Background & Aims: Many therapeutic attempts have demonstrated to be ineffective in reducing the severity of congenital chloride diarrhea and its long-term complications. The short-chain fatty acid butyrate stimulates intestinal water and ion absorption through a variety of mechanisms, including the activation of a parallel Cl -/butyrate and Na +/H + exchanger. In this case report, we report the therapeutic efficacy of butyrate on an 11-year-old patient affected by congenital chloride diarrhea. Methods: The efficacy of increasing doses of oral butyrate (from 50 to 100 mg/kg/day) was investigated through the daily evaluation of stool volume, bowel movements, fecal incontinence, serum, and stool electrolytes concentrations. The modifications in transepithelial intestinal ion transport elicited by butyrate were examined by rectal dialysis study. Results: A butyrate dose of 100 mg/kg/day induced a normalization of stool pattern and of serum and fecal electrolytes concentration. The rectal dialysis study demonstrated a proabsorptive effect induced by butyrate on Na +, Cl -, and K + intestinal transport. Butyrate therapy was well tolerated during the entire 12-month observation period, and the stool pattern and fecal and serum ion concentrations remained stable within the normal ranges. No clinical adverse events or episodes of dehydration requiring hospital care were observed. Conclusions: Butyrate could be effective in treating congenital chloride diarrhea. It is easily administered, useful in preventing severe dehydration episodes, and may be a promising therapeutic approach for a long-term treatment in this rare and severe condition.

Butyrate as an effective treatment of congenital chloride diarrhea / Roberto Berni, Canani; Terrin, Gianluca; Pia, Cirillo; Giuseppe, Castaldo; Francesco, Salvatore; Giuseppe, Cardillo; Anna, Coruzzo; Riccardo, Troncone. - In: GASTROENTEROLOGY. - ISSN 0016-5085. - 127:2(2004), pp. 630-634. [10.1053/j.gastro.2004.03.071]

Butyrate as an effective treatment of congenital chloride diarrhea

TERRIN, GIANLUCA;
2004

Abstract

Background & Aims: Many therapeutic attempts have demonstrated to be ineffective in reducing the severity of congenital chloride diarrhea and its long-term complications. The short-chain fatty acid butyrate stimulates intestinal water and ion absorption through a variety of mechanisms, including the activation of a parallel Cl -/butyrate and Na +/H + exchanger. In this case report, we report the therapeutic efficacy of butyrate on an 11-year-old patient affected by congenital chloride diarrhea. Methods: The efficacy of increasing doses of oral butyrate (from 50 to 100 mg/kg/day) was investigated through the daily evaluation of stool volume, bowel movements, fecal incontinence, serum, and stool electrolytes concentrations. The modifications in transepithelial intestinal ion transport elicited by butyrate were examined by rectal dialysis study. Results: A butyrate dose of 100 mg/kg/day induced a normalization of stool pattern and of serum and fecal electrolytes concentration. The rectal dialysis study demonstrated a proabsorptive effect induced by butyrate on Na +, Cl -, and K + intestinal transport. Butyrate therapy was well tolerated during the entire 12-month observation period, and the stool pattern and fecal and serum ion concentrations remained stable within the normal ranges. No clinical adverse events or episodes of dehydration requiring hospital care were observed. Conclusions: Butyrate could be effective in treating congenital chloride diarrhea. It is easily administered, useful in preventing severe dehydration episodes, and may be a promising therapeutic approach for a long-term treatment in this rare and severe condition.
2004
cld; congenital chloride diarrhea; scfas; short-chain fatty acids; slc26a3; solute carrier family 26; member 3 gene
01 Pubblicazione su rivista::01a Articolo in rivista
Butyrate as an effective treatment of congenital chloride diarrhea / Roberto Berni, Canani; Terrin, Gianluca; Pia, Cirillo; Giuseppe, Castaldo; Francesco, Salvatore; Giuseppe, Cardillo; Anna, Coruzzo; Riccardo, Troncone. - In: GASTROENTEROLOGY. - ISSN 0016-5085. - 127:2(2004), pp. 630-634. [10.1053/j.gastro.2004.03.071]
File allegati a questo prodotto
Non ci sono file associati a questo prodotto.

I documenti in IRIS sono protetti da copyright e tutti i diritti sono riservati, salvo diversa indicazione.

Utilizza questo identificativo per citare o creare un link a questo documento: https://hdl.handle.net/11573/402344
 Attenzione

Attenzione! I dati visualizzati non sono stati sottoposti a validazione da parte dell'ateneo

Citazioni
  • ???jsp.display-item.citation.pmc??? 27
  • Scopus 82
  • ???jsp.display-item.citation.isi??? 71
social impact