The effect of telehealth on quality of life and psychological outcomes over a 12-month period in a diabetic cohort within the Whole Systems Demonstrator cluster randomised trial

Background: Much is written about the promise of telehealth and there is great enthusiasm about its potential. However, many studies of telehealth do not meet orthodox quality standards and there are few studies examining quality of life in diabetes as an outcome. Objective: To assess the impact of home-based telehealth (remote monitoring of physiological, symptom and self-care behavior data for long-term conditions) on generic and disease-specific health-related quality of life, anxiety, and depressive symptoms over 12 months in patients with diabetes. Remote monitoring provides the potential to improve quality of life, through the reassurance it provides patients. Methods: The study focused on participant-reported outcomes of patients with diabetes within the Whole Systems Demonstrator (WSD) Telehealth Questionnaire Study, nested within a pragmatic cluster-randomized trial of telehealth (the WSD Telehealth Trial), held across 3 regions of England. Telehealth was compared with usual-care, with general practice as the unit of randomization. Participant-reported outcome measures (ShortForm 12, EuroQual-5D, Diabetes Health Profile scales, Brief State-Trait Anxiety Inventory, and Centre for Epidemiological Studies Depression Scale) were collected at baseline, short-term (4 months) and long-term (12months) follow-ups. Intention-to-treat analyses testing treatment effectiveness, were conducted using multilevel models controlling for practice clustering and a range of covariates. Analyses assumed participants received their allocated treatment and were conducted for participants who completed the baseline plus at least one follow-up assessment (n=317). Results: Primary analyses showed differences between telehealth and usual care were small and only reached significance for 1 scale [dibetes health profile-disinhibited eating, P=.006). The magnitude of differences between trial arms did not reach the trial-defined minimal clinically important difference of 0.3 standard deviations for any outcome. Effect sizes (Hedge's g) ranged from 0.015 to 0.143 for Generic quality of life (QoL) measures and 0.018 to 0.394 for disease specific measures. Conclusions: Second generation home-based telehealth as implemented in the WSD evaluation was not effective in the subsample of people with diabetes. Overall, telehealth did not improve or have a deleterious effect quality of life or psychological outcomes for patients with diabetes over a 12-month period

A qualitative study of Telehealth patient information leaflets (TILs) : are we giving patients enough information?

BACKGROUND: The provision of patient information leaflets regarding telehealth has been perceived by potential consumers as a strategy to promote awareness and adoption of telehealth services. However, such leaflets need to be designed carefully if adoption and awareness among potential users is to be promoted. Therefore, the aims of this study were: first, to see how telehealth was portrayed in some of the existing telehealth leaflets (THLs). Second, to explore patients' perceptions of the existing THLs and their engagement with the concept and how THLs can be optimised. METHODS: A two-step approach was employed to address the aims of this study. The first phase involved the use of discourse analysis to compare 12 electronically and publically available THLs, with the existing THL guidance "Involve Yorkshire and Humber". The second phase involved conducting 14 semi-structured interviews with potential telehealth users/patients to gauge their perception and engagement with the concept, using the two leaflets that were mostly matching with the guidance used. Six interviews were audio-recorded and eight had detailed jotted notes. The interviews were transcribed and thematically analysed to identify key themes. RESULTS: The discourse analysis showed certain gaps and variations within the screened leaflets when addressing the following aspects: cost of the telehealth service, confidentiality, patients' choices in addition to equipment use and technical support. Analysis of the interviews revealed patients' need for having clear and sufficient information about the telehealth service within the THLs; in addition to, patients' preference for the use of simpler terminologies for telehealth description and the provision of clear simple texts with pictorial presentations. The interviews also revealed certain limitations against adoption of telehealth by the participants, such as: lack of privacy and confidentiality of information, fear of technology breakdown and equipment failure, loss of face-to-face contact with healthcare professionals and being too dependent on the telehealth service. CONCLUSION: The current study showed a great variation among the screened THLs and highlighted certain gaps within the content and presentation of these leaflets. However, the study also highlighted certain key issues to be considered when designing THLs in the future to enhance telehealth uptake and use by patients

Longitudinal in vivo MRI in a Huntington’s disease mouse model: global atrophy in the absence of white matter microstructural damage

Huntington’s disease (HD) is a genetically-determined neurodegenerative disease. Characterising neuropathology in mouse models of HD is commonly restricted to cross-sectional ex vivo analyses, beset by tissue fixation issues. In vivo longitudinal magnetic resonance imaging (MRI) allows for disease progression to be probed non-invasively. In the HdhQ150 mouse model of HD, in vivo MRI was employed at two time points, before and after the onset of motor signs, to assess brain macrostructure and white matter microstructure. Ex vivo MRI, immunohistochemistry, transmission electron microscopy and behavioural testing were also conducted. Global brain atrophy was found in HdhQ150 mice at both time points, with no neuropathological progression across time and an elective sparing of the cerebellum. In contrast, no white matter abnormalities were detected from the MRI images or electron microscopy images alike. The relationship between motor function and MR-based structural measurements was different for the HdhQ150 and wild-type mice, although there was no relationship between motor deficits and histopathology. Widespread neuropathology prior to symptom onset is consistent with patient studies, whereas the absence of white matter abnormalities conflicts with patient data. The myriad reasons for this inconsistency require further attention to improve the translatability from mouse models of disease

Fishing the Molecular Bases of Treacher Collins Syndrome

Treacher Collins syndrome (TCS) is an autosomal dominant disorder of craniofacial development, and mutations in the TCOF1 gene are responsible for over 90% of TCS cases. The knowledge about the molecular mechanisms responsible for this syndrome is relatively scant, probably due to the difficulty of reproducing the pathology in experimental animals. Zebrafish is an emerging model for human disease studies, and we therefore assessed it as a model for studying TCS. We identified in silico the putative zebrafish TCOF1 ortholog and cloned the corresponding cDNA. The derived polypeptide shares the main structural domains found in mammals and amphibians. Tcof1 expression is restricted to the anterior-most regions of zebrafish developing embryos, similar to what happens in mouse embryos. Tcof1 loss-of-function resulted in fish showing phenotypes similar to those observed in TCS patients, and enabled a further characterization of the mechanisms underlying craniofacial malformation. Besides, we initiated the identification of potential molecular targets of treacle in zebrafish. We found that Tcof1 loss-of-function led to a decrease in the expression of cellular proliferation and craniofacial development. Together, results presented here strongly suggest that it is possible to achieve fish with TCS-like phenotype by knocking down the expression of the TCOF1 ortholog in zebrafish. This experimental condition may facilitate the study of the disease etiology during embryonic development

Mouse models of neurodegenerative disease: preclinical imaging and neurovascular component.

Neurodegenerative diseases represent great challenges for basic science and clinical medicine because of their prevalence, pathologies, lack of mechanism-based treatments, and impacts on individuals. Translational research might contribute to the study of neurodegenerative diseases. The mouse has become a key model for studying disease mechanisms that might recapitulate in part some aspects of the corresponding human diseases. Neurode- generative disorders are very complicated and multifacto- rial. This has to be taken in account when testing drugs. Most of the drugs screening in mice are very di cult to be interpretated and often useless. Mouse models could be condiderated a ‘pathway models’, rather than as models for the whole complicated construct that makes a human disease. Non-invasive in vivo imaging in mice has gained increasing interest in preclinical research in the last years thanks to the availability of high-resolution single-photon emission computed tomography (SPECT), positron emission tomography (PET), high eld Magnetic resonance, Optical Imaging scanners and of highly speci c contrast agents. Behavioral test are useful tool to characterize di erent ani- mal models of neurodegenerative pathology. Furthermore, many authors have observed vascular pathological features associated to the di erent neurodegenerative disorders. Aim of this review is to focus on the di erent existing animal models of neurodegenerative disorders, describe behavioral tests and preclinical imaging techniques used for diagnose and describe the vascular pathological features associated to these diseases

Platelet monoamine oxidase B activity in Parkinson's disease: a re-evaluation

An increase in platelet monoamine oxidase (MAO) B activity in drug-free parkinsonians (n = 6) compared with healthy controls (n = 10) has been confirmed using both phenylethylamine (PEA) and dopamine as substrates, reaching statistical significance in the case of PEA oxidising activity (p < 0.05). Thus, certain reports of raised platelet MAO B activity towards PEA but decreased activity towards dopamine in parkinsonians, raising the possibility of the existence of an abnormal form of MAO B in this condition, cannot be supported