L.W.A. van Suijlekom-Smit

Long-term follow-up on effectiveness and safety of etanercept in juvenile idiopathic arthritis: the Dutch national register

Objective: We undertook an observational study to obtain a complete overview of the long-term effectiveness and safety of etanercept in patients with different juvenile idiopathic arthritis (JIA) subtypes. Methods: At baseline we collected patient and disease characteristics of all Dutch patients with JIA who started treatment with etanercept. Disease activity was evaluated (at start of the study, after 3 months and then yearly) according to the JIA core set of the American College of Rheumatology paediatric definition for 30, 50 and 70% improvement (ACR Pedi 30, 50 and 70). Use of etanercept and concomitant drugs was monitored. Adverse events were recorded. Results: We included 146 patients with JIA with a median follow-up of 2.5 years per patient (range 0.3–7.3). JIA subtypes represented: 27% systemic, 8% polyarticular rheumatoid factor positive, 38% polyarticular rheumatoid factor negative, 19% oligoarticular extended, 3% enthesitis-related and 5% psoriatica. Most patients (77%) met the criteria of the ACR Pedi 30 in the first 3 months of treatment. For the majority of patients this improvement was sustained; 53 (36%) of all patients met the remission criteria. No other second-line agents were needed in 43 patients. Although patients with systemic JIA responded initially less to etanercept therapy than patients from other subtypes, those who did respond showed equal effectiveness in the long term. Serious adverse events rate was low (0.029 per patient year). Conclusions: Etanercept is effective and safe in JIA, even for a large proportion of the patients with systemic JIA. The greatest improvement occurred in the first 3 months of treatment, and was sustained for a long time in most patients (up to 75 months).

Facioskeletal changes in children with juvenile idiopathic arthritis

Objective: To investigate the facioskeletal morphology in patients with juvenile idiopathic arthritis (JIA) with and without temporomandibular joint (TMJ) involvement. Methods: Eighty five patients were included. TMJ involvement was defined by orthopantomogram alterations. Lateral cephalograms were used to determine linear and angular measurements and occlusion. Results: Patients regardless of their TMJ status had a 67% chance for retrognathia and a 52% chance for posterior rotation of the mandible and, respectively, 82% and 58% if TMJ involvement were present. Changes were not uniformly distributed among the different subtypes. Conclusion: Patients with JIA have an altered facial morphology, especially in the presence of TMJ involvement.

Intra-articular steroids in pauciarticular juvenile chronic arthritis, type 1.

The use of intra-articular steroids in one or both knees was evaluated in 21 children with type 1 pauciarticular juvenile chronic arthritis (JCA). The beneficial effect of the injection was noted within 3 days with no significant adverse reactions. Remission exceeding 6 months was seen in 70% of the knees and the arthritis remained inactive during the follow up period in 37%. The beneficial effect of the injection did not correlate with sex, age of onset or the presence of antinuclear antibodies or HLA-B27 antigen and there was no relationship with the size of involved joints at onset, the ESR at onset, or the presence of uveitis. Intra-articular corticosteroids in this type of JCA may provide prompt relief of swelling and pain and reduce the need for other forms of therapy. Remission was long lasting in the majority of the children.

Major improvements in health-related quality of life during the use of etanercept in patients with previously refractory juvenile idiopathic arthritis

Objective: To evaluate changes in health-related quality of life (HRQoL) in patients with refractory juvenile idiopathic arthritis (JIA) who are being treated with etanercept. Methods: 53 patients with JIA from seven Dutch centres were included. HRQoL was measured by the Childhood Health Assessment Questionnaire (CHAQ), Child Health Questionnaire (CHQ) and Health Utilities Index mark 3 (HUI3) at the start and after 3, 15 and 27 months of treatment. At the same time points the following JIA disease activity variables were collected; physician’s global assessment through the visual analogue scale (VAS), number of active and limited joints and erythrocyte sedimentation rate. A statistical method linear mixed models was used to assess outcomes over time. Results: During etanercept treatment both disease-specific and generic HRQoL outcomes improved dramatically. Significant improvements were shown after 3 months and these improvements continued at least up to 27 months of treatment. The disease-specific CHAQ, including VAS pain and wellbeing, showed a significant improvement in all domains. The generic health-profile measure CHQ improved for all the health concepts except for “family cohesion”, which was normal. The generic preference-based HUI3 showed impairment and, subsequently, significant improvement in the more specific domains (“pain”, “ambulatory”, “dexterity”). In accordance disease activity variables also improved significantly over time. Conclusion: This study shows that the HRQoL of patients with refractory JIA can be substantially improved by the use of etanercept for all aspects impaired by JIA. Information on HRQoL is crucial to understand the complete impact of etanercept treatment on patients with JIA and their families.

Reliability and validity of health status measurement by the TAPQOL.

Background: In addition to clinical measures in the evaluation of paediatric interventions, health related quality of life (HRQoL) is an important outcome. The TAPQOL (TNO-AZL Preschool children Quality of Life) was developed to measure HRQoL in preschool children. It is a generic instrument consisting of 12 scales that cover the domains physical, social, cognitive, and emotional functioning. Aims: To evaluate the feasibility, score distribution, internal consistency, test-retest reliability, and discriminative and concurrent validity of the TAPQOL multi-item scales in preschool children, aged 2–48 months. Also to evaluate the feasibility, reliability, and validity separately for infants (2–12 months old) and toddlers (12–48 months old). Methods: Parents of a random general population sample of 500 preschool children were sent a questionnaire by mail. A random subgroup of 159 parents who participated received a retest after two weeks. Results: The response rate was 83% at the test and 75% at the retest. There were few missing answers. Six scales showed ceiling effects. Nine scales had Cronbach’s alphas >0.70. In general, score distributions and Cronbach’s alphas were comparable for infants and toddlers. Test-retest showed no significant differences in mean scale scores; two scales had intra-class correlations <0.50. Five scales showed significant differences between children with no conditions versus children with two or more parent reported chronic conditions. Conclusion: Results showed that the TAPQOL is a feasible instrument to measure HRQoL and support the reliability and discriminative validity of the majority of its scales for infants as well as toddlers.

Influenza vaccination in asthmatic children: effects on quality of life and symptoms

This study aimed to detect the effect of influenza vaccination on quality of life, symptomatology and spirometry in asthmatic children. A randomised double-blind placebo-controlled trial in 696 (296 in 1999–2000 and 400 in 2000–2001) asthmatic children aged 6–18 yrs, which were vaccinated with either vaccine or placebo, was performed. Children participated for only one influenza season. They recorded symptoms in a diary and reported when symptom scores reached a predefined severity level. If this occurred research nurses visited them twice, first to take a pharyngeal swab and spirometry, and a week later to assess quality of life over the past illness week. Compared with placebo, vaccination improved health-related quality of life in the weeks of illness related to influenza-positive swabs. However, no effect was found for respiratory symptoms recorded in the diaries during those weeks. Similarly, no differences were found for quality of life in all weeks of illness or for respiratory symptoms throughout the seasons. Influenza vaccination was found to have a moderately beneficial effect on quality of life in influenza-positive weeks of illness in children with asthma.

Bone mineral density and body composition and influencing factors in children with rheumatic diseases treated with corticosteroids.

In rheumatic diseases the use of corticosteroids (CS), immobility, or the disease itself, may cause osteoporosis and growth retardation. We evaluated bone mineral density (BMD) by dual energy X-ray absorptiometry (DXA), growth and physical activity in 27 children with rheumatic disease, all treated with high dose CS for at least one year, in a cross-sectional design. BMD SDS was significantly lower than zero: -1.02 for total body and -1.49 for lumbar spine measurement. SDS for fat mass was higher than zero. In multiple regression analysis the Child Health Assessment Questionnaire score significantly correlated with BMD of the lumbar spine. There was no significant correlation with cumulative dose or duration of CS treatment. Height SDS decreased during treatment to -1.57 (p <0.001 compared to 0). In conclusion, BMD and body composition in children with rheumatic disease treated with CS are influenced by physical activity, as well as corticosteroid treatment and type of rheumatic disease.

Incidence of temporomandibular involvement in juvenile idiopathic arthritis

Objective: Temporomandibular joint (TMJ) involvement is a frequent feature in cross‐sectional prevalence studies among juvenile idiopathic arthritis (JIA) patients. The cross‐sectional design makes it almost impossible to study the incidence. Follow‐up data on TMJ involvement are sparse. In this study patients were reviewed with an interval of a minimum of 1 year and a maximum of 2 years to study the yearly incidence of TMJ involvement and to obtain follow‐up data on TMJ involvement and orthopantomogram (OPT) alterations. Methods: Children with JIA from a previous study on TMJ involvement were included. OPTs were scored according to Rohlins grading system (grade 0–5). A paediatric rheumatologist measured the level of disease activity during the interval. Results: Eighty‐nine of the 97 patients were included in this study with a mean follow‐up of 14 months. The yearly incidence of TMJ involvement was 7.1% in patients with JIA. Improvement on the OPT was seen in 27 patients (66%), and 19 of these patients no longer showed any signs of TMJ involvement. Worsening on the OPT was seen in four patients (10%). Disease activity was significantly lower in the improved patients than in the patients with worsening. Conclusion: Condylar lesions due to arthritis can improve over time, indicating a regenerative capacity of the mandibular condyle. As condylar improvement seems to be associated with low disease activity, it is important to consider the TMJ when deciding on a therapeutic regimen.

Impetigo: incidence and treatment in Dutch general practice in 1987 and 2001—results from two national surveys

Background  Impetigo is a common skin infection in children. The epidemiology is relatively unknown, and the choice of treatment is subject to debate.

Measuring Chronic Pain in Children, an Exploration

The study focused on the feasibility and validity of pain instruments and the optimal period of diary registration for measuring chronic pain intensity of 13 children. Highly positive associations were found between the registration of pain on a Visual Analogue Scale and on the Postoperative Pain Measure for Parents. For children under medical treatment for chronic limb pain a one-week diary registration suffices.