Treatment for hip displacement in children and youth with cerebral palsy (CP) is dependent upon when the problem is detected. Hip surveillance aims to identify hip displacement early through systematic screening and, together with timely orthopaedic intervention, can eliminate the need for salvage hip procedures. Here we report the impact on surgical practice of 1) hip surveillance program advocacy and knowledge translation efforts and 2) initial population-based program implementation. A retrospective review was completed of all children with CP undergoing surgery for hip displacement at a provincial tertiary pediatric hospital in the years 2004 to 2018. Date and type of surgery, age at surgery, Gross Motor Function Classification System (GMFCS) level, and pre-operative migration percentages (MP) were collected. Surgeries were categorized as soft-tissue release, reconstructive, and salvage procedures. Results were collected for three time periods: historical (September 2004- June 2010), hip surveillance advocacy and knowledge translation (July 2010- August 2015), and post provincial hip surveillance program implementation (September 2015 – December 2018). A total of 261 surgeries on 321 hips were reviewed. The rate of salvage procedures dropped from 29% (24) of surgeries performed in the historical period to 12% (14) during the period that included targeted hip surveillance knowledge translation and development of provincial guidelines. Since implementation of the surveillance program, salvage procedures have accounted for 7% (4) of interventions performed; all of these were in patients new to the province or existing patients. During the three study periods, reconstructive surgeries accounted for 64%, 81%, and 80% of the interventions performed. The mean initial MP at time of reconstructive surgery has decreased from 66% (SD 20%) in the historical time period to 57% (SD 25%) and 57% (SD 22%) in the subsequent time periods. There were a greater number of children at GMFCS levels III and IV in the two more recent time periods suggesting surveillance may assist in identifying children at these levels of impairment. The rate of soft-tissue surgeries was low during all time periods with the number trending higher since program implementation (7%, 7%, 10%). Knowledge translation and use of standardized surveillance guidelines can have a significant impact on the prevalence of salvage hip surgeries. Centers should utilize existing surveillance guidelines and educate key stakeholders about the importance of hip surveillance in the absence of a formal hip surveillance program

The patient's subjective experience of disease is an increasing focus in health care delivery. Health-related quality of life (HRQoL) is defined as a “functional effect of a medical condition and its consequent treatment”; it is both self-reported and multi-dimensional. While functional outcome is well researched among the soft tissue sarcoma (STS) population, few studies have focused on HRQoL, which gives a broader understanding of the psychological, somatic, social and physical toll of cancer and its treatment from the patient's viewpoint. The biologic and anatomic heterogeneity of sarcomas are considerable, just as the treatments are diverse, we surmise that the indicators of patient HRQoL differ and are not captured in existing generic HRQoL tools for cancer. The study objectives were to explore the domains of HRQoL and functioning in adult patients diagnosed with extremity STS from the patient's perspective from active care through survivorship through qualitative inquiry, so as to form the basis for the development of a patient-derived, sarcoma-specific, preference based HRQoL tool. Study design is a sequential exploratory mixed methods study of patient experience in localized or metastatic adult extremity STS (2007 and 2017). The study was conducted at a high-volume sarcoma centre. Qualitative descriptive design was grounded in an integrated knowledge translation approach and aimed at identifying HRQoL domains through in-person and electronic focus groups, and individual semi-structured interviews in both English and French (N=28). The interview guide topics were selected based on existing knowledge about PROs and HRQoL life, including (a) impact of diagnosis on employment or acquisition of academic/vocational skills; (b) physical and psychological functioning; (c) symptom burden; (d) treatment preferences; (e) knowledge of and use of existing resources; (f) impact on family time and resources; and (g) overall experience. Data was analyzed using inductive thematic networks approach using the qualitative software N-Vivo 12. Codes were generated by 2 independent qualitative experts capturing key concepts of HRQoL that is impacted by STS. Basic themes were clustered into organizing themes, and merged into global domains. Attention was paid to deviant cases and within-group dynamics during focus group discussion analysis. Discrepancies or inconsistencies in coding were resolved in consensus meetings. Final sample size was determined when data saturation was reached and no new themes emerged. Qualitative reduction of identified items to reach a consensus framework was facilitated by a moderator during multi-disciplinary panel meetings comprised of sarcoma experts, patient partners, allied health staff and other stakeholders. Twenty-nine patients with biopsy-proven localized or metastatic STS of the extremity participated (69% lower extremity STS; mean age 56 years, 25% with local recurrence, 21% metastatic, 18% amputation). Inductive thematic network analysis revealed five function-related domains HRQoL for patients with STS. The functional domains were mapped to the Wilson & Cleary Model and experience domains were mapped to the Picker Institute's Through Patient's Eyes model. This is a critical step toward developing disease specific outcome measures. Patient-centered research is crucial to understanding the impact of surgery, adjuvant therapy and the associated complications for patients with extremity STS, and thereby improving the quality of care provision. This study offers a unique perspective on what domains and sub domains are most impactful on HRQoL and provides the basis for our on-going development of a disease-specific, preference-based HRQoL measure

Introduction. Adult tendon injuries occur very frequently, but injured tendon heals very slowly and the mechanisms of the slow-healing response to injury are still largely unknown. Currently, the main barrier is our insufficient understanding of the mechanisms responsible for homeostasis, regeneration and repair of adult tendon. This gap in knowledge translates to a lack of experimental models. Therefore, using the combination of state-of-the-art genetic approaches, we have established novel cell biological tools to advance the understanding of tendon biology. Materials and Methods. Adult mouse tendon progenitor lines and Adult mouse tenocyte lines: Primary adult tenocytes were isolated from Achilles tendon in Scleraxis(fl/fl)/Scleraxis-GFP/p21(−/−) mice, then CD90.2- and subsequent Sca1-positive cells were sorted by Flow Cytometry. Then Scleraxis-null progenitor lines were generated by the treatment of those cells with adenovirus-Cre. Adult Scleraxis(+/+) and Scleraxis-null tenocyte lines were also generated from Scleraxis(fl/fl)/Scleraxis-GFP/p21(−/−) mice. To establish Scleraxis-Flag overexpressing tenocyte lines, Scleraxis and Flag-tag fusion-protein expression construct was generated and transfected into Scleraxis-null tenocytes (Scleraxis transgenic mouse strains were provided by Dr Ronen Schweitzer). Scleraxis antibody: DNA coding mouse Scleraxis residues were obtained by PCR, then the recombinant protein was expressed, immunized in rabbits, and an affinity-purified antibody was generated. Results. Established parental progenitor lines highly expressed Sca1 (98.9%), CD90.2 (97.3%), and CD44 (99.8%) and were almost negative for ScxGFP (2.3%). Interestingly, Scleraxis-null progenitors showed significantly increased clonogenicity. Furthermore, when stimulated toward mesenchymal lineages, Scleraxis-null progenitors enhanced differentiation into chondrocytes. Our Scleraxis antibody reacted with lysates from cells expressing Scleraxis-Flag fusion proteins (∼30 kDa), whereas it did not react with Scleraxis-null cells by Western analysis. Immunofluorescence analysis of adult mouse Achilles tendons further confirmed intense Scleraxis protein expression in wild-type tenocytes, whereas considerably decreased expression of Scleraxis was evident in Cre-treated Scleraxis(fl/fl) tenocytes. Discussion. These novel tools will be the promising resources to get an insight into molecular framework for Scleraxis in adult tendons. It is anticipated that the establishment of experimental models using these resources will fill major gaps in the current knowledge of adult tendon biology and will facilitate development of novel strategies to treat adult tendon injury

Construction of a functional skeleton is accomplished through co-ordination of the developmental processes of chondrogenesis, osteogenesis, and synovial joint formation. Infants whose movement in utero is reduced or restricted and who subsequently suffer from joint dysplasia (including joint contractures) and thin hypo-mineralised bones, demonstrate that embryonic movement is crucial for appropriate skeletogenesis. This has been confirmed in mouse, chick, and zebrafish animal models, where reduced or eliminated movement consistently yields similar malformations and which provide the possibility of experimentation to uncover the precise disturbances and the mechanisms by which movement impacts molecular regulation. Molecular genetic studies have shown the important roles played by cell communication signalling pathways, namely Wnt, Hedgehog, and transforming growth factor-beta/bone morphogenetic protein. These pathways regulate cell behaviours such as proliferation and differentiation to control maturation of the skeletal elements, and are affected when movement is altered. Cell contacts to the extra-cellular matrix as well as the cytoskeleton offer a means of mechanotransduction which could integrate mechanical cues with genetic regulation. Indeed, expression of cytoskeletal genes has been shown to be affected by immobilisation. In addition to furthering our understanding of a fundamental aspect of cell control and differentiation during development, research in this area is applicable to the engineering of stable skeletal tissues from stem cells, which relies on an understanding of developmental mechanisms including genetic and physical criteria. A deeper understanding of how movement affects skeletogenesis therefore has broader implications for regenerative therapeutics for injury or disease, as well as for optimisation of physical therapy regimes for individuals affected by skeletal abnormalities.

Cite this article: Bone Joint Res 2015;4:105–116