PDF herunterladen
CC BY 4.0 · European J Pediatr Surg Rep. 2022; 10(01): e45-e48
DOI: 10.1055/s-0041-1742154
Case Report

Hindgut Duplication in an Infant with Omphalocele–Exstrophy–Imperforate Anus–Spinal Defects (OEIS) Complex

Timothy F. Tirrell 
1   Department of Surgery, Boston Children's Hospital, Boston, Massachusetts, United States
,
Farokh R. Demehri
1   Department of Surgery, Boston Children's Hospital, Boston, Massachusetts, United States
,
Craig W. Lillehei
1   Department of Surgery, Boston Children's Hospital, Boston, Massachusetts, United States
,
Joseph G. Borer
2   Department of Urology, Boston Children's Hospital, Boston, Massachusetts, United States
,
Benjamin C. Warf
3   Department of Neurosurgery, Boston Children's Hospital, Boston, Massachusetts, United States
,
Belinda H. Dickie
1   Department of Surgery, Boston Children's Hospital, Boston, Massachusetts, United States
› Institutsangaben
› Weitere Informationen
Auch verfügbar auf
   Lizenzen und Reprints

Abstract

Introduction The congenital anomaly of omphalocele, cloacal exstrophy, imperforate anus, and spinal abnormalities (OEIS complex) is rare but well recognized. Hindgut duplications are also uncommon and are not known to be associated with OEIS. We describe a neonate with OEIS who was found to have fully duplicated blind-ending hindguts.

Case Report A premature infant boy with OEIS underwent first-stage closure on day of life 6, which included excision of the omphalocele sac, separation of the cecal plate and bladder halves, tubularization of the cecal plate, hindgut rescue with end colostomy, and joining of the bladder halves. Cecal plate inspection revealed two hindgut structures that descended distally, one descended midline into the pelvis along the sacrum and the second laterally along the left border of the sacrum. Both lumens connected to the cecal plate and had separate mesenteries. In an effort to maximize the colonic mucosal surface area, the hindgut segments were unified through a side-to-side anastomosis, creating a larger caliber hindgut. The cecal plate was tubularized and an end colostomy was created. Bowel function returned and he was discharged home on full enteral feeds.

Discussion This case represents a cooccurrence of two extremely rare and complex congenital anomalies. The decision to unify the distinct hindguts into a single lumen was made in an effort to combine the goals of management for both OEIS and alimentary duplications. The hindgut is abnormal in OEIS and should be assessed carefully during repair.

Keywords

OEIS - cloacal exstrophy - enteric duplication


Publikationsverlauf

Eingereicht: 12. Mai 2020

Angenommen: 16. Oktober 2020

Artikel online veröffentlicht:
10. März 2022

© 2022. The Author(s). This is an open access article published by Thieme under the terms of the Creative Commons Attribution License, permitting unrestricted use, distribution, and reproduction so long as the original work is properly cited. (https://creativecommons.org/licenses/by/4.0/)

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Rickham PP. Vesico-intestinal fissure. Arch Dis Child 1960; 35 (179) 97-102
    CrossrefPubMedGoogle Scholar
  • 2 Lund DP, Hendren WH. Cloacal exstrophy: a 25-year experience with 50 cases. J Pediatr Surg 2001; 36 (01) 68-75
    CrossrefPubMedGoogle Scholar
  • 3 Davidoff AM, Hebra A, Balmer D, Templeton Jr. JM, Schnaufer L. Management of the gastrointestinal tract and nutrition in patients with cloacal exstrophy. J Pediatr Surg 1996; 31 (06) 771-773
    CrossrefPubMedGoogle Scholar
  • 4 Sawaya D, Gearhart JP. Gastrointestinal reconstruction and outcomes for patients with the OEIS complex. Semin Pediatr Surg 2011; 20 (02) 123-125
    CrossrefPubMedGoogle Scholar
  • 5 Husmann DA, McLorie GA, Churchill BM, Ein SH. Management of the hindgut in cloacal exstrophy: terminal ileostomy versus colostomy. J Pediatr Surg 1988; 23 (12) 1107-1113
    CrossrefPubMedGoogle Scholar
  • 6 Russell KW, Holcomb III GW. Alimentary tract duplications. In: Holcomb III GW, Murphy JP, St Peter SD. eds. Ashcraft's Pediatric Surgery. 7th ed.. New York, NY: Elsevier; 2020: 629-640
    Google Scholar
  • 7 Ravitch MM. Hind gut duplication; doubling of colon and genital urinary tracts. Ann Surg 1953; 137 (05) 588-601
    CrossrefPubMedGoogle Scholar
  • 8 Hsu H, Gueng MK, Tseng YH, Wu CC, Liu PH, Chen CCC. Adenocarcinoma arising from colonic duplication cyst with metastasis to omentum: A case report. J Clin Ultrasound 2011; 39 (01) 41-43
    CrossrefPubMedGoogle Scholar
  • 9 Orr MM, Edwards AJ. Neoplastic change in duplications of the alimentary tract. Br J Surg 1975; 62 (04) 269-274
    CrossrefPubMedGoogle Scholar
  • 10 Blank G, Königsrainer A, Sipos B, Ladurner R. Adenocarcinoma arising in a cystic duplication of the small bowel: case report and review of literature. World J Surg Oncol 2012; 10: 55
    CrossrefPubMedGoogle Scholar
  • 11 Emery JA, Campbell JR, Hodges CV. Duplication of the hindgut: low male imperforate anus and unilateral exstrophy of the bladder. J Urol 1974; 112 (04) 532-535
    CrossrefPubMedGoogle Scholar
  • 12 Haq S, Nasrullah A, Ahmed I, Ghazanfar H, Sheikh AB, Akhtar A. Hindgut duplication: a unique case of six perineal openings. Cureus 2017; 9 (07) e1433
    PubMedGoogle Scholar
  • 13 Taghizadeh A, Qteishat A, Cuckow PM. Restoring hindgut continuity in cloacal exstrophy: a valuable method of optimising bowel length. Eur J Pediatr Surg 2009; 19 (03) 141-144
    Artikel in Thieme ConnectPubMedGoogle Scholar
  • 14 Fullerton BS, Sparks EA, Hall AM. et al. Growth morbidity in patients with cloacal exstrophy: a 42-year experience. J Pediatr Surg 2016; 51 (06) 1017-1021
    CrossrefPubMedGoogle Scholar