Clinical Evaluation of the Nanoduct Sweat Test System in the Diagnosis of Cystic Fibrosis After Newborn Screening

Overview

Journal Eur J Pediatr

Specialty Pediatrics

Date 2015 Feb 14

PMID 25678232

Citations 10

Authors

Annette Vernooij-van Langen

Edward Dompeling

Jan-Bart Yntema

Bert Arets

Harm Tiddens

Gerard Loeber

Jeannette Dankert-Roelse

Affiliations

Soon will be listed here.

Abstract

Unlabelled: After a positive newborn screening test for cystic fibrosis (CF), a sweat test is performed to confirm the diagnosis. The success rate of the generally acknowledged methods (Macroduct/Gibson and Cooke) in newborns varies between 73 and 99%. The Nanoduct sweat test system is easier to perform and less sweat is needed. The main aim of this study was to measure the success rate of the Nanoduct compared to current approved sweat test methods in a newborn population. After informed consent of the parents, newborns with a positive screening test for CF were included. The Macroduct or Gibson and Cooke and Nanoduct were performed in all infants, during the same appointment. The chloride concentration was determined by standard coulorimetry; conductivity was measured directly and converted to a NaCl molarity. One hundred eight newborns were included: 17 with CF, 7 with cystic fibrosis transmembrane regulator (CFTR)-related metabolic syndrome (CRMS), and 84 healthy children. The success rate of the Nanoduct was 93% and for the Macroduct/Gibson and Cooke 79% (McNemar, p = 0.002). The Nanoduct detected the same CF patients as the Macroduct/Gibson and Cooke; one CF patient had an equivocal result for both tests, and no patients were missed. The area under the receiver operating characteristic curve for detection of CF with the Nanoduct was 0.999, with ideal cutoff levels of 91 and 66 mmol/l, comparable to former studies.

Conclusion: The success rate of the Nanoduct to collect sufficient sweat in infants was higher compared to the Macroduct and Gibson and Cooke.

Citing Articles

Diagnosing cystic fibrosis in low- and middle-income countries: challenges and strategies.

Fuhrer M, Zampoli M, Abriel H Orphanet J Rare Dis. 2024; 19(1):482.

PMID: 39707455 PMC: 11662431. DOI: 10.1186/s13023-024-03506-1.

Diagnosing Cystic Fibrosis in the 21st Century-A Complex and Challenging Task.

Anton-Paduraru D, Azoicai A, Trofin F, Mindru D, Murgu A, Bocec A Diagnostics (Basel). 2024; 14(7).

PMID: 38611676 PMC: 11012009. DOI: 10.3390/diagnostics14070763.

Single-centre prospective evaluation of the first 5 years of cystic fibrosis newborn screening in Germany.

Gesenhues F, Michel K, Greve T, Roschinger W, Gothe F, Nubling J ERJ Open Res. 2024; 10(2).

PMID: 38444668 PMC: 10910348. DOI: 10.1183/23120541.00699-2023.

Exploring the Potential of Nanotechnology in Pediatric Healthcare: Advances, Challenges, and Future Directions.

Omidian H, Mfoafo K Pharmaceutics. 2023; 15(6).

PMID: 37376032 PMC: 10303369. DOI: 10.3390/pharmaceutics15061583.

Electrical aspects of skin as a pathway to engineering skin devices.

Abe Y, Nishizawa M APL Bioeng. 2021; 5(4):041509.

PMID: 34849444 PMC: 8604566. DOI: 10.1063/5.0064529.

References

Kleyn M, Korzeniewski S, Grigorescu V, Young W, Homnick D, Goldstein-Filbrun A . Predictors of insufficient sweat production during confirmatory testing for cystic fibrosis. Pediatr Pulmonol. 2010; 46(1):23-30. DOI: 10.1002/ppul.21318. View

Parad R, Marie Comeau A, Dorkin H, Dovey M, Gerstle R, Martin T . Sweat testing infants detected by cystic fibrosis newborn screening. J Pediatr. 2005; 147(3 Suppl):S69-72. DOI: 10.1016/j.jpeds.2005.08.015. View

Sezer R, Aydemir G, Akcan A, Paketci C, Karaoglu A, Aydinoz S . Nanoduct sweat conductivity measurements in 2664 patients: relationship to age, arterial blood gas, serum electrolyte profiles and clinical diagnosis. J Clin Med Res. 2013; 5(1):34-41. PMC: 3564566. DOI: 10.4021/jocmr1191w. View

Eng W, LeGrys V, Schechter M, Laughon M, Barker P . Sweat-testing in preterm and full-term infants less than 6 weeks of age. Pediatr Pulmonol. 2005; 40(1):64-7. DOI: 10.1002/ppul.20235. View

Slieker M, Uiterwaal C, Sinaasappel M, Heijerman H, van der Laag J, van der Ent C . Birth prevalence and survival in cystic fibrosis: a national cohort study in the Netherlands. Chest. 2005; 128(4):2309-15. DOI: 10.1378/chest.128.4.2309. View

Sands D, Oltarzewski M, Nowakowska A, Zybert K . Bilateral sweat tests with two different methods as a part of cystic fibrosis newborn screening (CF NBS) protocol and additional quality control. Folia Histochem Cytobiol. 2010; 48(3):358-65. DOI: 10.2478/v10042-010-0044-9. View

Lezana J, Vargas M, Karam-Bechara J, Aldana R, Furuya M . Sweat conductivity and chloride titration for cystic fibrosis diagnosis in 3834 subjects. J Cyst Fibros. 2004; 2(1):1-7. DOI: 10.1016/S1569-1993(02)00146-7. View

Tluczek A, Koscik R, Farrell P, Rock M . Psychosocial risk associated with newborn screening for cystic fibrosis: parents' experience while awaiting the sweat-test appointment. Pediatrics. 2005; 115(6):1692-703. DOI: 10.1542/peds.2004-0275. View

Williams S, Nussbaum E, Chin T, Do P, Singh K, Randhawa I . Diagnosis of cystic fibrosis in the kindred of an infant with CFTR-related metabolic syndrome: importance of follow-up that includes monitoring sweat chloride concentrations over time. Pediatr Pulmonol. 2014; 49(3):E103-8. DOI: 10.1002/ppul.22918. View

10.

Sims E, Clark A, McCormick J, Mehta G, Connett G, Mehta A . Cystic fibrosis diagnosed after 2 months of age leads to worse outcomes and requires more therapy. Pediatrics. 2007; 119(1):19-28. DOI: 10.1542/peds.2006-1498. View

11.

Losty H, Wheatley H, Doull I . The evaluation of a novel conductometric device for the diagnosis of cystic fibrosis. Ann Clin Biochem. 2006; 43(Pt 5):375-81. DOI: 10.1258/000456306778520025. View

12.

Moran J, Quirk K, Duff A, Brownlee K . Newborn screening for CF in a regional paediatric centre: the psychosocial effects of false-positive IRT results on parents. J Cyst Fibros. 2006; 6(3):250-4. DOI: 10.1016/j.jcf.2006.09.001. View

13.

LeGrys V . Assessment of sweat-testing practices for the diagnosis of cystic fibrosis. Arch Pathol Lab Med. 2001; 125(11):1420-4. DOI: 10.5858/2001-125-1420-AOSTPF. View

14.

Mayell S, Munck A, Craig J, Sermet I, Brownlee K, Schwarz M . A European consensus for the evaluation and management of infants with an equivocal diagnosis following newborn screening for cystic fibrosis. J Cyst Fibros. 2008; 8(1):71-8. DOI: 10.1016/j.jcf.2008.09.005. View

15.

Vernooij-van Langen A, Loeber J, Elvers B, Triepels R, Gille J, van der Ploeg C . Novel strategies in newborn screening for cystic fibrosis: a prospective controlled study. Thorax. 2012; 67(4):289-95. DOI: 10.1136/thoraxjnl-2011-200730. View

16.

Mastella G, Di Cesare G, Borruso A, Menin L, Zanolla L . Reliability of sweat-testing by the Macroduct collection method combined with conductivity analysis in comparison with the classic Gibson and Cooke technique. Acta Paediatr. 2000; 89(8):933-7. DOI: 10.1080/080352500750043378. View

17.

Barben J, Ammann R, Metlagel A, Schoeni M . Conductivity determined by a new sweat analyzer compared with chloride concentrations for the diagnosis of cystic fibrosis. J Pediatr. 2005; 146(2):183-8. DOI: 10.1016/j.jpeds.2004.08.085. View

18.

De Boeck K, Wilschanski M, Castellani C, Taylor C, Cuppens H, Dodge J . Cystic fibrosis: terminology and diagnostic algorithms. Thorax. 2005; 61(7):627-35. PMC: 2104676. DOI: 10.1136/thx.2005.043539. View

19.

Heeley M, Woolf D, HEELEY A . Indirect measurements of sweat electrolyte concentration in the laboratory diagnosis of cystic fibrosis. Arch Dis Child. 2000; 82(5):420-4. PMC: 1718326. DOI: 10.1136/adc.82.5.420. View

20.

Farrell P, Koscik R . Sweat chloride concentrations in infants homozygous or heterozygous for F508 cystic fibrosis. Pediatrics. 1996; 97(4):524-8. View