PDF herunterladen
CC BY 4.0 · European J Pediatr Surg Rep. 2021; 09(01): e33-e36
DOI: 10.1055/s-0041-1726868
Case Report

Ulcerative Colitis of the Neovagina in a Toddler with Cloaca and Chronic Kidney Disease

Marta Erculiani
1   Division of Pediatric Surgery, Presidio Ospedaliero di Treviso, Treviso, Veneto, Italy
,
Cinzia Zanatta
1   Division of Pediatric Surgery, Presidio Ospedaliero di Treviso, Treviso, Veneto, Italy
,
Enrico Vidal
2   Division of Pediatrics, Presidio Ospedaliero Universitario Santa Maria della Misericordia, Udine, Friuli-Venezia Giulia, Italy
,
Stefano Martelossi
3   Division of Pediatrics, Presidio Ospedaliero di Treviso, Treviso, Veneto, Italy
,
Paola Midrio
1   Division of Pediatric Surgery, Presidio Ospedaliero di Treviso, Treviso, Veneto, Italy
› Institutsangaben
› Weitere Informationen
   Lizenzen und Reprints

Abstract

The case of a toddler with long-channel cloaca, mild chronic kidney disease (CKD) due to renal dysplasia, and early onset of ulcerative colitis (UC) is herein reported. The patient underwent definitive repair of cloaca, that included vaginal elongation with colon, at 5 months of age and was admitted for episodes of vaginal bleeding at 22 months of age. A vaginoscopy revealed a severe inflammation of the colonic neovagina. As rectal bleeding was also noticed, she underwent a colonscopy that showed the same macroscopic inflammatory picture. Neovaginal and colonic biopsies confirmed UC. The mother turned out to be affected by UC since adolescence. The patient is now on oral therapy with mesalazine and topical steroid and mesalazine in the neovagina. The association between cloaca and inflammatory bowel disease (IBD) is anecdotal, but the family history of IBD should be considered when planning the surgical reconstruction of patients with cloaca. In this patient, the occurrence of UC may require a new neovagina in the future and the concomitance of CKD may complicate the overall management due to the potential nephrotoxicity of drugs used for UC therapy.

Keywords

cloaca - neovagina - anorectal malformation - diagnosis - ulcerative - colitis


Publikationsverlauf

Eingereicht: 09. Mai 2020

Angenommen: 04. November 2020

Artikel online veröffentlicht:
18. Mai 2021

© 2021. 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 Levitt MA, Peña A. Anorectal malformations. Orphanet J Rare Dis 2007; 2: 33
    CrossrefPubMedGoogle Scholar
  • 2 Levitt MA, Peña A. Cloacal malformations: lessons learned from 490 cases. Semin Pediatr Surg 2010; 19 (02) 128-138
    CrossrefPubMedGoogle Scholar
  • 3 Peiro JL, Scorletti F, Sbragia L. Prenatal diagnosis of cloacal malformation. Semin Pediatr Surg 2016; 25 (02) 71-75
    CrossrefPubMedGoogle Scholar
  • 4 Gabarain G, Garcia-Naveiro R, Ponsky TA, Boulanger SC, Parry RL. Ulcerative colitis of the neovagina as a postsurgical complication of persistent cloaca. J Pediatr Surg 2012; 47 (01) e19-e22
    CrossrefPubMedGoogle Scholar
  • 5 Descos L, Gillon J, André C. et al. Simultaneous onset of ulcerative colitis in the rectum and in a segment of colon used for colpopoiesis: report of a case. Dis Colon Rectum 1981; 24 (07) 532-534
    CrossrefPubMedGoogle Scholar
  • 6 Froese DP, Haggitt RC, Friend WG. Ulcerative colitis in the autotransplanted neovagina. Gastroenterology 1991; 100 (06) 1749-1752
    CrossrefPubMedGoogle Scholar
  • 7 Hennigan TW, Theodorou NA. Ulcerative colitis and bleeding from a colonic vaginoplasty. J R Soc Med 1992; 85 (07) 418-419
    PubMedGoogle Scholar
  • 8 Malka D, Anquetil C, Ruszniewski P. Ulcerative colitis in a sigmoid neovagina. N Engl J Med 2000; 343 (05) 369
    CrossrefPubMedGoogle Scholar
  • 9 Lima M, Ruggeri G, Randi B. et al. Vaginal replacement in the pediatric age group: a 34-year experience of intestinal vaginoplasty in children and young girls. J Pediatr Surg 2010; 45 (10) 2087-2091
    CrossrefPubMedGoogle Scholar
  • 10 Webster T, Appelbaum H, Weinstein TA, Rosen N, Mitchell I, Levine JJ. Simultaneous development of ulcerative colitis in the colon and sigmoid neovagina. J Pediatr Surg 2013; 48 (03) 669-672
    CrossrefPubMedGoogle Scholar
  • 11 Carvalho R, Dilworth P, Docimo S, Cuffari C. Crohn disease of the neovagina and augmented bladder in a child born with cloacal exstrophy. J Pediatr Gastroenterol Nutr 2009; 48 (01) 106-109
    CrossrefPubMedGoogle Scholar
  • 12 Ungaro R, Mehandru S, Allen PB, Peyrin-Biroulet L, Colombel JF. Ulcerative colitis. Lancet 2017; 389 (10080): 1756-1770
    CrossrefPubMedGoogle Scholar
  • 13 Ordás I, Eckmann L, Talamini M, Baumgart DC, Sandborn WJ. Ulcerative colitis. Lancet 2012; 380 (9853): 1606-1619
    CrossrefPubMedGoogle Scholar
  • 14 Lynch WD, Hsu R. Ulcerative colitis. In: Statpearls. Treasure Island (FL): StatPearls Publishing LLC; 2020
    Google Scholar
  • 15 Chapman-Kiddell CA, Davies PS, Gillen L, Radford-Smith GL. Role of diet in the development of inflammatory bowel disease. Inflamm Bowel Dis 2010; 16 (01) 137-151
    CrossrefPubMedGoogle Scholar
  • 16 Cabrera JM, Sato TT. Medical and surgical management of pediatric ulcerative colitis. Clin Colon Rectal Surg 2018; 31 (02) 71-79
    Artikel in Thieme ConnectPubMedGoogle Scholar
  • 17 Kelley-Quon LI, Jen HC, Ziring DA. et al. Predictors of proctocolectomy in children with ulcerative colitis. J Pediatr Gastroenterol Nutr 2012; 55 (05) 534-540
    CrossrefPubMedGoogle Scholar
  • 18 Bischoff A, Levitt MA, Breech L, Hall J, Peña A. Vaginal switch--a useful technical alternative to vaginal replacement for select cases of cloaca and urogenital sinus. J Pediatr Surg 2013; 48 (02) 363-366
    CrossrefPubMedGoogle Scholar
  • 19 Caldwell BT, Wilcox DT. Long-term urological outcomes in cloacal anomalies. Semin Pediatr Surg 2016; 25 (02) 108-111
    CrossrefPubMedGoogle Scholar
  • 20 Mutalib M. Renal involvement in paediatric inflammatory bowel disease. Pediatr Nephrol 2019
    PubMedGoogle Scholar
  • 21 Corica D, Romano C. Renal involvement in inflammatory bowel diseases. J Crohn's Colitis 2016; 10 (02) 226-235
    CrossrefPubMedGoogle Scholar
  • 22 Oikonomou KA, Kapsoritakis AN, Stefanidis I, Potamianos SP. Drug-induced nephrotoxicity in inflammatory bowel disease. Nephron Clin Pract 2011; 119 (02) c89-c94 , discussion c96
    CrossrefPubMedGoogle Scholar