Gastric Lactobezoar in an Infant with Cri-du-chat Syndrome

 Primary Author: Adame, Fiona DO

 Mentor(s): Jain, Sunil MD; Rupp, Richard MD

 Abstract:

Gastric lactobezoar (GLB) is a collection of acid insoluble material in enterally fed infants5. Abdominal distension and reflux are the most common presentations and are seen in two-thirds of all cases. Almost half of GLB occurs in premature infants. The pathogenesis of GLB is not clear, however several factors are considered to contribute GLB formation such as prematurity, low birth weight, and use of high caloric formula rich in fat and calcium. GLB may also be a complication of gastric hypomotility, which can be caused by various etiologies, including Cri-du-chat syndrome. Cri-du-chat syndrome is a genetic condition that presents with impaired growth and development, breathing and feeding difficulties, as well as characteristic facial appearance and hallmark high-pitched cry. We are presenting the first reported case of GLB in a premature infant with Cri-du-chat syndrome.

 References:

1 Bajorek, S., Basaldua, R., McGoogan, K., Miller, C., & Sussman, C. (2012). Neonatal Gastric Lactobezoar: Management with N-Acetylcysteine. Case Reports in Pediatrics, 2012, 3. https://doi.org/10.1155/2012/412412

 2 Bos, M., Wijnen, R., & Blaauw, I. (2013). Gastric pneumatosis and rupture caused by lactobezoar. Pediatrics International, 55(6).

 3 Heinz-Erian, P., Gassner, I., Klein-Franke, A., Jud, V., Trawoeger, R., Niederwanger, C., … Scholl-Buergi, S. (2012). Gastric lactobezoar - a rare disorder?(Review)(Clinical report). Orphanet Journal of Rare Diseases, 7.

 4 Hunt JN, Knox MT: A relation between the chain length of fatty acids and the slowing of gastric emptying. J Physiol 1968, 194:327-336

 5 Levkoff AH, Gadsden RH, Hennigar GR, Webb CM: Lactobezoar and gastric perforation in a neonate. J Pediatr 1970, 77:875-877

 6 Schreiner RL, Brady MS, Franken EA, Stevens DC, Lemons JA, Gresham EA: Increased incidence of lactobezoars in low birth weight infants. Am J Dis Child 1979, 133:936-940. 3,10,22 and Nutrition of the Preterm Infant. Edited by: Tsang RC, Uauy R, Koletzko B, Zlotkin S. Cincinnati, OH: Digital Education Publishing; 2005:415-416

 7 Sparks, B., & Kesavan, A. (2014). Treatment of a Gastric Lactobezoar with N-Acetylcysteine. Case Reports in Gastrointestinal Medicine, 2014, 3. https://doi.org/10.1155/2014/254741

 8 Wexler, H., & Poole, C. (1976). Lactobezoar, a complication of overconcentrated milk formula. Journal of Pediatric Surgery, 11(2), 261–262. https://doi.org/10.1016/0022-3468(76)90300-6

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