title: Hypothalamic involvement and insufficient sex steroid supplementation are associated with low bone mineral density in women with childhood onset craniopharyngioma.
abstract: BACKGROUND
  Data on bone mineral density (BMD) are lacking in adults with childhood onset (CO)-craniopharyngioma (CP) with hypothalamic damage from the tumor. In patients with CO GH deficiency, BMD increases during GH treatment.
  
  
  OBJECTIVE
  The aims were to evaluate BMD in adults with CO-CPs on complete hormone replacement, including long-term GH and to evaluate the impact of hypothalamic damage on these measures.
  
  
  METHODS
  BMD (dual-energy X-ray absorptiometry), markers of bone turn over, physical activity and calcium intake were assessed in 39 CO-CP adults (20 women), with a median age of 28 (17-57) years, in comparison with matched population controls.
  
  
  RESULTS
  Late puberty induction was recorded in both genders, but reduced androgen levels in females only. Only CP women had lower BMD (P=0.03) at L2-L4, and reduced Z-scores at femoral neck (P=0.004) and L2-L4 (P=0.004). Both genders had increased serum leptin levels (P=0.001), which significantly correlated negatively with BMD at L2-L4 (P=0.003; r=-0.5) and 45% of CP women had Z-score levels ≤-2.0 s.d. Furthermore, 75% of those with a Z-score ≤-2.0 s.d. had hypothalamic involvement by the tumor. Calcium intake (P=0.008) and physical activity (P=0.007) levels were reduced in CP men only. Levels of ostecalcin and crossLaps were increased in CP men only.
  
  
  CONCLUSIONS
  Despite continuous GH therapy, low BMD was recorded in CO-CP females. Insufficient estrogen and androgen supplementation during adolescence was the main cause, but hypothalamic involvement with consequent leptin resistance was also strongly associated with low BMD in both genders.
authors.affiliation: Department of Internal Medicine, Central Hospital, SE 291 85 Kristianstad, Sweden.
authors.forename: Helene
authors.initials: H
authors.lastname: Holmer
authors.forename: Vera
authors.initials: V
authors.lastname: Popovic
authors.forename: Bertil
authors.initials: B
authors.lastname: Ekman
authors.forename: Cecilia
authors.initials: C
authors.lastname: Follin
authors.forename: Ann Britt
authors.initials: AB
authors.lastname: Siversson
authors.forename: Eva Marie
authors.initials: EM
authors.lastname: Erfurth
mesh_terms: D015502:Absorptiometry, Photon; D000293:Adolescent; D000328:Adult; D015519:Bone Density; D002136:Calcium, Dietary / Q000008:administration & dosage; D015331:Cohort Studies; D003397:Craniopharyngioma / Q000188:drug therapy / Q000503:physiopathology* / Q000601:surgery; D005260:Female; D012739:Gonadal Steroid Hormones / Q000008:administration & dosage*; D020249:Hormone Replacement Therapy; D019382:Human Growth Hormone / Q000008:administration & dosage*; D006801:Humans; D008297:Male; D008875:Middle Aged; D009043:Motor Activity; D015675:Osteocalcin / Q000097:blood; D010911:Pituitary Neoplasms; D011628:Puberty, Delayed / Q000209:etiology; D012737:Sex Factors; D013739:Testosterone / Q000097:blood
chemical_list: D002136:Calcium, Dietary; D012739:Gonadal Steroid Hormones; D015675:Osteocalcin; D019382:Human Growth Hormone; D013739:Testosterone