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Handwriting Analysis in Children and Adolescents with Hemophilia: A Pilot Study.

ABSTRACT: BACKGROUND:Handwriting is a complex task that requires the integrity of different sensorimotor components to be performed successfully. Patients with hemophilia suffer from recurrent joint bleeds that may occur in the elbow, causing elbow dysfunction with handwriting performance impairment. In our study, we described instrumental dysgraphia that is related to functional disturbances. This pilot study aims to evaluate the handwriting performance in a group of patients with hemophilia. METHODS:The study was performed at the Angelo Bianchi Bonomi Hemophilia and Thrombosis Center in Milan. Boys with severe and moderate hemophilia A and B regularly followed-up at that Center, with age between 6-19 years, were eligible. Patients were invited to the Center for one multidisciplinary evaluation of the upper limbs that included: Clinical examination, surface electromyography, and handwriting assessment. RESULTS:All patients, but one, completed handwriting assessment. Overall, 14/19 (74%) had abnormal handwriting, which was overt instrumental dysgraphia in six (32%). There was no difference in Hemophilia Joint Health Score (HJHS) between dysgraphic and non-dysgraphic boys, while surface electromyography (sEMG) revealed a prevalence of flexor muscles of the upper limb in dysgraphic as compared with non-dysgraphic boys. CONCLUSIONS:The rather high prevalence of instrumental dysgraphia found in this pilot study deserves a further development of this preliminary experience by increasing the number of examined patients and comparing them with a control group, including quality of life and psychological assessment.

SUBMITTER: Pasta G

PROVIDER: S-EPMC7697872 | biostudies-literature | 2020 Nov

REPOSITORIES: biostudies-literature

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Handwriting Analysis in Children and Adolescents with Hemophilia: A Pilot Study.

Pasta Gianluigi G Mancuso Maria Elisa ME De Felice Filomena F Seuser Alexander A Annunziata Salvatore S Peyvandi Flora F Santagostino Elena E Mosconi Mario M Seuser Axel A

Journal of clinical medicine 20201114 11

<h4>Background</h4>Handwriting is a complex task that requires the integrity of different sensorimotor components to be performed successfully. Patients with hemophilia suffer from recurrent joint bleeds that may occur in the elbow, causing elbow dysfunction with handwriting performance impairment. In our study, we described instrumental dysgraphia that is related to functional disturbances. This pilot study aims to evaluate the handwriting performance in a group of patients with hemophilia.<h4> ...[more]

PMID: 33202574

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Project description:IntroductionEarly evaluation of writing readiness is essential to predict and prevent handwriting difficulties and its negative influences on school occupations. An occupation-based measurement for kindergarten children has been previously developed: Writing Readiness Inventory Tool In Context (WRITIC). In addition, to assess fine motor coordination two tests are frequently used in children with handwriting difficulties: the modified Timed Test of In-Hand Manipulation (Timed TIHM) and the Nine-Hole Peg Test (9-HPT). However, no Dutch reference data are available.AimTo provide reference data for (1) WRITIC, (2) Timed-TIHM and (3) 9-HPT for handwriting readiness assessment in kindergarten children.MethodsThree hundred and seventy-four children from Dutch kindergartens in the age of 5 to 6.5 years (5.6±0.4 years, 190 boys/184 girls) participated in the study. Children were recruited at Dutch kindergartens. Full classes of the last year were tested, children were excluded if there was a medical diagnosis such as a visual, auditory, motor or intellectual impairment that hinder handwriting performance. Descriptive statistics and percentiles scores were calculated. The score of the WRITIC (possible score 0-48 points) and the performance time on the Timed-TIHM and 9-HPT are classified as percentile scores lower than the 15th percentile to distinguish low performance from adequate performance. The percentile scores can be used to identify children that are possibly at risk developing handwriting difficulties in first grade.ResultsWRITIC scores ranged from 23 to 48 (41±4.4), Timed-TIHM ranged from 17.9 to 64.5 seconds (31.4± 7.4 seconds) and 9-HPT ranged from 18.2 to 48.3 seconds (28.4± 5.4). A WRITIC score between 0-36, a performance time of more than 39.6 seconds on the Timed-TIHM and more than 33.8 seconds on the 9-HPT were classified as low performance.ConclusionThe reference data of the WRITIC allow to assess which children are possibly at risk developing handwriting difficulties.

Project description:BACKGROUND:Future infertility is a significant concern for survivors of childhood and adolescent cancer. Children and adolescents may have the opportunity to undergo fertility preservation (FP) procedures (which preserve gonadal tissue or gametes for future use) prior to the cancer treatment. However, the decision is very complex, as it is often made by parents as proxy decision makers at the time of cancer diagnosis, and is time-sensitive (needing to occur before the cancer treatment begins). Furthermore, FP procedures in children and adolescents are experimental and cannot guarantee future fertility. An uninformed decision may result in future decision regret. OBJECTIVE:This study aimed to assess the acceptability, usability, and feasibility of a Web-based FP decision aid (DA) in parents of children and adolescents with cancer and clinicians. Fertility knowledge and decision regret were compared in families who reviewed the DA compared with those who did not. METHODS:The Web-based DA was developed according to the International Patient Decision Aid Standards. A cross-sectional study of parents of patients with cancer, who discussed fertility, and clinicians at a tertiary children's hospital was undertaken. The acceptability, usability, and feasibility of the DA were assessed using a pre-post survey design. Measures included the validated Decision Regret Scale, a purpose-designed fertility-related knowledge scale, questions regarding satisfaction with the DA, and open-ended responses for additional feedback. Furthermore, clinicians involved in FP were also invited to review the DA. RESULTS:We enrolled 34 parents and 11 clinicians in this study. Participants who reviewed the DA (15 parents and 11 clinicians) expressed satisfaction with its content and functionality. Parents reported an improved understanding of cancer treatments, infertility, and FP procedures and did not report greater decision regret after DA review. Most parents (13/15, 86%) would recommend the DA to other parents. All clinicians had a consensus that this was a valid and relevant information source for all involved in fertility care. CONCLUSIONS:It is an international standard of care to discuss the impact of cancer treatment on fertility before cancer treatment. This is the first fertility DA for parents of children and adolescents with cancer and is found to be relevant and acceptable by parents and clinicians. This DA has the potential to help support parents to make informed fertility-related decisions for their children and adolescents. However, future research is needed to assess the impact of the DA on prospective decision making.

Project description:BackgroundTo assess the feasibility, acceptability, safety, and short-term benefits of a tailored cardiac rehabilitation program for children and adolescents with long QT syndrome (LQTS).MethodsEight participants, aged between 6 and 18, with a positive LQTS genotype and impaired cardiorespiratory fitness, were enrolled in a 12-week centre-based cardiac rehabilitation program. The program included supervised exercise training group sessions (aerobic, resistance, and outdoor activities) and patient education workshops. Feasibility, acceptability, and safety of the program were prospectively monitored. Feedback from the parents, children, and professionals involved was collected from qualitative interviews. Short-term effects on cardiorespiratory fitness, muscle fitness, physical activity, and health-related quality of life (HRQoL) were measured between baseline and the end of the program.ResultsRetention (88% with one participant dropping out) and adherence (79%) rates were good, and no cardiac events occurred during the 12-week intervention period. Participants, parents, and healthcare professionals expressed a high level of satisfaction with the program. A significant increase between the beginning and the end of the program was observed for ventilatory anaerobic threshold (21.7±5.2 vs. 28.7±5.1 mL/kg/min, P=0.01, effect size=0.89), grip strength, (18±5.3 Kg vs. 20±4.7 Kg, P=0.02, effect size=0.90), lower limb explosive strength (142±36.5 cm vs. 148±24 cm, P=0.02, effect size=0.90), and parent-reported physical health dimension of HRQoL (65.6±9.75 vs. 84.4±20.35, P=0.03, effect size=0.87).ConclusionsA 12-week tailored centre-based cardiac rehabilitation program was feasible, acceptable, and safe for children with LQTS. Cardiac rehabilitation for children with LQTS presents a new approach aligned with secondary prevention in youth with cardiac diseases.Trial registrationThe trial was registered at Clinicaltrials.gov (NCT05964322, registration date: 27/07/2023).

Project description:BackgroundFirst graders born prematurely perform poorly on handwriting speed and legibility. However, whether there are specific legibility factors in which preterm children demonstrate difficulty remains unknown. In addition, handwriting performance beyond the first grade and the influence of sex on handwriting performance in preterm children are still unclear. We aimed to investigate the influence of prematurity and sex on multiple dimensions of handwriting in grade two and to identify the contributors to performance.MethodsSixty-three preterm (34 boys and 29 girls) and 67 full-term (27 boys and 40 girls) peers in grade two were included. Class teachers were asked to complete the Chinese Handwriting Evaluation Form. A subgroup of 39 preterm children received assessments on intelligence, visual perception, tactile and kinesthetic sensation, and fine motor skills. Their inattention behavior was rated using a maternal self-report with a behavioral scale.ResultsBoys born prematurely exhibited poorer performance in the speed dimension than full-term boys (p = 0.008), whereas there was comparable performance in the two groups of girls (p = 0.221). In the dimensions related to legibility, preterm boys (32.4%) had a higher percentage of children with difficulty in the construction dimension than the other groups (preterm girls: 6.9%, full-term boys: 7.4%, full-term girls: 5.0%). However, no group difference was found in the dimensions of accuracy and directionality. Of the sensory-perceptual-motor factors, attention was the most significant predictor of accuracy in performance (p = 0.046) and speed dimensions (p = 0.001) in preterm children.ConclusionsBoys appear to be vulnerable to the adverse impacts of preterm birth in terms of performance in the dimensions of speed and construction in grade two. Based on the significant contribution of attention to handwriting performance in preterm children, assessment and intervention in the area of attention is strongly suggested for preterm children with handwriting problems.

Dataset Information

Handwriting Analysis in Children and Adolescents with Hemophilia: A Pilot Study.

Publications

Handwriting Analysis in Children and Adolescents with Hemophilia: A Pilot Study.

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