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Management of a pseudarthrosis with sagittal malalignment in a patient with ochronotic spondyloarthropathy

  • Wael Alkasem
  • Louis Boissiere
  • Ibrahim Obeid
  • Anouar BourghliEmail author
Grand Rounds

Abstract

Purpose

Ochronotic spondyloarthropathy is an uncommon disease, and its association to sagittal malalignment in the context of a pseudarthrosis has never been described.

Methods

We present the case of a 56-year-old female, who underwent previously L4L5 laminectomy for central canal stenosis and started later on to complain of progressively severe low back pain with a significant forward imbalance while walking. X-rays showed non-compensated sagittal malalignment due to thoracolumbar kyphosis, CT scan revealed multilevel central intradiscal calcifications with important vacuum disc at L4L5, and MRI showed T1 and T2 hypointensity signal at the same level with bone marrow oedema. Alkaptonuric ochronosis was suspected and confirmed by the presence of homogentisic acid in the urine, and the diagnosis of L4L5 pseudarthrosis with associated severe sagittal malalignment in the context of ochronotic spondyloarthropathy was established.

Results

The patient underwent surgery with a posterior-only approach with a long-segment pedicle screw construct from T10 to the pelvis with a 360° fusion with a cage at L4L5. Samples taken from the disc and ligaments confirmed the diagnosis of ochronotic spondyloarthropathy macroscopically and microscopically. She could walk on day 2 with a satisfactory clinical and radiological result at 2 years.

Conclusion

This is the first case in the literature to describe a post-laminectomy pseudarthrosis leading to a significant sagittal malalignment in a patient with ochronotic spondyloarthropathy. Management of such a case is challenging as the spine is partially ankylosed; therefore, a long construct is advisable to avoid ankylosing disorders related complications.

Keywords

Alkaptonuria Ochronotic spondyloarthropathy Pseudarthrosis Sagittal alignment Kyphosis 

Notes

Compliance with ethical standards

Conflict of interest

There are no conflicts of interest for this case report.

References

  1. 1.
    Phornphutkul C, Introne WJ, Perry MB, Bernardini I, Murphey MD, Fitzpatrick DL, Anderson PD, Huizing M, Anikster Y, Gerber LH, Gahl WA (2002) Natural history of alkaptonuria. N Engl J Med 347:2111–2121.  https://doi.org/10.1056/NEJMoa021736 CrossRefGoogle Scholar
  2. 2.
    Laskar FH, Sargison KD (1970) Ochronotic arthropathy. A review with four case reports. J Bone Joint Surg Br 52:653–666CrossRefGoogle Scholar
  3. 3.
    Jebaraj I, Chacko BR, Chiramel GK, Matthai T, Parameswaran A (2013) A simplified staging system based on the radiological findings in different stages of ochronotic spondyloarthropathy. Indian J Radiol Imaging 23:101–105.  https://doi.org/10.4103/0971-3026.113628 CrossRefGoogle Scholar
  4. 4.
    Balaban B, Taskaynatan M, Yasar E, Tan K, Kalyon T (2006) Ochronotic spondyloarthropathy: spinal involvement resembling ankylosing spondylitis. Clin Rheumatol 25:598–601.  https://doi.org/10.1007/s10067-005-0038-8 CrossRefGoogle Scholar
  5. 5.
    Millea TP, Segal LS, Liss RG, Stauffer ES (1992) Spine fracture in ochronosis. Report of a case. Clin Orthop Relat Res 281:208–211Google Scholar
  6. 6.
    Donaldson CJ, Mitchell SL, Riley LH 3rd, Kebaish KM (2019) “As black as ink”: a case of alkaptonuria-associated myelopathy and a review of the literature. Spine (Phila Pa 1976) 44:E53–E59.  https://doi.org/10.1097/BRS.0000000000002755 CrossRefGoogle Scholar
  7. 7.
    Akeda K, Kasai Y, Kawakita E, Matsumura Y, Kono T, Murata T, Uchida A (2008) Thoracic myelopathy with alkaptonuria. Spine (Phila Pa 1976) 33:E62–E65.  https://doi.org/10.1097/BRS.0b013e31816046ea CrossRefGoogle Scholar
  8. 8.
    Elgafy H, Lukens SB (2014) Hyperextension injury in a patient with ochronotic spondyloarthropathy. Spine J 14:1802–1803.  https://doi.org/10.1016/j.spinee.2014.03.032 CrossRefGoogle Scholar
  9. 9.
    Yucetas SC, Ucler N (2018) Black-colored ligamentum flavum due to alkaptonuria. J Neurol Surg A Cent Eur Neurosurg.  https://doi.org/10.1055/s-0038-1675784 Google Scholar
  10. 10.
    Emel E, Karagoz F, Aydin IH, Hacisalihoglu S, Seyithanoglu MH (2000) Alkaptonuria with lumbar disc herniation: a report of two cases. Spine (Phila Pa 1976) 25:2141–2144CrossRefGoogle Scholar
  11. 11.
    Kalevski SK, Haritonov DG, Peev NA (2007) Alcaptonuria with lumbar disc prolapse: case study and review of the literature. Spine J 7:495–498.  https://doi.org/10.1016/j.spinee.2006.06.399 CrossRefGoogle Scholar
  12. 12.
    Rahimizadeh A, Soufiani H, Hassani V, Rahimizadeh A (2017) Symptomatic pseudarthrosis in ochronotic spine: case report. J Neurosurg Spine 26:220–228.  https://doi.org/10.3171/2016.5.SPINE15671 CrossRefGoogle Scholar
  13. 13.
    Chen LH, Kao FC, Niu CC, Lai PL, Fu TS, Chen WJ (2005) Surgical treatment of spinal pseudoarthrosis in ankylosing spondylitis. Chang Gung Med J 28:621–628Google Scholar
  14. 14.
    Kim KT, Lee SH, Suk KS, Lee JH, Im YJ (2007) Spinal pseudarthrosis in advanced ankylosing spondylitis with sagittal plane deformity: clinical characteristics and outcome analysis. Spine (Phila Pa 1976) 32:1641–1647.  https://doi.org/10.1097/BRS.0b013e318074c3ce CrossRefGoogle Scholar
  15. 15.
    Fang D, Leong JC, Ho EK, Chan FL, Chow SP (1988) Spinal pseudarthrosis in ankylosing spondylitis. Clinicopathological correlation and the results of anterior spinal fusion. J Bone Joint Surg Br 70:443–447CrossRefGoogle Scholar
  16. 16.
    Caron T, Bransford R, Nguyen Q, Agel J, Chapman J, Bellabarba C (2010) Spine fractures in patients with ankylosing spinal disorders. Spine (Phila Pa 1976) 35:E458–E464.  https://doi.org/10.1097/BRS.0b013e3181cc764f CrossRefGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2019

Authors and Affiliations

  • Wael Alkasem
    • 1
  • Louis Boissiere
    • 2
  • Ibrahim Obeid
    • 2
  • Anouar Bourghli
    • 3
    Email author
  1. 1.Neurosurgery and Spinal DepartmentZheen International HospitalErbīlIraq
  2. 2.Orthopedic Spinal Surgery Unit 1Bordeaux Pellegrin HospitalBordeauxFrance
  3. 3.Orthopedic and Spinal Surgery DepartmentKingdom HospitalRiyadhSaudi Arabia

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