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Article ; Online: Critically appraised paper: A bespoke multidisciplinary intervention is no more effective than standard management for children with symptomatic joint hypermobility for improving pain [commentary].

Pacey, Verity

2020 Volume 66, Issue 2, Page(s) 129

MeSH term(s)	Child ; Humans ; Joint Instability ; Pain
Language	English
Publishing date	2020-04-11
Publishing country	Netherlands
Document type	Journal Article ; Comment
ZDB-ID	2543915-7
ISSN	1836-9561 ; 1836-9553 ; 0004-9514
ISSN (online)	1836-9561
ISSN	1836-9553 ; 0004-9514
DOI	10.1016/j.jphys.2020.02.001
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Article ; Online: The Targeted Motor Control Screening Tool is Valid for Four-Year-Old Children.

Brown, Laura / Bacon, Amanda / Pacey, Verity / Ilhan, Emre

Physical therapy

2024

Abstract: Objective: The objective was to determine the validity of the Targeted Motor Control (TMC) screening tool with the Neurosensory Motor Developmental Assessment (NSMDA) in 4-year-old children.: Methods: In this single cohort observational study, ... ...

Abstract	Objective: The objective was to determine the validity of the Targeted Motor Control (TMC) screening tool with the Neurosensory Motor Developmental Assessment (NSMDA) in 4-year-old children. Methods: In this single cohort observational study, children (3 years 9 months to 4 years 5 months) completed the TMC and the NSMDA in a randomized order 5 to 14 days apart. Results: Seventy-six children (mean age = 4 years 2 months; SD = 2.5 months; n = 35 male) completed both assessments. Forty-two children performed within the normal range on the NSMDA. There were significant and positive moderate correlations between item totals overall and for each area on the NSMDA and the TMC (r = 0.40 to 0.61) and between the NSMDA functional grade for each area and the corresponding TMC areas (r = 0.47 to 0.67). However, the correlation between the NSMDA sensorimotor functional grade and the TMC sensory score was significant but low and positive (r = 0.35). The optimal cut-off score for detecting children at risk of atypical development on the TMC was a score of <9 (n = 42) (sensitivity = 82.4%; specificity = 66.7%) with a positive likelihood ratio of 2.47 (95% CI = 1.57 to 3.89) and a negative likelihood ratio of 0.26 (95% CI = 0.12 to 0.56). Conclusions: The TMC is a valid screening tool to identify 4-year-old children at risk of motor delay. Impact: Early identification of developmental concerns using a validated screening tool is recommended. The TMC is a valid performance-based screening tool that can be used to identify children at risk of atypical motor development who would benefit from further developmental assessment so that, if indicated, timely intervention can be implemented.
Language	English
Publishing date	2024-05-14
Publishing country	United States
Document type	Journal Article
ZDB-ID	415886-6
ISSN	1538-6724 ; 0031-9023
ISSN (online)	1538-6724
ISSN	0031-9023
DOI	10.1093/ptj/pzae071
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Article ; Online: The use and effectiveness of exercise for managing postural orthostatic tachycardia syndrome in young adults with joint hypermobility and related conditions: A scoping review.

Peebles, Karen C / Jacobs, Charl / Makaroff, Logan / Pacey, Verity

Autonomic neuroscience : basic & clinical

2024 Volume 252, Page(s) 103156

Abstract: Purpose: Postural Orthostatic Tachycardia Syndrome (POTS) is a form of dysautonomia. It may occur in isolation, but frequently co-exists in individuals with hypermobile variants of Ehlers-Danlos Syndrome (EDS) and related conditions (chronic fatigue ... ...

Abstract	Purpose: Postural Orthostatic Tachycardia Syndrome (POTS) is a form of dysautonomia. It may occur in isolation, but frequently co-exists in individuals with hypermobile variants of Ehlers-Danlos Syndrome (EDS) and related conditions (chronic fatigue syndrome [CFS] and fibromyalgia). Exercise is recommended for non-pharmacological POTS management but needs to be individualised. This scoping review explores the current literature on use and effectiveness of exercise-based management for POTS, with specific focus on individuals with joint hypermobility and related conditions who experience hypermobility, and/or pain, and/or fatigue. Methods: A systematic search, to January 2023, of Medline, EMBASE, AMED, CINAHL and the Cochrane library was conducted. Studies that reported on adolescents and adults who had been diagnosed with POTS using standard criteria and underwent an exercise-based training intervention were included. Results: Following full-text screening, 10 articles were identified (2 randomised control trials, 4 comparative studies and 4 case reports). One comparative study reported a small subset of participants with EDS and one case report included an individual diagnosed with CFS; the remainder investigated a wider POTS population. Overall, 3 months of endurance followed by resistance exercise, graduating from the horizontal-to-upright position reduced POTS symptoms and improved quality-of-life. Conclusion: The findings highlight a paucity of higher-level studies documenting exercise for POTS management in people with joint hypermobility and related conditions. Results from the wider POTS population demonstrate exercise is safe and effective. Large, well-designed clinical studies exploring exercise for POTS management adapting to meet the complex musculoskeletal and non-musculoskeletal features of symptomatic joint hypermobility are needed.
MeSH term(s)	Adolescent ; Humans ; Young Adult ; Postural Orthostatic Tachycardia Syndrome/therapy ; Fatigue Syndrome, Chronic ; Joint Instability/therapy ; Ehlers-Danlos Syndrome/complications ; Ehlers-Danlos Syndrome/therapy ; Exercise
Language	English
Publishing date	2024-02-13
Publishing country	Netherlands
Document type	Case Reports ; Journal Article ; Review
ZDB-ID	2020105-9
ISSN	1872-7484 ; 1566-0702
ISSN (online)	1872-7484
ISSN	1566-0702
DOI	10.1016/j.autneu.2024.103156
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Article: How Schroth Therapists Vary the Implementation of Schroth Worldwide for Adolescents with Idiopathic Scoliosis: A Mixed Methods Study.

Marchese, Rosemary / Ilhan, Emre / Pacey, Verity

Journal of clinical medicine

2023 Volume 12, Issue 18

Abstract: 1) Background: Schroth is a type of physiotherapeutic scoliosis specific exercise (PSSE) prescribed to adolescents with idiopathic scoliosis (AIS). Studies have investigated the effectiveness of Schroth but are yet to elucidate how Schroth is applied ... ...

Abstract	(1) Background: Schroth is a type of physiotherapeutic scoliosis specific exercise (PSSE) prescribed to adolescents with idiopathic scoliosis (AIS). Studies have investigated the effectiveness of Schroth but are yet to elucidate how Schroth is applied clinically and the factors that influence their prescription. (2) Methods: A mixed methods design was used comprising an anonymous survey and semi-structured interviews of Schroth therapists who treated AIS and who were publicly listed on the Barcelona Scoliosis Physical Therapy School or the International Schroth 3-dimensional Scoliosis Therapy School websites. The survey included 64 questions covering demographics, session and treatment characteristics, and whether therapists included other treatment modalities in their clinical practice. A convenience sample of survey participants were invited to participate in a semi-structured interview to further explore the factors that influenced their prescription of Schroth for AIS. Results from the survey were analyzed descriptively (n, %), whereas inductive thematic analysis was used for the interviews. (3) Results: of the 173 survey respondents (18% response rate), most were from Europe and North America (64.0%), female (78.6%), physiotherapists (96.0%), and worked in private settings (72.3%). Fifty-two per cent of participants used other types of PSSE as an adjunct to Schroth, the Scientific Exercise Approach to Scoliosis (SEAS) being the most frequently used (37.9%). Non-PSSE methods were used 'at some point' as an adjunct by 98.8% of participants, including massage and other soft tissue techniques (80.9%), Pilates (46.6%), and Yoga (31.5%). The Schroth techniques used by all survey respondents included breathing and pelvic corrections. Seven participants were interviewed, but data saturation was achieved after only four interviews. Thematic analysis revealed four, inter-related broad themes describing the factors that influenced Schroth prescription for AIS: (1) the adolescent as a whole, including physical, emotional and mental characteristics, and patient goals, (2) family, including parent relationship with the adolescent and the motivation of parents in regard to Schroth, (3) the systems within which the treatment was being offered, such as vicinity to the clinic and the presence of financial insurance support, and (4) therapist characteristics, such as their training and experience. (4) Conclusions: Schroth therapists worldwide use a variety of adjunctive methods to treat AIS. Therapists prescribing Schroth exercises to AIS consider the complex interplay of intra-, inter- and extra-personal factors in clinical practice. These considerations move beyond the three components of evidence-based practice of research, patient preferences, and clinical expertise, towards a systems-based reflection on exercise prescription.
Language	English
Publishing date	2023-09-19
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2662592-1
ISSN	2077-0383
ISSN	2077-0383
DOI	10.3390/jcm12186063
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Article ; Online: The Impact of Podiatric Intervention on the Quality of Life and Pain in Children and Adolescents with Hypermobility.

Maarj, Muhammad / Pacey, Verity / Tofts, Louise / Clapham, Matthew / Coda, Andrea

International journal of environmental research and public health

2023 Volume 20, Issue 17

Abstract: The purpose of this study was to evaluate the effect of custom-made orthotics on pain, health-related quality of life (HRQoL), function and fatigue in children and adolescents with generalised joint hypermobility (GJH) and lower limb pain. Fifty-three ... ...

Abstract	The purpose of this study was to evaluate the effect of custom-made orthotics on pain, health-related quality of life (HRQoL), function and fatigue in children and adolescents with generalised joint hypermobility (GJH) and lower limb pain. Fifty-three children aged 5-18 years were fitted with custom-made polypropylene orthotics. Visual analogue scale (VAS) assessed lower limb pain severity, Paediatric Quality of Life Inventory assessed HRQoL and fatigue and six-minute walk test (6 MWT) measured functional endurance at baseline, at 1 month and 3 months post-intervention. A mixed model including a random intercept for participant and a fixed effect for time was used to assess differences in outcomes over time. Fifty-two children completed the study (mean age 10.6-years). Children reported significantly reduced pain (mean VAS reduction -27/100, 95%CI: -33, -21), improved HRQoL (mean total improvement 11/100, 95%CI: 7, -15), functional capacity (mean 6MWT improvement 27 m, 95%CI: 18, -36) and fatigue (mean total improvement 13/100, 95%CI: 9, -17) after 1 month of wearing the custom-made orthotics. From 1 month to 3 months there was further statistically but not clinically significant reduction in pain while benefit on other outcomes was maintained. In this study, children with GJH reported reduced lower limb pain, improved HRQoL, functional endurance and fatigue after a month post-fitting of custom-made orthotics which was maintained over a 3 month period. Orthotics were well-tolerated with no serious adverse events reported.
MeSH term(s)	Humans ; Adolescent ; Child ; Quality of Life ; Fatigue ; Joint Instability ; Medicine ; Pain
Language	English
Publishing date	2023-08-22
Publishing country	Switzerland
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	2175195-X
ISSN	1660-4601 ; 1661-7827
ISSN (online)	1660-4601
ISSN	1661-7827
DOI	10.3390/ijerph20176623
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Article ; Online: Subgroups of Idiopathic Clubfoot Can Predict Short-term Outcomes.

Quinlan, Rachal / Pacey, Verity / Ilhan, Emre / Gibbons, Paul / Gray, Kelly

Journal of pediatric orthopedics

2023 Volume 43, Issue 5, Page(s) 326–331

Abstract: Background: The Pirani scale is used for the assessment of Ponseti-managed clubfoot. Predicting outcomes using the total Pirani scale score has varied results, however, the prognostic value of midfoot and hindfoot components remains unknown. The purpose ...

Abstract	Background: The Pirani scale is used for the assessment of Ponseti-managed clubfoot. Predicting outcomes using the total Pirani scale score has varied results, however, the prognostic value of midfoot and hindfoot components remains unknown. The purpose was to (1) determine the existence of subgroups of Ponseti-managed idiopathic clubfoot based on the trajectory of change in midfoot and hindfoot Pirani scale scores, (2) identify time points, at which subgroups can be distinguished, and (3) determine whether subgroups are associated with the number of casts required for correction and need for Achilles tenotomy. Methods: Medical records of 226 children with 335 idiopathic clubfeet, over a 12-year period, were reviewed. Group-based trajectory modeling of the Pirani scale midfoot score and hindfoot score identified subgroups of clubfoot that followed statistically distinct patterns of change during initial Ponseti management. Generalized estimating equations determined the time point, at which subgroups could be distinguished. Comparisons between groups were determined using the Kruskal-Wallis test for the number of casts required for correction and binary logistic regression analysis for the need for tenotomy. Results: Four subgroups were identified based on the rate of midfoot-hindfoot change: (1) fast-steady (61%), (2) steady-steady (19%), (3) fast-nil (7%), and (4) steady-nil (14%). The fast-steady subgroup can be distinguished at the removal of the second cast and all other subgroups can be distinguished at the removal of the fourth cast [ H (3) = 228.76, P < 0.001]. There was a significant statistical, not clinical, difference in the total number of casts required for correction across the 4 subgroups [median number of casts 5 to 6 in all groups, H (3) = 43.82, P < 0.001]. Need for tenotomy was significantly less in the fast-steady (51%) subgroup compared with the steady-steady (80%) subgroup [ H (1) = 16.23, P < 0.001]; tenotomy rates did not differ between the fast-nil (91%) and steady-nil (100%) subgroups [ H (1) = 4.13, P = 0.04]. Conclusions: Four distinct subgroups of idiopathic clubfoot were identified. Tenotomy rate differs between the subgroups highlighting the clinical benefit of subgrouping to predict outcomes in Ponseti-managed idiopathic clubfoot. Level of evidence: Level II, prognostic.
MeSH term(s)	Child ; Humans ; Infant ; Clubfoot/diagnosis ; Clubfoot/surgery ; Treatment Outcome ; Casts, Surgical ; Foot ; Tenotomy/methods
Language	English
Publishing date	2023-03-13
Publishing country	United States
Document type	Journal Article
ZDB-ID	604642-3
ISSN	1539-2570 ; 0271-6798
ISSN (online)	1539-2570
ISSN	0271-6798
DOI	10.1097/BPO.0000000000002382
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Article ; Online: Cutaneous functional units prediction in outcomes of early and intensive splinting following palmar burn injury in young children: a prospective study.

Thomas, Rhianydd / Wicks, Stephanie / Dale, Marita / Toose, Claire / Pacey, Verity

Burns : journal of the International Society for Burn Injuries

2023 Volume 50, Issue 3, Page(s) 717–729

Abstract: Background: Palmar burn injuries are common in young children and can result in contracture.: Methods: A prospective longitudinal study describes outcomes of palm and digit extension splint use following burn in 75 children (83 hands) aged < 5 years ... ...

Abstract	Background: Palmar burn injuries are common in young children and can result in contracture. Methods: A prospective longitudinal study describes outcomes of palm and digit extension splint use following burn in 75 children (83 hands) aged < 5 years and determines whether specific cutaneous functional units (CFUs) are associated with early signs of contracture (ESC). Outcomes were assessed up to 9-18 months following burn. Routine clinical data was collected at therapy reviews. Results: Children were splinted > 12 h/day for a mean of 158 days following burn. The mean time to splint cessation was 264 days following burn. Fourteen hands developed ESC (17%): 12 hands had full ROM restored following conservative management, 2 hands (3%) progressed to contracture. Hands that developed ESC had greater healing time (p = 0.002), greater number of CFUs affected (p < 0.001), and greater number of immediate first webspace and extended first webspace CFUs affected (p = 0.002, p < 0.001 respectively). ESC risk increases for each day to heal (odds ratio [OR] 1.1, 95% CI 1.0-1.2) and each CFU in extended first webspace (OR 2.8, 95% CI 1.5-5.0). Conclusion: Early and intensive splinting following palmar burn results in excellent ROM. Burns involving more CFUs or the first webspace are associated with ESC.
MeSH term(s)	Child ; Humans ; Child, Preschool ; Prospective Studies ; Burns/complications ; Burns/therapy ; Burns/diagnosis ; Longitudinal Studies ; Skin ; Contracture/etiology ; Contracture/therapy
Language	English
Publishing date	2023-12-27
Publishing country	Netherlands
Document type	Journal Article
ZDB-ID	197308-3
ISSN	1879-1409 ; 0305-4179
ISSN (online)	1879-1409
ISSN	0305-4179
DOI	10.1016/j.burns.2023.12.009
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Article ; Online: Medical complications in children with achondroplasia.

Armstrong, Jennifer A / Pacey, Verity / Tofts, Louise J

Developmental medicine and child neurology

2022 Volume 64, Issue 8, Page(s) 989–997

Abstract: Aim: To determine the rates of medical investigations, complications, interventions, and outcomes in children with achondroplasia.: Method: Children and adolescents with achondroplasia born between 2000 and 2019, aged between 0 and 18 years of age, ... ...

Abstract	Aim: To determine the rates of medical investigations, complications, interventions, and outcomes in children with achondroplasia. Method: Children and adolescents with achondroplasia born between 2000 and 2019, aged between 0 and 18 years of age, and seen at The Children's Hospital at Westmead skeletal dysplasia clinic were included. Data were collected retrospectively from clinical records. Standard descriptive statistics were used for analysis. Results: The study included 108 participants, 58 males and 50 females. Ninety-nine participants (91.7%) entered the study at birth. The other nine (8.3%) participants entered the study after birth (mean age = 2 years 4 months, SD = 1 year 8 months). The median age of exit from the study was 8 years 8 months (IQR = 8 years 9 months) with a median follow-up of 8 years 8 months (IQR = 8 years 9 months). Fifty-two (48%) participants presented with craniocervical stenosis, 15 (13.9%) with hydrocephalus, 66 (61.1%) with hearing impairment, 44 (40.7%) with sleep-disordered breathing, 46 (42.6%) with lower-limb malalignment, 24 (22.2%) with thoracolumbar kyphosis, 10 (9.3%) with symptomatic spinal stenosis, 12 (11.1%) with obesity, and 16 (14.8%) who had at least one admission for respiratory illness. Two children died during the study period. Interpretation: We report contemporary rates of medical complications in an Australian population of children with achondroplasia. Recommendations for surveillance in clinical practice are discussed. This information will help guide clinicians with their expectant management of achondroplasia and provide prognostic information to the families of children with achondroplasia.
MeSH term(s)	Achondroplasia/complications ; Achondroplasia/epidemiology ; Adolescent ; Australia ; Child ; Child, Preschool ; Female ; Humans ; Infant ; Infant, Newborn ; Kyphosis/complications ; Male ; Retrospective Studies ; Spinal Stenosis
Language	English
Publishing date	2022-03-02
Publishing country	England
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	80369-8
ISSN	1469-8749 ; 0012-1622
ISSN (online)	1469-8749
ISSN	0012-1622
DOI	10.1111/dmcn.15194
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Article ; Online: Hypermobility syndromes in children and adolescents: Assessment, diagnosis and multidisciplinary management.

Nicholson, Leslie Lorenda / Chan, Cliffton / Tofts, Louise / Pacey, Verity

Australian journal of general practice

2022 Volume 51, Issue 6, Page(s) 409–414

Abstract: Background: Managing children and adolescents with syndromic hypermobility and their parents is challenging in the context of a standard consultation. The multi-organ involvement of the syndromes, the evolving classification criteria and their highly ... ...

Abstract	Background: Managing children and adolescents with syndromic hypermobility and their parents is challenging in the context of a standard consultation. The multi-organ involvement of the syndromes, the evolving classification criteria and their highly heterogeneous presentation complicate both diagnosis and management. Objective: The purpose of this article is to provide an overview of the diagnosis, management and support of children with hypermobility syndromes, including the role of the multidisciplinary team and common associated features. Discussion: Optimising the quality of life of people with syndromic hypermobility begins with the early identification of their signs and symptoms. Diagnosis, ideally by adolescence, provides affected children and their parents with validation and directs their focus to management aimed at mitigating the socioeconomic, educational and health-related quality of life impact of these conditions.
MeSH term(s)	Adolescent ; Child ; Humans ; Joint Instability/complications ; Joint Instability/diagnosis ; Joint Instability/therapy ; Quality of Life ; Syndrome
Language	English
Publishing date	2022-05-30
Publishing country	Australia
Document type	Journal Article
ZDB-ID	2924889-9
ISSN	2208-7958 ; 2208-794X
ISSN (online)	2208-7958
ISSN	2208-794X
DOI	10.31128/AJGP-03-21-5870
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Article ; Online: Impaired muscle parameters in adults with mild to severe types of osteogenesis imperfecta: a cross-sectional study.

Coussens, Marie / Lapauw, Bruno / De Wandele, Inge / Malfait, Fransiska / Pocovi, Natasha C / Pacey, Verity / Calders, Patrick

Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research

2024 Volume 39, Issue 3, Page(s) 260–270

Abstract: Impaired muscle parameters may further compromise the already compromised skeleton in individuals with OI. This cross-sectional study aimed to compare muscle function and body composition in adults with various OI types and healthy controls. Sixty-eight ... ...

Abstract	Impaired muscle parameters may further compromise the already compromised skeleton in individuals with OI. This cross-sectional study aimed to compare muscle function and body composition in adults with various OI types and healthy controls. Sixty-eight adults with OI (mean age 42.2 yr; 27 men) and 68 healthy age- and sex-matched controls were recruited. Maximal isometric muscle force was assessed by handheld dynamometry (hand grip, hip flexors, shoulder abductors, and ankle dorsiflexors), muscle endurance by posture maintenance tests (shoulder abduction, hip flexion, and wall sit), and functional lower limb strength by 30-s chair rise test. In a sub cohort, dynamic muscle function (peak power and force) was assessed by a ground reaction force plate, and lean and fat mass, muscle and fat cross-sectional area (CSA), and muscle density by dual-energy X-ray absorptiometry and peripheral quantitative computed tomography. Multiple linear regression models were fitted with group (OI type I, III, IV/V, or controls), country, sex, and age in the fixed effects part. Overall, adults with various types of OI had lower isometric, endurance, and functional muscle strength (mean difference [MD] = OI type I: 19-43%, OI type IV/V: 25-68%, OI type III: 20-72%) compared to controls. Furthermore, adults with OI type I had lower dynamic muscle function (peak force [MD = 25-29%] and power [MD = 18-60%]), lean mass (MD = 10-17%), muscle CSA (MD = 9-21%), and muscle density (MD = 2-3%) but higher adiposity indices (MD = 24-42%) compared to controls. Functional lower limb strength and maximal muscle force were significantly different between OI types, whereas muscle endurance was not. To conclude, adults with OI present with markedly impaired muscle function which may partially be explained by their altered body composition. Our findings emphasize the need for proper assessment of various muscle parameters and (research into) appropriate and safe muscle strengthening approaches in this population.
MeSH term(s)	Male ; Adult ; Humans ; Osteogenesis Imperfecta ; Cross-Sectional Studies ; Hand Strength ; Absorptiometry, Photon/methods ; Muscle, Skeletal
Language	English
Publishing date	2024-03-18
Publishing country	England
Document type	Journal Article
ZDB-ID	632783-7
ISSN	1523-4681 ; 0884-0431
ISSN (online)	1523-4681
ISSN	0884-0431
DOI	10.1093/jbmr/zjae003
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