Integrating perspectives of transgender and gender-diverse youth, family members, and professionals to support their health and wellbeing - a mixed-method study protocol.

BACKGROUND: The current literature highlights a strong link between the poor health outcomes of transgender and gender diverse (TGD) individuals and their negative experiences in various areas of life. Most of these publications rely on adults' memories, lacking a focus on the current experiences and needs of young transgender and gender-diverse individuals. Furthermore, previous studies on support for these young people often solely consider the perspectives of TGD adults or professionals and rarely involve parents' viewpoints. METHODS: This study will use a mixed sequential method with a participatory approach. Firstly, the qualitative phase will explore the difficulties and needs of TGD (15-20 years old) and of the families and professionals who support them. Results from this part will be used to develop the questionnaire for the quantitative phase, with the help of a community board. Secondly, based on participatory epidemiological research, the quantitative phase will use an intersectional perspective to measure the impact of individual and structural factors on the quality of life and well-being of transgender and gender-diverse young people. Finally, a co-creation phase will be undertaken to formulate recommendations based on the results of the first two phases. DISCUSSION: This research aims at better understanding the influence of gender identity on the quality of life and health of TGD young people and their families and to identify protective and risk factors that affect their vulnerabilities. ETHICS AND DISSEMINATION: This study has been approved by the Ethics Committee of the Erasme Faculty Hospital (CCB B4062023000140). As this research is participatory and part of a PhD dissertation, we aim to disseminate the results through our partners' networks and structures locally, and internationally through conferences and peer-reviewed journals.

Approach to the Patient: Hormonal Therapy in Transgender Adults With Complex Medical Histories.

While endocrinologists continue to initiate gender-affirming hormone therapy (GAHT) in healthy transgender and gender diverse (TGD) patients, they may also encounter more TGD patients in their clinics with complex medical histories that influence the patient-provider shared decision-making process for initiating or continuing GAHT. The purpose of this Approach to the Patient article is to describe management considerations in 2 adults with thromboembolic disease and 2 adults with low bone mineral density in the setting of feminizing and masculinizing GAHT.

Changes in depression symptom profile with gender-affirming hormone use in transgender persons.

BACKGROUND: Women show higher prevalence of depression and different symptomatology than men, possibly influenced by sex hormones. Many transgender persons, who face a high risk of depression, use Gender-Affirming Hormone Therapy (GAHT), but the impact of GAHT on depressive symptom profiles is unknown. METHODS: This study examined depressive symptoms in transgender persons before GAHT and after 3- and 12 months of GAHT. We used the Inventory of Depressive Symptomatology-Self Report to assess depressive symptoms, exploratory factor analysis (EFA) to assess symptom clusters, and linear mixed models to assess changes in symptom clusters. RESULTS: This study included 110 transmasculine (TM) and 89 transfeminine (TF) participants. EFA revealed four symptom clusters: mood, anxiety, lethargy, and somatic symptoms. Changes in total depressive symptoms significantly differed between TM and TF groups. After 3 months of GAHT, TM participants reported improvement in lethargy (-16 %; 95%CI: -29 %; -2 %), and after 12 months TF participants reported worsening in low mood (24 %; 95%CI: 3 %; 51 %), but absolute score changes were modest. Neither group showed changes in anxiety or somatic symptoms. LIMITATIONS: This study had limited sample sizes at 12 months follow-up and did not include relevant biological or psychosocial covariates. DISCUSSION: Changes in depressive symptoms after GAHT use differ in TM and TF persons: TM persons report slight improvements in lethargy, whereas TF persons report a slight increase in low mood. Starting GAHT represents a significant life event with profound social and physical effects, and further research should assess social and biological effects of GAHT on mood-related symptoms.

CBLL1 is hypomethylated and correlates with cortical thickness in transgender men before gender affirming hormone treatment.

Gender identity refers to the consciousness of being a man, a woman or other condition. Although it is generally congruent with the sex assigned at birth, for some people it is not. If the incongruity is distressing, it is defined as gender dysphoria (GD). Here, we measured whole-genome DNA methylation by the Illumina © Infinium Human Methylation 850k array and reported its correlation with cortical thickness (CTh) in 22 transgender men (TM) experiencing GD versus 25 cisgender men (CM) and 28 cisgender women (CW). With respect to the methylation analysis, TM vs. CW showed significant differences in 35 CpGs, while 2155 CpGs were found when TM vs. CM were compared. With respect to correlation analysis, TM showed differences in methylation of CBLL1 and DLG1 genes that correlated with global and left hemisphere CTh. Both genes were hypomethylated in TM compared to the cisgender groups. Early onset TM showed a positive correlation between CBLL1 and several cortical regions in the frontal (left caudal middle frontal), temporal (right inferior temporal, left fusiform) and parietal cortices (left supramarginal and right paracentral). This is the first study relating CBLL1 methylation with CTh in transgender persons and supports a neurodevelopmental hypothesis of gender identity.

Gender-affirming treatment and mental health diagnoses in Danish transgender persons: a nationwide register-based cohort study.

IMPORTANCE: Gender affirming treatment aims to improve mental health. OBJECTIVE: To investigate longitudinal mental health outcomes in Danish transgender persons. DESIGN: National register-based cohort study in Danish transgender persons with diagnosis code of "gender identity disorder" during the period 2000-2021. PARTICIPANTS: Five age-matched controls of the same sex at birth and five age-matched controls of the other sex at birth were included for each transgender person. MAIN OUTCOMES: Diagnosis codes of mental and behavioral disorders and/or prescription of psychopharmacological agents until June 2022. RESULTS: The cohort included 3812 transgender persons with median age (interquartile range) 19 (15; 24) years for persons assigned female at birth (AFAB, N = 1993) and 23 (19; 33) years for persons assigned male at birth (AMAB, N = 1819) and 38 120 controls. Follow up duration was up to 10 years with mean (standard deviation) 4.5 (4.3) years. In transgender persons AFAB compared to control women, the odds ratio (OR) (95% confidence interval) for mental and behavioral disorders was 6.7 (5.5; 8.1) before the index date, 9.9 (8.4; 11.7) at 1 year, 5.8 (4.4; 7.7) at 5 years, and 3.4 (2.1; 7.5) at 8 years follow up. In transgender persons AMAB compared to control men, corresponding ORs were 5.0 (4.0; 6.4), 11.3 (9.3; 13.7), 4.8 (3.5; 6.5), and 6.6 (4.2; 10.3) at 8 years follow up (all P < .001). CONCLUSION: The OR for mental health disorders was higher in transgender persons compared to controls and remained elevated throughout follow up, especially in transgender persons AMAB.

The association between newborn cord blood steroids and ambient prenatal exposure to air pollution: findings from the ENVIRONAGE birth cohort.

Knowledge of whether prenatal exposure to ambient air pollution disrupts steroidogenesis is currently lacking. We investigated the association between prenatal ambient air pollution and highly accurate measurements of cord blood steroid hormones from the androgenic pathway.This study included 397 newborns born between the years 2010 and 2015 from the ENVIRONAGE cohort in Belgium of whom six cord blood steroid levels were measured: 17α-hydroxypregnenolone, 17α-hydroxyprogesterone, dehydroepiandrosterone, pregnenolone, androstenedione, and testosterone. Maternal ambient exposure to PM2.5 (particles with aerodynamic diameter ≤ 2.5 µm), NO2, and black carbon (BC) were estimated daily during the entire pregnancy using a high-resolution spatiotemporal model. The associations between the cord blood steroids and the air pollutants were tested and estimated by first fitting linear regression models and followed by fitting weekly prenatal exposures to distributed lag models (DLM). These analyses accounted for possible confounders, coexposures, and an interaction effect between sex and the exposure. We examined mixture effects and critical exposure windows of PM2.5, NO2 and BC on cord blood steroids via the Bayesian kernel machine regression distributed lag model (BKMR-DLM).An interquartile range (IQR) increment of 7.96 µg/m3 in PM2.5 exposure during pregnancy trimester 3 was associated with an increase of 23.01% (99% confidence interval: 3.26-46.54%) in cord blood levels of 17α-hydroxypregnenolone, and an IQR increment of 0.58 µg/m³ in BC exposure during trimester 1 was associated with a decrease of 11.00% (99% CI: -19.86 to -0.012%) in cord blood levels of androstenedione. For these two models, the DLM statistics identified sensitive gestational time windows for cord blood steroids and ambient air pollution exposures, in particular for 17α-hydroxypregnenolone and PM2.5 exposure during trimester 3 (weeks 28-36) and for androsterone and BC exposure during early pregnancy (weeks 2-13) as well as during mid-pregnancy (weeks 18-26). We identified interaction effects between pollutants, which has been suggested especially for NO2.Our results suggest that prenatal exposure to ambient air pollutants during pregnancy interferes with steroid levels in cord blood. Further studies should investigate potential early-life action mechanisms and possible later-in-life adverse effects of hormonal disturbances due to air pollution exposure.

Cystatin C-Based eGFR Changes during Gender-Affirming Hormone Therapy in Transgender Individuals.

BACKGROUND: Men with CKD tend to experience a faster eGFR decline than women, potentially because of sex hormones. Limited research exists regarding the effect of gender-affirming hormone therapy (GAHT) on kidney function. Furthermore, monitoring kidney function during GAHT is challenging because serum creatinine is confounded by body composition. To investigate the relationship between sex hormones and kidney function, we studied the changes of serum creatinine and serum cystatin C, a filtration marker less affected by sex, during 1 year of GAHT. METHODS: As part of the European Network for the Investigation of Gender Incongruence study, we measured serum creatinine and serum cystatin C in 260 transgender women and 285 transgender men before and 12 months after initiating GAHT. Transgender women received estradiol plus cyproterone acetate, while transgender men received testosterone. Cystatin C-based eGFR was calculated using the full-age-spectrum equation. RESULTS: In transgender women, cystatin C decreased by 0.069 mg/L (95% confidence interval [CI], 0.049 to 0.089), corresponding with a 7 ml/min per 1.73 m 2 increase in eGFR. In transgender men, cystatin C increased by 0.052 mg/L (95% CI, 0.031 to 0.072), corresponding with a 6 ml/min per 1.73 m 2 decrease in eGFR. Creatinine concentrations decreased (-0.065 mg/dl; 95% CI, -0.076 to -0.054) in transgender women and increased (+0.131 mg/dl; 95% CI, 0.119 to 0.142) in transgender men. Changes in creatinine-based eGFR varied substantially depending on the sex used in the equation. CONCLUSIONS: In this cohort of transgender individuals, cystatin C-based eGFR increased with estradiol and antiandrogen therapy and decreased with testosterone therapy.

Testosterone administration affects 1H-MRS metabolite spectra in transgender men.

BACKGROUND: Recently, a variety of studies using different neuroimaging techniques attempted to identify the existence of a brain endophenotype in people with gender dysphoria (GD). However, despite mounting neuroimaging work, brain gender differences and effects of gender-affirming hormone therapy (GAHT) at the metabolite level remain understudied. METHODS: Thirty-one transgender men (TM) before and after testosterone administration (7.7 months ± 3.5 months), relative to 30 cisgender men (CM) and 35 cisgender women (CW) underwent magnetic resonance spectroscopy (1H-MRS) at two time points. Two brain regions were assessed, i.e. the lateral parietal cortex and the amygdala/anterior hippocampus. Associated metabolites that were measured include N-acetyl aspartate (NAA), creatine (Cr), choline (Cho), glutamate and glutamine (Glx), myo-inositol (mI), glycine (Gly) and their respective ratios. RESULTS: A critical time by group interaction revealed an effect of GAHT in the lateral parietal cortex of TM. MI+Gly/Cr ratios decreased upon initiation of GAHT. In addition, NAA/Cr and Cho/Cr ratios were lower in CW when compared to CM in the lateral parietal cortex. Glx levels and Glx/Cr ratios in TM differed from those in CW in the amygdala/anterior hippocampus. Interestingly, pubertal age of onset of gender dysphoria (i.e. GD) in TM differentially affected testosterone-mediated effects on Cr concentration and NAA/Cr ratios when compared to childhood and adult GD onset in the amygdala/anterior hippocampus. CONCLUSION: This 1H-MRS study demonstrated that testosterone administration shifts mI+Gly/Cr ratios in the parietal cortex. In the amygdala/anterior hippocampus, modulation of metabolite concentrations by age of onset of GD is suggestive for a possible developmental trend.

Clinical presentation of sporadic and hereditary pheochromocytoma/paraganglioma.

Pheochromocytomas (PHEO) and paragangliomas (PGL) can occur sporadic or within genetic predisposition syndromes. Despite shared embryology, there are important differences between PHEO and PGL. The aim of this study was to describe the clinical presentation and disease characteristics of PHEO/PGL. A retrospective analysis of consecutively registered patients diagnosed with or treated for PHEO/PGL in a tertiary care centre was performed. Patients were compared according to anatomic location (PHEO vs PGL) and genetic status (sporadic vs hereditary). In total, we identified 38 women and 29 men, aged 50 ± 19 years. Of these, 42 (63%) had PHEO, and 25 (37%) had PGL. Patients with PHEO presented more frequently with sporadic than hereditary disease (45 years vs 27 (77%) vs 8 (23%)) than patients with PGL (9 (36%) vs 16 (64%), respectively) and were older at diagnosis (55 ± 17 vs 40 ± 18 years, P = 0.001), respectively). About half of the cases in both PHEO and PGL were diagnosed due to disease-related symptoms. In patients with PHEO, tumour diameter was larger (P = 0.001), metanephrine levels higher (P = 0.02), and there was more frequently a history of cardiovascular events than in patients with PGL. In conclusion, we found that patients with PGL more frequently have a hereditary predisposition than those with PHEO, contributing to the fact that diagnosis is generally made earlier in PGL. Although diagnosis in both PHEO and PGL was mostly due to related symptoms, patients with PHEO more often presented with cardiovascular comorbidities than those with PGL which might relate to a higher number of functionally active tumours in the former.

Sexual Motivation in Persons with Multiple Sclerosis: A Controlled Cross-Sectional Study.

Purpose: Sexual motives are major determinants of sexual behaviour. It has been known that sexual motives may vary according to circumstances. Multiple sclerosis (MS) is a chronic disease causing a broad range of symptoms and disabilities, that often interfere with sexual activities. We aimed to investigate the sexual motives in persons with MS. Patients and Methods: Cross-sectional study in 157 persons with MS and 157 controls matched for age, gender, relationship, duration of relationship and educational status via propensity score matching. The Reasons for Having Sex (YSEX) questionnaire assessed the proportion with which a person had engaged in sexual intercourse for each of 140 distinct motives to have sex. Estimated mean differences in scores for four primary factors (Physical, Goal attainment, Emotional, Insecurity) and 13 sub-factors, and sexual satisfaction and importance of sex were calculated as Average Treatment Effect of the Treated using 99% confidence intervals. Results: Persons with MS reported a lower proportion of engaging in sex compared with the controls for the factors Physical (-0.29), Emotional (-0.23) and Insecurity (-0.10); and for the physical sub-factors Pleasure (-0.48), Experience seeking (-0.32), Stress reduction (-0.24), and Physical desirability (-0.16), the emotional sub-factors Love and commitment (-0.27) and Expression (-0.17), and the insecurity sub-factor Self-esteem boost (-0.23). In the control group seven of the top 10 sexual motives were physical versus five in the MS group. The importance of sex was lower in the MS group (-0.68). Conclusion: Findings of this controlled cross-sectional study suggest a reduction in the number of sexual motives in persons with MS, especially of physical motives related to pleasure and experience seeking. Health care professionals may consider assessing sexual motivation when dealing with persons with MS who suffer from decreased sexual desire or another sexual dysfunction.

Physiological declines in sex steroid exposure in relation to changes in body composition in healthy men-a prospective cohort study.

OBJECTIVE: Androgen levels decline from early adulthood and decreases are steeper in men with increasing body mass index. It is, however, unclear to what extent changes in other indices of body composition and metabolism associate with changes in sex steroid levels in healthy men. Therefore, this study investigated longitudinal changes in body composition and metabolic health in relation to sex steroid levels in healthy adult men. DESIGN: This is a longitudinal, population-based study. A total of 676 healthy men aged 24-46 years were measured at baseline and after ±12 years. METHODS: Serum sex hormone-binding globulin (SHBG) was measured by immunoassay, testosterone (T), estradiol (E2), and dihydrotestosterone byliquid chromatography with tandem mass spectrometry (LC-MS/MS), calculated free T and calculated free E2 (cFE2), and homeostasis model assessment for insulin resistance (HOMA-IR) were calculated. Grip strength was measured by hand-grip dynamometry. Body composition was determined using dual-energy X-ray absorptiometry and peripheral quantitative computed tomography. RESULTS: Mean fat mass (FM), lean mass (LM), and HOMA-IR increased (all P < .001). Decreasing androgen and SHBG levels was associated with increasing FM, whereas decreasing (cF)E2 levels were associated with decreasing FM (all P < .005). Decreasing (cF)E2 levels and increasing SHBG levels associated with decreasing LM (all P < .002). Changes in sex steroid levels and HOMA-IR or grip strength were not interrelated. CONCLUSION: Aging leads to increases in FM indices and insulin resistance, whereas changes in parameters of LM are less unequivocal. In healthy adult men, physiological changes in sex steroid exposure clearly correlate with changes in adiposity but not so with lean mass, insulin resistance, or grip strength. CLINICAL TRIAL: The SIBEX study was registered on ClinicalTrials.gov (#NVT02997033).

Sex hormones, insomnia, and sleep quality: Subjective sleep in the first year of hormone use in transgender persons.

STUDY OBJECTIVES: Transgender persons can use gender-affirming hormone therapy (GAHT) to align their physical appearance with their identified gender. Many transgender persons report poor sleep, but the effects of GAHT on sleep are unknown. This study examined the effects of a 12 months of GAHT use on self-reported sleep quality and insomnia severity. METHODS: A sample of 262 transgender men (assigned female at birth, started masculinizing hormone use) and 183 transgender women (assigned male at birth, started feminizing hormone use), completed self-report questionnaires on insomnia (range 0-28), sleep quality (range 0-21) and sleep onset latency, total sleep time and sleep efficiency before start of GAHT and after 3, 6, 9, and 12 months of GAHT. RESULTS: Reported sleep quality showed no clinically significant changes after GAHT. Insomnia showed significant but small decreases after 3 and 9 months of GAHT in trans men (-1.11; 95%CI: -1.82; -0.40 and -0.97; 95%CI: -1.81; -0.13, respectively) but no changes in trans women. In trans men, reported sleep efficiency decreased by 2.8% (95%CI: -5.5%; -0.2%) after 12 months of GAHT. In trans women, reported sleep onset latency decreased by 9 min (95%CI: -15; -3) after 12 months of GAHT. CONCLUSIONS: These findings show that 12 months of GAHT use did not result in clinically significant changes in insomnia or sleep quality. Reported sleep onset latency and reported sleep efficiency showed small to modest changes after 12 months of GAHT. Further studies should focus on underlying mechanisms by which GAHT could affect sleep quality.

Investigating Levothyroxine Use and Its Association with Thyroid Health in Patients with Hypothyroidism: A Community Pharmacy Study.

Background: To gain maximum therapeutic effect while minimizing side effects, it is imperative for patients with hypothyroidism to use their levothyroxine (LT4) correctly, such as adhering to the prescribed regimen. Little is currently known about how patients actually use LT4 in real life. We investigated the use of LT4, as well as the thyroid health (thyrotropin [TSH] and health-related quality of life [HR-QoL]), and evaluated if proper LT4 use is associated with better thyroid health. Methods: A cross-sectional observational study was conducted in a Belgian community sample of adults using LT4 for hypothyroidism since ≥2 years. Participants completed a self-administered questionnaire on patient characteristics, self-reported adherence to LT4, timing of intake, and co-medication. They also completed the thyroid-specific patient-reported outcome (ThyPRO-39) questionnaire, measuring the HR-QoL. Pharmacy dispensing data were used to calculate the medication possession ratio (MPR). Results: We included 856 participants (mean age 61.4 ± 14.3 years, 86% [740/856] females). Approximately one in four participants (138/563) had out-of-range TSH levels. Generally, ThyPRO-39 scores were in the lower part of the range (indicating better HR-QoL), with the scales "emotional susceptibility" and "tiredness" showing the worst scores. Approximately 28% (178/632) of the participants were classified as non-adherent (MPR <80%), corresponding to at least 73 cumulative days per year without LT4 intake. Twenty-five percent (212/854) of participants self-reported non-adherence, with unintentional non-adherence (forgetfulness) most frequently reported (21.9%, 187/854). Only 39% (329/836) of participants complied with the recommendation of ingesting LT4 ≥ 30 minutes before eating. Additionally, 7% (58/856) of participants concurrently used molecules that bind to LT4, without applying the recommended dosing interval. There was no significant correlation between LT4 usage (adherence, timing of intake, and interaction with complex forming drugs) and TSH or HR-QoL. Conclusions: We found that many participants with hypothyroidism did not use their LT4 as effectively as possible, particularly with respect to timing of administration. However, the participants' HR-QoL seemed largely satisfactory, and there was no significant correlation between correctly using LT4 and thyroid health.

Gender minority stress in transgender people: a major role for social network.

BACKGROUND: Gender minority individuals, on average, experience higher rates of mental health problems. Mounting work suggests that gender minority stress (GMS) contributes to mental health outcomes in transgender/gender-nonconforming individuals. AIM: We assessed whether GMS decreased in transgender people after initiating gender-affirming hormone therapy (GAHT), and we identified social predictors and hormonal associations for GMS at 2 time points. METHODS: GMS was surveyed through self-report questionnaires tapping into proximal and distal stressors and coping constructs following the minority stress framework. Eighty-five transgender persons wishing to undertake hormonal interventions were assessed prospectively at start of GAHT and after 7.7 ± 3.5 months (mean ± SD). Sixty-five cisgender persons served as a control group. OUTCOMES: (1) Proximal stressors were surveyed by the Beck Depression Inventory II, State-Trait Anxiety Inventory, Scale for Suicide Ideation, Suicidal Thoughts/Attempts, Stigma Consciousness Questionnaire, and Perceived Stress Scale; (2) distal stressors by the Everyday Discrimination Scale; and (3) coping constructs by the Resilience Scale, social network, social standing, and Marlowe Crowne Social Desirability Scale. RESULTS: Transgender people experienced higher rates of proximal stressors (Beck Depression Inventory II, State-Trait Anxiety Inventory, Scale for Suicide Ideation, Suicidal Thoughts/Attempts, Perceived Stress Scale) and had lower protective factors (social standing) prior to and during GAHT than cisgender people. Social network and resilience were lower in transgender people relative to cisgender peers only at baseline. Prospectively, decreasing trait anxiety was observed in transgender people. Social factors were adequate predictors of multiple GMS constructs. Specifically, a major role for social network emerged. As for hormonal associations, only serum estradiol levels in transgender women with GAHT were negatively associated with trait anxiety and suicidal thoughts/attempts but positively with resilience and social desirability. CLINICAL IMPLICATIONS: Stimulating a social environment supportive of diverse identities, particularly by investing in social networks as a resource for resilience, is likely to alleviate GMS. STRENGTHS AND LIMITATIONS: Longer duration of interventions with sex steroid treatment, with continued resilience-enhancing strategies, is needed to observe further alleviation of GMS in transgender persons. Also, objective and subjective GMS identification with heteronormative attitudes and beliefs should be surveyed for good measure when assessing GMS. CONCLUSION: Transgender people experienced more GMS throughout study visits than cisgender people did. With a relatively short period of GAHT, some significant changes in and predictors for experienced GMS emerged.

Long-term cardiometabolic morbidity in young adults with classic 21-hydroxylase deficiency congenital adrenal hyperplasia.

PURPOSE: To study the current practice for assessing comorbidity in adults with 21-hydroxylase CAH and to assess the prevalence of comorbidity in these adults. METHODS: A structured questionnaire was sent to 46 expert centres managing adults with CAH. Information collected included current therapy and surveillance practice with a particular focus on osteoporosis/osteopaenia, hyperlipidaemia, type 2 diabetes/hyperinsulinaemia, hypertension, CV disease, obesity. RESULTS: Of the 31 (67%) centres from 15 countries that completed the survey, 30 (97%) screened for hypertension by measuring blood pressure, 30 (97%) screened for obesity, 26 (84%) screened for abnormal glucose homoeostasis mainly by using Hb1Ac (73%), 25 (81%) screened for osteoporosis mainly by DXA (92%), 20 (65%) screened for hyperlipidaemia and 6 (19%) screened for additional CV disease. Of the 31 centres, 13 provided further information on the six co-morbidities in 244 patients with a median age of 33 yrs (range 19, 94). Of these, 126 (52%) were females and 174 (71%) received fludrocortisone in addition to glucocorticoids. Of the 244 adults, 73 (30%) were treated for at least one comorbidity and 15 (21%) for more than 2 co-morbidities. Of 73, the patients who were treated for osteoporosis/osteopaenia, hyperlipidaemia, type 2 diabetes/hyperinsulinaemia, hypertension, CV disease, obesity were 43 (59%), 17 (23%), 16 (22%), 10 (14%), 8 (11), 3 (4%) respectively. CONCLUSION: Cardiometabolic and bone morbidities are not uncommon in adults with CAH. There is a need to standardise the screening for these morbidities from early adulthood and to explore optimal therapy through routine collection of standardised data.

PAI-BEL: a Belgian multicentre survey of primary adrenal insufficiency.

Objective: Primary adrenal insufficiency (PAI) is a rare disease with an increasing prevalence, which may be complicated by life-threatening adrenal crisis (AC). Good quality epidemiological data remain scarce. We performed a Belgian survey to describe the aetiology, clinical characteristics, treatment regimens, comorbidities and frequency of AC in PAI. Methods: A nationwide multicentre study involving 10 major university hospitals in Belgium collected data from adult patients with known PAI. Results: Two hundred patients were included in this survey. The median age at diagnosis was 38 years (IQR 25-48) with a higher female prevalence (F/M sex ratio = 1.53). The median disease duration was 13 years (IQR 7-25). Autoimmune disease was the most common aetiology (62.5%) followed by bilateral adrenalectomy (23.5%) and genetic variations (8.5%). The majority (96%) of patients were treated with hydrocortisone at a mean daily dose of 24.5 ± 7.0 mg, whereas 87.5% of patients also received fludrocortisone. About one-third of patients experienced one or more AC over the follow-up period, giving an incidence of 3.2 crises per 100 patient-years. There was no association between the incidence of AC and the maintenance dose of hydrocortisone. As high as 27.5% of patients were hypertensive, 17.5% had diabetes and 17.5% had a diagnosis of osteoporosis. Conclusion: This study provides the first information on the management of PAI in large clinical centres in Belgium, showing an increased frequency of postsurgical PAI, a nearly normal prevalence of several comorbidities and an overall good quality of care with a low incidence of adrenal crises, compared with data from other registries.

Doege-Potter syndrome in a patient with a giant abdominal solitary fibrous tumor: a case report and review of the literature.

A 63-year-old man with spells of reduced consciousness in the morning and a giant abdominal mass presented to our institution for a second opinion. Investigation revealed non-diabetic hypoinsulinemic hypoglycemic events. Removal of the abdominal mass solved the hypoglycemia. Anatomopathological examination confirmed a solitary fibrous tumor (SFT). Doege-Potter syndrome was diagnosed. Doege-Potter syndrome is a potentially life-threatening rare paraneoplastic syndrome characterized by recurrent hypoinsulinemic hypoglycemia due to the overproduction of a prohormone form of insulin-like growth factor-II (pro-IGF-II) from a solitary fibrous tumor. First, we describe the clinical, laboratory and radiologic findings of the case. Second, a brief literature review on Doege-Potter syndrome is provided.

Intonation Parameters in Gender Diverse People.

OBJECTIVES/HYPOTHESIS: Although intonation is often addressed in speech training for gender diverse individuals, the relationship between intonation and femininity/masculinity ratings remains unclear. The aim of this study was to examine differences in intonation parameters in gender diverse individuals. Moreover, the relationship between acoustic intonation parameters and femininity/masculinity ratings was investigated. METHODS: Speech samples of semistructured speech were elicited from cisgender (cis) (107 ciswomen, 104 cis men), transgender (trans) (19 trans women, 10 trans men), and non-binary (n = 11) individuals using a prosody protocol. An objective acoustic analysis was performed to compare intonation parameters (upward/downward/flat intonation shift, general and final intonation shift, general fundamental frequency range, fundamental frequency variation index) between groups. In addition, a listening experiment was conducted, consisting of a cis and gender diverse listening panel (n = 41). The listeners were asked to rate the femininity/masculinity of speech samples (n = 57) using a visual analogue scale. Correlational analyses were used to examine the relationship between intonation parameters and femininity/masculinity ratings. RESULTS: Similarity was found in the intonation parameters of participants with a similar gender identity. In non-binary speakers, no significant differences in acoustic intonation parameters were found between these speakers and the cisgender speakers. In addition, no significant correlations were found between the acoustic intonation parameters and the femininity/masculinity ratings in the groups with cis men, cis women, and non-binary participants. However, moderate to strong significant correlations were found between acoustic intonation parameters and femininity/masculinity ratings in the trans participants. CONCLUSIONS: Intonation is a "speech marker" that distinguishes between groups with a different gender identity. No relationship was observed between intonation and femininity/masculinity ratings for cisgender and non-binary speakers. However, the significant relationship between these parameters for transgender participants (trans men and trans women) provides evidence for intonation exercises in gender affirming voice, speech, and communication training, and therefore contribute to evidence-based intonation training in transgender persons.