Reumatología Clínica Reumatología Clínica
Reumatol Clin 2017;13:118-9 - Vol. 13 Núm.2 DOI: 10.1016/j.reuma.2015.12.007
Images in Clinical Rheumatology
Structural damage to the hip in systemic juvenile idiopathic arthritis: A case of regression with Anakinra
Daño estructural de la cadera en la artritis idiopática juvenil sistémica: un caso de regresión con Anakinra
Francisca Aguiara,, , Iva Britoa,b
a Rheumatology Department, Centro Hospitalar São João, Porto, Portugal
b Faculty of Medicine of Porto University, Porto, Portugal
Recibido 24 octubre 2015, Aceptado 30 diciembre 2015
Clinical case

A 15 years old caucasian girl was diagnosed with systemic juvenile idiopathic arthritis (sJIA) by the age of four and was initially treated with nonsteroidal anti-inflammatory drugs and oral corticosteroids (prednisolone 1mg/kg/day). Although there was some clinical improvement, the disease progressed with 1–2 articular and systemic exacerbations a year and, in the beginning of the year 2007, when she was 7 years old, she started weekly methotrexate (10mg/m2).

Despite methotrexate treatment escalation (up to 25mg/m2/week subcutaneously) the patient had persistent disease activity and developed severe left coxitis (Fig. 1) in the beginning of 2009, which did not respond to ultrasound guided joint injection with triamcinolone hexacetonide. Due to the persistence of arthritis and elevated inflammatory markers she was started on Anakinra (1mg/kg/day) in July 2009.

Fig. 1.

Anteroposterior (Panel A) and frog-leg lateral (Panel B) radiographs of the pelvis showing left hip joint space narrowing, subchondral cysts of the left femoral head and acetabular sclerosis (*).

Since then there has been sustained improvement with resolution of clinical symptoms, including complete imagiological regression of coxitis (Fig. 2), which allowed discontinuation of methotrexate and corticosteroids. The patient is in clinical remission on medication with Anakinra 100mg/day (2–2.5mg/kg/day) since 2011.

Fig. 2.

Anteroposterior (Panel A) and frog-leg lateral (Panel B) radiographs of the pelvis showing normal left hip joint space with discrete acetabular sclerosis.

Discussion

sJIA is an autoinflammatory rheumathologic disease that accounts for 5–10% of all patients classified as JIA.1 Hip involvement in sJIA is relatively common and is a cause of significant functional impairment and a marker of poor prognosis.2,3 A significant number of patients with sJIA has persistent disease despite the treatments used.

Anakinra is a recombinant form of human IL-1 receptor antagonist (IL-1Ra) that is recommended as first line disease-modifying therapy in sJIA selected patients and in refractory disease.4 In this case, the authors report the therapeutic success with Anakinra in a patient with refractory systemic and articular disease, emphasizing the regression of structural damage of the hip joint, which is rarely reported in these cases.

Conflict of interest

The authors declare that there are no conflicts of interest.

Ethical disclosuresProtection of human and animal subjects

The authors declare that no experiments were performed on humans or animals for this study.

Confidentiality of data

The authors declare that no patient data appear in this article.

Right to privacy and informed consent

The authors declare that no patient data appears in this article.

References
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P.J. Gowdie,S.M. Tse
Juvenile idiopathic arthritis
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W.P. Bekkering,R. ten Cate,L.W. Suijlekom-Smit,D. Mul,E.A. van der Velde,C.H. van den Ende
The relationship between impairments in joint function and disabilities in independent function in children with systemic juvenile idiopathic arthritis
J Rheumatol, 28 (2001), pp. 1099-1105
3
C. Modesto,P. Woo,J. García-Consuegra,R. Merino,M. Gracía-Granero,C. Arnal
Systemic onset juvenile chronic arthritis, polyarticular pattern and hip involvement as markers for a bad prognosis
Clin Exp Rheumatol, 19 (2001), pp. 211-217
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S. Ringold,P.F. Weiss,T. Beukelman,E.M. Dewitt,N.T. IIowite,Y. Kimura
Update of the 2011 American College of Rheumatology recommendations for the treatment of juvenile idiopathic arthritis: recommendations for the medical therapy of children with systemic juvenile idiopathic arthritis and tuberculosis screening among children receiving biologic medications
Arthritis Rheum, 65 (2013), pp. 2499-2512 http://dx.doi.org/10.1002/art.38092
Corresponding author.
Copyright © 2016. Elsevier Espa??a, S.L.U. and Sociedad Espa??ola de Reumatolog??a y Colegio Mexicano de Reumatolog??a
Reumatol Clin 2017;13:118-9 - Vol. 13 Núm.2 DOI: 10.1016/j.reuma.2015.12.007