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1.
Artigo em Inglês | MEDLINE | ID: mdl-35445450

RESUMO

AIMS: Parents of children with spinal muscular atrophy (SMA) often struggle with the all-consuming nature of the demands of caring for a child with substantial physical needs. Our aim was to explore experiences, challenges and needs of parents of a child with SMA in a COVID-19 pandemic situation. METHOD: Nineteen parents of 21 children (15 months to 13 years of age) with SMA types 1-3 participated in semi-structured interviews in June to July 2020. The interviews were analysed using inductive thematic analysis. RESULTS: Parents mentioned the protection of the health and well-being of the child as the central perspective and driving force during the COVID-19 pandemic. Three subthemes were identified: (1) responsibility, (2) balancing vulnerability and resilience and (3) (in)security. Some parents focused on the positive aspects during the lockdown, such as continuation of nusinersen treatment and family life. Some parents described helpful and positive cognitions to cope with the situation. In general, parents described a need for information with regard to COVID-19 and their child with SMA and a need for discussing their dilemmas and insecurities with a healthcare professional. INTERPRETATION: Parents put the health and well-being of their children first during the pandemic. From this study, we learned that parents of children with SMA need information and value direct contact with a healthcare professional to share their dilemmas and insecurities. The dialogue can help to empower parents in the conflicts and decisions they have to make during a pandemic.

2.
Int J Geriatr Psychiatry ; 37(5)2022 Mar 14.
Artigo em Inglês | MEDLINE | ID: mdl-35362219

RESUMO

OBJECTIVES: Amyotrophic Lateral Sclerosis (ALS) is a systemic and terminal disorder of the central nervous system which causes paralysis of limbs, respiratory and bulbar muscles, impacting on physical, communication, cognitive and behavioural functioning. Informal caregivers play a key role in the care of people with ALS. This study aimed to explore experiences of burden along with any beneficial aspects of caregiving in ALS. An understanding of both burden and benefit is important to support the informal caregiver and the person with ALS. METHODS/DESIGN: This exploratory mixed methods study characterizes two groups of informal caregivers in Ireland (n = 76) and the Netherlands (n = 58). In a semi-structured interview, quantitative data were collected in the form of standardized measures assessing psychological distress, quality of life and burden. Qualitative data were collected from an open ended question, in which caregivers identified positive aspects in their caregiving experience. These data types were purposefully mixed in the analysis and interpretation stages, to provide a greater depth of evidence through diverse research lenses. RESULTS: The caregiver cohorts were predominantly female (69%) and spouse/partners (84%) of the person with ALS. Greater levels of self-assessed burden were found among the caregivers in the Netherlands (p < 0.05), and higher levels of quality of life among the cohort from Ireland (p < 0.05). Themes generated through qualitative analysis identified caregiver satisfaction, ability to meet the patient's needs and the (re) evaluation of meaning and existential aspects of life as positive aspects of caregiving. Existential factors were identified frequently by the caregivers in Ireland, and personal satisfaction and meeting their care recipient's needs by caregivers in the Netherlands. Three percent of all respondents reported there was nothing positive about caregiving. CONCLUSIONS: Based on our findings, we suggest that both burden and the presence of positive factors should be evaluated and monitored. The possibility of concurrent positive and challenging experiences should be considered in the design and delivery of supportive interventions for informal caregivers.

3.
BMC Psychol ; 10(1): 72, 2022 Mar 17.
Artigo em Inglês | MEDLINE | ID: mdl-35300726

RESUMO

BACKGROUND: Amyotrophic lateral sclerosis (ALS), progressive muscular atrophy (PMA) and primary lateral sclerosis (PLS), together referred to as ALS, are life-limiting diagnoses affecting not only patients but also the families surrounding them, especially when dependent children are involved. Despite previous research highlighting the vulnerability of children in these families, they are, as yet, often overlooked in healthcare. Efforts are needed to better support children in families living with ALS, both directly and through strengthening parents in their parental role. This study sought to gain a better understanding of parental and children's experiences, struggles and support needs in families living with ALS. METHODS: Semi-structured interviews were conducted with 8 parents with ALS, 13 well parents and 15 children, together representing 17 families. Interview data were analyzed using qualitative content analysis. RESULTS: Three major themes were identified relating to (1) ALS-related transformations in families' homes, activities, roles and relationships, that trigger (2) distress among families, which, in turn, evokes (3) emotional, psychological, educational and practical support needs. For emotional and practical support, parents and children mainly rely on their own family and social network, whereas they seek educational and psychological support from healthcare professionals. CONCLUSIONS: Our findings imply that ALS care professionals may foster family adjustment to living with ALS, most notably through encouraging parents to engage in a dialogue with their children about the many transformations, struggles and needs imposed by ALS and teaching them how to start the dialogue.


Assuntos
Esclerose Amiotrófica Lateral , Esclerose Amiotrófica Lateral/psicologia , Criança , Família/psicologia , Pessoal de Saúde/psicologia , Humanos , Pais/psicologia , Pesquisa Qualitativa
4.
J Neurol ; 2022 Feb 07.
Artigo em Inglês | MEDLINE | ID: mdl-35129626

RESUMO

BACKGROUND: Home-monitoring of spirometry has the potential to improve care for patients with a motor neuron disease (MND) by enabling early detection of respiratory dysfunction and reducing travel burden. Our aim was to evaluate the validity and feasibility of home-monitoring vital capacity (VC) in patients with MND. METHODS: We included 33 patients with amyotrophic lateral sclerosis, progressive muscular atrophy or primary lateral sclerosis who completed a 12-week home-monitoring protocol, consisting of 4-weekly unsupervised home assessments of VC and a functional rating scale. At baseline, during a home visit, patients/caregivers were trained in performing a VC test, and the investigator performed a supervised VC test, which was repeated at final follow-up during a second home visit. Validity of the unsupervised VC tests was evaluated by the differences between supervised and unsupervised VC tests, and through Bland-Altman 95% limits-of-agreement. Feasibility was assessed by means of a survey of user-experiences. RESULTS: The 95% limits-of-agreement were [- 14.3; 11.7] %predicted VC, and 88% of unsupervised VC tests fell within 10%predicted of supervised VC. 88% of patients experienced VC testing as easy and not burdensome, however, 15% patients did not think their VC test was performed as well as in the clinic. 94% of patients would like home-monitoring of VC in MND care. DISCUSSION: Unsupervised VC testing at home, with prior face-to-face training, is a valid and time-efficient method for the remote monitoring of respiratory function, and well-accepted by patients with MND and their caregivers.

5.
Artigo em Inglês | MEDLINE | ID: mdl-34949141

RESUMO

Objective: Uniform data collection is fundamental for multicentre clinical trials. We aim to determine the variability, between ALS trial centers, in the prevalence of unexpected or implausible improvements in the revised ALS functional rating scale (ALSFRS-R) score, and its associations with individual patient and item characteristics.Methods: We used data from two multicentre studies to estimate the prevalence of an unexpected increase or implausible improvement in the ALSFRS-R score, defined as an increase of 5 points or more between two consecutive, monthly visits. For each patient with a 5-point or more increase, we evaluated the individual contribution of each ALSFRS-R item.Results: Longitudinal ALSFRS-R scores, originating from 114 trial centers enrolling a total of 1,240 patients, were analyzed. A 5-point or more increase in ALSFRS-R total score was found in 151 (12.2%) patients, with prevalence per study center ranging from 0% to 83%. Bulbar onset, faster disease progression at enrollment, and a lower ALSFRS-R score at baseline were associated with a sudden 5-point or more increase in the ALSFRS-R total score. ALSFRS-R items 2 (saliva), 9 (stairs), 10 (dyspnea), and 11 (orthopnea) were the primary drivers when a 5-point or more increase occurred.Conclusions: Sudden 5-point or more increases in ALSFRS-R total scores between two consecutive visits are relatively common. These sudden increases were not found to occur with equal frequency in trial centers; which underscores the need for amending existing standard operating procedures toward a universal version and monitoring of data quality during the study, in multicentre research.

6.
Brain Sci ; 11(12)2021 Nov 30.
Artigo em Inglês | MEDLINE | ID: mdl-34942899

RESUMO

The ENCALS survival prediction model offers patients with amyotrophic lateral sclerosis (ALS) the opportunity to receive a personalized prognosis of survival at the time of diagnosis. We explored experiences of patients with ALS, caregivers, and physicians with discussing personalized prognosis through interviews with patients and their caregivers, and in a focus group of physicians. Thematic analysis revealed four themes with seven subthemes; these were recognized by the focus group. First, tailored communication: physician's communication style and information provision mediated emotional impact and increased satisfaction with communication. Second, personal factors: coping style, illness experiences, and information needs affected patient and caregiver coping with the prognosis. Third, emotional impact ranged from happy and reassuring to regret. Fourth, regaining control over the future: participants found it helpful in looking towards the future, and emphasized the importance of quality over quantity of life. Personalized prognosis can be discussed with minimal adverse emotional impact. How it is communicated-i.e., tailored to individual needs-is as important as what is communicated-i.e., a good or poor prognosis. Discussing personalized prognosis may help patients with ALS and their caregivers regain control over the future and facilitate planning of the future (care). For many patients, quality of life matters more than quantity of time remaining.

7.
J Med Internet Res ; 23(9): e28766, 2021 09 22.
Artigo em Inglês | MEDLINE | ID: mdl-34550089

RESUMO

Despite recent and potent technological advances, the real-world implementation of remote digital health technology in the care and monitoring of patients with motor neuron disease has not yet been realized. Digital health technology may increase the accessibility to and personalization of care, whereas remote biosensors could optimize the collection of vital clinical parameters, irrespective of patients' ability to visit the clinic. To facilitate the wide-scale adoption of digital health care technology and to align current initiatives, we outline a road map that will identify clinically relevant digital parameters; mediate the development of benefit-to-burden criteria for innovative technology; and direct the validation, harmonization, and adoption of digital health care technology in real-world settings. We define two key end products of the road map: (1) a set of reliable digital parameters to capture data collected under free-living conditions that reflect patient-centric measures and facilitate clinical decision making and (2) an integrated, open-source system that provides personalized feedback to patients, health care providers, clinical researchers, and caregivers and is linked to a flexible and adaptable platform that integrates patient data in real time. Given the ever-changing care needs of patients and the relentless progression rate of motor neuron disease, the adoption of digital health care technology will significantly benefit the delivery of care and accelerate the development of effective treatments.


Assuntos
Doença dos Neurônios Motores , Tecnologia Biomédica , Cuidadores , Pessoal de Saúde , Humanos , Doença dos Neurônios Motores/diagnóstico , Doença dos Neurônios Motores/terapia , Tecnologia
8.
Artigo em Inglês | MEDLINE | ID: mdl-34486902

RESUMO

OBJECTIVE: To describe current practices and barriers and support needs in gastrostomy indication and decision-making amongst rehabilitation physicians of ALS care teams in the Netherlands. Methods: Cross-sectional online survey of rehabilitation physicians of ALS care teams in the Netherlands. Survey items covered current practices in timing of indication (i.e. indicators and criteria), goals, initiating discussion about gastrostomy, and criteria for preferred method of placement; and barriers and support needs in indication and decision-making. Descriptive analysis was used for quantitative responses, thematic, and content analysis for qualitative data. Results: Twenty-nine physicians (41%) of 27 ALS care teams (71%) responded. Timing of indication: physicians agreed on important indicators but not cutoff values/criteria. Goals: optimizing nutritional status (100%), ensuring safe food-intake (72%), and reducing effort of meals (59%). Initiating discussion about gastrostomy: 52% introduces the topic early after diagnosis, 48% at indication. Criteria for method of placement included physician preference (69%), availability of service (21%), lower complication risk (17%), contraindication (59%), and patient preference (24%). Reported barriers (69% of respondents) were: patient readiness (52%), timing of indication (31%), and organizational barriers (18%). Support needs (62%): evidence-based timing of indication (35%) and tailored patient education (31%). Conclusions: There is practice variation in the timing of first introduction of gastrostomy and preferred method of placement, but agreement on goals and indicators . More evidence on optimal timing of gastrostomy placement is needed. However, until then early and regular discussion of the topic of gastrostomy and better patient information may promote patient readiness and support patient choice.

9.
Brain Sci ; 11(8)2021 Aug 20.
Artigo em Inglês | MEDLINE | ID: mdl-34439713

RESUMO

Amyotrophic lateral sclerosis (ALS)/motor neuron disease (MND) is a systemic and fatal neurodegenerative condition for which there is currently no cure. Informal caregivers play a vital role in supporting the person with ALS, and it is essential to support their wellbeing. This multi-centre, mixed methods descriptive exploratory study describes the complexity of burden and self-defined difficulties as described by the caregivers themselves. Quantitative and qualitative data were collected during face-to-face interviews with informal caregivers from centres in the Netherlands, England, and Ireland. Standardised measures assessed burden, quality of life, and psychological distress; furthermore, an open-ended question was asked about difficult aspects of caregiving. Most caregivers were female, spouse/partners, and lived with the person with ALS for whom they provided care. Significant differences between national cohorts were identified for burden, quality of life, and anxiety. Among the difficulties described were the practical issues associated with the caregiver role and emotional factors such as witnessing a patient's health decline, relationship change, and their own distress. The mixed-methods approach allows for a more nuanced understanding of the burden and difficulties experienced. It is important to generate an evidence base to support the psychosocial wellbeing and brain health of informal caregivers.

10.
Neurorehabil Neural Repair ; 35(2): 131-144, 2021 02.
Artigo em Inglês | MEDLINE | ID: mdl-33410388

RESUMO

Background. Many stroke survivors suffer from leg muscle paresis, resulting in asymmetrical gait patterns, negatively affecting balance control and energy cost. Interventions targeting asymmetry early after stroke may enhance recovery of walking. Objective. To determine the feasibility and preliminary efficacy of up to 10 weeks of gait training assisted by multichannel functional electrical stimulation (MFES gait training) applied to the peroneal nerve and knee flexor or extensor muscle on the recovery of gait symmetry and walking capacity in patients starting in the subacute phase after stroke. Methods. Forty inpatient participants (≤31 days after stroke) were randomized to MFES gait training (experimental group) or conventional gait training (control group). Gait training was delivered in 30-minute sessions each workday. Feasibility was determined by adherence (≥75% sessions) and satisfaction with gait training (score ≥7 out of 10). Primary outcome for efficacy was step length symmetry. Secondary outcomes included other spatiotemporal gait parameters and walking capacity (Functional Gait Assessment and 10-Meter Walk Test). Linear mixed models estimated treatment effect postintervention and at 3-month follow-up. Results. Thirty-seven participants completed the study protocol (19 experimental group participants). Feasibility was confirmed by good adherence (90% of the participants) and participant satisfaction (median score 8). Both groups improved on all outcomes over time. No significant group differences in recovery were found for any outcome. Conclusions. MFES gait training is feasible early after stroke, but MFES efficacy for improving step length symmetry, other spatiotemporal gait parameters, or walking capacity could not be demonstrated. Trial Registration. Netherlands Trial Register (NTR4762).


Assuntos
Terapia por Estimulação Elétrica , Transtornos Neurológicos da Marcha/reabilitação , Espasticidade Muscular/reabilitação , Avaliação de Resultados em Cuidados de Saúde , Reabilitação do Acidente Vascular Cerebral , Acidente Vascular Cerebral/terapia , Idoso , Estudos de Viabilidade , Feminino , Seguimentos , Transtornos Neurológicos da Marcha/etiologia , Humanos , Masculino , Pessoa de Meia-Idade , Espasticidade Muscular/etiologia , Músculo Esquelético , Nervo Fibular , Projetos Piloto , Método Simples-Cego , Acidente Vascular Cerebral/complicações
11.
Muscle Nerve ; 63(5): 678-682, 2021 05.
Artigo em Inglês | MEDLINE | ID: mdl-33501670

RESUMO

INTRODUCTION: In multifocal motor neuropathy (MMN), knowledge about the pattern of treatment response in a wide spectrum of muscle groups, distal as well as proximal, after intravenous immunoglobulin (IVIg) initiation is lacking. METHODS: Hand-held dynamometry data of 11 upper and lower limb muscles, from 47 patients with MMN was reviewed. Linear mixed models were used to determine the treatment response after IVIg initiation and its relationship with initial muscle weakness. RESULTS: All muscle groups showed a positive treatment response after IVIg initiation. Changes in SD scores ranged from +0.1 to +0.95. A strong association between weakness at baseline and the magnitude of the treatment response was found. DISCUSSION: Improved muscle strength in response to IVIg appears not only in distal, but to a similar degree also in proximal muscle groups in MMN, with the largest response in muscle groups that show the greatest initial weakness.


Assuntos
Imunoglobulinas Intravenosas/uso terapêutico , Força Muscular/efeitos dos fármacos , Debilidade Muscular/tratamento farmacológico , Polineuropatias/tratamento farmacológico , Adulto , Feminino , Humanos , Imunoglobulinas Intravenosas/administração & dosagem , Masculino , Pessoa de Meia-Idade , Força Muscular/fisiologia , Dinamômetro de Força Muscular , Debilidade Muscular/fisiopatologia , Polineuropatias/fisiopatologia , Resultado do Tratamento
12.
J Neurol ; 268(5): 1738-1746, 2021 May.
Artigo em Inglês | MEDLINE | ID: mdl-33355879

RESUMO

BACKGROUND: We aimed to determine (1) the test-retest reliability of a newly developed portable fixed dynamometer (PFD) as compared to the hand-held dynamometer (HHD) in patients with motor neuron disease (MND) and (2) the PFD's ability to reduce possible examiner-induced ceiling effects. METHODS: Test-retest reliability of isometric muscle strength of the quadriceps was measured in patients with MND and non-neurological controls using the HHD and PFD. Reliability was estimated by the intraclass correlation coefficient (ICC) and standard error of measurement (SEM) using linear mixed effects models, and the Bland-Altman method of agreement. RESULTS: In total, 45 patients with MND and 43 healthy controls were enrolled in this study. The ICC of the PFD was excellent and similar in both patients and controls (ICC Patients 99.5% vs. ICC Controls 98.6%) with a SEM of 6.2%. A strong examiner-induced ceiling effect in HHD was found when the participant's strength exceeded that of examiner. Employing the PFD increased the range of muscle strength measurements across individuals nearly twofold from 414 to 783 N. CONCLUSIONS: Portable fixed dynamometry may significantly reduce examiner-induced ceiling effects, optimize the standardization of muscle strength testing, and maximize reliability. Ultimately, PFD may improve the delivery of care due to its potential for unsupervised, home-based assessments and reduce the burden to the patient of participating in clinical trials for MND or other neuromuscular diseases.


Assuntos
Contração Isométrica , Doença dos Neurônios Motores , Humanos , Força Muscular , Dinamômetro de Força Muscular , Reprodutibilidade dos Testes
13.
BMC Neurol ; 20(1): 446, 2020 Dec 14.
Artigo em Inglês | MEDLINE | ID: mdl-33308184

RESUMO

BACKGROUND: Personalized ENCALS survival prediction model reliably estimates the personalized prognosis of patients with amyotrophic lateral sclerosis. Concerns were raised on discussing personalized prognosis without causing anxiety and destroying hope. Tailoring communication to patient readiness and patient needs mediates the impact of prognostic disclosure. We developed a communication guide to support physicians in discussing personalized prognosis tailored to individual needs and preferences of people with ALS and their families. METHODS: A multidisciplinary working group of neurologists, rehabilitation physicians, and healthcare researchers A) identified relevant topics for guidance, B) conducted a systematic review on needs of patients regarding prognostic discussion in life-limiting disease, C) drafted recommendations based on evidence and expert opinion, and refined and finalized these recommendations in consensus rounds, based on feedback of an expert advisory panel (patients, family member, ethicist, and spiritual counsellor). RESULTS: A) Topics identified for guidance were 1) filling in the ENCALS survival model, and interpreting outcomes and uncertainty, and 2) tailoring discussion to individual needs and preferences of patients (information needs, role and needs of family, severe cognitive impairment or frontotemporal dementia, and non-western patients). B) 17 studies were included in the systematic review. C) Consensus procedures on drafted recommendations focused on selection of outcomes, uncertainty about estimated survival, culturally sensitive communication, and lack of decisional capacity. Recommendations for discussing the prognosis include the following: discuss prognosis based on the prognostic groups and their median survival, or, if more precise information is desired, on the interquartile range of the survival probability. Investigate needs and preferences of the patients and their families for prognostic disclosure, regardless of cultural background. If the patient does not want to know their prognosis, with patient permission discuss the prognosis with their family. If the patient is judged to lack decisional capacity, ask the family if they want to discuss the prognosis. Tailor prognostic disclosure step by step, discuss it in terms of time range, and emphasize uncertainty of individual survival time. CONCLUSION: This communication guide supports physicians in tailoring discussion of personalized prognosis to the individual needs and preferences of people with ALS and their families.


Assuntos
Esclerose Amiotrófica Lateral , Comunicação , Educação de Pacientes como Assunto/métodos , Relações Médico-Paciente , Revelação da Verdade , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Prognóstico
14.
J Neurol ; 267(11): 3310-3318, 2020 Nov.
Artigo em Inglês | MEDLINE | ID: mdl-32577867

RESUMO

BACKGROUND: Poor monitoring of respiratory function may lead to late initiation of non-invasive ventilation (NIV) in patients with motor neuron diseases (MND). Monitoring could be improved by remotely assessing hypoventilation symptoms between clinic visits. We aimed to determine which patient-reported hypoventilation symptoms are best for screening reduced respiratory function in patients with MND, and compared them to the respiratory domain of the amyotrophic lateral sclerosis functional rating scale (ALSFRS-R). METHODS: This prospective multi-center study included 100 patients with MND, who were able to perform a supine vital capacity test. Reduced respiratory function was defined as a predicted supine vital capacity ≤ 80%. We developed a 14-item hypoventilation symptom questionnaire (HYSQ) based on guidelines, expert opinion and think-aloud interviews with patients. Symptoms of the HYSQ were related to dyspnea, sleep quality, sleepiness/fatigue and pneumonia. The diagnostic performances of these symptoms and the ALSFRS-R respiratory domain were determined from the receiver operating characteristic (ROC) curves, area under the curve (AUC), sensitivity, specificity, predictive values and accuracy. RESULTS: Dyspnea-related symptoms (dyspnea while eating/talking, while lying flat and during light activity) were combined into the MND Dyspnea Scale (MND-DS). ROC curves showed that the MND-DS had the best diagnostic performance, with the highest AUC = 0.72, sensitivity = 75% and accuracy = 71%. Sleep-quality symptoms, sleepiness/fatigue-related symptoms and the ALSFRS-R respiratory domain showed weak diagnostic performance. CONCLUSION: The diagnostic performance of the MND-DS was better than the respiratory domain of the ALSFRS-R for screening reduced respiratory function in patients with MND, and is, therefore, the preferred method for (remotely) monitoring respiratory function.


Assuntos
Esclerose Amiotrófica Lateral , Doença dos Neurônios Motores , Insuficiência Respiratória , Esclerose Amiotrófica Lateral/complicações , Esclerose Amiotrófica Lateral/diagnóstico , Dispneia/diagnóstico , Dispneia/etiologia , Humanos , Doença dos Neurônios Motores/complicações , Doença dos Neurônios Motores/diagnóstico , Medidas de Resultados Relatados pelo Paciente , Estudos Prospectivos
15.
Artigo em Inglês | MEDLINE | ID: mdl-32362155

RESUMO

Objective: To evaluate whether a blended (face-to-face and online) psychosocial support program for caregivers of patients with Amyotrophic Lateral Sclerosis (ALS) and Progressive Muscular Atrophy (PMA), aimed at enhancing feeling of control over caregiving, reduces psychological distress. Methods: A randomized controlled trial using a wait-list control design was conducted. Caregiver-patient dyads were randomly assigned to either the support program (n = 74) or to a wait-list control group (n = 74). The support program, based on Acceptance and Commitment Therapy, consists of 1 face-to-face contact, 6 online guided modules and 1 telephone contact. Participants filled in questionnaires at baseline, 3 and 6 months. Caregivers' feeling of control over caregiving was assessed using two self-efficacy measures. Primary outcome was caregivers' psychological distress. Secondary outcomes included caregiver burden, caregiver quality of life and patients' quality of life and psychological distress. Intention-to-treat and per-protocol analyses were performed using linear mixed models. Results: The support program had no effect on the primary or secondary outcomes, despite a significant positive effect on the intervening variable self-efficacy with regard to control over thoughts. Almost half of the caregivers did not complete the intervention with the most frequently reported reason being lack of time. Caregivers who completed the intervention evaluated the support program positively. Conclusions: The support program did not reduce distress of partners of patients with ALS/PMA but may be beneficial by increasing feeling of control over the caregiving situation. The high level of intervention drop outs may have limited the ability to detect an intervention effect.Trial registration: Netherlands Trial Registry NTR5734, registered 28 March 2016.


Assuntos
Terapia de Aceitação e Compromisso , Esclerose Amiotrófica Lateral , Atrofia Muscular Espinal , Esclerose Amiotrófica Lateral/terapia , Cuidadores , Humanos , Sistemas de Apoio Psicossocial , Qualidade de Vida
16.
Artigo em Inglês | MEDLINE | ID: mdl-32003245

RESUMO

Objective: To evaluate the use of telehealth as part of specialized care for patients with amyotrophic lateral sclerosis (ALS) and the user experiences of patients and healthcare professionals. Methods: Fifty patients with ALS were recruited from a single specialist center and used telehealth, consisting of an ALS-app for self-monitoring and messaging, alerts for symptom-worsening, and nurse practitioner follow-up. Patients self-monitored their well-being (daily report), body weight (weekly) and functional status (monthly). The use of the telehealth service was evaluated through adoption rate, dropout rate and adherence to self-monitoring. User-experiences were collected through online surveys among 23 patients and nine healthcare professionals, and interviews with 12 patients. Results: The adoption rate was 80%, dropout rate 4% and median follow-up was 11 months. Good adherence was seen in 49% of patients for well-being, 83% for body weight and 87% for functional assessment. For patients who discontinued using telehealth due to the end-of-life phase, median time between last measurement and death was 19 days. The majority of patients experienced using telehealth as easy, helpful, not burdensome, and reported satisfaction with flexible clinic visits and the continuity of care. Healthcare professionals reported that telehealth was of added value in ALS-care. Conclusions: ALS-care supplemented by home-monitoring and nurse practitioner follow-up was shown to be suitable and widely accepted by patients and healthcare professionals in our ALS clinic. Success factors were low self-monitoring burden, a user-friendly platform and the provision of personalized feedback. Further research is needed to replicate these findings in other ALS clinics.


Assuntos
Esclerose Amiotrófica Lateral/terapia , Tutoria/métodos , Monitorização Fisiológica/métodos , Telemedicina/métodos , Idoso , Esclerose Amiotrófica Lateral/psicologia , Estudos de Coortes , Estudos de Viabilidade , Feminino , Seguimentos , Humanos , Masculino , Pessoa de Meia-Idade , Monitorização Fisiológica/psicologia , Profissionais de Enfermagem/psicologia , Satisfação do Paciente , Estudos Prospectivos
17.
Artigo em Inglês | MEDLINE | ID: mdl-31878794

RESUMO

Objective: We aimed to provide an overview of telehealth used in the care for patients with amyotrophic lateral sclerosis (ALS), and identify the barriers to and facilitators of its implementation. Methods: We searched Pubmed and Embase to identify relevant articles. Full-text articles with original research reporting on the use of telehealth in ALS care, were included. Data were synthesized using the Consolidation Framework for Implementation Research. Two authors independently screened articles based on the inclusion criteria. Results: Sixteen articles were included that investigated three types of telehealth: Videoconferencing, home-based self-monitoring and remote NIV monitoring. Telehealth was mainly used by patients with respiratory impairment and focused on monitoring respiratory function. Facilitators for telehealth implementation were a positive attitude of patients (and caregivers) toward telehealth and the provision of training and ongoing support. Healthcare professionals were more likely to have a negative attitude toward telehealth, due to the lack of personal evaluation/contact and technical issues; this was a known barrier. Other important barriers to telehealth were lack of reimbursement and cost-effectiveness analyses. Barriers and facilitators identified in this review correspond to known determinants found in other healthcare settings. Conclusions: Our findings show that telehealth in ALS care is well-received by patients and their caregivers. Healthcare professionals, however, show mixed experiences and perceive barriers to telehealth use. Challenges related to finance and legislation may hinder telehealth implementation in ALS care. Future research should report the barriers and facilitators of implementation and determine the cost-effectiveness of telehealth.


Assuntos
Esclerose Amiotrófica Lateral/terapia , Cuidadores , Aceitação pelo Paciente de Cuidados de Saúde , Telemedicina/métodos , Comunicação por Videoconferência , Esclerose Amiotrófica Lateral/psicologia , Cuidadores/psicologia , Cuidadores/tendências , Pessoal de Saúde/psicologia , Pessoal de Saúde/tendências , Humanos , Aceitação pelo Paciente de Cuidados de Saúde/psicologia , Insuficiência Respiratória/psicologia , Insuficiência Respiratória/terapia , Telemedicina/tendências , Comunicação por Videoconferência/tendências
18.
Psychol Health Med ; 25(3): 319-330, 2020 03.
Artigo em Inglês | MEDLINE | ID: mdl-31747791

RESUMO

Caregivers of Amyotrophic Lateral Sclerosis (ALS) and Progressive Muscular Atrophy (PMA) patients often experience psychological distress. Yet, it is unclear which factors explain the variance in psychological distress. This study seeks to evaluate how care demands and perceived control over caregiving influence psychological distress using moderation and mediation analysis. Data were collected as part of a RCT and 148 partners of patients with ALS or PMA were included. Psychological distress was assessed using the Hospital Anxiety and Depression Scale. Care demands were operationalized as physical functioning (Amyotrophic Lateral Sclerosis Functional Rating Scale-Revised) and behavioural changes of the patient (Amyotrophic Lateral Sclerosis-Frontotemporal Dementia-Questionnaire). Perceived control over caregiving was assessed using items adapted from the Job Content Questionnaire. Results showed that more behavioural changes and lower perceived control over caregiving were associated with higher levels of psychological distress in caregivers. Patients' physical functioning was not significantly related to caregivers' psychological distress. No moderation or mediation effects were found of perceived control over caregiving on the relationship between demand and psychological distress. Monitoring, psychoeducation and caregiver support with regard to behavioural changes in patients, seem to be important for the wellbeing of caregivers. Caregivers' perceived control might be a target for future interventions.


Assuntos
Esclerose Amiotrófica Lateral/enfermagem , Cuidadores/psicologia , Atrofia Muscular Espinal/enfermagem , Angústia Psicológica , Cônjuges/psicologia , Estresse Psicológico/psicologia , Idoso , Feminino , Humanos , Masculino , Pessoa de Meia-Idade
19.
Dev Med Child Neurol ; 62(1): 21-27, 2020 01.
Artigo em Inglês | MEDLINE | ID: mdl-31334566

RESUMO

AIM: To evaluate and synthesize the evidence for effects of upper extremity surgery (UES) on activities and participation of children and adolescents with cerebral palsy (CP). METHOD: The databases MEDLINE, Embase, and PsycINFO were searched for publications up to September 2018. Studies included were comparative studies with or without concurrent comparison groups or case series with pretest/posttest outcomes with a minimal sample size of 10 participants; those that reported the effects of UES with a follow-up time of at least 5 months; those including patients diagnosed with CP aged up to 20 years; and those that used a validated activity-based instrument. Risk of bias was assessed using the ROBINS-I (Risk Of Bias In Non-randomised Studies - of Interventions) tool and quality assessment was performed using the Grading of Recommendations Assessment, Development and Evaluation. RESULTS: Twelve studies, involving 310 children and adolescents, were included. The ability and perception of the patient to use the hand(s) and perform activities (measured with the Shriners Hospital Upper Extremity Evaluation, Assisting Hand Assessment, and House Functional Classification) improved significantly after UES. The quality of evidence was very low for each of the activity outcomes of interest. INTERPRETATION: The very low evidence prohibits recommendations on the use of UES to guide clinical practice. More high-quality comparative studies are needed to obtain better insight into the effects of UES on activities and participation. WHAT THIS PAPER ADDS: Low quality of evidence for effects of upper extremity surgery (UES) on activities and participation. Limited evidence for improvement in activities and participation after UES.


EFECTOS DE LA CIRUGÍA DE LA EXTREMIDAD SUPERIOR SOBRE LAS ACTIVIDADES Y LA PARTICIPACIÓN DE NIÑOS CON PARÁLISIS CEREBRAL: UNA REVISIÓN SISTEMÁTICA: OBJETIVO: Evaluar y sintetizar la evidencia de los efectos de la cirugía de extremidades superiores (UES) sobre las actividades y la participación de niños y adolescentes con parálisis cerebral (PC). MÉTODO: En las bases de datos MEDLINE, Embase y PsycINFO se buscaron publicaciones hasta septiembre de 2018. Los estudios incluidos fueron estudios comparativos con o sin grupos de comparación concurrentes o series de casos con resultados de prueba previa / prueba posterior con un tamaño de muestra mínimo de 10 participantes; aquellos que informaron los efectos de UES con un tiempo de seguimiento de al menos 5 meses; aquellos que incluyen pacientes diagnosticados con PC de hasta 20 años; y aquellos que utilizaron un instrumento validado basado en actividades. El riesgo de sesgo se evaluó mediante la herramienta ROBINS-I (Riesgo de sesgo en estudios no aleatorios - Intervenciones) y la evaluación de la calidad se realizó mediante la evaluación, desarrollo y evaluación de la calificación de las recomendaciones. RESULTADOS: Se incluyeron 12 estudios con 310 niños y adolescentes. La capacidad y la percepción del paciente para usar la/s mano/s, y realizar las actividades (medidas con la Evaluación de la extremidad superior del Hospital Shriners, la Evaluación de la mano auxiliar y la Clasificación funcional de la casa) mejoraron significativamente después de la UES. La calidad de la evidencia fue muy baja para cada uno de los resultados de actividad de interés. INTERPRETACIÓN: La evidencia muy baja previene realizar recomendaciones sobre el uso de UES para guiar la práctica clínica. Se necesitan más estudios comparativos de alta calidad para obtener una mejor comprensión de los efectos de UES en las actividades y la participación.


EFEITOS DE CIRURGIA DO MEMBRO SUPERIOR EM ATIVIDADES E PARTICIPAÇÃO DE CRIANÇAS COM PARALISIA CEREBRAL: UMA REVISÃO SISTEMÁTICA: OBJETIVO: Avaliar e sintetizar a evidência dos efeitos de cirurgia do membro superior (CMS) sobre as atividades e participação de crianças com paralisia cerebral (PC). MÉTODO: As bases de dados MEDLINE, Embase, e PsycINFO foram pesquisadas quanto a publicações até setembro de 2018. Foram incluídos estudos comparativos com ou sem grupos de comparação concorrentes ou séries de casos com resultados pré/pós-teste com um tamanho amostral mínimo de 10 participantes; aqueles que reportaram os efeitos de CMS com tempo de acompanhamento de pelo menos 5 meses; aqueles incluindo pacientes com diagnóstico de PC e até 20 anos de idade; e aqueles que usaram um instrumento válido para avaliar atividade. O risco de viés foi avaliado usando o instrumento RVENA-I (Risco de viés em estudos não aleatorizados - de intervenções) e a avaliação da qualidade foi realizada com a Pontuação de Recomendações, Avaliação, Desenvolvimento e Mensuração. RESULTADOS: Doze estudos, envolvendo 310 crianças e adolescentes, foram incluídos. A capacidade e percepção do paciente em utilizar as mãos e realizar atividades (mensuradas com a Avaliação do Hospital Shriners para a extremidade superior, a Avaliação da Mão Auxiliar, e a Classificação Funcional de House) melhoraram significantemente após CMS. A qualidade da evidência foi muito baixa para os resultados de atividade de interesse. INTERPRETAÇÃO: A evidência muito baixa proíbe recomendações sobre o uso de CMS para guiar a prática clínica. Mais estudos comparativos de alta qualidade são necessários para obter mais informações a respeito dos efeitos de CMS nas atividades e participação.


Assuntos
Atividades Cotidianas , Paralisia Cerebral/cirurgia , Avaliação de Resultados em Cuidados de Saúde , Participação Social , Extremidade Superior/cirurgia , Adolescente , Criança , Humanos
20.
Eur J Pediatr ; 178(12): 1883-1892, 2019 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-31620888

RESUMO

Although essential for providing optimal adolescent patient support, knowledge of the impact of Marfan syndrome in adolescence is limited. To explore adolescents' perceived impact of Marfan syndrome on (physical) functioning (activities, participation), disability (limitations, restrictions), contextual factors and support needs, we interviewed 19 adolescents with Marfan syndrome. Audio-recordings were transcribed, coded and analysed using thematic analysis. Identified themes were "difficulties in keeping up with peers" and "being and feeling different from peers". Furthermore, an adolescent Marfan syndrome-specific International Classification of Functioning, Disability and Health for Children and Youth (ICF-CY) model derived from the data describing the adolescent perceived impact of Marfan syndrome on functioning, disability and its contextual factors. Adolescents perceived problems in keeping up with peers in school, sports, leisure and friendships/relationships, and they could not meet work requirements. Moreover, participants perceived to differ from peers due to their appearance and disability. Contextual factors: coping with Marfan syndrome, self-esteem/image, knowledge about Marfan syndrome, support from family/friends/teachers, ability to express needs and peer-group acceptation acted individually as barrier or facilitator for identified themes.Conclusion: Adolescents with Marfan syndrome perceived limitations and restrictions in (physical) functioning. They perceived problems in keeping up with peers and perceived to differ from peers due to their appearance and disability. This warrants awareness and tailored physical, psychosocial, educational and environmental support programmes to improve (physical) functioning and empowerment of adolescents with Marfan syndrome.What is known:• Marfan syndrome is a hereditary connective tissue disorder.• Marfan syndrome affects multiple systems.What is new:• Adolescents with Marfan syndrome perceive (1) problems in keeping up with peers in school, sports, leisure, friendships/relationships and work (2) to differ from peers due to their appearance and disability.• An adolescent Marfan syndrome-specific International Classification of Functioning, Disability and Health for Children and Youth model derived from the data describing the adolescent perceived impact of Marfan syndrome on functioning, disability and contextual factors.


Assuntos
Atividades Cotidianas , Síndrome de Marfan/fisiopatologia , Síndrome de Marfan/psicologia , Qualidade de Vida , Adolescente , Avaliação da Deficiência , Feminino , Humanos , Masculino , Países Baixos , Grupo Associado , Autoimagem , Inquéritos e Questionários
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