PubMed Health⌕ Search

Biomedical subjects

N T Ilowite

Publications and source records attributed to N T Ilowite.

At least 19 recordsLinked to original sources

Etanercept in children with polyarticular juvenile rheumatoid arthritis. Pediatric Rheumatology Collaborative Study Group.

BACKGROUND: We evaluated the safety and efficacy of etanercept, a soluble tumor necrosis factor receptor (p75):Fc fusion protein, in children with polyarticular juvenile rheumatoid arthritis who did not tolerate or had an inadequate response to methotrexate. METHODS: Patients 4 to 17 years old received 0.4 mg of etanercept per kilogram of body weight subcutaneously twice weekly for up to three months in the initial, open-label part of a multicenter trial. Those who responded to treatment then entered a double-blind study and were randomly assigned to receive either placebo or etanercept for four months or until a flare of the disease occurred. A response was defined as an improvement of 30 percent or more in at least three of six indicators of disease activity, with no more than one indicator worsening by more than 30 percent. RESULTS: At the end of the open-label study, 51 of the 69 patients (74 percent) had had responses to etanercept treatment. In the double-blind study, 21 of the 26 patients who received placebo (81 percent) withdrew because of disease flare, as compared with 7 of the 25 patients who received etanercept (28 percent) (P=0.003). The median time to disease flare with placebo was 28 days, as compared with more than 116 days with etanercept (P<0.001). In the double-blind study, there were no significant differences between the two treatment groups in the frequency of adverse events. CONCLUSIONS: Treatment with etanercept leads to significant improvement in patients with active polyarticular juvenile rheumatoid arthritis. Etanercept is well tolerated by pediatric patients.

Activities of Daily Living↗

Premature atherosclerosis in systemic lupus erythematosus.

Premature atherosclerosis is a recognized complication of systemic lupus erythematosus (SLE). Since life expectancy in SLE is improving, premature atherosclerosis is emerging as an ever more important clinical issue. Atherosclerosis begins in the pediatric age group, and interventions directed at prevention should begin in childhood as well. Possible etiologies include dyslipoproteinemia (DL) from the underlying chronic inflammatory disease or from corticosteroid therapy, hypercoagulation due to antiphospholipid antibodies or nephrotic syndrome, vasculitis, and hypertension. A relationship between DL and presence of anticardiolipin antibodies (aCL) has been reported. Dietary therapy is helpful, but many patients continue to have significant DL after both dietary modification and fish oil supplementation. Lipid lowering drugs may be indicated in this subgroup. Potential mechanisms of the DL are discussed. Other strategies to prevent atherosclerosis and its complications are reviewed.

Age of Onset↗

Family-level coping in juvenile rheumatoid arthritis: assessing the utility of a quantitative family interview.

OBJECTIVE: To explore the viability of a quantitative family interview to describe family-level coping strategies used to deal with juvenile rheumatoid arthritis (JRA)-related stressors for early and late adolescents. METHOD: A structured interview protocol with 30 adolescents with JRA and family members assessed ways JRA disrupts or changes family functioning. Emotional reactions, sequential phases of family response, and treatment adherence were discussed. Interviews were coded for family-level coping. To assess adjustment, family members completed the Youth Self Report and the Family Environment Scale. The pediatric rheumatologist provided medical information. RESULTS: The family interview produced both quantitative and qualitative data. Families reported multiple JRA-related stressors (mean 6.6). For many adolescents, treatment adherence was problematic. Families used all 3 types of coping strategies (appraisal-, problem-, and emotion-focused) to varying degrees. Problem-focused approaches were most commonly used and included seeking support (used by 73% of families), self-reliance (70%), and family coordination (70%) for dealing with specific problems, and seeking information about JRA (67%). Emotion-focused approaches, such as impulsive outbursts and diminished awareness of others' feelings, were associated with problematic adjustment. Few differences were found between the families of early and late adolescents. CONCLUSION: The quantitative family interview has the potential to be a useful tool in documenting JRA-related stressors, family-level coping processes, and how family-level coping is associated with treatment adherence and psychosocial adjustment.

Adaptation, Psychological↗

Discontinuation of methotrexate treatment in juvenile rheumatoid arthritis.

OBJECTIVE: Children with juvenile rheumatoid arthritis (JRA) treated with methotrexate (MTX) were examined for their course after the discontinuation of the drug to define the relapse and remission rates and to identify predictors of relapse. METHODOLOGY: A retrospective chart review of all patients with JRA was conducted in two pediatric rheumatology centers. A total of 101 patients being treated with MTX were identified. Dose, response to the drug, and length of time until reaching a state of complete control were noted. The outcome of patients with a complete response in whom the drug was discontinued was examined with regards to length of time to relapse or continued remission. RESULTS: In 25 patients, MTX was discontinued after reaching complete control of the disease. There were no statistically significant predictors of response to MTX identified. Of 25 whose MTX was discontinued, relapse occurred in 13 (52%) after a mean of 11 months after discontinuation. There was no significant difference among patients who relapsed or those who remained in remission as to sex, subtype of JRA, number of months to complete control, or number of months in complete control until discontinuing MTX. Patients younger than 41/2 years at diagnosis were found to be more likely to relapse than patients diagnosed at a later age. In 10 of the patients who relapsed, complete control was induced within a mean of 7 months after restarting MTX. CONCLUSION: The optimal time for discontinuing MTX in children with JRA who have achieved complete control is unknown. Relapse occurred in approximately half of the patients in whom MTX was discontinued. Because response to reinstitution of the drug is good, it is reasonable to discontinue MTX after prolonged complete control. It remains to be seen whether the relapse rate can be improved by waiting for longer periods of time in complete control before its discontinuation.

Adolescent↗

Methotrexate treatment of Wegener granulomatosis in children.

Wegener granulomatosis traditionally has been treated with glucocorticoids and cyclophosphamide. Both the disease and its treatments are associated with significant morbidity and mortality rates. There has been an effort to find effective but less toxic alternative treatments. We describe three children with Wegener granulomatosis who responded well to treatment with glucocorticoids and methotrexate, similar to a regimen used in adults.

Adolescent↗

Hyperlipidemia and the rheumatic diseases.

Premature atherosclerosis is a recognized complication of systemic lupus erythematosus. Atherosclerosis begins in the pediatric age group, and interventions directed toward prevention should begin in childhood as well. A possible cause of premature atherosclerosis is dyslipoproteinemia from the underlying chronic inflammatory disease or from corticosteroid therapy. A relationship between dyslipoproteinemia and anticardiolipin antibodies has been demonstrated. Dietary therapy is helpful, but many patients continue to have significant dyslipoproteinemia after both dietary modification and fish oil supplementation. Lipid-lowering drugs may be indicated in this subgroup. Potential mechanisms of dyslipoproteinemia are discussed.

Animals↗

Immunoglobulin M immunoblot for diagnosis of Borrelia burgdorferi infection in patients with acute facial palsy.

We used immunoblotting to improve the specificity of the serologic diagnosis of Lyme borreliosis in cases of acute facial palsy. Twelve of 15 patients (80%) with suspected Lyme borreliosis, versus 0 of 10 controls, were positive by immunoglobulin M immunoblotting of acute-phase sera and 3 were negative, including 2 with borderline enzyme immunoassay results. Immunoglobulin M immunoblotting is a useful test to confirm Borrelia burgdorferi infection in patients with acute facial palsy and a positive enzyme immunoassay result.

Acute Disease↗

Lack of autoantibody expression in children born to mothers with silicone breast implants.

OBJECTIVE: We determined systematically the prevalence of autoantibodies in children born to mothers with silicone breast implants and the relationships with clinical symptoms and methods of exposure. METHODS: Autoantibody expression was determined in 80 children born to mothers with silicone implants and in 42 controls. A clinical assessment score was assigned to each patient. Antinuclear antibodies as well as antibodies to mitochondrial, smooth muscle, striational, myocardial, parietal cell, reticulin tissues, or subcellular compartments were measured by indirect fluorescent assay. Antibodies to nRNP (U1-RNP/snRNP); Sm; SS-A; SS-B; Scl-70; thyroid microsome; immunoglobulin (Ig)G, IgM, and IgA antibodies to cardiolipin; and antibodies to native and denatured human types I and II collagen were measured by enzyme-linked immunosorbent assay. Serum complement components C3 and C4 and IgM rheumatoid factor were measured by nephelometry. RESULTS: Autoantibody prevalence was not significantly different between children born to mothers with silicone implants and controls. The presence of autoantibodies was not related to the children's clinical symptoms or to the method of exposure. CONCLUSIONS: Determination of autoantibody production is of limited clinical utility in the evaluation of children born to mothers with silicone breast implants.

Adolescent↗

Increased urinary NO3(-) + NO2- and neopterin excretion in children breast fed by mothers with silicone breast implants: evidence for macrophage activation.

OBJECTIVE: To determine whether children breast fed by mothers with silicone implants (BFSI) have increased urinary excretion of nitric oxide (NO) metabolites and neopterin, whether these are associated with esophageal dysmotility, and whether in vitro incubation of macrophages with silicone increases NO synthesis. METHODS: In a case-control study based on laboratory investigation, 38 BFSI children (17 male, 21 female, mean age 7.1 +/- 3.6 years, range 0.5-16.5) were compared with 30 controls (14 male, 16 female, mean age 8.4 +/- 3.5 years, range 2.5-17). Urinary NO was quantitated using the Griess reaction. Urinary neopterin was determined by radioimmunoassay. Murine macrophages were cultured with or without silicone and NO production assayed. RESULTS: Urinary NO and neopterin were significantly increased in BFSI children compared with controls. There was a significant inverse relationship between urinary neopterin excretion and the severity of esophageal dysfunction. In vitro nitrite production was nearly 60% higher in macrophages grown on silicone compared to other growth conditions. CONCLUSION: BFSI children have evidence of macrophage activation and this is associated with esophageal dysmotility. In vitro data support the proposal that silicone exposure causes macrophage activation.

Adolescent↗

Muscle, reticuloendothelial, and late skin manifestations of Lyme disease.

In addition to classic organ system involvement, Lyme disease may be characterized by myositis, liver and spleen involvement, and atypical cutaneous manifestations. Myositis is characteristically localized near an involved joint or localized neuropathy. Nuclear imaging with gallium-67 may be useful for detection. Myositis responds to treatment with intravenous or oral antibiotics. Patients with erythema migrans have been observed to have liver function test abnormalities in the absence of symptomatic hepatitis. Splenomegaly has been noted infrequently in patients with Lyme disease. Chronic cutaneous manifestations of Lyme disease--including erythema migrans, acrodermatitis chronica atrophicans, and lymphadenosis benigna cutis--have been observed more frequently in Europe than in the United States. It appears that they are caused primarily by the Borrelia afzelii genomic group of Borrelia burgdorferi, which has been found exclusively in Europe.

Europe↗

Effects of dietary modification and fish oil supplementation on dyslipoproteinemia in pediatric systemic lupus erythematosus.

OBJECTIVE: To determine if a program of dietary modification and fish oil supplementation is effective in treating the dyslipoproteinemia in pediatric systemic lupus erythematosus (SLE). METHODS: Prospective clinical trial where each patient serves as his/her own control. Twenty-four consecutive adolescents fulfilling SLE classification criteria were screened with fasting lipid profiles. Patients were identified as having dyslipoproteinemia of active disease or of corticosteroid therapy. Patients were treated for 6 weeks with dietary modification and if dyslipoproteinemia did not normalize with another 6 weeks of dietary modification and fish oil supplementation. RESULTS: Seventeen patients (71%) had dyslipoproteinemia; 10 of active disease, 4 of steroid therapy; 3 with a combined pattern. Eleven patients underwent dietary modification. There was a significant decrease in serum triglyceride concentrations (p < 0.05). Total cholesterol, low density lipoprotein cholesterol, and high density lipoprotein cholesterol did not change significantly. A further significant decline in serum triglycerides was achieved with fish oil supplementation (p < 0.05). Five of the 11 patients who underwent treatment continued to have dyslipoproteinemia. CONCLUSION: Dyslipoproteinemia is common in pediatric SLE. Dietary modification and fish oil supplementation appear to be effective in improving serum lipid profiles, and blinded studies are warranted. a significant number of patients may require pharmacologic therapy for persistent dyslipoproteinemia to prevent complications of premature atherosclerosis.

Adolescent↗

Interpretation of immunoblot in pediatric Lyme arthritis.

OBJECTIVE: To determine the pattern of bands present on an immunoblot (Western blot) in children with Lyme arthritis. METHODS: Sera from 20 children with Lyme arthritis from an endemic area were assayed, and the results compared with those obtained in 162 control sera from the same area. The study was retrospective, serum bank based. RESULTS: Eighty-seven percent of control sera revealed no bands on immunoblot, and all had < or = 4 bands. All Lyme arthritis sera revealed > or = 5 bands (mean 8.4, range 5-13 bands). Bands of molecular weight 25, 28, 39, 47, 50, and 93 kDa were seen in patients and not in controls. There was no correlation between duration of arthritis or activity of arthritis at presentation and number of bands present. CONCLUSION: Criteria were derived for a positive immunoblot based on the number and molecular weights of bands seen in our laboratory. These are the presence of 5 or more bands, of which at least one is an apparently specific band (25, 28, 39, 47, 50, or 93 kDa). Results of immunoblot are useful to confirm clinically suspected Lyme arthritis.

Adolescent↗

Sclerodermalike esophageal disease in children breast-fed by mothers with silicone breast implants.

OBJECTIVE: To determine whether breast-fed children of mothers with silicone implants are at increased risk for the development of sclerodermalike esophageal involvement compared with children not exposed to silicone implants. DESIGN: Case-series [corrected]. SETTING: Referral-based pediatric gastroenterology clinic. PATIENTS: Eleven children (mean age, 6.0 years; range, 1.5 to 13 years; six boys and five girls) referred for abdominal pain who were born to mothers who had silicone breast implants (eight breast-fed children and three bottle-fed) were compared with 17 patients (mean age, 10.7 years; range, 2 to 18 years; 11 boys and six girls) with abdominal pain who were not exposed to silicone implants. METHODS: All children underwent esophageal manometry and upper intestinal endoscopy with esophageal biopsy and were tested for antinuclear antibody and autoantibodies to Scl-70, centromere, ribonucleoprotein, Sm, Ro, La, and phospholipid. RESULTS: Six of the eight breast-fed children from mothers with silicone implants had significantly abnormal esophageal motility with nearly absent peristalsis in the distal two thirds of the esophagus and decreased lower sphincter pressure. Upper esophageal pressures and motility were normal. Compared with controls, the breast-fed children had significantly decreased lower sphincter pressure and abnormal esophageal wave propagation. These manometric abnormalities were not seen in the three bottle-fed children. There was no difference in the expression of autoantibodies in the breast-fed children compared with the bottle-fed children or controls. CONCLUSIONS: A relationship appears to exist between breast-feeding by mothers with silicone implants and abnormal esophageal motility. Studies evaluating larger numbers of children are needed to determine the extent of the risk.

Abdominal Pain↗

The rheumatoid factor cross-reactive idiotype in juvenile rheumatoid arthritis: role of the CD5-positive B cell.

The major rheumatoid factor cross-reactive idiotype (RCRI), which was defined by prototypic monoclonal IgM rheumatoid factors (RF) from Waldenstrom's macroglobulinemia patient Wa, is present on 60% of all monoclonal IgM RF paraproteins. The RCRI is expressed in high frequency by pokeweed mitogen-derived plasma cells (PWM-PCs) and in high concentration in the sera from adults with rheumatoid arthritis (RA) who express RF in their sera. Unlike adults with RA, most children with juvenile rheumatoid arthritis (JRA) are seronegative for RF as detected by classic IgG binding assays. In the experiments summarized herein, we demonstrated that approximately 2/3 of JRA patients who are seronegative for RF, express the RCRI in high concentration in their sera and in high frequency among their PWM-PCs. Expression of this idiotype could not be attributed to expression of hidden IgM RF, or IgA RF, and may be expressed on a parallel set of immunoglobulin molecules, related to RFs, but lacking the ability to bind to IgG. There is an increased number of circulating CD5+ B cells in patients with JRA but there was no significant relationship between CD5+ B cell numbers and serum RCRI concentration, suggesting that in this disease, RCRI bearing immunoglobulins may also be produced by non-CD5+ B cells or by a small subset of CD5+ B cells.

Adolescent↗

Panniculitis and fever in children.

We describe three children with panniculitis and associated systemic manifestations including fever. Histopathologic features, such as the presence of lobular or septal inflammation, presence of vasculitis, character of the cellular infiltrate, and presence of erythrophagocytosis, were useful in classifying this group of panniculitides. In one patient with subcutaneous polyarteritis nodosa, corticosteroid therapy was effective; in two other patients with histiocytic cytophagic panniculitis, there were poor responses to steroids, intravenously administered immune globulin, dapsone, or antimalarial drugs; cyclosporine was very effective and appeared to be the drug of choice.

Child↗