Skip to content
2000
Volume 21, Issue 5
  • ISSN: 1573-3971
  • E-ISSN: 1875-6360

Abstract

Introduction

Unlike restrictive pulmonary function and apical fibrobullous disease, diffuse interstitial lung disease is scarce in patients with ankylosing spondylitis (AS). We present a systematic review of the association between AS and SS. We also report a new case of SS revealed by interstitial lung disease in AS patients treated with tumor necrosis factor (TNF) inhibitors.

Materials and Methods

The systematic review was performed according to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guideline using the MEDLINE and SCOPUS databases and included case reports and case series describing the association between AS and SS.

Results

There were sixty-three patients, including our case: 16 males and 47 females. The mean age was 49.2 years. The mean SpA duration was 14.1 years. The mean delay between SpA and SS was 12.8 years (0-27). SS was diagnosed after SpA in 62% of cases (n=39). It preceded SpA in 36.5% (n=23) and was concomitant with SpA in 1 case. All patients had sicca symptoms. Minor salivary gland biopsy, showed focal sialadenitis grade III or grade IV in the Chisholm classification in 20 patients. Anti-nuclear antibody was positive in 75.8% of cases. Among them, anti-SSA and anti-SSB were positive in 44.4% and 35.3% of cases. Except for our patient, no patient had interstitial lung disease. SS extra glandular manifestations were reported in 12 cases.

Conclusion

The occurrence of Sjögren’s syndrome is uncommon in patients with ankylosing spondylitis. This association has been reported in the literature, suggesting a pathogenetic link between these two diseases. This association should be considered in ankylosing spondylitis patients with diffuse interstitial lung disease. Knowing this association is necessary for therapeutic adjustment.

Our study has some limitations. Publication bias was the major bias in our study. Indeed, we only included case reports and case series describing the association between SpA and SS. We did not search for unpublished work. Moreover, the follow-up was not specified in most included articles.

© 2025 Bentham Science Publishers
Loading

Article metrics loading...

/content/journals/crr/10.2174/0115733971315532240920065136
2024-11-05
2025-12-22

  • From This Site

    /content/journals/crr/10.2174/0115733971315532240920065136
    dcterms_title,dcterms_subject,pub_keyword
    -contentType:Contributor -contentType:Concept -contentType:Institution
    10
    5
Loading full text...

Full text loading...

References

  1. ZochlingJ. BrandtJ. BraunJ. The current concept of spondyloarthritis with special emphasis on undifferentiated spondyloarthritis.Rheumatology200544121483149110.1093/rheumatology/kei04716091395
     [Google Scholar]
  2. RudwaleitM. van der HeijdeD. LandewéR. AkkocN. BrandtJ. ChouC.T. DougadosM. HuangF. GuJ. KirazliY. Van den BoschF. OlivieriI. RoussouE. ScarpatoS. SørensenI.J. Valle-OñateR. WeberU. WeiJ. SieperJ. The assessment of spondylo arthritis international society classification criteria for peripheral spondyloarthritis and for spondyloarthritis in general.Ann. Rheum. Dis.2011701253110.1136/ard.2010.13364521109520
     [Google Scholar]
  3. El MaghraouiA. Extra-articular manifestations of ankylosing spondylitis: Prevalence, characteristics and therapeutic implications.Eur. J. Intern. Med.201122655456010.1016/j.ejim.2011.06.00622075279
     [Google Scholar]
  4. KanathurN. Lee-ChiongT. Pulmonary manifestations of ankylosing spondylitis.Clin. Chest Med.201031354755410.1016/j.ccm.2010.05.00220692546
     [Google Scholar]
  5. ZhangL.W. ZhouP.R. WeiP. CongX. WuL.L. HuaH. Expression of interleukin-17 in primary Sjögren’s syndrome and the correlation with disease severity: A systematic review and meta-analysis.Scand. J. Immunol.2018874e1264910.1111/sji.1264929476557
     [Google Scholar]
  6. TaamsL.S. SteelK.J.A. SrenathanU. BurnsL.A. KirkhamB.W. IL-17 in the immunopathogenesis of spondyloarthritis.Nat. Rev. Rheumatol.201814845346610.1038/s41584‑018‑0044‑230006601
     [Google Scholar]
  7. WhaleyK. ChisholmD.M. WilliamsonJ. DickW.C. NukiG. BuchananW.W. Sjögren’s syndrome in psoriatic arthritis, ankylosing spondylitis and Reiter’s syndrome.Acta Rheumatol. Scand.1971171-410511410.3109/rhe1.1971.17.issue‑1‑4.165579064
     [Google Scholar]
  8. ClaymanM.D. ReinertsenJ.L. Ankylosing spondylitis with subsequent development of rheumatoid arthritis, Sjögren’s syndrome, and rheumatoid vasculitis.Arthritis Rheum.197821338338910.1002/art.1780210315646836
     [Google Scholar]
  9. CollinsP. RogersS. JacksonJ. McCARTANB. Psoriasis, psoriatic arthritis and the possible association with Sjogren’s syndrome.Br. J. Dermatol.1992126324224510.1111/j.1365‑2133.1992.tb00652.x1554600
     [Google Scholar]
  10. BrandtJ. RudwaleitM. EggensU. MertzA. DistlerA. SieperJ. BraunJ. Increased frequency of Sjögren’s syndrome in patients with spondyloarthropathy.J. Rheumatol.19982547187249558175
     [Google Scholar]
  11. Scotto di FazanoC. GriloR.M. VergneP. CoyralD. InaouiR. BonnetC. BertinP. TrèvesR. Is the relationship between spondyloarthropathy and Sjögren’s syndrome in women coincidental? A study of 13 cases.Joint Bone Spine200269438338710.1016/S1297‑319X(02)00414‑112184435
     [Google Scholar]
  12. BrandtJ. MaierT. RudwaleitM. KühlU. HiepeF. SieperJ. BraunJ. Co-occurrence of spondyloarthropathy and connective tissue disease: Development of Sjögren’s syndrome and mixed connective tissue disease (MCTD) in a patient with ankylosing spondylitis.Clin. Exp. Rheumatol.2002201808411892716
     [Google Scholar]
  13. TosunM. KarkucakM. CapkinE. CakirbayH. GulerM. TopbasM. Coexisting ankylosing spondylitis and Sjogren’s syndrome: A case report.Rheumatol. Int.200525647848010.1007/s00296‑005‑0598‑615761725
     [Google Scholar]
  14. ChangH.K. BangK.T. LeeB.H. KimJ.H. BaeK.W. KimM.J. KimS.K. Concurrence of Sjögren’s syndrome in a patient with Chlamydia-induced reactive arthritis; An unusual finding.Korean J. Intern. Med.200621211611910.3904/kjim.2006.21.2.11616913441
     [Google Scholar]
  15. KobakS. Celebi KobakA. KabasakalY. DoganavsargilE. Sjögren’s syndrome in patients with ankylosing spondylitis.Clin. Rheumatol.200626217317510.1007/s10067‑006‑0255‑916547690
     [Google Scholar]
  16. JarrotP.A. ArcaniR. DarmonO. RoudierJ. CauchoisR. MazodierK. JeanR. BalandraudN. KaplanskiG. Axial articular manifestations in primary sjögren syndrome: Association with spondyloarthritis.J. Rheumatol.20214871037104610.3899/jrheum.20018932669446
     [Google Scholar]
  17. BruzzeseV MarreseC ScolieriP PepeJ. Psoriatic spondyloarthritis and Sjögren syndrome: A casual association?Reumatismo202072317017210.4081/reumatismo.2020.1287
     [Google Scholar]
  18. ZhaoGW HuangLF LiD ZengY Ankylosing spondylitis coexists with rheumatoid arthritis and Sjögren’s syndrome: A case report with literature review.Clin. Rheumatol.20214052083208610.1007/s10067‑020‑05350‑7
     [Google Scholar]
  19. WhittinghamK. BarnesS. GardinerC. Tools to measure quality of life and carer burden in informal carers of heart failure patients: A narrative review.Palliat. Med.201327759660710.1177/026921631347717923442879
     [Google Scholar]
  20. ChisholmD.M. MasonD.K. Labial salivary gland biopsy in Sjögren’s disease.J. Clin. Pathol.196821565666010.1136/jcp.21.5.6565697370
     [Google Scholar]
  21. VoulgarelisM. TzioufasA.G. Current aspects of pathogenesis in Sjögren’s syndrome.Ther. Adv. Musculoskelet. Dis.20102632533410.1177/1759720X1038143122870458
     [Google Scholar]
  22. MoutsopoulosH.M. ChusedT.M. MannD.L. KlippelJ.H. FauciA.S. FrankM.M. LawleyT.J. HamburgerM.I. Sjögren’s syndrome (Sicca syndrome): Current issues.Ann. Intern. Med.1980922_Part_121222610.7326/0003‑4819‑92‑2‑2127352730
     [Google Scholar]
  23. SloumaM. KharratL. TezegdentiA. DhahriR. GhazouaniE. GharsallahI. Pro-inflammatory cytokines in spondyloarthritis: A case-control study.Expert Rev. Clin. Immunol.202420665566310.1080/1744666X.2024.230408038205504
     [Google Scholar]
  24. SloumaM. KharratL. GharsallahI. Interleukin-22 and the diagnosis of axial spondyloarthritis.Int. J. Rheum. Dis.202225223623710.1111/1756‑185X.1427534970838
     [Google Scholar]
  25. SloumaM. KharratL. TezegdentiA. MetouiL. DhahriR. GhazouaniE. CheourE. GharsallahI. LouzirB. Increased serum interleukin 22 levels in patients with axial spondyloarthritis.Expert Rev. Clin. Immunol.202319112312910.1080/1744666X.2023.214256336326666
     [Google Scholar]
  26. MozS. AitaA. BassoD. RamondaR. PlebaniM. PunziL. Spondyloarthritis: Matrix metalloproteinasesas biomarkers of pathogenesis and response to tumor necrosis factor (TNF) inhibitors.Int. J. Mol. Sci.201718483010.3390/ijms1804083028420081
     [Google Scholar]
  27. SloumaM., Bouzid, S., Dhahri, R., Rahmouni, S., Litaiem, N., Gharsallah, I., Metoui, L., Louzir, B. Matrix metalloproteinases; a biomarker of disease activity and prognosis in spondyloarthritis: A narrative review. Curr. Rev. Clin. Experimen. Pharmacol.2023181313810.2174/277243281766622011311280935049445
     [Google Scholar]
  28. ReveilleJ.D. ZhouX. LeeM. WeismanM.H. YiL. GenslerL.S. ZouH. WardM.M. IshimoriM.L. LearchT.J. HeD. RahbarM.H. WangJ. BrownM.A. HLA class I and II alleles in susceptibility to ankylosing spondylitis.Ann. Rheum. Dis.2019781667310.1136/annrheumdis‑2018‑21377930341055
     [Google Scholar]
  29. LiuS. ZhangJ. LiuM. ChenQ. WangS. WangD. SunL. New-onset primary Sjögren’s syndrome following exposure to severe acute respiratory syndrome coronavirus 2: A retrospective cohort study.Rheumatol. Autoimmun.202441374610.1002/rai2.12113
     [Google Scholar]
  30. SloumaM. AbbesM. KharratL. GharsallahI. Post-COVID-19 reactive arthritis.Clin. Rheumatol.20224182609261010.1007/s10067‑022‑06241‑935788920
     [Google Scholar]
  31. SloumaM. AbbesM. MehmliT. DhahriR. MetouiL. GharsallahI. LouzirB. Reactive arthritis occurring after COVID-19 infection: A narrative review.Infection2023511374510.1007/s15010‑022‑01858‑z35655110
     [Google Scholar]
  32. SloumaM. MhemliT. AbbesM. TrikiW. DhahriR. MetouiL. GharsallahI. LouzirB. Rheumatoid arthritis occurring after coronavirus disease 2019 (COVID-19) infection: Case based review.Egypt. Rheumatol.202244327527810.1016/j.ejr.2022.03.00137521095
     [Google Scholar]
  33. BerdalG. HalvorsenS. van der HeijdeD. MoweM. DagfinrudH. Restrictive pulmonary function is more prevalent in patients with ankylosing spondylitis than in matched population controls and is associated with impaired spinal mobility: A comparative study.Arthritis Res. Ther.2012141R1910.1186/ar369922277377
     [Google Scholar]
  34. DincerU. CakarE. KiralpM.Z. BozkanatE. KilacH. DursunH. The pulmonary involvement in rheumatic diseases: pulmonary effects of ankylosing spondylitis and its impact on functionality and quality of life.Tohoku J. Exp. Med.2007212442343010.1620/tjem.212.42317660708
     [Google Scholar]
  35. El MaghraouiA. ChaouirS. AbidA. BezzaA. TabacheF. AchemlalL. AbouzahirA. GhafirD. OhayonV. ArchaneM.I. Lung findings on thoracic high-resolution computed tomography in patients with ankylosing spondylitis. Correlations with disease duration, clinical findings and pulmonary function testing.Clin. Rheumatol.200423212312810.1007/s10067‑003‑0845‑815045626
     [Google Scholar]
  36. Sampaio-BarrosP.D. CerqueiraE.M.F.P. RezendeS.M. MaedaL. CondeR.A. ZanardiV.A. BértoloM.B. de Menezes NetoJ.R. SamaraA.M. Pulmonary involvement in ankylosing spondylitis.Clin. Rheumatol.200726222523010.1007/s10067‑006‑0286‑216572281
     [Google Scholar]
  37. IbinaiyeP.O. SalawuF. Pulmonary manifestations of ankylosing spondylitis treated as pulmonary tuberculosis: A case report and review of literature.Niger. Postgrad. Med. J.200916427427610.4103/1117‑1936.18141320037624
     [Google Scholar]
  38. RosenowE. StrimlanC.V. MuhmJ.R. FergusonR.H. Pleuropulmonary manifestations of ankylosing spondylitis.Mayo Clin. Proc.19775210641649909316
     [Google Scholar]
  39. LeeC.C. LeeS.H. ChangI.J. LuT.C. YuanA. ChangT.A. TsaiK.C. ChenW.J. Spontaneous pneumothorax associated with ankylosing spondylitis.Rheumatology200544121538154110.1093/rheumatology/kei07716105907
     [Google Scholar]
  40. RumancikW.M. FiroozniaH. DavisM.S.Jr LeitmanB.S. GolimbuC. RafiiM. McCauleyD.I. Fibrobullous disease of the upper lobes: An extraskeletal manifestation of ankylosing spondylitis.J. Comput. Tomogr.19848322522910.1016/0149‑936X(84)90066‑36744925
     [Google Scholar]
  41. CohenA.A. NatelsonE.A. FechnerR.E. Fibrosing interstitial pneumonitis in ankylosing spondylitis.Chest197159436937110.1378/chest.59.4.3695551581
     [Google Scholar]
  42. CampbellA.H. MacDonaldC.B. Upper lobe fibrosis associated with ankylosing spondylitis.Br. J. Dis. Chest19655929010110.1016/S0007‑0971(65)80017‑114283439
     [Google Scholar]
  43. UffmannM. KienerH.P. BankierA.A. BaldtM.M. ZontsichT. HeroldC.J. Lung manifestation in asymptomatic patients with primary Sjögren syndrome: Assessment with high resolution CT and pulmonary function tests.J. Thorac. Imaging200116428228910.1097/00005382‑200110000‑0000911685093
     [Google Scholar]
  44. WangY. HouZ. QiuM. YeQ. Risk factors for primary Sjögren syndrome-associated interstitial lung disease.J. Thorac. Dis.20181042108211710.21037/jtd.2018.03.12029850114
     [Google Scholar]
  45. LohrmannC. UhlM. WarnatzK. GhanemN. KotterE. SchaeferO. LangerM. High-resolution CT imaging of the lung for patients with primary Sjögren’s syndrome.Eur. J. Radiol.200452213714310.1016/j.ejrad.2004.01.00615489070
     [Google Scholar]
  46. DeheinzelinD. CapelozziV.L. KairallaR.A. Barbas FilhoJ.V. SaldivaP.H. de CarvalhoC.R. Interstitial lung disease in primary Sjögren’s syndrome. Clinical-pathological evaluation and response to treatment.Am. J. Respir. Crit. Care Med.1996154379479910.1164/ajrccm.154.3.88106218810621
     [Google Scholar]
  47. DongX. ZhouJ. GuoX. LiY. XuY. FuQ. LuY. ZhengY. A retrospective analysis of distinguishing features of chest HRCT and clinical manifestation in primary Sjögren’s syndrome-related interstitial lung disease in a Chinese population.Clin. Rheumatol.201837112981298810.1007/s10067‑018‑4289‑630242640
     [Google Scholar]
  48. LiebowA.A. CarringtonC.B. Diffuse pulmonary lymphoreticular infiltrations associated with dysproteinemia.Med. Clin. North Am.197357380984310.1016/S0025‑7125(16)32278‑74540729
     [Google Scholar]
  49. Ramos-CasalsM. Brito-ZerónP. SerorR. BootsmaH. BowmanS.J. DörnerT. GottenbergJ.E. MarietteX. TheanderE. BombardieriS. De VitaS. MandlT. NgW.F. KruizeA. TzioufasA. VitaliC. EULAR Sjögren Syndrome Task Force Characterization of systemic disease in primary Sjögren’s syndrome: EULAR-SS Task Force recommendations for articular, cutaneous, pulmonary and renal involvements.Rheumatology201554122230223810.1093/rheumatology/kev20026231345
     [Google Scholar]
  50. MyersJ.L. ColbyT.V. Pathologic manifestations of bronchiolitis, constrictive bronchiolitis, cryptogenic organizing pneumonia, and diffuse panbronchiolitis.Clin. Chest Med.199314461162210.1016/S0272‑5231(21)00925‑48313666
     [Google Scholar]
  51. MüllerN.L. WhiteD.A. JiangH. GemmaA. Diagnosis and management of drug-associated interstitial lung disease.Br. J. Cancer200491S2S24S3010.1038/sj.bjc.660206415340375
     [Google Scholar]
  52. AtzeniF. BoiardiL. SallìS. BenucciM. Sarzi-PuttiniP. Lung involvement and drug-induced lung disease in patients with rheumatoid arthritis.Expert Rev. Clin. Immunol.20139764965710.1586/1744666X.2013.81117323899235
     [Google Scholar]
  53. PavyS. ConstantinA. PhamT. GossecL. MaillefertJ.F. CantagrelA. CombeB. FlipoR.M. GoupilleP. Le LoëtX. MarietteX. PuéchalX. SchaeverbekeT. SibiliaJ. TebibJ. WendlingD. DougadosM. Methotrexate therapy for rheumatoid arthritis: clinical practice guidelines based on published evidence and expert opinion.Joint Bone Spine200673438839510.1016/j.jbspin.2006.01.00716626993
     [Google Scholar]
  54. KremerJ.M. AlarcónG.S. WeinblattM.E. KaymakcianM.V. MacalusoM. CannonG.W. PalmerW.R. SundyJ.S. St ClairE.W. AlexanderR.W. SmithG.J. AxiotisC.A. Clinical, laboratory, radiographic, and histopathologic features of methotrexate-associated lung injury in patients with rheumatoid arthritis: A multicenter study with literature review.Arthritis Rheum.199740101829183710.1002/art.17804010169336418
     [Google Scholar]
  55. Sieiro SantosC. Álvarez CastroC. MoralesM.C. Díez ÁlvarezE. Anti-TNF-α-induced lupus syndrome.Z. Rheumatol.202180548148610.1007/s00393‑021‑00983‑833709168
     [Google Scholar]
  56. SkalkouA. PelechasE. VoulgariP.V. DrososA.A. TNF-induced Lupus. A case-based review.Curr. Rheumatol. Rev.2022181728210.2174/157339711766621110209433034727862
     [Google Scholar]
  57. ShovmanO. TamarS. AmitalH. WatadA. ShoenfeldY. Diverse patterns of anti-TNF-α-induced lupus: Case series and review of the literature.Clin. Rheumatol.201837256356810.1007/s10067‑017‑3884‑229063464
     [Google Scholar]
  58. Perez-AlvarezR. Perez-de-LisM. Diaz-LagaresC. Pego-ReigosaJ.M. RetamozoS. BoveA. Brito-ZeronP. BoschX. Ramos-CasalsM. Interstitial lung disease induced or exacerbated by TNF-targeted therapies: Analysis of 122 cases.Semin. Arthritis Rheum.201141225626410.1016/j.semarthrit.2010.11.00221277618
     [Google Scholar]
  59. NakashitaT. AndoK. KanekoN. TakahashiK. MotojimaS. Potential risk of TNF inhibitors on the progression of interstitial lung disease in patients with rheumatoid arthritis.BMJ Open201448e00561510.1136/bmjopen‑2014‑00561525125479
     [Google Scholar]
  60. KooB.S. HongS. KimY.J. KimY.G. LeeC.K. YooB. Mortality in patients with rheumatoid arthritis-associated interstitial lung disease treated with an anti-tumor necrosis factor agent.Korean J. Intern. Med.201530110410910.3904/kjim.2015.30.1.10425589842
     [Google Scholar]
/content/journals/crr/10.2174/0115733971315532240920065136
Loading
Sjögren’s Syndrome and Ankylosing Spondylitis Association: A Case-Based Review
Curr. Rheumatol. Rev. 21, 535 (2025); https://doi.org/10.2174/0115733971315532240920065136
/content/journals/crr/10.2174/0115733971315532240920065136
/content/journals/crr/10.2174/0115733971315532240920065136
Loading

Data & Media loading...

Supplements

Supplementary material, along with the published article, is available on the publisher's website.

file format download Download

  • Article Type:     Review Article
Keyword(s): ankylosing spondylitis; interstitial lung disease, tumor necrosis factor, anti-nuclear antibody; Sjögren’s syndrome; spondyloarthritis

Most Cited Most Cited RSS feed

This is a required field
Please enter a valid email address
Approval was a Success
Invalid data
An Error Occurred
Approval was partially successful, following selected items could not be processed due to error
Please enter a valid_number test