Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat

S. C. Marks, A. Wojtowicz, M. Szperl, E. Urbanowska, C. A. Mackay, W. Wiktor-Jedrzejczak, E. R. Stanley, S. L. Aukerman

Research output: Contribution to journalArticle

77 Citations (Scopus)

Abstract

The toothless (tl/tl) mutation in the rat results in a paucity of osteoclasts and osteopetrosis that cannot be corrected by bone marrow transplantation. In the present study we demonstrate that tl/tl rats also have profound deficiencies of femoral, peritoneal, and pleural cavity macrophages. Further-more, the macrophage colony stimulating activity of post-endotoxin sera from tl/tl rats is substantially reduced, suggesting that, as in the case of the op mutation in mice, the basis of the tl mutation is a deficiency of the macrophage growth factor, colony stimulating factor-1 (CSF-1). Consistent with this suggestion, treatment of tl/tl rats from birth for up to six weeks with CSF-1 reduced the osteopetrosis, increased body weight, and permitted tooth eruption. In addition, CSF-1 treatment induced large numbers of osteoclasts in tl/tl bones and macrophages in the peritoneal cavity and bone marrow. Persistence of metaphyseal sclerosis, however, indicated that the disease was not totally corrected by this treatment. These studies indicate that the basis of the tl mutation is most likely another CSF-1 deficiency, and further emphasize the role of this growth factor in osteoclast differentiation.

Original languageEnglish (US)
Pages (from-to)89-93
Number of pages5
JournalBone
Volume13
Issue number1
DOIs
StatePublished - 1992

Fingerprint

Macrophage Colony-Stimulating Factor
Tooth
Osteoclasts
Macrophages
Osteopetrosis
Mutation
Peritoneal Cavity
Intercellular Signaling Peptides and Proteins
Tooth Eruption
Pleural Cavity
Sclerosis
Thigh
Bone Marrow Transplantation
Endotoxins
Therapeutics
Bone Marrow
Body Weight
Parturition
Bone and Bones
Serum

Keywords

  • Colony Stimulating Factor-1 (CSF-1)
  • Macrophage
  • Macrophage-Colony Stimulating Factor (M-CSF)
  • Osteoclast
  • Osteopetrosis
  • Rat
  • Tooth eruption

ASJC Scopus subject areas

  • Physiology
  • Hematology

Cite this

Marks, S. C., Wojtowicz, A., Szperl, M., Urbanowska, E., Mackay, C. A., Wiktor-Jedrzejczak, W., ... Aukerman, S. L. (1992). Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat. Bone, 13(1), 89-93. https://doi.org/10.1016/8756-3282(92)90365-4

Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat. / Marks, S. C.; Wojtowicz, A.; Szperl, M.; Urbanowska, E.; Mackay, C. A.; Wiktor-Jedrzejczak, W.; Stanley, E. R.; Aukerman, S. L.

In: Bone, Vol. 13, No. 1, 1992, p. 89-93.

Research output: Contribution to journalArticle

Marks, SC, Wojtowicz, A, Szperl, M, Urbanowska, E, Mackay, CA, Wiktor-Jedrzejczak, W, Stanley, ER & Aukerman, SL 1992, 'Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat', Bone, vol. 13, no. 1, pp. 89-93. https://doi.org/10.1016/8756-3282(92)90365-4
Marks, S. C. ; Wojtowicz, A. ; Szperl, M. ; Urbanowska, E. ; Mackay, C. A. ; Wiktor-Jedrzejczak, W. ; Stanley, E. R. ; Aukerman, S. L. / Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat. In: Bone. 1992 ; Vol. 13, No. 1. pp. 89-93.
@article{063fe3c9cfb844e487b41159de120505,
title = "Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat",
abstract = "The toothless (tl/tl) mutation in the rat results in a paucity of osteoclasts and osteopetrosis that cannot be corrected by bone marrow transplantation. In the present study we demonstrate that tl/tl rats also have profound deficiencies of femoral, peritoneal, and pleural cavity macrophages. Further-more, the macrophage colony stimulating activity of post-endotoxin sera from tl/tl rats is substantially reduced, suggesting that, as in the case of the op mutation in mice, the basis of the tl mutation is a deficiency of the macrophage growth factor, colony stimulating factor-1 (CSF-1). Consistent with this suggestion, treatment of tl/tl rats from birth for up to six weeks with CSF-1 reduced the osteopetrosis, increased body weight, and permitted tooth eruption. In addition, CSF-1 treatment induced large numbers of osteoclasts in tl/tl bones and macrophages in the peritoneal cavity and bone marrow. Persistence of metaphyseal sclerosis, however, indicated that the disease was not totally corrected by this treatment. These studies indicate that the basis of the tl mutation is most likely another CSF-1 deficiency, and further emphasize the role of this growth factor in osteoclast differentiation.",
keywords = "Colony Stimulating Factor-1 (CSF-1), Macrophage, Macrophage-Colony Stimulating Factor (M-CSF), Osteoclast, Osteopetrosis, Rat, Tooth eruption",
author = "Marks, {S. C.} and A. Wojtowicz and M. Szperl and E. Urbanowska and Mackay, {C. A.} and W. Wiktor-Jedrzejczak and Stanley, {E. R.} and Aukerman, {S. L.}",
year = "1992",
doi = "10.1016/8756-3282(92)90365-4",
language = "English (US)",
volume = "13",
pages = "89--93",
journal = "Bone",
issn = "8756-3282",
publisher = "Elsevier Inc.",
number = "1",

}

TY - JOUR

T1 - Administration of colony stimulating factor-1 corrects some macrophage, dental, and skeletal defects in an osteopetrotic mutation (toothless, tl) in the rat

AU - Marks, S. C.

AU - Wojtowicz, A.

AU - Szperl, M.

AU - Urbanowska, E.

AU - Mackay, C. A.

AU - Wiktor-Jedrzejczak, W.

AU - Stanley, E. R.

AU - Aukerman, S. L.

PY - 1992

Y1 - 1992

N2 - The toothless (tl/tl) mutation in the rat results in a paucity of osteoclasts and osteopetrosis that cannot be corrected by bone marrow transplantation. In the present study we demonstrate that tl/tl rats also have profound deficiencies of femoral, peritoneal, and pleural cavity macrophages. Further-more, the macrophage colony stimulating activity of post-endotoxin sera from tl/tl rats is substantially reduced, suggesting that, as in the case of the op mutation in mice, the basis of the tl mutation is a deficiency of the macrophage growth factor, colony stimulating factor-1 (CSF-1). Consistent with this suggestion, treatment of tl/tl rats from birth for up to six weeks with CSF-1 reduced the osteopetrosis, increased body weight, and permitted tooth eruption. In addition, CSF-1 treatment induced large numbers of osteoclasts in tl/tl bones and macrophages in the peritoneal cavity and bone marrow. Persistence of metaphyseal sclerosis, however, indicated that the disease was not totally corrected by this treatment. These studies indicate that the basis of the tl mutation is most likely another CSF-1 deficiency, and further emphasize the role of this growth factor in osteoclast differentiation.

AB - The toothless (tl/tl) mutation in the rat results in a paucity of osteoclasts and osteopetrosis that cannot be corrected by bone marrow transplantation. In the present study we demonstrate that tl/tl rats also have profound deficiencies of femoral, peritoneal, and pleural cavity macrophages. Further-more, the macrophage colony stimulating activity of post-endotoxin sera from tl/tl rats is substantially reduced, suggesting that, as in the case of the op mutation in mice, the basis of the tl mutation is a deficiency of the macrophage growth factor, colony stimulating factor-1 (CSF-1). Consistent with this suggestion, treatment of tl/tl rats from birth for up to six weeks with CSF-1 reduced the osteopetrosis, increased body weight, and permitted tooth eruption. In addition, CSF-1 treatment induced large numbers of osteoclasts in tl/tl bones and macrophages in the peritoneal cavity and bone marrow. Persistence of metaphyseal sclerosis, however, indicated that the disease was not totally corrected by this treatment. These studies indicate that the basis of the tl mutation is most likely another CSF-1 deficiency, and further emphasize the role of this growth factor in osteoclast differentiation.

KW - Colony Stimulating Factor-1 (CSF-1)

KW - Macrophage

KW - Macrophage-Colony Stimulating Factor (M-CSF)

KW - Osteoclast

KW - Osteopetrosis

KW - Rat

KW - Tooth eruption

UR - http://www.scopus.com/inward/record.url?scp=0026608325&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=0026608325&partnerID=8YFLogxK

U2 - 10.1016/8756-3282(92)90365-4

DO - 10.1016/8756-3282(92)90365-4

M3 - Article

C2 - 1581113

AN - SCOPUS:0026608325

VL - 13

SP - 89

EP - 93

JO - Bone

JF - Bone

SN - 8756-3282

IS - 1

ER -