Skip to main content
Erschienen in: Pediatric Nephrology 4/2013

01.04.2013 | Original Article

Prediction of steroid-sparing agent use in childhood idiopathic nephrotic syndrome

verfasst von: Jérôme Harambat, Astrid Godron, Sandrine Ernould, Claire Rigothier, Brigitte Llanas, Sandrine Leroy

Erschienen in: Pediatric Nephrology | Ausgabe 4/2013

Einloggen, um Zugang zu erhalten

Abstract

Background

About half of children with steroid-sensitive idiopathic nephrotic syndrome (INS) will develop steroid dependency or a frequently relapsing course requiring steroid-sparing agents (SSA). Because of the adverse effects of prolonged steroid treatment, the early identification of children at high risk of requiring SSA may be a useful diagnostic tool to tailor the therapeutic strategy. The aim of this study was to identify predictors of the need for SSA and derive a predictive model.

Methods

This was a retrospective hospital-based cohort study which included all children with steroid-responsive INS followed for at least 4.5 months. Cox regression modeling and decision curve analysis were performed.

Results

A total of 120 children (81 boys) with INS were included and followed up for a median time of 6.7 (range 0.4–24.1) years. Median age at diagnosis was 3.4 years. Seventy-two (60 %) children required a SSA after a median time of 10 months following initial diagnosis. Male children, age at disease onset, methylprednisolone pulse use, and time to achieve first remission were significantly associated with the outcome. Time to achieve remission only remained significant after adjustment: hazard ratio (HR) = 1.9 [95 % confidence interval (CI) 1.5–2.5] if considered as a continuous variable, and HR = 4.1 (95 % CI 1.9–8.6) when dichotomized using the 9-day threshold. The area under the receiver operating curve of the related predictive model was 0.81 (95 % CI 0.74–0.89), and the decision curve analysis demonstrated that this model performed better than any other strategy.

Conclusions

Time to first remission is a strong predictor of the need for SSA in pediatric INS. Further prospective and impact studies are warranted to confirm the accuracy and benefit of our prediction model.
Anhänge
Nur mit Berechtigung zugänglich
Literatur
2.
Zurück zum Zitat [No authors listed] (1978) A report of the International Study of Kidney Disease in Children. Nephrotic syndrome in children: prediction of histopathology from clinical and laboratory characteristics at time of diagnosis. Kidney Int 13:159–165 [No authors listed] (1978) A report of the International Study of Kidney Disease in Children. Nephrotic syndrome in children: prediction of histopathology from clinical and laboratory characteristics at time of diagnosis. Kidney Int 13:159–165
3.
Zurück zum Zitat Tarshish P, Tobin JN, Bernstein J, Edelmann CR Jr (1997) Prognostic significance of the early course of minimal change nephrotic syndrome: report of the International Study of Kidney Disease in Children. J Am Soc Nephrol 8:769–776PubMed Tarshish P, Tobin JN, Bernstein J, Edelmann CR Jr (1997) Prognostic significance of the early course of minimal change nephrotic syndrome: report of the International Study of Kidney Disease in Children. J Am Soc Nephrol 8:769–776PubMed
4.
Zurück zum Zitat Hodson EM, Willis NS, Craig JC (2008) Non-corticosteroid treatment for nephrotic syndrome in children. Cochrane Database Syst Rev 1:CD002290 Hodson EM, Willis NS, Craig JC (2008) Non-corticosteroid treatment for nephrotic syndrome in children. Cochrane Database Syst Rev 1:CD002290
5.
Zurück zum Zitat Barratt TM, Soothill JF (1970) Controlled trial of cyclophosphamide in steroid-sensitive relapsing nephrotic syndrome of childhood. Lancet 2:479–482PubMedCrossRef Barratt TM, Soothill JF (1970) Controlled trial of cyclophosphamide in steroid-sensitive relapsing nephrotic syndrome of childhood. Lancet 2:479–482PubMedCrossRef
6.
Zurück zum Zitat Latta K, von Schnakenburg C, Ehrich JH (2001) A meta-analysis of cytotoxic treatment for frequently relapsing nephrotic syndrome in children. Pediatr Nephrol 16:271–282PubMedCrossRef Latta K, von Schnakenburg C, Ehrich JH (2001) A meta-analysis of cytotoxic treatment for frequently relapsing nephrotic syndrome in children. Pediatr Nephrol 16:271–282PubMedCrossRef
7.
Zurück zum Zitat British Association for Paediatric Nephrology (1991) Levamisole for corticosteroid-dependent nephrotic syndrome in childhood. Lancet 337:1555–1557 British Association for Paediatric Nephrology (1991) Levamisole for corticosteroid-dependent nephrotic syndrome in childhood. Lancet 337:1555–1557
8.
Zurück zum Zitat Ponticelli C, Edefonti A, Ghio L, Rizzoni G, Rinaldi S, Gusmano R, Lama G, Zacchello G, Confalonieri R, Altieri P, Bettinelli A, Maschio G, Cinotti GA, Fuiano G, Schena FP, Castellani A, Delia Casa-Alberighi O (1993) Cyclosporin versus cyclophosphamide for patients with steroid-dependent and frequently relapsing idiopathic nephrotic syndrome: a multicentre randomized controlled trial. Nephrol Dial Transplant 8:1326–1333PubMed Ponticelli C, Edefonti A, Ghio L, Rizzoni G, Rinaldi S, Gusmano R, Lama G, Zacchello G, Confalonieri R, Altieri P, Bettinelli A, Maschio G, Cinotti GA, Fuiano G, Schena FP, Castellani A, Delia Casa-Alberighi O (1993) Cyclosporin versus cyclophosphamide for patients with steroid-dependent and frequently relapsing idiopathic nephrotic syndrome: a multicentre randomized controlled trial. Nephrol Dial Transplant 8:1326–1333PubMed
9.
Zurück zum Zitat Hogg RJ, Fitzgibbons L, Bruick J, Bunke M, Ault B, Baqi N, Trachtman H, Swinford R (2006) Mycophenolate mofetil in children with frequently relapsing nephrotic syndrome: A report from the Southwest Pediatric Nephrology Study Group. Clin J Am Soc Nephrol 1:1173–1178PubMedCrossRef Hogg RJ, Fitzgibbons L, Bruick J, Bunke M, Ault B, Baqi N, Trachtman H, Swinford R (2006) Mycophenolate mofetil in children with frequently relapsing nephrotic syndrome: A report from the Southwest Pediatric Nephrology Study Group. Clin J Am Soc Nephrol 1:1173–1178PubMedCrossRef
10.
Zurück zum Zitat Guigonis V, Dallocchio A, Baudouin V, Dehennault M, Hachon-Le Camus C, Afanetti M, Groothoff J, Llanas B, Niaudet P, Nivet H, Raynaud N, Taque S, Ronco P, Bouissou F (2008) Rituximab treatment for severe steroid- or cyclosporine-dependent nephrotic syndrome: a multicentric series of 22 cases. Pediatr Nephrol 23:1269–1279PubMedCrossRef Guigonis V, Dallocchio A, Baudouin V, Dehennault M, Hachon-Le Camus C, Afanetti M, Groothoff J, Llanas B, Niaudet P, Nivet H, Raynaud N, Taque S, Ronco P, Bouissou F (2008) Rituximab treatment for severe steroid- or cyclosporine-dependent nephrotic syndrome: a multicentric series of 22 cases. Pediatr Nephrol 23:1269–1279PubMedCrossRef
11.
Zurück zum Zitat Constantinescu AR, Shah HB, Foote EF, Weiss LS (2000) Predicting first-year relapses in children with nephrotic syndrome. Pediatrics 105:492–495PubMedCrossRef Constantinescu AR, Shah HB, Foote EF, Weiss LS (2000) Predicting first-year relapses in children with nephrotic syndrome. Pediatrics 105:492–495PubMedCrossRef
12.
Zurück zum Zitat Yap HK, Han EJ, Heng CK, Gong WK (2001) Risk factors for steroid dependency in children with idiopathic nephrotic syndrome. Pediatr Nephrol 16:1049–1052PubMedCrossRef Yap HK, Han EJ, Heng CK, Gong WK (2001) Risk factors for steroid dependency in children with idiopathic nephrotic syndrome. Pediatr Nephrol 16:1049–1052PubMedCrossRef
13.
Zurück zum Zitat Andersen RF, Thrane N, Noergaard K, Rytter L, Jespersen B, Rittig S (2010) Early age at debut is a predictor of steroid-dependent and frequent relapsing nephrotic syndrome. Pediatr Nephrol 25:1299–1304PubMedCrossRef Andersen RF, Thrane N, Noergaard K, Rytter L, Jespersen B, Rittig S (2010) Early age at debut is a predictor of steroid-dependent and frequent relapsing nephrotic syndrome. Pediatr Nephrol 25:1299–1304PubMedCrossRef
14.
Zurück zum Zitat Vivarelli M, Moscaritolo E, Tsalkidis A, Massella L, Emma F (2010) Time for initial response to steroids is a major prognostic factor in idiopathic nephrotic syndrome. J Pediatr 156:965–971PubMedCrossRef Vivarelli M, Moscaritolo E, Tsalkidis A, Massella L, Emma F (2010) Time for initial response to steroids is a major prognostic factor in idiopathic nephrotic syndrome. J Pediatr 156:965–971PubMedCrossRef
15.
Zurück zum Zitat Letavernier B, Letavernier E, Leroy S, Baudet-Bonneville V, Bensman A, Ulinski T (2008) Prediction of high-degree steroid dependency in pediatric idiopathic nephrotic syndrome. Pediatr Nephrol 23:2221–2226PubMedCrossRef Letavernier B, Letavernier E, Leroy S, Baudet-Bonneville V, Bensman A, Ulinski T (2008) Prediction of high-degree steroid dependency in pediatric idiopathic nephrotic syndrome. Pediatr Nephrol 23:2221–2226PubMedCrossRef
16.
Zurück zum Zitat Bérard E, Broyer M, Dehennault M, Dumas R, Eckart P, Fischbach M, Loirat C, Martinat L, Pediatric Society of Nephrology (2005) Corticosensitive nephrotic syndrome (or nephrosis) in children. Therapeutic guideline proposed by the Pediatric Society of Nephrology. Nephrol Ther 1:150–156PubMedCrossRef Bérard E, Broyer M, Dehennault M, Dumas R, Eckart P, Fischbach M, Loirat C, Martinat L, Pediatric Society of Nephrology (2005) Corticosensitive nephrotic syndrome (or nephrosis) in children. Therapeutic guideline proposed by the Pediatric Society of Nephrology. Nephrol Ther 1:150–156PubMedCrossRef
17.
Zurück zum Zitat Royston P, Altman DG, Sauerbrei W (2006) Dichotomizing continuous predictors in multiple regression: a bad idea. Stat Med 25:127PubMedCrossRef Royston P, Altman DG, Sauerbrei W (2006) Dichotomizing continuous predictors in multiple regression: a bad idea. Stat Med 25:127PubMedCrossRef
18.
Zurück zum Zitat Vickers AJ, Elkin EB (2006) Decision curve analysis: a novel method for evaluating prediction models. Med Decis Making 26:565–574PubMedCrossRef Vickers AJ, Elkin EB (2006) Decision curve analysis: a novel method for evaluating prediction models. Med Decis Making 26:565–574PubMedCrossRef
19.
Zurück zum Zitat Niaudet P (2009) Long-term outcome of children with steroid-sensitive idiopathic nephrotic syndrome. Clin J Am Soc Nephrol 4:1547–1548PubMedCrossRef Niaudet P (2009) Long-term outcome of children with steroid-sensitive idiopathic nephrotic syndrome. Clin J Am Soc Nephrol 4:1547–1548PubMedCrossRef
20.
Zurück zum Zitat Rüth EM, Landolt MA, Neuhaus TJ, Kemper MJ (2004) Health-related quality of life and psychosocial adjustment in steroid-sensitive nephrotic syndrome. J Pediatr 145:778–783PubMedCrossRef Rüth EM, Landolt MA, Neuhaus TJ, Kemper MJ (2004) Health-related quality of life and psychosocial adjustment in steroid-sensitive nephrotic syndrome. J Pediatr 145:778–783PubMedCrossRef
21.
Zurück zum Zitat Kabuki N, Okugawa T, Hayakawa H, Tomizawa S, Kasahara T, Uchiyama M (1998) Influence of age at onset on the outcome of steroid-sensitive nephrotic syndrome. Pediatr Nephrol 12:467–470PubMedCrossRef Kabuki N, Okugawa T, Hayakawa H, Tomizawa S, Kasahara T, Uchiyama M (1998) Influence of age at onset on the outcome of steroid-sensitive nephrotic syndrome. Pediatr Nephrol 12:467–470PubMedCrossRef
22.
Zurück zum Zitat Fujinaga S, Hirano D, Nishizaki N (2011) Early identification of steroid dependency in Japanese children with steroid-sensitive nephrotic syndrome undergoing short-term initial steroid therapy. Pediatr Nephrol 26:485–486PubMedCrossRef Fujinaga S, Hirano D, Nishizaki N (2011) Early identification of steroid dependency in Japanese children with steroid-sensitive nephrotic syndrome undergoing short-term initial steroid therapy. Pediatr Nephrol 26:485–486PubMedCrossRef
23.
Zurück zum Zitat MacHardy N, Miles PV, Massengill SF, Smoyer WE, Mahan JD, Greenbaum L, Massie S, Yao L, Nagaraj S, Lin JJ, Wigfall D, Trachtman H, Hu Y, Gipson DS (2009) Management patterns of childhood-onset nephrotic syndrome. Pediatr Nephrol 24:2193–2201PubMedCrossRef MacHardy N, Miles PV, Massengill SF, Smoyer WE, Mahan JD, Greenbaum L, Massie S, Yao L, Nagaraj S, Lin JJ, Wigfall D, Trachtman H, Hu Y, Gipson DS (2009) Management patterns of childhood-onset nephrotic syndrome. Pediatr Nephrol 24:2193–2201PubMedCrossRef
24.
Zurück zum Zitat Ehrich JH, Brodehl J (1993) Long versus standard prednisone therapy for initial treatment of idiopathic nephrotic syndrome in children. Arbeitsgemeinschaft für Pädiatrische Nephrologie. Eur J Pediatr 152:357–361PubMedCrossRef Ehrich JH, Brodehl J (1993) Long versus standard prednisone therapy for initial treatment of idiopathic nephrotic syndrome in children. Arbeitsgemeinschaft für Pädiatrische Nephrologie. Eur J Pediatr 152:357–361PubMedCrossRef
25.
Zurück zum Zitat Hoyer PF, Brodehl J (2006) Initial treatment of idiopathic nephrotic syndrome in children: prednisone versus prednisone plus cyclosporine A: a prospective, randomized trial. J Am Soc Nephrol 17:1151–1157PubMedCrossRef Hoyer PF, Brodehl J (2006) Initial treatment of idiopathic nephrotic syndrome in children: prednisone versus prednisone plus cyclosporine A: a prospective, randomized trial. J Am Soc Nephrol 17:1151–1157PubMedCrossRef
Metadaten
Titel
Prediction of steroid-sparing agent use in childhood idiopathic nephrotic syndrome
verfasst von
Jérôme Harambat
Astrid Godron
Sandrine Ernould
Claire Rigothier
Brigitte Llanas
Sandrine Leroy
Publikationsdatum
01.04.2013
Verlag
Springer-Verlag
Erschienen in
Pediatric Nephrology / Ausgabe 4/2013
Print ISSN: 0931-041X
Elektronische ISSN: 1432-198X
DOI
https://doi.org/10.1007/s00467-012-2365-8

Weitere Artikel der Ausgabe 4/2013

Pediatric Nephrology 4/2013 Zur Ausgabe

Update Pädiatrie

Bestellen Sie unseren Fach-Newsletter und bleiben Sie gut informiert.