Gallstones in an infant with pyloric stenosis

Abstract

'Simultaneous occurrence of pyloric stenosis and multiple small gallstones has been reported only once previously, though jaundice is frequently observed in patients with congenital hypertrophic pyloric stenosis'. Gallstone disease is rare in infancy. 1-5 Usually, some underlying aetiology is identifiable when gallstones are diagnosed in younger children, for example, total parenteral nutrition, ileal resection, sepsis, dehydration etc. 2,5,6 However, only one case of gallstones with pyloric stenosis (PS) has been reported so far in the English literature.7 It is surprising as the infants with PS have two strong predisposing factors for gallstone formation; namely, hyperbilirubinemia (in 2-17% patients) 4 and dehydration and/or starvation.A 1-month-old first-born male child was brought with history of recurrent non-bilious vomiting for 10 days and presence of visible peristalsis from left to right side in epigastrium. He was born of a full-term normal delivery and had no significant antenatal history. Icterus was noted for 5 days before presentation and was confined to the face. Physical examination revealed an icteric, malnourished and moderately dehydrated infant with visible peristalsis in the epigastrium but no palpable lump. The clinical diagnosis of PS was considered and was substantiated by sonographic findings of thickened pyloric muscle (5 mm) and an elongated pyloric canal (18 mm). Another remarkable finding was the presence of multiple small stones in the gall bladder (Fig. 1). The liver architecture was normal and the common bile duct (CBD) was not dilated. Blood acid-base examination and biochemistry revealed hypokalemic alkalosis and indirect hyperbilirubinemia with a total bilirubin level of 6 mg%. Coomb's test was negative and C-reactive proteins were not increased. A diagnosis of infantile hypertrophic pyloric stenosis (IHPS) with cholelithiasis was made. Fluid resuscitation was started and once the neonate was stabilized, he underwent pyloromyotomy.At laparotomy through small transverse supra-umbilical rectus splitting incision, the diagnosis of IHPS was confirmed and Ramstedt's pyloromyotomy was performed. Multiple small gallstones (1-3 mm in size) were palpable in the gall bladder. Extrahepatic ducts including the CBD were normal. No stone was felt at the lower end of the CBD. It was decided to continue the expectant treatment for the gallstones because no congenital ductal anomaly was found. Following pyloromyotomy, feeding was resumed after 36 h. The neonate was discharged 3 days postoperatively. On follow up sonography, the gallstones were found to be decreasing in size over time and disappeared fully by 12 months. The patient is growing well, has no icterus and is still on 6 monthly review.

Secondary pseudohypoaldosteronism causing cardiopulmonary arrest and cholelithiasis

“…Cholelithiasis is rare in infancy, but several predisposing factors to cholelithiasis in infancy have been identified, such as prematurity, prolonged fasting, parenteral nutrition, use of furosemide, ceftriaxone therapy, sepsis, or dehydration . It was reported that pyloric stenosis, Bartter syndrome and PHA are related to cholelithiasis in infancy, a condition in which dehydration is likely to develop. In the current case, those risk factors were not present, except for possible chronic dehydration.…”