Some recent clinical evidence shows that Monoarticular forms, with arthritis limited to one joint, currently included according to ILAR criteria in the Oligoarticular subtype, are distinct from forms involving 2 to 4 joints instead, but little is known about the clinical course and risk factors for arthritis relapse in the two clinical entities. Aim: The objectives of the following study are: to compare in the two groups (Mono and Oligo) some clinical-laboratory characteristics, clinical course in terms of arthritis relapse according to current therapy, pubertal stage and other factors (sex, uveitis, ANA, SILB).Materials and methods: We performed a retrospective analysis of prospectively collected data from patients with a diagnosis of Oligoarticular JIA (ILAR criteria) followed at the Pediatric Rheumatology Unit of Padua. Patients were studied by dividing them into two groups: subjects with Monoarticular JIA, with exclusive involvement of one joint for at least 5 years after disease onset, and subjects with Oligoarticular JIA, with involvement of 2 to 4 joints. Each patient was evaluated at onset, then every 3-4 months depending on clinical status. Variables assessed were gender, age, presence of BJHS ≥5/9 Beigton criteria, presence of ANA (positivity with titer > 1/80 on Hep2 cells), presence and site of active arthritis, presence of uveitis, therapy, and outcome. Arthritis relapse was defined as the recurrence of active arthritis after at least 3 months of remission, with the need to intensify current therapy. Puberty status was defined by the onset of menarche in females and the presence of pubic and axillary hair and a testicular volume ≥12 ml in males. The average age of onset of puberty in the Italian population is 10 and 12 years, respectively.Results: A total of 343 patients were studied,118 with Monoarticular JIA and 225 with OligoJIA. MonoJIA had a higher proportion of male subjects, lower frequency of uveitis and ANA+, and higher frequency of BJHS. During follow-up, 98.7% of patients with Oligo JIA have at least one arthritis relapse, while more than one in two subjects with mono JIA never relapses. The average frequency of arthritis relapses/year/patient was 0.11 in Mono and 0.47 in Oligo, thus relapsing on average 4.27 times more than Monoarticular forms. In the merged group (mono and oligo), 32.8 percent of relapses occurred while being treated with DMARDs. Differential analysis for the two groups showed that in Mono, 90.6% of relapses occurred without therapy or on treatment with AIDs, compared with 64.9% in Oligo. In total, relapses in DMARDs amounted to 9.3% of relapses in Mono JIA, while in Oligo more than 1 in 3 (35.1%). None of the variables under analysis (gender, uveitis, ANA+ and SILB) is associated with a different frequency of arthritis relapse/year in the two groups. The mean annual frequency of arthritis relapses was significantly lower in the post-pubertal period than in the pre-pubertal phase in both Mono and Oligo. Puberty turns out to be a positive element in clinical evolution from the point of view of joint manifestations. Finally, Oligo more frequently require second-line treatments during follow-up and have a less benign long-term outcome than Monoarticular forms. Conclusions: Mono JIA has distinct features compared to Oligo JIA: later onset, greater presence of SILB, lower frequency of uveitis and ANA+, need for less aggressive and predominantly local therapies, and more benign prognosis. Nearly all patients with Oligoarthritis experience at least one arthritis relapse, whereas in Mono, about one in two patients experience no relapse throughout follow-up. Oligo JIA is more aggressive in terms of arthritis relapses/year, with suboptimal response to secondline treatments, greater use of DMARDs during follow-up, and worse final outcome. Puberty appears to be a positive prognostic factor with a significant reduction in the frequency of relapses in post-pubertal phase in the two forms.
Alcune recenti evidenze cliniche dimostrano che che le forme Monoarticolari, incluse secondo i criteri ILAR nel sottotipo Oligoarticolare, siano distinte dalle forme che coinvolgono da 2 a 4 articolazioni, ma poco è conosciuto riguardo al decorso clinico e ai fattori di rischio di ricaduta di artrite nelle due entità cliniche. Scopo: Gli obiettivi del seguente studio sono: confrontare nei due gruppi (Mono e Oligo) alcune caratteristiche clinico-laboratoristiche, il decorso clinico in termini di ricadute di artrite in base alla terapia in atto, stadio puberale e altri fattori (sesso, uveite, ANA, SILB).Materiali e metodi: Si è effettuata un’analisi retrospettiva di una raccolta dati prospettica di pazienti con diagnosi di AIG Oligoarticolare (criteri ILAR) seguiti presso la Reumatologia Pediatrica di Padova. I pazienti sono stati studiati suddividendoli in due gruppi: soggetti con AIG Monoarticolare, artrite in una articolazione per almeno 5 anni dall’esordio, e soggetti con AIG Oligoarticolare, con artrite in 2-4 articolazioni. Ogni paziente è stato valutato all’esordio, quindi ogni 3-4 mesi a seconda dello stato clinico. Le variabili valutate sono state: sesso, età, presenza di SILB, ≥5/9 criteri di Beigton), presenza di ANA (positività con titolo > 1/80 su Hep2 cells), presenza e sede di artrite attiva, presenza di uveite, terapia e outcome. La ricaduta di artrite è stata definita come la ricomparsa di artrite attiva dopo almeno 3 mesi di remissione, con la necessità di intensificare la terapia in atto. Lo stato di pubertà è stato definito dalla comparsa del menarca nelle femmine e dalla presenza di peli pubici e ascellari e un volume testicolare ≥12 ml nei maschi. L’età media di inizio della pubertà nella popolazione italiana è rispettivamente di 10 e 12 anni.Risultati:Sono stati studiati 343 pazienti,118 con AIG Mono e 225 con AIG Oligoarticolare. L’AIG Mono presenta una maggior percentuale di soggetti di sesso maschile, minor frequenza di uveite e di ANA+ e maggior frequenza di SILB. Durante il follow-up il 98,7% dei pazienti con AIG Oligo presenta almeno una ricaduta di artrite, mentre più di un soggetto su due con AIG Mono non ricade mai. La frequenza media di ricadute di artrite/anno/paziente è risultata di 0,11 nelle Mono e di 0,47 nelle Oligo, che ricadono quindi mediamente 4,27 volte in più delle forme Mono. Nel gruppo accorpato il 32,8% delle ricadute sono avvenute con DMARDs. Dall’analisi dei due gruppi è emerso che nelle Mono il 90,6% delle ricadute è avvenuta senza terapia o con AIDs, rispetto al 64,9% nelle Oligo. Le ricadute in DMARDs ammontano al 9,3% delle ricadute nell’AIG Mono, nell’Oligo invece a più di una ricaduta su 3 (35,1%). Nessuna delle variabili in analisi (genere, uveite, ANA+ e SILB) è associata a una differente frequenza di ricadute di artrite/anno nei due gruppi. La frequenza media annua di ricadute di artrite è risultata minore nel periodo post-puberale rispetto alla fase pre-puberale sia nelle Mono che nelle Oligo. La pubertà si rivela essere un elemento positivo nell’evoluzione clinica dal punto di vista delle manifestazioni articolari. Infine le Oligo necessitano più frequentemente trattamenti di seconda linea durante il follow-up e presentano un outcome a lungo termine meno benigno rispetto alle forme Mono. Conclusioni: L’AIG Mono ha caratteristiche distinte rispetto all’AIG Oligo, ovvero: esordio più tardivo, maggior presenza di SILB, minor frequenza di uveite e ANA+, necessità di terapie meno aggressive e prevalentemente di tipo locale e prognosi più benigna. L’AIG Oligo risulta più aggressiva in termini di ricadute di artrite/anno, con una risposta subottimale ai trattamenti di seconda linea, maggior ricorso a trattamenti con DMARDs durante il follow-up e peggior outcome finale. La pubertà risulta un fattore prognostico positivo con una significativa riduzione della frequenza di ricadute in età post-puberale.
Fattori di rischio per ricaduta di artrite nell’AIG Monoarticolare e Oligoarticolare
RIGONI, CHIARA
2022/2023
Abstract
Some recent clinical evidence shows that Monoarticular forms, with arthritis limited to one joint, currently included according to ILAR criteria in the Oligoarticular subtype, are distinct from forms involving 2 to 4 joints instead, but little is known about the clinical course and risk factors for arthritis relapse in the two clinical entities. Aim: The objectives of the following study are: to compare in the two groups (Mono and Oligo) some clinical-laboratory characteristics, clinical course in terms of arthritis relapse according to current therapy, pubertal stage and other factors (sex, uveitis, ANA, SILB).Materials and methods: We performed a retrospective analysis of prospectively collected data from patients with a diagnosis of Oligoarticular JIA (ILAR criteria) followed at the Pediatric Rheumatology Unit of Padua. Patients were studied by dividing them into two groups: subjects with Monoarticular JIA, with exclusive involvement of one joint for at least 5 years after disease onset, and subjects with Oligoarticular JIA, with involvement of 2 to 4 joints. Each patient was evaluated at onset, then every 3-4 months depending on clinical status. Variables assessed were gender, age, presence of BJHS ≥5/9 Beigton criteria, presence of ANA (positivity with titer > 1/80 on Hep2 cells), presence and site of active arthritis, presence of uveitis, therapy, and outcome. Arthritis relapse was defined as the recurrence of active arthritis after at least 3 months of remission, with the need to intensify current therapy. Puberty status was defined by the onset of menarche in females and the presence of pubic and axillary hair and a testicular volume ≥12 ml in males. The average age of onset of puberty in the Italian population is 10 and 12 years, respectively.Results: A total of 343 patients were studied,118 with Monoarticular JIA and 225 with OligoJIA. MonoJIA had a higher proportion of male subjects, lower frequency of uveitis and ANA+, and higher frequency of BJHS. During follow-up, 98.7% of patients with Oligo JIA have at least one arthritis relapse, while more than one in two subjects with mono JIA never relapses. The average frequency of arthritis relapses/year/patient was 0.11 in Mono and 0.47 in Oligo, thus relapsing on average 4.27 times more than Monoarticular forms. In the merged group (mono and oligo), 32.8 percent of relapses occurred while being treated with DMARDs. Differential analysis for the two groups showed that in Mono, 90.6% of relapses occurred without therapy or on treatment with AIDs, compared with 64.9% in Oligo. In total, relapses in DMARDs amounted to 9.3% of relapses in Mono JIA, while in Oligo more than 1 in 3 (35.1%). None of the variables under analysis (gender, uveitis, ANA+ and SILB) is associated with a different frequency of arthritis relapse/year in the two groups. The mean annual frequency of arthritis relapses was significantly lower in the post-pubertal period than in the pre-pubertal phase in both Mono and Oligo. Puberty turns out to be a positive element in clinical evolution from the point of view of joint manifestations. Finally, Oligo more frequently require second-line treatments during follow-up and have a less benign long-term outcome than Monoarticular forms. Conclusions: Mono JIA has distinct features compared to Oligo JIA: later onset, greater presence of SILB, lower frequency of uveitis and ANA+, need for less aggressive and predominantly local therapies, and more benign prognosis. Nearly all patients with Oligoarthritis experience at least one arthritis relapse, whereas in Mono, about one in two patients experience no relapse throughout follow-up. Oligo JIA is more aggressive in terms of arthritis relapses/year, with suboptimal response to secondline treatments, greater use of DMARDs during follow-up, and worse final outcome. Puberty appears to be a positive prognostic factor with a significant reduction in the frequency of relapses in post-pubertal phase in the two forms.File | Dimensione | Formato | |
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https://hdl.handle.net/20.500.12608/47022