Neurotoxicity of cyclosporine A in children with steroid-resistant nephrotic syndrome: is cytotoxic edema really an unfavorable predictor of permanent neurological damage?

Summary

Background

Cyclosporine A-associated neurotoxicity has been reported mainly after organ transplantation. Only a small number of children with steroid-resistant nephrotic syndrome and cyclosporine A-associated neurotoxicity have been reported.

Patients

We report three children, aged 4, 11, and 15, with steroid-resistant nephrotic syndrome and cyclosporine A-associated neurotoxicity. In two of the patients, primary diagnosis was idiopathic nephrotic syndrome, and in one it was IgA nephropathy. Magnetic resonance with diffusion-weighted imaging, combined with quantification of apparent diffusion coefficient values, showed lesions caused by cytotoxic edema indicating irreversible brain damage. Nonetheless, the patients fully recovered clinically and radiologically after prompt discontinuation of cyclosporine A.

Conclusions

Neurotoxic effects should be suspected in any child with nephrotic syndrome treated with cyclosporine A in whom sudden neurological symptoms occur. Cytotoxic edema is a rare finding in pediatric patients. However, even in such cases with seemingly irreversible brain damage, full recovery without permanent neurological sequels is possible with prompt cyclosporine A discontinuation and supportive therapy.

This is a preview of subscription content, access via your institution.

Fig. 1

References

  1. 1.

    Gijtenbeek JM, van den Bent MJ, Vecht CJ. Cyclosporine neurotoxicity: a review. J Neurol. 1999;246:339–46.

    CAS  Article  PubMed  Google Scholar 

  2. 2.

    Lombel RM, Hodson EM, Gipson DS. Treatment of steroid-resistant nephrotic syndrome in children: new guidelines from KDIGO. Pediatr Nephrol. 2013;28:409–14.

    Article  PubMed  Google Scholar 

  3. 3.

    Jeong MH, Lee JH, Yum MS, Ko TS, Park SY. A case of posterior reversible encephalopathy syndrome during cyclosporine therapy in a child with steroid resistant nephrotic syndrome. J Korean Soc Pediatr Nephrol. 2007;11:92–9.

    Article  Google Scholar 

  4. 4.

    Saeed B, Abou-Zor N, Amer Z, Kanani I, Hilal M. Cyclosporin-A induced posterior revesible encephalopathy syndrome. Saudi J Kidney Dis Transpl. 2008;19:439–42.

    PubMed  Google Scholar 

  5. 5.

    Sakai N, Kawasaki Y, Imaizumi T, Kanno S, et al. Two patients with focal segmental glomerulosclerosis complicated by cyclosporine-induced reversible posterior leukoencephalopathy syndrome. Clin Nephrol. 2010;73:482–6.

    CAS  Article  PubMed  Google Scholar 

  6. 6.

    Taque S, Peudenier S, Gie S, et al. Central neurotoxicity of cyclosporine in two children with nephrotic syndrome. Pediatr Nephrol. 2004;19:276–80.

    Article  PubMed  Google Scholar 

  7. 7.

    Zhang Y, Zhou J, Chen Y. Posterior reversible encephalopathy syndrome in a child with steroid-resistant nephrotic syndrome: a case report and review of literature. Int J Clin Exp Pathol. 2014;7:4433–7.

    PubMed  PubMed Central  Google Scholar 

  8. 8.

    Ishikura K, Hamasaki Y, Skai T, Hataya H, Mak RH, Honda M. Posterior reversible encephalopathy syndrome in children with kidney diseases. Pediatr Nephrol. 2012;27:375–84.

    Article  PubMed  Google Scholar 

  9. 9.

    Iyer RS, Chaturvedi A, Pruthi S, Khanna PC, Ishak GE. Medication neurotoxicity in children. Pediatr Radiol. 2011;41:1455–64.

    Article  PubMed  Google Scholar 

  10. 10.

    Ishikura K, Hamasaki Y, Sakai T, et al. Children with posterior reversible encephalopathy syndrome associated with atypical diffusion-weighted imaging and apparent diffusion coefficient. Clin Exp Nephrol. 2011;15:275–80.

    Article  PubMed  Google Scholar 

Download references

Author information

Affiliations

Authors

Corresponding author

Correspondence to Danica Batinić MD, PhD.

Ethics declarations

Conflict of interest

D. Batinić, D. Milošević, B. Filipović-Grčić, M. Topalović-Grković, N. Barišić and D. Turudić declare that they have no competing interests.

Ethical standards

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. Informed consent was obtained from the patients’ parents.

Rights and permissions

Reprints and Permissions

About this article

Verify currency and authenticity via CrossMark

Cite this article

Batinić, D., Milošević, D., Filipović-Grčić, B. et al. Neurotoxicity of cyclosporine A in children with steroid-resistant nephrotic syndrome: is cytotoxic edema really an unfavorable predictor of permanent neurological damage?. Wien Klin Wochenschr 129, 579–582 (2017). https://doi.org/10.1007/s00508-017-1221-z

Download citation

Keywords

  • Cyclosporine A neurotoxicity
  • Steroid-resistant nephrotic syndrome
  • Children