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An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme

Aim: Sickle cell disease (SCD) is the most frequent monogenic disease worldwide; ~5–7% of the world population carry a hemoglobin disorder trait. In the US, one in every 1,941 newborns has SCD, whereas one in every 3,000 newborns in France is affected - resulting in 385 new cases and 5,883 newly ide...

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Autores principales: Rémus, Christelle, Stanislas, Aurélie, Bouazza, Naïm, Gauthereau, Valérie, Polak, Michel, Blanche, Stéphane, Niakaté, Assa, Gluckman, Eliane, Tréluyer, Jean-Marc, Munnich, Arnold, Girot, Robert, Cavazzana, Marina
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7318296/
https://www.ncbi.nlm.nih.gov/pubmed/32637386
http://dx.doi.org/10.3389/fped.2020.00300
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author Rémus, Christelle
Stanislas, Aurélie
Bouazza, Naïm
Gauthereau, Valérie
Polak, Michel
Blanche, Stéphane
Niakaté, Assa
Gluckman, Eliane
Tréluyer, Jean-Marc
Munnich, Arnold
Girot, Robert
Cavazzana, Marina
author_facet Rémus, Christelle
Stanislas, Aurélie
Bouazza, Naïm
Gauthereau, Valérie
Polak, Michel
Blanche, Stéphane
Niakaté, Assa
Gluckman, Eliane
Tréluyer, Jean-Marc
Munnich, Arnold
Girot, Robert
Cavazzana, Marina
author_sort Rémus, Christelle
collection PubMed
description Aim: Sickle cell disease (SCD) is the most frequent monogenic disease worldwide; ~5–7% of the world population carry a hemoglobin disorder trait. In the US, one in every 1,941 newborns has SCD, whereas one in every 3,000 newborns in France is affected - resulting in 385 new cases and 5,883 newly identified carriers per year. The objective of the present study was to evaluate three different ways of providing information to parents at risk of having a child with SCD, with a view to increasing the parental screening rate and decreasing the number of new cases per year in France. Method: In a randomized study, we contacted 300 couples of parents after their child had been identified as a SCD carrier in the French national newborn screening programme: 100 couples received an information letter (the standard procedure in France: arm A), 100 couples received a letter and then a follow-up phone call (arm B), and 100 received a letter and then three follow-up text messages at 5-day intervals (arm C). The primary endpoint was the number of parents in each arm screened in the 120 days after the letter had been sent. In a modified intention-to-treat analysis, the screening rate was 17% in arm A, 35% in arm B, and 30% in arm C. Results: Telephone and text message follow-ups were associated with higher screening rates, compared with no follow-up. After being informed of their child's carrier status, some parents had consulted a healthcare professional but had not been referred for screening (16% in arm A, 19% in arm B, and 13% in arm C). Conclusion: A letter followed by a phone call or three text messages is more effective than a letter alone for informing parents at risk of having a child with SCD. The effective implementation of this follow-up programme probably requires better training of all the healthcare professionals involved.
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spelling pubmed-73182962020-07-06 An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme Rémus, Christelle Stanislas, Aurélie Bouazza, Naïm Gauthereau, Valérie Polak, Michel Blanche, Stéphane Niakaté, Assa Gluckman, Eliane Tréluyer, Jean-Marc Munnich, Arnold Girot, Robert Cavazzana, Marina Front Pediatr Pediatrics Aim: Sickle cell disease (SCD) is the most frequent monogenic disease worldwide; ~5–7% of the world population carry a hemoglobin disorder trait. In the US, one in every 1,941 newborns has SCD, whereas one in every 3,000 newborns in France is affected - resulting in 385 new cases and 5,883 newly identified carriers per year. The objective of the present study was to evaluate three different ways of providing information to parents at risk of having a child with SCD, with a view to increasing the parental screening rate and decreasing the number of new cases per year in France. Method: In a randomized study, we contacted 300 couples of parents after their child had been identified as a SCD carrier in the French national newborn screening programme: 100 couples received an information letter (the standard procedure in France: arm A), 100 couples received a letter and then a follow-up phone call (arm B), and 100 received a letter and then three follow-up text messages at 5-day intervals (arm C). The primary endpoint was the number of parents in each arm screened in the 120 days after the letter had been sent. In a modified intention-to-treat analysis, the screening rate was 17% in arm A, 35% in arm B, and 30% in arm C. Results: Telephone and text message follow-ups were associated with higher screening rates, compared with no follow-up. After being informed of their child's carrier status, some parents had consulted a healthcare professional but had not been referred for screening (16% in arm A, 19% in arm B, and 13% in arm C). Conclusion: A letter followed by a phone call or three text messages is more effective than a letter alone for informing parents at risk of having a child with SCD. The effective implementation of this follow-up programme probably requires better training of all the healthcare professionals involved. Frontiers Media S.A. 2020-06-19 /pmc/articles/PMC7318296/ /pubmed/32637386 http://dx.doi.org/10.3389/fped.2020.00300 Text en Copyright © 2020 Rémus, Stanislas, Bouazza, Gauthereau, Polak, Blanche, Niakaté, Gluckman, Tréluyer, Munnich, Girot and Cavazzana. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Pediatrics
Rémus, Christelle
Stanislas, Aurélie
Bouazza, Naïm
Gauthereau, Valérie
Polak, Michel
Blanche, Stéphane
Niakaté, Assa
Gluckman, Eliane
Tréluyer, Jean-Marc
Munnich, Arnold
Girot, Robert
Cavazzana, Marina
An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title_full An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title_fullStr An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title_full_unstemmed An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title_short An Evaluation of Three Ways of Communicating Carrier Status Results to the Parents of Children in a Neonatal Sickle Cell Screening Programme
title_sort evaluation of three ways of communicating carrier status results to the parents of children in a neonatal sickle cell screening programme
topic Pediatrics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7318296/
https://www.ncbi.nlm.nih.gov/pubmed/32637386
http://dx.doi.org/10.3389/fped.2020.00300
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