Browsing by Author "Lindenschot, M."

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Perceive, Recall, Plan and Perform (PRPP)-Assessment Based on Parent-Provided Videos of Children with Mitochondrial Disorder: Action Design Research on Implementation Challenges.
Lindenschot, M. ; Groot, I.J.M. de ; -van der Sanden, M.W.G. Nijhuis ; Steultjens, E.M.J. ; Graff, M.J.L. ; Koene, S.
2023, Article / Letter to editor (Physical and Occupational Therapy in Pediatrics, vol. 43, iss. 1, (2023), pp. 74-92)AIMS: Performing the Perceive, Recall, Plan and Perform (PRPP)-Assessment, using video material of everyday life, seems sensible to lower the patient burden, enhance ecological validity, and provide care at a distance. However, receipt of adequate video material is not self-evident and assessing videos can be challenging. Therefore, this study aims to optimize the process of gaining video material and to optimize the PRPP-Assessment based on parent-provided videos. METHODS: An action design research method was used, focusing on implementation of the PRPP-Assessment based on parent-provided videos within the care of children with a mitochondrial disorder or similar symptoms. RESULTS: Five cycles were conducted. To receive input, the cycles used videos of nine children performing activities, written feedback, and semi-structured interviews and focus groups comprising parents (nn=n13), a teacher (nn=n1), occupational therapists (nn=n16), and other professionals (nn=n2) . This led to successful implementation of the PRPP-Assessment. General lessons were learned on (1) instructing parents; (2) handling video material; (3) PRPP-Assessment based on parent-provided videos; and (4) PRPP-Assessment of children (with limited functional abilities). CONCLUSIONS: Lessons learned should be implemented in practice and are incorporated into a manual to guide the implementation of video-based observations with PRPP-Assessment in practice.
Evidence for effect of l-serine, a novel therapy for GRIN2B-related neurodevelopmental disorder.
Hollander, B. den ; Veenvliet, A.R.J. ; Lindenschot, M. ; Essen, P. van ; Peters, G. ; Santos-Gómez, A. ; Olivella, M. ; Altafaj, X. ; Brands, M.M. ; Jacobs, B.A.W. ; Karnebeek, C.D. van et al. sine al.
2023, Article / Letter to editor (Molecular Genetics and Metabolism, vol. 138, iss. 3, (2023), pp. 107523)RATIONALE: To date, causal therapy is potentially available for GRIN2B-related neurodevelopmental disorder (NDD) due to loss-of-function (LoF) variants in GRIN2B, resulting in dysfunction of the GluN2B subunit-containing N-methyl-d-aspartate receptor (NMDAR). Recently, in vitro experiments showed that high doses of NMDAR co-agonist d-serine has the potential to boost the activity in GluN2B LoF variant-containing NMDARs. Initial reports of GRIN2B-NDD patients LoF variants, treated with l-serine using different regimens, showed varying effects on motor and cognitive performance, communication, behavior and EEG. Here, this novel treatment using a standardized protocol with an innovative developmental outcome measure is explored further in an open-label observational GRIN2B-NDD study. METHODS: Initially, in vitro studies were conducted in order to functionally stratify two de novo GRIN2B variants present in two female patients (18 months and 4 years old). Functional studies showed that both variants are LoF, and thus the patients were treated experimentally according to an approved protocol with oral l-serine (500 mg/kg/day in 4 doses) for a period of 12 months. Both patients showed a heterogeneous clinical phenotype, however overlapping symptoms were present: intellectual developmental disability (IDD), behavioral abnormalities and hypotonia. Outcome measures included laboratory tests, quality of life, sleep, irritability, stool, and performance skills, measured by, among others, the Perceive-Recall-Plan-Perform System of Task Analysis (PRPP-Assessment). RESULTS: Both patients tolerated l-serine without adverse effects. In one patient, improvement in psychomotor development and cognitive functioning was observed after 12 months (PRPP mastery score 10% at baseline, 78% at twelve months). In the most severe clinically affected patient no significant objective improvement in validated outcomes was observed. Caregivers of both patients reported subjective increase of alertness and improved communication skills. CONCLUSION: Our observational study confirms that l-serine supplementation is safe in patients with GRIN2B-NDD associated with LoF variants, and may accelerate psychomotor development and ameliorate cognitive performance in some but not all patients. The PRPP-Assessment, a promising instrument to evaluate everyday activities and enhance personalized and value-based care, was not performed in the severely affected patient, meaning that possible positive results may have been missed. To generate stronger evidence for effect of l-serine in GRIN2B-NDD, we will perform placebo-controlled n-of-1 trials.
The reliability and validity of the perceive, recall, plan and perform assessment in children with a mitochondrial disorder.
Lindenschot, M. ; Koene, S. ; Nott, M.T. ; -van der Sanden, M.W.G. Nijhuis ; Groot, I.J.M. de ; Steultjens, E.M.J. ; Graff, M.J.L.
2022, Article / Letter to editor (Disability and Rehabilitation, (2022), pp. 1-14)PURPOSE: To investigate the reliability and validity of the Perceive, Recall, Plan and Perform System of Task Analysis (PRPP-Assessment) by parent-provided videos of children with mitochondrial disorders. METHODS: Videos of 13 children performing 3-7 activities each were the subject of study, resulting in 65 activities. These were scored using the PRPP-Assessment by 11 raters. Internal consistency was calculated with Cronbach's alpha. Intra-rater reliability was evaluated by Bland-Altman Plots on 44 PRPP-Assessment scores. Inter-rater reliability was calculated with ICC(Agreement) on 128 PRPP-Assessment scores. Construct validity was assessed by comparing the PRPP-Assessment scores to the Canadian Occupational Performance Measure using Cohen's Kappa. PRPP-Assessments scores were evaluated with a multi-faceted Rasch Analysis. RESULTS: Internal consistency was high (0.92). Intra-rater reliability was sufficient to be good (92-96% within the 95%-Limits of the Agreement). The ICC(Agreement) for stage 1 Mastery Score showed acceptable inter-rater reliability (0.646). Stage 2 of the PRPP-Assessment showed low ICCs due to a lack of variability within the sample. Four out of six hypotheses on validity were accepted. Rasch's analysis demonstrated sound goodness-of-fit, and supported the validity of the PRPP-Assessment. CONCLUSION: The PRPP-Assessment by parent-provided videos in this heterogenic group showed sufficient to good psychometric properties. In practice, careful task selection and formulating criterion is recommended.Implications for RehabilitationPRPP-Assessment by parent-provided videos is reliable and valid in the complex, heterogenous group of children with mitochondrial disordersThe PRPP-Assessment is suitable for children with mitochondrial disorders as it showed to contain familiar, functional and meaningful tasks and activities that fit with their level of functioningProfessionals should be aware that parents might not be used to the criterium-based frame of reference of the PRPP-AssessmentWhen applying the PRPP-Assessment, it is recommended to be careful in task selection and formulating the criterion.
Tailored interviewing to uncover the perspectives ofchildren with multiple disabilities on daily activities: Aqualitative analyses of interview methods and interviewerskills
Steultjens, E.M.J. ; Lindenschot, M. ; Diepeveen, S.J.H. ; Zajec, J. ; Groot, I. de ; Nijhuis-van der Sanden, M.W.G. ; Koene, S. ; Graff, Maud
2022, Article / Letter to editor (Australian Occupational Therapy Journal, (2022), pp. 1-15)
Hoe ga je in gesprek met kinderen over de betekenis van hun handelen?
Schot, M. ; Lindenschot, M. ; Velde, A.
2020, Article / Letter to editor (Ergotherapie Magazine, vol. 5, (2020), pp. 40-44)
Everyday Activities for Children with Mitochondrial Disorder: A Retrospective Chart Review.
Lindenschot, M. ; Groot, I. de ; Koene, S. ; Satink, T. ; Steultjens, E.M.J. ; Nijhuis-van der Sanden, M.W.G.
2018, Article / Letter to editor (Occupational Therapy International, (2018))
The perspectives and values of children with a mitochondrial disorder with regard to everyday activities.
Lindenschot, M. ; Steultjens, E.M.J. ; Zajec, J. ; Nijhuis-van der Sanden, M.W.G. ; Koene, S. ; Groot, I. de
2018, Article / Letter to editor (Journal of Developmental and Physical Disabilities, (2018), pp. 1-26)
Hoofdstuk 28: Ergotherapie
Lindenschot, M. ; Rijnbeek, D. ; Satink, T. ; Nijhuis-van der Sanden, M.W.G.
2017, Part of book or chapter of book (Spaans, J; van der Horst, H.; Visser, S. (ed.), Handboek behandeling van somatisch onvoldoende verklaarde lichamelijke klachten)
Uitkomsten van de ‘Cognitive Orientation to daily Occupational Performance’ (CO-OP) voor kinderen met DCD: Multipele Casestudie – Kwalitatief deel
Lindenschot, M. ; Kottorp, A. ; Steultjens, E.M.J.
2013, Article / Letter to editor (Wetenschappelijk Tijdschrift voor Ergotherapie, vol. 6, iss. 2, (2013), pp. 26-36)
Uitkomsten van de ‘Cognitive Orientation to daily Occupational Performance’ (CO-OP) voor kinderen met DCD : Multipele Casestudie - Kwantitatief deel
Lindenschot, M. ; Kottorp, A. ; Steultjens, E.M.J.
2012, Article / Letter to editor (Wetenschappelijk Tijdschrift voor Ergotherapie, vol. 5, iss. 4, (2012), pp. 24-38)Volgens internationale schattingen is de prevalentie van de diagnose Developmental Coordination Disorder (DCD) minstens 6% voor kinderen in de leeftijd van 5 tot 11 jaar (1). De American Psychiatric Association (APA) heeft DCD gedefinieerd als “The essential feature of DCD is a marked impairment in the development of motor coordination (…) that significantly interferes with academic achievement or activities of daily living” (1). Vrij vertaald naar het Nederlands betekent dit: ‘een aantoonbare verstoring in de ontwikkeling van de motorische coördinatie (…) dat significant interfereert met de schoolse prestaties of met algemene dagelijkse activiteiten’. Vanwege het effect van DCD op het dagelijks leven van kinderen en hun familie (2) is het noodzakelijk dat we interventies toepassen met als doel het dagelijks leven te verbeteren (3).

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Browsing by Author "Lindenschot, M."

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