Assessment of Orofacial Symptoms in Juvenile Idiopathic Arthritis: Validation of a Consensus-Based Short Patient Questionnaire.

OBJECTIVE: To develop, validate, and test the performance of patient-reported outcomes (PROs) in a short patient questionnaire (< 5 minutes in length) in order to assess the multidimensional aspects of orofacial symptoms related to juvenile idiopathic arthritis (JIA) among patients aged ≥ 10 years. METHODS: The study was conducted by an interdisciplinary task force from the Temporomandibular Joint Juvenile Arthritis Working Group. The project consisted of a multiphased approach including the following: (1) conceptual phase with online international survey of 167 healthcare workers, (2) item generation and drafting of preliminary questionnaire(s) (ie, face validity), (3) cognitive script interview and probing (ie, content validity; 16 patients with JIA), (4) assessment of construct validity (ie, convergence and divergence; 53 patients with JIA), (5) test of reliability, and (6) test of clinical performance and psychometric characteristics (95 patients with JIA). RESULTS: In total, 7 PROs were included in the final patient questionnaire: (1) pain frequency, (2) pain intensity, (3) pain location, (4) jaw function, (5) specific questions related to symptoms and dysfunction, (6) changes in face and jaw pain since last visit, and (7) changes in jaw function since last visit. In total, 80% of the patients were able to complete the questionnaire in less than 5 minutes. CONCLUSION: We have developed and validated a short patient questionnaire to assess the multidimensional aspects of JIA-related orofacial symptoms. The PROs included in our questionnaire show acceptable validity and reliability. The questionnaire is applicable to routine monitoring of subjects with JIA, as well as future research studies.

Standardizing Terminology and Assessment for Orofacial Conditions in Juvenile Idiopathic Arthritis: International, Multidisciplinary Consensus-based Recommendations.

OBJECTIVE: To propose multidisciplinary, consensus-based, standardization of operational terminology and method of assessment for temporomandibular joint (TMJ) involvement in juvenile idiopathic arthritis (JIA). METHODS: Using a sequential expert group-defined terminology and methods-of-assessment approach by (1) establishment of task force, (2) item generation, (3) working group consensus, (4) external expert content validity testing, and (5) multidisciplinary group of experts final Delphi survey consensus. RESULTS: Seven standardized operational terms were defined: TMJ arthritis, TMJ involvement, TMJ arthritis management, dentofacial deformity, TMJ deformity, TMJ symptoms, and TMJ dysfunction. CONCLUSION: Definition of 7 operational standardized terms provides an optimal platform for communication across healthcare providers involved in JIA-TMJ arthritis management.

A Comparison of Radiographic Joint Space Width Measurements Versus Ultrasonographic Assessment of Cartilage Thickness in Children with Juvenile Idiopathic Arthritis.

OBJECTIVE: Joint space narrowing (JSN) is a measurable outcome of tissue degeneration in arthritis. JSN is usually assessed by conventional radiography. Ultrasonographic (US) measurement of joint cartilage thickness has been validated in healthy children, and US measurement of the distal femoral cartilage has been validated in a group of patients with juvenile idiopathic arthritis (JIA). Our aim was to compare the measures of cartilage thickness of the proximal cartilage site in the second metacarpophalangeal (MCP), second proximal interphalangeal (PIP), and knee joints as assessed by US to joint space width (JSW) as measured by computerized radiography in children with JIA. METHODS: The study included 74 children with JIA aged 5-15 years (median 11.3 yrs). MCP and PIP joints were assessed at one midline spot. Knee joints were assessed at the medial and lateral femoral condylar areas. Only the proximal cartilage site in the joints was assessed by US, whereas the complete JSW was assessed by radiography. RESULTS: We assessed 136 second MCP, 138 second PIP, and 146 knee joints. We found a high level of agreement between US and radiographic measures of cartilage thickness and JSW: r = 0.82-0.86 (second MCP), r = 0.50-0.55 (second PIP), and r = 0.52-0.81 (knee); p < 0.001 for all 8 assessed sites. CONCLUSION: US measurements of cartilage thickness of the proximal site of the second MCP, second PIP, and knee joints correlated well with radiographic JSW measurements in the finger and knee joints of children with JIA. However, US does not measure the distal cartilage, which may limit its use in the assessment of JSN.

The Danish version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR).

The Juvenile Arthritis Multidimensional Assessment Report (JAMAR) is a new parent/patient-reported outcome measure that enables a thorough assessment of the disease status in children with juvenile idiopathic arthritis (JIA). We report the results of the cross-cultural adaptation and validation of the parent and patient versions of the JAMAR in the Danish language. The reading comprehension of the questionnaire was tested in ten JIA parents and patients. Each participating centre was asked to collect demographic, clinical data and the JAMAR in 100 consecutive JIA patients or all consecutive patients seen in a 6-month period and to administer the JAMAR to 100 healthy children and their parents. The statistical validation phase explored descriptive statistics and the psychometric issues of the JAMAR: the three Likert assumptions, floor/ceiling effects, internal consistency, Cronbach's alpha, interscale correlations, test-retest reliability and construct validity (convergent and discriminant validity). A total of 303 JIA patients (7.9% systemic, 35% oligoarticular, 22.1% RF negative polyarthritis, 35% other categories) and 99 healthy children, were enrolled in three centres. The JAMAR components discriminated well healthy subjects from JIA patients. All JAMAR components revealed good psychometric performances. In conclusion, the Danish version of the JAMAR is a valid tool for the assessment of children with JIA and is suitable for use both in routine clinical practice and clinical research.

Assessment of dentofacial growth deviation in juvenile idiopathic arthritis: Reliability and validity of three-dimensional morphometric measures.

INTRODUCTION: Patients with juvenile idiopathic arthritis (JIA) and involvement of the temporomandibular joint (TMJ) often experience abnormal facial growth. Three-dimensional (3D) assessment of dentofacial growth deviation has become more common with advancement and commercialization of imaging technologies. However, no standardized guidelines exist for interpretation of 3D imaging in patients with JIA. The aim of this study was to propose and validate morphometric measures for the 3D radiographic assessment of dentofacial growth deviation in patients with JIA to enhance: 1) Description of dentofacial growth deviation; 2) Treatment planning; 3) Longitudinal follow-up. METHODS: The study was conducted in a standardized sequential-phased approach involving: 1) Preliminary decision-making; 2) Item generation; 3) Test of content-validity; 4) Test of reliability; 5) Test of construct validity; 6) Establishment of final recommendations. RESULTS: Twenty-one morphometric measures were evaluated. Based on results of reliability and validity-testing including subjects with JIA (n = 70) and non-JIA controls (n = 19), seven measures received a "high recommendation" score. Those measures were associated with posterior mandibular height, occlusal cant, mandibular asymmetry, mandibular inclination, and anterior/posterior lower face height. Nine other measures were "moderately recommended" and five received a "somewhat recommendation" score. CONCLUSION: Seven morphometric measures were considered very useful in the 3D assessment of growth deviation in patients with TMJ disease associated with JIA. These variables can be used to standardize the description of dentofacial deformities and to plan corrective interventions.

Models of Care for addressing chronic musculoskeletal pain and health in children and adolescents.

Chronic musculoskeletal pain among children and adolescents is common and can negatively affect quality of life. It also represents a high burden on the health system. Effective models of care for addressing the prevention and management of pediatric musculoskeletal pain are imperative. This chapter will address the following key questions: (1) Why are pediatric-specific models of pain care needed? (2) What is the burden of chronic musculoskeletal pain among children and adolescents? (3) What are the best practice approaches for early identification and prevention of chronic musculoskeletal pain in children and adolescents? (4) What are the recommended strategies for clinical management of chronic pain, including pharmacological, physical, psychological and complementary, and alternative approaches? (5) What are the most effective strategies for implementing models of pain care across different care settings? (6) What are the research priorities to improve models of care for children and adolescents with chronic musculoskeletal pain?

Cartilage thickness of the knee joint in juvenile idiopathic arthritis: comparative assessment by ultrasonography and magnetic resonance imaging.

OBJECTIVE: The functional disability experienced in juvenile idiopathic arthritis (JIA) is primarily caused by joint effusion, synovial membrane hypertrophy, and periarticular soft tissue edema, leading to the degeneration of the osteocartilaginous structures because of the inflammatory process in the synovium. The ability to visualize the inflammatory changes and hence the ensuing osteocartilaginous degeneration is, therefore, of great importance in pediatric rheumatology. Ultrasonography (US) has been validated as a tool for measuring cartilage thickness in healthy children and, previously, we have found good agreement with the measures obtained by magnetic resonance imaging (MRI). Our aim is to validate and compare US with MRI measurements of distal femoral cartilage thickness in the knee joint at the medial condyle, lateral condyle, and intercondylar spots in children with JIA, and to locate the best spot for imaging comparisons. METHODS: One knee from each of 23 children with oligoarticular JIA were investigated by both MRI and US. Outcome measures of imaging procedures were distal femoral cartilage thickness. RESULTS: We found a high level of agreement between MRI and US measurements of mean cartilage thickness, and Rho values between modalities were high (between 0.70 and 0.86, p < 0.05 for all). We found a thinner cartilage thickness at the medial condyle in comparison to the other investigated points. Evaluation of anatomical landmarks for optimal measurement of cartilage thickness was found to be the intercondylar spot, which was easier to locate in addition to a smaller variance around the mean for that anatomical measuring point. CONCLUSION: US measurements of distal femoral cartilage thickness are highly correlated to MRI measurements. The intercondylar notch of the distal femoral cartilage may be the best anatomical point for cartilage thickness measurements of the knee. US is a reliant and nonexpensive, non-invasive modality for visualization of childhood femoral cartilage.

Assessment of disease activity in juvenile idiopathic arthritis. The number and the size of joints matter.

OBJECTIVE: Variables for assessment of disease activity of juvenile idiopathic arthritis (JIA) were studied, in order to develop a disease activity score for children with JIA. METHODS: One randomly chosen hospital visit was studied for each of 312 patients with JIA, with regard to disease activity variables. The physician global assessment score visual analog scale (physician GA) was used as a dependent variable in comparisons between potential disease activity variables. Previous studies have shown this variable to be the most sensitive to changes in JIA disease activity and to be comparable between patients. RESULTS: Based on Spearman's rank order correlation the number of active joints had a strong association with the physician GA. The median physician GA score rose markedly for each active large joint, but less for small joints, although small joints were also statistically important in assessing disease activity. Among the laboratory data, the erythrocyte sedimentation rate, C-reactive protein level, and platelet count showed weak correlations to the physician GA. CONCLUSION: In preparation of a disease activity score for children with JIA the importance of both the number and size of joints involved needs further evaluation.