Resting energy expenditure, pulmonary
inflammation, and genotype in the
early course of cystic fibrosis
M. A. Thomson, MRCP, R. W. Wilmott, MD, C. Wainwright, FRACP,
B. Masters, MB, FRACP, P. J. Francis, MD, FRACP, and
R. W. Shepherd, MD, FRACP
From the Children's Nutrition Research Centre, Royal Children's Hospital, Brisbane, Austra-
lia, the Department of Pediatrics, Division of Pulmonary Medicine, Children's Hospital Med-
ical Center, Cincinnati, Ohio, and the Cystic FibrosisClinics, Royal Children's and Mater
Children's Hospitals, Brisbane, Australia
To further investigate the early course of cystic fibrosis (CF), specifically to exam-
ine factors contributing to energy imbalance, we examined resting energy
expenditure (REE) (by indirect calorimetry) per unit body weight and metaboli-
cally active body cell mass (by total body potassium), in relation to CF genotype
(by genomic DNA analysis), CF pancreatic phenotype, and markers of pulmonary
inflammation (from bronchoalveolar lavage fluid). Eighteen subjects with pres-
ymptomatic CF who were less than 2 years of age (n = 1I, ±F508/~F508 genotype;
n = 15, pancreatic insufficiency phenotype), identified by newborn screening,
were compared with age-, sex-, and length-matched control subjects (n = 13).
Those with the ~F508/~F508 genotype had significantly higher mean REE ex-
pressed per unit body weight (125%) and body cell mass (I 15%; p <0.03). Those
with other genotypes (n = 7) did not, as a group, have significantly different mean
REEs, but individuals with known "severe" genotypes had REEsin the high range
and those with a pancreatic-sufficient phenotype had significantly lower REEthan
those with a pancreatic-insufficient phenotype (p <0.05), and REEswere in the
normal range. Examination of bronchoalveolar lavage fluid revealed positive
culture results (7/10) but variable colony counts, neutrophil percentages, and
concentrations of interleukin-8 and interleukin-113 equally in both CF genotype
groups. These markers of pulmonary inflammation were not correlated, individ-
ually or collectively, with REEor genotype. We conclude that genotypic variations
in energy balance are detectable early in CF unrelated to lung inflammation.
Subclinical defects in body composition and pulmonary integrity occur early in
CF and, in combination with increased cellular metabolic activity, have important
clinical implications with respect to early diagnosis and management. (J Pediatr
1996; 129:367-73)
Supported in part by the Cystic Fibrosis Association of Queensland
and by the Royal Children's Hospital Foundation (Dr. Thomson,
Research Fellow).
Submitted for publication July 25, 1995; accepted April 23, 1996.
Reprint requests: R. W. Shepherd, MD, FRACP, Associate Profes-
sor, Children's Nutrition Research Centre, Royal Children's Hos-
pital, Brisbane, Queensland 4029, Australia.
Copyright © 1996 by Mosby-Year Book, Inc.
0022-3476/96/$5.00 + 0 9/21/74505
Malnutrition and nutritional growth retardation is a common
complication of cystic fibrosis known to affect prognosis
adversely.l' 2 The observation that raised energy expenditure
contributes to energy imbalance is now well documented. 3-I3
In infants with presymptomatic CF, resting energy expendi-
ture may be increased, 5' 6 and we suggested previously that
total energy expenditure is increased] although this possi-
bility is disputed. 13 In older subjects, it appears that there
may be compensation for increased REE by reducing spon-
367