{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T00:15:29Z","timestamp":1792282529039,"version":"4.4.0"},"reference-count":80,"publisher":"The Company of Biologists","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2015,1,1]]},"abstract":"<jats:p>Microphthalmos is a rare congenital anomaly characterized by reduced eye size and visual deficits of variable degrees. Sporadic and hereditary microphthalmos has been associated to heterozygous mutations in genes fundamental for eye development. Yet, many cases are idiopathic or await the identification of molecular causes. Here we show that haploinsufficiency of Meis1, a transcription factor with an evolutionary conserved expression in the embryonic trunk, brain and sensory organs, including the eye, causes microphthalmic traits and visual impairment, in adult mice. By combining the analysis of Meis1 loss-of-function and conditional Meis1 functional rescue with ChIP-seq and RNA-seq approaches we show that, in contrast to Meis1 preferential association with Hox-Pbx binding sites in the trunk, Meis1 binds to Hox\/Pbx-independent sites during optic cup development. In the eye primordium, Meis1 coordinates, in a dose-dependent manner, retinal proliferation and differentiation by regulating genes responsible for human microphthalmia and components the Notch signalling pathway. In addition, Meis1 is required for eye patterning by controlling a set of eye territory-specific transcription factors, so that in Meis1\u2212\/\u2212 embryos boundaries among the different eye territories are shifted or blurred. We thus propose that Meis1 is at the core of a genetic network implicated in eye patterning\/microphthalmia, itself representing an additional candidate for syndromic cases of these ocular malformations.<\/jats:p>","DOI":"10.1242\/dev.122176","type":"journal-article","created":{"date-parts":[[2015,8,8]],"date-time":"2015-08-08T01:12:41Z","timestamp":1438996361000},"source":"Crossref","is-referenced-by-count":30,"title":["Meis1 coordinates a network of genes implicated in eye development and microphthalmia"],"prefix":"10.1242","author":[{"given":"S\u00e9verine","family":"Marcos","sequence":"first","affiliation":[{"name":"Centro de Biolog\u00eda Molecular \u201cSevero Ochoa\u201d, CSIC-UAM, c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"},{"name":"CIBER de Enfermedades Raras (CIBERER), c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Monica","family":"Gonz\u00e1lez-L\u00e1zaro","sequence":"additional","affiliation":[{"name":"Departamento de Desarrollo y Reparaci\u00f3n Cardiovascular, Centro Nacional de Investigaciones Cardiovasculares (CNIC), c\/ Melchor Fern\u00e1ndez Almagro, 3, E-28029 Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leonardo","family":"Beccari","sequence":"additional","affiliation":[{"name":"Centro de Biolog\u00eda Molecular \u201cSevero Ochoa\u201d, CSIC-UAM, c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"},{"name":"CIBER de Enfermedades Raras (CIBERER), c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Carramolino","sequence":"additional","affiliation":[{"name":"Departamento de Desarrollo y Reparaci\u00f3n Cardiovascular, Centro Nacional de Investigaciones Cardiovasculares (CNIC), c\/ Melchor Fern\u00e1ndez Almagro, 3, E-28029 Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Jesus","family":"Martin-Bermejo","sequence":"additional","affiliation":[{"name":"Centro de Biolog\u00eda Molecular \u201cSevero Ochoa\u201d, CSIC-UAM, c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"},{"name":"CIBER de Enfermedades Raras (CIBERER), c\/ Nicol\u00e1s Cabrera, 1, E-28049 Madrid, Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Oana","family":"Amarie","sequence":"additional","affiliation":[{"name":"Institute of Developmental Genetics Helmholtz Center Munich; 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