Skip to main content

Advertisement

Log in

Currarino Triad With Dual Pathology in the Presacral Mass

Report of a Case

  • Case Report
  • Published:
Diseases of the Colon & Rectum

Abstract

PURPOSE: Currarino triad, which comprises >anorectal stenosis, anterior sacral defect, and a presacral mass, is an uncommon cause of constipation in children and adults. The presacral mass in this triad is most often caused by an anterior sacral meningocele, a teratoma, or an enterogenous cyst, but rarely may be caused by dual pathology. A neonate with Currarino triad and dual pathology in the presacral mass is described in this report. METHOD: A male Chinese neonate, who presented with abdominal distention and constipation on the second day of life, was found to have features of Currarino triad. Colostomy was done in the neonatal period, and the presacral mass was excised by posterior sagittal perineal approach at the age of six months. RESULTS: The excised presacral mass consisted of an anterior meningocele and a teratoma. The patient continued to have constipation during follow-up and required anorectoplasty to correct residual anorectal stenosis. At the time of this report the patient was three years old and growing normally with normal anorectal function. DISCUSSION: Of a total of about 200 cases of complete Currarino triad found in the literature, in only 22 patients did the presacral mass contain both meningocele and teratoma. The features of these 22 patients and the current views on the surgical management of Currarino triad are discussed.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Similar content being viewed by others

References

  1. G Currarino D Coin T Votteler (1981) ArticleTitleTriad of anorectal, sacral and presacral anomalies Am J Radiol 137 395–398

    Google Scholar 

  2. J Kochling G Pistor MS Brands R Nasir WR Lanksh (1996) ArticleTitleThe Currarino syndrome-hereditary transmitted syndrome of anorectal, sacral and presacral anomalies. Case report and review of the literature Eur J Pediatr Surg 6 114–119

    Google Scholar 

  3. Y Iinuma M Iwafuchi M Uchiyama et al. (2000) ArticleTitleA case of Currarino triad with familial sacral bony deformities Pediatr Surg Int 16 134–135

    Google Scholar 

  4. SC Lee YS Chun SE Jung KW Park WK Kim (1997) ArticleTitleCurrarino triad; anorectal malformations, sacral bony abnormality and presacral mass A review of 11 cases J Pediatr Surg 32 58–61

    Google Scholar 

  5. DS Kirks DF Merten HC Filtson WJ Oakes (1984) ArticleTitleThe Currarino triad Pediatr Radiol 14 220–225

    Google Scholar 

  6. JA Crameri WD Ford LL Morris (1995) ArticleTitleFamilial triad of anorectal, sacrococcygeal and presacral anomalies that includes sacrococcygeal teratomas Pediatr Surg Int 10 350–353

    Google Scholar 

  7. VD Yates RS Wilroy GL Whitington JC Simmons (1983) ArticleTitleAnterior sacral defects J Pediatr 102 239–242

    Google Scholar 

  8. DS O’Riordain PR O’Connell WO Kirwan (1991) ArticleTitleHereditary sacral agenesis with presacral mass and anorectal stenosis Br J Surg 78 536–538

    Google Scholar 

  9. HA Heij CG Moorman-Voestermans A Vos CM Kneepkens (1990) ArticleTitleTriad of anorectal stenosis, sacral anomaly and presacral mass. a remediable cause of severe constipation Br J Surg 77 102–104

    Google Scholar 

  10. JL Werner H Taybi (1970) ArticleTitlePresacral masses in childhood AJR Am J Roentgenol 109 403–410

    Google Scholar 

  11. M Oren B Lorber SH Lee RC Truex SuffixJr AR Gennaro (1977) ArticleTitleAnterior sacral meningocele Dis Colon Rectum 20 492–505

    Google Scholar 

  12. KW Ashcraft T Holder (1974) ArticleTitleHereditary presacral teratoma J Pediatr Surg 9 691–697

    Google Scholar 

  13. RE Sonnino S Chou FM Guttman (1989) ArticleTitleHereditary sacrococcygeal teratomas J Pediatr Surg 24 1074–1075

    Google Scholar 

  14. H Brem BL Beaver PM Colombani et al. (1989) ArticleTitleNeonatal diagnosis of a presacral mass in the presence of congenital anal stenosis and partial sacral agenesis J Pediatr Surg 24 1076–1078

    Google Scholar 

  15. Y Tsuchida W Watanasupt T Nakajo (1989) ArticleTitleAnorectal malformations associated with a presacral tumor and sacral defect Pediatr Surg Int 4 398–402

    Google Scholar 

  16. MS Dias RG Azizkhan (1998) ArticleTitleA novel embryogenetic mechanism for Currarino’s triad Pediatr Neurosurg 28 223–229

    Google Scholar 

  17. SA Lynch PM Bond AJ Copp et al. (1995) ArticleTitleA gene for autosomal dominant sacral agenesis maps to the holoprosencephaly region at 7q36 Nat Genet 11 93–95

    Google Scholar 

  18. M Masuno K Imaizumi N Aida et al. (1996) ArticleTitleCurrarino triad with terminal deletion 7Q35-qter J Med Genet 33 877–878

    Google Scholar 

  19. M Seri G Martucciello L Paleari et al. (1999) ArticleTitleExclusion of the sonic Hedgehog gene as responsible for Currarino syndrome and anorectal malformations with sacral hypodevelopment Hum Genet 104 108–110

    Google Scholar 

  20. J Kochling M Karbasiyan A Reis (2001) ArticleTitleSpectrum of mutations and genotype-phenotype analysis in Currarino syndrome Eur J Hum Genet 9 599–605

    Google Scholar 

  21. GF Gudinchet P Maeder T Laurent B Meyrat P Schnyder (1997) ArticleTitleMagnetic resonance detection of myelodysplasia in children with Currarino triad Pediatr Radiol 27 903–907

    Google Scholar 

  22. N Otagiri Y Matsumoto Y Yoshida (2000) ArticleTitlePosterior approach for Currarino syndrome with anterior sacral meningocele J Pediatr Surg 35 1112–1114

    Google Scholar 

  23. S Somuncu E Ariturk O Iyigun et al. (1997) ArticleTitleA case of anterior sacral meningocele totally excised using the posterior sagittal approach J Pediatr Surg 32 730–732

    Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

About this article

Cite this article

Thambidorai, C.R., Muin, I., Razman, J. et al. Currarino Triad With Dual Pathology in the Presacral Mass. Dis Colon Rectum 46, 974–977 (2003). https://doi.org/10.1007/s10350-004-6696-9

Download citation

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10350-004-6696-9

Keywords

Navigation