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Child's Nervous System
Published in: Child's Nervous System 4/2024

Open Access 11-12-2023 | Fecal Incontinence | Research

Disparities in indications and outcomes reporting for pediatric tethered cord surgery: The need for a standardized outcome assessment tool

Authors: Matthew C. Findlay, Samuel Tenhoeve, Skyler A. Terry, Rajiv R. Iyer, Douglas L. Brockmeyer, Michael P. Kelly, John R. W. Kestle, David Gonda, Vijay M. Ravindra

Published in: Child's Nervous System | Issue 4/2024

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Abstract

Purpose

Tethered cord syndrome (TCS) is characterized by abnormal attachment of the spinal cord neural elements to surrounding tissues. The most common symptoms include pain, motor or sensory dysfunction, and urologic deficits. Although TCS is common in children, there is a significant heterogeneity in outcomes reporting. We systematically reviewed surgical indications and postoperative outcomes to assess the need for a grading/classification system.

Methods

PubMed and EMBASE searches identified pediatric TCS literature published between 1950 and 2023. Studies reporting surgical interventions, ≥ 6-month follow-up, and ≥ 5 patients were included.

Results

Fifty-five studies representing 3798 patients were included. The most commonly reported non-urologic symptoms were nonspecific lower-extremity motor disturbances (36.4% of studies), lower-extremity/back pain (32.7%), nonspecific lower-extremity sensory disturbances (29.1%), gait abnormalities (29.1%), and nonspecific bowel dysfunction/fecal incontinence (25.5%). Urologic symptoms were most commonly reported as nonspecific complaints (40.0%). After detethering surgery, retethering was the most widely reported non-urologic outcome (40.0%), followed by other nonspecific findings: motor deficits (32.7%), lower-extremity/back/perianal pain (18.2%), gait/ambulation function (18.2%), sensory deficits (12.7%), and bowel deficits/fecal incontinence (12.7%). Commonly reported urologic outcomes included nonspecific bladder/urinary deficits (27.3%), bladder capacity (20.0%), bladder compliance (18.2%), urinary incontinence/enuresis/neurogenic bladder (18.2%), and nonspecific urodynamics/urodynamics score change (16.4%).

Conclusion

TCS surgical literature is highly variable regarding surgical indications and reporting of postsurgical outcomes. The lack of common data elements and consistent quantitative measures inhibits higher-level analysis. The development and validation of a standardized outcomes measurement tool—ideally encompassing both patient-reported outcome and objective measures—would significantly benefit future TCS research and surgical management.
Literature
1.
Yamada S, Iacono RP, Andrade T, Mandybur G, Yamada BS (1995) Pathophysiology of tethered cord syndrome. Neurosurg Clin N Am 6:311–323PubMedCrossRef
2.
Yamada S, Won DJ, Yamada SM (2004) Pathophysiology of tethered cord syndrome: correlation with symptomatology. Neurosurg Focus 16(2):E6PubMedCrossRef
3.
Yamada S, Zinke DE, Sanders D (1981) Pathophysiology of “tethered cord syndrome.” J Neurosurg 54:494–503PubMedCrossRef
4.
Agarwalla PK, Dunn IF, Scott RM, Smith ER (2007) Tethered cord syndrome. Neurosurg Clin N Am 18:531–547PubMedCrossRef
5.
Barutçuoğlu M, Selçuki M, Umur AS, Mete M, Gurgen SG, Selcuki D (2016) Scoliosis may be the first symptom of the tethered spinal cord. Indian J Orthop 50:80–86PubMedPubMedCentralCrossRef
6.
Jackson T, Jones A, Miller N, Georgopoulos G (2019) Clubfoot and tethered cord syndrome: results of treatment with the Ponseti Method. J Pediatr Orthop 39:318–321PubMedCrossRef
7.
Lew SM, Kothbauer KF (2007) Tethered cord syndrome: an updated review. Pediatr Neurosurg 43:236–248PubMedCrossRef
8.
Shukla M, Sardhara J, Sahu RN, Sharma P, Behari S, Jaiswal AK, Srivastava AK, Mehrotra A, Das KK, Bhaisora KS (2018) Adult versus pediatric tethered cord syndrome: clinicoradiological differences and its management. Asian J Neurosurg 13:264–270PubMedPubMedCentralCrossRef
9.
Bhimani AD, Selner AN, Patel JB, Hobbs JG, Esfahani DR, Behbahani M, Zayyad Z, Nikas D, Mehta AI (2019) Pediatric tethered cord release: an epidemiological and postoperative complication analysis. Journal of Spine Surgery 5:337–350PubMedPubMedCentralCrossRef
10.
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 21:516–521PubMed
11.
Hoffman HJ, Hendrick EB, Humphreys RP (1976) The tethered spinal cord: its protean manifestations, diagnosis and surgical correction. Childs Brain 2:145–155PubMed
12.
13.
McVeigh LG, Anokwute MC, Chen S, Jea A (2022) Spinal column shortening for tethered cord syndrome: a systematic review and individual patient data meta-analysis. J Neurosurg Pediatr 29:624–633PubMedCrossRef
14.
Stewart LA, Clarke M, Rovers M, Riley RD, Simmonds M, Stewart G, Tierney JF (2015) Preferred reporting items for systematic review and meta-analyses of individual participant data: the PRISMA-IPD Statement. JAMA 313:1657–1665PubMedCrossRef
15.
16.
Abrahamsson K, Olsson I, Sillén U (2007) Urodynamic findings in children with myelomeningocele after untethering of the spinal cord. J Urol 177: 331–334. discussion 334
17.
Altiok H, Riordan A, Graf A, Krzak J, Hassani S (2016) Response of scoliosis in children with myelomeningocele to surgical release of tethered spinal cord. Top Spinal Cord Inj Rehabil 22:247–252PubMedPubMedCentralCrossRef
18.
Alzahrani A, Alsowayan O, Farmer JP, Capolicchio JP, Jednak R, El-Sherbiny M (2016) Comprehensive analysis of the clinical and urodynamic outcomes of secondary tethered spinal cord before and after spinal cord untethering. J Pediatr Urol 12:101.e101-106CrossRef
19.
Balkan E, Kiliç N, Avşar I, Boyaci S, Aksoy K, Doğruyol H (2001) Urodynamic findings in the tethered spinal cord: the effect of tethered cord division on lower urinary tract functions. Eur J Pediatr Surg 11:116–119PubMedCrossRef
20.
Beaumont A, Muszynski CA, Kaufman BA (2007) Clinical significance of terminal syringomyelia in association with pediatric tethered cord syndrome. Pediatr Neurosurg 43:216–221PubMedCrossRef
21.
Behbahani M, Shlobin N, Rosen C, Yerkes E, Swaroop V, Lam S, Bowman R (2020) Multidisciplinary management of tethered spinal cord syndrome in children: operationalizing an outpatient patient-centered workflow. J Multidiscip Healthc 13:1283–1290PubMedPubMedCentralCrossRef
22.
Broderick KM, Munoz O, Herndon CD, Joseph DB, Kitchens DM (2015) Utility of urodynamics in the management of asymptomatic tethered cord in children. World J Urol 33:1139–1142PubMedCrossRef
23.
Cha S, Wang KC, Park K, Shin HI, Lee JY, Chong S, Kim K (2018) Predictive value of intraoperative bulbocavernosus reflex during untethering surgery for post-operative voiding function. Clin Neurophysiol 129:2594–2601PubMedCrossRef
24.
Chern JJ, Dauser RC, Whitehead WE, Curry DJ, Luerssen TG, Jea A (2011) The effect of tethered cord release on coronal spinal balance in tight filum terminale. Spine (Phila Pa 1976) 36: E944–949
25.
Cornette L, Verpoorten C, Lagae L, Van Calenbergh F, Plets C, Vereecken R, Casaer P (1998) Tethered cord syndrome in occult spinal dysraphism: timing and outcome of surgical release. Neurology 50:1761–1765PubMedCrossRef
26.
Destro F, Canazza L, Meroni M, Selvaggio G, Parazzini C, Valentini L, Riccipetitoni G (2018) Tethered cord and anorectal malformations: a case series. Eur J Pediatr Surg 28:484–490PubMedCrossRef
27.
Edström E, Wesslén C, Fletcher-Sandersjöö A, Elmi-Terander A, Sandvik U (2022) Filum terminale transection in pediatric tethered cord syndrome: a single center, population-based, cohort study of 95 cases. Acta Neurochir (Wien) 164:1473–1480PubMedCrossRef
28.
Erkan K, Unal F, Kiris T, Karalar T (2000) Treatment of terminal syringomyelia in association with tethered cord syndrome: clinical outcomes with and without syrinx drainage. Neurosurg Focus 8(3):E9PubMedCrossRef
29.
Geyik M, Geyik S, Şen H, Pusat S, Alptekin M, Yılmaz AE, Nazik M, Erkutlu İ (2016) Urodynamic outcomes of detethering in children: experience with 46 pediatric patients. Childs Nerv Syst 32:1079–1084PubMedCrossRef
30.
Glenn C, Cheema AA, Safavi-Abbasi S, Gross NL, Martin MD, Mapstone TB (2015) Spinal cord detethering in children with tethered cord syndrome and Chiari type 1 malformations. J Clin Neurosci 22:1749–1752PubMedCrossRef
31.
Guerra LA, Pike J, Milks J, Barrowman N, Leonard M (2006) Outcome in patients who underwent tethered cord release for occult spinal dysraphism. J Urol 176:1729–1732PubMedCrossRef
32.
Hayashi T, Kimiwada T, Kohama M, Shirane R, Tominaga T (2018) Minimally invasive surgical approach to filum lipoma. Neurol Med Chir (Tokyo) 58:132–137PubMedCrossRef
33.
Hayashi T, Kimiwada T, Shirane R, Tominaga T (2022) Retethering risk in pediatric spinal lipoma of the conus medullaris. J Neurosurg Pediatr 29:342–349PubMedCrossRef
34.
Inoue M, Uchida K, Otake K, Nagano Y, Shimura T, Hashimoto K, Matsushita K, Koike Y, Matsubara T, Kusunoki M (2017) Long-term functional outcome after untethering surgery for a tethered spinal cord in patients with anorectal malformations. Pediatr Surg Int 33:995–999PubMedCrossRef
35.
Lagae L, Verpoorten C, Casaer P, Vereecken R, Fabry G, Plets C (1990) Conservative versus neurosurgical treatment of tethered cord patients. Z Kinderchir 45(Suppl 1):16–17PubMed
36.
Lavallée LT, Leonard MP, Dubois C, Guerra LA (2013) Urodynamic testing—is it a useful tool in the management of children with cutaneous stigmata of occult spinal dysraphism? J Urol 189:678–683PubMedCrossRef
37.
Lee JY, Phi JH, Cheon JE, Kim SK, Kim IO, Cho BK, Wang KC (2012) Preuntethering and postuntethering courses of syringomyelia associated with tethered spinal cord. Neurosurgery 71:23–29PubMedCrossRef
38.
Lee SB, Im YJ, Jung JH, Do MT, Lee JY, Wang KC, Park K (2022) Clinical and urodynamic features of secondary tethered cord syndrome: how can they be found longitudinally? Neurourol Urodyn 41:365–374PubMedCrossRef
39.
McGirt MJ, Mehta V, Garces-Ambrossi G, Gottfried O, Solakoglu C, Gokaslan ZL, Samdani A, Jallo GI (2009) Pediatric tethered cord syndrome: response of scoliosis to untethering procedures. J Neurosurg Pediatr 4:270–274PubMedCrossRef
40.
McLone DG, Herman JM, Gabrieli AP, Dias L (1990) Tethered cord as a cause of scoliosis in children with a myelomeningocele. Pediatr Neurosurg 16:8–13PubMedCrossRef
41.
Mehta VA, Gottfried ON, McGirt MJ, Gokaslan ZL, Ahn ES, Jallo GI (2011) Safety and efficacy of concurrent pediatric spinal cord untethering and deformity correction. J Spinal Disord Tech 24:401–405PubMedCrossRef
42.
Metcalfe PD, Luerssen TG, King SJ, Kaefer M, Meldrum KK, Cain MP, Rink RC, Casale AJ (2006) Treatment of the occult tethered spinal cord for neuropathic bladder: results of sectioning the filum terminale. J Urol 176: 1826–1829. discussion 1830
43.
Meyrat BJ, Vernet O, Berger D, de Tribolet N (1993) Pre- and postoperative urodynamic and anorectal manometric findings in children operated upon for a primary tethered cord. Eur J Pediatr Surg 3:309–312PubMedCrossRef
44.
Mualem W, Nathani KR, Durrani S, Zamanian C, Ghaith AK, Michalopoulos GD, Rotter J, Daniels D, Bydon M (2023) Utilizing pre- and postoperative radiological parameters to predict surgical outcomes following untethering for tethered cord syndrome in a pediatric population. J Neurosurg Pediatr 31:159–168PubMedCrossRef
45.
Nogueira M, Greenfield SP, Wan J, Santana A, Li V (2004) Tethered cord in children: a clinical classification with urodynamic correlation. J Urol 172:1677–1680. discussion 1680
46.
Ogiwara H, Joko M, Takado M, Uematsu K, Kameda M, Sasaki N, Kitagawa M, Morota N (2015) Duration of the horizontal decubitus position for prevention of cerebrospinal fluid leakage following transection of a tight filum terminale. J Neurosurg Pediatr 15:461–464PubMedCrossRef
47.
Ostling LR, Bierbrauer KS, Ct K (2012) Outcome, reoperation, and complications in 99 consecutive children operated for tight or fatty filum. World Neurosurg 77:187–191PubMedCrossRef
48.
Sadrameli SS, Chu JK, Chan TM, Steele WJ, Curry DJ, Lam SK (2019) Minimally invasive tubular tethered cord release in the pediatric population. World Neurosurg 128:e912–e917PubMedCrossRef
49.
Samuels R, McGirt MJ, Attenello FJ, Garcés Ambrossi GL, Singh N, Solakoglu C, Weingart JD, Carson BS, Jallo GI (2009) Incidence of symptomatic retethering after surgical management of pediatric tethered cord syndrome with or without duraplasty. Childs Nerv Syst 25:1085–1089PubMedCrossRef
50.
Schoenmakers MA, Gooskens RH, Gulmans VA, Hanlo PW, Vandertop WP, Uiterwaal CS, Helders PJ (2003) Long-term outcome of neurosurgical untethering on neurosegmental motor and ambulation levels. Dev Med Child Neurol 45:551–555PubMedCrossRef
51.
Seki T, Hida K, Yano S, Sasamori T, Hamauch S, Koyanagi I, Houkin K (2016) Surgical outcome of children and adolescents with tethered cord syndrome. Asian Spine J 10:940–944PubMedPubMedCentralCrossRef
52.
53.
Selden NR, Nixon RR, Skoog SR, Lashley DB (2006) Minimal tethered cord syndrome associated with thickening of the terminal filum. J Neurosurg 105:214–218PubMed
54.
Shahjouei S, Hanaei S, Habibi Z, Hoseini M, Ansari S, Nejat F (2016) Randomized clinical trial of acetazolamide administration and/or prone positioning in mitigating wound complications following untethering surgeries. J Neurosurg Pediatr 17:659–666PubMedCrossRef
55.
Sim J, Shim Y, Kim KH, Kim SK, Lee JY (2021) Features of the filum terminale in tethered cord syndrome with focus on pathology. J Korean Neurosurg Soc 64:585–591PubMedCrossRef
56.
Stavrinou P, Kunz M, Lehner M, Heger A, Müller-Felber W, Tonn JC, Peraud A (2011) Children with tethered cord syndrome of different etiology benefit from microsurgery-a single institution experience. Childs Nerv Syst 27:803–810PubMedCrossRef
57.
Steinbok P, MacNeily AE, Hengel AR, Afshar K, Landgraf JM, Hader W, Pugh J (2016) Filum section for urinary incontinence in children with occult tethered cord syndrome: a randomized, controlled pilot study. J Urol 195:1183–1188PubMedCrossRef
58.
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 22:838–842PubMedCrossRef
59.
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 39:773–777PubMedCrossRef
60.
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 17:408–411PubMedCrossRef
61.
Udayakumaran S, Rathod CT (2018) Tailored strategies to manage cerebrospinal fluid leaks or pseudomeningocele after surgery for tethered cord syndrome. World Neurosurg 114:e1049–e1056PubMedCrossRef
62.
Udayakumaran S, Karthika KS, Nair NS, George M, Gopinath S (2021) Prognostication of the neurological outcome of tethered cord based on intraoperative neuromonitoring findings: how close can we get? Br J Neurosurg. online ahead of print
63.
Valentini LG, Babini M, Cordella R, Beretta E, Destro F, Murabito P, Caldiroli D, Devigili G, Selvaggio G (2021) Early de-tethering: analysis of urological and clinical consequences in a series of 40 children. Childs Nerv Syst 37:941–949PubMedCrossRef
64.
65.
Yener S, Thomas DT, Hicdonmez T, Dagcinar A, Bayri Y, Kaynak A, Dagli TE, Tugtepe H (2015) The effect of untethering on urologic symptoms and urodynamic parameters in children with primary tethered cord syndrome. Urology 85:221–226PubMedCrossRef
66.
Meyrat BJ, Tercier S, Lutz N, Rilliet B, Forcada-Guex M, Vernet O (2003) Introduction of a urodynamic score to detect pre- and postoperative neurological deficits in children with a primary tethered cord. Childs Nerv Syst 19:716–721PubMedCrossRef
67.
Robbins JW, Lundy PA, Gard AP, Puccioni MJ (2015) Perineal pain secondary to tethered cord syndrome: retrospective review of single institution experience. Childs Nerv Syst 31:2141–2144PubMedCrossRef
68.
Wang H, Xu T, Sun J, Wang Y, Sun K, Xu X, Zhang B, Guo Y, Shi J (2019) Homogeneous spinal-shortening axial decompression as a revision surgery after untethering surgery in pediatric patients with tethered cord syndrome. World Neurosurg 124:e702–e706PubMedCrossRef
69.
Bowman RM, Mohan A, Ito J, Seibly JM, McLone DG (2009) Tethered cord release: a long-term study in 114 patients. J Neurosurg Pediatr 3:181–187PubMedCrossRef
70.
Kestle JRW, Riva-Cambrin J (2019) Prospective multicenter studies in pediatric hydrocephalus. J Neurosurg Pediatr 23:135–141PubMedCrossRef
71.
Xu K, He J, Wang L (2022) A systematic review and meta-analysis of minimally invasive surgery in children with occult tethered cord syndrome. Transl Pediatr 11:403–410PubMedPubMedCentralCrossRef
72.
O’Connor KP, Smitherman AD, Milton CK, Palejwala AH, Lu VM, Johnston SE, Homburg H, Zhao D, Martin MD (2020) Surgical treatment of tethered cord syndrome in adults: a systematic review and meta-analysis. World Neurosurgery 137:e221–e241PubMedCrossRef
73.
Lyden P, Brott T, Tilley B, Welch KM, Mascha EJ, Levine S, Haley EC, Grotta J, Marler J (1994) Improved reliability of the NIH Stroke Scale using video training. NINDS TPA Stroke Study Group Stroke 25:2220–2226
74.
Jain S, Iverson LM (2023) Glasgow Coma Scale. StatPearls Publishing, Treasure Island (FL), StatPearls
75.
Hunt W, Hess R (1968) Surgical risk as related to time of intervention in the repair of intracranial aneurysms. J Neurosurg 28:14–20PubMedCrossRef
76.
Roberts TT, Leonard GR, Cepela DJ (2017) Classifications in brief: American Spinal Injury Association (ASIA) Impairment Scale. Clin Orth Relat Res 475:1499–1504CrossRef
77.
78.
Biffl WL, Moore EE, Offner PJ, Brega KE, Franciose RJ, Burch JM (1999) Blunt carotid arterial injuries: implications of a new grading scale. J Trauma Acute Care Surg 47:845CrossRef
79.
Fang Y, Pei Z, Chen H, Wang R, Feng M, Wei L, Li J, Zhang H, Wang S (2021) Diagnostic value of Knosp grade and modified Knosp grade for cavernous sinus invasion in pituitary adenomas: a systematic review and meta-analysis. Pituitary 24:457–464PubMedCrossRef
80.
Fisher CM, Kistler JP, Davis JM (1980) Relation of cerebral vasospasm to subarachnoid hemorrhage visualized by computerized tomographic scanning. Neurosurgery 6:1–9PubMedCrossRef
81.
Spetzler RF, Martin NA (1986) A proposed grading system for arteriovenous malformations. J Neurosurg 65:476–483PubMedCrossRef
82.
Kulkarni AV, Drake JM, Mallucci CL, Sgouros S, Roth J, Constantini S (2009) Endoscopic third ventriculostomy in the treatment of childhood hydrocephalus. J Pediatr 155:254-259.e251PubMedCrossRef
83.
84.
Quinn TJ, Dawson J, Walters M (2008) Dr John Rankin; his life, legacy and the 50th anniversary of the Rankin Stroke Scale. Scott Med J 53:44–47PubMedCrossRef
85.
Yao M, Simoes A (2023) Urodynamic testing and interpretation. StatPearls Publishing, Treasure Island (FL), StatPearls
Metadata
Title
Disparities in indications and outcomes reporting for pediatric tethered cord surgery: The need for a standardized outcome assessment tool
Authors
Matthew C. Findlay
Samuel Tenhoeve
Skyler A. Terry
Rajiv R. Iyer
Douglas L. Brockmeyer
Michael P. Kelly
John R. W. Kestle
David Gonda
Vijay M. Ravindra
Publication date
11-12-2023
Publisher
Springer Berlin Heidelberg
Published in
Child's Nervous System / Issue 4/2024
Print ISSN: 0256-7040
Electronic ISSN: 1433-0350
DOI
https://doi.org/10.1007/s00381-023-06246-y

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