title: Bone health of children with intestinal failure measured by dual energy X-ray absorptiometry and digital X-ray radiogrammetry.
abstract: Children with intestinal failure (IF) receiving long-term parenteral nutrition (PN) are at risk of developing low bone mineral density (BMD). Next to the dual energy X-ray absorptiometry (DXA) method, digital X-ray radiogrammetry (DXR) using the BoneXpert software has become available to obtain the Bone Health Index (BHI) in hand radiographs. In this study we 1) evaluated the prevalence of low BMD in children with IF using DXA and DXR, 2) compared DXA and DXR results, and 3) aimed to identify factors associated with low BMD.
  
  
  
  A retrospective study was performed including all children with IF between 2000 and 2015 who underwent a DXA measurement and/or a hand radiograph. Z-scores of BMD total body (BMD TB) and lumbar spine (BMD LS), bone mineral apparent density (BMAD) and bone health index (BHI) were collected. A low BMD and low BHI were defined as a Z-score ≤ -2. DXA and DXR results were compared for cases in which a DXA and hand radiograph were performed within a 6 months' interval.
  
  
  
  Forty-six children were included. Overall, 24.3% of the children had a low BMD at the first DXA at a median age of 6 years; correction for growth failure (n = 6) reduced this to 16.2%. Fifty percent had a low BHI at the first hand radiograph. Median DXA and BHI Z-scores were significantly lower than reference scores. Age, duration of PN and surgical IF were related to lower Z-scores at the first DXA. Paired DXA and DXR results (n = 18) were compared, resulting in a Cohen's kappa of 0.746 ('substantial') for BMD TB. Spearman's correlation coefficient for BHI and BMD TB Z-scores was 0.856 (p < 0.001). Hand radiography had a sensitivity of 90% and specificity of 86% (BMD TB).
  
  
  
  Up to 50% of the children had a low BMD. Children with IF have a significantly poorer bone health than the reference population, also after weaning off PN. Bone health assessment by DXA and DXR showed good agreement, especially for Z-scores ≤ -2. DXR assessment using BoneXpert software seems to be feasible for monitoring of bone health in children with IF.
authors.affiliation: Department of Pediatric Gastroenterology, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands.
authors.forename: Esther
authors.initials: E
authors.lastname: Neelis
authors.affiliation: Department of Pediatric Gastroenterology, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands.
authors.forename: Noortje
authors.initials: N
authors.lastname: Rijnen
authors.affiliation: Department of Nuclear Medicine, Erasmus Medical Center, The Netherlands.
authors.forename: Johanna
authors.initials: J
authors.lastname: Sluimer
authors.affiliation: Department of Pediatric Surgery, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands; Department of Dietetics, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands.
authors.forename: Joanne
authors.initials: J
authors.lastname: Olieman
authors.affiliation: Department of Biostatistics, Erasmus Medical Center, The Netherlands.
authors.forename: Dimitris
authors.initials: D
authors.lastname: Rizopoulos
authors.affiliation: Department of Pediatric Surgery, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands.
authors.forename: René
authors.initials: R
authors.lastname: Wijnen
authors.affiliation: Department of Pediatric Gastroenterology, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands; Department of Pediatric Gastroenterology, Leiden University Medical Center - Willem Alexander Children's Hospital, Leiden, The Netherlands.
authors.forename: Edmond
authors.initials: E
authors.lastname: Rings
authors.affiliation: Department of Pediatric Gastroenterology, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands.
authors.forename: Barbara
authors.initials: B
authors.lastname: de Koning
authors.affiliation: Department of Pediatric Gastroenterology, Erasmus Medical Center - Sophia Children's Hospital, Rotterdam, The Netherlands. Electronic address: j.hulst@erasmusmc.nl.
authors.forename: Jessie
authors.initials: J
authors.lastname: Hulst
mesh_terms: D015502:Absorptiometry, Photon / Q000379:methods*; D015519:Bone Density / Q000502:physiology*; D002648:Child; D005260:Female; D006225:Hand / Q000000981:diagnostic imaging; D006801:Humans; D007410:Intestinal Diseases / Q000150:complications / Q000178:diet therapy / Q000473:pathology*; D007422:Intestines / Q000473:pathology; D008159:Lumbar Vertebrae / Q000000981:diagnostic imaging; D008297:Male; D010024:Osteoporosis / Q000150:complications / Q000000981:diagnostic imaging*; D010288:Parenteral Nutrition; D011859:Radiography / Q000379:methods; D015203:Reproducibility of Results; D012189:Retrospective Studies; D012680:Sensitivity and Specificity; D014965:X-Rays
keywords: Bone mineral density; Digital X-ray radiogrammetry; Dual energy X-ray absorptiometry; Intestinal failure; Parenteral nutrition