Recognition of the Cornelia de Lange syndrome phenotype with facial dysmorphology novel analysis

被引:89
作者
Basel-Vanagaite, L. [1 ,2 ,3 ,4 ]
Wolf, L. [3 ,4 ]
Orin, M. [4 ]
Larizza, L. [5 ,6 ]
Gervasini, C. [5 ,6 ]
Krantz, I. D. [7 ,8 ]
Deardoff, M. A. [7 ,8 ]
机构
[1] Schneider Childrens Med Ctr Israel, Dept Med Genet, Rabin Med Ctr, Beilinson Campus,39 Jabotinsky St, IL-49101 Petah Tiqwa, Israel
[2] Felsenstein Med Res Ctr, Petah Tiqwa, Israel
[3] Tel Aviv Univ, IL-69978 Tel Aviv, Israel
[4] FDNA Inc, Boston, MA USA
[5] Ist Auxol Italiano, Lab Med Cytogenet & Mol Genet, Milan, Italy
[6] Univ Milan, Dept Hlth Sci, Med Genet, Milan, Italy
[7] Childrens Hosp Philadelphia, Div Human Mol Genet, Philadelphia, PA 19104 USA
[8] Univ Penn, Perelman Sch Med, Philadelphia, PA 19104 USA
关键词
automated facial recognition; CdLS; clinical genetics; dysmorphology; FDNA; FETAL ALCOHOL SPECTRUM; GENOMIC IMBALANCES; DELETION SYNDROME; MUTATIONS; NIPBL; REARRANGEMENTS; FEATURES; CDLS;
D O I
10.1111/cge.12716
中图分类号
Q3 [遗传学];
学科分类号
071007 [遗传学];
摘要
Facial analysis systems are becoming available to healthcare providers to aid in the recognition of dysmorphic phenotypes associated with a multitude of genetic syndromes. These technologies automatically detect facial points and extract various measurements from images to recognize dysmorphic features and evaluate similarities to known facial patterns (gestalts). To evaluate such systems' usefulness for supporting the clinical practice of healthcare professionals, the recognition accuracy of the Cornelia de Lange syndrome (CdLS) phenotype was examined with FDNA's automated facial dysmorphology novel analysis (FDNA) technology. In the first experiment, 2D facial images of CdLS patients with either an NIPBL or SMC1A gene mutation as well as non-CdLS patients which were assessed by dysmorphologists in a previous study were evaluated by the FDNA technology; the average detection rate of experts was 77% while the system's detection rate was 87%. In the second study, when a new set of NIPBL, SMC1A and non-CdLS patient photos was evaluated, the detection rate increased to 94%. The results from both studies indicated that the system's detection rate was comparable to that of dysmorphology experts. Therefore, utilizing such technologies may be a useful tool in a clinical setting.
引用
收藏
页码:557 / 563
页数:7
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