Clinical Characteristics of Patients with Palindromic Rheumatism: Is There any Difference Between Seronegative and Seropositive Patients?

Abstract

Palindromic rheumatism (PR) is a clinical entity, which is characterized by remittent flares of pain, swelling and erythema around the joints that do not lead to persistent damage. In the literature, the frequencies of rheumatoid factor (RF) positivity and anti-cyclic citrullinated peptide (anti-CCP) antibody positivity have been reported as 39-68% in patients with PR. To our knowledge, studies investigating the comparison of clinical features of seronegative and seropositive patients are scarce. We aimed to assess demographic data and clinical characteristics of PR patients who followed in our clinic and to compare these features between seronegative and seropositive patients. The current study was designed as retrospective and cross-sectional. Thirty-one patients were screened and 21 patients who met the inclusion and exclusion criteria were included in the study. The median age, median follow-up duration, and median flare duration were 43 years, 18 months, and 3 days, respectively. Twelve patients were in seronegative group while 9 patients were in seropositive group. The median age was higher in seropositive patients than in seronegative patients (55 vs. 42 years, p=0.023). Even though the duration of flare and smoking cigarette were higher in seropositive group, there was not statistically significant difference between groups (4 vs. 2 days and 28.3 vs. 9.7 packs/years, respectively). Comorbidities were common in seronegative patients but there was not statistically significant difference between groups. Mean lymphocyte count was statistically significantly higher in seropositive group (p=0.037). In conclusion, the median age and mean lymphocyte count were higher in seropositive group than in seronegative group.

Keywords

• Anti-cyclic citrullinated peptide
• rheumatoid factor
• rheumatoid arthritis
• palindromic rheumatism

Palindromik Romatizma Tanılı Hastaların Klinik Özellikleri: Seropozitif ve Seronegatif Hastalar Arasında Fark Var mı?

Abstract

Palindromik romatizma (PR), eklemlerde kalıcı hasara neden olmaksızın alevlenmeler şeklinde ağrı, şişlik ve eritem ile karakterize klinik bir sendromdur. Literatürde, PR hastalarının %39-68’inde Romatoid faktör (RF) ve antisiklik sitrüline peptid (anti-CCP) antikor pozitifliği bulunmaktadır. Literatürde seropozitif ve seronegatif PR hastalarının klinik özelliklerini karşılaştıran çok az çalışma olup hala aydınlatılmamış birçok nokta vardır. Bu çalışmada kliniğimizde takipli PR tanılı hastaların demografik ve klinik özelliklerinin değerlendirilmesi, seropozitif ve seronegatif PR hastalarının karşılaştırılması planlandı. Çalışma retrospektif, kesitsel bir çalışma olarak tasarlandı. Otuz bir hasta tarandı ve dahil etme ve dışlama kriterlerini karşılayan 21 hasta çalışmaya alındı. Hastaların ortanca yaşı 43 yıl, ortanca takip süreleri 18 ay ve ortanca alevlenme süresi 3 gündü. Seronegatif grupta 12 hasta yer alırken seropozitif grupta 9 hasta mevcuttu. Seropozitif grubun ortanca yaş değeri daha yüksekti (55 yıl vs 42 yıl, p=0,023). Alevlenme süresi ve sigara içme süresi seropozitif grupta daha uzun olmasına rağmen istatistiksel olarak anlamlı bulunmadı (Sırasıyla, 4 gün vs 2 gün ve 28,3 paket/yıl vs 9,7 paket/yıl). Eşlik eden komorbideteler seronegatif hastalarda daha fazla olmasına rağmen istatistiksel olarak anlamlı değildi. Ortalama lenfosit değerleri ise seropozitif grupta anlamlı daha yüksekti (p=0,037). Sonuç olarak, seropozitif PR hastalarının ortanca yaşları negatif olan hastalara göre daha yüksek olduğu ve ortalama lenfosit değerlerinin seropozitif hastalarda daha yüksek olduğu bulundu.

Keywords

• Antisiklik sitrüline peptid
• romatoid faktör
• romatoid artrit
• palindromik romatizma

References

1. 1. Mankia K, Emery P. Palindromic rheumatism as part of the rheumatoid arthritis continuum. Nat Rev Rheumatol. 2019 Nov;15(11):687-695. doi: 10.1038/s41584-019-0308-5. Epub 2019 Oct 8. PMID: 31595059.
2. 2. Pasero G, Barbieri P. Palindromic rheumatism: you just have to think about it! Clin Exp Rheumatol. 1986;4(3):197–199.
3. 3. Sanmartí R, Haro I, Cañete JD. Palindromic rheumatism: a unique and enigmatic entity with a complex relationship with rheumatoid arthritis. Expert Rev Clin Immunol. 2021 Apr;17(4):375-384. doi: 10.1080/1744666X.2021.1899811. Epub 2021 Mar 17. PMID: 33666522.
4. 4. Gonzalez-Lopez L, Gamez-Nava JI, Jhangri GS, Ramos-Remus C, Russell AS, Suarez-Almazor ME. Prognostic factors for the development of rheumatoid arthritis and other connective tissue diseases in patients with palindromic rheumatism. J Rheumatol. 1999;26(3):540-545.
5. 5. Ahn JK, Hwang J, Seo GH. Incidence and risk of developing rheumatic diseases in 19,724 patients with palindromic rheumatism in South Korea: A nationwide population-based study. Joint Bone Spine. 2021;88(3):105128. doi:10.1016/j.jbspin.2020.105128
6. 6. Mankia K, Emery P. Palindromic rheumatism as part of the rheumatoid arthritis continuum. Nat Rev Rheumatol. 2019;15(11):687-695. doi:10.1038/s41584-019-0308-5
7. 7. Sanmartí R, Frade-Sosa B, Morlà R, et al. Palindromic Rheumatism: Just a Pre-rheumatoid Stage or Something Else?. Front Med (Lausanne). 2021;8:657983. Published 2021 Mar 25. doi:10.3389/fmed.2021.657983
8. 8. Castellanos-Moreira R, Rodriguez-Garcia SC, Cabrera-Villalba S, et al. Anti-carbamylated protein antibody isotype pattern differs between palindromic rheumatism and rheumatoid arthritis. Ther Adv Musculoskelet Dis. 2020;12:1759720X20978139. Published 2020 Dec 10. doi:10.1177/1759720X20978139

9. 9. Cañete JD, Arostegui JI, Queiró R, et al. An unexpectedly high frequency of MEFV mutations in patients with anti-citrullinated protein antibody-negative palindromic rheumatism. Arthritis Rheum. 2007;56(8):2784-2788. doi:10.1002/art.22755
10. 10. Cuervo A, Sanmartí R, Ramírez J, et al. Palindromic rheumatism: Evidence of four subtypes of palindromic-like arthritis based in either MEFV or rheumatoid factor/ACPA status [published online ahead of print, 2021 Jun 5]. Joint Bone Spine. 2021;88(6):105235. doi:10.1016/j.jbspin.2021.105235
11. 11. Hannonen P, Mütt.nen T, Oka M. Palindromic rheumatism: a clinical survey of sixty patients. Scandinavian J Rheumatol.1987;16(6):413–420.
12. 12. Cabrera-Villalba S, Ramirez J, Salvador G, et al. Is there subclinical synovitis in patients with palindromic rheumatism in the intercritical period? A clinical and ultrasonographic study according to anticitrullinated protein antibody status. J Rheumatol. 2014;41 (8):1650–1655.
13. 13. Khabbazi A, Hajialiloo M, Kolahi S, Soroosh M, Esalatmanesh K, Sharif S. A multicenter study of clinical and laboratory findings of palindromic rheumatism in Iran. Int J Rheum Dis. 2012;15(4):427-430. doi:10.1111/j.1756-185X.2012.01739.x
14. 14. Mankia K, D'Agostino MA, Wakefield RJ, et al. Identification of a distinct imaging phenotype may improve the management of palindromic rheumatism. Ann Rheum Dis. 2019;78(1):43-50. doi:10.1136/annrheumdis-2018-214175
15. 15. Chen HH, Chao WC, Liao TL, Lin CH, Chen DY. Risk of autoimmune rheumatic diseases in patients with palindromic rheumatism: A nationwide, population-based, cohort study. PLoS One. 2018;13(7):e0201340. Published 2018 Jul 26. doi:10.1371/journal.pone.0201340