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The Bone & Joint Journal
Vol. 106-B, Issue 3 | Pages 277 - 285
1 Mar 2024
Pinto D Hussain S Leo DG Bridgens A Eastwood D Gelfer Y

Aims. Children with spinal dysraphism can develop various musculoskeletal deformities, necessitating a range of orthopaedic interventions, causing significant morbidity, and making considerable demands on resources. This systematic review aimed to identify what outcome measures have been reported in the literature for children with spinal dysraphism who undergo orthopaedic interventions involving the lower limbs. Methods. A PROSPERO-registered systematic literature review was performed following PRISMA guidelines. All relevant studies published until January 2023 were identified. Individual outcomes and outcome measurement tools were extracted verbatim. The measurement tools were assessed for reliability and validity, and all outcomes were grouped according to the Outcome Measures Recommended for use in Randomized Clinical Trials (OMERACT) filters. Results. From 91 eligible studies, 27 individual outcomes were identified, including those related to clinical assessment (n = 12), mobility (n = 4), adverse events (n = 6), investigations (n = 4), and miscellaneous (n = 1). Ten outcome measurement tools were identified, of which Hoffer’s Functional Ambulation Scale was the most commonly used. Several studies used unvalidated measurement tools originally developed for other conditions, and 26 studies developed new measurement tools. On the OMERACT filter, most outcomes reported pathophysiology and/or the impact on life. There were only six patient- or parent-reported outcomes, and none assessed the quality of life. Conclusion. The outcomes that were reported were heterogenous, lack validation and failed to incorporate patient or family perceptions. Until outcomes can be reported unequivocally, research in this area will remain limited. Our findings should guide the development of a core outcome set, which will allow consistency in the reporting of outcomes for this condition. Cite this article: Bone Joint J 2024;106-B(3):277–285


The Bone & Joint Journal
Vol. 102-B, Issue 4 | Pages 495 - 500
1 Apr 2020
Milligan DJ Cosgrove AP

Aims. To monitor the performance of services for developmental dysplasia of the hip (DDH) in Northern Ireland and identify potential improvements to enhance quality of service and plan for the future. Methods. This was a prospective observational study, involving all infants treated for DDH between 2011 and 2017. Children underwent clinical assessment and radiological investigation as per the regional surveillance policy. The regional radiology data was interrogated to quantify the use of ultrasound and ionizing radiation for this population. Results. Evidence-based changes were made to the Northern Ireland screening programme, including an increase in ultrasound scanning capacity and expansion of nurse-led screening clinics. The number of infant hip ultrasound scans increased from 4,788 in 2011, to approximately 7,000 in 2013 and subsequent years. The number of hip radiographs on infants of less than one year of age fell from 7,381 to 2,208 per year. There was a modest increase in the treatment rate from 10.9 to 14.3 per 1,000 live births but there was a significant reduction in the number of closed hip reductions. The incidence of infants diagnosed with DDH after one year of age was 0.30 per 1,000 live births over the entire period. Conclusion. Improving compliance with the regional infant hip screening protocols led to reduction in operative procedures and reduced the number of pelvic radiographs of infants. We conclude that performance monitoring of screening programmes for DDH is essential to provide a quality service. Cite this article: Bone Joint J 2020;102-B(4):495–500


The Bone & Joint Journal
Vol. 104-B, Issue 9 | Pages 1081 - 1088
1 Sep 2022
Behman AL Bradley CS Maddock CL Sharma S Kelley SP

Aims

There is no consensus regarding optimum timing and frequency of ultrasound (US) for monitoring response to Pavlik harness (PH) treatment in developmental dysplasia of the hip (DDH). The purpose of our study was to determine if a limited-frequency hip US assessment had an adverse effect on treatment outcomes compared to traditional comprehensive US monitoring.

Methods

This study was a single-centre noninferiority randomized controlled trial. Infants aged under six months whose hips were reduced and centred in the harness at initiation of treatment (stable dysplastic or subluxable), or initially decentred (subluxated or dislocated) but reduced and centred within four weeks of PH treatment, were randomized to our current standard US monitoring protocol (every clinic visit) or to a limited-frequency US protocol (US only at end of treatment). Groups were compared based on α angle and femoral head coverage at the end of PH treatment, acetabular indices, and International Hip Dysplasia Institute (IHDI) grade on one-year follow-up radiographs.


The Journal of Bone & Joint Surgery British Volume
Vol. 91-B, Issue 10 | Pages 1388 - 1393
1 Oct 2009
Aird JJ Hogg A Rollinson P

In 1937 Blount described a series of 28 patients with ‘Tibia vara’. Since then, a number of deformities in the tibia and the femur have been described in association with this condition. We analysed 14 children with Blount’s disease who were entered into a cross-sectional study. Their mean age was 10 (2 to 18). They underwent a clinical assessment of the rotational profile of their legs and a CT assessment of the angle of anteversion of their hips (femoral version). We compared our results to previously published controls. A statistically significant increase in femoral anteversion was noted in the affected legs, with on average the femurs in patients with Blount’s disease being 26° more anteverted than those in previously published controls. We believe this to be a previously unrecognised component of Blount’s disease, and that the marked intoeing seen in the disease may be partly caused by internal femoral version, in addition to the well-recognised internal tibial version


The Bone & Joint Journal
Vol. 102-B, Issue 5 | Pages 611 - 617
1 May 2020
Leo DG Jones H Murphy R Leong JW Gambling T Long AF Laine J Perry DC

Aims

To identify a suite of the key physical, emotional, and social outcomes to be employed in clinical practice and research concerning Perthes' disease in children.

Methods

The study follows the guidelines of the COMET-Initiative (Core Outcome Measures in Effectiveness Trials). A systematic review of the literature was performed to identify a list of outcomes reported in previous studies, which was supplemented by a qualitative study exploring the experiences of families affected by Perthes’ disease. Collectively, these outcomes formed the basis of a Delphi survey (two rounds), where 18 patients with Perthes’ disease, 46 parents, and 36 orthopaedic surgeons rated each outcome for importance. The International Perthes Study Group (IPSG) (Dallas, Texas, USA (October 2018)) discussed outcomes that failed to reach any consensus (either ‘in’ or ‘out’) before a final consensus meeting with representatives of surgeons, patients, and parents.


The Journal of Bone & Joint Surgery British Volume
Vol. 85-B, Issue 4 | Pages 572 - 577
1 May 2003
Theologis TN Harrington ME Thompson N Benson MKD

The aim of this study was to define objectively gait function in children with treated congenital talipes equinovarus (CTEV) and a good clinical result. The study also attempted an analysis of movement within the foot during gait. We compared 20 children with treated CTEV with 15 control subjects. Clinical assessment demonstrated good results from treatment. Three-dimensional gait analysis provided kinematic and kinetic data describing movement and moments at the joints of the lower limb during gait. A new method was used to study movement within the foot during gait. The data on gait showed significantly increased internal rotation of the foot during walking which was partially compensated for by external rotation at the hip. A mild foot drop and reduced plantar flexor power were also observed. Dorsiflexion at the midfoot was significantly increased, which probably compensated for reduced mobility at the hindfoot. Patients treated for CTEV with a good clinical result should be expected to have nearly normal gait and dynamic foot movement, but there may be residual intoeing, mild foot drop, loss of plantar flexor power with compensatory increased midfoot dorsiflexion and external hip rotation


The Bone & Joint Journal
Vol. 100-B, Issue 6 | Pages 806 - 810
1 Jun 2018
Choudry QA Paton RW

Aims

The aim of this prospective cohort study was to evaluate the effectiveness of the neonatal hip instability screening programme.

Patients and Methods

The study involved a four-year observational assessment of a neonatal hip screening programme. All newborns were examined using the Barlow or Ortolani manoeuvre within 72 hours of birth; those with positive findings were referred to a ‘one-stop’ screening clinic for clinical and sonographic assessment of the hip. The results were compared with previous published studies from this unit.


The Bone & Joint Journal
Vol. 100-B, Issue 9 | Pages 1249 - 1252
1 Sep 2018
Humphry S Thompson D Price N Williams PR

Aims

The significance of the ‘clicky hip’ in neonatal and infant examination remains controversial with recent conflicting papers reigniting the debate. We aimed to quantify rates of developmental dysplasia of the hip (DDH) in babies referred with ‘clicky hips’ to our dedicated DDH clinic.

Patients and Methods

A three-year prospective cohort study was undertaken between 2014 and 2016 assessing the diagnosis and treatment outcomes of all children referred specifically with ‘clicky hips’ as the primary reason for referral to our dedicated DDH clinic. Depending on their age, they were all imaged with either ultrasound scan or radiographs.


The Bone & Joint Journal
Vol. 99-B, Issue 11 | Pages 1533 - 1536
1 Nov 2017
Nie K Rymaruk S Paton RW

Aims

A clicky hip is a common referral for clinical and sonographic screening for developmental dysplasia of the hip (DDH). There is controversy regarding whether it represents a true risk factor for pathological DDH. Therefore a 20-year prospective, longitudinal, observational study was undertaken to assess the relationship between the presence of a neonatal clicky hip and pathological DDH.

Patients and Methods

A total of 362 infants from 1997 to 2016 were referred with clicky hips to our ‘one-stop’ paediatric hip screening clinic. Hips were assessed clinically for instability and by ultrasound imaging using a simplified Graf/Harcke classification. Dislocated or dislocatable hips were classified as Graf Type IV hips.


The Bone & Joint Journal
Vol. 99-B, Issue 9 | Pages 1250 - 1255
1 Sep 2017
Talbot C Adam J Paton R

Aims

Despite the presence of screening programmes, infants continue to present with late developmental dysplasia of the hip (DDH), the impact of which is significant. The aim of this study was to assess infants with late presenting dislocation of the hip despite universal clinical neonatal and selective ultrasound screening.

Patients and Methods

Between 01 January 1997 to 31 December 2011, a prospective, longitudinal study was undertaken of a cohort of 64 670 live births. Late presenting dislocation was defined as presentation after three months of age. Diagnosis was confirmed by ultrasound and plain radiography. Patient demographics, referral type, reason for referral, risk factors (breech presentation/strong family history) and clinical and radiological findings were recorded.


The Bone & Joint Journal
Vol. 97-B, Issue 11 | Pages 1572 - 1576
1 Nov 2015
Donnelly KJ Chan KW Cosgrove AP

Developmental dysplasia of the hip (DDH) should be diagnosed as early as possible to optimise treatment. The current United Kingdom recommendations for the selective screening of DDH include a clinical examination at birth and at six weeks. In Northern Ireland babies continue to have an assessment by a health visitor at four months of age. As we continue to see late presentations of DDH, beyond one year of age, we hypothesised that a proportion had missed an opportunity for earlier diagnosis. We expect those who presented to our service with Tonnis grade III or IV hips and decreased abduction would have had clinical signs at their earlier assessments.

We performed a retrospective review of all patients born in Northern Ireland between 2008 and 2010 who were diagnosed with DDH after their first birthday. There were 75 856 live births during the study period of whom 645 children were treated for DDH (8.5 per 1000). The minimum follow-up of our cohort from birth, to detect late presentation, was four years and six months. Of these, 32 children (33 hips) were diagnosed after their first birthday (0.42 per 1000).

With optimum application of our selective screening programme 21 (65.6%) of these children had the potential for an earlier diagnosis, which would have reduced the incidence of late diagnosis to 0.14 per 1000. As we saw a peak in diagnosis between three and five months our findings support the continuation of the four month health visitor check. Our study adds further information to the debate regarding selective versus universal screening.

Cite this article: Bone Joint J 2015;97-B:1572–6.


The Bone & Joint Journal
Vol. 97-B, Issue 2 | Pages 265 - 269
1 Feb 2015
Mace J Paton RW

Over a 15-year prospective period, 201 infants with a clinically unstable hip at neonatal screening were subsequently reviewed in a ‘one stop’ clinic where they were assessed clinically and sonographically. Their mean age was 1.62 weeks (95% confidence interval (CI) 1.35 to 1.89). Clinical neonatal hip screening revealed a sensitivity of 62% (mean, 62.6 95%CI 50.9 to 74.3), specificity of 99.8% (mean, 99.8, 95% CI 99.7 to 99.8) and positive predictive value (PPV) of 24% (mean, 26.2, 95% CI 19.3 to 33.0). Static and dynamic sonography for Graf type IV dysplastic hips had a 15-year sensitivity of 77% (mean, 75.8 95% CI 66.9 to 84.6), specificity of 99.8% (mean, 99.8, 95% CI 99.8 to 99.8) and a PPV of 49% (mean, 55.1, 95% CI 41.6 to 68.5). There were 36 infants with an irreducible dislocation of the hip (0.57 per 1000 live births), including six that failed to resolve with neonatal splintage.

Most clinically unstable hips referred to a specialist clinic are female and stabilise spontaneously. Most irreducible dislocations are not identified from this neonatal instability group. There may be a small subgroup of females with instability of the hip which may be at risk of progression to irreducibility despite early treatment in a Pavlik harness.

A controlled study is required to assess the value of neonatal clinical screening programmes.

Cite this article: Bone Joint J 2015;97-B:265-9.


The Bone & Joint Journal
Vol. 96-B, Issue 5 | Pages 701 - 706
1 May 2014
Dartnell J Gough M Paterson JMH Norman-Taylor F

Proximal femoral resection (PFR) is a proven pain-relieving procedure for the management of patients with severe cerebral palsy and a painful displaced hip. Previous authors have recommended post-operative traction or immobilisation to prevent a recurrence of pain due to proximal migration of the femoral stump. We present a series of 79 PFRs in 63 patients, age 14.7 years (10 to 26; 35 male, 28 female), none of whom had post-operative traction or immobilisation.

A total of 71 hips (89.6%) were reported to be pain free or to have mild pain following surgery. Four children underwent further resection for persistent pain; of these, three had successful resolution of pain and one had no benefit. A total of 16 hips (20.2%) showed radiographic evidence of heterotopic ossification, all of which had formed within one year of surgery. Four patients had a wound infection, one of which needed debridement; all recovered fully. A total of 59 patients (94%) reported improvements in seating and hygiene.

The results are as good as or better than the historical results of using traction or immobilisation. We recommend that following PFR, children can be managed without traction or immobilisation, and can be discharged earlier and with fewer complications. However, care should be taken with severely dystonic patients, in whom more extensive femoral resection should be considered in combination with management of the increased tone.

Cite this article: Bone Joint J 2014; 96-B:701–6.


The Bone & Joint Journal
Vol. 95-B, Issue 6 | Pages 846 - 850
1 Jun 2013
Price KR Dove R Hunter JB

Most centres in the United Kingdom adopt a selective screening programme for developmental dysplasia of the hip (DDH) based on repeated clinical examination and selective ultrasound examination. The Newborn Infant Physical Examination protocol implemented in 2008 recommends a first examination at birth and then a second and final examination at six to ten weeks of age. Due to concerns over an increase in late presentations we performed a retrospective review of our 15-year results to establish if late presentation increases treatment requirements. Of children presenting before six weeks of age, 84% were treated successfully with abduction bracing, whereas 86% of children presenting after ten months eventually required open reduction surgery. This equates to a 12-fold increase in relative risk of requiring open reduction following late presentation. Increasing age at presentation was associated with an increase in the number of surgical procedures, which are inevitably more extensive and complex, with a consequent increased in cost per patient. The implementation of an opportunistic examination at three to five months could help to reduce the unintended consequences of the Newborn Infant Physical Examination programme.

Cite this article: Bone Joint J 2013;95-B:846–50.


The Bone & Joint Journal
Vol. 95-B, Issue 5 | Pages 706 - 713
1 May 2013
Westberry DE Davids JR Anderson JP Pugh LI Davis RB Hardin JW

At our institution surgical correction of symptomatic flat foot deformities in children has been guided by a paradigm in which radiographs and pedobarography are used in the assessment of outcome following treatment. Retrospective review of children with symptomatic flat feet who had undergone surgical correction was performed to assess the outcome and establish the relationship between the static alignment and the dynamic loading of the foot.

A total of 17 children (21 feet) were assessed before and after correction of soft-tissue contractures and lateral column lengthening, using standardised radiological and pedobarographic techniques for which normative data were available.

We found significantly improved static segmental alignment of the foot, significantly improved mediolateral dimension foot loading, and worsened fore-aft foot loading, following surgical treatment. Only four significant associations were found between radiological measures of static segmental alignment and dynamic loading of the foot.

Weakness of the plantar flexors of the ankle was a common post-operative finding. Surgeons should be judicious in the magnitude of lengthening of the plantar flexors that is undertaken and use techniques that minimise subsequent weakening of this muscle group.

Cite this article: Bone Joint J 2013;95-B:706–13.


The Bone & Joint Journal
Vol. 95-B, Issue 5 | Pages 699 - 705
1 May 2013
Chin KF Misra VP Sicuri GM Fox M Sinisi M

We investigated the predictive value of intra-operative neurophysiological investigations in obstetric brachial plexus injuries. Between January 2005 and June 2011 a total of 32 infants of 206 referred to our unit underwent exploration of the plexus, including neurolysis. The findings from intra-operative electromyography, sensory evoked potentials across the lesion and gross muscular response to stimulation were evaluated. A total of 22 infants underwent neurolysis alone and ten had microsurgical reconstruction. Of the former, one was lost to follow-up, one had glenoplasty and three had subsequent nerve reconstructions. Of the remaining 17 infants with neurolysis, 13 (76%) achieved a modified Mallet score > 13 at a mean age of 3.5 years (0.75 to 6.25). Subluxation or dislocation of the shoulder is a major confounding factor. The positive predictive value and sensitivity of the intra-operative EMG for C5 were 100% and 85.7%, respectively, in infants without concurrent shoulder pathology. The positive and negative predictive values, sensitivity and specificity of the three investigations combined were 77%, 100%, 100% and 57%, respectively.

In all, 20 infants underwent neurolysis alone for C6 and three had reconstruction. All of the former and one of the latter achieved biceps function of Raimondi grade 5. The positive and negative predictive values, sensitivity and specificity of electromyography for C6 were 65%, 71%, 87% and 42%, respectively.

Our method is effective in evaluating the prognosis of C5 lesion. Neurolysis is preferred for C6 lesions.

Cite this article: Bone Joint J 2013;95-B:699–705.


The Journal of Bone & Joint Surgery British Volume
Vol. 94-B, Issue 11 | Pages 1579 - 1582
1 Nov 2012
Abdelaziz TH Samir S Magdy W

A total of 35 children with Erb’s palsy and shoulder abduction of < 90° underwent transfer of teres major. In 18 cases (group 1) a trapezius transfer was added (combined procedure). In 17 cases (group 2) teres major transfer was carried out in isolation (single procedure). The mean gain in abduction was 67.2° (60° to 80°) in group 1 and 37.6° (20° to 70°) in group 2, which reached statistical significance (p < 0.001).

Group 2 was further divided into those who had deltoid power of < M3 (group 2a) and those with deltoid power ≥ M3 (group 2b). The difference in improvement of abduction between groups 2a and group 2b was statistically significant (p < 0.001) but the difference between group 2b and group 1 was not (p = 0.07).

We recommend the following protocol of management: in children with abduction ≥ 90° a single procedure is indicated. In children with abduction < 90°: a combined procedure is indicated if deltoid power is < M3 and a single procedure is indicated if deltoid power is ≥ M3. If no satisfactory improvement is achieved, the trapezius can be transferred at a later stage.


The Journal of Bone & Joint Surgery British Volume
Vol. 93-B, Issue 8 | Pages 1122 - 1125
1 Aug 2011
Chandrasenan J Klezl Z Bommireddy R Calthorpe D

We retrospectively reviewed the records of 16 children treated for spondylodiscitis at our hospital between 2000 and 2007. The mean follow-up was 24 months (12 to 38). There was a mean delay in diagnosis in hospital of 25 days in the ten children aged less than 24 months. At presentation only five of the 16 children presented with localising signs and symptoms. Common presenting symptoms were a refusal to walk or sit in nine children, unexplained fever in six, irritability in five, and limping in four. Plain radiography showed changes in only seven children. The ESR was the most useful investigation when following the clinical course of the disease. Positive blood cultures were obtained in seven children with Staphylococcus aureus being isolated in five. Antibiotics were used in 14 children and spinal bracing in six. Children with spondylodiscitis often present with a confusing clinical picture leading to late diagnosis.

The early use of MRI in the investigation of children with an atypical picture may avoid unnecessary delay in starting treatment and possibly prevent long-term problems. All except one of our children had made a complete clinical recovery at final follow-up. However, all six children in the > 24-month age group showed radiological evidence of degenerative changes which might cause problems in the future.


The Journal of Bone & Joint Surgery British Volume
Vol. 93-B, Issue 8 | Pages 1134 - 1139
1 Aug 2011
Schindeler A Birke O Yu NYC Morse A Ruys A Baldock PA Little DG

Congenital pseudarthrosis of the tibia is an uncommon manifestation of neurofibromatosis type 1 (NF1), but one that remains difficult to treat due to anabolic deficiency and catabolic excess. Bone grafting and more recently recombinant human bone morphogenetic proteins (rhBMPs) have been identified as pro-anabolic stimuli with the potential to improve the outcome after surgery. As an additional pharmaceutical intervention, we describe the combined use of rhBMP-2 and the bisphosphonate zoledronic acid in a mouse model of NF1-deficient fracture repair.

Fractures were generated in the distal tibiae of neurofibromatosis type 1-deficient (Nf1+/−) mice and control mice. Fractures were open and featured periosteal stripping. All mice received 10 μg rhBMP-2 delivered in a carboxymethylcellulose carrier around the fracture as an anabolic stimulus. Bisphosphonate-treated mice also received five doses of 0.02 mg/kg zoledronic acid given by intraperitoneal injection.

When only rhBMP but no zoledronic acid was used to promote repair, 75% of fractures in Nf1+/− mice remained ununited at three weeks compared with 7% of controls (p < 0.001). Systemic post-operative administration of zoledronic acid halved the rate of ununited fractures to 37.5% (p < 0.07).

These data support the concept that preventing bone loss in combination with anabolic stimulation may improve the outcome following surgical treatment for children with congenital pseudarthoris of the tibia and NF1.


The Journal of Bone & Joint Surgery British Volume
Vol. 93-B, Issue 4 | Pages 542 - 547
1 Apr 2011
Bonnard C Fournier J Babusiaux D Planchenault M Bergerault F de Courtivron B

This study evaluated the results of a physeal-sparing technique of intra-articular anterior cruciate ligament (ACL) reconstruction in skeletally immature patients, with particular reference to growth disturbance. Between 1992 and 2007, 57 children with a mean age of 12.2 years (6.8 to 14.5) underwent ACL reconstruction using the same technique. At a mean of 5.5 years (2 to 14) after surgery, 56 patients underwent clinical and radiological evaluation. At that time, 49 patients (87.5%) had reached bony maturity and 53 (95%) achieved A or B according to the IKDC 2000 classification. Four patients had stopped participation in sports because of knee symptoms, and three patients (5.4%) had a subsequent recurrent ACL injury. There was no clinical or radiological evidence of growth disturbance after a mean growth in stature of 20.0 cm (3 to 38).

This study demonstrates that ACL reconstruction sparing the physes in children is a safe technique protecting against meniscal tears and giving better results than reconstruction in adults, without causing significant growth disturbance.