Standardized instruments for assessing psychiatric, behavioral, sexual problems, and quality of life in male and female adults with disorders of sex development in Indonesia

Introduction

Disorders of Sex Development (DSD) are a group of congenital medical conditions defined as atypical development of chromosomal, gonadal, and anatomical sex.¹ Pathophysiologically, DSD are divided into two main groups; 1) Anomalies in sex determination caused by chromosomal and gonadal anomalies, 2) Anomalies in sex differentiation caused by the anomalies in enzyme and hormones.² Based on the karyotype, DSD can be divided into 46-XY DSD, 46-XX DSD, and chromosomal DSD. The 46-XY DSD includes gonadal dysgenesis, androgen biosynthesis defect, androgen insensitivity, hypospadias of unknown origin, and epispadias. The 46-XX DSD includes monogenic forms of primary ovarian insufficiency, disorder of androgen excess, and Mayer-Rokitansky-Küster-Hauser (MRKH) syndrome. Chromosomal DSD includes 45-X (Turner syndrome), 47-XXY (Klinefelter syndrome), and mixed gonadal dysgenesis.³ It is estimated that genital anomalies happen every 4500-5000 births, and this condition requires a long-term management strategy that involves a multidisciplinary approach and tight cooperation with the family.^4,5

Due to the external genitalia abnormalities presenting in DSD patients, it is very common to have sex and gender difference in DSD patients.⁶ Therefore, psychiatric, behavioral, and gender-associated problems could arise in adults and children with DSD. Adults with DSD, especially those assigned as women. A previous study reported a lower sexual satisfaction and self-satisfaction in female DSD adults due to dissatisfaction with their own bodies.⁷

Ambiguity of gender occurring in DSD adults may create a problem of social identity. Adults with DSD may undergo verbal or even physical bullying which causes them to withdraw from the society. Consequently, adults with DSD also have a higher prevalence of people suffering from anxiety and depression, which was associated with the experience of shame, body dissatisfaction, and overall low self-esteem.⁸ Therefore, a structured assessment for each variable is needed for research to correctly measure psychiatric and sexual problems and the overall quality of life in adults with DSD. A validated and reliable instrument and technical considerations such as time to completion and adaptation to a specific population are essential in deciding which tool to choose for each purpose. This study aims to review structured questionnaire instruments that have been used to assess psychiatric, behavioral, sexual problems, and quality of life in adults with DSD.

Assesment of psychiatric and behavioral problem

Bajszczak, et al. study used the General Health Questionnaire (GHQ-28).⁹ This questionnaire has been a widely used screening instrument that can detect a wide spectrum of psychological disorders, especially the anxiety/depression spectrum. Consisting of 28 items, GHQ-28 is divided into four subdomains; somatic symptoms, anxiety and insomnia, disorder of daily function, and symptom of depression. Each item is a four scale from 0 to 3 (Likert scale), with a total of 21 scores in each scale, with higher scores indicating more disturbing symptoms. This questionnaire was originally developed by Goldberg et al. (1970) to detect the psychiatric disorder in a primary setting.^10,11 This questionnaire has been translated into different language and has been validated in various clinical settings. In the original paper, Goldberg, et al. used people from the population of London and found that with a cutoff of 4/5, this questionnaire shows satisfactory correlation of 0.77 with a specificity of 87% and sensitivity of 91.4%, and a misclassification rate of 11%.¹⁰ In a study conducted in Saudi Arabia, the same cutoff of 4/5 was found, although with lower specificity and sensitivity of 72% and 74% respectively, and a higher misclassification rate of 27%.¹² This questionnaire was already adopted into Indonesian language. In a study using samples from students in Central Java, Syafitri, et al. found that GHQ-28 had satisfactory reliability with overall Cronbach’s alpha of 0.897 and Cronbach’s alpha in each subscale A, B, C, and D were 0.831, 0.841, 0.688, and 0.833. Correlation coefficient of total score to each subscale ranges from 0.693 to 0.836, showing moderate to a high positive correlation.¹³ Specificity and sensitivity study in Indonesia was conducted on shorter version of GHQ-28 named GHQ-12. Conducted in six public health centers in Central Java, cutoff of ≥11 in Likert’s scale gave sensitivity of 81% and specificity of 62% and had satisfactory reliability with Cronbach’s alpha of 0.863.¹⁴ Overall, this questionnaire is quick, easy to use, and had satisfactory reliability and moderate sensitivity and specificity.

Brief Symptoms Inventory (BSI) is one of the most used questionnaires assessing psychiatric/behavioral problems in adults with DSD with a total of three studies using this tool.^15–17 BSI is a 53-items questionnaire used to measure general psychological well-being which consists of nine domains; somatization, obsessive-compulsive behavior, interpersonal sensitivity, depression, anxiety, hostility, phobic anxiety, paranoid ideation, and psychoticism. Each item is on a 5-point scale from 0 to 4 with a higher score indicating more severe symptoms. There are three scoring systems in BSI: Global Severity Index (GSI) which summed all scores, Positive Symptoms Total (PST), which count all items with a non-zero response, and Positive Symptom Distress Index (PSDI) which is the sum of the values of the items receiving non-zero responses divided by the PST. Scores are interpreted by comparison to age-appropriate normative data and divided into persentiles with a cutoff of T-scores >63.¹⁸ BSI-53 was reported to have good internal consistency reliability for all nine domain with Cochrane alpha ranging from 0.71 to 0.85 and good test-retest reliability for nine symptoms ranging from 0.68 to 0.91 and 0.90 for the Global Severity Index.¹⁸ No study about BSI adaptation into Indonesian language was found. This questionnaire offers a robust psychological assessment with scores interpreted based on normative data although it is relatively hard to use because of the number of items it contains. Derogatis, et al. offers a shortened BSI version into just 18 questions (BSI-18) for easier and more practical uses, although no study using BSI-18 in DSD patient was found.¹⁹ This shortened version of BSI questionnaire also offers good internal consistency with Cronbach’s alpha score for GSI was 0.93.²⁰

Achenbach and Rescorla’s Adult Behavior Checklist (ABCL) was used by D’alberton, et al. to assess behavior problems and competencies in adults.²¹ This tool was part of the Achenbach System of Empirically Based Assessment (ASEBA) to assess adaptive functioning and problem in adult aged 18 to 90+ years. This questionnaire is broken down into two parts; cross-informant syndrome and DSM-oriented scales. The cross-informant syndrome is further broken down into many subdomains; anxious/depressed, withdrawn/depressed, somatic complaints, social problems, thought problems (psychotic and obsessive spectrum), attention problems, rule-breaking behavior, and aggressive behavior. The DSM-oriented scales are broken down into six subdomains; affective problems, anxiety problems, somatic problems, attention deficit/hyperactivity problems, oppositional defiant problems, and conduct problems. This questionnaire generates a total score (TS) which can be divided into Internalizing score (IS) and externalizing score (ES) and divided into three ranges; normal range (< 64 for TS, IS, and < 60 ES), borderline (65-69 for TS, IS, and 60-63 ES), and pathological (≥ 70 for TS, IS and ≥ 64 for ES).²² Psychometric analysis of ABCL in a Chinese population shows the satisfactory result with factor loading were within 0.6-0.8 with moderate-to-high cross-informant and DSM-oriented scale correlation coefficient.²³ Unfortunately, no version adopted into Indonesian was available. ABCL offers a robust assessment of the adult behavioral problem with diverse subdomains, although its main disadvantage is the complicated nature of this tool.

Rosenberg Self-esteem Scale (RSES) is a quick and easy to uses questionnaire to assess global self-esteem defined as an overall evaluation of its own worth as a human being. As the name implies, this tool was first developed by Rosenberg, et al. back in 1965. RSES is a simple tool with 10 items, with each item is a 4-point scale from 0 to 3. With a score ranging 0 to 30, higher score indicates higher self-esteem.²⁴ In the original version, half of the questions are positively worded while the other half are negatively worded. This version had been tested for its validity and reliability in many settings and on average has been found to be effective.^25,26 However the negatively worded items are attributed to low factor loading, especially the question “I wish I could have more respect for myself”.²⁷ The revised version of this question based on a study in Thailand was, “I think I am able to give myself more respect” showed similar reliability as the original version while having better model fit with Goodness fit index of 0.970 and normed fit index of 0.964.²⁸ This questionnaire had been adopted into Indonesian language and its construct validity had been tested by Maroqi, et al. (2018). Based on this study, it was found that 7 out of 10 questions are unidimensional and therefore acceptable as assessment tools. This study found that questions 2, 5, and 8 had to be dropped due to the low loading factor and its T-value being < 1.96.²⁹ This questionnaire offers an easy to use and quick assessment of its respondent self-worth. However, its main disadvantages lie in some question that is negatively worded that has low factor loading even in its Indonesian version.

Mini International Neuropsychiatric Interview plus (MINI +) was used by Engberg, et al. to assess psychiatric problems in adult women with 46-XY and 46-XX.³⁰ This tool is a structured interview that allows the physician to diagnose 23 DSM-IV Axis I psychiatric diseases experienced at any time in life, including the specific time during a study with an estimated completion time of 45 minutes per subject. This questionnaire is an extended version of the MINI tools that are made for clinical settings that has a completion time of 15 minutes. Generally, MINI was divided into two categories; MINI patient patient-rated (MINI-PR) and MINI clinician-rated (MINI-CR). Compared to semi structured interview of diagnosis based on DSM-IV as a gold standard, MINI-CR offers a good or excellent kappa value (0.60-0.90) indicating high reliability with sensitivity of 70% or higher for all diagnosis but three values (dysthymia, obsessive compulsive disorder, and current drug dependencies) and specificity and negative predictive value of 85% or higher across all diagnosis. Patient-generated MINI-PR, on the other hand, had lower kappa value than MINI-CR (0.45-0.59) indicating lower reliability for major depressive episode, lifetime mania, panic disorder, agoraphobia, and psychotic disorder. MINI-PR was found to have high reliability (0.64-0.70) in diagnosing alchohol dependence, drug dependence, and anorexia and had poor reliability (< 0.45) for dysthymia, simple phobia, and social phobia.³¹ MINI, but not MINI+ was used several times and adopted into Indonesian version to screened depression, anxiety, and psychosis.^32,33 validity and reliability assessment of MINI Indonesian version to diagnosed depression, anxiety, and psychotis was conducted by Idaiani, et al. In this study it was found that MINI had a sensitivity between 60-80% and positive predictive value of 30-60%. It was shown to have moderate reliability (kappa value 0.62 and 0.76) for depression and anxiety.³² MINI+ offers a comprehensive diagnosis of all 23 axis I psychiatric disorders based on DSM-IV. Its main disadvantages is the long completion time of 45 minutes per subject. MINI in the other hand offer quicker but less comprehensive diagnosis with shorter completion time of 15 minutes although no study of MINI assessment in DSD patient was found. No study of MINI+ adaptation to Indonesian language and its validity/reliability study was found

Hospital anxiety and depression scale (HADS) was used by Liedmeier, et al. to assessed anxiety and depression in adult with DSD.⁷ Focused in the dimension of depression and anxiety, HADS is a seven items questionnaire with four-point scale from 0 (“definitely”) to 3 (“not at all”). Score cutoff for this instrument was divided into three categories; mild (score 8-10), moderate (11-14), and severe (15-21).³⁴ This questionnaire is widely used and has been adopted to various language such as Swedish, Chinese, French, Dutch, Portugenese, and Arabic. Most factor analysis of studies assessing HADS validity/reliability demonstrated two factor solution and in good accordance with the HADS subscale for anxiety (HADS-A) and depression (HADS-D). Cronbach’s alpha for HADS-A varied within 0.68 to 0.93 and for HADS-D varied within 0.67 to 0.90). Overall studies agrees to put score of 8 as a cutoff point with sensitivity and specificity around 80%, comparable to GHQ-28 that, assessed in the same domain of anxiety and depression.³⁵ HADS had been adopted into Indonesian version and its validity and reliability had been tested in patients with stroke. In this study it was shown that HADS-A had an inter-rater agreement of 0.706 and HADS-D had score of 0.681.³⁶ The tool offers a quick, easy to use depression and anxiety screening tools with high sensitivity and specificity and is already adopted into Indonesian version.

Body image scale (BIS) is a 30-items that assesses the satisfaction of various body parts on a five point scale from 1 (“very satisfied”) to 5 (“very dissatisfied”). An individual body image was thought to be influenced by many factors including personality type, social norm, cultural, and gender. A study of validity and reliability was conducted in patients with Inflammatory bowel disease (IBD) and cancer patients. In sample with IBD underwent stoma forming surgery, Internal consistency BIS was demonstrated with Cronbach’s alpha of 0.93, convergent validity with correlation coefficient of 0.64 (p < 0.001), and test-retest reliability of 0.82 (p < 0.001).³⁷ A systematic review of validity and reliability of BIS in cancer patients was conducted. In this systematic review, it was shown that the structural validity of BIS was rated sufficient with three studies support unidimensionality of the scale. Internal consistency using Cronbach’s alpha values ranged from 0.86-0.96. Reliability using test-retest shows value ranging from 0.85 to 0.92 indicating sufficient result.³⁸ This questionnaire has been adopted into an Indonesian version named “Skala Citra Tubuh”. In this study reliability is lower than any other study yet still acceptable with Cronbach’s alpha score of 0.696.³⁹ Compared to RSES, BIS is less commonly used but offers a moderate structural validity, internal consistency, and reliability and more focused on external appearance than RSES.

To assess ADHD, Liedmeier, et al. and de Vries, et al. both used the Adult ADHD self-report scale (ASRS).^7,8 This questionnaire was developed by WHO and consist of two versions; one is ASRS full version which has 18 items and ASRS screener which has 6 items each with 5-point scale ranging from “never” to “very often”. One score can be given if respondent answer for “sometimes, often or very often” in the first three questions and “often or very often” for the second three questions.⁴⁰ Liemeier, et al. and de Vries, et al. studies both incorporated ASRS screener which has 6 items.^7,8 Although shortened, ASRS screener outperformed the 18-items ASRS due to wide variations in symptoms-level concordance (cohen kappa in the range of 0.16-0.81). ASRS screener was shown to have sensitivity of 68.7%, specificity of 99.5%, and kappa of 0.76.⁴⁰ ASRS screener had an internal consistency reliability of 0.63-0.72 and had a test-retest reliability of 0.58-0.77.⁴¹ Unfortunately no study adapting this tool into Indonesian version is available yet.

To assess autism in patient with DSD, both Liedmeier, et al. and de Vries, et al. incorporated Short Autism Spectrum Quotient (AQ-10).^7,8 This 10-item questionnaire was adapted from Autism Spectrum Quotient long version with 50 items. In this questionnaire, each item is rated on a four point scale from “definitely agree” to “definitely disagree” with score of more than 6 indicating autistic symptoms. Using this cutoff, AQ-10 had a sensitivity of 93%, specificity of 95%, positive predictive value of 86%. This questionnaire showed high internal consistency with Cronbach’s alpha of 0.85.⁴² No adaptation into an Indonesian version was available yet. This questionnaire offers a quick screening of autism symptoms with very high specificity and sensitivity and was shown to be highly reliable.

The “Coping with DSD” questionnaire is the only questionnaire specific to be used in the population of adult with DSD in this review. Originally this questionnaire was developed by Kleinemeier, et al. in 2010. This questionnaire consisted of nine statement each with a four point scale of 1 (“completely true”) to 4 (“not true at all”). The nine statements are divided into two factors; openness and shame/stigmatization which a higher score indicates more open coping wiuth DSD and less shame/stigmatization.⁴³ Study regarding the validity and reliability of the original questionnaire was available yet. The main disadvantages of this questionnaire is that this tool is relatively new and highly specific for DSD population and thus makes the research regarding its usage very limited.

Assessment of quality of life

To assess quality of life, the WHOQOL questionnaire developed by World Health Organization is the most commonly used instrument for this purposes. Many studies used this instrument to assess quality of life (QoL) in adult patients with DSD.^7,21,44–46 This instrument, named WHOQOL-BREF, is the shortened version of the WHOQOL-100 quality of life assessment. It has 24 items divided into four subdomains; physical health, psychological, social relationship, and environment. Based on the original study by the WHOQOL group in 1998, it has satisfactory internal consistency with Cronbach’s alpha for each domain ranging from 0.72 to 0.88. Its test-retest reliability was ranging from 0.66 for physical health, 0.72 for psychological, 0.76 for social relationships and 0.87 for environment.⁴⁷ This instrument is already used widely and has been adopted into several language including Indonesian. In the study by Anisah, et al. published in 2019 for the population of Tuberculosis patient, WHOQOL-BREF showed good internal consistency, discriminant validity, and construct validity in all its four domain.⁴⁸ For the general population of adults in Indonesia, a study by Resmiya adopted WHOQOL-BREF into an Indonesian version named “Kualitas hidup Indonesia”. This version of WHOQOL had 30 items divided into 9 factors; spirituality, meaning of life, life achievement, work ethics, academics and knowledge, prosocial, social relationships, physical health, and psychological. This instrument showed high reliability coefficient with Cronbach’s alpha of 0.88. Mean correlation coefficient for each domain ranging from 0.311 to 0.429 indicating low correlation between subdomain.⁴⁹ Overall, WHOQOL-BREF offers a quick use self-reported general assessment of respondent quality of life with satisfactory reliability. It is by far the most commonly used instrument to measure QoL and had been adopted into the Indonesian language either by direct translation or with modification.

Short-form 36 is another instrument used to measure quality of life. For DSD patient, it has been used by Ros, et al. to measure QoL in adult with DSD.⁵⁰ As the name implies, this questionnaire consist of 36 items developed to measure health in 8 subdomains; physical functioning (10 questions), role physical functioning (4 questions), bodily pain (2 questions), general health (5 questions), vitality (4 questions), role emotional functioning (3 questions), social functioning (2 questions), and mental health (5 questions). This instrument scores range from 0 – 100 with higher score indicates better health status. Unlike any other instrument previously talked, the instrument is privately owned by The Medical Outcomes Trust, Health Assessment Lab, Quality Metric Incorporated, and Optum Incorporated, which holds all of its copyrights and trademarks.⁵¹ In the primary care setting, validity test for SF-36 had been conducted by Brazier, et al. in 1992. In this study, SF-36 was shown to be reliable with Cronbach’s alpha greater than 0.85 and reliability coefficient greater than 0.75. It was able to detect low level of ill health in patients who had scored 0 (good health) in another QoL instrument which is Nottingham health profile.⁵² A study of the Taiwanese population showed that SF-36 and WHOQOL had different constructs. In this study it was shown that SF-36 measures health-related QoL while WHOQOL measures more global QoL. Therefore clinicians and researchers should properly define which QoL wants to be measured to determine whether to use SF-36 or WHOQOL.⁵³ SF-36 had been adopted into an Indonesian version. A cross-sectional study in population with Rheumatoid Arthritis showed that the Indonesian version of SF-36 showed acceptable reliability in all domains except vitality (Cronbach’s alpha 0.52). Convergent and discriminant validities are good in almost all domains except for question 9, 16, 27, and 35.⁵⁴ SF-36 offers an alternative to WHOQOL to measure QoL with more focused on health-related QoL than WHOQOL. It has acceptable reliability and has also been adopted into the Indonesian language. The main disadvantage of this instrument is that the Indonesian version still needs revision due to its low reliability in one domain and several questions do not meet convergent and discriminant validities.

Assessment of gender role

To measure gender-related psychological problems, there are several instruments that have been used for population of adults with DSD. Hines, et al. incorporated Core Gender Identity, Sexual Orientation, and Gender Role Behavior to measures gender-related psychological problems in adult with DSD.⁵⁵ This questionnaire is a 12-items that is divided into three subdomains; core gender identity (the sense of self as male or female), sexual orientation (preferences for sexual partners of the same or other sex), and gender role behavior (participation in stereotypical masculine and feminine activities). This questionnaire measures gender identity problems in the past 12 months and lifetime. Likert scaling from 1 (always) to 7 (never) was used in items 1 – 8 and scaling from 1 (exclusively heterosexual) to 5 (exclusively homosexual) was used in items 9 – 12. In this questionnaire, lower scores indicates more sex-typical gender role behavior, more satisfaction with the current gender assignment, and more heterosexual tendencies. This questionnaire is an original tool made by Hines, et al. in 2003.⁵⁵ Ediati, et al. adopted it into Indonesian (Bahasa Indonesia) version in 2015. In this revised version, additional items to measure subject wishes for social gender role change were added each for the version for women and the version for men. In total, the Indonesian version comprised 14 items and had two gender versions (male and female) for the participant based on the gender they identified with. For the Indonesian version of the gender role questionnaire, principal component analysis generated two component explaining 74.9% of total variance. Items 1-4 are loaded on factor 1 that reflects gender identity and gender role behavior while item 5-14 are loaded on factor 2 which reflect cross-gender identity and cross-gender role behavior. Internal consistency for both factors is good (Cronbach’s alpha were 0.88 and 0.96, respectively).⁵⁶ This tool offers a comprehensive gender role assessment and has been used for DSD patients. Its main advantages are already adapted and revised in the Indonesian version with high reliability for all items.

Utrecht Gender Dysphoria Scale (UGDS) is another tool that can be used to measure gender dysphoria for both adults and children. This questionnaire consists of 12 items and has two versions each for males and female based on the basis of gender assigned at birth.⁵⁷ This instrument shows good intrernal consistency with Cronbach’s alpha of 0.80 for both the female and male versions. This instrument also showed excellent discriminant validity between transexual and non-transexual subjects (p < 0.001).⁵⁷ To meet the demand for the LGBTQ community arising lately, UGDS was further revised to encompass all the gender spectrum into UGDS – Gender spectrum (UGDS-GS). The revision added several items and made this instrument into 18 items tool with each tool using Likert-type scale.⁵⁸ Unfortunately, no study adopting this tools into an Indonesian version was found during the literature search.

Summary

Table 1 summarizes studies assessing gender roles, QoL, and psychiatric problems in adults with DSD. A total of 15 studies were found with various questionnaires/tools used in each studies. For adult studies, there are different tools to assess psychiatric problems, quality of life, and gender role. In addition, in adults, sexual function should be assessed due to the nature of DSD itself. Psychiatric problems are prevalent in the adult with DSD. Usually self-reported, many studies showed increased prevalence of various psychiatric problems in adults with DSD such as ADHD in Turner syndrome, increased prevalence of depression and anxiety, behavioral problem, social incompetencies, and suicidal tendencies.⁸ There are several structured or semi-structured questionnaires to assess psychiatric problems that had been used for adults with DSD.

Recommendations

Studies in adults with DSD are more abundant when compared to studies in DSD children. For psychiatric problems, the recommendation lies on the specific purposes or area of psychiatric problems that are being researched. To assess psychiatric problems with focused on anxiety or depression, the author recommends GHQ-28. This questionnaire has been a widely used screening instrument that focuses on the anxiety/depression spectrum. Originally created by Goldberg in 1970, This questionnaire has been widely used and translated into different languages and already validated in various clinical settings. Overall, this study offers a comprehensive psychiatric assessment focused on the anxiety/depression spectrum, consisting of 28 items divided into four subdomain; somatic symptoms, anxiety and insomnia, disorder of daily function, and symptom of depression. Validity studies show that this questionnaire is valid, shows satisfying reliability, and has high sensitivity and specificity. This tool also already been adapted into Indonesian language and shown to have high reliability with overall Cronbach’s alpha of 0.897. Overall this questionnaire is quick, easy to use, and had satisfactory reliability and high sensitivity and specificity with Indonesian adaptation available.

If the research demands an all-in-one tool that can screen many psychiatric problems comprehensively, the author recommends the Mini International Neuropsychiatric Interview plus (MINI+). This tool is a structured interview that allows the physician to diagnosed 23 DSM-IV Axis I psychiatric disease experienced in any time in life, including the specific time during a study with an estimated completion time of 45 minutes per subject. This questionnaire offers two different subjective approaches by having two variants; MINI patient-rated (MINI-PR) based on the patients perspective and MINI clinician-rated (MINI-CR) based on clinician perspective. Many studies did a reliability test for MINI+ to diagnose many different psychiatric problems. For MINI-CR, it was shown to have good reliability to diagnose many psychiatric problems with the exception of dysthymia, obsessive compulsive disorder, and current drug dependencies. MINI-PR is especially reliable to diagnose alcohol dependence, drug dependence, and anorexia, with low reliability when diagnosing dysthymia, simple phobia, and social phobia. Therefore depends on what diagnosis the researcher wants to diagnosed, MINI+ could either be good or bad as a research tool. Additional plus point for this tool is that it was already adapted into the Indonesian language although validity and reliability tests are limited to diagnosing depression, anxiety, and psychosis. Additional study of validity and reliability are needed when the Indonesian version of MINI+ is being used to diagnose symptoms other than depression, anxiety, and psychosis. Its main disadvantages is the long completion time of 45 minutes per subject.

To assess a specific problem of self-esteem, the author recommends Rosenberg Self-esteem Scale (RSES). This tools is more common in research than body image scale and more easy to use with only 10-items in total. This questionnaire is shown to be reliable. Its main disadvantage is the items that are phrased negatively often have low factor loading that means low validity. Some studies revised the negative words into a positive sentence, and the result shows similar reliability to the original version while having a better goodness fit index. This study already adapted into Bahasa Indonesia with a construct validity test shows 7 out of 10 items had acceptable loading factor. Questions 2,5, and 8 are dropped due to low loading factor. In conclusion, This questionnaire offers an easy to use and quick assessment of its respondent self-worth. However, its main disadvantages lie in some question that are negatively worded and have low factor loading even in its Indonesian version.

To assesses autism and ADHD in DSD patient, there are tools like Short Autism Spectrum Quotient (AQ-10) and used Adult ADHD self-report scale (ASRS). Both are an easy to use and quick tools to assessed either ADHD or autism spectrum. Studies also shows that this study had good reliability with high specificity. However, the author does not recommend these tools mainly because no Indonesian adaptation is available yet and because the utility of these tools overlap with MINI+ that has an Indonesian adaptation. Although both MINI+, ASRS, and AQ-10 still need validity and reliability, the already adapted MINI+ means that the translation process of adaptation are already done for MINI+. However for researcher concerned by the 45-minute completion time of MINI+, ASRS and AQ-10 could be an alternative due to the short and quick nature of these tools although it means that additional translation projects and validity-reliability tests need to be conducted first.

To assesses quality of life in adults with DSD, a literature search founds two tools that been used in adult with DSD; WHOQOL and short-form 36. The author recommends the use of WHOQOL over SF-36 for this purpose due to several reasons. The first reason is that WHOQOL developed by the World Health Organization (WHO) is more commonly used in research than SF-36. WHOQOL that been used in study of DSD adults is WHOQOL-BREF which is a shortened version of WHOQOL-100. This questionnaire is relatively short and quick to use with a total of 24 items divided into four subdomains; physical health, psychological, social relationship, and environment. This tools shows satisfactory internal consistency with Cronbach’s alpha for each domain ranging from 0.72 to 0.88. Its test-retest reliability ranged from 0.66 for physical health, 0.72 for psychological, 0.76 for social relationships and 0.87 for the environment. WHOQOL is widely used and has already been adapted into an Bahasa Indonesia version named “Kualitas Hidup Indonesia”. This version of WHOQOL had 30 items divided into 9 factors; spirituality, meaning of life, life achievement, work ethics, academics and knowledge, prosocial, social relationships, physical health, and psychological. This instrument showed a high reliability coefficient with Cronbach’s alpha of 0.88. On the other hand, SF-36 while being longer than WHOQOL, the main concern in this questionnaire is that this is the only questionnaire which is privately owned. Therefore, there will be additional administration and legal procedure needed before using this tool. Another disadvantages of SF-36 when compared to WHOQOL is the low reliability of its Indonesian adaptation with several questions not meeting convergent and discriminant validities and therefore it still needs revision.

Differences in sex and gender are to be expected in adults with DSD. In some countries, the difference between sex and gender may create some sociocultural problems, even induce verbal or physical bullying.⁵⁹ Some countries with strong culture of binary gender may also force adults with DSD to comply with one of the “typical” gender (male or female).⁵⁹ Theferore, assessment of gender role is of the utmost importance for adults with DSD. For gender role assessment of adults with DSD, literature searching found two questionnaires; Core Gender Identity, Sexual Orientation, and Gender Role Behavior and Utrecht Gender Dysphoria Scale (UGDS). Both are relatively short and quick to use with only 12 items. Overall recommendation goes to Core Gender Identity, Sexual Orientation, and Gender Role Behavior due to several reasons. This questionnaire, although short, comprehensively assesses gender role problems in three domains; core gender identity (the sense of self as male or female), sexual orientation (preferences for sexual partners of the same or other sex), and gender role behavior (participation in stereotypical masculine and feminine activities). Core Gender Identity, Sexual Orientation, and Gender Role Behavior measures gender roles in the past 12 months and lifetime. The main advantages of this tool is the avaibility of an Indonesian version that shows acceptable construct validity and good internal consistency. However UDGS also had its own advantages. This questionnaire already revised to encompasses LGBTQ community into UDGS-Gender Spectrum (UDGS-GS). Unfortunately despite this advantages, there is currently no Indonesian adaptation available and therefore need an additional adaptation, validity, and reliability study before UDGS can be applied to the Indonesian population.

Conclusion

There are a plethora of structured questionnaire tools used to measure psychiatric or behavioral problems, quality of life, and gender roles in both adults and children with DSD each with its own advantages and disadvantages. Some of the tools already been adopted into an Indonesian version and its validity and reliability had been tested.

For psychiatric problems for adults with DSD, the recommendation lies on the specific purposes or the area of psychiatric problems that are being researched. To assess psychiatric problems with focus on anxiety or depression, the author recommends GHQ-28. If the research demands an all-in-one tool that can screened many psychiatric problems comprehensively, the author recommends Mini International Neuropsychiatric Interview plus (MINI+). To assess self-esteem, the author recommends Rosenberg Self-esteem Scale (RSES). To assess quality of life in adults with DSD, WHOQOL is widely used and is also the authors’ recommendation. To assess gender role in adult with DSD, Core Gender Identity, Sexual Orientation, and Gender Role Behavior is recommended.