title: Pediatric short bowel syndrome and subsequent development of inflammatory bowel disease: an illustrative case and literature review.
abstract: Short bowel syndrome (SBS) in neonates is an uncommon but highly morbid condition. As SBS survival increases, physiologic complications become more apparent. Few reports in the literature elucidate outcomes for adults with a pediatric history of SBS. We present a case report of a patient, born with complicated gastroschisis resulting in SBS at birth, who subsequently developed symptoms and pathologic changes of inflammatory bowel disease (IBD) as an adult. The patient lived from age 7, after a Bianchi intestinal lengthening procedure, to age 34 independent of parenteral nutrition (PN), but requiring hydration fluid via G-tube. He was then diagnosed with IBD, after presenting with weight loss, diarrhea, and malabsorption, which required resumption of PN and infliximab treatment. This report adds to a small body of the literature which points to a connection between SBS in neonates and subsequent diagnosis of IBD. Recent evidence suggests that SBS and IBD have shared features of mucosal immune dysfunction and altered intestinal microbiota. We review current treatment options for pediatric SBS as well as multidisciplinary and coordinated transition strategies. We conclude that there may be an etiologic connection between SBS and IBD and that this knowledge may impact outcomes and approaches to care.
authors.affiliation: Division of Pediatric Surgery, Department of Surgery, Children's Healthcare of Atlanta, Emory University School of Medicine, 1405 Clifton Road NE, Atlanta, GA, 30322, USA. kmccurd@emory.edu.
authors.forename: Katherine J
authors.initials: KJ
authors.lastname: Baxter
authors.affiliation: Division of General and GI Surgery, Department of Surgery, Emory University School of Medicine, Atlanta, GA, USA.
authors.forename: Jahnavi K
authors.initials: JK
authors.lastname: Srinivasan
authors.affiliation: Division of Endocrinology, Metabolism and Lipids, Department of Medicine, Emory University School of Medicine, Atlanta, GA, USA.
authors.forename: Thomas R
authors.initials: TR
authors.lastname: Ziegler
authors.affiliation: Division of Gastroenterology, Department of Medicine, Emory University School of Medicine, Atlanta, GA, USA.
authors.forename: Tanvi
authors.initials: T
authors.lastname: Dhere
authors.affiliation: Division of Pediatric Surgery, Department of Surgery, Children's Healthcare of Atlanta, Emory University School of Medicine, 1405 Clifton Road NE, Atlanta, GA, 30322, USA.
authors.forename: Richard R
authors.initials: RR
authors.lastname: Ricketts
authors.affiliation: Division of Pediatric Surgery, Department of Surgery, Children's Healthcare of Atlanta, Emory University School of Medicine, 1405 Clifton Road NE, Atlanta, GA, 30322, USA.
authors.forename: Megan M
authors.initials: MM
authors.lastname: Durham
mesh_terms: D002648:Child; D005440:Fluid Therapy; D005765:Gastrointestinal Agents / Q000627:therapeutic use; D020139:Gastroschisis / Q000150:complications*; D006801:Humans; D007231:Infant, Newborn; D015212:Inflammatory Bowel Diseases / Q000209:etiology / Q000628:therapy*; D000069285:Infliximab / Q000627:therapeutic use; D008297:Male; D010289:Parenteral Nutrition, Total; D012778:Short Bowel Syndrome / Q000209:etiology / Q000628:therapy*
chemical_list: D005765:Gastrointestinal Agents; D000069285:Infliximab
keywords: Inflammatory bowel disease; Longitudinal intestinal lengthening and tailoring procedure (Bianchi procedure); Malabsorption; Short bowel syndrome