Sacroiliitis in children and adolescents with familial Mediterranean fever

Kaçmaz, Hülya; Aldemir, Esin; Tanatar, Ayşe; Karadağ, Şerife Gül; Çakan, Mustafa; Sönmez, Hafize Emine; Ayaz, Nuray Aktay

doi:10.1186/s42358-021-00188-2

Cited by 5 publications

(11 citation statements)

References 16 publications

(20 reference statements)

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“…Für diese Erkrankung weist das Kind ein ungewöhnliches Alter auf; für die – ebenfalls mit einer axialen Beteiligung einhergehende – Enthesitis-assoziierte Arthritis liegt das durchschnittliche Alter bei 13,2 ± 2,6. Die Altersverteilung der SI bei FMF lag in derselben Studie bei 7,2 ± 5,05 3 .…”

Section: Diskussionunclassified

“…Mit einer Prävalenz von 2,6 % entwickeln nur wenige Kinder mit FMF im Verlauf der Erkrankung eine SI 3 , 11 , 12 . Die häufigste Variante bei der molekulargenetischen Untersuchung von Patienten mit SI und FMF ist Met694Val im MEFV Gen, wie auch in unserem Fall.…”

Section: Diskussionunclassified

“…Im Verlauf de -Enthesitis-assoziierte Arthritis liegt das durchschnittliche Alter bei 13,2 ± 2,6. Die Altersverteilung der SI bei FMF lag in derselben Studie bei 7,2 ± 5,05 [3].…”

Section: Falldarstellungunclassified

“…Im Jahr 1987 wurde der erste Fall mit FMF und SI beschrieben [10]. In einer Studie über Patienten mit FMF und SI hatten 44,5 % eine Enthesitis, 36,5 % Fersenschmerzen und 17,4 % untere Rückenschmerzen [3].…”

Section: Falldarstellungunclassified

“…Dies gilt als eine eindeutig pathologische Variante und ist i. d. R. mit einem schweren Phänotyp und frühzeitigem Krankheitsausbruch verbunden [13]. Im Vergleich zu Patienten mit einer Spondyloarthropathie sind Patienten mit FMF und SI meistens HLA-B27 negativ [3].…”

Section: Falldarstellungunclassified

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Mehr als nur Wachstumsschmerzen?

Sabo¹,

Heinkele²,

Blankenburg³

et al. 2022

Arthritis und Rheuma

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Section: Diskussionunclassified

“…Im Verlauf de -Enthesitis-assoziierte Arthritis liegt das durchschnittliche Alter bei 13,2 ± 2,6. Die Altersverteilung der SI bei FMF lag in derselben Studie bei 7,2 ± 5,05 [3].…”

Section: Falldarstellungunclassified

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Mehr als nur Wachstumsschmerzen?

Sabo¹,

Heinkele²,

Blankenburg³

et al. 2022

Arthritis und Rheuma

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Childhood-Onset Sacroiliitis

Avar-Aydın¹,

Özçakar²,

Şahap

et al. 2023

J Clin Rheumatol

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Objective The aims of this study were to describe disease associations of magnetic resonance imaging (MRI)–confirmed and clinically symptomatic sacroiliitis in pediatric patients with rheumatic diseases and to examine the relationship between patient characteristics and MRI findings of the sacroiliac joint (SIJ). Methods Demographic and clinical data were extracted from the electronic medical records of the patients with sacroiliitis followed in the last 5 years. Active inflammatory and structural damage lesions of the SIJ-MRI were examined by the modified Spondyloarthritis Research Consortium of Canada scoring system, and correlation analysis of these results with clinical characteristics was evaluated. Results A total of 46 symptomatic patients were found to have MRI-proven sacroiliitis of 3 different etiologies: juvenile idiopathic arthritis (JIA) (n = 17), familial Mediterranean fever (FMF) (n = 14), and chronic nonbacterial osteomyelitis (CNO) (n = 8). Seven patients, FMF and JIA (n = 6) and FMF and CNO (n = 1), had a co-diagnosis that might cause sacroiliitis. Although inflammation scores and structural damage lesions did not statistically differ between the groups, capsulitis and enthesitis on the MRI were more frequently detected in the CNO group. There was a negative correlation between symptom onset and inflammation scores of bone marrow edema. Disease composite scores and acute phase reactants were correlated with MRI inflammation scores. Conclusions We demonstrated that JIA, FMF, and CNO were the major rheumatic causes of sacroiliitis in children originating from the Mediterranean region. Quantitative MRI scoring tools can be used to assess the inflammation and damage of the SIJ in rheumatic diseases, show discrepancies between them, and have an important correlation with various clinical and laboratory features.

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Sacroiliitis in familial Mediterranean fever: A rare joint involvement of the disease

Özçelik,

Çelikel,

Tekin

et al. 2024

J Paediatrics Child Health

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AimFamilial Mediterranean fever (FMF) is a monogenic autoinflammatory disease characterised by recurrent episodes of fever and polyserositis. Sacroiliac joint involvement is rare in FMF patients. The purpose of this study was to evaluate the demographic, clinical, laboratory and imaging findings of patients with FMF who developed sacroiliitis.MethodsThe files of paediatric patients aged 0–18 years who were followed up with a diagnosis of FMF were retrospectively reviewed. FMF patients with evidence of sacroiliitis on magnetic resonance imaging (MRI) were included in the study.ResultsAmong 1062 FMF patients, 22 (12 males; median age 8.5) (2.1%) of them were found to have sacroiliitis. FMF was diagnosed before sacroiliitis in nine (40.9%) patients and after in 13 (59.1%) patients. The most common symptom in patients with sacroiliitis was low back pain (n = 21, 95.5%). In MEFV gene analysis, M694V was found in 16 (72.7%) patients and was the most common mutation. MRI showed evidence of sacroiliitis in all patients. All patients were using colchicine. Patients with FMF‐associated sacroiliitis, remission was achieved with non‐steroidal anti‐inflammatory drugs in 12 (54.5%), conventional disease‐modifying antirheumatic drugs in six (27.3%) and tumour necrosis factor inhibitor treatment in four (31.8%). Four (31.8%) patients experienced sacroiliitis when colchicine incompatible and four (31.8%) patients experienced sacroiliitis while using biologic agents for colchicine‐resistant FMF.ConclusionsFMF‐associated sacroiliitis should be considered especially in patients with M694V mutation if they have symptoms such as low back pain. Colchicine‐resistant FMF patients should be evaluated for sacroiliitis symptoms at each visit.

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Sacroiliitis in children and adolescents with familial Mediterranean fever

Cited by 5 publications

References 16 publications

Mehr als nur Wachstumsschmerzen?

Mehr als nur Wachstumsschmerzen?

Childhood-Onset Sacroiliitis

Sacroiliitis in familial Mediterranean fever: A rare joint involvement of the disease

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