Cimaz, Rolando

Hofer, Michaël F.

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EditorialIs cervical spine involvement in juvenile polyarthritisunder-recognized?Detecting cervical abnormalities earlyThis editorial refers to Radiological cervical spineinvolvement in young adults with polyarticular juvenileidiopathic arthritis, by M Elhai, J Wipff, R Bazeli et al.,doi:10.1093/rheumatology/kes054, on pages 267275.Elhai et al.’s [1] study evaluates the frequency of radio-logical cervical spine involvement in young adults withpolyarticular JIA (pJIA) compared with RA patients. Theauthors show that about two-thirds of patients in bothgroups had radiographic abnormalities in the cervicalspine, including anterior atlantoaxial subluxation, erosionof the odontoid process, C1C2 arthritis and apophysealjoint arthritis. Lesions were found even in patients withoutclinical symptoms and their presence correlated positivelywith the severity of disease.The similar prevalence of structural cervical spinelesions in young adults with pJIA and RA highlights theimportance of this complication for the long-term follow-upof juvenile polyarthritis. This aspect of pJIA outcome needsfurther study because literature on the topic is relativelyscarce, in contrast to the abundant number of papers oncervical problems in RA. Another striking point of thisstudy is the high percentage of asymptomatic patientspresenting with radiological cervical lesions. Only 42% ofpatients without neck complaints or findings on examin-ation had a normal cervical scan; accordingly, the authorsraise the question of the frequency of systematic cervicalspine radiological controls during follow-up in pJIA.According to the ILAR classification [2], polyarthritis pa-tients with onset before the age of 16 years are dividedinto two different categories, depending on the result ofthe RF [2]. RF-negative polyarthritis corresponds to a het-erogeneous group of patients with symmetrical or asym-metrical joint involvement, presence or absence of ANAs,and includes 1012% of the JIA cases [3, 4]. TheRF-positive polyarticular category is considered to be anearly form of RA and represents 23% of JIA patients [3].The long-term prognosis of pJIA patients can vary butoutcome studies with a follow-up of at least 10 yearshave shown that active arthritis is still present in3967% of patients [5]. In particular, the latter studyshowed that children who did not present with a periodof disease inactivity in the 5 years of disease onset hadmore long-term joint damage. In another study, patientswith RF-positive polyarthritis or early evidence of jointdamage had a higher risk of persistent active arthritisduring follow-up [6].In Elhai et al.’s [1] study, RF-positive patients repre-sented 60% of the cohort: more than 10 times theexpected frequency in normal JIA distribution, indicatingan over-representation of this category. The high propor-tion of RF-positive patients is likely to have influenced theresults of the study and could explain similar cervicalspine involvement in JIA and RA patients. Interestingly,the frequency of a positive RF was similar in patientswith or without cervical involvement; these data suggestthat RF-negative patients had a similarly high rate of cer-vical spine abnormalities, which may be explained by asignificantly longer duration of disease course in compari-son with that in RF-positive patients (17.3 vs 9.8 years).Imaging evaluation of children with polyarthritis is usu-ally indicated according to clinical findings, mainly inpatients with joint inflammation that persists through treat-ment. Plain radiography is the standard imaging techniquefor monitoring abnormalities in peripheral joints, but thereare no recommendations for timing intervals during thefollow-up of JIA patients [7]. Cervical spine involvementhas been described in different forms of JIA, includingpolyarthritis, systemic arthritis and enthesitis-related arth-ritis. In 2002, Laiho et al. [8] reported cervical spine struc-tural abnormalities in 62% of 159 JIA patients, whounderwent radiographic examination because of necksymptoms or as a part of a pre-operative routine. Morethan 80% of the patients suffered from polyarthritis, and25% of them were RF positive. Elhai et al.’s [1] study hadsimilar findings but the patient cohort was quite different,since two-thirds of the patients were treated with abiotherapy and half of them did not show any neck symp-toms or signs.Many children and adolescents with rheumatic diseasesare treated by paediatricians who are specialists inrheumatology in paediatric settings; care for young pa-tients will be adjusted to factors related to their age, likegrowth, family, and school. The paediatrician will transferthe patient to an adult clinic at the end of their adoles-cence and will not see their long-term outcome. Transitionclinics have recently been developed to offer a smoothtransfer to adult care, which is essential for continuity ofcare. Transition clinics have another major advantage inthat they highlight the outcome of JIA patients during earlyadulthood. The information gathered from young adult JIApatients is useful in improving the care provided to chil-dren with JIA. Elhai et al.’s [1] study shows that cervicalinvolvement in pJIA is common and can occur even in theabsence of symptoms. This finding underlines the import-ance for clinicians of detecting minor cervical symptomsin JIA patients. The bias of the study could be that sub-jects with mild disease, which can go into long-term! The Author 2012. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: journals.permissions@oup.comRHEUMATOLOGYRheumatology 2013;52:221222doi:10.1093/rheumatology/kes273Advance Access publication 25 October 2012EDITORIALremission, are less likely to seek medical care. In light ofthis, should pJIA patients with only minor cervical symp-toms still undergo regular cervical imaging? Prospectivetrials need to be done before we can make clearrecommendations.In Elhai et al.’s [1] study, mean disease duration beforethe introduction of the biological agents was 8.2 years,since biotherapies were not licensed for JIA at diseaseonset in the majority of the patients studied. These newtreatments have improved the control of joint inflammationand we expect that they will decrease the high incidenceof cervical spine involvement in future. Finally, even if plainX-rays are easy to obtain, newer imaging techniques,such as MRI with contrast, could be useful as they aremore sensitive in detecting joint inflammation (Fig. 1)and pre-destructive changes [7]. They are now themethod of choice in detecting cervical spine involvementin patients with pJIA. Early diagnosis of cervical abnorm-alities would be important in selecting pJIA patients to bemore aggressively treated and for whom protective meas-ures should be implemented.Disclosure statement: The authors have declared no con-flicts of interest.Michae¨l F. Hofer1,2 and Rolando Cimaz31Paediatric Rheumatology of Western Switzerland, PaediatricDepartment, CHUV, University Hospital, Lausanne,2Paediatric Department, HUG, Geneva, Switzerland and3AOU Meyer, Firenze, Italy.Accepted 6 September 2012Correspondence to: Michae¨l F. Hofer, PaediatricRheumatology, Department of Paediatrics, BH11-CHUV,CH-1011 Lausanne, Switzerland.E-mail: michael.hofer@chuv.chReferences1 Elhai M, Wipff J, Bazeli R et al. Radiological cervicalspine involvement in young adults with polyarticularjuvenile idiopathic arthritis. Rheumatology 2012, AdvanceAccess published 17 April 2012 doi:10.1093/rheumatology/kes054.2 Petty RE, Southwood TR. International League ofAssociations for Rheumatology classification of juvenileidiopathic arthritis: second revision, Edmonton, 2001.Rheumatology 2004;31:2.3 Modesto C, Anto´n J, Rodriguez B et al. Incidence andprevalence of juvenile idiopathic arthritis in Catalonia(Spain). Scand J Rheumatol 2010;39:4729.4 Ravelli A, Varnier GC, Oliveira S et al. Antinuclearantibodypositive patients should be grouped as aseparate category in the classification of juvenileidiopathic arthritis. Arthritis Rheum 2011;63:26775.5 Magnani A, Pistorio A, Magni-Manzoni S et al.Achievement of a state of inactive disease at least once inthe first 5 years predicts better outcome of patients withpolyarticular juvenile idiopathic arthritis. J Rheumatol2009;36:62834.6 Ringold S, Kristy D, Seidel K, Koepsell T, Wallace CA.Inactive disease in polyarticular juvenile idiopathic arthritis:current patterns and associations. Rheumatology 2009;48:9727.7 Breton S, Jousse-Joulin S, Finel E et al. Imagingapproaches for evaluating peripheral joint abnormalities injuvenile idiopathic arthritis. Semin Arthritis Rheum 2012;41:698711.8 Laiho K, Savolainen A, Kautiainen H, Kekki P, Kauppi M.The cervical spine in juvenile chronic arthritis. Spine J2002;2:8994.FIG. 1 MRI with cervical involvement.(a) Without contrast (T1-weighted) and (b) with contrast (T2 fat-saturation) of a 15-year-old girl with pJIA: periodontoideffusion, vertebral bone abnormalities and articular contrast enhancement.222 www.rheumatology.oxfordjournals.orgEditorial

Is cervical spine involvement in juvenile polyarthritis under-recognized?

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