Literature DB >> 34144198

Expectations and experience: Parent and patient perspectives regarding treatment for Severe Combined Immunodeficiency (SCID).

Heather Smith1, Christopher Scalchunes2, Morton J Cowan3, Jennifer Puck3, Jennifer Heimall4.   

Abstract

INTRODUCTION: Infants with SCID are treated with hematopoietic cell transplantation (HCT) or gene therapy (GT). Caregiver perceptions of pre-treatment counseling and understanding of durability of HCT/GT are poorly understood.
METHODS: A survey was designed and distributed to families of patients with SCID. Topics in the questionnaire included SCID genotype and treatment, family recollections of pre-treatment counseling and present clinical status.
RESULTS: 151 surveys were analyzed. 132 were treated with HCT, 19 with GT. From counseling received, 37% expected HCT/GT would lead to "cure"; 43% expected HCT/GT would last a lifetime. Of 136 living patients, 59% reported overall good health but 65% reported some persistent health challenges.
CONCLUSIONS: For some, interpretation of the word "cure" varied, leading to misunderstanding regarding need for continued medical evaluations and additional therapies. Clear communication regarding the importance of lifelong follow-up, no matter the treatment outcome, will help to optimize good health and quality of life.
Copyright © 2021 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  Gene therapy; Genetic counseling; Hematopoietic stem cell transplant; Patient perspective; Patient reported outcomes; Severe Combined Immunodeficiency

Mesh:

Year:  2021        PMID: 34144198      PMCID: PMC8559521          DOI: 10.1016/j.clim.2021.108778

Source DB:  PubMed          Journal:  Clin Immunol        ISSN: 1521-6616            Impact factor:   10.190


  20 in total

1.  Hematopoietic stem cell transplantation for severe combined immunodeficiency in the neonatal period leads to superior thymic output and improved survival.

Authors:  Laurie A Myers; Dhavalkumar D Patel; Jennifer M Puck; Rebecca H Buckley
Journal:  Blood       Date:  2002-02-01       Impact factor: 22.113

2.  Communication Skills Training for Physicians Improves Patient Satisfaction.

Authors:  Adrienne Boissy; Amy K Windover; Dan Bokar; Matthew Karafa; Katie Neuendorf; Richard M Frankel; James Merlino; Michael B Rothberg
Journal:  J Gen Intern Med       Date:  2016-02-26       Impact factor: 5.128

Review 3.  Current Knowledge and Priorities for Future Research in Late Effects after Hematopoietic Stem Cell Transplantation (HCT) for Severe Combined Immunodeficiency Patients: A Consensus Statement from the Second Pediatric Blood and Marrow Transplant Consortium International Conference on Late Effects after Pediatric HCT.

Authors:  Jennifer Heimall; Jennifer Puck; Rebecca Buckley; Thomas A Fleisher; Andrew R Gennery; Benedicte Neven; Mary Slatter; Elie Haddad; Luigi D Notarangelo; K Scott Baker; Andrew C Dietz; Christine Duncan; Michael A Pulsipher; Mort J Cowan
Journal:  Biol Blood Marrow Transplant       Date:  2017-01-06       Impact factor: 5.742

4.  Immunological reconstitution of sex-linked lymphopenic immunological deficiency.

Authors:  R A Gatti; H J Meuwissen; H D Allen; R Hong; R A Good
Journal:  Lancet       Date:  1968-12-28       Impact factor: 79.321

5.  Validity and consequence of informed consent in pediatric bone marrow transplantation: The parental experience.

Authors:  Jan M Benedict; Christy Simpson; Conrad V Fernandez
Journal:  Pediatr Blood Cancer       Date:  2007-11       Impact factor: 3.167

Review 6.  Informed consent in pediatric research.

Authors:  Tom Leibson; Gideon Koren
Journal:  Paediatr Drugs       Date:  2015-02       Impact factor: 3.022

7.  Long-term outcome after hematopoietic stem cell transplantation of a single-center cohort of 90 patients with severe combined immunodeficiency.

Authors:  Bénédicte Neven; Sandrine Leroy; Hélène Decaluwe; Francoise Le Deist; Capucine Picard; Despina Moshous; Nizar Mahlaoui; Marianne Debré; Jean-Laurent Casanova; Liliane Dal Cortivo; Yoann Madec; Salima Hacein-Bey-Abina; Geneviève de Saint Basile; Jean-Pierre de Villartay; Stéphane Blanche; Marina Cavazzana-Calvo; Alain Fischer
Journal:  Blood       Date:  2009-01-23       Impact factor: 22.113

8.  Human Inborn Errors of Immunity: 2019 Update on the Classification from the International Union of Immunological Societies Expert Committee.

Authors:  Stuart G Tangye; Waleed Al-Herz; Aziz Bousfiha; Talal Chatila; Charlotte Cunningham-Rundles; Amos Etzioni; Jose Luis Franco; Steven M Holland; Christoph Klein; Tomohiro Morio; Hans D Ochs; Eric Oksenhendler; Capucine Picard; Jennifer Puck; Troy R Torgerson; Jean-Laurent Casanova; Kathleen E Sullivan
Journal:  J Clin Immunol       Date:  2020-01-17       Impact factor: 8.317

9.  Communication skills training for physicians improves health literacy and medical outcomes among patients with hypertension: a randomized controlled trial.

Authors:  Seyedeh Belin Tavakoly Sany; Fatemeh Behzhad; Gordon Ferns; Nooshin Peyman
Journal:  BMC Health Serv Res       Date:  2020-01-23       Impact factor: 2.655

10.  Communication Skills of Physicians and Patients' Satisfaction.

Authors:  Mohammad-Hossein Biglu; Farnaz Nateq; Morteza Ghojazadeh; Ali Asgharzadeh
Journal:  Mater Sociomed       Date:  2017-09
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