Background Human milk feeding protects against oxidative stress-induced damage in preterm neonates, including severe multifactorial diseases such as retinopathy of prematurity (ROP), necrotizing enterocolitis (NEC), and bronchopulmonary dysplasia (BPD). The carotenoids, which are not found in formula milk, might play a key role in these actions. Methods A multicenter, double-blind, randomized controlled trial was conducted in three tertiary Italian neonatal intensive care units. All preterm infants <32 +6 weeks' gestational age were eligible and were randomized to a single, oral, daily 0.5-mL dose of carotenoid supplementation (0.14 mg lutein + 0.0006 mg zeaxanthin) or placebo (5% glucose solution) from birth till 36 weeks' corrected gestational age. Primary outcomes were threshold ROP, NEC > second stage, and BPD. Surveillance for detection of these diseases and for intolerance/adverse effects was performed.Results No treatment-related adverse effect was documented in the 229 analyzed infants, whose clinical/demographical characteristics were similar in the two groups. Threshold ROP incidence did not significantly differ in treated (6.2%) versus not treated infants (10.3%; p ¼ 0.18). The same occurred for NEC (1.7% versus 5.1%; p ¼ 0.15) and BPD (4.5% versus 10.3%; p ¼ 0.07). Noteworthy, the progression rate from early ROP stages to threshold ROP was decreased by 50% (0.30 versus 0.44; p ¼ 0.23). Conclusion Lutein/zeaxanthin supplementation in preterm infants is well tolerated. No significant effect was seen on threshold ROP, NEC, or BPD. The decreasing trends of these outcomes in the treatment group need to be assessed and confirmed on larger sample-sizes.
We report a case of posterior non-arteritic ischaemic optic neuropathy (NAION) causing bilateral visual loss in a 7-month-old female infant, after a therapeutic course with sildenafil, a phosphodiesterase type 5 inhibitors (PDE5i). The patient was affected by a complex cyanotic congenital heart defect and had undergone cavopulmonary anastomosis (Glenn operation) 3 months ago. After 2 months of recurring chylothorax, a course of oral sildenafil was administered, with the hypothesis that pulmonary vascular resistances were increased. Approximately 4 weeks later the acute onset of visual worsening and poor pupillary light reflex prompted the diagnosis of posterior NAION. Despite a rapid cessation of PDE5i and systemic treatment with corticosteroids, no visual recovery was noticed at 2-year follow-up. NAION has been associated with PDE5i therapy in adults, but to the best of our knowledge it is almost unheard of in children. We suggest close monitoring of visual function in children undergoing treatment with sildenafil.
Background:Juvenile idiopathic arthritis (JIA) is the most common paediatric rheumatic disease. Its most threatening complication is represented by uveitis, which could cause severe visual impairment if not diagnosed and treated promptly. It is an asymptomatic uveitis and the diagnosis is only instrumental. Therefore, regular ophthalmologic surveillance is crucial in the management of JIA. To date there are no specific predictive markers of uveitis development among JIA patients, including serologic subsets. Anti-Nuclear Antibodies (ANA) positive patients have the highest risk of iridocyclitis, but ANA are not specific. They could be found in patients with JIA without uveitis, in many other rheumatologic and inflammatory conditions and also in healthy subjects (6-12% of children). Anti-DFS70 antibodies (ANA forming a specific pattern in immunofluorescence) have been taken into consideration, but their role has not yet been established in a pediatric setting. Currently, there are few reports, involving small groups of patients and with non-univocal results. Some studies report anti-DFS70 antibodies more frequently in children with rheumatological diseases, in particular JIA-related uveitis; on the contrary, others describe them in healthy ANA positive patients.Objectives:1) To evaluate the correlation between anti-DFS70 autoantibodies and the risk of developing uveitis in a cohort of patients with JIA ANA + in pediatric age;2) to compare the prevalence of anti-DFS70 in patients with JIA, with a group of healthy ANA + children, to better define the role of these autoantibodies (potential risk factor or a protective factor for the development of AARDs in children?)Methods:We evaluated retrospectively 51 patients with JIA ANA +. We divided these patients in two groups, according to the presence (n=11) or the absence (n=40) of uveitis. For each patient we evaluated: gender, current age, age at diagnosis, type of JIA, therapy, presence of other diseases, dosage of ANA (with IF-Hep2), research of anti-ENA and anti-DFS70 antibodies (with chemiluminescence). Subsequently the whole group of patients with JIA was compared with a control group of healthy subjects aged ≤ 18 years (n=30), followed in the pediatric rheumatology clinic for occasional finding of ANA positivity, without pathologies at the moment of the study (in particular without rheumatological or autoimmune diseases).Results:Among patients with JIA without uveitis, anti-DFS70 autoantibodies were positive only in one patient. Anti-DFS70 were negative in all patients with JIA and uveitis. The difference between the two groups is not significant (p=1). In the group of healthy patients 6/30 (20%) presented a positivity of anti-DFS70 autoantibodies, in the absence of anti-ENA.Conclusion:Our data revealed no correlation between the positivity of anti-DFS70 autoantibodies and the risk of uveitis in patients with JIA ANA +, even if the number of patients is small. Further studies are needed to identify a reliable predictive marker of uveitis risk in JIA patients. The finding of a significant greater prevalence of anti-DFS70 autoantibodies in healthy ANA + subjects allows to suppose that this autoantibody could represent a possible protective marker for development of AARDs in asymptomatic children with isolated ANA positivity, as for adults. To confirm this hypothesis, it would be useful to carry on the study prospectively, encompassing children with other rheumatological diseases, and prolonging the clinical and laboratory follow up.References:[1]Clarke S, Sen ES, Ramanan AV. Juvenile idiopathic arthritis-associated uveitis. Ped Reumatol 2016; 14: 27.[2]Seeling CA, Bauer O, Seeling H-P. Autoantibodies Against DFS70/LEDGF Exclusion Markers for Systemic Autoimmune Rheumatic Diseases (SARD). Clin. Lab 2016;62:499- 517.[3]Schmeling H et al. Autoantibodies to Dense Fine Speckles in Pediatric Diseases and Controls. The Journal of Rheumatology 2015;42(12):2419-2426.Disclosure of Interests:None declared
BackgroundJuvenile idiopathic arthritis (JIA) is the most common rheumatic disease in childhood and uveitis is one its major extra-articular manifestation. Children with JIA and a positive antinuclear antibodies (ANA) are known to be at risk to develop severe uveitis, which is usually asymptomatic and can lead to blindness if misdiagnosed. To date the finding of markers of uveitis is still a challenge. In adults anti-DFS70, ANA which have indirect immunofluorescence pattern described as dense fine spleckles (DFS pattern) and bind a 70kDa protein in immunoblot or chemioluminescence, are reported to be a marker of otherwise healthy individuals among asymptomatic ANA positive patients (in the absence of anti-Extractable Nuclear Antigens antibodies). The role of anti-DFS70 in children is yet not established even though but it has also been reported with a remarkably high frequency in unhealthy children (localized scleroderma, juvenile dermatomyositis and uveitis); it has also been detected in 2.1% of healthy children.ObjectivesThe aim of our observational study is to evaluate the correlation between uveitis and anti-DFS70 antibodies in children with JIA.Methods36 paediatric patients (24 females, 12 males) affected by JIA admitted to the Rheumatology Unit of Verona were evaluated. For each patient the following data were analysed: JIA subtype, ANA positivity, anti-DFS positivity, presence of uveitis diagnosed by a pediatric ophthalmologist.ResultsIn our series oligoarticular ANA + JIA was the predominant subtype (25 cases, 70% of total JIA cases), followed by oligoarticular ANA – subtype (6 cases, 16.5% of total), poliarticular ANA – subtype (3 cases, 8% of total) and poliarticular ANA + (2 cases, 5.5% of total).All 6 patients (16.5%), who developed mono- or bilateral uveitis, were affected by oligoarticular ANA + JIA and presented DFS ANA pattern, confirming the evidence that DFS ANA pattern is the most common pattern associated with uveitis. Only one patient presented anti-ENA antibodies encompassing anti-DFS70 positivity. None of the other five patients presented antibodies anti-DFS70, regardless of the clinical history of uveitis.ConclusionDSF ANA pattern remains the most common pattern seen in JIA patients and seems to be an hallmark of uveitis. In our series we found that neither JIA nor the risk of uveitis in JIA correlates with anti-DFS70 isolated positivity. The role of these antibodies in children remains unclear. Further studies are necessary to identify a reliable biomarker to guide ophthalmologic screening in JIA patients in order to identify children likely to develop uveitis.References[1] Conrad K, et al. The Clinical Relevance of Anti-DFS70 Autoantibodies. Clinic Rev Allerg Immunol. 2017; 52: 202.[2] Malyavantham K, et al. Analysis of DFS70 pattern and impact on ANA screening using a novel HEp-2 ELITE/DFS70 knockout substrate. Auto Immun highlights 2017; 8: 3[3] Mariz, et al. Pattern on the antinuclear antibody-HEp-2 is a critical parameter for discriminating antinuclear antibody-positive healthy indivi...
BackgroundTherapy of oligoarticular JIA is based on intrarticular injections of steroids, methotrexate and biotechnological therapy. ANAs-positive oligoarticular JIA patients with an early onset of disease have a consistent risk to develop uveites.ObjectivesThe primary aim of this study is to evaluate longitudinally the effect of non-systemic therapy and of systemic immmunomodulating drugs (e.g MTX) on ANAs in JIA patients; secondary occurance of iridocyclitis was evaluated.MethodsMonocentric retrospective not randomised study of 40 patients affected by oligoarticular JIA (ILAR classification) with ANA positivity at the baseline (T0, time of diagnosis). Patient of Group 1 received only intra-articular infiltrations or NSAID. Patients of Group 2 were treated with DMARDs (most of them with subcutaneous MTX 15 mg/m2/week) or MTX +biotechnlogical therapies. The assay for ANAs (I.I.F.) was assessed in all patients at T0, at T1 (12 months) and T2 (24 months). At 24 months was also recorded the occurence of uveitis during the follow up. Exclusion criteria were the ANAs negativity at T0 and other subgroups of JIA.ResultsTwenty-one patients (52,5%) were treated with non-systemic therapy (Group 1), and ninenteen (47,5%) with systemic immunomodulating therapy (Group 2). In Group 2 fifteen subjects were treated with MTX, one with MTX plus cyclosporine, one with only cyclosporine, two with MTX plus biotechnological agent (etarnecept and adalimumab). At T1, in Group 1 only one patient out of 20 (4,8%) became ANAs negative versus 42,1% (8/19) of patients in Group 2 (p 0.0033). At T2 the incidence of ANAs positivity did not change in Group 1 (only 1/21 ANAs negative), while 42,1% of patients treated with systemic therapies were ANA negative (p 0.006). Three patients were lost at the follow up. The two patients who received bDMARDs in addition to MTX remained ANAs-positive both in T1 and T2. At the end of follow-up period eight uveitis have occured, six in ANAs positive patients of Group 1 (6/21, 28,5%) and two in ANA negatives patients in Group 2 (2/19, 10,5%).ConclusionsANAs-positive patients treated with methotrexate seems to have higher possibility to became ANAs-negative versus patients treated with non-systemic immunomodulating therapy. It’s known that MTX might prevent onset of uveitis in JIA, as shown in our results. Demostrating a negativization of antinuclear antibodies using MTX therapy could help to add a role of this drug in the disease history of the oligoarthritis.Disclosure of InterestNone declared
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