Skip to main content

Advertisement

Log in

Growth Parameters of Turkish Children With an Autoinflammatory Disease Before and After Canakinumab Treatment

  • Research Paper
  • Published:
Indian Pediatrics Aims and scope Submit manuscript

Abstract

Objective

To evaluate the effect of canakinumab on growth parameters of patients with autoinflammatory diseases.

Methods

This retrospective study included Colchicine resistant familial Mediterranean fever (FMF), Mevalonate kinase deficiency (MKD), Tumor necrosis factor receptor-associated periodic fever syndrome (TRAPS), Deficiency of adenosine deaminase 2 (DADA2) patients treated with canakinumab for at least six consecutive months.

Results

Eleven patients with FMF, 9 with MKD, 3 with TRAPS, and 1 with DADA2 were included. The median age (range) at diagnosis and drug initiation was 6.06 (1.45–16.06) years and 9.72 (1.82–19.11) years, respectively. The mean weight, height, and BMI SD scores significantly increased after canakinumab. There were significant improvements in laboratory parameters and disease activities. However, growth parameters after the drug did not differ according to gender, the duration of diagnostic delay, and age at the diagnosis.

Conclusion

Canakinumab seems to have a positive effect on growth in patients with autoinflammatory diseases by controlling disease activity and inflammation.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. Oda H, Kastner DL. Genomics, Biology, and Human Illness: Advances in the monogenic autoinflammatory diseases. Rheum Dis Clin North Am. 2017;43:327–45.

    Article  Google Scholar 

  2. Kisla Ekinci RM, Balci S, Bisgin A, Hershfield M, Atmis B, Dogruel D, et al. Renal Amyloidosis in Deficiency of Adenosine Deaminase 2: Successful Experience with Canakinumab. Pediatrics. 2018;142:e20180948.

    Article  Google Scholar 

  3. van der Burgh R, Ter Haar NM, Boes ML, Frenkel J. Mevalonate kinase deficiency, a metabolic autoinflammatory disease. Clin Immunol. 2013;147:197–206.

    Article  CAS  Google Scholar 

  4. ter Haar NM, Oswald M, Jeyaratnam J, Anton J, Barron KS, Brogan PA, et al. Recommendations for the management of autoinflammatory diseases. Ann Rheum Dis. 2015;74:1636–44.

    Article  CAS  Google Scholar 

  5. de Benedetti F, Gattorno M, Anton J, Ben-Chetrit E, Frenkel J, Hoffman HM, et al. Canakinumab for the treatment of autoinflammatory recurrent fever syndromes. N Engl J Med. 2018;378:1908–19.

    Article  CAS  Google Scholar 

  6. Ozen S, Kone-Paut I, Gül A. Colchicine resistance and intolerance in familial mediterranean fever: Definition, causes, and alternative treatments. Semin Arthritis Rheum. 2017;47:115–20.

    Article  CAS  Google Scholar 

  7. Balci S, Ekinci RMK, Bayazit AK, Melek E, Dogruel D, Altintas DU, et al. Juvenile systemic lupus erythematosus: A single-center experience from southern Turkey. Clin Rheumatol. 2019;38:1459–68.

    Article  Google Scholar 

  8. Kisla Ekinci RM, Balci S, Akay E, Dogruel D, Altintas DU, Yilmaz M. Disease severity and genotype affect physical growth in children with familial mediterranean fever. Arch Rheumatol. 2019;34:i–vi. [Epub ahead of print]

    Google Scholar 

  9. Gutierrez-Suarez R, Ruperto N, Gastaldi R, Pistorio A, Felici E, Burgos-Vargas R, et al. A proposal for a pediatric version of the systemic lupus international collaborating clinics/american college of rheumatology damage index based on the analysis of 1,015 patients with juvenile-onset systemic lupus erythematosus. Arthritis Rheum. 2006;54:2989–96.

    Article  Google Scholar 

  10. Zung A, Barash G, Zadik Z, Barash J. Familial mediterranean fever and growth: Effect of disease severity and colchicine treatment. J Pediatr Endocrinol Metab. 2006;19:155–60.

    Article  Google Scholar 

  11. Yoldas TC, Cakar N, Basaran O, Acar B, Uncu N, Caycy FS. The effect of colchicine and disease severity on physical growth in children with familial mediterranean fever. Clin Rheumatol. 2016;35:1603–07.

    Article  Google Scholar 

  12. Livneh A, Langevitz P, Zemer D, Zaks N, Kees S, Lidar T, et al. Criteria for the diagnosis of familial mediterranean fever. Arthritis Rheum. 1997;40:1879–85.

    Article  CAS  Google Scholar 

  13. Ozen S, Demirkaya E, Duzova A, Erdogan O, Erken E, Gul A, et al. FMF50: A score for assessing outcome in familial mediterranean fever. Ann Rheum Dis. 2014;73:897–901.

    Article  Google Scholar 

  14. Piram M, Koné-Paut I, Lachmann HJ, Frenkel J, Ozen S, Kuemmerle-Deschner J, et al. Validation of the autoinflammatory diseases activity index (AIDAI) for hereditary recurrent fever syndromes. Ann Rheum Dis. 2014;73:2168–73.

    Article  Google Scholar 

  15. Neyzi O, Gunoz A, Furman A, Bundak R, Gökçay G, Darendeliler F, et al. Weight, height, head circumference and body mass index references for Turkish children. Cocuk Sagligi ve Hastaliklari Derg. 2008;51:1–14. [article in Turkish]

    Google Scholar 

  16. Kisla Ekinci RM, Balci S, Dogruel D, Altintas DU, Yilmaz M. Canakinumab in children with familial mediterranean fever: A single-center, retrospective analysis. Paediatr Drugs. 2019;21:389–95.

    Article  Google Scholar 

  17. Balci S, Kisla Ekinci RM, Melek E, Atmyp B, Bisgin A, Yilmaz M. Phenotypic variability in two patients with tumor necrosis factor receptor associated periodic fever syndrome emphasizes a rare manifestation: Immunoglobulin a nephropathy. Eur J Med Genet. 2019;Oct 3:103780. [Epub ahead of print]

    Google Scholar 

  18. Basaran O, Uncu N, Celikel BA, Taktak A, Gur G, Cakar N. Interleukin-1 targeting treatment in familial Mediterranean fever: an experience of pediatric patients. Mod Rheumatol. 2015;25:621–4.

    Article  CAS  Google Scholar 

Download references

Funding

Funding: None

Author information

Authors and Affiliations

Authors

Contributions

Contributors: SB, MY: conceptualized and designed the study, drafted the initial manuscript, and reviewed and revised the manuscript; RMKE,DD: collected data, carried out the initial analyses, and reviewed and revised the manuscript; DUA, DD: designed the data collection instruments, and coordinated and supervised data collection, and critically reviewed the manuscript. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.

Corresponding author

Correspondence to Sibel Balci.

Ethics declarations

Competing interest: None stated.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Balci, S., Ekinci, R.M.K., Dogruel, D. et al. Growth Parameters of Turkish Children With an Autoinflammatory Disease Before and After Canakinumab Treatment. Indian Pediatr 57, 637–640 (2020). https://doi.org/10.1007/s13312-020-1892-9

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s13312-020-1892-9

Keywords

Navigation