Outcome Of Disease In Children Research Articles

20161631, Phillippi - Race and income affects outcomes in juvenile dermatomyositis

The relationship of race and income, the latter as a surrogate for socioeconomic status, on disease outcomes has been widely researched in many diseases and chronic conditions. For example black (and Hispanic) race is associated with a worse outcome in pediatric systemic lupus erythematosus. In this volume of The Journal, Phillippi et al investigated the relationship of these factors to the functional outcome and development of calcinosis as a measurement of morbidity (and perhaps disease control) in juvenile dermatomyositis, a rare disease with an annual incidence of 1-4 per million children. Calcinosis, if severe, can result in substantial disability. In a cross sectional study based on 438 children reported by North American pediatric rheumatologists to the Childhood Arthritis and Rheumatology Research Alliance Legacy Registry, they found that both black race and low income (defined as <$50 000 per year) independently negatively affected patient function and was positively associated with the development of calcinosis. It is important to state that despite these statistically significant disparities, the functional outcome of the patients, including those from black race and low income, was generally good. The cause of these disparities could not be ascertained from this study and can include genetic/pharmacogenetic and environmental effects, as well as patient-related factors such as adherence, type of insurance, or other unknown factors. Disparities did not appear to be related to access to expert medical care or differences in treatments prescribed. This study also demonstrates the importance of using well designed registries to investigate rare diseases in “real life.” Registries, used for many years, especially in oncology, are now used more frequently in childhood rheumatic diseases. Newly developed registries that include prospectively collected data and biologic specimens will improve the quality of these studies and enable us to answer some of the questions left open in this study. Article page 38 ▶ Race, Income, and Disease Outcomes in Juvenile DermatomyositisThe Journal of PediatricsVol. 184PreviewTo determine the relationships among race, income, and disease outcomes in children with juvenile dermatomyositis (JDM). Full-Text PDF

Race, Income, and Disease Outcomes in Juvenile Dermatomyositis

To determine the relationships among race, income, and disease outcomes in children with juvenile dermatomyositis (JDM). Data from 438 subjects with JDM enrolled in the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Legacy Registry were analyzed. Demographic data included age, sex, race, annual family income, and insurance status. Clinical outcomes included muscle strength, presence of rash, calcinosis, weakness, physical function, and quality of life measures. Disease outcomes were compared based on race and income. Minority subjects were significantly more likely to have low annual family income and significantly worse scores on measures of physical function, disease activity, and quality of life measures. Subjects with lower annual family income had worse scores on measures of physical function, disease activity, and quality of life scores, as well as weakness. Black subjects were more likely to have calcinosis. Despite these differences in outcome measures, there were no significant differences among the racial groups in time to diagnosis or duration of disease. Using calcinosis as a marker of disease morbidity, black race, annual family income <$50 000 per year, negative antinuclear antibody, and delay in diagnosis >12 months were associated with calcinosis. Minority race and lower family income are associated with worse morbidity and outcomes in subjects with JDM. Calcinosis was more common in black subjects. Further studies are needed to examine these associations in more detail, to support efforts to address health disparities in subjects with JDM and improve disease outcomes.

Tissue and peripheral eosinophilia as predictors for disease outcome in children with ulcerative colitis

BackgroundEosinophils are implicated in the pathogenesis of ulcerative colitis. AimsTo evaluate the magnitude of mucosal and blood eosinophils in newly diagnosed pediatric ulcerative colitis patients and its significance in predicting disease outcomes. MethodsWe retrospectively evaluated colorectal biopsies of 96 pediatric patients with ulcerative colitis and 50 age- and sex-matched controls. Samples were taken from diseased areas of the colon and examined by a gastrointestinal pathologist. The most inflamed site was used for assessment of mucosal eosinophils. ResultsSamples from 96 diagnostic and 70 follow-up colonoscopies were evaluated. Median age was 13.3 years (IQR 10.1–15.3). Median duration of follow-up was 12.8 years (IQR 7.2–17.1). Median number of tissue eosinophils at diagnosis was 45 (IQR 22–73) compared to 10 eosinophils (IQR 8–25) during histologic remission (p<0.0001). Peripheral absolute eosinophil counts correlated with tissue inflammation and eosinophilia (p=0.001). Mucosal eosinophilic infiltration (p=0.02) and peripheral eosinophilia (p=0.04) was associated with clinical severity at diagnosis. Multivariate analysis showed that severe eosinophilic infiltration is associated with corticosteroid therapy following diagnosis (p=0.04) but not with long-term risk for step-up therapy or colectomy. ConclusionTissue and peripheral eosinophilia correlate with ulcerative colitis severity at diagnosis and with short-term corticosteroid requirement but not with long-term outcomes.

Effects of a Cognitive Behavioral Therapy Intervention Trial to Improve Disease Outcomes in Children with Inflammatory Bowel Disease.

Studies testing the efficacy of behavioral interventions to modify psychosocial sequelae of inflammatory bowel disease in children are limited. This report presents outcomes through a 6-month follow-up from a large randomized controlled trial testing the efficacy of a cognitive behavioral intervention for children with inflammatory bowel disease and their parents. One hundred eighty-five children aged 8 to 17 years with a diagnosis of Crohn's disease or ulcerative colitis and their parents were randomized to one of two 3-session conditions: (1) a social learning and cognitive behavioral therapy condition or (2) an education support condition designed to control for time and attention. There was a significant overall treatment effect for school absences due to Crohn's disease or ulcerative colitis (P < 0.05) at 6 months after treatment. There was also a significant overall effect after treatment for child-reported quality of life (P < 0.05), parent-reported increases in adaptive child coping (P < 0.001), and reductions in parents' maladaptive responses to children's symptoms (P < 0.05). Finally, exploratory analyses indicated that for children with a higher level of flares (2 or more) prebaseline, those in social learning and cognitive behavioral therapy condition experienced a greater reduction in flares after treatment. This trial suggests that a brief cognitive behavioral intervention for children with inflammatory bowel disease and their parents can result in improved child functioning and quality of life, and for some children may decrease disease activity.

Effects of a Cognitive Behavioral Therapy Intervention Trial to Improve Disease Outcomes in Children with Inflammatory Bowel Disease:

Effects of a Cognitive Behavioral Therapy Intervention Trial to Improve Disease Outcomes in Children with Inflammatory Bowel Disease:

Clinical profile and outcome of kawasaki disease in children in himalayan region of North India

Background: Kawasaki disease (KD) is an important cause of acquired heart disease with significant morbidity if not diagnosed and managed appropriately. It is usually under-diagnosed due to lack of knowledge regarding this entity among pediatricians and dermatologists. In this retrospective, tertiary care teaching institute based study clinical profile and outcome of KD in children from Himalayan region of north India was appraised. Methods: Clinical details of all the cases diagnosed as KD over 3 years from January 2011 to December 2013 were collected from inpatient files and outpatient follow-up clinical records. Results: Twelve children were diagnosed to have KD out of which seven had classical KD and five had incomplete KD. Mean age at presentation was 2.45 years and male to female ratio was 1.4:1. All children were treated with intravenous immunoglobulin and aspirin. Two out of 12 children had residual coronary artery disease at 6 weeks, 3 and 6 months of follow-up echocardiography. Conclusions: KD remains an important differential diagnosis for fever, rash, and lymphadenopathy in children <5 years.

The Clinical Features and Long-term Sequelae of Invasive Meningococcal Disease in Children

We evaluated the outcome of invasive meningococcal disease in children <15 years of age (n = 181). Neisseria meningitidis serogroup B comprised 78% of bacterial isolates. Case fatality rate was 11.6%. In follow-up interviews (115/160 survivors, 72%), at least 1 long-term sequela was reported in 38/115 children (33%), including learning-academic difficulties (22.6%), hearing impairment (7%), neurologic (12.2%), behavioral (14.8%) and motor (10.4%) deficits.

Association of a Genetic Variant of Carnitine Palmitoyltransferase 1A with Infections in Alaska Native Children

To evaluate whether the arctic variant (c.1436C→T) of carnitine palmitoyltransferase type 1A (CPT1A) is associated with a higher incidence of adverse health outcomes in Alaska Native infants and children. We evaluated health measures from birth certificates (n = 605) and Alaska Medicaid billing claims (n = 427) collected from birth to 2.5 years of age for a cohort of Alaska Native infants with known CPT1A genotype. To account for geographic variations in gene distribution and other variables, data also were evaluated in cohorts. When analysis was restricted to residents of nonhub communities in Western and Northern Alaska, children homozygous for the arctic variant experienced more episodes of lower respiratory tract infection than did heterozygous or noncarrier children (5.5 vs 3.7, P = .067) and were more likely to have had otitis media (86% vs 69%, 95% CI 1.4-8.9). Associations were weaker for more homogeneous cohorts. The association of the arctic variant of CPT1A with infectious disease outcomes in children between birth and 2.5 years of age suggests that this variant may play a role in the historically high incidence of these health outcomes among indigenous Arctic populations; further studies will need to assess if this association was confounded by other risk factors.

Mo1974 Effect of Biologics on the Long Term Outcomes of Inflammatory Bowel Disease in Children: A 25-Year Follow-up Study

Mo1974 Effect of Biologics on the Long Term Outcomes of Inflammatory Bowel Disease in Children: A 25-Year Follow-up Study

Allogeneic hematopoietic cell transplantation outcomes for children and young adults with treatment-related myelodysplastic syndrome or acute

Objective: Therapy-related myeloid malignancies (tMDS or tAML) are well-known complications of cancer therapy for which outcomes are poor. Whether clinical characteristics can predict disease outcome in children and young adults, as they do in older adults, is not well known. Method: Twenty-five children with tMDS/tAML underwent allogeneic hematopoietic stem cell transplantation (allo-HCT) at the University of Minnesota between 1990 and 2010. Twenty-one patients received myeloablative conditioning. Graft sources included umbilical cord blood in fourteen patients [single (n=9), double (n=5)] and bone marrow in 11. Prior to transplant twenty-one patients received AML-directed chemotherapy and 19 were in complete remission at the time of allo-HCT. Twelve patients had high-risk cytogenetic abnormalities involving 11q23 (n=6), chromosome 5 (n=4) and/or chromosome 7 (n=5). Result: The 5-year OS and DFS was 23% (CI 95%: 9-41%) and 17% (CI 95%: 5-35%) respectively. Transplant related mortality (TRM) and relapse at 1-year were 39% (CI 95%: 19-59%) and 22% (CI 95%: 5-38%). We found no significant differences in survival based on remission status, pre-transplant cytogenetics or conditioning regimen. Conclusion: Outcomes of children and young adults with tMDS/tAML continue to be poor with TRM and relapse remaining the major obstacles to cure.

Factors associated with disease outcome in children at Kenyatta National Hospital (KNH)

Globally, 10.5 million children die annually. Approximately 6 million of Kenya’s population are under five years with a child mortality rate of 74 per 1000 live births. Children attending Kenyatta National Hospital (KNH) have preventable and treatable illnesses. The aim of the study was to assess care taker factors associated with disease outcome among children at KNH. A cross sectional study was conducted; questionnaires were administered to the caretakers of the 156 children recruited in the study. Data entry was done in Microsoft Access and analyzed using statistical package for Social Science (SPSS). There was a significant association between diseases outcome and length of exclusive breast-feeding (P < 0.025), occupation of both fathers and mothers (P< 0.026 and P <0.001, respectively) in the priority disease outcomes. There was a statistically significant association between duration of illness and that of seeking treatment before visiting KNH, (P < 0.001). Children who stayed more than 2 months before seeking health care at KNH had a 3-fold risk of becoming priority cases. Using formal education as the reference category for mother occupation, the odds of priority outcome for unemployed compared to formal education was 10.4 and informal education compared to formal education was 7.6. Caretakers delayed in seeking health care, prior to admission. Results from this study suggest that occupation of parents was a major determinant in regard to disease outcome. Communities need to be empowered to obtain and access basic health care services. More studies need to be done to identify specific caretaker factors associated with childhood illnesses. This will enable to evaluate further the possible interventions in the health care delivery among children. A longitudinal study is crucial to follow up children post admission addressing specific illnesses. Occupation of parents, occupation of parents and length of exclusive breast-feeding was a major determinant in regard to disease category. Key words: Disease outcome, cross sectional, priority, emergency category.

Nailfold capillary density is importantly associated over time with muscle and skin disease activity in juvenile dermatomyositis

To investigate the longitudinal association of nailfold capillary density (NCD; as a potential marker of activity) with various other clinical measures of disease activity and to evaluate baseline NCD as a predictor of disease outcome in children with JDM. Data from 809 clinic visits from 92 JDM patients were prospectively collected at each clinic visit over a time period of 5.5 years. The number of capillaries per millimetre at the distal nailfold was scored using a stereomicroscope. Disease activity was determined using the Childhood Myositis Assessment Scale (CMAS) and a modification of the validated disease activity score (DAS), which included three skin (SDAS) and three muscle (MDAS) criteria. An inception cohort subgroup (n=28) with a baseline visit at diagnosis was analysed separately. Both DAS subscores, MDAS (β = -0.04437, P < 0.0001) and SDAS (β = -0.1589, P < 0.0001), as well as the CMAS (β = 0.02165, P < 0.0001) were significantly associated with loss of end row nailfold capillary over time (multiple regression mixed-model analysis). All patients in the inception subcohort showed a reduced baseline NCD (diagnostic sensitivity = 100%) that improved as the disease improved, but this did not predict longer term outcome or course of disease. NCD is a marker of skin and muscle disease activity, and is an important measure of disease activity changes from visit to visit. Determination of capillary density may be useful when making treatment decisions. A decrease in NCD may be considered for inclusion in the diagnostic criteria due to its high sensitivity.

Thrombomodulin and von Willebrand Factor: Relation to Endothelial Dysfunction and Disease Outcome in Children with Acute Lymphoblastic Leukemia

The severe endothelial dysfunction in children with acute lymphoblastic leukemia (ALL) can result from the disease itself, from treatment, or from other conditions (e.g. sepsis). The aim of this study was to determine the levels of markers of endothelial activation in children with ALL and to assess their potential prognostic value. Fifty-two children with ALL, 19 children with ALL 1–10 years after the completion of therapy, and 28 healthy children were studied. In children with ALL, there was a significant increase in thrombomodulin (TM) and von Willebrand factor (vWF) levels during the acute phase of the disease and during treatment. Children with an unfavorable outcome had higher levels of TM. In conclusion, severe endothelial dysfunction is present during the acute phase of ALL and during treatment and appears to result from the disease itself. Serum TM and vWF levels might represent additional, but not independent, prognostic markers in childhood ALL.

Synovial fluid proteins differentiate between the subtypes of juvenile idiopathic arthritis

Juvenile idiopathic arthritis (JIA) is a heterogeneous group of inflammatory diseases, and no clinically useful prognostic markers to predict disease outcome in children with JIA are currently available. Synovial fluid likely reflects the proteins present in the inflamed synovium. The purpose of this study was to delineate the synovial fluid proteome and determine whether protein expression differs in the different subtypes of JIA. Synovial fluid samples obtained from children with oligoarticular JIA, polyarticular JIA, or systemic JIA were compared. Two-dimensional gel electrophoresis for protein separation and matrix-assisted laser desorption ionization-time-of-flight mass spectrometry and quadripole time-of-flight mass spectrometry for protein identification were used for this study. Synovial fluid cells were analyzed by polymerase chain reaction (PCR) for the presence of haptoglobin messenger RNA (mRNA). The synovial fluid proteome of the samples was delineated. The majority of proteins showed overexpression in JIA synovial fluid as compared with noninflammatory control samples. There were 24 statistically significantly differentially expressed spots (>2-fold change; P < 0.05) between the subtypes of JIA. PCR analysis revealed haptoglobin mRNA, suggesting that haptoglobin is locally produced in an inflamed joint in JIA. Despite the similar histologic appearance of inflamed joints in patients with different subtypes of JIA, there are differences in protein expression according to the subtype of JIA. Haptoglobin is differentially expressed between the subtypes of JIA and is locally produced in an inflamed joint in JIA. Haptoglobin and other differentially expressed proteins may be potential biomarkers in JIA.

A toll‐like receptor 4 variant is associated with fatal outcome in children with invasive meningococcal disease

Toll-like receptor 4 (TLR4) is the major endotoxin signalling receptor of the innate immune system and is required for efficient recognition of bacterial infections. Here, we analysed a possible association between the TLR4 variant Asp299Gly and disease outcome in children with invasive meningococcal disease. In total, 197 children with invasive meningococcal disease were analysed for the TLR4 Asp299Gly variant. Genotyping results were correlated with mortality, the frequency of ventilation support, application of inotropic substances, skin grafting, and limb loss. The overall Asp299Gly allele frequency was 9.4%. Detection of a heterozygous Asp299Gly TLR4 mutation was significantly associated with fatal outcome (non-survivor group: 31.6% vs. survivor group: 12.1%; p = 0.021) and was even more pronounced in patients with disease onset less than 24 months of age (non-survivor group: 42.8% vs. survivor group: 10.2%; p = 0.006). In this age group, ventilation support was also more frequent in patients with the Asp299Gly genotype (37.5% vs. 6.2%). Our data suggest that the heterozygous TLR4 Asp299Gly genotype is associated with an increased mortality in children with invasive meningococcal disease.

Pityriasis lichenoides: the differences between children and adults

Pityriasis lichenoides (PL) is a skin disease that affects both children and adults. Anecdotally, it is said to run a more benign course in children, with a frequent tendency to self-resolution. However, to our knowledge, there have been no published studies comparing PL in both age groups. To evaluate the clinicopathological features, overall efficacy of treatments and disease outcomes in children and adults diagnosed with PL. A retrospective review of records was undertaken on all patients diagnosed with PL at two regional centres during an 8-year period (from 1998 to 2006). For each individual, data were collected on age, sex, number of lesions, lesional morphology and distribution, symptoms, histopathological features, treatment modalities (and response), overall follow-up and length of remission. We recorded 25 children (median age 8 years, range 2-18) and 32 adults (median age 40 years, range 20-65) with PL. All the children and adults had more than 20 scaly, papular lesions. Children had greater lesional body involvement than adults. Lesions on the legs and trunk were present in 23 children (92%) compared with 19 adults (59%) (P < 0.01) and facial involvement was observed more commonly in children (n = 10, 40%) compared with adults (n = 8, 25%). Dyspigmentation was significantly more common in children (n = 18, 72%) compared with adults (n = 6, 19%) (P < 0.001). Topical corticosteroids were used by 16 children (64%) and 18 adults (56%) but only half in each group found these effective. Eight children (32%) were treated with erythromycin, with only two (25%) clearing, and one of these subsequently relapsing. In contrast, four adults (13%) received antibiotics, with three (75%) clearing and none relapsing thereafter. Ultraviolet B phototherapy was used in eight children (32%), with seven (88%) completely or almost clearing, but four (57%) subsequently relapsed. Fourteen adults (44%) received phototherapy; 10 (71%) completely cleared and only two of these (20%) subsequently relapsed. Strikingly, after a median disease duration of 30 months, only five children (20%) went into complete remission compared with 25 adults (78%) (P < 0.001). This is the first study to compare PL in children and adults. Our findings suggest that, compared with adults, PL in children is more likely to run an unremitting course, with greater lesional distribution, more dyspigmentation and a poorer response to conventional treatment modalities.

Off‐label and unlicensed prescribing for children: have we made any progress?

Off‐label and unlicensed prescribing for children: have we made any progress?

Hemorrhagic Complications in Pediatric Hematologic Malignancies

Hematologic malignancies account for almost 40% of all cancers in children. Hemorrhage is the most common cause of early death in children with leukemia. Furthermore, major bleeding episodes lead to shorter survival and increased resource use. Potential risk factors for bleeding include hyperleukocytosis, immunophenotype of leukemia (especially acute promyelocytic leukemia), thrombocytopenia, and associated infections. Successful management of a bleeding episode is dependent on prompt identification of a child at high risk for bleeding, and should be directed at the replacement of blood products as well as to the aggressive therapy for underlying risk factors. Although in recent years there is a favorable decline in the hemorrhage-related mortality, the overall prevalence and the extent of morbidity posed by this potentially fatal complication, including disease outcome in children with hematologic malignancy, is largely unknown. In addition, there are no evidence-based guidelines for prophylactic blood or blood product transfusions. Prospective studies are required to define the epidemiology and risk factors predisposing children with hematologic malignancy to bleeding and to develop management guidelines.

Differences in disease outcomes between medicaid and privately insured children: Possible health disparities in juvenile rheumatoid arthritis

To determine the relationship between health insurance status and disease outcome in children with juvenile rheumatoid arthritis (JRA). JRA patients followed at a tertiary pediatric rheumatology center were assessed for the number of active joints and number of joints with limited range of motion. Disease activity, patient well-being, and pain were measured. Disability was assessed by the Childhood Health Assessment Questionnaire, health-related quality of life by the Pediatric Quality of Life Inventory (PedsQL) Generic Core Scale, and the PedsQL Rheumatology Module. Health care resource utilization was estimated based on the number of billing events for health services coded in administrative databases; these databases also provided information on patient health insurance status. Children insured by Medicaid or similar state programs for low-income families were considered to have Medicaid status. Disease outcomes of children with Medicaid status was compared with that of children with private health insurance. Forty (14%) of the 295 children with JRA had Medicaid status. Patients with Medicaid status were more often of nonwhite race (P < or = 0.04) and more frequently had a polyarticular or systemic disease course (P = 0.04) compared with other patients (n = 255). After correction for differences in disease duration, race, JRA onset, and JRA course between groups, children with Medicaid status continued to have significantly higher disability (P < 0.0003), and lower mean PedsQL Generic Core Scale scores (P < 0.05), while health resource utilization appeared similar between groups. Despite apparently similar health resource utilization and joint involvement, Medicaid status is associated with significantly lower health-related quality of life and higher disability in JRA.

Is hydrotherapy cost-effective? A randomised controlled trial of combined hydrotherapy programmes compared with physiotherapy land techniques in children with juvenile idiopathic arthritis

To compare the effects of combined hydrotherapy and land-based physiotherapy (combined) with land-based physiotherapy only (land) on cost, health-related quality of life (HRQoL) and outcome of disease in children with juvenile idiopathic arthritis (JIA). Also to determine the cost-effectiveness of combined hydrotherapy and land-based physiotherapy in JIA. A multicentre randomised controlled, partially blinded trial was designed with 100 patients in a control arm receiving land-based physiotherapy only (land group) and 100 patients in an intervention arm receiving a combination of hydrotherapy and land-based physiotherapy (combined group). Three tertiary centres in the UK. Patients aged 4-19 years diagnosed more than 3 months with idiopathic arthritides, onset before their 16th birthday, stable on medication with at least one active joint. Patients in the combined and land groups received 16 1-hour treatment sessions over 2 weeks followed by local physiotherapy attendances for 2 months. Disease improvement defined as a decrease of > or =30% in any three of six core set variables without there being a 30% increase in more than one of the remaining three variables was used as the primary outcome measure and assessed at 2 months following completion of intervention. Health services resource use (in- and outpatient care, GP visits, drugs, interventions, and investigations) and productivity costs (parents' time away from paid work) were collected at 6 months follow-up. HRQoL was measured at baseline and 2 and 6 months following intervention using the EQ-5D, and quality-adjusted life-years (QALYs) were calculated. Secondary outcome measures at 2 and 6 months included cardiovascular fitness, pain, isometric muscle strength and patient satisfaction. Seventy-eight patients were recruited into the trial and received treatment. Two months after intervention 47% patients in the combined group and 61% patients in the land group had improved disease with 11 and 5% with worsened disease, respectively. The analysis showed no significant differences in mean costs and QALYs between the two groups. The combined group had slightly lower mean costs (-6.91 pounds Sterling) and lower mean QALYs (-0.0478, 95% confidence interval -0.11294 to 0.0163 based on 1000 bootstrap replications). All secondary measures demonstrated a mean improvement in both groups, with the combined group showing greater improvements in physical aspects of HRQoL and cardiovascular fitness. JIA is a disease in which a cure is not available. This research demonstrates a beneficial effect from both combined hydrotherapy and land-based physiotherapy treatment and land-based physiotherapy treatment alone in JIA without any exacerbation of disease, indicating that treatments are safe. The caveat to the results of the cost-effectiveness and clinical efficacy analysis is that the restricted sample size could have prevented a true difference being detected between the groups. Nevertheless, there appears to be no evidence to justify the costs of building pools or initiating new services specifically for use in this disease. However, this conclusion may not apply to patients with unremitting active disease who could not be entered into the trial because of specified exclusion criteria. For this group, hydrotherapy or combined treatment may still be the only physiotherapy option. Further research is suggested into: the investigation and development of appropriate and sensitive outcome measures for use in future hydrotherapy and physiotherapy trials of JIA; preliminary studies of methodologies in complex interventions such as physiotherapy and hydrotherapy to improve recruitment and ensure protocol is acceptable to patients and carers; hydrotherapy in the most common paediatric user group, children with neurological dysfunction, ensuring appropriate outcome measures are available and methodologies previously tried; patient satisfaction and compliance in land-based physiotherapy and hydrotherapy and European studies of hydrotherapy in rare disorders such as JIA.