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Bone & Joint Research
Vol. 10, Issue 9 | Pages 619 - 628
27 Sep 2021
Maestro-Paramio L García-Rey E Bensiamar F Saldaña L

Aims. To investigate whether idiopathic osteonecrosis of the femoral head (ONFH) is related to impaired osteoblast activities. Methods. We cultured osteoblasts isolated from trabecular bone explants taken from the femoral head and the intertrochanteric region of patients with idiopathic ONFH, or from the intertrochanteric region of patients with osteoarthritis (OA), and compared their viability, mineralization capacity, and secretion of paracrine factors. Results. Osteoblasts from the intertrochanteric region of patients with ONFH showed lower alkaline phosphatase (ALP) activity and mineralization capacity than osteoblasts from the same skeletal site in age-matched patients with OA, as well as lower messenger RNA (mRNA) levels of genes encoding osteocalcin and bone sialoprotein and higher osteopontin expression. In addition, osteoblasts from patients with ONFH secreted lower osteoprotegerin (OPG) levels than those from patients with OA, resulting in a higher receptor activator of nuclear factor kappa-light-chain-enhancer of activated B cells (NF-κB) ligand (RANKL)-to-OPG ratio. In patients with ONFH, osteoblasts from the femoral head showed reduced viability and mineralized nodule formation compared with osteoblasts from the intertrochanteric region. Notably, the secretion of the pro-resorptive factors interleukin-6 and prostaglandin E. 2. as well as the RANKL-to-OPG ratio were markedly higher in osteoblast cultures from the femoral head than in those from the intertrochanteric region. Conclusion. Idiopathic ONFH is associated with a reduced mineralization capacity of osteoblasts and increased secretion of pro-resorptive factors. Cite this article: Bone Joint Res 2021;10(9):619–628


The Bone & Joint Journal
Vol. 105-B, Issue 4 | Pages 439 - 448
15 Mar 2023
Hong H Pan X Song J Fang N Yang R Xiang L Wang X Huang C

Aims. The prevalence of scoliosis is not known in patients with idiopathic short stature, and the impact of treatment with recombinant human growth hormone on those with scoliosis remains controversial. We investigated the prevalence of scoliosis radiologically in children with idiopathic short stature, and the impact of treatment with growth hormone in a cross-sectional and retrospective cohort study. Methods. A total of 2,053 children with idiopathic short stature and 4,106 age- and sex-matched (1:2) children without short stature with available whole-spine radiographs were enrolled in the cross-sectional study. Among them, 1,056 with idiopathic short stature and 790 controls who had radiographs more than twice were recruited to assess the development and progression of scoliosis, and the need for bracing and surgery. Results. In the cross-sectional study, there was an unexpectedly higher prevalence of scoliosis (33.1% (681/2,053) vs 8.52% (350/4,106)) in children with idiopathic short stature compared with controls (odds ratio 3.722; p < 0.001), although most cases were mild. In the longitudinal study, children with idiopathic short stature had a higher risk of the development and progression of scoliosis than the controls. Among children with idiopathic short stature without scoliosis at baseline, treatment with growth hormone significantly increased the risk of developing scoliosis (p = 0.015) and the need for bracing (p < 0.001). Among those with idiopathic short stature and scoliosis at baseline, treatment with growth hormone did not increase the risk of progression of the scoliosis, the need for bracing, or surgery. Conclusion. The impact of treatment with growth hormone on scoliosis in children with idiopathic short stature was considered controllable. However, physicians should pay close attention to the assessment of spinal curves in these children. Cite this article: Bone Joint J 2023;105-B(4):439–448


Bone & Joint Open
Vol. 2, Issue 4 | Pages 255 - 260
15 Apr 2021
Leo DG Russell A Bridgens A Perry DC Eastwood DM Gelfer Y

Aims. This study aims to define a set of core outcomes (COS) to allow consistent reporting in order to compare results and assist in treatment decisions for idiopathic clubfoot. Methods. A list of outcomes will be obtained in a three-stage process from the literature and from key stakeholders (patients, parents, surgeons, and healthcare professionals). Important outcomes for patients and parents will be collected from a group of children with idiopathic clubfoot and their parents through questionnaires and interviews. The outcomes identified during this process will be combined with the list of outcomes previously obtained from a systematic review, with each outcome assigned to one of the five core areas defined by the Outcome Measures Recommended for use in Randomized Clinical Trials (OMERACT). This stage will be followed by a two round Delphi survey aimed at key stakeholders in the management of idiopathic clubfoot. The final outcomes list obtained will then be discussed in a consensus meeting of representative key stakeholders. Conclusion. The inconsistency in outcomes reporting in studies investigating idiopathic clubfoot has made it difficult to define the success rate of treatments and to compare findings between studies. The development of a COS seeks to define a minimum standard set of outcomes to collect in all future clinical trials for this condition, to facilitate comparisons between studies and to aid decisions in treatment. Cite this article: Bone Jt Open 2021;2(4):255–260


The Bone & Joint Journal
Vol. 104-B, Issue 6 | Pages 758 - 764
1 Jun 2022
Gelfer Y Davis N Blanco J Buckingham R Trees A Mavrotas J Tennant S Theologis T

Aims. The aim of this study was to gain an agreement on the management of idiopathic congenital talipes equinovarus (CTEV) up to walking age in order to provide a benchmark for practitioners and guide consistent, high-quality care for children with CTEV. Methods. The consensus process followed an established Delphi approach with a predetermined degree of agreement. The process included the following steps: establishing a steering group; steering group meetings, generating statements, and checking them against the literature; a two-round Delphi survey; and final consensus meeting. The steering group members and Delphi survey participants were all British Society of Children’s Orthopaedic Surgery (BSCOS) members. Descriptive statistics were used for analysis of the Delphi survey results. The Appraisal of Guidelines for Research & Evaluation checklist was followed for reporting of the results. Results. The BSCOS-selected steering group, the steering group meetings, the Delphi survey, and the final consensus meeting all followed the pre-agreed protocol. A total of 153/243 members voted in round 1 Delphi (63%) and 132 voted in round 2 (86%). Out of 61 statements presented to round 1 Delphi, 43 reached ‘consensus in’, no statements reached ‘consensus out’, and 18 reached ‘no consensus’. Four statements were deleted and one new statement added following suggestions from round 1. Out of 15 statements presented to round 2, 12 reached ‘consensus in’, no statements reached ‘consensus out’, and three reached ‘no consensus’ and were discussed and included following the final consensus meeting. Two statements were combined for simplicity. The final consensus document includes 57 statements allocated into six successive stages. Conclusion. We have produced a consensus document for the treatment of idiopathic CTEV up to walking age. This will provide a benchmark for standard of care in the UK and will help to reduce geographical variability in treatment and outcomes. Appropriate dissemination and implementation will be key to its success. Cite this article: Bone Joint J 2022;104-B(6):758–764


Aims. The study was undertaken to compare the efficacy of Woodcast splints and plaster-of-Paris casts in maintaining correction following sequential manipulation of idiopathic clubfeet. Methods. In this randomized prospective trial, 23 idiopathic clubfeet were immobilized with plaster-of-Paris casts and 23 clubfeet were immobilized with a splint made of Woodcast that encircled only two-thirds the circumference of the limb. The number of casts or splints needed to obtain full correction, the frequency of cast or splint-related complications, and the time taken for application and removal of the casts and splints were compared. Results. The mean number of casts required to obtain full correction of the deformity (Pirani Score 0) was 4.35 (95% confidence interval (CI) 3.74 to 4.95) when plaster-of-Paris was used and 4.87 (95% CI 4.33 to 5.41) when the Woodcast splint was used (p = 0.190). The time required for application and removal of the Woodcast splint were significantly less than that required for application and removal of plaster-of-Paris casts (p < 0.001). Woodcast splint-related complications were not more frequent than plaster-of-Paris cast related complications. Conclusion. Though Woodcast splints covering two-thirds of the circumference of the lower limbs of infants were effective in maintaining the correction of clubfoot deformity during serial manipulation and casting treatment, the superiority of Woodcasts over plaster-of-Paris could not be established. Cite this article: Bone Joint J 2020;102-B(10):1399–1404


Bone & Joint Open
Vol. 3, Issue 1 | Pages 98 - 106
27 Jan 2022
Gelfer Y Leo DG Russell A Bridgens A Perry DC Eastwood DM

Aims. To identify the minimum set of outcomes that should be collected in clinical practice and reported in research related to the care of children with idiopathic congenital talipes equinovarus (CTEV). Methods. A list of outcome measurement tools (OMTs) was obtained from the literature through a systematic review. Further outcomes were collected from patients and families through a questionnaire and interview process. The combined list, as well as the appropriate follow-up timepoint, was rated for importance in a two-round Delphi process that included an international group of orthopaedic surgeons, physiotherapists, nurse practitioners, patients, and families. Outcomes that reached no consensus during the Delphi process were further discussed and scored for inclusion/exclusion in a final consensus meeting involving international stakeholder representatives of practitioners, families, and patient charities. Results. In total, 39 OMTs were included from the systematic review. Two additional OMTs were identified from the interviews and questionnaires, and four were added after round one Delphi. Overall, 22 OMTs reached ‘consensus in’ during the Delphi and two reached ‘consensus out’; 21 OMTs reached ‘no consensus’ and were included in the final consensus meeting. In all, 21 participants attended the consensus meeting, including a wide diversity of clubfoot practitioners, parent/patient representative, and an independent chair. A total of 21 outcomes were discussed and voted upon; six were voted ‘in’ and 15 were voted ‘out’. The final COS document includes nine OMTs and two existing outcome scores with a total of 31 outcome parameters to be collected after a minimum follow-up of five years. It incorporates static and dynamic clinical findings, patient-reported outcome measures, and a definition of CTEV relapse. Conclusion. We have defined a minimum set of outcomes to draw comparisons between centres and studies in the treatment of CTEV. With the use of these outcomes, we hope to allow more meaningful research and a better clinical management of CTEV. Cite this article: Bone Jt Open 2022;3(1):98–106


The Bone & Joint Journal
Vol. 106-B, Issue 7 | Pages 735 - 743
1 Jul 2024
Gelfer Y Cavanagh SE Bridgens A Ashby E Bouchard M Leo DG Eastwood DM

Aims. There is a lack of high-quality research investigating outcomes of Ponseti-treated idiopathic clubfeet and correlation with relapse. This study assessed clinical and quality of life (QoL) outcomes using a standardized core outcome set (COS), comparing children with and without relapse. Methods. A total of 11 international centres participated in this institutional review board-approved observational study. Data including demographics, information regarding presentation, treatment, and details of subsequent relapse and management were collected between 1 June 2022 and 30 June 2023 from consecutive clinic patients who had a minimum five-year follow-up. The clubfoot COS incorporating 31 parameters was used. A regression model assessed relationships between baseline variables and outcomes (clinical/QoL). Results. Overall, 293 patients (432 feet) with a median age of 89 months (interquartile range 72 to 113) were included. The relapse rate was 37%, with repeated relapse in 14%. Treatment considered a standard part of the Ponseti journey (recasting, repeat tenotomy, and tibialis anterior tendon transfer) was performed in 35% of cases, with soft-tissue release and osteotomies in 5% and 2% of cases, respectively. Predictors of relapse included duration of follow-up, higher initial Pirani score, and poor Evertor muscle activity. Relapse was associated with poorer outcomes. Conclusion. This is the first multicentre study using a standardized COS following clubfoot treatment. It distinguishes patients with and without relapse in terms of clinical outcomes and QoL, with poorer outcomes in the relapse group. This tool allows comparison of treatment methods and outcomes, facilitates information sharing, and sets family expectations. Predictors of relapse encourage us to create appropriate treatment pathways to reduce relapse and improve outcome. Cite this article: Bone Joint J 2024;106-B(7):735–743


Bone & Joint Open
Vol. 1, Issue 8 | Pages 457 - 464
1 Aug 2020
Gelfer Y Hughes KP Fontalis A Wientroub S Eastwood DM

Aims. To analyze outcomes reported in studies of Ponseti correction of idiopathic clubfoot. Methods. A systematic review of the literature was performed to identify a list of outcomes and outcome tools reported in the literature. A total of 865 studies were screened following Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines, and 124 trials were included in the analysis. Data extraction was completed by two researchers for each trial. Each outcome tool was assigned to one of the five core areas defined by the Outcome Measures Recommended for use in Randomized Clinical Trials (OMERACT). Bias assessment was not deemed necessary for the purpose of this paper. Results. In total, 20 isolated outcomes and 16 outcome tools were identified representing five OMERACT domains. Most outcome tools were appropriately designed for children of walking age but have not been embraced in the literature. The most commonly reported isolated outcomes are subjective and qualitative. The quantitative outcomes most commonly used are ankle range of motion (ROM), foot position in standing, and muscle function. Conclusions. There is a diverse range of outcomes reported in studies of Ponseti correction of clubfoot. Until outcomes can be reported unequivocally and consistently, research in this area will be limited. Completing the process of establishing and validating COS is the much-needed next step. Cite this article: Bone Joint Open 2020;1-8:457–464


The Bone & Joint Journal
Vol. 96-B, Issue 9 | Pages 1264 - 1268
1 Sep 2014
Gelfer Y Dunkley M Jackson D Armstrong J Rafter C Parnell E Eastwood DM

Previous studies have identified clinical and demographic risk factors for recurrence in the treatment of idiopathic clubfoot (congenital talipes equinovarus). Evertor muscle activity is not usually considered amongst them. This study aimed to evaluate whether recurrence could be predicted by demographic, clinical and gait parameters. From a series of 103 children with clubfeet, 67 had completed a follow-up of two years: 41 male and 26 female, 38 with idiopathic and 29 with non-idiopathic deformities. The mean age was 3.2 years (2.1 to 6.3). Primary correction was obtained in all 38 children (100%) with an idiopathic deformity, and in 26 of 29 patients (90%) with a non-idiopathic deformity. Overall, 60 children (90%) complied with the abduction brace regime. At a mean follow-up of 31.4 months (24 to 62), recurrence was noted in six children (15.8%) in the idiopathic and 14 children (48.3%) in the non-idiopathic group. Significant correlation was found between poor evertor activity and recurrence in both groups. No statistically significant relationship was found between the rate of recurrence and the severity of the initial deformity, the age at the time of treatment, the number of casts required or the compliance with the brace. After correction of idiopathic and non-idiopathic clubfoot using the Ponseti method, only poor evertor muscle activity was statistically associated with recurrence. The identification of risk factors for recurrent deformity allows clinicians to anticipate problems and advocate early additional treatment to improve muscle balance around the ankle. Cite this article: Bone Joint J 2014;96-B:1264–8


The Bone & Joint Journal
Vol. 96-B, Issue 10 | Pages 1424 - 1426
1 Oct 2014
Mayne AIW Bidwai AS Beirne P Garg NK Bruce CE

We report the effect of introducing a dedicated Ponseti service on the five-year treatment outcomes of children with idiopathic clubfoot. Between 2002 and 2004, 100 feet (66 children; 50 boys and 16 girls) were treated in a general paediatric orthopaedic clinic. Of these, 96 feet (96%) responded to initial casting, 85 requiring a tenotomy of the tendo-Achillis. Recurrent deformity occurred in 38 feet and was successfully treated in 22 by repeat casting and/or tenotomy and/or transfer of the tendon of tibialis anterior, The remaining 16 required an extensive surgical release. . Between 2005 and 2006, 72 feet (53 children; 33 boys and 20 girls) were treated in a dedicated multidisciplinary Ponseti clinic. All responded to initial casting: 60 feet (83.3%) required a tenotomy of the tendo-Achillis. Recurrent deformity developed in 14, 11 of which were successfully treated by repeat casting and/or tenotomy and/or transfer of the tendon of tibialis anterior. The other three required an extensive surgical release. . Statistical analysis showed that children treated in the dedicated Ponseti clinic had a lower rate of recurrence (p = 0.068) and a lower rate of surgical release (p = 0.01) than those treated in the general clinic. This study shows that a dedicated Ponseti clinic, run by a well-trained multidisciplinary team, can improve the outcome of idiopathic clubfoot deformity. Cite this article: Bone Joint J 2014;96-B:1424–6


The Journal of Bone & Joint Surgery British Volume
Vol. 89-B, Issue 3 | Pages 378 - 381
1 Mar 2007
Lourenço AF Morcuende JA

The Ponseti method of treating club foot has been shown to be effective in children up to two years of age. However, it is not known whether it is successful in older children. We retrospectively reviewed 17 children (24 feet) with congenital idiopathic club foot who presented after walking age and had undergone no previous treatment. All were treated by the method described by Ponseti, with minor modifications. The mean age at presentation was 3.9 years (1.2 to 9.0) and the mean follow-up was for 3.1 years (2.1 to 5.6). The mean time of immobilisation in a cast was 3.9 months (1.5 to 6.0). A painless plantigrade foot was obtained in 16 feet without the need for extensive soft-tissue release and/or bony procedures. Four patients (7 feet) had recurrent equinus which required a second tenotomy. Failure was observed in five patients (8 feet) who required a posterior release for full correction of the equinus deformity. We conclude that the Ponseti method is a safe, effective and low-cost treatment for neglected idiopathic club foot presenting after walking age


The Bone & Joint Journal
Vol. 98-B, Issue 9 | Pages 1270 - 1275
1 Sep 2016
Park S Kang S Kim JY

Aims. Our aim was to investigate the predictive factors for the development of a rebound phenomenon after temporary hemiepiphysiodesis in children with genu valgum. Patients and Methods. We studied 37 limbs with idiopathic genu valgum who were treated with hemiepiphyseal stapling, and with more than six months remaining growth at removal of the staples. All children were followed until skeletal maturity or for more than two years after removal of the staples. Results. On multivariate logistic regression analysis, the rate of correction, body mass index (BMI), age, and initial valgus angle were significantly associated with a rebound phenomenon. With those characteristics, a predictive model for rebound was generated using recursive partitioning analysis. Children with a rapid rate of correction had the most frequent and severe rebound phenomenon (incidence 79%, mean 4°), whereas those with a slow rate of correction had less rebound when they had low BMI (43%, 2°) and none when the BMI was ≥ 21 kg/m. 2. . Conclusion. This is the first study to evaluate a predictive model for a rebound phenomenon after temporary hemiepiphysiodesis in children with idiopathic genu valgum. Cite this article: Bone Joint J 2016;98-B:1270–5


The Journal of Bone & Joint Surgery British Volume
Vol. 91-B, Issue 10 | Pages 1400 - 1402
1 Oct 2009
Imam S Aldridge C Lyall H

Whereas avascular necrosis of the scaphoid after a fracture is well-documented, idiopathic avascular necrosis of the scaphoid (Preiser’s disease) is rare. Little is known of the aetiology of the condition and even less about the best course of management. We describe a rare case of bilateral Preiser’s disease. Possible aetiological factors and a summary of the current concepts of management are discussed


The Journal of Bone & Joint Surgery British Volume
Vol. 90-B, Issue 4 | Pages 512 - 514
1 Apr 2008
Nicoletti S Salama A Stanley D

We present a case of idiopathic osteonecrosis of the humeral capitellum in a 44-year-old female in the absence of any associated risk factors. Arthroscopy was undertaken to remove the loose bodies and debride the capitellum, with a satisfactory outcome


The Journal of Bone & Joint Surgery British Volume
Vol. 82-B, Issue 6 | Pages 881 - 884
1 Aug 2000
Schaefer D Hefti F

We used a combined cuboid/cuneiform osteotomy to treat residual adductus deformity in idiopathic and secondary club feet. The mean follow-up for 27 feet (22 idiopathic, four arthrogrypotic and one related to amniotic band syndrome) was 5.0 years (2.0 to 9.8). All healed uneventfully except for one early wound infection. No further surgery was required in the 22 idiopathic club feet but four of five with secondary deformity needed further surgery. At follow-up all patients with idiopathic and two with secondary club feet were free from pain and satisfied with the result. In the idiopathic feet, adductus of the forefoot, as measured by the calcaneal second metatarsal angle, improved on average from 20.7 ± 2.0° to 8.9 ± 1.8° (p < 0.05). In four feet, with a follow-up of more than six years, there was complete recurrence of the deformity. In the secondary club feet, there was no improvement of the adductus. We conclude that in most, but not all, idiopathic club feet a cuboid/cuneiform osteotomy can provide satisfactory correction of adductus deformity. Those with secondary deformity require other procedures


The Journal of Bone & Joint Surgery British Volume
Vol. 77-B, Issue 4 | Pages 596 - 601
1 Jul 1995
Feldbrin Z Gilai A Ezra E Khermosh O Kramer U Wientroub S

We performed electrophysiological studies on both legs of 52 children, aged from 3 months to 15 years, with idiopathic club foot. In only nine (17%) was no abnormality found. Isolated peroneal nerve damage was seen in 14 (27%). Abnormality of both peroneal and posterior tibial nerves was found in five (10%). Four patients (8%) had evidence of isolated spinal-cord dysfunction, whereas combined spinal-cord and peripheral-nerve lesions were seen in 14 (27%). Six patients (11%) had variable neurogenic electrophysiological patterns. In 13 patients in whom the studies were repeated neither progression nor improvement of the electrophysiological parameters was observed. Pathological electrophysiological findings were found in 66% of conservatively-treated patients. In the 43 patients treated surgically, all 16 with fair and poor results had pathological electrophysiological findings and 12 required further operations. Multiplicity of the pathological findings was related to the severity of the deformity of the foot; normal studies represent a good prognostic sign. Electrophysiological studies are useful in idiopathic club foot with residual deformities after conservative or operative treatment. Our findings support the theory that muscle imbalance is an aetiological factor in idiopathic club foot


The Journal of Bone & Joint Surgery British Volume
Vol. 61-B, Issue 2 | Pages 138 - 143
1 May 1979
Inoue A Ono K

We have studied the histological appearances of forty femoral heads with idiopathic avascular necrosis. The characteristic histopathological changes of recurrent necrosis were present in 83 per cent. Recurrent necrosis occurred widely after revascularisation had progressed as far as the subchondral zone. The aetiology of idiopathic avascular necrosis of the femoral head may be a chronic condition which produces repeated infarction. The deformation and incomplete revascularisation of the femoral head may be due to repeated episodes of infarction as well as to mechanical factors related to weight-bearing


The Journal of Bone & Joint Surgery British Volume
Vol. 88-B, Issue 10 | Pages 1385 - 1387
1 Oct 2006
Changulani M Garg NK Rajagopal TS Bass A Nayagam SN Sampath J Bruce CE

We report our initial experience of using the Ponseti method for the treatment of congenital idiopathic club foot. Between November 2002 and November 2004 we treated 100 feet in 66 children by this method. The standard protocol described by Ponseti was used except that, when necessary, percutaneous tenotomy of tendo Achillis were performed under general anaesthesia in the operating theatre and not under local anaesthesia in the out-patient department. The Pirani score was used for assessment and the mean follow-up time was 18 months (6 to 30). The results were also assessed in terms of the number of casts applied, the need for tenotomy of tendo Achillis and recurrence of the deformity. Tenotomy was required in 85 of the 100 feet. There was a failure to respond to the initial regimen in four feet which then required extensive soft-tissue release. Of the 96 feet which responded to initial casting, 31 (32%) had a recurrence, 16 of which were successfully treated by repeat casting and/or tenotomy and/or transfer of the tendon of tibialis anterior. The remaining 15 required extensive soft-tissue release. Poor compliance with the foot-abduction orthoses (Denis Browne splint) was thought to be the main cause of failure in these patients


The Journal of Bone & Joint Surgery British Volume
Vol. 88-B, Issue 3 | Pages 377 - 379
1 Mar 2006
Barker SL Lavy CBD

Achilles tenotomy is a recognised step in the Ponseti technique for the correction of idiopathic congenital talipes equinovarus in most percutaneous cases. Its use has been limited in part by concern that the subsequent natural history of the tendon is unknown. In a study of 11 tendons in eight infants, eight tendons were shown to be clinically intact and ten had ultrasonographic evidence of continuity three weeks after tenotomy. At six weeks after tenotomy all tendons had both clinical and ultrasonographic evidence of continuity


The Bone & Joint Journal
Vol. 95-B, Issue 10 | Pages 1308 - 1316
1 Oct 2013
Stokes OM Luk KDK

Adolescent idiopathic scoliosis affects about 3% of children. Non-operative measures are aimed at altering the natural history to maintain the size of the curve below 40° at skeletal maturity. The application of braces to treat spinal deformity pre-dates the era of evidence-based medicine, and there is a paucity of irrefutable prospective evidence in the literature to support their use and their effectiveness has been questioned.

This review considers this evidence. The weight of the evidence is in favour of bracing over observation. The most recent literature has moved away from addressing this question, and instead focuses on developments in the design of braces and ways to improve compliance.

Cite this article: Bone Joint J 2013;95-B:1308–16.