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Pyridostigmine in Pediatric Intestinal Pseudo-obstruction: Case Report of a 2-year Old Girl and Literature Review

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Di Nardo Giovanni, Viscogliosi Federica, Esposito Francesco, Stanghellini Vincenzo, Villa Maria Pia, Parisi Pasquale, Morlando Alessia, Calo Girolamo, De Giorgio Roberto,
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 ( Di Nardo Giovanni ) 
Sapienza University of Rome School of Medicine and Psychology

 ( Viscogliosi Federica ) 
University of Naples Federico II Department of Translational Medical Science
 ( Esposito Francesco ) 
Santobono-Pausilipon Children¡¯s Hospital Radiology Unit
 ( Stanghellini Vincenzo ) 
University of Bologna Department of Medical and Surgical Sciences
 ( Villa Maria Pia ) 
Sapienza University of Rome School of Medicine and Psychology
 ( Parisi Pasquale ) 
Sapienza University of Rome School of Medicine and Psychology
 ( Morlando Alessia ) 
University of Naples Federico II Department of Translational Medical Science
 ( Calo Girolamo ) 
University of Ferrara Department of Medical Sciences
 ( De Giorgio Roberto ) 
University of Ferrara Department of Medical Sciences

Abstract


Pediatric chronic intestinal pseudo-obstruction is a rare disorder characterized by a severe impairment of gastrointestinal motility leading to intestinal obstruction symptoms in the absence of mechanical causes. The diagnosis is usually clinical and diagnostic work is usually aimed to rule out mechanical obstruction and to identify any underlying diseases. Treatment is challenging and requires a multidisciplinary effort. In this manuscript we describe the youngest child successfully treated with the orally administrable, longacting, reversible anti-cholinesterase drug, pyridostigmine. Like other drugs belonging to cholinesterase inhibitors, pyridostigmine enhances gut motility by increasing acetylcholine availability in the enteric nervous system and neuro-muscular junctions. Based on the direct evidence from the reported case, we reviewed the current literature on the use of pyridostigmine in severe pediatric dysmotility focusing on intestinal pseudo-obstruction. The overall data emerged from the few published studies suggest that pyridostigmine is an effective and usually well tolerated therapeutic options for patients with intestinal pseudo-obstruction. More specifically, the main results obtained by pyridostigmine included marked reduction of abdominal distension, reduced need of parenteral nutrition, and improvement of oral feeding. The present case and review on pyridostigmine pave the way for eagerly awaited future randomized controlled studies testing the efficacy of cholinesterase inhibitors in pediatric severe gut dysmotility.

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Cholinesterase inhibitors; Enteric nervous system; Gastrointestinal motility; Intestinal pseudo-obstruction; Pyridostigmine bromide

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