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C57BL/6JCya-Lystem1/Cya
Common Name:
Lyst-KO
Product ID:
S-KO-03031
Background:
C57BL/6JCya
Product Type
Age
Genotype
Sex
Quantity
Price:
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Basic Information
Strain Name
Lyst-KO
Strain ID
KOCMP-17101-Lyst-B6J-VA
Gene Name
Lyst
Product ID
S-KO-03031
Gene Alias
D13Sfk13; beige; bg
Background
C57BL/6JCya
NCBI ID
17101
Modification
Conventional knockout
Chromosome
13
Phenotype
MGI:107448
Document
Click here to download >>
Application
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Rare Disease Data Center >>
Note
Note: When using this mouse strain in a publication, please cite “C57BL/6JCya-Lystem1/Cya mice (Catalog S-KO-03031) were purchased from Cyagen.”
Strain Description
Ensembl Number
ENSMUST00000110559
NCBI RefSeq
NM_010748
Target Region
Exon 5
Size of Effective Region
~3.4 kb
Detailed Document
Click here to download >>
Overview of Gene Research
LYST, the lysosomal trafficking regulator, is crucial for the regulation of membrane dynamics and intracellular trafficking of lysosomes and lysosome-related organelles (LROs). Lysosomes and LROs are dynamic organelles involved in maintaining cellular hemostasis and regulating cellular functions, and LYST-mediated vesicle trafficking is essential for their proper function [1].

Mutations in the LYST gene cause Chédiak-Higashi syndrome, an autosomal recessive immunodeficiency. In LYST-deficient human neuronal models and neurons differentiated from CHS patients' induced pluripotent stem cells, there is lysosome depletion with hyperelongated tubules extruding from enlarged autolysosomes, suggesting LYST is needed for correct fission/scission of autolysosome tubules during autophagic lysosome reformation to maintain lysosomal homeostasis [2]. In a mouse model of Chediak-Higashi syndrome, LYST deficiency leads to accumulation of photoreceptor outer segment phagosomes in retinal pigment epithelial cells, reduced retinal adhesion, and elevated levels of certain proteases and oxidative stress markers [3]. The yeast LYST homolog Bph1, when absent, causes lipidated Atg8 to accumulate on endosomes, reduced selective autophagy flux, and defective endocytosis, indicating a role in maintaining endosomal functioning [4].

In conclusion, LYST is key for maintaining lysosomal and endosomal functions. Studies using models with LYST deficiency, such as human neuronal models, patient-derived iPSCs, and mouse models, have revealed its importance in autophagic lysosome reformation, retinal function, and endosomal processes. These findings contribute to understanding the pathogenesis of Chédiak-Higashi syndrome and potentially other related diseases [1,2,3,4].

References:

1. Turner, Mackenzie E, Che, Jingru, Mirhaidari, Gabriel J M, Best, Cameron A, Barker, Jenny C. 2024. The lysosomal trafficking regulator "LYST": an 80-year traffic jam. In Frontiers in immunology, 15, 1404846. doi:10.3389/fimmu.2024.1404846. https://pubmed.ncbi.nlm.nih.gov/38774881/

2. Serra-Vinardell, Jenny, Sandler, Maxwell B, De Pace, Raffaella, Sharma, Prashant, Malicdan, May Christine V. 2023. LYST deficiency impairs autophagic lysosome reformation in neurons and alters lysosome number and size. In Cellular and molecular life sciences : CMLS, 80, 53. doi:10.1007/s00018-023-04695-x. https://pubmed.ncbi.nlm.nih.gov/36707427/

3. Ji, Xiaojie, Zhao, Lihong, Umapathy, Ankita, Naggert, Jürgen K, Nishina, Patsy M. 2022. Deficiency in Lyst function leads to accumulation of secreted proteases and reduced retinal adhesion. In PloS one, 17, e0254469. doi:10.1371/journal.pone.0254469. https://pubmed.ncbi.nlm.nih.gov/35239671/

4. Vargas Duarte, Prado, Hardenberg, Ralph, Mari, Muriel, González Montoro, Ayelén, Ungermann, Christian. 2022. The yeast LYST homolog Bph1 is a Rab5 effector and prevents Atg8 lipidation at endosomes. In Journal of cell science, 135, . doi:10.1242/jcs.259421. https://pubmed.ncbi.nlm.nih.gov/35343566/

Quality Control Standard
Sperm Test

Pre-cryopreservation: Measurement of sperm concentration, determination of sperm viability.

Post-cryopreservation: A vial of cryopreserved sperms is selected for in-vitro fertilization from each batch.

Environmental Standards:SPF
Available Region:Global
Source:Cyagen
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