Limited Alleviation of Lysosomal Acid Lipase Deficiency by Deletion of Matrix Metalloproteinase 12

Int J Mol Sci. 2024 Oct 13;25(20):11001. doi: 10.3390/ijms252011001.

Abstract

Lysosomal acid lipase (LAL) is the only known enzyme that degrades cholesteryl esters and triglycerides at an acidic pH. In LAL deficiency (LAL-D), dysregulated expression of matrix metalloproteinase 12 (MMP-12) has been described. The overexpression of MMP-12 in myeloid lineage cells causes an immune cell dysfunction resembling that of Lal knockout (Lal KO) mice. Both models develop progressive lymphocyte dysfunction and expansion of myeloid-derived suppressor (CD11b+ Gr-1+) cells. To study whether MMP-12 might be a detrimental contributor to the pathology of LAL-D, we have generated Lal/Mmp12 double knockout (DKO) mice. The phenotype of Lal/Mmp12 DKO mice closely resembled that of Lal KO mice, while the weight and morphology of the thymus were improved in Lal/Mmp12 DKO mice. Cytological examination of blood smears showed a mildly reversed lymphoid-to-myeloid shift in DKO mice. Despite significant decreases in CD11b+ Ly6G+ cells in the peripheral blood, bone marrow, and spleen of Lal/Mmp12 DKO mice, the hematopoietic bone marrow progenitor compartment and markers for neutrophil chemotaxis were unchanged. Since the overall severity of LAL-D remains unaffected by the deletion of Mmp12, we conclude that MMP-12 does not represent a viable target for treating the inflammatory pathology in LAL-D.

Keywords: CD11b+ Ly6G+ cells; LAL; chronic inflammation; lymphoid-to-myeloid shift; lysosomal storage disease; matrix metalloproteinase 12; myeloid-derived suppressor cells.

MeSH terms

  • Animals
  • CD11b Antigen / metabolism
  • Disease Models, Animal
  • Gene Deletion
  • Matrix Metalloproteinase 12* / genetics
  • Matrix Metalloproteinase 12* / metabolism
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout*
  • Neutrophils / metabolism
  • Sterol Esterase* / deficiency
  • Sterol Esterase* / genetics
  • Sterol Esterase* / metabolism
  • Wolman Disease* / genetics
  • Wolman Disease* / pathology

Substances

  • Matrix Metalloproteinase 12
  • Sterol Esterase
  • lysosomal acid lipase, mouse
  • CD11b Antigen
  • matrix metallopeptidase 12, mouse