論文 - 詳細
| RRC ID | 87579 |
|---|---|
| 著者 | Weasner BM, Stinchfield MJ, Weasner BP, Takada S, O'Connor MB, Kumar JP, Newfeld SJ. |
| タイトル | Genetic analyses enabled by the Fourth Chromosome Resource Projectreveal unexpected mutant phenotypes and suggest new disease models. |
| ジャーナル | G3 (Bethesda) |
| Abstract |
To date the Fourth Chromosome Resource Project (FCRP) has deposited over 850 stocks for the genetic analysis of its 79 protein coding genes. Here we employ those stocks to reveal unexpected phenotypes for multiple exemplar genes. Expression studies of the transcript and protein provide clues to an adult function for maverick, a gene that despite prior efforts has remained inscrutable. Loss of function studies reveal an adult brain phenotype for the well studied signal transducer legless and the first phenotype for datilographo, a gene with no prior mutations. Marked clones with a new eyeless null allele in the larval brain elicited the first heterochronic phenotype in flies. Mutant clones of myoglianin encompassing the entire adult glial blood brain barrier elicited overgrowth of the underlying optic lobes. Complete clones of one glial layer within the two layer barrier were obtained for multiple genes and provide an opportunity to interrogate barrier crossing mechanisms for neurological therapeutics. In overexpression studies, rough eye phenotypes were generated by two human genes known to cause autosomal dominant neurological diseases. How YY1 and GRM1 haploinsufficiency leads to inherited cognitive impairment and spinocerebellar ataxia visible from birth is currently unknown. Fly eye phenotypes provide a tractable disease model for understanding their mechanism of action. Taken together, the ease with which mutant phenotypes were revealed suggests that a considerable amount of interesting biology remains to be uncovered on the fourth chromosome. |
| 公開日 | 2026-3-28 |
| DOI | 10.1093/g3journal/jkag077 |
| PII | 8554074 |
| PMID | 41902692 |
| リソース情報 | |
| ショウジョウバエ | DGRC#119078 DGRC#119079 DGRC#119080 DGRC#119081 DGRC#119082 DGRC#119083 DGRC#119084 DGRC#119085 "DGRC#118953 DGRC#119086 DGRC#118954 DGRC#119087 DGRC#118955 DGRC#119088 DGRC#118956 DGRC#119089 DGRC#118957 DGRC#118958 DGRC#119090 DGRC#118959 DGRC#119194 DGRC#118960 DGRC#118961 DGRC#119195 DGRC#118962 DGRC#119196 DGRC#118963 DGRC#118964 DGRC#119197 DGRC#118965 DGRC#118966 DGRC#118967 DGRC#119198 DGRC#118968 DGRC#119199 DGRC#118969 DGRC#118970 DGRC#119200 DGRC#118971 DGRC#118972 DGRC#119201 DGRC#118973 DGRC#118978 DGRC#118974 DGRC#118975 DGRC#118979 DGRC#118976 DGRC#118980 DGRC#118977 DGRC#118960 DGRC#118982 DGRC#119136 DGRC#119137 DGRC#118983 DGRC#119150 DGRC#119151 DGRC#119152 DGRC#119153 DGRC#119310 DGRC#119311 DGRC#119075 DGRC#119076 DGRC#119077 DGRC#118984 DGRC#118985 DGRC#118986 DGRC#118987 DGRC#118988 DGRC#118989 DGRC#118990 DGRC#118991 DGRC#118992 DGRC#118993 DGRC#118994 DGRC#118995 DGRC#118996 DGRC#118997 DGRC#118998 DGRC#118999 DGRC#119000 DGRC#119001 DGRC#119003 DGRC#119004 DGRC#119005 DGRC#119006 DGRC#119016 DGRC#119017 DGRC#119022 DGRC#119019 DGRC#119020 DGRC#119024 DGRC#119018 DGRC#119021 DGRC#119023 DGRC#119002 DGRC#119006 DGRC#119138 DGRC#119139 DGRC#119140 DGRC#119141 DGRC#119142 DGRC#119143 DGRC#119144 DGRC#119145 DGRC#119146 DGRC#119147 DGRC#119148 DGRC#119149 DGRC#118981 DGRC#119774 DGRC#119776 DGRC#119778 DGRC#119780 DGRC#119782 DGRC#119784 DGRC#119786 DGRC#119788 DGRC#119790 DGRC#119792 DGRC#119794 DGRC#119775 DGRC#119777 DGRC#119779 DGRC#119781 DGRC#119783 DGRC#119785 DGRC#119787 DGRC#119789 DGRC#119791 DGRC#119793 DGRC#119795" |