Key words Müllerian agenesis - PCOS (polycystic ovary syndrome) - CAIS (complete androgen insensitivity
syndrome) - support group
Schlüsselwörter MRKH-Syndrom (Mayer-Rokitansky-Küster-Hauser) - PCOS (polyzystisches Ovarialsyndrom)
- CAIS (komplette Androgenresistenz) - Selbsthilfe
Introduction
Persons with different sexual characteristics often report a feeling of being “different
from others” or of “not being normal”. This has been described both for individuals
with different primary sexual characteristics and for those with different secondary
sexual characteristics [1 ], [2 ], [3 ]. In such cases, contact to other affected individuals or attending support groups
can be experienced as supportive and de-stigmatising [4 ]. However, the authors of this study did not find any published reports on how individuals
with complete androgen insensitivity syndrome (CAIS), Mayer-Rokitansky-Küster-Hauser
syndrome (MRKHS) or polycystic ovary syndrome (PCOS) experience contact to other affected
individuals, what they find to be helpful, and whether some individuals felt that
such contact also had negative aspects.
Description of the syndromes investigated
The investigated diagnostic groups consisted of syndromes which result in changes
to female sexual characteristics or female sexual development and which are accompanied
by infertility (CAIS and MRKHS) or reduced fertility (PCOS).
CAIS and MRKHS are rare disorders and are classified as belonging to the group of
disorders of sex development, DSD [5 ], [6 ].
The prevalence of CAIS is reported to be approximately 1 in 20 000 live births with
an 46,XY karyotype [7 ]. Despite the 46,XY karyotype, persons with CAIS are phenotypically female with external
female genitalia due to complete insensitivity of cells to androgens. As the testes
develop and release anti-Müllerian hormone, the uterus does not develop and the vagina
ends blindly in a pouch. The diagnosis is frequently made in puberty because of primary
amenorrhoea; breasts develop normally but pubic and underarm hair is either lacking
or reduced [8 ]. The sexual identity of persons with CAIS is usually described as unambiguously
female [8 ], [9 ], however there are also other reports in the literature indicating an experienced
sexual identity that differs from that of other women [10 ], [11 ]. Personal contacts with
affected persons (when preparing the study) also showed that some did not feel the
term “woman” to be a suitable description, which is why we have used gender-neutral
terms to describe this group in our study.
MRKHS or Müllerian agenesis occurs in around 1 of 5000 female births [12 ]. It is characterised by agenesis of the uterus and vagina in women with an XX karyotype
and is presumably of polygenetic origin. The ovaries are fully developed and functional
[13 ]. Due to the undeveloped or extremely shortened vagina, vaginal sexual intercourse
without previous medical treatment is not possible in most cases.
PCOS is one of the most common endocrine disorders in women of reproductive age; reports
on its prevalence range from 5 to 17.8 % (using the Rotterdam criteria), depending
on the diagnostic criteria [14 ], [15 ]. In addition to hyperandrogenism and oligorrhoea or amenorrhoea, other external
characteristics include acne, obesity, hirsutism and alopecia [16 ]. Some women with PCOS show a “masculinisation” of their external appearance.
What all of these diagnostic groups have in common are changes to certain female sexual
characteristics or to their sexual development. However, the syndromes differ considerably
with respect to the impact they have on the individual affected, particularly with
regard to the wish to have children (persons with CAIS cannot have their own genetic
children; in some countries, women with MRKHS may have genetic children if they enlist
the help of a surrogate mother; women with PCOS are usually subfertile but pregnancy
is possible for some women with PCOS). The widely differing prevalence of the syndromes
(CAIS and MRKHS are rare disorders, PCOS is relatively widespread) also needs to be
taken into account when considering the results of this study.
Contact to other affected individuals
Contact to other affected persons can take very different forms. In addition to individual
personal contacts, e.g. during a stay in hospital, individuals may also be in contact
with a group (group therapy, various support groups; cf. also [17 ], [18 ]). Contacts increasingly also come about via the internet, in the form of private
e-mail contacts or discussion forums (cf. [19 ], [20 ]).
The central task of self-help groups has been described as the provision of social
and emotional support as well as the provision of information [21 ].
Overall, contact to self-help/support groups has been experienced differently; some
people find it helpful, others reject it [2 ], [22 ], [37 ].
For people with CAIS and PCOS it has been reported that the experience of “otherness”
or isolation associated with the diagnosis can be reduced through contact to other
affected persons [2 ], [21 ]. For intersex persons in particular, the importance of meeting “similar others”
for the development of a positive identity was emphasised [23 ]. Our search of the literature did not find any comparable studies on self-help groups
or contacts to other affected persons for persons with MRKHS. However, results obtained
from an interdisciplinary Patient Day indicated that women with MRKHS also judged
the exchange of experiences with other affected persons to be positive [20 ].
As providing information above and beyond the respective individual diagnosis is also
an important task of support groups, this study also looks at how participants evaluated
the information on various aspects of the disorder that was provided by their treating
physician and whether this was related to their contact to other affected persons.
Psychological distress
There are a number of studies on psychological distress in persons with CAIS, MRKHS
and PCOS, some of which point to increased levels of distress. However, the results
are somewhat contradictory [24 ]. Studies have reported increased psychological distress but also completely unremarkable
levels of distress in individual groups (CAIS: [25 ], [26 ], MRKHS: [27 ], [28 ], [29 ], PCOS [30 ], [31 ]).
Individual results appear to indicate that persons in support groups cannot be taken
as representative for the patient group as a whole [32 ]. One of the hypotheses for this is that persons with particularly high levels of
distress are more likely to seek contact to other affected individuals or support
groups. To assess this more precisely, our study recorded the levels of psychological
distress of the persons taking part in the study. The study then examined whether
there was any correlation between the level of psychological distress and contact
to other affected persons (as well as how this contact was rated).
Aim of this study
This study aimed to examine contact to other affected persons with CAIS, MRKHS and
PCOS and its possible correlation with psychological distress. In addition to the
frequency of such contacts for the individual diagnostic groups, the study also aimed
to investigate whether such contacts were considered helpful and whether contact to
others was desired. Accordingly, the following null hypotheses were proposed for the
study: the 3 diagnostic groups do not differ from each other with regard to levels
of psychological distress, frequency of contact, the assessment of this contact and
the desire for contact. The frequency of contact to other affected persons, the assessment
of this contact and the desire for contact are not related to the level of psychological
distress; the information received from the treating physician also has no impact
on these variables.
Method
Data collection and participants
Data was collected between March 2010 and July 2011 as part of a research project
on “Androgens quality of life and femininity in persons with CAIS, MRKHS and PCOS”
at the University Clinic Hamburg-Eppendorf (supported by the Else Kröner Fresenius
Stiftung; project leader: Prof. Dr. Richter-Appelt; the project was approved by the
Ethics Commission of the local medical association). Participants (minimum age: 18
years) were recruited from all over Germany through their physicians and the hospitals
where they received treatment (primarily through the Gynaecological University Hospital
of Tübingen), through support groups (primarily the support group “Intersexuelle Menschen
e. V.”), through the projectʼs website and with the help of appeals launched in professional
journals. Participation consisted of filling out a questionnaire which had been compiled
from standardised instruments and answering questions which the investigators developed
themselves however space was
also provided for the participantsʼ own comments. Confirmation of the respective diagnoses
was conducted in 2 stages. The data given in the questionnaires was checked for plausibility
and for its agreement with the diagnosis participants stated they had received. In
addition, the participantʼs physician was contacted and asked to confirm the diagnosis
as well as to provide additional medical findings (the precondition for this was that
participants authorised their physician to release their medical records). Questionnaires
which could not be clearly classified into one of the diagnostic groups were excluded
from the analysis. Questionnaires which had been filled out but did not include the
participantʼs consent to take part in the study were also excluded.
For a better comparison of certain variables (relationship status, occupation), additional
data from a non-clinical sample (n = 932 women) was also reviewed; this data had been
collected in the form of an online survey conducted as part of the research project.
Questionnaire
Information about contact to other affected persons was collected using questions
developed by the researchers themselves. Recorded data included information as to
whether such contacts had taken place, whether the contact was considered helpful
and – if there had been no contact – whether the participant desired such contact.
The last two questions included space for comments by the participants themselves.
Participants were also asked whether they felt that they had been given sufficient
information by their treating physician about the diagnostics used, treatment, the
consequences of the diagnosis and long-term consequences. An index (0–4) was compiled
from the answers to these 4 points which showed how well the study participants felt
they had been informed overall (0 = not sufficiently on any point; 4 = well informed
on all points).
Psychological distress was assessed using the Brief Symptom Inventory (BSI, German
version [33 ]). The questionnaire recorded individual psychological distress over the past week
using 53 different items. The participant rated the extent to which they felt impaired
by various medical conditions on a 5-point Likert scale (0 = not at all; 4 = very
strongly). The answers were summarised in 9 scales (Somatisation, Obsessive-Compulsive,
Interpersonal Sensitivity, Depression, Anxiety, Hostility, Phobic Anxiety, Paranoid
Ideation, Psychoticism). In addition, an overall score was calculated which reflected
the overall level of psychological distress.
The resulting values were converted into standardised T-values using the standard
values given in the manual [33 ], which permitted a direct comparison to be made with the non-clinical reference
samples (mean T-value for distribution is 50, standard deviation is 10. A value of
50 in a patient sample would thus correspond exactly to the mean value of the non-clinical
reference group; a value of 70 would be 2 standard deviations above the mean value
of the reference group and would thus point to markedly higher levels of distress).
In accordance with the BSI manual, persons who had T-values of 63 or more in their
overall score or in at least 2 subscales were classified as “clinical cases” with
conspicuously higher levels of psychological distress.
Statistical analysis
Statistical analysis was conducted using the PASW 18 software (SPSS). χ2 -tests for 2 × 2 and 3 × 2 cross-classified tables were used to calculate the relationship
between categorical variables. If statistical requirements were not met (expected
frequency of individual cells < 5), exact χ2 -tests (Fisher-Yates test) were used for calculation. Differences between groups were
compared using Mann-Whitney U-test.
The results of the Brief Symptom Inventory (BSI) were recorded as standardised T-values,
which were calculated based on the reference data of the test manual [35 ] (mean distribution of the T-values is 50, standard deviation is 10).
Any additional comments by participants were assessed qualitatively. Frequencies of
the issues mentioned were calculated.
Results
Description of samples
A total of 126 questionnaires were completed, of which 5 had to be excluded due to
formal faults (no consent given for participation in the study). Six more questionnaires
were excluded due to missing or inappropriate diagnosis (no clear diagnosis of CAIS,
MRKHS or PCOS). Thus, the data of 115 persons was included in the final evaluation
(11 persons with CAIS; 49 women with MRKHS; 55 women with PCOS).
The study participants heard about the research project from a number of different
sources. In the group with CAIS, the majority (63.6 %; 7/11) heard about it through
their support groups. In the MRKHS group, this proportion was distinctly lower (2 %;
1/49), most persons in this group (65.3 %; 32 of 49) were advised of the study by
the hospital they attended or by their general practitioner. In the PCOS group, 78.2 %
(43/55) heard about the study from the hospital they attended or from the physician
treating them on an outpatient basis; only one person (1.8 %) was informed about the
study by her support group. A detailed summary is given in [Table 1 ].
Table 1 How did study participants hear about the research project?
How did you hear of our study?
CAIS (n = 11)
MRKHS (n = 49)
PCOS (n = 55)
N
%
N
%
n
%
* 25 women with MRKHS (51 %) heard of the study through their treatment at Tübingen
University Hospital.
University clinic/specialised clinic/centre
1
9.1 %
28
57.1 %*
31
56.4 %
Gynaecologist
–
–
2
4.1 %
5
9.1 %
Another physician
–
–
2
4.1 %
7
12.7 %
Support group
7
63.6 %
1
2.0 %
1
1.8 %
Another affected person
1
9.1 %
2
4.1 %
1
1.8 %
Internet
1
9.1 %
6
12.2 %
2
3.6 %
Other
1
9.1 %
–
–
1
1.8 %
Not specified
–
–
8
16.3 %
7
12.7 %
The average age of participants with CAIS was 38.7 years (± 9.6); the average age
of participants with MRKHS was 23.6 years (± 5.8) and that of participants with PCOS
was 29.1 years (± 4.2).
At the time of the study, 54.5 % of the participants with CAIS were living with a
partner (5 with a male partner, 1 with a female partner). In the MRKHS group, 75.5 %
were in a relationship, and 87.3 % of the PCOS group were in a relationship. The proportion
of persons living with a partner differed significantly between the diagnostic groups
(Fisher-Yates test: p-value = 0.037) (reference value for the non-clinical sample:
73 % living with a partner).
All participants with CAIS were either working or in education or training. In the
MRKHS group, 45 persons (91.8 %) were either working or in education or training (or
at school/university), 4 (8.2 %) were unemployed. Of the participants with PCOS, 40
(72.7 %) were working or in education or training, 7 women (12.7 %) were on maternity
leave or had taken parental leave, 3 (5.5 %) described themselves as housewives, 4
persons (7.3 %) were unemployed, and 1 (1.8 %) was incapable of working (reference
value for the non-clinical sample: 71.5 % employed or in education or training; 3.3 %
unemployed; 2.5 % incapable of working or had taken early retirement for invalidity;
22.6 % other).
Psychological distress
In accordance with the definition of the BSI for “clinical levels of psychological
distress”, 6 persons (54.5 %) in the study group with CAIS were classified as suffering
from higher levels of psychological distress. In the MRKHS group the BSI could only
be evaluated for 48 participants; of these 48 participants, 26 (54.2 %) were considered
to have higher levels of psychological distress. In the PCOS group, 29 persons (52.7 %)
showed higher levels of psychological distress. The distribution between the diagnostic
groups did not differ significantly (χ2 = 0.027; df = 2; p-value = 0.987).
The results for the overall BSI value (GSI, Global Severity Index) and the 9 subscales
for each diagnostic group are given in [Table 2 ].
Table 2 BSI results (T-valuesa ) for each diagnostic group.
CAIS (n = 11)
MRKHS (n = 48b )
PCOS (n = 55)
MW
(SD)
MW
(SD)
MW
(SD)
a Mean = 50, standard deviation = 10b The BSI results could not be calculated for one of the persons with MRKHS due to
a lack of data.c Participants (number and percentage), who fulfilled the criteria for “clinically
relevant level of psychological distress”: GSI T-value ≥ 63, or T-value ≥ 63 in 2 or more subscales).
Overall GSI score
60.91
(10.30)
57.25
(14.15)
59.04
(12.98)
Subscales
Somatisation
56.73
(11.16)
52.90
(10.66)
54.35
(11.11)
Obsessive-Compulsive
59.36
(9.78)
54.21
(11.82)
55.84
(10.76)
Interpersonal Sensitivity
61.64
(10.46)
58.06
(11.48)
59.65
(11.09)
Depression
60.36
(12.38)
55.56
(13.18)
58.98
(12.49)
Anxiety
57.27
(11.22)
53.33
(12.31)
53.95
(11.76)
Hostility
60.09
(11.20)
59.50
(10.83)
59.87
(9.85)
Phobic Anxiety
56.64
(12.46)
54.31
(11.42)
53.02
(10.90)
Paranoid Ideation
54.00
(11.14)
57.29
(11.52)
56.85
(15.56)
Psychoticism
59.91
(11.31)
56.63
(12.02)
56.16
(10.89)
Clinical casesc
n
(%)
n
(%)
n
(%)
6
54.5 %
26
54.2 %
29
52.7 %
Contact to other affected persons
All of the study participants with CAIS reported that they were in contact with other
affected persons. In the MRKHS group, only 48 women answered this question; of these,
30 (62.5 %) had contact to other affected persons. Among the group with PCOS only
6 persons (10.9 %) were in contact with other affected women. The proportion of people
with contact to other affected persons depended significantly on the respective diagnosis
(Fisher-Yates test: p-value<0.001)
Was contact to others rated as helpful?
When study participants did have contact to other affected persons, the majority (87.2 %)
of the participants experienced such contact as helpful. Of the participants with
CAIS who had contact to other affected persons, 81.8 % (9/11) experienced this contact
as helpful. In the group with MKRHS the percentage of women who felt contact was helpful
was 90 % (27/30); in the group with PCOS this figure was 83.3 % (5/6). The percentage
did not correlate significantly with the individual diagnosis (Fisher-Yates test:
p-value = 0.596).
A total of 41 participants in the study included additional comments on how they assessed
contact with other affected persons. People who rated contact as helpful most frequently
cited the experience of not being alone (13 mentions) as their reason for such contact.
The exchange of experience and information on the diagnosis were also listed as important
(6 mentions), followed by psychological support and the greater understanding they
received from other affected persons who had had similar experiences (5 mentions each).
Other comments on the benefits of contact included being able to talk openly about
the diagnosis (1 mention) and being able to let oneʼs guard down in a group of other
persons with the diagnosis (1 mention).
If the contact was not rated as beneficial, the reasons for this, cited in 3 cases,
included the focus on negative topics in the support group, while 2 cases experienced
contacts to other affected persons as stressful. One person with CAIS reported a feeling
of “not belonging” and of exclusion even when meeting other persons with a diagnosis
of CAIS. Other expectations and practical reasons (distance from the place of residence)
were also cited as explanations for why the contact was not felt to be helpful.
Typical comments for the different categories are listed in [Table 3 ].
Table 3 Reasons given for the respective answer: contact helpful yes/no.
Contact to other affected persons …
Category
Typical comment (original text)
Was helpful because:
Feeling of not being alone
“You find out that you are not alone with the diagnosis”
Similar experience/more understanding
“You have someone who has gone through the same things as you have yourself. That
is incredibly helpful.”
Exchange of experience/information
“Was able to find out about surgery from them”
Psychological support
“I felt/feel it to be a place where someone is there for me on bad days.”
Changed the way of dealing with the diagnosis
“Thanks to the forum I can now talk more openly about it”
Able to show weakness
“You realise that you can sometimes allow yourself to show weakness”
Was
not
helpful because:
The focus was on negative aspects
“However, I feel one should not only occupy oneself with the diagnosis and the problems.
At one stage this ʼcontinual rehashing of problemsʼ with other affected persons got
to be too much for me.”
Contact is stressful
“At the moment it is more stressful, because I experience it as very inflexible and
it does not help me progress further”
Had other expectations
“I was too unrealistic”
Practical reasons
“Unfortunately because of the distance, benefit only limited”
Do people wish for contact with other affected persons?
Individuals who had no contact with other affected persons were asked whether they
wished for such contact. In the group with MRKHS 52.9 % (9 of 17) wished for such
contact, in the PCOS group 39.6 % (19 of 48) wished for such contact. This distribution
did not differ significantly between the 3 diagnostic groups (Fisher-Yates test: p-value = 0.561).
Thirty persons appended additional comments regarding their wish for contact to other
affected persons. If contact was desired, 9 persons stated their reason as wanting
to hear about othersʼ experience of the diagnosis and wishing to exchange experiences.
One woman with PCOS hoped to obtain more information about her diagnosis.
Persons who did not wish for contact to other affected persons usually stated that
they did not feel it to be necessary for them (6 mentions). Three women rejected such
contact because they did not want to be reminded of their diagnosis. Two women with
MRKHS did not want any contact because they considered this would be too distressing
and were afraid that discussions would focus too much on negative topics.
[Table 4 ] gives a summary of typical comments on the respective topics.
Table 4 Reasons given for the respective answer: contact desired yes/no.
Contact to other affected persons …
Category
Typical comment
Is desired because:
Hear about other experiences/exchange
“Would like to hear about othersʼ experience with it and how they deal with it”
More information
“Want to find out more about this disease”
Is
not
desired because:
Not felt to be necessary
“I can deal with it quite well”
Contact experienced as stressful
“Contact would be too stressful, real.”
Do not want to think about diagnosis
Partly try not to think about it”
Correlation between contacts to other affected persons and psychological distress
In the overall group, no significant correlation was found between higher levels of
psychological distress (based on the criteria for “clinical cases”) and contact to
other affected persons (analysis of the 3 diagnostic groups taken together; χ2 = 0.365; df = 1; p-value = 0.546). This also applied when the diagnostic groups were
analysed separately (CAIS: no calculation, all individuals have contact to others;
MRKHS: χ2 = 0.065; df = 1; p-value = 0.798; PCOS: Fisher-Yates test: p-value = 0.197).
Similarly, in the group which had contact to other affected persons, no correlation
was found between the level of psychological distress and evaluation of the contact
as helpful (analysis of all 3 diagnostic groups taken together; χ2 = 3.375; df = 2; p-value = 0.185).
This also applied to the individual diagnostic groups (CAIS: Fisher-Yates test: p-value = 1.000;
MRKHS: Fisher-Yates test: p-value = 0.483; PCOS: Fisher-Yates test: p-value = 1.000).
For the overall group, the wish to have contact with other affected persons depended
significantly on the level of psychological distress (Fisher-Yates test: p-value = 0.024).
In the group of clinically distressed persons 57.6 % (19/33) wished for contact, while
the wish for contact with other affected persons was distinctly lower in the group
with clinically unremarkable levels of distress (28.1 %; 9/32). The following correlations
were found for the different diagnostic groups. This calculation was not made for
the CAIS group as all persons in the group had contacts to other affected persons.
There was no correlation in the group with MRKHS (Fisher-Yates test: p-value = 1.000).
In the group with PCOS the level of psychological distress was found to correlate
significantly with the wish for contact to other affected persons (Fisher-Yates test:
p-value = 0.007). Of the women with clinically significant levels of psychological
stress (“clinical cases”), 60.9 % (14/23)
wished to have contact to other affected persons, while in the group without clinically
relevant levels of distress, only 20 % (5/25) expressed this wish.
A summary of the results is given in [Fig. 1 ].
Fig. 1 Summarised representation of contact to other affected persons. Each column on the
left shows the distribution of answers in the group with no clinical signs of psychological
distress, while the column on the right shows the results for the group with clinical
signs of psychological distress. The respective share for every answer is shown: 1.
Individuals had contact to others and this contact was considered helpful (green);
2. Individuals had contact to others and this contact was considered not helpful (yellow); 3. Individuals have no contact but would like contact (orange);
4. Individuals have no contact but do not wish for contact (red); 5. Not specified or ambivalent answers (grey). The graph
shows that the proportion of the group with contact to others (green and yellow areas)
differs considerably between the 3 diagnostic groups. Most persons who had contacts
to others considered them to be helpful (green area). The wish to have
contact was expressed most often by women with a diagnosis of PCOS and clinical signs
of psychological distress (column on the right, red area).
Information obtained from the treating physician
How well individuals felt that their physician had informed them differed significantly
between the diagnostic groups (Fisher-Yates test: p-value < 0.001). Overall, the extent
of information provided by the physician was rated highest by the MRKHS group (median = 4),
and lowest by the CAIS group (median = 0). With a median value of 3, the rating given
by the PCOS group was between that of the 2 other groups.
No difference was found between persons who had contact to other affected persons
and those who did not with regard to their statements about the extent of the information
provided by their physician (U = 1442; p-value = 0.426). There was also no difference
with regard to the information provided by their physician between persons who considered
contact to be helpful and those who did not (U = 43.5; p-value = 0.112; however it
should be noted here that the group of persons who did not consider contact to be
helpful was very small, with only 4 persons). Similarly, no difference in the assessment
of the extent to which information was provided by the physician was found between
the group who wished for contact compared to the group of persons who did not (U = 478.5;
p-value = 0.718).
The level of psychological distress (BSI overall value) did also not correlate with
the patientsʼ assessment of the extent of information provided by their physicians.
Discussion
Central results and comparison with other studies
The null hypotheses formulated at the beginning regarding the level of psychological
distress, the assessment of contact to affected persons as being helpful, and the
wish to have contacts to other affected persons were not rejected by the results of
the study. The 3 diagnostic groups did not differ with regard to these variables.
The null hypothesis regarding the frequency of contact must be rejected as the 3 groups
differed significantly from each other. In the sample investigated, all persons with
CAIS had contacts to other affected persons with CAIS, in the group of women with
MRKHS the majority reported being in contact with other persons with MRKHS while,
in comparison, in the group of women with PCOS a much lower proportion of women reported
being in contact with other women with PCOS. Of the 3 diagnostic groups investigated,
PCOS is the most common; overall more women are diagnosed with this syndrome. Given
the fact that the incidence of women with PCOS is
higher, one could conjecture that women with PCOS would also have more opportunity
to establish contact to other affected women or to join support groups, as the choices
available for this fairly common diagnosis are greater. The existing data however
tell a rather different story. When interpreting the results it must be borne in mind
that the participants recruited heard about the study in many different ways. Persons
with CAIS were primarily informed about the study through the offices of support groups
for XY women; accordingly, they were the group which most frequently cited having
contact to other affected persons. We can safely assume that the results for the group
with CAIS are not representative and that the percentage of persons with contact to
other persons with the same diagnosis is probably overestimated based on the sample
of people in our study. To compare these results, we looked at the Hamburg study on
intersexuality; in this study 50 % of
participants reported that they had some experience of support groups [34 ]. For women with PCOS another study cited a figure of 51.5 % (17/33) of persons who
had described attending a support group [31 ]. Since in our study, participants were partly recruited through the nationwide network
of PCOS support groups, it can be assumed that the percentage of women with PCOS who
attend support groups was overestimated. The percentage of persons with a particular
diagnosis who have contact to other affected persons or to support groups would have
to be investigated in larger, more representative studies. The ways in which potential
study participants are recruited play a very important role.
A correlation between the level of psychological distress and the desire to have contact
with other affected persons was only found in the group with PCOS; the desire to have
contact was voiced more frequently by women with higher levels of psychological distress.
There were no differences between the diagnostic groups with regard to the frequency
of such contact or the assessment of contact.
In the sample investigated, there was no indication that primarily persons with particularly
high levels of psychological distress sought contact to other persons with the same
diagnosis or to support groups. This is in accordance with the results of Jauca et
al. [31 ] who, in their study of 33 women with PCOS, also did not report any significant differences
with regard to the level of psychological distress between persons who attended support
groups and those who did not.
The diagnostic groups did differ in their assessment of the information provided by
their attending physician. The group of women with MRKHS reported the highest levels
of satisfaction with the extent of information they had received, followed by the
group of women with PCOS; persons with CAIS reported the lowest level. No correlation
was found between the extent of information received from their attending physician
and the frequency of contact, the assessment of such contact, or the desire for contact.
Using the examples ‘androgen insensitivity’ and ‘adrenogenital syndrome’ (AGS, one
of a group of intersex conditions) Warne reported that support groups went through
various phases during which the focus of the group changed (from obtaining information
and supporting other members of the group to lobbying for social change) [35 ]. In our study, contact to other affected persons were investigated in a more general
sense (not only contact to support groups), and it was therefore not possible to make
any statements about changing phases within groups. It is conceivable however that
the backdrop to the assessment of contacts as “not helpful” by individual persons
could be a lack of fit between the needs of the individual and the current phase of
the group.
In this context it is important to point out again the differences between the 3 diagnostic
groups investigated, particularly with respect to the potential to have genetic children
of oneʼs own. Women with PCOS can become pregnant, whereas persons with CAIS and MRKHS
cannot. Depending on the phase of life they are in, becoming pregnant and the medical
support they may require could play a key role for women with PCOS. This would probably
also be reflected in the topics participants put forward for discussion in support
groups. Which topics individual persons in support groups wish to talk about and which
areas they wish to learn more about were not investigated in this study. Future studies
could investigate which topics persons in support groups wish (or do not wish) to
talk about and which topics they experience as particularly helpful, depending on
their current circumstances, for the different diagnostic groups.
In a report which included her own experiences, an affected physician described her
contacts to an AGS action group of affected persons and emphasised the importance
of the different generations within the support group [36 ]. Medical advances and changes in the problems experienced by patients mean that
it is necessary to look at self-help groups over 10 to 20-year periods [36 ]. This aspect was not included in our study and could be investigated in more detail
in subsequent studies: Did the participant have any contact to persons of similar
age or going through the same stage of treatment? Was contact to “experienced” persons
with the same diagnosis felt to be particularly helpful? Or could the experiences
of older affected persons not be compared to the individualʼs own situation because
the treatment available had changed?
The reasons given for the assessment of contact to others listed above more or less
correspond to the range of topics described in a qualitative study on support groups
for PCOS [21 ] with the difference that, in our study, negative aspects were also mentioned (e.g.
the stress experienced through contact to other affected persons).
Overall, it was noticeable that the percentage of persons with higher levels of psychological
distress in all 3 diagnostic groups was more than 50 % and thus relatively high. These
figures correspond to the results of the Hamburg Intersex Study [25 ]; in the Hamburg study, 59 % of participants fulfilled the criteria for clinically
relevant levels of distress. Jauca et al. described similar results for women with
PCOS [31 ]; they reported a figure of 42.4 % for persons with clinically higher levels of psychological
distress. This once again highlights the importance of ancillary diagnostic investigations
in persons with different sexual characteristics as these could help identify patients
with high levels of psychological distress and provide them with support or therapy.
Limitations
As already discussed above, the manner in which participants were contacted and recruited
into the study plays an important role for the interpretation of our results. Most
of the participants with CAIS heard about the study through a support group; we therefore
have to assume that their results regarding the frequency of contact with other affected
persons are skewed. Due to the way in which we chose to recruit participants into
the study (not a random sample, only persons interested in taking part in the study
were included), we cannot assume that the results are representative for any of the
3 groups. The results reflect the perceptions of some persons with these diagnoses
and should not be generalised. They offer a first insight into the subject matter
and provide starting points for further investigations.
The differentiated examination of the proband sample resulted in a number of subgroups,
some of which were quite small. Thus, despite the initially large sample group, this
may have resulted in correlations between individual variables not being visible in
the results.
This study did not investigate the differences between the 3 syndromes in detail;
in particular, the differing importance for affected persons was not examined. Only
one specific aspect was highlighted here. The data described here gives a first overview
of contact to other affected persons with CAIS, MRKHS and PCOS. To assess individual
impact factors in more detail and potentially make recommendations for different patient
groups, the following points need to be investigated in further studies: In which
context did contact take place? For how long? With regard to support groups: How was
the group organised? What goals did the group have? What were the expectations on
joining a support group?
Conclusion
The majority of participants in our study reported that contact to other affected
persons was helpful; however, no correlation between contacts and the level of psychological
distress was found in any of the diagnostic groups. The current data do not permit
any recommendations to be made on which persons would benefit from contact to a support
group and which would not. To formulate any such recommendation a further study would
be necessary to differentiate between the types of contact and examine them in connection
with the specific characteristics of affected persons.
It should be noted, however, that among the women with PCOS, those with clinically
relevant levels of psychological distress more frequently expressed the wish to have
contact with other affected persons. This group could benefit from information on
what services are available. However, it should be noted that some participants also
reported negative experiences associated with contacts to other affected persons.
This issue also needs to be broached when counselling patients.
Practical implications
Some women with different sexual characteristics report that they benefit from contact
to other affected individuals. In particular women with PCOS and clinically relevant
levels of psychological distress expressed more frequently the wish to have contact
to others. For this group, it could be beneficial to address the topic of self-help
groups during treatment and to advise patients of suitable groups available to them.
Acknowledgement
We would like to thank everyone who was involved in the study; our particular thanks
go to the people who participated in the study who were prepared to share their experiences
with us.
We would like to thank the Else Kröner Fresenius Stiftung for financially supporting
this research project.
