Skip to main content
SpringerLink
Log in

Selective dorsal rhizotomy for spasticity of genetic etiology

Child's Nervous System Aims and scope Submit manuscript

Cite this article

Abstract

Objective Selective dorsal rhizotomy (SDR) is most commonly applied in the context of the treatment of the spastic diplegic variant of cerebral palsy (CP). Its role in the treatment of spasticity associated with other conditions is not well-established. We sought to review outcomes following SDR for the treatment of functionally limiting spasticity in the setting of a genetic etiology. Methods A systematic literature review was performed using the databases Ovid Medline, Embase, Cochrane Library, and PubMed based on the PRISMA guidelines. Articles were included if they described the application of SDR for spasticity of genetic etiology. Reported outcomes pertaining to spasticity and gross motor function following SDR were summarized. Results Five articles reporting on 16 patients (10 males, 6 females) met the inclusion criteria, of which four reported on SDR for hereditary spastic paraplegia (HSP) and four on syndromic patients or other inherited diseases, with an overall follow-up ranging from 11 to 252 months. These individuals were found to have several genetic mutations including ALS2, SPG4, and SPG3A. The mean age at the time of surgery was 14.9 years (median 10 years, range 3–37 years). Conclusions Although all patients experienced a reduction in spasticity, the long-term gross motor functional outcomes objectively assessed at last follow-up were heterogeneous. There may be a role for SDR in the context of static genetic disorders causing spasticity. Further evidence is required prior to the widespread adoption of SDR for such disorders as, based on the collective observations of this review, spasticity is consistently reduced but the long-term effect on gross motor function remains unclear.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1

References

  1. Aquilina K, Graham D, Wimalasundera N (2015) Selective dorsal rhizotomy: an old treatment re-emerging. Arch Dis Child 100:798–802

    Article  Google Scholar 

  2. Graham D, Aquilina K, Mankad K, Wimalasundera N (2018) Selective dorsal rhizotomy: current state of practice and the role of imaging. Quant Imaging Med Surg 8:209–218

    Article  Google Scholar 

  3. Davidson B, Fehlings D, Milo-Manson G, Ibrahim GM (2019) Improving access to selective dorsal rhizotomy for children with cerebral palsy. CMAJ 191:E1205–e1206

    Article  Google Scholar 

  4. Summers J, Coker B, Eddy S, Elstad M, Bunce C, Bourmpaki E, Pennington M, Aquilina K, Cawker S, Edwards R, Goodden J, Hawes S, McCune K, Pettorini B, Smith J, Sneade C, Vloeberghs M, Patrick H, Powell H, Verity C, Peacock JL (2019) Selective dorsal rhizotomy in ambulant children with cerebral palsy: an observational cohort study. Lancet Child Adolesc Health 3:455–462

    Article  Google Scholar 

  5. McLaughlin J, Bjornson K, Temkin N, Steinbok P, Wright V, Reiner A, Roberts T, Drake J, O'Donnell M, Rosenbaum P, Barber J, Ferrel A (2002) Selective dorsal rhizotomy: meta-analysis of three randomized controlled trials. Dev Med Child Neurol 44:17–25

    Article  Google Scholar 

  6. Steinbok P, Reiner AM, Beauchamp R, Armstrong RW, Cochrane DD, Kestle J (1997) A randomized clinical trial to compare selective posterior rhizotomy plus physiotherapy with physiotherapy alone in children with spastic diplegic cerebral palsy. Dev Med Child Neurol 39:178–184

    Article  CAS  Google Scholar 

  7. Wright FV, Sheil EM, Drake JM, Wedge JH, Naumann S (1998) Evaluation of selective dorsal rhizotomy for the reduction of spasticity in cerebral palsy: a randomized controlled trial. Dev Med Child Neurol 40:239–247

    Article  CAS  Google Scholar 

  8. McLaughlin JF, Bjornson KF, Astley SJ, Hays RM, Hoffinger SA, Armantrout EA, Roberts TS (1994) The role of selective dorsal rhizotomy in cerebral palsy: critical evaluation of a prospective clinical series. Dev Med Child Neurol 36:755–769

    Article  CAS  Google Scholar 

  9. Nicolini-Panisson RD, Tedesco AP, Folle MR, Donadio MVF (2018) Selective dorsal rhizotomy in cerebral palsy: selection criteria and postoperative physical therapy protocols. Revista paulista de pediatria : orgao oficial da Sociedade de Pediatria de Sao Paulo 36:9

    Article  Google Scholar 

  10. Funk JF, Panthen A, Bakir MS, Gruschke F, Sarpong A, Wagner C, Lebek S, Haberl EJ (2015) Predictors for the benefit of selective dorsal rhizotomy. Res Dev Disabil 37:127–134

    Article  Google Scholar 

  11. Gump WC, Mutchnick IS, Moriarty TM (2013) Selective dorsal rhizotomy for spasticity not associated with cerebral palsy: reconsideration of surgical inclusion criteria. Neurosurg Focus 35:E6

    Article  Google Scholar 

  12. Fink JK (2014) Hereditary spastic paraplegia: clinical principles and genetic advances. Semin Neurol 34:293–305

    Article  Google Scholar 

  13. Racis L, Tessa A, Pugliatti M, Storti E, Agnetti V, Santorelli FM (2014) Infantile-onset ascending hereditary spastic paralysis: a case report and brief literature review. Eur J Paediatr Neurol 18:235–239

    Article  Google Scholar 

  14. D'Amore A, Tessa A, Casali C, Dotti MT, Filla A, Silvestri G, Antenora A, Astrea G, Barghigiani M, Battini R, Battisti C, Bruno I, Cereda C, Dato C, Di Iorio G, Donadio V, Felicori M, Fini N, Fiorillo C, Gallone S, Gemignani F, Gigli GL, Graziano C, Guerrini R, Gurrieri F, Kariminejad A, Lieto M, Marques LourenCo C, Malandrini A, Mandich P, Marcotulli C, Mari F, Massacesi L, Melone MAB, Mignarri A, Milone R, Musumeci O, Pegoraro E, Perna A, Petrucci A, Pini A, Pochiero F, Pons MR, Ricca I, Rossi S, Seri M, Stanzial F, Tinelli F, Toscano A, Valente M, Federico A, Rubegni A, Santorelli FM (2018) Next generation molecular diagnosis of hereditary spastic paraplegias: an Italian cross-sectional study. Front Neurol 9:981

    Article  Google Scholar 

  15. Parodi L, Fenu S, Stevanin G, Durr A (2017) Hereditary spastic paraplegia: more than an upper motor neuron disease. Rev Neurol 173:352–360

    Article  CAS  Google Scholar 

  16. Lo Giudice T, Lombardi F, Santorelli FM, Kawarai T, Orlacchio A (2014) Hereditary spastic paraplegia: clinical-genetic characteristics and evolving molecular mechanisms. Exp Neurol 261:518–539

    Article  CAS  Google Scholar 

  17. Parodi L, Coarelli G, Stevanin G, Brice A, Durr A (2018) Hereditary ataxias and paraparesias: clinical and genetic update. Curr Opin Neurol 31:462–471

    Article  Google Scholar 

  18. Kai M, Yongjie L, Ping Z (2014) Long-term results of selective dorsal rhizotomy for hereditary spastic paraparesis. J Clin Neurosci 21:116–120

    Article  Google Scholar 

  19. Sharma J, Bonfield C, Steinbok P (2016) Selective dorsal rhizotomy for hereditary spastic paraparesis in children. Child’s Nerv Syst 32:1489–1494

    Article  Google Scholar 

  20. Buizer AI, van Schie PEM, Bolster EAM, van Ouwerkerk WJ, Strijers RL, van de Pol LA, Stadhouder A, Becher JG, Vermeulen RJ (2017) Effect of selective dorsal rhizotomy on daily care and comfort in non-walking children and adolescents with severe spasticity. Eur J Paediatr Neurol 21:350–357

    Article  CAS  Google Scholar 

  21. Cole GF, Farmer SE, Roberts A, Stewart C, Patrick JH (2007) Selective dorsal rhizotomy for children with cerebral palsy: the Oswestry experience. Arch Dis Child 92:781–785

    Article  CAS  Google Scholar 

  22. Grunt S, Becher JG, Vermeulen RJ (2011) Long-term outcome and adverse effects of selective dorsal rhizotomy in children with cerebral palsy: a systematic review. Dev Med Child Neurol 53:490–498

    Article  Google Scholar 

  23. van Schie PE, Schothorst M, Dallmeijer AJ, Vermeulen RJ, van Ouwerkerk WJ, Strijers RL, Becher JG (2011) Short- and long-term effects of selective dorsal rhizotomy on gross motor function in ambulatory children with spastic diplegia. J Neurosurg Pediatr 7:557–562

    Article  Google Scholar 

  24. Rushna Raza* RL (2019) Approach to managing spasticity in heriditary spastic paraplegia 101136/archdischild-2019-epa181

  25. Grootveld LR, van Schie PE, Buizer AI, Jeroen Vermeulen R, van Ouwerkerk WJ, Strijers RL, Becher JJ (2016) Sudden falls as a persistent complication of selective dorsal rhizotomy surgery in children with bilateral spasticity: report of 3 cases. J Neurosurg Pediatr 18:192–195

    Article  Google Scholar 

  26. Mazarakis NK, Vloeberghs MH (2016) Spasticity secondary to Leigh syndrome managed with selective dorsal rhizotomy: a case report. Child’s Nerv Syst 32:1745–1748

    Article  CAS  Google Scholar 

  27. Buizer AI (2019) Selective dorsal rhizotomy in children with cerebral palsy. Lancet Child Adolesc Health 3:438–439

    Article  Google Scholar 

  28. Finsterer J, Loscher W, Quasthoff S, Wanschitz J, Auer-Grumbach M, Stevanin G (2012) Hereditary spastic paraplegias with autosomal dominant, recessive, X-linked, or maternal trait of inheritance. J Neurol Sci 318:1–18

    Article  Google Scholar 

  29. Novak I, McIntyre S, Morgan C, Campbell L, Dark L, Morton N, Stumbles E, Wilson SA, Goldsmith S (2013) A systematic review of interventions for children with cerebral palsy: state of the evidence. Dev Med Child Neurol 55:885–910

    Article  Google Scholar 

  30. Pucks-Faes E, Dobesberger J, Hitzenberger G, Matzak H, Mayr A, Fava E, Genelin E, Saltuari L (2019) Intrathecal baclofen in hereditary spastic paraparesis. Front Neurol 10:901

    Article  Google Scholar 

  31. Margetis K, Korfias S, Boutos N, Gatzonis S, Themistocleous M, Siatouni A, Dalivigka Z, Flaskas T, Stranjalis G, Boviatsis E, Sakas D (2014) Intrathecal baclofen therapy for the symptomatic treatment of hereditary spastic paraplegia. Clin Neurol Neurosurg 123:142–145

    Article  Google Scholar 

  32. Nordmark E, Josenby AL, Lagergren J, Andersson G, Stromblad LG, Westbom L (2008) Long-term outcomes five years after selective dorsal rhizotomy. BMC Pediatr 8:54

    Article  Google Scholar 

  33. Orrell RW (1993) ALS2-related disorders. In: Adam MP, Ardinger HH, Pagon RA, Wallace SE, Bean LJH, Stephens K, Amemiya A (eds) GeneReviews((R)). University of Washington, Seattle University of Washington, Seattle GeneReviews is a registered trademark of the University of Washington, Seattle. All rights reserved., Seattle (WA)

    Google Scholar 

  34. Jeffery SMT, Markia B, Pople IK, Aquilina K, Smith J, Mohamed AZ, Burchell A, Jenkins L, Walsh P, Clark N, Sacree J, Cramp M, Babiker MOE, Atherton WG, Clarke A, Edwards RJ (2019) Surgical outcomes of single-level bilateral selective dorsal rhizotomy for spastic diplegia in 150 consecutive patients. World Neurosurg

  35. Klebe S, Stevanin G, Depienne C (2015) Clinical and genetic heterogeneity in hereditary spastic paraplegias: from SPG1 to SPG72 and still counting. Rev Neurol 171:505–530

    Article  CAS  Google Scholar 

  36. da Graca FF, de Rezende TJR, Vasconcellos LFR, Pedroso JL, Barsottini OGP, Franca MC Jr (2018) Neuroimaging in hereditary spastic paraplegias: current use and future perspectives. Front Neurol 9:1117

    Article  Google Scholar 

  37. de Souza PVS, de Rezende Pinto WBV, de Rezende Batistella GN, Bortholin T, Oliveira ASB (2017) Hereditary spastic paraplegia: clinical and genetic hallmarks. Cerebellum (London, England) 16:525–551

    Article  Google Scholar 

  38. Peacock WJ, Arens LJ, Berman B (1987) Cerebral palsy spasticity. Selective posterior rhizotomy. Pediatr Neurosci 13:61–66

    Article  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Division of Neurosurgery, The Hospital for Sick Children, 555 University Ave, Suite 1503, Toronto, Ontario, M5G 1X8, Canada

    Laura-Nanna Lohkamp, Ian Coulter & George M. Ibrahim

Authors
  1. Laura-Nanna Lohkamp
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Ian Coulter
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. George M. Ibrahim
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Laura-Nanna Lohkamp.

Ethics declarations

Conflict of interest

The authors do not have any conflicts of interest.

Additional information

Publisher’s note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and Permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Lohkamp, LN., Coulter, I. & Ibrahim, G.M. Selective dorsal rhizotomy for spasticity of genetic etiology. Childs Nerv Syst 36, 1357–1365 (2020). https://doi.org/10.1007/s00381-020-04601-x

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00381-020-04601-x

Keywords

search

Navigation