Sex-, age-, and height-specific reference curves for the 6-min walk test in healthy children and adolescents

Saraff, Vrinda; Schneider, Johanna; Colleselli, Valeria; Ruepp, Monika; Rauchenzauner, Markus; Neururer, Sabrina; Geiger, Ralf; Högler, Wolfgang

doi:10.1007/s00431-014-2454-8

Cited by 33 publications

(21 citation statements)

References 13 publications

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“…It is interesting to note that BMI markedly decreased in patients 1 and 2. These patients underwent the greatest improvement in height and, although, unfortunately, our study does not provide quantitatively measured objective data on musculoskeletal function (15), this positive effect on weight and body composition likely resulted from the higher physical activity of children, a fact disclosed by the patients' parents in the clinical interviews. Likewise, families reported the interruption of frequent oral doses of phosphate as an appreciable benefit linked to the new treatment.…”

Section: Discussionmentioning

confidence: 75%

Positive Response to One-Year Treatment With Burosumab in Pediatric Patients With X-Linked Hypophosphatemia

et al. 2020

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Section: Discussionmentioning

confidence: 75%

Positive Response to One-Year Treatment With Burosumab in Pediatric Patients With X-Linked Hypophosphatemia

et al. 2020

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“…Such an evaluation should include an assessment of limb length and alignment (in both the coronal and sagittal planes) as well as the torsional profile of the lower limb. Yearly assessment of the 6-minute walk test (6MWT) in patients >5-6 years might help to quantify the functional consequences of XLH on bone and muscles 77 . Patients should have at least twice-yearly dental examinations after tooth eruption, orthodontic evaluation around the age of 12 years and an extended dental evaluation with transition to adult care.…”

Section: Follow-up Of Patients With Xlhmentioning

confidence: 99%

“…Postoperative assessment of the axis correction should be documented at 12 months. Functional assessment should be performed according to the World Health Organization (WHO) International Classification of Function using tools such as the Pediatric Outcomes Data Collection Instrument (PODCI) or the 6MWT 77,170 . As patients transition to adult services, a full orthopaedic clinical and radiographic assessment enables definition of any residual deformity and facilitates appropriate follow-up arrangements.…”

Section: Assessment Of Surgical Outcomesmentioning

confidence: 99%

“…-minute walk test77 ; ALP, alkaline phosphatase; BAP, bone alkaline phosphatase; eGFR , estimated glomerular filtration rate195,196 ; ICD, intercondylar distance (reference values are given here 73 ); IMD, intermalleolar distance; NA , not applicable; PTH, parathyroid hormone; QOL , quality of life; TmP/GFR , maximum rate of renal tubular reabsorption of phosphate per glomerular filtration rate. a These examinations should also be performed at the time of transition to adult care.…”

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confidence: 99%

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Clinical practice recommendations for the diagnosis and management of X-linked hypophosphataemia

et al. 2019

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X-linked hypophosphataemia (XLH) is an X-linked dominant disorder caused by mutations in PHEX (located at Xp22.1), which encodes a cell-surface-bound protein-cleavage enzyme (phosphate-regulating neutral endopeptidase PHEX), predominantly expressed in osteoblasts, osteocytes and teeth (odontoblasts and cementoblasts). XLH is the most common cause of inherited phosphate wasting, with an incidence of 3.9 per 100,000 live births and a prevalence ranging from 1.7 per 100,000 children to 4.8 per 100,000 persons (children and adults) 1-3. Although the pathogenesis of XLH is not fully understood, animal studies indicate that loss of Phex function results in enhanced secretion of the phosphaturic hormone fibroblast growth factor 23 (FGF23), with osteocytes being the primary source of FGF23 production 4. These effects explain most of the characteristic features of the disease, including renal phosphate wasting with consequent hypophosphataemia, diminished synthesis of active vitamin D (1,25(OH) 2 vitamin D), rickets, osteomalacia, odontomalacia and disproportionate short stature 4-6. Patients usually develop clinical symptoms during the first or second year of life. Early treatment with oral phosphate supplementation and active vitamin D heals rickets, limits dental abscess formation and prevents progressive growth failure, but in a substantial proportion of patients treatment is unsuccessful and/or associated with adverse effects (for example, hyper parathyroidism and nephrocalcinosis) 7,8. Up to two-thirds of children with XLH require lower limb surgery 9-12. Conventional therapy further stimulates FGF23 levels and thereby renal phosphate wasting, resulting in a vicious circle, which might limit its efficacy 6,13-15. Adult patients with XLH are at risk of complications such as early osteoarthritis, enthesopathies, spinal stenosis, pseudofractures and hearing loss, which might limit quality of life 16-18. In 2018, burosumab, a fully human monoclonal IgG1 antibody neutralizing FGF23, was approved by health authorities for the treatment of patients with

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“…Various functional tests are being used, for example, the 6-minute walk test [45,46] , Bruininks-Oseretsky Test of Motor Proficiency, Second Edition [47] , the Gross Motor Function Measure [48] , the Childhood Health Assessment Questionnaire [49] , and the widely used FACES Pain Scale [50] . Specific muscle force and power tests include the chair-rising test, mechanography (legs) [51,52] and grip force testing by dynamometry [53] , amongst others.…”

Section: Diagnostic Tools For Mobility Muscle and Functionmentioning

confidence: 99%

Osteoporosis in Children with Chronic Disease

Högler

Ward²

2015

Calcium and Bone Disorders in Children and Adolescents

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Serious illness in children and its therapy can cause osteoporosis, manifesting as vertebral and nonvertebral fractures, pain, skeletal deformity and temporary or even permanent loss of ambulation. In contrast to adults, skeletal growth in children offers tremendous potential to recover bone mineral density and to reshape fractured vertebral bodies, even without bone-targeted therapy, provided that bone health threats are transient and residual growth is sufficient. Here, we outline the principles of bone strength development and the risk factors for osteoporosis due to various paediatric systemic illnesses. We also explain why the approach to the diagnosis and monitoring of childhood osteoporosis has moved away from a bone density-centric focus to a more functional assessment. Finally, we discuss the best candidates for and current approaches to the treatment of osteoporosis in children.

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Sex-, age-, and height-specific reference curves for the 6-min walk test in healthy children and adolescents

Abstract: These reference curves will allow more accurate grading of mobility and exercise capacity in sick or disabled children and monitoring the effects of intervention or treatment.

Cited by 33 publications

References 13 publications

Positive Response to One-Year Treatment With Burosumab in Pediatric Patients With X-Linked Hypophosphatemia

Positive Response to One-Year Treatment With Burosumab in Pediatric Patients With X-Linked Hypophosphatemia

Clinical practice recommendations for the diagnosis and management of X-linked hypophosphataemia

Osteoporosis in Children with Chronic Disease

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