TY - JOUR
T1 - Animal models for cystic fibrosis
T2 - A systematic search and mapping review of the literature – Part 1: genetic models
AU - Leenaars, Cathalijn H.C.
AU - De Vries, Rob B.M.
AU - Heming, Anna
AU - Visser, Damian
AU - Holthaus, David
AU - Reijmer, Joey
AU - Elzinga, Janneke
AU - Kempkes, Rosalie W.M.
AU - Punt, Carine
AU - Beumer, Wouter
AU - Meijboom, Franck L.B.
AU - Ritskes-Hoitinga, Merel
PY - 2020
Y1 - 2020
N2 - Animal models for cystic fibrosis (CF) have enhanced our understanding of the pathology and contributed to the development of new treatments. In the field of CF, many animal models have been developed and described. To our knowledge, thus far, none of the reviews of CF animal models has used a systematic methodology. A systematic approach to creating model overviews can lead to an objective, evidence-based choice of an animal model for new research questions. We searched Pubmed and Embase for the currently available animal models for CF. Two independent reviewers screened the results. We included all primary studies describing an animal model for CF. After duplicate removal, 12,304 publications were left. Because of the large number of models, in the current paper, only the genetic models are presented. A total of 636 publications were identified describing genetic animal models for CF in mice, pigs, ferrets, rats and zebrafish. Most of these models have an altered Cftr gene. An overview of basic model characteristics and outcome measures for these genetic models is provided, together with advice on using these data. As far as the authors are aware, this is one of the largest systematic mapping reviews on genetic animal models for CF. It can aid in selecting a suitable model and outcome measures. In general, the reporting quality of the included publications was poor. Further systematic reviews are warranted to determine the quality and translational value of these models further.
AB - Animal models for cystic fibrosis (CF) have enhanced our understanding of the pathology and contributed to the development of new treatments. In the field of CF, many animal models have been developed and described. To our knowledge, thus far, none of the reviews of CF animal models has used a systematic methodology. A systematic approach to creating model overviews can lead to an objective, evidence-based choice of an animal model for new research questions. We searched Pubmed and Embase for the currently available animal models for CF. Two independent reviewers screened the results. We included all primary studies describing an animal model for CF. After duplicate removal, 12,304 publications were left. Because of the large number of models, in the current paper, only the genetic models are presented. A total of 636 publications were identified describing genetic animal models for CF in mice, pigs, ferrets, rats and zebrafish. Most of these models have an altered Cftr gene. An overview of basic model characteristics and outcome measures for these genetic models is provided, together with advice on using these data. As far as the authors are aware, this is one of the largest systematic mapping reviews on genetic animal models for CF. It can aid in selecting a suitable model and outcome measures. In general, the reporting quality of the included publications was poor. Further systematic reviews are warranted to determine the quality and translational value of these models further.
KW - Animal models
KW - cystic fibrosis
KW - outcome measures
KW - search string
KW - systematic map
UR - http://www.scopus.com/inward/record.url?scp=85071459579&partnerID=8YFLogxK
U2 - https://doi.org/10.1177/0023677219868502
DO - https://doi.org/10.1177/0023677219868502
M3 - Review article
C2 - 31411127
SN - 0023-6772
VL - 54
SP - 330
EP - 340
JO - Laboratory animals
JF - Laboratory animals
IS - 4
ER -