscales were expected.                  from 0.44 in GH to 0.90 in Ba (Tab. III). Cronbach’s alpha
Spearman’s correlation coefficient rho for ranked variables            was not applicable to Pa, since it is a single-element do-
was used for all correlation studies to obtain a robust estima-        main. The procedure ‘Cronbach’s alpha if item is deleted’
tion even in case of non-normal distribution 19. The level of          showed improvement of alpha coefficient if item 9, 11 and
statistical significance was considered reached if p-value was         21 was deleted in Ba, FD and En respectively (Tab. III). No
< 0.05 in all procedures.                                              floor or ceiling effect was found considering the total score
                                                                       since no patients scored 0 or 100 at PANQOL-It, whereas
                                                                       ceiling effect was observed in Pa, Ax, GH, Ba. ICC ob-
Results                                                                tained in the two distinct administrations resulted in 0.75
A cohort of 124 outpatients was recruited to take part in              for PANQOL-It total score and ranged from 0.42 for En to
the study. Twenty-two patients (17.7%) were managed                    0.94 for Pa (Tab. III).
conservatively with wait and scan policy, while 98 (79.0%)
and 4 (3.2%) underwent surgical resection or stereotactic              Construct validity
radiosurgery with Gamma-Knife at least six months before               Tumour size and facial nerve involvement significantly
recruitment respectively. Epidemiological features of the              correlated with FD. In addition, very weak and weak cor-
samples are represented in Table I. No significant differ-             relations were observed with tumour maximum diameter
ences in age, gender, and hearing function were observed               (rho = -0.20; p < 0.05) and tumour extension (rho = -0.30;
among the various treatment groups, whereas tumour size                p < 0.01), respectively, and moderate correlation was ob-
expressed with maximum diameter and Koos classification,               served with the facial involvement (rho = -0.53; p < 0.01).
facial involvement, tinnitus referred and self-perceived un-           No correlation was observed between hearing function and
steadiness were significantly different across the various             HL (rho = -0.09; p = 0.322). In contrast, the aforementioned
treatment groups. Figure 2 shows results for the total score           domain was significantly correlated with complaints of tin-
and the seven PANQOL-It domains in the study sample.                   nitus (rho = -0.22; p < 0.05). In addition, weak correlations
                                                                       were observed between tinnitus and other domains such
Reliability                                                            as Ax (rho = -0.40; p < 0.01), GH (rho = -0.34; p < 0.01)
Significant correlations were observed across PANQOL-                  and En (rho = -0.39; p < 0.01). The correlation between Ba
It domains, except between Pa and GH (Tab. II). Correla-               and self-perceived unsteadiness was significant but weak
tions in general ranged from weak to moderate, whereas all             (rho = -0.25; p < 0.01) and the same was observed between
domains showed moderate to strong correlations with the                the same symptom and Ax (rho = -0.22; p < 0.05).
PANQOL-It total score. Cronbach’s alpha coefficient was
0.92 in the total score and the procedure ‘Cronbach’s alpha            Criterion-related validity
if item is deleted’ pointed out that removal of each item              Significant correlations were reported across DASS21
did not improve the alpha coefficient of the instrument.               subscales and PANQOL-It domains. In detail, moderate
Specifically, the Cronbach’s alpha for each domain ranged              to strong correlations were observed between Ax and De



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E. Apa et al.




Table I. Epidemiological features of the sample.
 Variable                                Total                        Surgery                 Conservative             Gamma-Knife                Significance
                                       (n = 124)                      (n = 98)                  (n = 22)                 (n = 4)
    Gender
      Female                              54 (43.5%)                 8 (36.4%)                 46 (46.9%)                 0 (0.0%)                 p = 0.135a
      Male                                70 (56.5%)                 14 (63.6%)                52 (53.1%)                4 (100.0%)
    Age
      Years                                 59.65                      63.00                     58.57                      67.50                  p = 0.132 b
                                        (12.26; 22-80)             (11.29; 36-79)            (12.45; 22-80)             (7.51; 61-74)
    Tumour size (mm)
       d < 10                             16 (12.9%)                 10 (45.5%)                 6 (6.1%)                   0 (0.0%)                     ⁎⁎a
       d = 10-20                          52 (41.9%)                 12 (54.5%)                36 (36.7%)                4 (100.0%)
       d > 20                             56 (45.2%)                   0 (0.0%)                56 (57.1%)                  0 (0.0%)
       I Koos                             16 (12.9%)                 10 (45.5%)                 6 (6.1%)                   0 (0.0%)                     ⁎⁎a
       II Koos                            34 (27.4%)                 8 (36.4%)                 24 (24.5%)                 2 (50.0%)
       III Koos                           52 (41.9%)                  4 (18.2%)                46 (46.9%)                 2 (50.0%)
       IV Koos                            22 (17.7%)                   0 (0.0%)                22 (22.4%)                  0 (0.0%)
    Hearing function
       A                                  36 (29.0%)                 6 (27.3%)                 28 (28.6%)                2 (50.0%)                 p = 0.325a
       B                                  16 (12.9%)                  2 (9.1%)                 14 (14.3%)                 0 (0.0%)
       C                                  46 (37.1%)                 12 (54.5%)                32 (32.7%)                2 (50.0%)
       D                                  26 (21.0%)                  2 (9.1%)                 24 (24.5%)                 0 (0.0%)
    Tinnitus
       Yes                                38 (30.6%)                 14 (63.6%)                22 (22.4%)                2 (50.0%)                      ⁎⁎a
       No                                 86 (69.4%)                  8 (36.4%)                76 (77.6%)                2 (50.0%)
    Unsteadiness
       Yes                                68 (54.8%)                 10 (45.5%)                44 (44.9%)                2 (50.0%)                      ⁎a
       No                                 56 (45.2%)                 12 (54.5%)                54 (55.1%)                2 (50.0%)
    Facial involvement
       I HB                               44 (35.5%)                 20 (90.9%)                20 (20.4%)                4 (100.0%)                     ⁎⁎a
       II HB                              26 (21.0%)                  2 (9.1%)                 26 (26.5%)                 0 (0.0%)
       III HB                             24 (19.4%)                  0 (0.0%)                 22 (22.4%)                 0 (0.0%)
       IV HB                              22 (17.7%)                  0 (0.0%)                 22 (22.4%)                 0 (0.0%)
       V HB                                6 (4.8%)                   0 (0.0%)                  6 (6.1%)                  0 (0.0%)
       VI HB                               2 (1.6%)                   0 (0.0%)                  2 (2.0%)                  0 (0.0%)
a
 Chi-square, bANOVA test, ⁎ p value < 0.05, ⁎⁎ p value < 0.01.
HB: House-Brackmann grading system; d: maximum diameter. Hearing Function according to the American Academy of Otolaryngology – Head and Neck Surgery Foundation clas-
sification 13.


Table II. Correlations between PANQOL-It domain scores.
                                Balance      Hearing loss                Facial dysfunction           Energy         General health          Anxiety           Pain
    Balance                            1.00
    Hearing loss                       0.32**            1.00
    Facial dysfunction                 0.31**            0.38**                  1.00
    Energy                             0.54**            0.51**                  0.50**               1.00
    General health                     0.41**            0.35**                  0.25**               0.54**              1.00
    Anxiety                            0.48**            0.48**                  0.24**               0.61**              0.57**              1.00
    Pain                               0.24**            0.29**                   0.20*               0.23**               0.13               0.37**          1.00
    Total score                        0.65**            0.68**                  0.58**               0.80**              0.68**              0.78**          0.54**
* p value < 0.05, ** p value < 0.01.




134
                                                                                             First Italian disease-specific quality-of-life instrument for vestibular schwannoma




Table III. PANQOL-It reliability.
                                            Cronbach’s alpha          “Cronbach’s alpha if item            Item          Floor          Ceiling            ICC (95 % CI)
                                               coefficient                  is deleted”                   deleted        effect         effect
 Balance                                            0.90                            0.92                     9            0.0%          17.7%           0.79 (0.63-0.88)**
 Hearing loss                                       0.77                              -                      -            1.6%           8.1%           0.72 (0.51-0.84)**
 Facial dysfunction                                 0.49                            0.66                    11            0.0%          24.2%           0.56 (0.24-0.75)**
 Energy                                             0.79                            0.83                    21            0.0%          12.9%           0.42 (-0.01-0.67)*
 General health                                     0.44                              -                      -            4.8%          3.2%            0.43 (0.01-0.67)*
 Anxiety                                            0.88                              -                      -            1.6%          37.1%           0.90 (0.83-0.94)**
 Pain                                                NA                              NA                     NA            9.7%          45.7%           0.94 (0.89-0.96)**
 Total score                                        0.92                              -                      -            0.0%           0.0%           0.77 (0.59-0.87)**
* p value < 0.05, ** p value < 0.01.
NA: not applicable; ICC: Intraclass Correlation Coefficient; IC: confidence interval.



(rho = -0.63; p < 0.01), Anxiety (rho = -0.59; p < 0.01),                                        was representative of all subjects suffering from or treated
Stress (rho = -0.55; p < 0.01), between GH and Depression                                        for VS. Since the first six months of time after diagnosis is
(rho = -0.56; p < 0.01), Anxiety (rho = -0.51; p < 0.01), and                                    associated with poor quality of life, especially with regards
between En and Depression (rho = -0.60; p < 0.01), Anxiety                                       to anxiety, considering only patients at least six months af-
(rho = -0.47; p < 0.01), Stress (rho = - 0.51; p < 0.01). In ad-                                 ter treatment may provide the benefit of assessing patients
dition, moderate to strong correlations were observed also                                       with a stable quality of life 20. Three groups were identified
between WHODAS II-D1 and GH (rho = -0.44; p < 0.01)                                              according to the treatment and, even if no significant differ-
and En (rho = -0.60; p < 0.01).                                                                  ences in age, gender, and hearing function were observed
                                                                                                 among the three groups, they were significantly different
Discussion                                                                                       with relation to tumour size, facial involvement, tinnitus
The objective of the current study was to validate the PAN-                                      and self-perceived unsteadiness. This result was similar to
QOL into the Italian language. For this purpose, the per-                                        the results obtained by McLaughlin et al. 21. Since the ob-
mission by the original authors was obtained and interna-                                        jective was not to examine the quality of life across treat-
tional standards established by IQOLA were adopted. The                                          ment groups, it was not a limitation in the current study.
instrument was translated using the iterative translation                                        The means and standard deviations of PANQOL-It do-
process and psychometric properties were subsequently                                            mains in the sample yielded similar results to the original
assessed by administering PANQOL-It to 124 outpatients.                                          study and other validation studies, with the exception of
The number of subjects recruited was in accordance with                                          GH (Tab. IV).
the original article and other validation studies 5-9. Only                                      Responses to this dimension were smaller in the present
patients in follow-up for VS or who underwent surgery or                                         study, reflecting worse self-perceived general health. In ad-
stereotactic radiosurgery at least six months before recruit-                                    dition, for FD, En, Pa and the total score the results were
ment were considered. This was crucial for two reasons:                                          slightly smaller. This may be regarded as a limitation be-
only stable patients were taken into account and the sample                                      cause, according to item response theory, the equivalence of


Table IV. Means and standard deviations of PANQOL domains in the current study and those in the original article and other validation studies.
                    Original version 5       Dutch version 6    Spanish version 7       French version 8 Japanese version 9                Current study
 Balance                        72.9 (20.5)                66.0 (29.4)                  68.36 (29.57)          62.8 (28.2)               86 (18)              69.2 (27.8)
 Hearing loss                   63.8 (22.2)                41.3 (27.3)                  66.43 (28.33)          57.2 (25.3)               72 (22)              54.6 (26.2)
 Facial dysfunction             85.4 (18.9)                83.6 (21.3)                  75.30 (24.29)          84.2 (15.4)               92 (14)              70.5 (24.8)
 Energy                         67.6 (23.0)                66.2 (28.9)                  66.97 (28.79)          69.3 (25.7)               79 (20)              63.2 (27.8)
 General health                 68.3 (21.3)                60.4 (22.1)                  53.24 (15.73)          59.7 (19.9)               59 (18)              44.8 (27.5)
 Anxiety                        73.5 (20.4)                71.3 (25.2)                  80.09 (29.57)          72.4 (23.2)               83 (19)              76.6 (26.7)
 Pain                           77.7 (28.7)                70.4 (35.9)                  74.07 (35.0)           78.9 (32.6)               80 (25)              68.2 (35.8)
 Total score                    72.8 (15.6)                 69 (21)                      69.2 (20.8)                -                    79 (14)              63.9 (19.1)




                                                                                                                                                                            135
E. Apa et al.




translations is supported when items in different languages
are placed similarly on the scale 14. Nevertheless, more than
acceptable reliability was demonstrated for the PANQOL-It.
With the exception between Pa and GH, weak to moderate
significant correlations were observed across PANQOL-It
domains, while all dimensions showed moderate to strong
correlations with the total score. The internal consistency
of PANQOL-It was compared with the internal consistency
of PANQOL and the Dutch, Spanish, French and Japanese
versions 6-9. Since the total score is the average of the seven
subscales, the internal consistency was also assessed by cal-
culating Cronbach’s alpha coefficient for each domain sepa-
rately. The alpha was good for Ba and Ax, and acceptable for                      Figure 3. Internal consistency of PANQOL domains in different versions of
HL and En (Tab. V). A coefficient of less than 0.7 was ob-                        the instrument.
tained for GH according to the Dutch, French and Japanese                         Usa: original version of PANQOL 5; Ned: Dutch version 6; Spa: Spanish version
                                                                                  7
                                                                                   ; Fra: French version 8; Jap: Japanese version 9; Ita: Italian version of the Penn
versions and for FD according to the French version. The                          Acoustic Neuroma Quality of Life Scale (PANQOL-It); Ba: balance domain; HL:
poor reliability obtained for those two dimensions could be                       hearing loss domain; FD: facial dysfunction domain; En: energy domain; GH:
explained by the small number of items that composed them,                        general health domain; Ax: anxiety domain; Pa: pain domain.
since GH and FD are composed of two and three items, re-
spectively. Moreover, in contrast with the original study and
other validation studies, in the current study a large propor-                    did not improve the alpha of the entire instruments. Thus,
tion of patients with facial involvement was observed 5-8. In                     we decided to remove none of them. No floor effect was ob-
fact, in the original article only 18 of 143 subjects present-                    served for PANQOL-It and its domains whereas a ceiling ef-
ed a facial involvement with HB more than I, while in the                         fect was reported in Pa, GH, Ba and Ax. It was also observed
Dutch, Spanish and French studies two, one and no patients                        in the original article for the same dimensions. Although it
with facial involvement were observed, respectively. On the                       is predictable for Pa and GH, since those domains are com-
other hand, in the current study 8 patients (6.4%) presented a                    posed of one and two items, respectively, it should be consid-
severe facial involvement or a total facial paralysis. Finally,                   ered a slight limit. In particular, this could lead to a difficulty
Cronbach’s alpha was not applicable to Pa because it is a sin-                    in distinguishing patients with the highest possible scores in
gle-element domain. Therefore, FD, GH and Pa represent-                           these domains. The test-retest reliability was used to mea-
ed the main drawbacks of PANQOL-It. Nonetheless, since                            sure the reproducibility of the instrument. It was adequate
Cronbach’s alpha coefficient of the total score was 0.92 and                      since a significant ICC between the scores of PANQOL-It
coefficients of each domain were within the range of other                        and its domains was observed. In the original article, the au-
validation studies, the internal consistency resulted excellent                   thors reported no meaningful correlations with tumour size
for the entire instrument and more than acceptable for each                       and audiometric data. The same was observed in the current
domain (Fig. 3). Even if the removal of items 9, 11 and 21                        study, since significant but very weak correlations were seen
from Ba, FD and En improved the alpha coefficient of the                          only between FD and tumour size and between HL and hear-
respective domains, the removal of the aforementioned items                       ing function. In contrast, moderate correlation was observed


Table V. Internal consistency (Cronbach’s alpha) of PANQOL domains in the current study and those in the original article and other validation studies.
                      Original version 5      Dutch version 6    Spanish version 7        French version 8 Japanese version 9                Current study
 Balance                      0.87                   0.94                  0.91                     0.90                     0.92                    0.90
 Hearing loss                 0.77                   0.75                  0.85                     0.68                     0.87                    0.77
 Facial dysfunction           0.71                   0.65                  0.67                     0.46                     0.78                    0.49
 Energy                       0.88                   0.91                  0.89                     0.89                     0.79                    0.79
 General health               0.73                   0.31                  0.67                     0.25                     0.42                    0.44
 Anxiety                      0.81                   0.88                  0.97                     0.72                     0.81                    0.88
 Pain                          NA                     NA                    NA                       NA                       NA                      NA
 Total score                    -                      -                   0.87                       -                      0.93                    0.92




136
                                                                   First Italian disease-specific quality-of-life instrument for vestibular schwannoma




between facial involvement and facial domain, which was                Conclusions
consistent with the original article. Significant but weak cor-
                                                                       In the current study, we translated and validated the Ital-
relations were also observed between Ba and self-perceived
                                                                       ian version of the PANQOL (PANQOL-It). While the sam-
unsteadiness, whereas weak to moderate correlations were
                                                                       ple showed smaller values in the seven dimensions and in
reported between complaints of tinnitus and HL, Ax, GH and
                                                                       the total score compared to the original article and other
En, in accordance with the previous study by Kojima et al. 3
                                                                       validations studies, the instrument presented more than ac-
They observed that tinnitus severity was the strongest pre-
                                                                       ceptable psychometric properties. Cronbach’s alpha coef-
dictor of quality of life in patients with VS managed with a
                                                                       ficient resulted excellent for the entire instrument and was
wait and see policy. In particular, it significantly correlated
                                                                       consistent for each domain with the original article and
with hearing impairment, anxiety and depression, whereas
                                                                       other validation studies. Therefore, its adoption is justified
unsteadiness and hearing impairment had significant but less
                                                                       for both clinical and research purposes for Italian-speaking
impact on quality of life. Moderate to strong significant cor-
                                                                       subjects. In order to properly interpret the results, clinicians
relations were observed between all subscales of DASS21
                                                                       need to be aware that the direction of the score is different
and Ax and En, and between Depression and Anxiety of
                                                                       for item 25, the only one for which the answer should be
DASS21 and GH. Those results were predictable since ma-
                                                                       counted not inversely.
ny items of PANQOL derived from the Hospital Anxiety and
Depression Scale, a reliable and valid screening instrument            Acknowledgements
for anxiety and depression intended for the hospital medical           The authors thank Micheal J. Ruckenstein MD and his
outpatient clinical setting 22. The inclusion of psychosocial          team, Department of Otorhinolaryngology Head & Neck
dimensions regarding anxiety, energy and general health rep-           Surgery, Hospital of the University of Pennsylvania, USA,
resents an absolutely helpful feature for clinicians since it          for their permission to use and translate into Italian lan-
allows for wider inferences to be made using the instrument.           guage the original version of the Penn Acoustic Neuroma
Moreover, since anxiety or depression are frequent problems            Quality of Life Scale and validate its Italian version (PAN-
in patients suffering for VS, it is important to assess these          QOL-It).
specific dimensions. In fact, Pruijn and colleagues observed
that anxiety and lack of energy were key factors and two of            Conflict of interest statement
the strongest predictors affecting and reducing the physical           This research did not receive any specific grant from fund-
and mental quality of life 23. In addition, moderate to strong         ing agencies in the public, commercial, or not-for-profit
correlations were observed between WHODAS II-D1 and                    sectors.
GH and En. The former is the cognition domain of an in-
strument aimed at measuring behavioural limitations and                Funding
restrictions to participation experienced by an individual. It         Internal departmental funding was used to support this
evaluates understanding and communicating and results ob-              study without commercial sponsorship. This research re-
served suggest a moderate to strong relationship to GH and             ceived no other specific grant from any funding agency in
En dimensions and the social functioning. Finally, only one            the public, commercial, or not-for-profit sectors.
aspect should be considered. Headache and any other symp-
toms that the patient might experience which were not asked            Author contributions
for in the questionnaire could influence the quality of life 23.       EA: investigation, data curation, statistical analysis, visual-
In addition, Bender et al., administering different question-          isation, writing – original draft; FM: conceptualisation, da-
naires to patients before surgery and at the first follow-up,          ta curation; CG: investigation, data curation; LS: resources,
observed a significantly worse quality of life in depressed            supervision; DM: conceptualisation, methodology, project
patients both before and after surgery 24. Thus, the exis-             administration, supervision, statistical analysis, resources;
tence of a disease-specific quality of life instrument such as         SP: supervision, writing – review & editing; RN: valida-
PANQOL does not preclude the concomitant use of generic                tion, visualisation, writing – review & editing.
instruments for assessing quality of life. The latter provide
broader comparisons and prevent potential confusion due to             Ethical consideration
psychological factors, whereas the former are typically more           The study was approved by the local Ethic Committee
responsive to change and provide more relevant information             (Protocol AOU 0019113/19 of 3rd July 2019) and was con-
to clinicians 25. Of course, in the research application the           ducted with permission from the authors, received by email
contextual use of both types of instruments is recommended.            on 14th January 2019, to use the original instrument. Writ-



                                                                                                                                                137
E. Apa et al.




ten informed consent was obtained from each participant                       13
                                                                                   Committee on Hearing and Equilibrium guidelines for the evaluation
and all procedures were in accordance with the ethical                             of hearing preservation in acoustic neuroma (vestibular schwanno-
                                                                                   ma). American Academy of Otolaryngology-Head and Neck Surgery
standards of the institutional research board and with the                         Foundation, INC. Otolaryngol Head Neck Surg 1995;113:179-180.
1964 Helsinki declaration and its later amendments.                                https://doi.org/10.1016/S0194-5998(95)70101-X
                                                                              14
                                                                                   Ware JE, Gandek BL, Keller SD; IQOLA Project Group. Evaluating
References                                                                         instruments used across-nationally; methods from the IQOLA Pro-
                                                                                   ject. In: Pilker B, editor. Quality of Life and Pharmacoeconomics in
1
     Reznitsky M, Petersen MMBS, West N, et al. Epidemiology of                    Clinical Trials. Second edition. Philadelphia, NY: Lippincott - Raven
     vestibular schwannomas – prospective 40-year data from an unse-               Publishers 1996. pp. 681-692.
     lected national cohort. Clin Epidemiol 2019;11:981-986. https://doi.
     org/10.2147/CLEP.S218670
                                                                              15
                                                                                   Federici S, Meloni F, Mancini A, et al. World Health Organisa-
2
     Foley RW, Shirazi S, Maweni RM, et al. Signs and symptoms of                  tion Disability Assessment Schedule II: contribution to the Ital-
     acoustic neuroma at initial presentation: an exploratory analysis.            ian validation. Disabil Rehabil 2009;31:553-564. https://doi.
     Cureus 2017;9:e1846. https://doi.org/10.7759/cureus.1846                      org/10.1080/09638280802240498
3
     Kojima T, Oishi N, Nishiyama T, et al. Severity of Tinnitus distress     16
                                                                                   Bottesi G, Ghisi M, Altoè G, et al. The Italian version of the de-
     negatively impacts quality of life in patients with vestibular schwan-        pression anxiety stress scales-21: factor structure and psychometric
     noma and mimics primary tinnitus. Front Neurol 2019;10:389. https://          properties on community and clinical samples. Compr Psychiatry
     doi.org/10.3389/fneur.2019.00389                                              2015;60:170-181. https://doi.org/10.1016/j.comppsych.2015.04.005
4
     Gauden A, Weir P, Hawthorne G, et al. Systematic review of quality       17
                                                                                   Hadjipanayis CG, Carlson ML, Link MJ, et al. Congress of neurologi-
     of life in the management of vestibular schwannoma. J Clin Neurosci           cal surgeons systematic review and evidence-based guidelines on sur-
     2011;18:1573-1584. https://doi.org/10.1016/j.jocn.2011.05.009                 gical resection for the treatment of patients with vestibular schwanno-
5
     Shaffer BT, Cohen MS, Bigelow DC, et al. Validation of a disease-             mas. Neurosurgery 2018;82:E40-E43. https://doi.org/10.1093/neuros/
     specific quality-of-life instrument for acoustic neuroma: the penn            nyx512
     acoustic neuroma quality-of-life scale. Laryngoscope 2010;120:1646-
     1654. https://doi.org/10.1002/lary.20988
                                                                              18
                                                                                   Terwee CB, Bot SD, de Boer MR, et al. Quality criteria were proposed
                                                                                   for measurement properties of health status questionnaires. J Clin Epi-
6
     van Leeuwen BM, Herruer JM, Putter H, et al. Validating the penn
     acoustic neuroma quality of life scale in a sample of dutch patients          demiol 2007;60:34-42. https://doi.org/10.1016/j.jclinepi.2006.03.012
     recently diagnosed with vestibular schwannoma. Otol Neurotol             19
                                                                                   Prion SK, Haerling KA. Making Sense of methods and measurement:
     2013;34:952-957. https://doi.org/10.1097/MAO.0b013e31828bb2bb                 spearman-rho ranked-order correlation coefficient. Clin Simul Nurs
7
     Medina MD, Carrillo A, Polo R, et al. Validation of the Penn Acous-           2014;10:535-536. https://doi.org/10.1016/J.ECNS.2014.07.005
     tic Neuroma Quality-of-Life Scale (PANQOL) for spanish-speaking          20
                                                                                   Carlson ML, Tombers NM, Kerezoudis P, et al. Quality of life within
     patients. Otolaryngol Head Neck Surg 2017;156:728-734. https://doi.           the first 6 months of vestibular schwannoma diagnosis with impli-
     org/10.1177/0194599816688640
                                                                                   cations for patient counseling. Otol Neurotol 2018;39:E1129-E1136.
8
     Oddon PA, Montava M, Salburgo F, et al. conservative treatment of             https://doi.org/10.1097/MAO.0000000000001999
     vestibular schwannoma: growth and penn acoustic neuroma quality of
     life scale in french language. Acta Otorhinolaryngol Ital 2017;37:320-
                                                                              21
                                                                                   McLaughlin EJ, Bigelow DC, Lee JY, et al. Quality of life in acous-
     327. https://doi.org/10.14639/0392-100X-1094                                  tic neuroma patients. Otol Neurotol 2015;36:653-656. https://doi.
9
     Nishiyama T, Oishi N, Kojima T, et al. Validation and multidimen-             org/10.1097/MAO.0000000000000674
     sional analysis of the japanese penn acoustic neuroma quality-of-life    22
                                                                                   Zigmond AS, Snaith RP. The hospital anxiety and depres-
     scale. Laryngoscope 2020;130:2885-2890. https://doi.org/10.1002/              sion scale. Acta Psychiatr Scand 1983;67:361-370. https://doi.
     lary.28488                                                                    org/10.1111/j.1600-0447.1983.tb09716.x
10
     Lucidi D, Fabbris C, Cerullo R, et al. Quality of life in vestibular     23
                                                                                   Pruijn IMJ, Kievit W, Hentschel MA, et al. What determines qual-
     schwannoma: a comparison of three surgical techniques. Eur Arch               ity of life in patients with vestibular schwannoma? Clin Otolaryngol
     Otorhinolaryngol 2022;279:1795-1803. https://doi.org/10.1007/                 2021;46:412-420. https://doi.org/10.1111/coa.13691
     s00405-021-06855-w
                                                                              24
                                                                                   Bender M, Tatagiba M, Gharabaghi A. Quality of life after vestibu-
11
     Erickson NJ, Schmalz PGR, Agee BS, et al. Koos classification of ves-
     tibular schwannomas: a reliability study. Neurosurgery 2019;85:409-           lar schwannoma surgery: a question of perspective. Front Oncol
     414. https://doi.org/10.1093/neuros/nyy409                                    2022;11:770789. https://doi.org/10.3389/fonc.2021.770789
12
     House JW, Brackmann DE. Facial nerve grading system. Oto-
                                                                              25
                                                                                   Guyatt GH, Feeny DH, Patrick DL. Measuring health-related
     laryngol Head Neck Surg 1985;93:146-147. https://doi.                         quality of life. Ann Intern Med 1993;118:622-629. https://doi.
     org/10.1177/019459988509300202                                                org/10.7326/0003-4819-118-8-199304150-00009




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                                                                                         First Italian disease-specific quality-of-life instrument for vestibular schwannoma




Appendix 1. The Italian Version of the Penn Acoustic Neuroma Quality-of-Life scale (PANQOL-It).
 La preghiamo di rispondere a ciascuna delle seguenti domande con un numero da 1 a 5 a seconda del grado di accordo con le affermazioni
 riportate.
                                                                                                          Non              Non    Non saprei              Sono            Sono
                                                                                                          sono         sono molto                       d’accordo         molto
                                                                                                       d’accordo        d’accordo                                       d’accordo
 1        La perdita d’udito ha influito negativamente sulle mie relazioni personali                        1               2         3                      4              5
 2        Ho difficoltà a seguire una conversazione a causa della perdita d’udito                           1               2         3                      4              5
 3        Non riesco a concentrarmi quando sento campanelli, fischi o altri rumori                          1               2         3                      4              5
 4        Ho un significativo problema di instabilità                                                       1               2         3                      4              5
 5        Mi sento instabile o senza equilibrio                                                             1               2         3                      4              5
 6        Quando cammino o sto in piedi, avverto un senso di rotazione o mi sento                           1               2         3                      4              5
          cadere
 7        Ho difficoltà a cambiare direzione quando cammino, a causa dei miei problemi                      1                2               3               4               5
          di equilibrio
 8        Ho difficoltà a camminare per casa al buio                                                        1                2               3               4               5
 9        A causa dei miei problemi di equilibrio, ho paura che gli altri mi credano                        1                2               3               4               5
          ubriaco
 10       A causa dell’aspetto della mia faccia, mi comporto in modo strano con le                          1                2               3               4               5
          persone
 11       Ho una eccessiva lacrimazione, o provo disagio o prurito ad un occhio                             1                2               3               4               5
 12       I miei problemi di mimica facciale influiscono negativamente sul mio modo di                      1                2               3               4               5
          parlare
 13       A causa del neurinoma dell’acustico, riesco ad avere meno successo di quanto                      1                2               3               4               5
          vorrei
 14       Avverto dolore alla testa dal lato del neurinoma dell’acustico                                    1                2               3               4               5
 15       Sono in qualche modo preoccupato del fatto che qualcosa di terribile stia per                     1                2               3               4               5
          accadere
 16       Ho spesso brutti pensieri                                                                         1                2               3               4               5
 17       Mi sento come se fossi rallentato                                                                 1                2               3               4               5
 18       Ho la sensazione di avere “farfalle nello stomaco”                                                1                2               3               4               5
 19       Mi faccio spesso prendere dal panico                                                              1                2               3               4               5
 20       A causa del neurinoma, mi sento spesso isolato                                                    1                2               3               4               5
 21       Provo difficoltà a concentrarmi sulle attività quotidiane, come leggere il                        1                2               3               4               5
          giornale o guardare la televisione
 22       Sono diventato più impaziente                                                                     1                2               3               4               5
 23       Ho perso energia o vitalità                                                                       1                2               3               4               5
 24       Ho difficoltà nel ricordare le cose                                                               1                2               3               4               5
 25       Ho una salute eccellente                                                                          1                2               3               4               5
 26       Ho il timore che il mio stato di salute peggiori negli anni a venire                              1                2               3               4               5
The scoring direction differs for item 25, the only one for which the answer should be counted not inversely. Items 4, 5, 6, 7, 8, 9 correspond to the balance domain (Ba), items 1, 2,
3, 20 correspond to the hearing loss domain (HL), item 10, 11, 12 correspond to the facial dysfunction domain (FD), items 13, 17, 21, 22, 23, 24 represent the energy domain
(En) and items 25, 26 the general health domain (GH), whereas items 15,16,18,19 and item 14 rank the magnitude of anxiety (Ax) and pain (Pa) domains respectively. The seven
scores are calculated on a normalised scale from 0 to 100, since worst to best quality of life experienced. The total score (Tot) is calculated as the equal average of domain scores.




                                                                                                                                                                                 139
