Effect of Colchicine-Resistant Familial Mediterranean Fever on Growth Parameters

Sözeri, Betül; Mır, Sevgı; Berdelı, Afig

doi:10.5606/tjr.2011.001

Cited by 2 publications

(4 citation statements)

References 29 publications

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“…Meanwhile, the growth retardation was attributed to sustained inflammation by the authors. 6 Because colchicine-resistant patients were successfully treated with concomitant canakinumab in our study, we did not investigate colchicine resistance as a parameter on growth.…”

Section: Discussionmentioning

confidence: 99%

“…Moreover, several studies including limited numbers of patients addressed the question whether physical growth is affected by genotype and disease severity or not. [5][6][7][8][9][10] However, all these studies utilized an ancient Tel Hashomer severity scoring system, which was not validated in the pediatric population. 11 Thus, in this study, we aimed to analyze the growth parameters in children with FMF according to disease characteristics including genotype and disease severity by a recently validated tool in relatively more patients.…”

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confidence: 99%

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Disease Severity and Genotype Affect Physical Growth in Children With Familial Mediterranean Fever

et al. 2019

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Objectives: This study aims to analyze the growth parameters in children with familial Mediterranean fever (FMF) according to disease characteristics including genotype and disease severity by a recently validated tool in relatively more patients. Patients and methods: This retrospective study included 126 patients with FMF (70 males, 56 females; mean age 7.3±3.6 years; range, 4.1 to 18 years). MEditerranean FeVer (MEFV) gene analysis was performed with a molecular diagnostics tool by using a next-generation sequencing platform. Disease severity was determined for the first visit by the validated tool in children, international severity scoring system for FMF. Growth parameters including weight and height were investigated after standard deviation (SD) scores were calculated by anthropometric references in Turkish children. Results: Median follow-up duration was 74.7 months (range, 7.5 to 169 months). Ninety-three patients (73.8%) had at least one M694V mutation in MEFV gene. Six patients (4.8%) had severe disease, 58 (46%) had intermediate severity, and 62 (49.2%) had mild disease. Mean height SD score was significantly lower at last visit than before colchicine treatment. Initial and last height and weight SD scores were lower in patients with at least one M694V mutation than those without. However, the difference was statistically significant for only initial height SD score. We also found statistically significant lower initial height, final height, and weight SD scores in patients with intermediate severity-severe disease activity than mild disease. Conclusion: We advise physicians to score disease severity prospectively and pay attention to patients with intermediate severity-severe disease to avoid growth disturbances.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

Disease Severity and Genotype Affect Physical Growth in Children With Familial Mediterranean Fever

et al. 2019

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“…It has been shown that FMF patients catch up to their growth with an effective colchicine treatment [24–26]. …”

Section: Discussion and Evaluationmentioning

confidence: 99%

“…Pro-inflammatory cytokines may modulate growth patterns in children with inflammatory diseases through both systemic and local effects of the GH/IGF-1 axes [ 23 ]. It has been shown that FMF patients catch up to their growth with an effective colchicine treatment [ 24 – 26 ].…”

Section: Discussion and Evaluationmentioning

confidence: 99%

The experience of canakinumab in renal amyloidosis secondary to Familial Mediterranean fever

et al. 2016

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IntroductionFamilial Mediterranean fever (FMF) is an autosomal recessive disease characterized by self-limited recurrent attacks of fever and serositis. Patients may develop renal amyloidosis. Colchicine prevents attacks and renal amyloidosis. Five to 10 % of the patients with FMF are resistant or intolerant to colchicine.Case descriptionHerein, we reported our experience with clinical-laboratory features and treatment responses of a pediatric FMF patient with amyloidosis treated with canakinumab. We observed a significant decrease in proteinuria and increase growth in the patient.Discussion and evaluationThe most serious complication of FMF is the development of AA type amyloidosis which is characterized by proteinuria. Colchicine is the prototype drug that decreases production of amyloidogenic precursor protein. Occasionally, colchicine inadequate patient is observed, as in our case. Canakinumab is a human anti-IL-1β monoclonal antibody. Previously, canakinumab efficacy were shown in a limited number of studies.ConclusionsOur data, though limited to only one patient, emphasize that therapeutic intervention with canakinumab seems to be improve kidney function in colchicine-resistant FMF with renal amyloidosis.

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Effect of Colchicine-Resistant Familial Mediterranean Fever on Growth Parameters

Cited by 2 publications

References 29 publications

Disease Severity and Genotype Affect Physical Growth in Children With Familial Mediterranean Fever

Disease Severity and Genotype Affect Physical Growth in Children With Familial Mediterranean Fever

The experience of canakinumab in renal amyloidosis secondary to Familial Mediterranean fever

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