Best Practices and Lessons Learned for Action Research in eHealth Design and Implementation: Literature Review.

BACKGROUND: Action research (AR) is an established research framework to introduce change in a community following a cyclical approach and involving stakeholders as coresearchers in the process. In recent years, it has also been used for eHealth development. However, little is known about the best practices and lessons learned from using AR for eHealth development. OBJECTIVE: This literature review aims to provide more knowledge on the best practices and lessons learned from eHealth AR studies. Additionally, an overview of the context in which AR eHealth studies take place is given. METHODS: A semisystematic review of 44 papers reporting on 40 different AR projects was conducted to identify the best practices and lessons learned in the research studies while accounting for the particular contextual setting and used AR approach. RESULTS: Recommendations include paying attention to the training of stakeholders' academic skills, as well as the various roles and tasks of action researchers. The studies also highlight the need for constant reflection and accessible dissemination suiting the target group. CONCLUSIONS: This literature review identified room for improvements regarding communicating and specifying the particular AR definition and applied approach.

Brain-Computer interfaces for communication: preferences of individuals with locked-in syndrome, caregivers and researchers.

OBJECTIVES: The development of Brain-Computer Interfaces to restore communication (cBCIs) in people with severe motor impairment ideally relies on a close collaboration between end-users and other stakeholders, such as caregivers and researchers. Awareness about potential differences in opinion between these groups is crucial for development of usable cBCIs and access technology (AT) in general. In this study, we compared the opinions of prospective cBCI users, their caregivers and cBCI researchers regarding: (1) what applications would users like to control with a cBCI; (2) what mental strategies would users prefer to use for cBCI control; and (3) at what stage of their clinical trajectory would users like to be informed about AT and cBCIs. METHODS: We collected data from 28 individuals with locked-in syndrome, 29 of their caregivers and 28 cBCI researchers. The questionnaire was supported with animation videos to explain different cBCI concepts, the utility of which was also assessed. RESULTS: Opinions of the three groups were aligned with respect to the most desired cBCI applications, but diverged regarding mental strategies and the timing of being informed about cBCIs. Animation videos were regarded as clear and useful tools to explain cBCIs and mental strategies to end-users and other stakeholders. CONCLUSIONS: Disagreements were clear between stakeholders regarding which mental strategies users prefer to use and when they would like to be informed about cBCIs. To move forward in the development and clinical implementation of cBCIs, it will be necessary to align the research agendas with the needs of the end-users and caregivers.IMPLICATIONS FOR REHABILITATIONBrain-Computer Interfaces may offer people with severe motor impairment a brain-based and muscle-independent approach to control communication-technology. The successful development of communication BCIs (cBCIs) relies on a close collaboration between end-users and other stakeholders, such as caregivers and researchers.Our work reveals that people with locked-in syndrome (end-users), their caregivers and researchers developing cBCIs agree that direct and private forms of communication are the most desired cBCI applications, but disagree regarding the preferred mental strategies for cBCI control and when to be informed about cBCIs.Animation videos are an effective tool for providing information to individuals, independent of their level of health literacy, regarding the concept of cBCIs and mental strategies for control.The misalignment in opinions of different groups of stakeholders about cBCIs strengthens the argument for a user-centered design approach in the development of cBCIs and access technology designed for daily life usage.

Ethical issues in brain-computer interface research, development, and dissemination.

The steadily growing field of brain-computer interfacing (BCI) may develop useful technologies, with a potential impact not only on individuals, but also on society as a whole. At the same time, the development of BCI presents significant ethical and legal challenges. In a workshop during the 4th International BCI meeting (Asilomar, California, 2010), six panel members from various BCI laboratories and companies set out to identify and disentangle ethical issues related to BCI use in four case scenarios, which were inspired by current experiences in BCI laboratories. Results of the discussion are reported in this article, touching on topics such as the representation of persons with communication impairments, dealing with technological complexity and moral responsibility in multidisciplinary teams, and managing expectations, ranging from an individual user to the general public. Furthermore, we illustrate that where treatment and research interests conflict, ethical concerns arise. On the basis of the four case scenarios, we discuss salient, practical ethical issues that may confront any member of a typical multidisciplinary BCI team. We encourage the BCI and rehabilitation communities to engage in a dialogue, and to further identify and address pressing ethical issues as they occur in the practice of BCI research and its commercial applications.

Involving Older Adults During COVID-19 Restrictions in Developing an Ecosystem Supporting Active Aging: Overview of Alternative Elicitation Methods and Common Requirements From Five European Countries.

Background: Information and communication technology solutions have the potential to support active and healthy aging and improve monitoring and treatment outcomes. To make such solutions acceptable, all stakeholders must be involved in the requirements elicitation process. Due to the COVID-19 situation, alternative approaches to commonly used face-to-face methods must often be used. One aim of the current article is to share a unique experience from the Pharaon project where due to the COVID-19 outbreak alternative elicitation methods were used. In addition, an overview of common functional, quality, and emotional goals identified by six pilot sites is presented to complement the knowledge about the needs of older adults. Methods: Originally planned face-to-face co-creation seminars were impossible to carry out, and all pilot sites chose alternative requirements elicitation methods that were most suitable in their situation. The elicited requirements were presented in the form of goal models. In one summary goal model, we provide an overview of common functional, quality, and emotional goals. Results: Different elicitation methods were combined based on the digital literacy of the target group and their access to digital tools. Methods applied without digital technologies were phone interviews, reviews of literature and previous projects, while by means of digital technologies online interviews, online questionnaires, and (semi-)virtual co-creation seminars were conducted. The combination of the methods allowed to involve all planned stakeholders. Virtual and semi-virtual co-creation seminars created collaborative environment comparable to face-to-face situations, while online participation helped to save the time of the participants. The most prevalent functional goals elicited were "Monitor health," "Receive advice," "Receive information." "Easy to use/comfortable," "personalized/tailored," "automatic/smart" were identified as most prevalent quality goals. Most frequently occurring emotional goals were "involved," "empowered," and "informed." Conclusion: There are alternative methods to face-to-face co-creation seminars, which effectively involve older adults and other stakeholders in the requirements elicitation process. Despite the used elicitation method, the requirements can be easily transformed into goal models to present the results in a uniform way. The common requirements across different pilots provided a strong foundation for representing detailed requirements and input for further software development processes.

Brain-Computer Interfaces for Communication: Preferences of Individuals With Locked-in Syndrome.

BACKGROUND: Brain-computer interfaces (BCIs) have been proposed as an assistive technology (AT) allowing people with locked-in syndrome (LIS) to use neural signals to communicate. To design a communication BCI (cBCI) that is fully accepted by the users, their opinion should be taken into consideration during the research and development process. OBJECTIVE: We assessed the preferences of prospective cBCI users regarding (1) the applications they would like to control with a cBCI, (2) the mental strategies they would prefer to use to control the cBCI, and (3) when during their clinical trajectory they would like to be informed about AT and cBCIs. Furthermore, we investigated if individuals diagnosed with progressive and sudden onset (SO) disorders differ in their opinion. METHODS: We interviewed 28 Dutch individuals with LIS during a 3-hour home visit using multiple-choice, ranking, and open questions. During the interview, participants were informed about BCIs and the possible mental strategies. RESULTS: Participants rated (in)direct forms of communication, computer use, and environmental control as the most desired cBCI applications. In addition, active cBCI control strategies were preferred over reactive strategies. Furthermore, individuals with progressive and SO disorders preferred to be informed about AT and cBCIs at the moment they would need it. CONCLUSIONS: We show that individuals diagnosed with progressive and SO disorders preferred, in general, the same applications, mental strategies, and time of information. By collecting the opinion of a large sample of individuals with LIS, this study provides valuable information to stakeholders in cBCI and other AT development.

Hearing the needs of clinical users.

In the past 10 years, brain-computer interfaces (BCIs) for controlling assistive devices have seen tremendous progress with respect to reliability and learnability, and numerous exemplary applications were demonstrated to be controllable by a BCI. Yet, BCI-controlled applications are hardly used for patients with neurologic or neurodegenerative disease. Such patient groups are considered potential end-users of BCI, specifically for replacing or improving lost function. We argue that BCI research and development still faces a translational gap, i.e., the knowledge of how to bring BCIs from the laboratory to the field is insufficient. BCI-controlled applications lack usability and accessibility; both constitute two sides of one coin, which is the key to use in daily life and to prevent nonuse. To increase usability, we suggest rigorously adopting the user-centered design in applied BCI research and development. To provide accessibility, assistive technology (AT) experts, providers, and other stakeholders have to be included in the user-centered process. BCI experts have to ensure the transfer of knowledge to AT professionals, and listen to the needs of primary, secondary, and tertiary end-users of BCI technology. Addressing both, usability and accessibility, in applied BCI research and development will bridge the translational gap and ensure that the needs of clinical end-users are heard, understood, addressed, and fulfilled.

An auditory brain-computer interface (BCI).

Brain-computer interfaces (BCIs) translate brain activity into signals controlling external devices. BCIs based on visual stimuli can maintain communication in severely paralyzed patients, but only if intact vision is available. Debilitating neurological disorders however, may lead to loss of intact vision. The current study explores the feasibility of an auditory BCI. Sixteen healthy volunteers participated in three training sessions consisting of 30 2-3 min runs in which they learned to increase or decrease the amplitude of sensorimotor rhythms (SMR) of the EEG. Half of the participants were presented with visual and half with auditory feedback. Mood and motivation were assessed prior to each session. Although BCI performance in the visual feedback group was superior to the auditory feedback group there was no difference in performance at the end of the third session. Participants in the auditory feedback group learned slower, but four out of eight reached an accuracy of over 70% correct in the last session comparable to the visual feedback group. Decreasing performance of some participants in the visual feedback group is related to mood and motivation. We conclude that with sufficient training time an auditory BCI may be as efficient as a visual BCI. Mood and motivation play a role in learning to use a BCI.

Depression and anxiety in individuals with amyotrophic lateral sclerosis: epidemiology and management.

Amyotrophic lateral sclerosis (ALS) is a fatal motor neuron disease with no curative treatment. Considering the devastating nature of the disease, a high prevalence of depression and anxiety in affected patients would be expected. A review of the literature shows prevalence rates for depression in ALS patients ranging from 0% to 44%, but studies using the structured interview according to DSM-IV criteria find highly consistent rates of 9-11%. Prevalence rates for anxiety in ALS range from 0% to 30%. Depression and anxiety appear to be not always properly addressed aspects of ALS, as there are only a few references in the literature about psychological and pharmacological interventions. Additionally, pharmacological antidepressant therapy is often not continuously monitored and its effectiveness remains unevaluated. A review of the literature and our own experiences show that there is a lack of psychological care and, to our knowledge, there is no specific psychological intervention method for ALS patients. Concerning pharmacological treatment of depression in patients with ALS, there is broad consensus among clinical experts that SSRIs and TCAs are helpful, but there have been no controlled clinical studies of these medications in ALS patients. TCAs can be prescribed if anticholinergic effects are desired simultaneously for treating pseudohypersalivation or insomnia. Anxiety is usually treated with anxiolytics, but again there have been no systematic studies of these drugs in patients with ALS. For psychological intervention we suggest a cognitive behavioural approach, which has to be integrated into an intervention programme that includes teaching of appropriate coping strategies and reappraisal skills and encourages engagement in activities that are still practicable and pleasant. We propose that the treatment of depression and anxiety should involve both cognitive behavioural therapy and pharmacological intervention. Pharmacological treatment should be strictly monitored for effectiveness. To date, no clinical trials are available that would allow us to recommend pharmacotherapy over psychotherapy or vice versa; however, evidence from other patient groups, such as elderly patients diagnosed with major depressive disorder, suggests that a combination of both therapies has the potential to also improve depression and anxiety in patients with ALS.

BCI Meeting 2005--workshop on signals and recording methods.

This paper describes the highlights of presentations and discussions during the Third International BCI Meeting in a workshop that evaluated potential brain-computer interface (BCI) signals and currently available recording methods. It defined the main potential user populations and their needs, addressed the relative advantages and disadvantages of noninvasive and implanted (i.e., invasive) methodologies, considered ethical issues, and focused on the challenges involved in translating BCI systems from the laboratory to widespread clinical use. The workshop stressed the critical importance of developing useful applications that establish the practical value of BCI technology.

Classifying EEG and ECoG signals without subject training for fast BCI implementation: comparison of nonparalyzed and completely paralyzed subjects.

We summarize results from a series of related studies that aim to develop a motor-imagery-based brain-computer interface using a single recording session of electroencephalogram (EEG) or electrocorticogram (ECoG) signals for each subject. We apply the same experimental and analytical methods to 11 nonparalysed subjects (eight EEG, three ECoG), and to five paralyzed subjects (four EEG, one ECoG) who had been unable to communicate for some time. While it was relatively easy to obtain classifiable signals quickly from most of the nonparalyzed subjects, it proved impossible to classify the signals obtained from the paralyzed patients by the same methods. This highlights the fact that though certain BCI paradigms may work well with healthy subjects, this does not necessarily indicate success with the target user group. We outline possible reasons for this failure to transfer.

Epidural electrocorticography for monitoring of arousal in locked-in state.

Electroencephalography (EEG) often fails to assess both the level (i.e., arousal) and the content (i.e., awareness) of pathologically altered consciousness in patients without motor responsiveness. This might be related to a decline of awareness, to episodes of low arousal and disturbed sleep patterns, and/or to distorting and attenuating effects of the skull and intermediate tissue on the recorded brain signals. Novel approaches are required to overcome these limitations. We introduced epidural electrocorticography (ECoG) for monitoring of cortical physiology in a late-stage amytrophic lateral sclerosis patient in completely locked-in state (CLIS). Despite long-term application for a period of six months, no implant-related complications occurred. Recordings from the left frontal cortex were sufficient to identify three arousal states. Spectral analysis of the intrinsic oscillatory activity enabled us to extract state-dependent dominant frequencies at <4, ~7 and ~20 Hz, representing sleep-like periods, and phases of low and elevated arousal, respectively. In the absence of other biomarkers, ECoG proved to be a reliable tool for monitoring circadian rhythmicity, i.e., avoiding interference with the patient when he was sleeping and exploiting time windows of responsiveness. Moreover, the effects of interventions addressing the patient's arousal, e.g., amantadine medication, could be evaluated objectively on the basis of physiological markers, even in the absence of behavioral parameters. Epidural ECoG constitutes a feasible trade-off between surgical risk and quality of recorded brain signals to gain information on the patient's present level of arousal. This approach enables us to optimize the timing of interactions and medical interventions, all of which should take place when the patient is in a phase of high arousal. Furthermore, avoiding low-responsiveness periods will facilitate measures to implement alternative communication pathways involving brain-computer interfaces (BCI).

Assessing attention and cognitive function in completely locked-in state with event-related brain potentials and epidural electrocorticography.

OBJECTIVE: Patients in the completely locked-in state (CLIS), due to, for example, amyotrophic lateral sclerosis (ALS), no longer possess voluntary muscle control. Assessing attention and cognitive function in these patients during the course of the disease is a challenging but essential task for both nursing staff and physicians. APPROACH: An electrophysiological cognition test battery, including auditory and semantic stimuli, was applied in a late-stage ALS patient at four different time points during a six-month epidural electrocorticography (ECoG) recording period. Event-related cortical potentials (ERP), together with changes in the ECoG signal spectrum, were recorded via 128 channels that partially covered the left frontal, temporal and parietal cortex. MAIN RESULTS: Auditory but not semantic stimuli induced significant and reproducible ERP projecting to specific temporal and parietal cortical areas. N1/P2 responses could be detected throughout the whole study period. The highest P3 ERP was measured immediately after the patient's last communication through voluntary muscle control, which was paralleled by low theta and high gamma spectral power. Three months after the patient's last communication, i.e., in the CLIS, P3 responses could no longer be detected. At the same time, increased activity in low-frequency bands and a sharp drop of gamma spectral power were recorded. SIGNIFICANCE: Cortical electrophysiological measures indicate at least partially intact attention and cognitive function during sparse volitional motor control for communication. Although the P3 ERP and frequency-specific changes in the ECoG spectrum may serve as indicators for CLIS, a close-meshed monitoring will be required to define the exact time point of the transition.

The Asilomar Survey: Stakeholders' Opinions on Ethical Issues Related to Brain-Computer Interfacing.

Brain-Computer Interface (BCI) research and (future) applications raise important ethical issues that need to be addressed to promote societal acceptance and adequate policies. Here we report on a survey we conducted among 145 BCI researchers at the 4th International BCI conference, which took place in May-June 2010 in Asilomar, California. We assessed respondents' opinions about a number of topics. First, we investigated preferences for terminology and definitions relating to BCIs. Second, we assessed respondents' expectations on the marketability of different BCI applications (BCIs for healthy people, BCIs for assistive technology, BCIs-controlled neuroprostheses and BCIs as therapy tools). Third, we investigated opinions about ethical issues related to BCI research for the development of assistive technology: informed consent process with locked-in patients, risk-benefit analyses, team responsibility, consequences of BCI on patients' and families' lives, liability and personal identity and interaction with the media. Finally, we asked respondents which issues are urgent in BCI research.

Fragmentation of slow wave sleep after onset of complete locked-in state.

Locked-in syndrome (LIS) as a result of brainstem lesions or progressive neurodegenerative disorders, such as amyotrophic lateral sclerosis (ALS), is a severe medical condition in which a person is fully conscious but unable to move or talk. LIS can transition into complete locked-in syndrome (CLIS) in which residual abilities to communicate through muscle twitches are entirely lost. It is unknown how CLIS affects circadian rhythm and sleep/wake patterns. Here we report a 39-year-old ALS patient who transitioned from LIS to CLIS while brain activity was continuously recorded using electrocorticography (ECoG) over one month. While we found no circadian rhythm in heart rate and body temperature, transition into CLIS was associated with increased fragmentation of slow wave sleep (SWS) across the day. Total time in SWS did not change. SWS fragmentation might reflect progressive circadian system impairment and should be considered as a factor further limiting communication capabilities in these patients.

A Dry EEG-System for Scientific Research and Brain-Computer Interfaces.

Although it ranks among the oldest tools in neuroscientific research, electroencephalography (EEG) still forms the method of choice in a wide variety of clinical and research applications. In the context of brain-computer interfacing (BCI), EEG recently has become a tool to enhance human-machine interaction. EEG could be employed in a wider range of environments, especially for the use of BCI systems in a clinical context or at the homes of patients. However, the application of EEG in these contexts is impeded by the cumbersome preparation of the electrodes with conductive gel that is necessary to lower the impedance between electrodes and scalp. Dry electrodes could provide a solution to this barrier and allow for EEG applications outside the laboratory. In addition, dry electrodes may reduce the time needed for neurological exams in clinical practice. This study evaluates a prototype of a three-channel dry electrode EEG system, comparing it to state-of-the-art conventional EEG electrodes. Two experimental paradigms were used: first, event-related potentials (ERP) were investigated with a variant of the oddball paradigm. Second, features of the frequency domain were compared by a paradigm inducing occipital alpha. Furthermore, both paradigms were used to evaluate BCI classification accuracies of both EEG systems. Amplitude and temporal structure of ERPs as well as features in the frequency domain did not differ significantly between the EEG systems. BCI classification accuracies were equally high in both systems when the frequency domain was considered. With respect to the oddball classification accuracy, there were slight differences between the wet and dry electrode systems. We conclude that the tested dry electrodes were capable to detect EEG signals with good quality and that these signals can be used for research or BCI applications. Easy to handle electrodes may help to foster the use of EEG among a wider range of potential users.

The influence of psychological state and motivation on brain-computer interface performance in patients with amyotrophic lateral sclerosis - a longitudinal study.

The current study investigated the effects of psychological well-being measured as quality of life (QoL), depression, current mood and motivation on brain-computer interface (BCI) performance in amyotrophic lateral sclerosis (ALS). Six participants with most advanced ALS were trained either for a block of 20 sessions with a BCI based on sensorimotor rhythms (SMR) or a block of 10 sessions with a BCI based on event-related potentials, or both. Questionnaires assessed QoL and severity of depressive symptoms before each training block and mood and motivation before each training session. The SMR-BCI required more training than the P300-BCI. The information transfer rate was higher with the P300-BCI (3.25 bits/min) than with the SMR-BCI (1.16 bits/min). Mood and motivation were related to the number of BCI sessions. Motivational factors, specifically challenge and mastery confidence, were positively related to BCI performance (controlled for the number of sessions) in tow participants, while incompetence fear was negatively related with performance in one participant. BCI performance was not related to motivational factors in three other participants nor to mood in any of the six participants. We conclude that motivational factors may be related to BCI performance in individual subjects and suggest that motivational factors and well-being should be assessed in standard BCI protocols. We also recommend using P300-based BCI as first choice in severely paralyzed patients who present with a P300 evoked potential.

A note on ethical aspects of BCI.

This paper focuses on ethical aspects of BCI, as a research and a clinical tool, that are challenging for practitioners currently working in the field. Specifically, the difficulties involved in acquiring informed consent from locked-in patients are investigated, in combination with an analysis of the shared moral responsibility in BCI teams, and the complications encountered in establishing effective communication with media.

A brain-computer interface controlled auditory event-related potential (p300) spelling system for locked-in patients.

Using brain-computer interfaces (BCI) humans can select letters or other targets on a computer screen without any muscular involvement. An intensively investigated kind of BCI is based on the recording of visual event-related brain potentials (ERP). However, some severely paralyzed patients who need a BCI for communication have impaired vision or lack control of gaze movement, thus making a BCI depending on visual input no longer feasible. In an effort to render the ERP-BCI usable for this group of patients, the ERP-BCI was adapted to auditory stimulation. Letters of the alphabet were assigned to cells in a 5 x 5 matrix. Rows of the matrix were coded with numbers 1 to 5, and columns with numbers 6 to 10, and the numbers were presented auditorily. To select a letter, users had to first select the row and then the column containing the desired letter. Four severely paralyzed patients in the end-stage of a neurodegenerative disease were examined. All patients performed above chance level. Spelling accuracy was significantly lower with the auditory system as compared with a similar visual system. Patients reported difficulties in concentrating on the task when presented with the auditory system. In future studies, the auditory ERP-BCI should be adjusted by taking into consideration specific features of severely paralyzed patients, such as reduced attention span. This adjustment in combination with more intensive training will show whether an auditory ERP-BCI can become an option for visually impaired patients.