Zhihao Li , Ning Wang , Haobo Wang , Lingling Yang , Yong Li , Lantao Gu , Can Fu , Pengpeng Yue , Honghao Yu
{"title":"单细胞转录组学揭示了galt基因编辑诱导小鼠肺损伤的机制","authors":"Zhihao Li , Ning Wang , Haobo Wang , Lingling Yang , Yong Li , Lantao Gu , Can Fu , Pengpeng Yue , Honghao Yu","doi":"10.1016/j.bbrc.2025.151780","DOIUrl":null,"url":null,"abstract":"<div><div>Galactosemia, caused by mutations in the <em>GALT</em> gene, leads to multi-organ damage. This study investigates the impact of <em>Galt c.847 + 1G > T mutation</em> on lung tissue using single-cell transcriptomics. We employed CRISPR/Cas9 to generate a <em>Galt</em> gene-edited mouse model with the <em>Galt</em> c. 847 + 1G > T mutation and assessed <em>Galt</em> expression through PCR and Western blotting. Histopathological analysis revealed significant structural lung changes, including alveolar congestion and inflammation. Single-cell RNA sequencing demonstrated a marked reduction in immune cells (NK, T, macrophages, B cells) and an increase in alveolar type II cells, vascular endothelial cells, and myofibroblasts in the GAL mouse. The increased abundance of alveolar type II cells indicated impaired differentiation and repair. Metabolic analysis revealed significant abnormalities linked to <em>Galt c.847 + 1G > T mutation</em>, with disruptions in TGF-β1, FGF, and Mif pathways contributing to cellular dysfunction and exacerbated lung injury. This model provides insights into the molecular mechanisms of lung injury in galactosemia, highlighting significant alterations in lung cell populations and key signaling pathways.</div></div>","PeriodicalId":8779,"journal":{"name":"Biochemical and biophysical research communications","volume":"763 ","pages":"Article 151780"},"PeriodicalIF":2.5000,"publicationDate":"2025-04-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Single-cell transcriptomics reveals the mechanisms of lung injury induced by galt gene editing in mouse\",\"authors\":\"Zhihao Li , Ning Wang , Haobo Wang , Lingling Yang , Yong Li , Lantao Gu , Can Fu , Pengpeng Yue , Honghao Yu\",\"doi\":\"10.1016/j.bbrc.2025.151780\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div>Galactosemia, caused by mutations in the <em>GALT</em> gene, leads to multi-organ damage. This study investigates the impact of <em>Galt c.847 + 1G > T mutation</em> on lung tissue using single-cell transcriptomics. We employed CRISPR/Cas9 to generate a <em>Galt</em> gene-edited mouse model with the <em>Galt</em> c. 847 + 1G > T mutation and assessed <em>Galt</em> expression through PCR and Western blotting. Histopathological analysis revealed significant structural lung changes, including alveolar congestion and inflammation. Single-cell RNA sequencing demonstrated a marked reduction in immune cells (NK, T, macrophages, B cells) and an increase in alveolar type II cells, vascular endothelial cells, and myofibroblasts in the GAL mouse. The increased abundance of alveolar type II cells indicated impaired differentiation and repair. Metabolic analysis revealed significant abnormalities linked to <em>Galt c.847 + 1G > T mutation</em>, with disruptions in TGF-β1, FGF, and Mif pathways contributing to cellular dysfunction and exacerbated lung injury. This model provides insights into the molecular mechanisms of lung injury in galactosemia, highlighting significant alterations in lung cell populations and key signaling pathways.</div></div>\",\"PeriodicalId\":8779,\"journal\":{\"name\":\"Biochemical and biophysical research communications\",\"volume\":\"763 \",\"pages\":\"Article 151780\"},\"PeriodicalIF\":2.5000,\"publicationDate\":\"2025-04-11\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Biochemical and biophysical research communications\",\"FirstCategoryId\":\"99\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S0006291X25004942\",\"RegionNum\":3,\"RegionCategory\":\"生物学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"BIOCHEMISTRY & MOLECULAR BIOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Biochemical and biophysical research communications","FirstCategoryId":"99","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0006291X25004942","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"BIOCHEMISTRY & MOLECULAR BIOLOGY","Score":null,"Total":0}
Single-cell transcriptomics reveals the mechanisms of lung injury induced by galt gene editing in mouse
Galactosemia, caused by mutations in the GALT gene, leads to multi-organ damage. This study investigates the impact of Galt c.847 + 1G > T mutation on lung tissue using single-cell transcriptomics. We employed CRISPR/Cas9 to generate a Galt gene-edited mouse model with the Galt c. 847 + 1G > T mutation and assessed Galt expression through PCR and Western blotting. Histopathological analysis revealed significant structural lung changes, including alveolar congestion and inflammation. Single-cell RNA sequencing demonstrated a marked reduction in immune cells (NK, T, macrophages, B cells) and an increase in alveolar type II cells, vascular endothelial cells, and myofibroblasts in the GAL mouse. The increased abundance of alveolar type II cells indicated impaired differentiation and repair. Metabolic analysis revealed significant abnormalities linked to Galt c.847 + 1G > T mutation, with disruptions in TGF-β1, FGF, and Mif pathways contributing to cellular dysfunction and exacerbated lung injury. This model provides insights into the molecular mechanisms of lung injury in galactosemia, highlighting significant alterations in lung cell populations and key signaling pathways.
期刊介绍:
Biochemical and Biophysical Research Communications is the premier international journal devoted to the very rapid dissemination of timely and significant experimental results in diverse fields of biological research. The development of the "Breakthroughs and Views" section brings the minireview format to the journal, and issues often contain collections of special interest manuscripts. BBRC is published weekly (52 issues/year).Research Areas now include: Biochemistry; biophysics; cell biology; developmental biology; immunology
; molecular biology; neurobiology; plant biology and proteomics