Performance of Tel-Hashomer, Livneh, pediatric and new Eurofever/PRINTO classification criteria for familial Mediterranean fever in a referral center

Rheumatol Int. 2020 Jan;40(1):21-27. doi: 10.1007/s00296-019-04463-w. Epub 2019 Oct 23.

Abstract

Until now, the diagnosis of familial Mediterranean fever (FMF) was based on validated subsets of clinical criteria, but recently new Eurofever/PRINTO classification criteria concerning genetic analyses were proposed. The study aimed to compare the performances of three validated diagnostic criteria (Tel-Hashomer, Livneh, pediatric criteria) and new Eurofever/PRINTO classification criteria. The medical charts of study and control groups were reviewed retrospectively. Patients were evaluated for three diagnostic criteria and new Eurofever/PRINTO classification criteria. Control group consists of patients with other autoinflammatory diseases. A total of 1291 patients were classified into three groups according to their mutations: group 1: 447 patients with homozygous mutations; group 2: 429 patients with compound heterozygous mutations; and group 3: 415 patients with one heterozygous mutation. Similar diagnostic utility was found according to Livneh criteria between groups. But, proportion of patients fulfilling Tel-Hashomer and pediatric criteria was higher in groups 1 and 2. According to Eurofever/PRINTO criteria, 98.2% of patients with homozygous mutations, 94.2% of patients with compound heterozygous mutations and 80.2% of patients with heterozygous mutations were classified as FMF. In control group, 99.2% of them fulfilled the Livneh criteria, 66.9% met the pediatric criteria and 0.8% satisfied the Tel-Hashomer criteria, while none of control patients met the Eurofever/PRINTO classification criteria. Performances of three validated diagnostic criteria and new Eurofever/PRINTO classification criteria for FMF were similar and provide high utility in diagnosing/classifying patients with homozygous and compound heterozygous mutations. However, both Eurofever/PRINTO classification criteria and Tel-Hashomer criteria had significantly lower performance in heterozygous patients.

Keywords: Autoinflammation; Classification criteria; Diagnostic criteria; Familial Mediterranean fever; MEFV.

MeSH terms

  • Adolescent
  • Arthritis / physiopathology
  • Case-Control Studies
  • Chest Pain / physiopathology
  • Child
  • Child, Preschool
  • Colchicine / therapeutic use
  • Consanguinity
  • Drug Resistance
  • Exons / genetics
  • Familial Mediterranean Fever / classification
  • Familial Mediterranean Fever / diagnosis*
  • Familial Mediterranean Fever / genetics
  • Familial Mediterranean Fever / physiopathology
  • Female
  • Hereditary Autoinflammatory Diseases / classification
  • Hereditary Autoinflammatory Diseases / diagnosis
  • Heterozygote*
  • Homozygote*
  • Humans
  • Male
  • Mutation
  • Pyrin / genetics*
  • Reproducibility of Results
  • Retrospective Studies
  • Severity of Illness Index
  • Tubulin Modulators / therapeutic use

Substances

  • MEFV protein, human
  • Pyrin
  • Tubulin Modulators
  • Colchicine