Massachusetts Chapter of the American College of Surgeons

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Pubertal Onset and Metabolic Bone Disease in Pediatric Intestinal Failure
Emily Nes, MD1,2, Katherine Culbreath, MD1,2, Gregory Keefe, MD1,2, Steven J. Staffa, MS2, Sam M. Han, MD1,2, E. Reese, MPH, RDN, LDN1,4, Biren P. Modi, MD, MPH1,2, Tom Jaksic, MD, PhD1,2, Christopher P. Duggan, MD, MPH1, Christina M. Jacobsen, MD, PhD3, Alexandra N. Carey, MD1,4
1Center for Advanced Intestinal Rehabilitation, Boston Children’s Hospital and Harvard Medical School, Boston, MA, USA; 2Department of Surgery, Boston Children’s Hospital and Harvard Medical School, Boston, MA, USA; 3Division of Endocrinology, Boston Children’s Hospital and Harvard Medical School, Boston, MA, USA; 4Division of Gastroenterology, Hepatology, and Nutrition, Boston Children’s Hospital and Harvard Medical School, Boston, MA, USA

Background: Bone mineral density (BMD) increases post-puberty in idiopathic juvenile osteoporosis (IJO); however, the effect on pediatric intestinal failure (IF) is unknown. This study aimed to identify the impact of puberty on BMD in pediatric IF.
Methods: This retrospective review of children in an interdisciplinary intestinal rehabilitation program included those with pediatric IF dependent on PN, who underwent puberty, and had at least two Dual-Energy X-Ray Absorptiometry scans. Metabolic bone disease (MBD) was defined as BMD Z-scores ? -2. Univariate and multivariable analyses identified predictors of MBD in pediatric IF.
Results: Forty-seven patients were evaluated. Thirty-three (70.2%) had MBD. On univariate analysis, predictors of lower BMD Z-scores included PN dependence (p=0.019), non-ambulatory status (p=0.02), and chronic intestinal pseudo-obstruction (p=0.004). Predictors of higher BMD Z-scores included higher weight-for-age Z-score (WAZ, p<0.001), height-for-age Z-score (p=0.002) and bone age Z-score (p=0.009), and midgut or segmental volvulus (p=0.018). Pubertal onset did not significantly impact BMD Z-scores (p=0.101), however, BMD Z-scores significantly worsened over time (p=0.014, Figure 1). Multivariable analysis identified WAZ as the only significant independent factor, with higher WAZ associated with higher BMD Z-scores (p<0.001).
Conclusion: Contrary to IJO, BMD Z-scores in pediatric IF continue to worsen over time despite pubertal onset. Higher WAZ was associated with higher BMD Z-scores in this population. This finding emphasizes the importance of nutritional optimization to promote weight gain and mitigate MBD risk in pediatric IF.


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