Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor

W. Wiktor-Jedrzejczak, E. Urbanowska, S. L. Aukerman, J. W. Pollard, E. R. Stanley, P. Ralph, A. A. Ansari, K. W. Sell, M. Szperl

Research output: Contribution to journalArticle

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Abstract

Mice that are mutant at the op locus have a severe deficiency of mononuclear phagocytes due to an inactivating mutation in the CSF-1 (macrophage colony-stimulating factor, M-CSF) gene. op/op mice are toothless, possessing skeletal abnormalities, a low body weight, and compromised fertility; they are osteopetrotic due to a deficiency of osteoclasts. The congenital osteopetrosis, toothless phenotype, osteoclast deficit, and the defects in splenic and femoral macrophages were corrected by routes of administration of human recombinant CSF-1 that maintained normal circulating CSF-1 concentrations. Early restoration of circulating CSF-1 was required for rescue of the toothless phenotype, but only partially restored body weight. In contrast, the deficiencies of pleural and peritoneal cavity macrophages and the reduced female fertility were not corrected by restoration of circulating CSF-1. These results suggest that although circulating CSF-1 is required for osteoclast and macrophage production, local synthesis and action of the growth factor are important for certain target cell populations.

Original languageEnglish (US)
Pages (from-to)1049-1054
Number of pages6
JournalExperimental Hematology
Volume19
Issue number10
StatePublished - 1991
Externally publishedYes

Fingerprint

Macrophage Colony-Stimulating Factor
Intercellular Signaling Peptides and Proteins
Osteoclasts
Fertility
Macrophages
Body Weight
Osteopetrosis
Phenotype
Pleural Cavity
Health Services Needs and Demand
Peritoneal Cavity
Peritoneal Macrophages
Phagocytes
Thigh
Mutation
Genes

Keywords

  • CSF-1
  • macrophages
  • op/op mice
  • osteopetrosis

ASJC Scopus subject areas

  • Cancer Research
  • Cell Biology
  • Genetics
  • Hematology
  • Oncology
  • Transplantation

Cite this

Wiktor-Jedrzejczak, W., Urbanowska, E., Aukerman, S. L., Pollard, J. W., Stanley, E. R., Ralph, P., ... Szperl, M. (1991). Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor. Experimental Hematology, 19(10), 1049-1054.

Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor. / Wiktor-Jedrzejczak, W.; Urbanowska, E.; Aukerman, S. L.; Pollard, J. W.; Stanley, E. R.; Ralph, P.; Ansari, A. A.; Sell, K. W.; Szperl, M.

In: Experimental Hematology, Vol. 19, No. 10, 1991, p. 1049-1054.

Research output: Contribution to journalArticle

Wiktor-Jedrzejczak, W, Urbanowska, E, Aukerman, SL, Pollard, JW, Stanley, ER, Ralph, P, Ansari, AA, Sell, KW & Szperl, M 1991, 'Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor', Experimental Hematology, vol. 19, no. 10, pp. 1049-1054.
Wiktor-Jedrzejczak W, Urbanowska E, Aukerman SL, Pollard JW, Stanley ER, Ralph P et al. Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor. Experimental Hematology. 1991;19(10):1049-1054.
Wiktor-Jedrzejczak, W. ; Urbanowska, E. ; Aukerman, S. L. ; Pollard, J. W. ; Stanley, E. R. ; Ralph, P. ; Ansari, A. A. ; Sell, K. W. ; Szperl, M. / Correction by CSF-1 of defects in the osteopetrotic op/op mouse suggests local, developmental, and humoral requirements for this growth factor. In: Experimental Hematology. 1991 ; Vol. 19, No. 10. pp. 1049-1054.
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