論文 - 詳細
| RRC ID | 79053 |
|---|---|
| 著者 | Martelli F, Lin J, Mele S, Imlach W, Kanca O, Barlow CK, Paril J, Schittenhelm RB, Christodoulou J, Bellen HJ, Piper MDW, Johnson TK. |
| タイトル | Identifying potential dietary treatments for inherited metabolic disorders using Drosophila nutrigenomics. |
| ジャーナル | Cell Rep |
| Abstract |
Inherited metabolic disorders are a group of genetic conditions that can cause severe neurological impairment and child mortality. Uniquely, these disorders respond to dietary treatment; however, this option remains largely unexplored because of low disorder prevalence and the lack of a suitable paradigm for testing diets. Here, we screened 35 Drosophila amino acid disorder models for disease-diet interactions and found 26 with diet-altered development and/or survival. Using a targeted multi-nutrient array, we examine the interaction in a model of isolated sulfite oxidase deficiency, an infant-lethal disorder. We show that dietary cysteine depletion normalizes their metabolic profile and rescues development, neurophysiology, behavior, and lifelong fly survival, thus providing a basis for further study into the pathogenic mechanisms involved in this disorder. Our work highlights the diet-sensitive nature of metabolic disorders and establishes Drosophila as a valuable tool for nutrigenomic studies for informing potential dietary therapies. |
| 巻・号 | 43(3) |
| ページ | 113861 |
| 公開日 | 2024-3-26 |
| DOI | 10.1016/j.celrep.2024.113861 |
| PII | S2211-1247(24)00189-X |
| PMID | 38416643 |
| PMC | PMC11037929 |
| MeSH | Amino Acid Metabolism, Inborn Errors* Animals Child Diet Drosophila Humans Infant Metabolic Diseases* / genetics Nutrigenomics |
| IF | 8.109 |
| オルトメトリクス指標 |
オルトメトリクス指標項目
|
| 最多言及媒体 | X(Twitter) |
| 各媒体での言及数の合計 | 21 |
| 過去6か月間でのオルトメトリクス指標の変動値 | 0.0 |
| リソース情報 | |
| ショウジョウバエ | CAS-0001 |