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Concurrent Joint Hypermobility Syndrome and Spondyloarthropathy

A New Spondyloarthropathy Subgroup With a Less Severe Disease Phenotype?

Shur, Natalie F. MBChB, BMedSci (Hons), MRes, MRCP(UK)*; Shur, Joshua D. MSci (Hons), MBBS, FRCR; Rennie, Winston J. MB, MSc, FRCR, MFSEM; Moorthy, Arumugam MB, MRCP (UK) Rheum, FRCP Edin

doi: 10.1097/RHU.0000000000000959
Original Article: PDF Only

Background The coexistence of joint hypermobility syndrome (JHS) and spondyloarthropathy (SpA) presents a challenging clinical conundrum due to the contradictory clinical signs that may be present. Classic features such as restricted spinal movement or early morning back stiffness may not be present. Timely diagnosis and appropriate management of these patients are difficult as they tend to have lower scores on validated objective measures.

Methods We performed a medical records review study to identify patients with both JHS and SpA who had presented to the Leicester Spondyloarthropathy clinic. Patients were diagnosed with axial SpA if they met the Assessment of SpondyloArthritis international Society classification criteria.1 Their imaging was reviewed by a consultant musculoskeletal radiologist.

Results Four cases were identified from the patient database (female; average age, 37.5 years). All patients presented with lower back pain or sacroiliac joint pain but preserved spinal movement with a negative Schober's test. Two had a history of symptoms for more than 10 years. All had a Beighton score of greater than 6. Three of the patients were HLA positive, and 3 had a positive family history. All patients thus far have had their symptoms adequately controlled on nonsteroidal anti-inflammatory drugs and physiotherapy.

Conclusions The coexistence of JHS and SpA is rare but important to recognize. These patients are difficult to diagnose as they may present late because of preserved spinal movements. It is unclear whether the preserved flexibility masks the true extent of disease or whether clinically they represent a less severe disease phenotype.

From the *Department of Rheumatology, University Hospitals of Leicester NHS Trust, Leicester Royal Infirmary, Leicester, United Kingdom;

Joint Department of Medical Imaging, University Health Network, Toronto, Canada;

Department of Radiology, University Hospitals of Leicester NHS Trust, Leicester Royal Infirmary; and

§Department of Medical Education, University of Leicester, Leicester, United Kingdom.

This study was classified as service evaluation, and therefore formal National Research Ethics Approval was unnecessary for this study.

Consent to publish from participants has been sought to use their anonymized information and images for academic publication.

Data sharing is not applicable to this article as no data sets were generated or analyzed during the current study.

The authors declare no conflict of interest.

Author Contributions: N.F.S. was responsible for data collection and preparing the manuscript draft. J.D.S. contributed to imaging interpretation and manuscript preparation. W.J.R. contributed to imaging interpretation and manuscript preparation. A.M. contributed to the conception of the study, data collection, and manuscript preparation. All authors approved the final manuscript.

Correspondence: Natalie F. Shur, MBChB, BMedSci (Hons), MRes, MRCP(UK), Department of Rheumatology, University Hospitals of Leicester NHS Trust, Leicester Royal Infirmary, Infirmary Square, United Kingdom LE1 5WW. E-mail:

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