Frequency, morbidity and equity - the case for increased research on male fertility.

Currently, most men with infertility cannot be given an aetiology, which reflects a lack of knowledge around gamete production and how it is affected by genetics and the environment. A failure to recognize the burden of male infertility and its potential as a biomarker for systemic illness exists. The absence of such knowledge results in patients generally being treated as a uniform group, for whom the strategy is to bypass the causality using medically assisted reproduction (MAR) techniques. In doing so, opportunities to prevent co-morbidity are missed and the burden of MAR is shifted to the woman. To advance understanding of men's reproductive health, longitudinal and multi-national centres for data and sample collection are essential. Such programmes must enable an integrated view of the consequences of genetics, epigenetics and environmental factors on fertility and offspring health. Definition and possible amelioration of the consequences of MAR for conceived children are needed. Inherent in this statement is the necessity to promote fertility restoration and/or use the least invasive MAR strategy available. To achieve this aim, protocols must be rigorously tested and the move towards personalized medicine encouraged. Equally, education of the public, governments and clinicians on the frequency and consequences of infertility is needed. Health options, including male contraceptives, must be expanded, and the opportunities encompassed in such investment understood. The pressing questions related to male reproductive health, spanning the spectrum of andrology are identified in the Expert Recommendation.

Developing a self-management intervention to manage hypermobility spectrum disorders (HSD) and hypermobile Ehlers-Danlos syndrome (hEDS): an analysis informed by behaviour change theory.

PURPOSE: Hypermobility Spectrum Disorders (HSD) and Hypermobile Ehlers-Danlos Syndrome (hEDS) are heritable connective tissue disorders associated with joint instability and pain, but with scant guidance for supporting patients. The aim was to determine recommendations for an HSD/hEDS self-management intervention. MATERIALS AND METHODS: Barriers to self-management were mapped onto the Theoretical Domains Framework (TDF) and Capability, Opportunity, Motivation-Behaviour (COM-B) model in a behavioural analysis. A modified Nominal Group Technique was used to prioritise behaviour change technique (BCT) interventions (n = 9 women). RESULTS: Possible BCTs incorporated. EDUCATION: Incorporating self-help strategies, education to improve their knowledge of HSD/hEDS, and how to judge information about HSD/hEDS. TRAINING: In activity pacing, assertiveness and communication skills, plus what to expect during pregnancy, when symptoms can worsen. ENVIRONMENTAL RESTRUCTURING AND ENABLEMENT: Support from occupational therapists to maintain independence at work and home. MODELLED BEHAVIOUR: That illustrates how other people with HSD/hEDS have coped with the psychosocial impact. CONCLUSIONS: This study is the first to apply theoretically-informed approaches to the management of HSD/hEDS. Participants indicated poor access to psychological support, occupational therapy and a lack of knowledge about HSD/hEDS. Future research should evaluate which intervention options would be most acceptable and feasible.Implications for rehabilitationPatients with Hypermobility Spectrum Disorders or Hypermobile Ehlers-Danlos Syndrome can be active partners in the co-design of behaviour change interventions.Behaviour change interventions should target psychological support and patient education, particularly patient information.Additional behaviour change interventions included environmental restructuring and enablement; adaptations to participants' environment with input from occupational therapy.Participants were keen to suggest opportunities for behavioural modelling; positive fist-person modelling narratives, written by those with HSD/hEDS, which addressed how they coped with the psychosocial impact of their condition.

A gender perspective on appearance-related concerns and its manifestations among persons born with unilateral cleft lip and palate.

Cleft lip and palate (CLP) affects a person's facial appearance and can cause appearance-related distress. Appearance-related distress and its manifestations are not fully understood. Women with CLP tend to experience a higher degree of distress as well as undergo more aesthetic revisions than men. The aim of this study was to investigate if self-reported appearance-related gender differences occur among adults born with unilateral CLP using the Derriford Appearance Scale 24 (DAS24). One hundred sixty two consecutive patients, 107 men and 55 women, with unilateral CLP and no associated syndromes treated at the same hospital were asked to answer the DAS24. A mixed methods approach was used to analyse the questionnaires both quantitively and qualitatively. Fifty men and 30 women participated, the results showed that the aspects of appearance considered most disturbing was cleft-related among both genders, most common  in regards to the nose. Three themes were found: acceptance, cleft features, and general appearance issues. The quantitative part showed that women reported higher appearance-related social anxiety and avoidance than men. This study demonstrated that appearance-related distress is present in both genders; however, women expressed more distress than men. Moreover, non-cleft as well as cleft features are of importance for self-reported dissatisfaction.

Psychometric Validation of the Brazilian Portuguese Version of the Derriford Appearance Scale-24 (DAS-24) for People Living with HIV/AIDS.

The changes in appearance of people living with HIV/AIDS (PLHA) interferes with how people around them react to their body, how social interactions take place, and how each person perceives and accepts their body. The definition of itself can be severely challenged when the body changes as a result of illness and the person does not look healthier anymore. People living with HIV/AIDS (PLHA) are an especially vulnerable group when it comes to "distress" and the psychosocial impact of appearance, yet the assessment of body image changes in these people was subjective in Brazil. The aim of this paper was to assess the psychometric properties of the Brazilian version of Derriford Appearance Scale 24 (DAS-24) for a sample of Brazilians living with HIV/AIDS. A sample of 400 patients were recruited from an HIV/AIDS ambulatory, aged between 18 and 78 years, of both sexes. The psychometric properties of DAS-24 were investigated while using confirmatory factor analysis (CFA), with unweighted least square estimation and listwise deletion for missing data. The adjustment of three structural models previously established for DAS-24 (single-factor, two-factor, and three-factor) was investigated. Evidences of construct validity-convergent and discriminant-and internal consistency-Cronbach's alpha and construct reliability-were also generated for the measure model. The results showed that the one-factor model had the best adjustment, after eliminating items 8, 17, and 20, and accepting the covariance of errors between items 4 and 10; 9 and 23; 11 and 14; and, 14 and 22. Additionally, validity and reliability evidence were satisfactory for the model. The Brazilian Portuguese version of DAS-24 seems to be a psychometrically sound scale for measuring body image distress for people living with HIV/AIDS (PLHA).

Validating a scale to measure engineers' perceived self-efficacy for engineering education outreach.

Education outreach in schools has been identified as a critical route to influence children's perceptions and capabilities for Science, Technology, Engineering, and Mathematics careers. Evidence suggests that providing non-teaching professionals like engineers with training programmes and structured experience can boost perceived self-efficacy to perform education outreach, which in turn means better quality and more frequent public engagement. A validated measure of the construct of perceived self-efficacy for engineering education outreach will be useful for effective science communication participation, research, and practise. This article presents the methods used to develop the Engineering Outreach Self-efficacy Scale (EOSS), along with initial reliability and validation results to support the scale's use. The 10-item scale was found to have good internal consistency and reliability (Cronbach's alpha α = .92) with a sample of 160 engineers. The scale had convergent validity with general self-efficacy. Engineers with more experience of education outreach had higher self-efficacy for engineering education outreach. There were no significant differences between male and female engineers. Initial test-retest results showed engineers receiving training in education outreach significantly improved their EOSS scores, indicating capability to detect change over time. It is hoped this scale will prove useful for further evaluation of engineering education outreach and public engagement with science activities.

A Systematic Review of the Effectiveness of Acceptance and Commitment Therapy (ACT) for Body Image Dissatisfaction and Weight Self-Stigma in Adults.

Body image dissatisfaction (BID) and weight self-stigma are prevalent and associated with physical and psychological ill-health. Acceptance and Commitment Therapy (ACT) is increasingly employed for both, yet little is known about its effectiveness. Searches of 12 databases identified six studies using online, face-to-face or self-help ACT interventions for BID or weight self-stigma, of varying duration and intensity. Their effectiveness and quality were evaluated. Two reported improved BID, three improved weight self-stigma, and one reported no impact on weight self-stigma. Methodological issues (small sample sizes, lack of allocation concealment, attention control and long-term follow up) impacted the validity of findings. Due to the small number of studies and poor study quality, the effectiveness of ACT for BID and weight self-stigma remains unclear. Nonetheless findings suggest psychological flexibility may facilitate reduction in BID and weight self-stigma and indicate that brief online as well as lengthy face-to-face delivery may be useful. Suggestions for further research are made.

Factor structure and convergent validity of the Derriford Appearance Scale-24 using standard scoring versus treating 'not applicable' responses as missing data: a Scleroderma Patient-centered Intervention Network (SPIN) cohort study.

OBJECTIVE: Valid measures of appearance concern are needed in systemic sclerosis (SSc), a rare, disfiguring autoimmune disease. The Derriford Appearance Scale-24 (DAS-24) assesses appearance-related distress related to visible differences. There is uncertainty regarding its factor structure, possibly due to its scoring method. DESIGN: Cross-sectional survey. SETTING: Participants with SSc were recruited from 27 centres in Canada, the USA and the UK. Participants who self-identified as having visible differences were recruited from community and clinical settings in the UK. PARTICIPANTS: Two samples were analysed (n=950 participants with SSc; n=1265 participants with visible differences). PRIMARY AND SECONDARY OUTCOME MEASURES: The DAS-24 factor structure was evaluated using two scoring methods. Convergent validity was evaluated with measures of social interaction anxiety, depression, fear of negative evaluation, social discomfort and dissatisfaction with appearance. RESULTS: When items marked by respondents as 'not applicable' were scored as 0, per standard DAS-24 scoring, a one-factor model fit poorly; when treated as missing data, the one-factor model fit well. Convergent validity analyses revealed strong correlations that were similar across scoring methods. CONCLUSIONS: Treating 'not applicable' responses as missing improved the measurement model, but did not substantively influence practical inferences that can be drawn from DAS-24 scores. Indications of item redundancy and poorly performing items suggest that the DAS-24 could be improved and potentially shortened.

A systematic review of the traits and cognitions associated with use of and belief in complementary and alternative medicine (CAM).

Complementary and alternative medicine (CAM) use is widespread despite the controversy over its effectiveness. Although previous reviews have examined the demographics and attitudes of CAM users, there is no existing review on the traits or cognitions which characterise either CAM users or those who believe in CAM effectiveness. The current systematic review set out to address these gaps in the literature by applying a narrative synthesis. A bibliographic search and manual searches were undertaken and key authors were contacted. Twenty-three papers were selected. The trait openness to experience was positively associated with CAM use but not CAM belief. Absorption and various types of coping were also positively associated with CAM use and belief. No other trait was reliably associated with CAM use or belief. Intuitive thinking and ontological confusions were positively associated with belief in CAM effectiveness; intuitive thinking was also positively associated with CAM use. Studies researching cognitions in CAM use/belief were mostly on non-clinical samples, whilst studies on traits and CAM use/belief were mostly on patients. The quality of studies varied but unrepresentative samples, untested outcome measures and simplistic statistical analyses were the most common flaws. Traits and cognition might be important correlates of CAM use and also of faith in CAM.

Toward a Conceptual and Methodological Shift in Craniofacial Research.

OBJECTIVE: To date, research investigating the psychological impact of craniofacial conditions has produced variability across outcomes. The aims of this article were to summarize the challenges that may contribute to this variability, and to offer alternative perspectives and approaches to guide future research and practice. DESIGN: A comprehensive evaluation of papers exploring adjustment to congenital craniofacial conditions was conducted. Methodological approaches and underlying conceptual issues were identified and summarized. RESULTS: The conceptual limitations identified include inherent challenges pertaining to the multifactorial and fluctuating nature of adjustment, a lack of consensus regarding the primary constituents of a positive outcome, scant use of appropriate models and theories, and a predominant focus on "deficits" over "strengths." The methodological shortcomings identified include a lack of representative samples, biomedical inclusion/exclusion criteria, inconsistency in measurement, a relative absence of the patient perspective, variability in approaches to data analysis and interpretation, and the failure to draw on knowledge from other disciplines and related fields of health research. Findings are believed to be relevant to all disciplines involved in craniofacial research and practice. CONCLUSIONS: Existing literature remains markedly affected by a range of conceptual and methodological challenges, despite these challenges being identified 25 years ago. The present article proposes that a shift in the way we conceptualize and study craniofacial conditions is needed, in order to construct a comprehensive understanding of adjustment to craniofacial conditions, and to address the key unanswered questions important to all stakeholders.

Do Psychosocial Interventions Improve Quality of Life and Wellbeing in Adults with Neuromuscular Disorders? A Systematic Review and Narrative Synthesis.

Quality of life and well-being are frequently restricted in adults with neuromuscular disorders. As such, identification of appropriate interventions is imperative. The objective of this paper was to systematically review and critically appraise quantitative studies (RCTs, controlled trials and cohort studies) of psychosocial interventions designed to improve quality of life and well-being in adults with neuromuscular disorders. A systematic review of the published and unpublished literature was conducted. Studies meeting inclusion criteria were appraised using a validated quality assessment tool and results presented in a narrative synthesis. Out of 3,136 studies identified, ten studies met criteria for inclusion within the review. Included studies comprised a range of interventions including: cognitive behavioural therapy, dignity therapy, hypnosis, expressive disclosure, gratitude lists, group psychoeducation and psychologically informed rehabilitation. Five of the interventions were for patients with Amyotrophic Lateral Sclerosis (ALS). The remainder were for patients with post-polio syndrome, muscular dystrophies and mixed disorders, such as Charcot-Marie-Tooth disease, myasthenia gravis and myotonic dystrophy. Across varied interventions and neuromuscular disorders, seven studies reported a short-term beneficial effect of intervention on quality of life and well-being. Whilst such findings are encouraging, widespread issues with the methodological quality of these studies significantly compromised the results. There is no strong evidence that psychosocial interventions improve quality of life and well-being in adults with neuromuscular disorders, due to a paucity of high quality research in this field. Multi-site, randomised controlled trials with active controls, standardised outcome measurement and longer term follow-ups are urgently required.

The effect of septorhinoplasty on quality of life and nasal function in Asians.

This is the first study that aimed to assess the effects of septorhinoplasty on quality of life (QOL) in an Asian population. The study consisted of 2 parts. First, the Derriford Appearance Scale 59 (DAS-59) was translated into Korean, and the reliability and validity were assessed by administering the Korean version of Derriford Appearance Scale 59 (DAS-59K) and 36-item short-form health survey to 88 inpatients scheduled for operations. Then, a prospective study was conducted which included 31 patients who underwent primary septorhinoplasty from October 2008 through May 2009. The changes in QOL and nasal symptoms were evaluated by comparing the preoperative and postoperative 3 month DAS-59K and nasal obstruction symptom evaluation scales. Principal component analysis of the DAS-59K showed an optimum 5-factor and the Cronbach α for each factor was greater than 0.7. Significant correlation was found between the DAS-59K and 36-item short-form health survey. Objective evaluation showed at least an improvement in every patient. After septorhinoplasty, there was improvement at scores related to general self-consciousness, negative self-concept, and physical stress (P < 0.05). The pattern of improvement differed by sex, age, and the presence of external nose deviation. Mean nasal obstruction symptom evaluation scores decreased significantly after surgery. The DAS-59K is a reliable and valid test, which can be a useful tool to assess individual response to living with problems of appearance. Septorhinoplasty improves both QOL and nasal function which should be taken into consideration in future counseling of individual patients expecting septorhinoplasty.

Human amnion epithelial cells repair established lung injury.

With a view to developing a cell therapy for chronic lung disease, human amnion epithelial cells (hAECs) have been shown to prevent acute lung injury. Whether they can repair established lung disease is unknown. We aimed to assess whether hAECs can repair existing lung damage induced in mice by bleomycin and whether the timing of cell administration influences reparative efficacy. In addition, we aimed to characterize the effect of hAECs on fibroblast proliferation and activation, investigating possible mechanisms of reparative action. hAECs were administered intraperitoneally (IP) either 7 or 14 days after bleomycin exposure. Lungs were assessed 7 days after hAEC administration. Bleomycin significantly reduced body weight and induced pulmonary inflammation and fibrosis at 14 and 21 days. Delivery of hAECs 7 days after bleomycin had no effect on lung injury, whereas delivery of hAECs 14 days after bleomycin normalized lung tissue density, collagen content, and α-SMA production, in association with a reduction in pulmonary leucocytes and lung expression of TGF-ß, PDGF-α, and PDGF-ß. In vitro, hAECs reduced proliferation and activation of primary mouse lung fibroblasts. Our findings suggest that the timing of hAEC administration in the course of lung disease may impact on the ability of hAECs to repair lung injury.

Incorporating user perspectives in the design of an online intervention tool for people with visible differences: face IT.

BACKGROUND: Individuals with visible differences can experience social anxiety in relation to their appearance. Social skills-based psychosocial interventions have to date shown only limited effectiveness at addressing their concerns. AIMS: To incorporate user perspectives in the development of an online psychosocial intervention, known as Face IT. METHOD AND RESULTS: Study one consisted of a needs assessment with 12 individuals with a visible difference and six health professionals in order to identify the difficulties experienced by those with visible difference and obtain feedback on the proposed content of Face IT. The findings demonstrated support for the social skills model and the use of an online intervention. Study two consisted of an empirical usability evaluation of Face IT with 14 potential users and 14 health professionals. Based on feedback from the participants, changes were made to the graphics and navigation of the programme. The clinical content has been made more acceptable. CONCLUSIONS: The findings indicate support for the importance of social skills-based psychosocial interventions for addressing the needs of those with a visible difference, and have allowed modifications to be made to Face IT ahead of a randomized controlled trial of effectiveness.

Attentional and interpretative biases in appearance concern: An investigation of biases in appearance-related information processing.

The present study examined associations between high levels of appearance concern and information processing biases in interpretation and attention. An opportunity sample (N=79) categorised ambiguous stimuli as related or unrelated to appearance. Participants then responded to the same stimuli in a modified visual dot-probe task assessing attentional bias. Participant responses were assessed in relation to level of appearance concern. The results indicated a valence specific bias towards interpretation of ambiguous stimuli as negative and appearance-related in individuals with higher levels of concern. There was also evidence of attentional bias towards information perceived as appearance-related in participants with higher levels of appearance concern. The study findings suggest that association between appearance-orientated information processing biases and level of appearance concern; this association may lead to mutually reinforcing bias and concern.

Neuropathological correlates of temporal pole white matter hyperintensities in CADASIL.

BACKGROUND AND PURPOSE: White matter (WM) hyperintensities on MRI or leukoaraiosis is characteristic of stroke syndromes. Increased MRI signals in the anterior temporal pole are suggested to be diagnostic for cerebral autosomal-dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL), with 90% sensitivity and 100% specificity. The structural correlates of these specific WM hyperintensities seen on T2-weighted and FLAIR sequences in the temporal pole of CADASIL are unclear. We assessed pathological changes in postmortem tissue from the temporal pole to reveal the cause of CADASIL-specific WM hyperintensities. METHODS: A combination of tinctorial and immunostaining approaches and in vitro imaging methods were used to quantify the extent of perivascular space (PVS), arteriosclerosis determined as the sclerotic index, WM myelination as the myelin index, and damage within the WM as accumulated degraded myelin basic protein in samples of the anterior temporal pole from 9 CADASIL and 8 sporadic subcortical ischemic vascular dementia cases, and 5 similarly aged (young) and 5 older controls. Luxol fast blue-stained serial sections from a CADASIL case were also used to reconstruct the temporal pole, which was then compared to the MR images. RESULTS: Luxol fast blue sections used to reconstruct the temporal pole revealed an abundance of enlarged PVS in the WM that topographically appeared as indistinct opaque regions. The mean and total areas of the PVS per WM area (%PVS) were significantly greater in CADASIL compared to the controls. The myelin index was severely reduced in CADASIL in relation to the subcortical ischemic vascular dementia and control sample that was consistent with increased immunoreactivity of degraded myelin basic protein, indicating myelin degeneration. Cerebral microvessels associated with the PVS exhibited a 4.5-fold greater number of basophilic (hyalinized) vessels and a 57% increase in the sclerotic index values in CADASIL subjects compared to young controls. A significant correlation between the quantity of hyalinized vessels and sclerotic index values was also apparent (P<0.05). CONCLUSIONS: Our findings suggest that MRI hyperintensities in the temporal pole of CADASIL patients are explained by enlarged PVS and degeneration of myelin accompanied by lack of drainage of the interstitial fluid rather than lacunar infarcts. Consistent with the lack of MR hypersignals in the temporal pole of older subcortical ischemic vascular dementia subjects, our observations imply greater progression of pathological changes in CADASIL patients.

Multiple intraosseous calvarial hemangiomas mimicking metastasis from renal cell carcinoma.

Renal cell carcinomas are known to metastasise to the bones in the form of lytic lesions. However, not all osteolytic lesions in patients with renal cell carcinoma are metastatic in nature. The report describes the case of a 68-year old lady who was diagnosed with a renal cell carcinoma 3 and half years back and treated with radical nephrectomy along with excision of an inferior vena cava tumour thrombus. The tumour was completely excised and she remained disease free till date. Subsequently, multiple lytic lesions were detected incidentally on the cranial vault, which on biopsy demonstrated intraosseous hemangioma. Though it is well known that renal cell carcinomas can metastasise to the bones in the form lytic lesions, it is important for clinicians to remember a few other differentials, one of which would be an intraosseous hemangioma, which is a benign pathology. Many times patients would be treated as having metastatic disease merely on radiological findings. In this case report, there was a high index of radiological suspicion for metastases, however establishing diagnosis by biopsy prevented overtreatment in this instance.