Surgical treatment of spinal stenosis in achondroplasia: Literature review comparing results in adults and paediatrics

Published:October 30, 2021DOI:https://doi.org/10.1016/j.jcot.2021.101672

      Abstract

      Background

      This study aims to assess the quantity and quality of available literature on surgical treatment outcomes of spinal stenosis in adult and paediatric achondroplasia patients through a systematic review of literature and to investigate the suitability of conducting a meta-analysis on outcomes of surgical treatment.

      Methods

      Online databases were searched according to PRISMA guidelines. No restrictions regarding study design, sample size, previous treatment, or publication date were implemented. The following terms: “Spinal stenosis”, “Spinal Decompression”, “Spinal fusion”, each term separately combined with the term “Achondroplasia” were used. Quality of the included studies were assessed used the Modified Coleman method.

      Results

      Five adult and four paediatric single-sample non-comparative studies were identified for inclusion (176 adult and 102 paediatric patients). Meta-analyses assessed the proportion of patients achieving full resolution of symptoms to be 0.51 (95% CI 0.00 to 1.00); the proportion of patients achieving full or partial resolution of symptoms to be 0.90 (95% CI 0.84 to 0.97); the proportion of procedures requiring re-operation to be 0.42 (95% CI 0.34 to 0.50; and the proportion of procedures involving dural tears to be 0.20 (95% CI 0.02 to 0.39). Statistical heterogeneity was very high for full resolution of symptoms and requirement for dural repair; and very low for other outcomes.

      Conclusions

      The available literature on this population and condition is sparse, highly heterogenous, and is generally of low quality limiting the value of meta-analysis. Overall, outcomes of surgical decompression of symptomatic spinal stenosis in achondroplasia patients show consistent degree of resolution of symptoms. Duration of symptoms prior to surgical treatment appears to play an important role in the overall outcome of treatment. Therefore, a delay in diagnosis and treatment can potentially be detrimental in achieving a better outcome.

      Keywords

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      References

        • Shiang R.
        • Thompson L.M.
        • Zhu Y.-Z.
        • et al.
        Mutations in the transmembrane domain of FGFR3 cause the most common genetic form of dwarfism, achondroplasia.
        Cell. 1994 Jul; 78: 335-342
        • Genetics Home Reference
        Health conditions: achondroplasia.
        ([Internet], [cited 2020 Aug 10]. Available from:)
        • Hall J.
        The natural history of achondroplasia.
        ([cited 2020 Jun 7])in: Nicoletti B. Kopits S.E. Ascani E. McKusick V.A. Dryburgh S.C. Human Achondroplasia [Internet]. Springer US, Boston, MA1988: 3-9 (Available from:)
        • Hurko O.
        • Pyeritz R.
        • Uematsu S.
        Neurological considerations in achondroplasia.
        Basic Life Sci. 1988; 48: 153-162
      1. Nelson MA. Kyphosis and lumbar stenosis in achondroplasia. In: Human Achondroplasia. Springer US; p. 305–311. (Basic Life Sciences).

        • Rimoin D.
        Clinical variability in achondroplasia.
        ([cited 2020 Jun 7])in: Nicoletti B. Kopits S.E. Ascani E. McKusick V.A. Dryburgh S.C. Human Achondroplasia [Internet]. Springer US, Boston, MA1988: 3-9 (Available from:)
        • Gigilio G.
        • Passariello R.
        • Pagnotta G.
        • Crostelli M.
        • Ascani E.
        Anatomy of the lumbar spine in achondroplasia.
        ([cited 2020 Jun 7])in: Nicoletti B. Kopits S.E. Ascani E. McKusick V.A. Dryburgh S.C. Human Achondroplasia [Internet]. Springer US, Boston, MA1988: 3-9 (Available from:)
        • Jeong S.-T.
        • Song H.-R.
        • Keny S.M.
        • Telang S.S.
        • Suh S.-W.
        • Hong S.-J.
        MRI study of the lumbar spine in achondroplasia: a morphometric analysis for the evaluation OF stenosis OF the canal.
        J Bone Joint Surg Br. 2006 Sep; 88-B: 1192-1196
        • Yukawa Y.
        • Lenke L.G.
        • Tenhula J.
        • Bridwell K.H.
        • Riew K.D.
        • Blanke K.
        A comprehensive study OF patients with surgically treated lumbar spinal stenosis with neurogenic claudication.
        J Bone Jt Surg-Am. 2002 Nov; 84: 1954-1959
        • Yamada H.
        • Nakamura S.
        • Tajima M.
        • Kageyama N.
        Neurological manifestations of pediatric achondroplasia.
        J Neurosurg. 1981 Jan; 54: 49-57
        • Ruiz-Garcia M.
        • Tovar-Baudin A.
        • Del Castillo-Ruiz V.
        • et al.
        Early detection of neurological manifestations in achondroplasia.
        Childs Nerv Syst. 1997 May 20; 13: 208-213
        • Nelson M.A.
        Spinal stenosis in achondroplasia.
        Proc Roy Soc Med. 1972 Nov; 65: 1028-1029
        • Hunter A.G.
        • Bankier A.
        • Rogers J.G.
        • Sillence D.
        • Scott C.I.
        Medical complications of achondroplasia: a multicentre patient review.
        J Med Genet. 1998 Sep 1; 35: 705-712
        • Coleman B.D.
        • Khan K.M.
        • Maffulli N.
        • Cook J.L.
        • Wark J.D.
        Studies of surgical outcome after patellar tendinopathy: clinical significance of methodological deficiencies and guidelines for future studies.
        Scand J Med Sci Sports. 2000 Feb; 10: 2-11
        • Jakobsen R.B.
        • Engebretsen L.
        • Slauterbeck J.R.
        An analysis of the quality of cartilage repair studies.
        J Bone Joint Surg Am. 2005 Oct; 87: 2232-2239
        • Thomeer R.T.W.M.
        • van Dijk J.M.C.
        Surgical treatment of lumbar stenosis in achondroplasia.
        J Neurosurg Spine. 2002 Apr; 96: 292-297
        • Vleggeert-Lankamp C.
        • Peul W.
        Surgical decompression of thoracic spinal stenosis in achondroplasia: indication and outcome: clinical article.
        J Neurosurg Spine. 2012 Aug; 17: 164-172
        • Carlisle E.S.
        • Ting B.L.
        • Abdullah M.A.
        • et al.
        Laminectomy in patients with achondroplasia: the impact of time to surgery on long-term function.
        Spine. 2011 May; 36: 886-892
        • Ain M.C.
        • Chang T.-L.
        • Schkrohowsky J.G.
        Laminectomies and achondroplasia: does body mass index influence surgical outcomes?.
        Am J Med Genet. 2007 May 15; 143A: 1032-1037
        • Pyeritz R.E.
        • Sack G.H.
        • Udvarhelyi G.B.
        • Opitz J.M.
        • Reynolds J.F.
        Thoracolumbosacral laminectomy in achondroplasia: long-term results in 22 patients.
        Am J Med Genet. 1987 Oct; 28: 433-444
        • Ain M.C.
        • Elmaci I.
        • Hurko O.
        • Clatterbuck R.E.
        • Lee R.R.
        • Rigamonti D.
        Reoperation for spinal restenosis in achondroplasia.
        J Spinal Disord. 2000 Apr; 13: 168-173
        • Bydon M.
        • Macki M.
        • Xu R.
        • Ain M.C.
        • Ahn E.S.
        • Jallo G.I.
        Spinal decompression in achondroplastic patients using high-speed drill versus ultrasonic bone curette: technical note and outcomes in 30 cases.
        J Pediatr Orthop. 2014 Dec; 34: 780-786
        • Sciubba D.M.
        • Noggle J.C.
        • Marupudi N.I.
        • et al.
        Spinal stenosis surgery in pediatric patients with achondroplasia.
        J Neurosurg. 2007 May; 106: 372-378
        • Baca K.E.
        • Abdullah M.A.
        • Ting B.L.
        • et al.
        Surgical decompression for lumbar stenosis in pediatric achondroplasia.
        J Pediatr Orthop. 2010 Aug; 30: 449-454
        • Herno A.
        • Airaksinen O.
        • Saari T.
        The long-term prognosis after operation for lumbar spinal stenosis.
        Scand J Rehabil Med. 1993 Dec; 25: 167-171
        • Herno A.
        • Airaksinen O.
        • Saari T.
        • Luukkonen M.
        Lumbar spinal stenosis: a matched-pair study of operated and non-operated patients.
        Br J Neurosurg. 1996 Jan; 10: 461-466
        • Airaksinen O.
        • Herno A.
        • Turunen V.
        • Saari T.
        • Suomlainen O.
        Surgical Outcome of 438 Patients Treated Surgically for Lumbar Spinal Stenosis: Spine. vol. 22. 1997 Oct: 2278-2282 (19)
        • Ain M.C.
        • Shirley E.D.
        • Pirouzmanesh A.
        • Hariri A.
        • Carson B.S.
        Postlaminectomy kyphosis in the skeletally immature achondroplast.
        Spine. 2006 Jan 15; 31: 197-201
        • Tafazal S.I.
        • Sell P.J.
        Incidental durotomy in lumbar spine surgery: incidence and management.
        Eur Spine J. 2005 Apr; 14: 287-290
        • West J.L.
        • Arnel M.
        • Palma A.E.
        • Frino J.
        • Powers A.K.
        • Couture D.E.
        Incidental durotomy in the pediatric spine population.
        J Neurosurg Pediatr. 2018 01; 22: 591-594
        • Shafi K.
        • Lovecchio F.
        • Sava M.
        • et al.
        Complications and revisions after spine surgery in patients with skeletal dysplasia: have we improved?.
        Global Spine J. 2021 Feb 18; (219256822199478)