Skip to main content

Sweat test practice in pediatric pulmonology after introduction of cystic fibrosis newborn screening

Abstract

The influence of the generalization of cystic fibrosis newborn screening (CFNBS) in France on sweat test (ST) prescription is unknown. In this French retrospective, descriptive, and multicenter study, we studied the indications, number, methods, and results of STs prescribed by a pediatric pulmonologist in children who had a negative CFNBS and an ST for respiratory symptoms in 2012. We included 502 children with 523 STs, performed with four different methods. The main indication was asthma (71.3 %), then chronic cough (52.4 %), atypical lower airway infections (42.2 %), and bronchiectasis (7 %). Four children had a diagnosis of CF (0.8 %), all presenting with chronic productive cough and recurrent respiratory infections.

Conclusion: Asthma is the most frequent indication of ST in our cohort. Because of the systematic CFNBS in France, some prescriptions should be avoided, particularly in case of severe or moderate asthma with no other associated symptom. Moreover, methods of STs often do not follow the guidelines and need standardization.

What is Known:
• Newborn screening (NBS) has become the most frequent circumstance of the diagnosis of cystic fibrosis (CF) in France after its generalization.
• The prescription of sweat test (ST) in children with respiratory symptoms who already had a negative NBS has not been studied.
What is New:
• In children with a negative CF NBS referred to a university hospital for respiratory diseases, despite important variations of ST methods, only 4 children among 502 have been diagnosed as CF.
• Despite recommendations, ST prescription should be avoided in children with moderate to severe asthma and no other associated symptom.

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

Abbreviations

CF:

Cystic fibrosis

NBS:

Newborn screening

ST:

Sweat test

References

  1. 1.

    Asilsoy S, Bayram E, Agin H, Apa H, Can D, Gulle S, Altinoz S (2008) Evaluation of chronic cough in children. Chest 134:1122–1128

    Article  PubMed  Google Scholar 

  2. 2.

    Baumer JH (2003) Evidence based guidelines for the performance of the sweat test for the investigation of cystic fibrosis in the UK. Arch Dis Child 88:1126–1127

    PubMed Central  CAS  Article  PubMed  Google Scholar 

  3. 3.

    Bellis G, Lemonnier L, Sponga M (2014) Registre Français de la Mucoviscidose - Bilan des données 2012 (Vaincre la Mucoviscidose et Ined)

  4. 4.

    Brémont F (2001) Definition and nosological aspects of chronic cough in children. Arch Pediatr 8(Suppl 3):597–599

    Article  PubMed  Google Scholar 

  5. 5.

    Chang AB, Glomb WB (2006) Guidelines for evaluating chronic cough in pediatrics: ACCP evidence-based clinical practice guidelines. Chest 129:260S–283S

    Article  PubMed  Google Scholar 

  6. 6.

    Cohen R, Just J, Koskas M, Bingen E, Boucherat M, Bourrillon A, Foucaud P, François M, Garnier JM, Guillot M, Ployet MJ, Schlemmer C, Gaudelus J (2005) Recurrent respiratory tract infections: how should we investigate and treat? Arch Pediatr 12:183–190

    CAS  Article  PubMed  Google Scholar 

  7. 7.

    Couriel J (2002) Assessment of the child with recurrent chest infections. Br Med Bull 61:115–132

    Article  PubMed  Google Scholar 

  8. 8.

    De Boeck K, Wilschanski M, Castellani C, Taylor C, Cuppens H, Dodge J, Sinaasappel M (2006) Cystic fibrosis: terminology and diagnostic algorithms. Thorax 61:627–635

    PubMed Central  Article  PubMed  Google Scholar 

  9. 9.

    Donato L, Gaugler C, Weiss L, Krieger P, Debry C (2001) Chronic cough in children: signs of serious disease and investigations. Arch Pediatr 8(Suppl 3):638–644

    Article  PubMed  Google Scholar 

  10. 10.

    Farrell PM, Rosenstein BJ, White TB, Accurso FJ, Castellani C, Cutting GR, Durie PR, Legrys VA, Massie J, Parad RB, Rock MJ, Campbell PW 3rd (2008) Guidelines for diagnosis of cystic fibrosis in newborns through older adults: cystic fibrosis foundation consensus report. J Pediatr 153:S4–S14

    PubMed Central  Article  PubMed  Google Scholar 

  11. 11.

    HAS (2006) Mucoviscidose. Protocole national de diagnostic et de soins pour une maladie rare. Available from: www.has-sante.fr

  12. 12.

    HAS (2009) Asthme de l’enfant de moins de 36 mois : diagnostic, prise en charge et traitement en dehors des épisodes aigus - recommandations professionnelles. Available from: www.has-sante.fr

  13. 13.

    HAS (2009) Le dépistage néonatal systématique de la mucoviscidose en France : état des lieux et perspectives après 5 ans de fonctionnement. Available from: www.has-sante.fr

  14. 14.

    Hubert D, Le Bourgeois M (2012) The respiratory disease of cystic fibrosis, from infancy to childhood. Arch Pediatr 19(Suppl 1):S17–S19

    Article  PubMed  Google Scholar 

  15. 15.

    Lee DS, Rosenberg MA, Peterson A, Makholm L, Hoffman G, Laessig RH, Farrell PM (2003) Analysis of the costs of diagnosing cystic fibrosis with a newborn screening program. J Pediatr 142:617–623

    Article  PubMed  Google Scholar 

  16. 16.

    LeGrys VA, Yankaskas JR, Quittell LM, Marshall BC, Mogayzel PJ Jr (2007) Diagnostic sweat testing: the cystic fibrosis foundation guidelines. J Pediatr 151:85–89

    Article  PubMed  Google Scholar 

  17. 17.

    Lødrup Carlsen KC, Hedlin G, Bush A, Wennergren G, de Benedictis FM, De Jongste JC, Baraldi E, Pedroletti C, Barbato A, Malmström K, Pohunek P, Pedersen S, Piacentini GL, Middelveld RJM, Carlsen KH (2011) Assessment of problematic severe asthma in children. Eur Respir J 37:432–440

    Article  PubMed  Google Scholar 

  18. 18.

    Marchac V, Scheinmann P, de Blic J (2004) Severe asthma in children: identification and investigation. Arch Pediatr 11(Suppl 2):80s–85s

    Article  PubMed  Google Scholar 

  19. 19.

    Munck A, Roussey M (2008) The French nationwide cystic fibrosis newborn screening program: strategy and results. Arch Pediatr 15(Suppl 1):S1–S6

    Article  PubMed  Google Scholar 

  20. 20.

    O’Donnell AE (2008) Bronchiectasis. Chest 134:815–823

    Article  PubMed  Google Scholar 

  21. 21.

    Reynaud-Gaubert M (1999) Respiratory disease in cystic fibrosis: from physiopathology to therapy. Kinesitherapy and pulmonary transplantation excluded. Rev Mal Respir 16:495–509

    CAS  PubMed  Google Scholar 

  22. 22.

    Rosen MJ (2006) Chronic cough due to bronchiectasis: ACCP evidence-based clinical practice guidelines. Chest 129:122S–131S

    Article  PubMed  Google Scholar 

  23. 23.

    Rosenstein BJ, Cutting GR (1998) The diagnosis of cystic fibrosis: a consensus statement. Cystic fibrosis foundation consensus panel. J Pediatr 132:589–595

    CAS  Article  PubMed  Google Scholar 

  24. 24.

    Rota M, Nguyen-Khoa T, Marchand M, Feldmann D, Dumont J, Khalfon D, Vassault A, Borgard J-P (2008) Sweat testing: review of technical requirements. Ann Biol Clin (Paris) 66:221–227

    CAS  Google Scholar 

  25. 25.

    Roussey M, Munck A (2009) Questions raised and answers provided since the French nationwide cystic fibrosis newborn screening program was initiated. Arch Pediatr 16:540–542

    CAS  Article  PubMed  Google Scholar 

  26. 26.

    Sermet-Gaudelus I, Munck A, Rota M, Roussey M, Feldmann D, Nguyen-Khoa T (2010) French guidelines for sweat test practice and interpretation for cystic fibrosis neonatal screening. Arch Pediatr 17:1349–1358

    CAS  Article  PubMed  Google Scholar 

Download references

Acknowledgments

We thank Dr. B. Borwell for reading and correcting this manuscript.

Conflict of interest

The authors declare that they have no conflict of interest.

Author information

Affiliations

Authors

Corresponding author

Correspondence to Céline Grimaldi.

Additional information

Communicated by Peter de Winter

Rights and permissions

Reprints and Permissions

About this article

Verify currency and authenticity via CrossMark

Cite this article

Grimaldi, C., Brémont, F., Berlioz-Baudoin, M. et al. Sweat test practice in pediatric pulmonology after introduction of cystic fibrosis newborn screening. Eur J Pediatr 174, 1613–1620 (2015). https://doi.org/10.1007/s00431-015-2579-4

Download citation

Keywords

  • Pediatric pulmonology
  • Sweat test
  • Prescription
  • Respiratory symptoms