The expanded spectrum of arthritis in children with familial Mediterranean fever

Avar-Aydın, Pınar Özge; Özçakar, Z. Bı̇rsı̇n; Aydın, Fatma; Karakas, Hatice Dilara; Çakar, Nilgün; Yalçınkaya, Fatoş

doi:10.1007/s10067-022-06082-6

Cited by 12 publications

(21 citation statements)

References 23 publications

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“…31 Sacroiliitis affects approximately 5% of the patients diagnosed with FMF. 7,32,33 Inflammatory back pain, radiographic sacroiliitis without vertebral involvement, and HLA-B27 negativity are the main characteristic features of FMF-related sacroiliitis. However, it was shown that FMF-associated autoinflammation exaggerate the tendency toward spondyloarthritis-associated disorders, and there was a strong association between FMF and ankylosing spondylitis.…”

Section: Discussionmentioning

confidence: 99%

“…A previous study in CNO showed that the presence of the MEFV gene mutations increased the risk of sacroiliitis. 26 Thus, the presence of severe MEFV gene mutations emerges as a predisposing and, possibly, a modifying factor for sacroiliitis in these types of diseases, particularly for patients originating from the Mediterranean region.…”

Section: Discussionmentioning

confidence: 99%

“…A recent report from the Childhood Arthritis and Rheumatology Research Alliance registry showed that 28% of the patients with either enthesitis-related arthritis or juvenile psoriatic arthritis had sacroiliitis, whereas only 38% of them were symptomatic 31 . Sacroiliitis affects approximately 5% of the patients diagnosed with FMF 7,32,33 . Inflammatory back pain, radiographic sacroiliitis without vertebral involvement, and HLA-B27 negativity are the main characteristic features of FMF-related sacroiliitis.…”

Section: Discussionmentioning

confidence: 99%

“…The regional and whole-body MRIs were used at the diagnosis of CNO, and yearly screening with whole-body MRI was performed at our center. The MEFV gene mutations were examined at the diagnosis of CNO, as an associated disease of FMF or MEFV gene carriage 26 . The clinical scoring system by Jansson et al 27 was calculated for this group of patients at the diagnosis of sacroiliitis.…”

Section: Methodsmentioning

confidence: 99%

“…The MEFV gene mutations were examined at the diagnosis of CNO, as an associated disease of FMF or MEFV gene carriage. 26 The clinical scoring system by Jansson et al 27 was calculated for this group of patients at the diagnosis of sacroiliitis.…”

Section: Patients With Chronic Nonbacterial Osteomyelitismentioning

confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Section: Patients With Chronic Nonbacterial Osteomyelitismentioning

confidence: 99%

See 3 more Smart Citations

Childhood-Onset Sacroiliitis

Avar-Aydın¹,

Özçakar²,

Şahap

et al. 2023

J Clin Rheumatol

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Spondyloarthritis in familial Mediterranean fever: a cohort study

Kasman

Duruöz

2022

Rheumatol Int

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Not easy-peasy to diagnose: familial Mediterranean fever unaccompanied by fever

et al. 2023

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PurposeClassical attacks of familial Mediterranean fever (FMF) are often accompanied by fever, but some of the patients have attacks without fever. This study aimed to compare the characteristics of FMF patients with and without fever during their attacks and draw attention to the different clinical presentations of FMF in children. MethodsMedical les of patients aged 0-18 years who were followed up with the diagnosis of FMF in two reference pediatric rheumatology centers were reviewed retrospectively. The patients were divided into two groups: Children who had had no fever in any of their attacks were assigned as group 1, and those who had fever during their attacks were classi ed as group 2. ResultsOut of 2003 patients evaluated, 191 (9.53%) patients had attacks not accompanied by fever and their median age at onset of symptoms (7.0 vs. 4.0 years, p < 0.001) and the median age at diagnosis (8.6 vs. 6.0 years, p < 0.001) were signi cantly higher, however group 2 had delay in diagnosis. The annual number of attacks and abdominal attacks were more common in group 2, arthritis, arthralgia, erysipelaslike rash, exercise-induced leg pain, and myalgia were more common in group 1. ConclusionThe data from the assessment of children with FMF attacks not accompanied with fever were presented for the rst time. Children with late age onset of FMF and dominance of musculoskeletal features may display attacks not accompanied with fever.

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The expanded spectrum of arthritis in children with familial Mediterranean fever

Cited by 12 publications

References 23 publications

Childhood-Onset Sacroiliitis

Childhood-Onset Sacroiliitis

Spondyloarthritis in familial Mediterranean fever: a cohort study

Not easy-peasy to diagnose: familial Mediterranean fever unaccompanied by fever

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