Mineral Content of Calcified Tissues
in Cystic Fibrosis Mice
**
LARA R. GAWENIS,
1
P AULETTE SPENCER,
4
LAURA S. HILLMAN,
3
MATTHEW C. HARLINE,
1
J. STEVEN
MORRIS,
2
AND LANE L. CLARKE*
,1
1
Dalton Cardiovascular Research Center and the Department
of Veterinary Biomedical Sciences,
2
Missouri University Research
Reactor Center,
3
Department of Child Health, University of
Missouri–Columbia, Columbia, MO 65211, and
4
the Department of Oral Biology, University of Missouri–Kansas
City School of Dentistry, Kansas City, MO 64108
Received December 26, 2000; Accepted February 16, 2001
ABSTRACT
Although abnormal hard tissue mineralization is a recognized
complication of cystic fibrosis (CF), the pathogenesis leading from the
defective cystic fibrosis transmembrane conductance regulator (CFTR)
protein is poorly understood. We hypothesized that CFTR plays a
direct role in the mineralization of bone and teeth and tested the
hypothesis using CF mouse models [CFTR(–) mice]. In vivo measure-
ments by dual-emission X-ray absorpitometry (DEXA) indicated that
bone mineral density (BMD) was reduced in CF mice as compared to
gender-matched littermates. However, no change was evident after
correction of BMD for the covariant of body weight. The latter finding
was confirmed in isolated femurs and nasal bones by standard dry-
ashing and instrumental neutron activation analysis (INAA). INAA of
the continuously growing hypsodont incisor teeth from CFTR(–) mice
revealed reduced Ca and normal P in the enamel layer—a finding con-
sistent with changes in the deciduous teeth of CF children. Interest-
ingly, enamel fluoride was increased in the CFTR(–) incisors and may
associate with abnormal enamel crystallite formation. The iron content
of the incisor enamel was reduced, explaining the loss of yellow pig-
mentation in CFTR(–) incisors. In contrast to the incisors, the mineral
© Copyright 2001 by Humana Press Inc.
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* Author to whom all correspondence and reprint requests should be addressed.
** Preliminary reports published in Pediatric Pulmonology, 14, 253A (1997)
and 15, 253A (1998).
Biological Trace Element Research 69 Vol. 83, 2001