Pediatric Surgery International

, Volume 35, Issue 2, pp 221–226 | Cite as

Tethered cord in children with anorectal malformations with emphasis on rectobladder neck fistula

  • I. SamukEmail author
  • A. Bischoff
  • E. Freud
  • A. Pena
Original Article



To find the incidence of tethered cord (TC) in patients with anorectal malformations (ARM) and to determine the relationship between bowel/urinary control and TC in a subset of patients with rectobladder neck fistula (RBNF).


The database of a tertiary medical center was retrospectively collected for all patients treated for ARM from 1980 to 2012. All patients with TC and RBNF were identified.


Among 790 patients, who underwent screening for TC, 285(36%) were diagnosed with TC. Eleven of 37 screened patients with RBNF were diagnosed with TC. The median follow-up period was 49 months (range 2–222 months). TC was diagnosed in 3/18(16.6%) patients with sacral ratio (SR) ≥ 0.7; 4/12(33.3%) with SR 0.41–0.69; and 4/7(57.1%) patients with SR 0–0.4. The association of TC in RBNF patients had a negative influence in the prognosis for bowel and urinary control.


The incidence of TC among patients with ARM is 36%. Incidence of TC among patients with RBNF correlates with SR value and is higher with lower SR. Patients with RBNF and TC have dismal prognosis for bowel control, unrelated to their SR status. Many unresolved questions related to the management of ARM patients with asymptomatic TC still remain.


Anorectal malformations Imperforate anus Tethered cord Rectobladder neck fistula 



This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflict of interest.


  1. 1.
    Tuuha SE, Aziz D, Drake J, Wales P, Kim PC (2004) Is surgery necessary for asymptomatic tethered cord in anorectal malformation patients? J Pediatr Surg 39:773–737CrossRefPubMedGoogle Scholar
  2. 2.
    Hoffman HJ, Hendrick EB, Humphreys RP (1976) The tethered spinal cord: its protean manifestations, diagnosis and surgical correction. Childs Brain 2:145–155PubMedGoogle Scholar
  3. 3.
    Kyrklund K, Pakarinen MP, Taskinen S, Kivisaari R, Rintala RJ (2016) Spinal cord anomalies in patients with anorectal malformations without severe sacral abnormalities or meningomyelocele: outcomes after expectant, conservative management. J Neurosurg Spine 25:782–789CrossRefPubMedGoogle Scholar
  4. 4.
    Levitt MA, Patel M, Rodriguez G, Gaylin DS, Pena A (1997) The tethered spinal cord in patients with anorectal malformations. J Pediatr Surg 32:462–468CrossRefPubMedGoogle Scholar
  5. 5.
    McLone DG, La Marca F (1997) The tethered spinal cord: diagnosis, significance and management. Semin Pediatr Neurol 4:192–208CrossRefPubMedGoogle Scholar
  6. 6.
    Haro H, Komori H, Okawa A, Kawabata S, Shinomiya K (2004) Long-term outcomes of surgical treatment for tethered cord syndrome. J Spinal Disord Tech 17:16–20CrossRefPubMedGoogle Scholar
  7. 7.
    Garceau GJ (1953) The filum terminale syndrome. J Bone Jt Surg 35:711–716CrossRefGoogle Scholar
  8. 8.
    Lew SM, Kothbauer KF (2007) Tethered cord syndrome: an updated review. Pediatr Neurosurg 43:236–248CrossRefPubMedGoogle Scholar
  9. 9.
    Shweikeh F, Al-Khouja L, Nuno M. Johnson P, Drazin D, Adamo MA (2015) Disparities in clinical and economic outcomes in children and adolescents following surgery for tethered cord syndrome in the United States. J Neurosurg Pediatr 15:427–433CrossRefPubMedGoogle Scholar
  10. 10.
    Golonka NR, Haga LJ, Keating RP, Eichelberger MR, Gilbert JC, Hartman GE, Powell DM, Vezina G, Newman KD (2002) Routine MRI evaluation of low imperforate anus reveals unexpected high incidence of tethered spinal cord. J Pediatr Surg 37:966–969CrossRefPubMedGoogle Scholar
  11. 11.
    Morimoto K, Takemoto O, Wakayama A (2003) Tethered cord associated with anorectal malformation. Pediatr Neurosurg 382:79–82CrossRefGoogle Scholar
  12. 12.
    Muthukumar N, Subramaniam B, Gnanaseelan T, Rathinam R, Thiruthavadoss A (2000) Tethered cord syndrome in children with anorectal malformations. J Neurosurg 92:626–630CrossRefPubMedGoogle Scholar
  13. 13.
    Scottoni F, Iacobelli BD, Zaccara AM, Totonelli G, Schingo AM, Bagolan P (2014) Spinal ultrasound in patients with anorectal malformations: is this the end of an era? Pediatr Surg Int 30:829–831CrossRefPubMedGoogle Scholar
  14. 14.
    Long FR, Hunter JV, Mahboubi S, Kalmus A, Templeton JM (1996) Tethered cord and associated vertebral anomalies in children and infants with imperforate anus: evaluation with MR Imaging and plain radiography. Radiology 200:377–382CrossRefPubMedGoogle Scholar
  15. 15.
    Tsuda T, Iwai N, Kimura O, Kubota Y, Ono S, Saskai Y (2007) Bowel function after surgery for anorectal malformations in patients with tethered spinal cord. Pediatr Surg Int 23:1171–1174CrossRefPubMedGoogle Scholar
  16. 16.
    Van Den Hondel D, Sloots C, de Jong THR, Lequin M, Winen R (2016) Screening and treatment of tethered spinal cord in anorectal malformation patients. Eur J Pediatr Surg 26:22–26PubMedGoogle Scholar
  17. 17.
    Warf BC, Scott RM, Barnes PD, Hendren WH (1993) Tethered spinal cord in patients with anorectal and urogenital malformations. Pediatr Neurosurg 19:25–30CrossRefPubMedGoogle Scholar
  18. 18.
    Teo ATK, Gan BK, Tung JSZ, Low Y, Seow WT (2012) Low-lying spinal cord and tethered cord syndrome in children with anoretcal malformations. Singapore Med J 53:570–576PubMedGoogle Scholar
  19. 19.
    Peña A (1995) Anorectal malformations Semin. Pediatr Surg 4:35–47Google Scholar
  20. 20.
    Uchida K, Inoue M, Matsubara T, Otake K, Koike Y, Okugawa Y, Kawamoto A, Miki C, Kusunoki M (2007) Evaluation and treatment for spinal cord tethering in patients with anorectal malformations. Eur J Pediatr Surg 17:408–411CrossRefPubMedGoogle Scholar
  21. 21.
    Kim SM. Chang HK, Lee MJ, Shim KW, Oh JT, Kim DS et al (2010) Spinal dysraphism with anorectal malformation: lumbosacral magnetic resonance imaging evaluation in 120 patients. J Pediatr Surg 45:769–776CrossRefPubMedGoogle Scholar
  22. 22.
    Samuk I, Bischoff A, Hall J, Levitt M, Pena A (2016) Anorectal malformation with rectobladder neck fistula: a distinct and challenging malformation. J Pediatr Surg 51:1592–1596CrossRefPubMedGoogle Scholar
  23. 23.
    Valentini LG, Selvaggio G, Erbetta A, Cordella R, Pecoraro MG, Bova S, Boni E, Beretta E, Furlanetto M (2013) Occult spinal dysraphism: lessons learned by retrospective analysis of 149 surgical cases about natural history, surgical indications, urodynamic testing, and intraoperative neurophysiological monitoring. Childs Nerv Syst 29:1657–1669CrossRefPubMedGoogle Scholar
  24. 24.
    Von Koch CS, Quinones-Hinojosa A, Gulati M, Lyon R, Peacock WJ, Yingling CD (2002) Clinical outcome in children undergoing tethered cord release utilizing intra-operative neurophysiological monitoring. Pediatr Neurosurg 37:81–86CrossRefGoogle Scholar
  25. 25.
    Vassilyadi M, Tataryn Z, Merziotis M (2012) Retethering in children after sectioning of the filum terminale. Pediatr Neurosurg 48:335–341CrossRefPubMedGoogle Scholar
  26. 26.
    Chern JJ, Tubbs RS, Patel AJ, Gordon AS, Bandt SK, Smyth MD, Jea A, Oakes WJ (2011) Preventing cerebrospinal fluid leak following transection of a tight filum terminale. J Neurosurg Pediatr 8:35–38CrossRefPubMedGoogle Scholar
  27. 27.
    Solmaz I, Izci Y, Albayrak B, Cetinalp E, Kural C, Sengul G, Gocmez C, Pusat S, Tuzun Y (2011) Tethered cord syndrome in childhood: special emphasis on the surgical technique and review of the literature with our experience. Turk Neurosurg 21:516–521PubMedGoogle Scholar
  28. 28.
    Strong MJ, Thompson EM, Roundy N, Selden NR (2015) Use of lumbar laminoplasty vs. laminotomy for transection of the filum terminale does not affect early complication rates or postoperative course. Childs Nerv Syst 31:597–601CrossRefPubMedGoogle Scholar
  29. 29.
    Thuy M, Chaseling R, Fowler A (2015) Spinal cord detethering procedures in children: a 5 year retrospective cohort study of the early post-operative course. J Clin Neurosci 22:838–842. CrossRefPubMedGoogle Scholar
  30. 30.
    Yong RL, Habrock-Bach TH, Vaughan M, Kestle JR, Steinbok P (2011) Symptomatic retethering of the spinal cord after section of a tight filum terminale. Neurosurgery 68:1594–1602CrossRefPubMedGoogle Scholar
  31. 31.
    Boemers TM, van Gool JD, de Jong TP (1995) Tethered spinal cord: the effect of neurosurgery on the lower urinary tract and male sexual function. Br J Urol 76:747–751CrossRefPubMedGoogle Scholar
  32. 32.
    Keating MA, Rink RC, Bauer SB, Krarup C, Dyro FM, Winston KR, Shillito J, Fischer EG, Retik AB (1998) Neurological implications of the changing approach in management of occult spinal lesions. J Urol 140:1299–1301CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  1. 1.Department of Pediatric and Adolescent Surgery, Schneider Children’s Medical Center, Petach Tikva, Affiliated with the Sackler Faculty of MedicineUniversity of Tel AvivTel AvivIsrael
  2. 2.International Center for Colorectal CareChildren’s Hospital ColoradoAuroraUSA

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