Annular pancreas in two consecutive siblings: an extremely rare case

Eur J Pediatr Surg. 2005 Oct;15(5):364-8. doi: 10.1055/s-2005-865838.

Abstract

Annular pancreas is the rare congenital anomaly where the pancreas forms a full or incomplete ring around the second segment of the duodenum, causing various degrees of stenosis or atresia. It is estimated that it appears in 1 out of 12 000-15 000 births of living neonates and until now, in the literature, only 6 cases have been reported among individuals of the same family. We present the case of two siblings, a boy and a girl, with annular pancreas from consecutive pregnancies of the same couple. Both neonates had a prenatal diagnosis of duodenal obstruction and they underwent duodenoduodenal, proximal transverse to distal longitudinal anastomosis. Furthermore, the girl had a mobile ascending colon. Their postoperative condition was perfect. The case we are reporting is an addition to the other 6 cases of familial presentation of annular pancreas and is similar to one of them. In these families, a total of 16 persons present this congenital anomaly while 14 are seemingly healthy. Twelve of the affected persons are female and 4 male. In conclusion, it can be stated that female individuals seem to have a greater propensity to transmit the disease to their descendants, compared to males, suggesting the possible action of an autosomal recessive sex-influenced gene. The recording of such rare family cases should be encouraged, in order to fully recognize a possible type of inherited transmission.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Anastomosis, Surgical
  • Digestive System Abnormalities / genetics
  • Duodenal Obstruction / congenital*
  • Duodenal Obstruction / diagnostic imaging
  • Duodenal Obstruction / surgery
  • Female
  • Humans
  • Infant, Newborn
  • Inheritance Patterns
  • Intestinal Atresia* / surgery
  • Male
  • Pancreas / abnormalities*
  • Pregnancy
  • Siblings
  • Ultrasonography, Prenatal