| Literature DB >> 29753093 |
Ester Garne1, Anke Rissmann2, Marie-Claude Addor3, Ingeborg Barisic4, Jorieke Bergman5, Paula Braz6, Clara Cavero-Carbonell7, Elizabeth S Draper8, Miriam Gatt9, Martin Haeusler10, Kari Klungsoyr11, Jennifer J Kurinczuk12, Nathalie Lelong13, Karen Luyt14, Catherine Lynch15, Mary T O'Mahony16, Olatz Mokoroa17, Vera Nelen18, Amanda J Neville19, Anna Pierini20, Hanitra Randrianaivo21, Judith Rankin22, Florence Rouget23, Bruno Schaub24, David Tucker25, Christine Verellen-Dumoulin26, Diana Wellesley27, Awi Wiesel28, Nataliia Zymak-Zakutnia29, Monica Lanzoni30, Joan K Morris31.
Abstract
Septo-optic nerve dysplasia is a rare congenital anomaly with optic nerve hypoplasia, pituitary hormone deficiencies and midline developmental defects of the brain. The clinical findings are visual impairment, hypopituitarism and developmental delays. The aim of this study was to report prevalence, associated anomalies, maternal age and other epidemiological factors from a large European population based network of congenital anomaly registries (EUROCAT). Data from 29 full member registries for the years 2005-2014 were included, covering 6.4 million births. There were 99 cases with a diagnosis of septo-optic dysplasia. The prevalence of septo-optic dysplasia in Europe was calculated to lie between 1.9 and 2.5 per 100,000 births after adjusting for potential under-reporting in some registries. The prevalence was highest in babies of mothers aged 20-24 years of age and was significantly higher in UK registries compared with other EUROCAT registries (P = 0.021 in the multilevel model) and the additional risk for younger mothers was significantly greater in the UK compared to the rest of Europe (P = 0.027). The majority of septo-optic dysplasia cases were classified as an isolated cerebral anomaly (N = 76, 77%). Forty percent of diagnoses occurred in fetuses with a prenatal diagnosis. The anomaly may not be visible at birth, which is reflected in that 57% of the postnatal diagnoses occurred over 1 month after birth. This is the first population based study to describe the prevalence of septo-optic dysplasia in Europe. Septo-optic dysplasia shares epidemiological patterns with gastroschisis and this strengthens the hypothesis of vascular disruption being an aetiological factor for septo-optic dysplasia.Entities:
Keywords: Associated anomalies; EUROCAT; Maternal age; Population based; Prevalence; Septo-optic dysplasia
Mesh:
Year: 2018 PMID: 29753093 DOI: 10.1016/j.ejmg.2018.05.010
Source DB: PubMed Journal: Eur J Med Genet ISSN: 1769-7212 Impact factor: 2.708