التفاصيل البيبلوغرافية
| العنوان: |
IgLON5 deficiency produces behavioral alterations in a knockout mouse model |
| المؤلفون: |
Landa, Jon, Serafim, Ana Beatriz, Alba, Mercedes, Maudes, Estibaliz, Molina-Porcel, Laura, Garcia-Serra, Anna, Mannara, Francesco, Dalmau, Josep, Graus, Francesc, Sabater, Lidia |
| المساهمون: |
Instituto de Salud Carlos III |
| المصدر: |
Frontiers in Immunology ; volume 15 ; ISSN 1664-3224 |
| بيانات النشر: |
Frontiers Media SA |
| سنة النشر: |
2024 |
| المجموعة: |
Frontiers (Publisher - via CrossRef) |
| مصطلحات موضوعية: |
Immunology, Immunology and Allergy |
| الوصف: |
Background Anti-IgLON5 disease is a neurological disorder characterized by autoantibodies against IgLON5 and pathological evidence of neurodegeneration. IgLON5 is a cell adhesion molecule of unknown function that is highly expressed in the brain. Our aim was to investigate the impact of IgLON5 loss-of-function in evaluating brain morphology, social behavior, and the development of symptoms observed in an IgLON5 knockout (IgLON5-KO) mouse model. Methods The IgLON5-KO mice were generated using CRISPR-Cas9 technology. Immunohistochemistry on fixed sagittal brain sections and Western blotting brain lysates were used to confirm IgLON5 silencing and to evaluate the presence of other cell surface proteins. Two- month-old IgLON5-KO and wild-type (WT) mice underwent a comprehensive battery of behavioral tests to assess 1) locomotion, 2) memory, 3) anxiety, 4) social interaction, and 5) depressive-like behavior. Brain sections were examined for the presence of anatomical abnormalities and deposits of hyperphosphorylated tau in young adult (2-month-old) and aged (22-month-old) mice. Results Mice did not develop neurological symptoms reminiscent of those seen in patients with anti-IgLON5 disease. Behavioral testing revealed that 2-month-old IgLON5-KO mice showed subtle alterations in motor coordination and balance. IgLON5-KO females exhibited hyperactivity during night and day. Males were observed to have depressive-like behavior and excessive nest-building behavior. Neuropathological studies did not reveal brain morphological alterations or hyperphosphorylated tau deposits. Conclusion IgLON5-KO mice showed subtle alterations in behavior and deficits in fine motor coordination but did not develop the clinical phenotype of anti-IgLON5 disease. |
| نوع الوثيقة: |
article in journal/newspaper |
| اللغة: |
unknown |
| DOI: |
10.3389/fimmu.2024.1347948 |
| DOI: |
10.3389/fimmu.2024.1347948/full |
| الإتاحة: |
https://doi.org/10.3389/fimmu.2024.1347948Test |
| حقوق: |
https://creativecommons.org/licenses/by/4.0Test/ |
| رقم الانضمام: |
edsbas.DC5B3E23 |
| قاعدة البيانات: |
BASE |
