Increased psychiatric morbidity in women with complete androgen insensitivity syndrome or complete gonadal dysgenesis

Engberg, Hedvig; Strandqvist, Anna; Nordenström, Anna; Butwicka, Agnieszka; Nordenskjöld, Agneta; Hirschberg, Angelica Lindén; Frisén, Louise

doi:10.1016/j.jpsychores.2017.08.009

Cited by 26 publications

(16 citation statements)

References 42 publications

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“…In two studies (Chen et al, 2015;Rabkin et al, 2000), authors compared patients with their caregivers and one reported higher frequencies of both depressive and anxiety disorders in patients (Chen et al, 2015). In four out of eight comparisons with patients with non-rare diseases, patients with rare diseases showed higher rates than controls (Gündel et al, 2003;Huey et al, 2010;Jenkins-Jones et al, 2018;Mozzetta et al, 2008), two studies found no differences (Engberg et al, 2017;Ozel-Kizil, Akbostanci, Ozguven, & Atbasoglu, 2008), and in two studies, the authors reported higher frequencies in the comparison group (Dias et al, 2011;Moore et al, 2016).…”

Section: Comparisons With Control Groupsmentioning

confidence: 95%

“…A sensitivity analysis excluding two studies with a quality of <40% revealed a pooled prevalence of 18.8% (95% CI 13.8-25.2%). The prevalence of any lifetime affective disorder ranged between 11.6% (Henningsen & Meinck, 2003) and 72.7% (Engberg et al, 2017) with a pooled prevalence of 46.1% (95% CI 35.8-56.8%) across 10 studies. The model showed significant heterogeneity (I 2 = 90%, p < 0.0001).…”

Section: Frequency Of Affective Disordersmentioning

confidence: 99%

“…All eight studies reporting comparisons with population-based samples found significant differences in terms of a higher psychopathological burden of patients with rare diseases (Engberg et al, 2017;Gündel et al, 2001Gündel et al, , 2003Hedgeman et al, 2017;Lu et al, 2017;Manti et al, 2016;Moore et al, 2016;Roos et al, 2016). In two studies (Chen et al, 2015;Rabkin et al, 2000), authors compared patients with their caregivers and one reported higher frequencies of both depressive and anxiety disorders in patients (Chen et al, 2015).…”

Section: Comparisons With Control Groupsmentioning

confidence: 99%

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Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

et al. 2021

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We aimed to identify the prevalence of affective and anxiety disorders across different rare disease and identify correlates of psychopathology. We performed a systematic review and meta-analysis. We systematically searched Medline, PSYNDEX, PsycINFO for observational studies examining clinically diagnosed affective and/or anxiety disorders in adults with rare chronic diseases. Two researchers reviewed titles and abstracts independently and, for eligible studies, independently extracted data. The prevalence rates were pooled using a random intercept logistic regression model. We published a review protocol (http://www.crd.york.ac.uk/PROSPERO/display_record.php?ID=CRD42018106614CRD42018106614). We identified and screened 34 402 records for eligibility and considered 39 studies in the qualitative and 37 studies in the quantitative analysis, including N = 5951 patients with 24 different rare diseases. Heterogeneity between studies was large. Prevalence rates ranged widely between studies, with pooled prevalence estimates of 13.1% (95% CI 9.6–17.7%; I2 = 87%, p < 0.001) for current and 39.3% (95% CI 31.7–47.4%; I2 = 84%, p < 0.001) for lifetime major depressive disorder, 21.2% (95% CI 15.4–28.6%; I2 = 90%, p < 0.001) for current and 46.1% (95% CI 35.8–56.8%; I2 = 90%, p < 0.001) for lifetime affective disorders, and 39.6% (95% CI 25.5–55.6%; I2 = 96%, p < 0.001) for current and 44.2% (95% CI 27.0–62.9%; I2 = 94%, p < 0.001) for lifetime anxiety disorders. Sensitivity analyses excluding studies of low quality revealed nearly the same results. We conducted the first systematic review examining affective and anxiety disorders in adults with different rare diseases and found high prevalence rates. Supporting patients in disease adjustment can be crucial for their overall health and well-being.

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Section: Comparisons With Control Groupsmentioning

confidence: 95%

Section: Frequency Of Affective Disordersmentioning

confidence: 99%

Section: Comparisons With Control Groupsmentioning

confidence: 99%

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Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

et al. 2021

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“…Thus, no conclusions can be drawn about the specific cause (mental, physical, and/or social health) that contributed to the participants’ ratings. However, it is known that individuals with DSD more often have mental health problems than the normal population [ 46 , 47 ]. This fact was also seen in a previously conducted analysis concerning the mental health of this study population [ 13 ].…”

Section: Discussionmentioning

confidence: 99%

Physical and Reported Subjective Health Status in 222 Individuals with XY Disorder of Sex Development

Gong

Raile

Słowikowska-Hilczer

et al. 2021

Journal of the Endocrine Society

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Context Little is known about the physical health of individuals with 46,XY disorders of sex development (DSD). Objective To assess physical and reported subjective health of individuals with XY DSD. Design and methods As part of the dsd-LIFE study, patients with an XY DSD condition were analyzed in different diagnosis groups for metabolic parameters, comorbidities, metabolic syndrome, bone outcomes, and reported subjective health. Findings were evaluated by descriptive statistics. Results A total of 222 patients with XY DSD were included with a mean age of 28.8±12.2 years, mean height of 175.3±7.7 cm, mean weight of 74.3±20.0 kg and mean BMI of 24.1±6.0 kg/m 2. Obesity rate was not increased when descriptively compared to Eurostat data. Fourteen patients had metabolic syndrome (14/175; 8.0%). In descriptive comparison to data from the DECODE study and WHO, subjects fared better in the categories waist circumference, glucose, triglyceride, cholesterol and high-density lipoprotein. Of participants with available bone health data, 19/122 (15.6%) patients had a Z-score ≤ - 2.0 at lumbar spine indicating lowered bone mineral density (BMD). Mostly gonadectomized individuals with complete androgen insensitivity syndrome (CAIS) and no estrogen therapy had lowered BMD at lumbar spine. Individuals with XY DSD performed poorly in the category subjective health in descriptive comparison to Eurostat data. Conclusion Participants reported a lower subjective health status compared to Eurostat data but their overall metabolic health status was good. Decreased BMD at lumbar spine was especially present in gonadectomized individuals with CAIS and no estrogen therapy.

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“…Similar to previous studies (22,(36)(37)(38)(39) we performed a retrospective case-control study to estimate the risk of neurodevelopmental and psychiatric disorders in individuals with TS. Odds ratios (OR) for each neurodevelopmental and psychiatric outcome were estimated from conditional logistic regression models and were strati ed on matched sets to account for matching by year and county of birth.…”

Section: :4 Statistical Analysesmentioning

confidence: 99%

Neurodevelopmental and Psychiatric Disorders in Females With Turner Syndrome: A Population-based Study

Avdic¹,

Butwicka

Nordenström

et al. 2020

Preprint

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Background: Turner syndrome is the result of a missing X chromosome, partially or completely, in phenotypic girls. This can cause an array of medical and developmental difficulties. The intelligence quote has previously been described as uneven but considered within normal range. Although a social, intellectual and psychiatric profile is described in females with Turner syndrome, it is unclear to what extent they meet the clinical criteria for neurodevelopmental or psychiatric diagnoses. The aim of this study was to examine the prevalence of neurodevelopmental and psychiatric disorders in females with Turner syndrome. Methods: A retrospective case-control study was performed with a total of 1392 females with Turner syndrome identified through the Swedish National Patient Register and compared with 1:100 age- and sex matched controls from the general population. The association between Turner syndrome and diagnoses of neurodevelopmental and/ or psychiatric disorders were calculated using conditional logistic regression and is presented as estimated risk (Odds ratio, OR, 95% Confidence interval, CI) in females with Turner syndrome compared with matched controls. Results: Females with Turner syndrome had higher risk of any neurodevelopmental or psychiatric disorder (OR 1.37, 95% Cl 1.20-1.57), an eightfold (OR 8.59, 95% CI 6.58-11.20) increased risk of intellectual disability and a fourfold (OR 4.26, 95% CI 2.94-6.18) increased risk of autism spectrum disorder compared with the controls. In addition, females with Turner syndrome had an increased risk of a diagnosis of psychotic disorders (OR 1.98, 95% Cl 1.36-2.88), eating disorders (OR 2.03, 95% Cl 1.42-2.91) and behavioral disorders (OR 2.01, 95% CI 1.35-2.99). Conclusions: Females with TS have an increased risk of being diagnosed with any neurodevelopmental and psychiatric disorder. This warrants extensive assessment of intellectual and cognitive functions from early ages and increased psychiatric vigilance should be a part of lifelong healthcare for females with TS.

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Increased psychiatric morbidity in women with complete androgen insensitivity syndrome or complete gonadal dysgenesis

Abstract: The increased psychiatric morbidity in women with CAIS and GD highlights the need for clinical awareness of the psychiatric vulnerability in these patients.

Cited by 26 publications

References 42 publications

Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

Affective and anxiety disorders in patients with different rare chronic diseases: a systematic review and meta-analysis

Physical and Reported Subjective Health Status in 222 Individuals with XY Disorder of Sex Development

Neurodevelopmental and Psychiatric Disorders in Females With Turner Syndrome: A Population-based Study

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