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Accepted: July 11, 2006 Published online: August 31, 2006
Psychosocial Functioning, Self-Perception and
Body Image and Their Auxologic Correlates in
Growth Hormone and Oestrogen-Treated Young
Adult Women with Turner Syndrome
Katrien Lagrou a Christelle Froidecoeur a Francisca Verlinde a
Margaretha Craen b Jean De Schepper c Inge François d Guy Massa a, e
on behalf of the Belgian Study Group of Paediatric Endocrinology
a Belgian Study Group of Paediatric Endocrinology (BSGPE), Departments of Paediatric Endocrinology,
Universities of b Gent , c Brussels , d Leuven , and e Virga Jesseziekenhuis Hasselt , Belgium
plaints, thought problems, delinquent behaviour). TS pa-
Adult ؒ Turner syndrome ؒ Psychosocial functioning ؒ Body
tients did not differ from the non-TS female group in their
image ؒ Oestrogen treatment ؒ Growth hormone treatment
bodily satisfaction. TS patients, particularly patients with a 45,X karyotype, perceived themselves as less socially com-petent. BMI was significantly related to the appraisal score
of the Body Attitude Scale, whereas height was not related
Few data are available on the psychosocial sta-
to any of the evaluated psychosocial parameters. Conclu-
tus of growth hormone (GH) and oestrogen treated women
The psychosocial adaptation of young adult women
with Turner syndrome (TS). In this study, we evaluated psy-
with TS, diagnosed at an early age and treated during child-
chosocial functioning, self-concept and body image in GH
hood with GH and oestrogens if indicated, appears to be
and oestrogen treated young adult women with TS and we
quite satisfactory. Follow-up of adult TS patients should not
studied the relationship with auxological parameters. Pa-
neglect the problem of overweight and associated psycho-
tients and Methods:
Thirty women with TS (mean 8 SD
age: 22.1 8 2.4 years), all treated with GH and oestrogens if indicated, and an age-matched reference group of 44 non-Turner female students (age: 20.5 8 2.1 years) completed 3 questionnaires evaluating, respectively, behavioural and
emotional problems (Young Adult Self Report), self-concept (Self Perception Profile for College Students) and body-im-
Turner syndrome (TS) is a genetic disorder caused by
age (Body Attitude Scale). Results:
TS patients did not report
the absence or structural abnormality of one X-chromo-
more behavioural and emotional problems compared to the
some, affecting approximately 1/2,000 females  . Al-
non-TS females except for attention problems; they even re-
though it has been well-established that adults with TS
ported fewer problems on some subscales (somatic com-
are susceptible to a large range of medical and psychoso-
Department of Paediatric Endocrinology, Virga Jesseziekenhuis
Tel. +32 11 309 864, Fax +32 11 309 898, E-Mail firstname.lastname@example.org
cial problems [2, 3] , psychological studies in adult women
Auxological and clinical data were collected from the ques-
with TS report a large variability regarding psychosocial tionnaire and completed with data from the medical record. Stud-
ied parameters were: karyotype, age and height at the start of GH
adaptation and functioning [4–10] . Moreover, the effects treatment, duration of GH treatment, age and height at arrest of
of height and height gain from growth hormone (GH) GH therapy, age at start of oestrogen therapy, age at menarche, treatment on psychosocial functioning still remain un-
final height and BMI. Height and BMI data were expressed as
clear [11–13] and the effects of oestrogen replacement standard deviation scores using the Flemish references  .
therapy have only been studied on self-concept and cog-
The test scores of the TS patient group were compared to those
of an age-matched (mean age: 20.5 8 2.1 years) reference group
nitive abilities in adolescents [14–16] . Recent studies sug-
of 44 female students (being trained in social work or nursing).
gest that adult height or height gain during GH therapy is not associated with quality-of-life scores, whereas old-
er age at menarche and the lack of sex hormone replace-
For the psychological evaluation, 3 standardized question-
ment during adult life are major determinants of psycho-
naires were used: behavioural and emotional problems were as-sessed by the Young Adult Self Report (YASR), self-perception by
logical well-being [17, 18] . In addition, Carel et al.  the Self Perception Profile for College Students (SPP) and body
found that the hearing status was a key predictor of image by the Bodily Attitude Scale (BAS).
health-related quality-of-life outcomes  .
The YASR is a questionnaire for young adults aged 18–30 years
In the present study, we evaluated the psychosocial developed by Achenbach  . The Dutch version of Verhulst 
functioning, the self-concept and the body image of was adapted to the Flemish language and the Belgian school sys-
tem  . We also developed a French version  , both versions
young adult women with TS treated during childhood with copyright permission of Achenbach and with back-transla-
with GH and oestrogens, if indicated. In addition, we tion by an independent bilingual translator. The YASR contains
studied the impact of several auxological parameters on a 119-item list of behaviours that might be problematic. Each be-these psychological parameters.
havioural item is rated as 0 for not true, 1 for somewhat or some-times true, and 2 for very true or often true. Higher scores indicate more problematic scores. This profile of behavioural problems can be interpreted by grouping the problem items into external-
Patients and Methods
izing (consisting of intrusive behaviour, aggressive behaviour, and delinquent behaviour); internalizing (consisting of anxious/
depressed and withdrawn) and total problems and/or viewing the
Young adult women with TS treated during childhood with
‘syndrome’ subscales, i.e., anxious/depressed, withdrawn, somat-
GH by paediatric endocrinologists belonging to the Belgian Study
ic complaints, thought problems, attention problems, intrusive
Group for Paediatric Endocrinology were contacted in order to
behaviour, aggressive behaviour, and delinquent behaviour. Re-
ask whether they were willing to participate in a psychological
sults were expressed as T-scores by comparison with the scores
study. A first part of the study consisted of a mailed questionnaire
obtained in a normative reference population  . Normalized
concerning medical care, health and psychosocial status to be
T-scores are assigned to the raw scores of a scale according to the
filled in at home  . A second part comprised a psychological
percentiles found for the raw scores in the normative sample.
evaluation taking place in the hospital where they were previ-
Mean 8 SD T-scores for the TS group and reference group were
ously or currently followed. The study was approved by the local
calculated, as well as the number of subjects with scores within
ethical committees of the participating centres and written in-
the clinical or pathological range (T-scores above 63 for the glob-
formed consent was obtained from all participants.
al behavioural scores and T-scores above 70 for the subscale
The inclusion criteria for the present study were: age between
scores) and within the borderline or problematic range (T-scores
18 and 23 years, patients with induced puberty after completion
between 60 and 63 for the global behavioural scores and T-scores
of at least the third year of pubertal induction and patients with
between 67 and 70 for the subscale scores).
spontaneous puberty at least 1 year after menarche. From the 102
The SPP was developed by Harter and Neemann  for the
patients who completed the mailed questionnaire 70 (69%) re-
ages 18–23 years and has been translated, adapted and back-trans-
ported to be willing to participate in the psychological evaluation.
lated into Dutch and French with permission of the author. This
Forty-nine of these 70 patients fulfilled the inclusion criteria of
questionnaire consists of 54 items subdivided into 12 subscales:
age and puberty. At final inclusion, 30/49 patients (61%) effec-
scholastic competence, intellectual ability, creativity, job compe-
tively accepted to participate in the psychological evaluation: 9
tence, athletic competence, physical appearance, peer acceptance,
patients could not be reached by phone or e-mail, 10 patients were
close friendship, romantic relationships, relationship with par-
not able or did not want to participate anymore mainly due to
ents, morality and sense of humour. In addition, the SPP also taps
changes in studies, working situation or residence.
global self-worth. Each of the content domains have four items per
All patients were treated during childhood with biosynthetic
subscale, while the self-worth subscale has six items. Each item
human GH at a dose of 0.34 mg/kg/week administered as a daily
describes two statements, one reflecting high competence, the
subcutaneous injection. In the patients without spontaneous pu-
other reflecting low competence. First the subject is asked which
berty, puberty was induced with low doses of ethinyl estradiol
kind of person he or she is most like; the subject then decides
(EE2) (starting dose, 50 ng/kg/day) usually after at least 2 years
whether that description is rather true or really true for him or
of GH therapy alone and at a minimum age of 11 years.
her. Each item is scored from 1 to 4, where a score of 1 indicates
low competence and a score of 4 reflects high competence. For
Auxological and clinical characteristics of the Turner pa-
each subscale the mean of the sum of the items is calculated.
The BAS is an originally Dutch questionnaire (‘Lichaams-
belevingsvragenlijst’) constructed by Baardman  in 1989 and
validated by Simis et al.  . Normative data are available for ages 12–22 years. We developed a French version by translating (and
back-translation by an independent bilingual translator) the Age at diagnosis, years
questionnaire from Dutch with permission of Koot. The BAS con-
sists of 45 items and are scored on the basis of a five-point Likert
scale. Appraisal, attribution and physical contact are the evalu-
ated subscales. The appraisal factor includes items such as ‘Are
you satisfied with the way your body looks?’ The attribution fac-
tor includes items such as ‘Do you think people avoid you because
of your appearance?’ The physical contact factor consists of items
such as ‘In general, how much do you like being touched by some-
body else?’ Raw scores go from a minimum of 45 to a maximum
of 225, higher scores indicating a more positive bodily image. For
each factor the mean of the sum of the items is calculated.
Auxological and clinical results are expressed as mean 8 SD.
Test scores are expressed as T-scores (YASR) or as the mean of the sum of the items (SPP and BAS). For the YASR the number of pa-
tients with T-scores within the borderline or clinical range was
also calculated. Differences in test scores between TS patients and
non-TS women were evaluated by the non-paired t
test, or by the Chi-Square test or Fisher’s exact test as appropriate. In addition,
a Of 26 patients who received EE2 treatment.
the results obtained in TS patients with 45,X karyotype were com-
b At last visit means at the end of GH treatment or at final
pared with those with another karyotype by the non-paired t test.
A p value ! 0.05 was considered significant for inter-group com-
c Current height and BMI are based upon patient’s report in
parisons. For the TS patient group potential associations between
test scores and auxological parameters were analysed by regres-sion analysis and expressed by the Pearson correlation coefficient. A p value ! 0.01 was considered as significant because of multiple testing. Statistical analysis was performed using SPSS software (version 13.0, SPSS Inc., USA).
m 2 (range: 17.2–46.7 kg/m2); 2 (7%) women were over-weight (BMI 1 25 kg/m2) and 4 (13%) were obese (BMI 1 30 kg/m2). Seventeen (57%) patients had a 45,X karyo-
type and 13 had other karyotypes (45X,46 Xi(Xq): n = 8; 45X,46XY: n = 2; 45X,46XX,46,Xi(Xq): n = 1; 45X,46,Xi
Characteristics of the Subjects and Treatment
(Xq),47 Xi(Xq): n = 1; 46X,delXp: n = 1). The education-
The auxological and clinical characteristics of the TS al level was rather high: 16 (53%) subjects had a diploma
patients are shown in table 1 . The age at evaluation was of higher secondary education and 10 (33%) had a college 22.1 8 2.4 years. Puberty was induced in 22 patients, or university degree, whereas only 3 (10%) patients had a whereas 8 patients reported to have developed puberty diploma of lower secondary education and 1 patient re-spontaneously; 4 of them, however, needed oestrogen ceived special education. No differences in clinical and treatment to have complete pubertal development; 4 pa-
auxological characteristics were observed among patients
tients had spontaneous menarche. Menarche occurred at
with induced or spontaneous puberty, neither among pa-
the age of 15.3 8 1.2 years in patients with EE2 treatment,
tients with the 45,X karyotype and those with other
and at respectively 11, 13, 13, and 15 years in those with karyotypes.
spontaneous menarche. Twenty-nine of the 30 women were currently taking oestrogens.
Psychosocial Functioning (Behavioural and Emotional
Final height was 152.6 8 4.4 cm (range: 145–161 cm).
All TS patients had a height below the population mean
The findings of the YASR are depicted in figure 1 A, B.
(166.5 cm) and 20 (67%) patients still had a height below Both TS patients and the non-TS reference group had the third percentile (155 cm). BMI was 24.1 8 5.9 kg/
mean problem scores within the normal range. TS wom-
Psychosocial Functioning of Young Adult
Fig. 1. a
Scores at the broad-band scales of
the YASR. The open bars represent the
scores of the non-TS females. * p ! 0.05.
Scores at the syndrome scales of the
YASR. The open bars represent the mean problem scores of the TS patients and the
black bars the mean problem scores of the non-TS females. * p ! 0.05.
en did not have more behavioural and emotional prob-
scores on several syndrome subscales and on the 3
lems compared to the non-TS females. On the contrary, broad-band scales. Significantly more TS patients com-TS patients had lower (less problematic) scores than the pared to the non-TS group had attention problem scores reference group on several subscales, i.e .
within the borderline range (7/30 (23%) vs. 2/44 (5%),
plaints (54.3 8 6.8 vs. 57.8 8 6.6, p = 0.03), thought prob-
lems (51.6 8 4.9 vs. 55.4 8 8.2, p = 0.02), and delinquent
When taking into account the karyotype, patients
behaviour (52.1 8 2.9 vs. 55.1 8 5.8, p = 0.01), as well as with 45,X karyotype had more problematic scores than on the broad-band externalizing scale (49.7 8 9.7 vs. 54.7
patients with other karyotypes only on the withdrawal
subscale (61.3 8 10.6 vs. 54.3 8 7.0, p = 0.049).
Table 2 shows that, when considering the number of
subjects with deviant behavioural problems scores, i.e.
YASR T-scores within the borderline or clinical range, a
The results of the SPP are shown in table 3 . TS patients
considerable number of TS patients, as well as of the
had scores comparable to those of the non-TS reference
non-TS reference group had problematic or pathological group on the majority of subscales, including intellectual
Number of subjects with problematic scores at the Young
Scores at the Self Perception Profile (SPP)
* p < 0.05; ** p < 0.02; *** p < 0.01.
Scores at the Bodily Attitude Scale (BAS)
Table 4 shows that with respect to body image as
measured by the BAS, TS patients’ bodily attitude scores were comparable to those of non-TS women for apprais-
al, attribution as well as for physical contact. On the BAS, no significant differences were found between pa-tients with the 45,X karyotype and those with other karyotypes.
ability, athletic competence, physical appearance and global self-worth. In comparison with the reference
Correlations between Test Scores and Auxological
group, TS women had lower scores for social acceptance
(2.7 8 0.7 vs. 3.0 8 0.6, p ! 0.05), romantic relationships
No associations were found between the YASR and
(2.1 8 0.7 vs. 2.5 8 0.6, p ! 0.05) and sense of humour SPP scores, and height or BMI. With respect to the BAS, (2.6 8 0.7 vs. 3.1 8 0.7, p ! 0.01), and higher scores for only a negative correlation was found between BMI and morality (3.2 8 0.4 vs. 3.0 8 0.4, p ! 0.02).
the appraisal score (r = –0.51; p ! 0.01). None of the test
Patients with the 45,X karyotype had lower scores scores was related to the ages at start of GH therapy, at
than those with other karyotypes for close friendship (2.7
start of oestrogen therapy or at menarche.
8 0.7 vs. 3.3 8 0.7, p ! 0.05), romantic relationships (1.9 8 0.6 vs. 2.4 8 0.7, p ! 0.05) and sense of humour (2.4 8 0.6 vs. 2.9 8 0.6, p ! 0.05).
Psychosocial Functioning of Young Adult
list in girls with TS, which remained unchanged during GH therapy  . The presence of attention problems in
This psychosocial evaluation of young adult women TS patients is a well-known feature already present at a
with TS reveals that these women have similar scores as very young age and being part of a larger cognitive-be-age-matched women without TS with respect to most of havioural profile. Attention problems can be associated the assessed domains. Although the TS patients in our with problems of hyperactivity, distractibility, clumsi-study had been treated during many years with GH and ness and/or poor school performance. Several authors re-oestrogens, they all had an adult height below the popula-
ported a TS specific neurocognitive phenotype consist-
tion mean, and two-thirds of the patients still had a height
ing of impaired visual-spatial and visual-perceptual abil-
below the third percentile. Hence, short stature does not ities, motor function, non-verbal memory, executive seem to affect psychosocial well-being at adulthood very function and attentional abilities [6, 27–31] .
Patients with the 45,X karyotype had more problem-
We decided to compare the data of our TS patient atic scores for withdrawal than patients with other karyo-
group with the data of an age-matched group of non-TS types. This might be due to the fact that patients with female students, which we consider as a reference group other karyotypes usually have fewer or less severe TS and not as a control group. Ideally, the results obtained in
symptoms and/or dysmorphic features impeding social
our TS group should be compared with data obtained in contact  . Further studies are needed to verify this hy-non-GH treated TS patients, but this was not feasible pothesis.
since we could not trace enough age-matched untreated
Regarding self-perception, TS patients had self-per-
TS patients to serve as a control group. We acknowledge ception scores comparable to the reference group on the that this is a shortcoming of the present study. As a con-
majority of subscales except for social acceptance, ro-
sequence, our results can only be extended to TS patients
mantic relations and sense of humour, globally in line
who have been diagnosed rather early in life and have with the Dutch results  , but somewhat in contrast to been treated with GH and oestrogens.
other previous studies reporting impaired self-esteem
With respect to the presence of behavioural and emo-
and self-confidence [4, 6, 27, 29] . Problems in social rela-
tional problems, TS patients had similar scores as the ref-
tionships, particularly in contact with the opposite sex, as
erence group on the majority of syndrome subscales and reflected by a lesser number of intimate relationships or even less problematic scores on some subscales (somatic marriages, later psychosexual milestones, problems with complaints, thought problems, delinquent behaviour) social cues have repeatedly been reported [4–9, 18] . Our and on the broad-band externalizing scale. Our findings TS patient group had a lower sense of humour, which are globally in line with the YASR data reported by van could be understood within the context of a lack of self-Pareren et al.
revealing comparable behavioural confidence in social relations as well as a lack of good
problem scores between TS women and the general pop-
understanding of social, especially affective, cues  .
ulation. However, their results were only based upon the Our TS patients had higher scores for morality than the broad-band scales. It is somewhat surprising that our TS non-TS reference group. These higher morality scores patients had lower scores on the somatic complaints sub-
could be seen as a way of compensation or coping, attach-
scale than the non-TS reference group as TS patients are ing more importance to or accentuating internal values often confronted with TS specific health problems [2, 3] . rather than external values such as physical appearance. This may be due to the fact that the YASR somatic com-
Among the TS group, patients with the 45,X karyotype
plaints subscale does not measure TS specific somatic had lower self-perception scores with respect to social problems such as hearing problems, cardiac disease, hy-
contact and sense of humour than TS patients with other
pothyroidism or hypertension. In addition, TS patients karyotypes. A similar interpretation as mentioned above have experience in adapting to these health problems, could be applied, i.e. patients with a 45,X karyotype hav-and probably tend to complain less about their body than
ing more pronounced TS symptoms  could experi-
ence more problems in social relations.
Significantly more TS patients than reference women
The satisfaction of the TS patients with body image
received scores on the YASR attention problems subscale and/or female appearance was not different from that of within the borderline and clinical range. In line with oth-
the non-TS females, with similar bodily attitudes scores
ers  , we previously reported elevated scores on the at-
for appraisal, attribution as well as physical contact on the
tention problems subscale of the Child Behaviour Check-
BAS, and comparable scores on the physical appearance
subscale of the SPP. The Dutch study reported a more ings such as different treatment regimens (late pubertal negative body image in the TS group compared to the induction, no oestrogen treatment at adult age), the choice population sample, but BAS attribution and physical con-
of the studied somatic and psychological parameters, and
tact scores were only slightly lower  . We suspect that age differences of the studied TS patients impeding a val-the non-problematic bodily attitudes scores in our TS id comparison of data from different publications.
group could be due to defence and/or coping mechanisms such as a tendency of minimization and denial of prob-lems with respect to physical appearance or femininity.
Since the TS specific physical characteristics and/or dys-morphic features cannot be treated or changed essential-
In conclusion, our findings revealed that young adult
ly, a way of coping, a way of adapting to this reality is try-
patients with TS, diagnosed at an early age and treated
ing to minimize the importance of physical appearance during childhood with GH and oestrogens if indicated, and to accentuate the importance of other aspects in did not report more behavioural and emotional problems life.
compared to age-matched non-TS women except atten-
When analyzing the potential somatic and clinical tion problems. TS patients even reported fewer problems
correlates of the psychological test scores in the TS pa-
than non-TS females at some subscales. Although TS may
tient group BMI, reflecting obesity, a common problem have an important impact on physical appearance, TS pa-in adult TS [2, 33] , was a more important determinant tients did not differ from non-TS women in their bodily than height. Bodily attitudes scores on the appraisal sub-
satisfaction. In line with previous studies, TS patients
scale of the BAS were inversely related to BMI, i.e. lower perceived themselves as less socially competent, particu-bodily appraisal scores were related to a higher BMI. Oth-
larly patients with a 45,X karyotype. BMI was the only
er clinical determinants such as height, age at start of GH
somatic correlate related to the bodily attitudes scores.
therapy, age at start of oestrogen therapy and age at men-
The medical and psychological follow-up of adult TS pa-
arche were not related to any of the test scores.
tients should not neglect the problem of overweight and
Our findings are in line with those from other studies associated psychosocial consequences.
demonstrating that height does not have a significant im-pact on psychological functioning [9, 13, 17, 18] . For oth-er somatic correlates discrepant results were found. In
contrast to our findings, Wide-Boman et al.  reported that BMI was not related to psychological well-being.
This work was supported by a research grant from the Belgian
Some studies demonstrated that late induction of puber-
Study Group for Paediatric Endocrinology. The authors express
ty and health problems associated with TS (hearing im-
their thanks to all the members of the BSGPE who invited patients to participate to this study. Part of this work was previously pub-
pairment and cardiac problems) had a negative impact on
lished in abstract form [Horm Res 2005;64(suppl 1):211] and was
quality of life or well-being [17, 18] . A complex interplay presented as a mini poster at the ESPE/LWPES 7th Joint Meeting of factors is probably responsible for these divergent find-
Paediatric Endocrinology, Lyon, september 2005.
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motor function in girls with Turner’s syn-
Turner’s syndrome in adulthood. Endocr
1. The average time it takes for a person to experience pain relief from aspirin is 25 minutes. A new ingredient is added to help speed up relief. Let µ denote the average time to obtain pain relief with the new product. An experiment is conducted to verify if the new product is better. What are the null and alternative hypotheses? (a) H 0 : µ = 25 vs. Ha : µ 25 (GO TO 2) (b) H
Fact Sheet Caregiving and Depression mental health professional ma help to prevent the Could the sadness, loneliness or anger you feel development of a more serious depression over time. today be a warning sign of depression? It’s possible. It is not unusual for caregivers to develop mild or Symptoms of Depression more serious depression as a result of the constant