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Neuropathic arthropathy of the shoulder secondary to operated syringomyelia: a case-based review

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Abstract

Neuropathic arthropathy (NA) is a long-term progressive disorder that causes joint destruction in the existence of a neurologic deficit. Minor injuries and fractures are commonly overlooked until the visible joint deterioration becomes persistent. Syringomyelia is one of the important causes of NA. The appearance of clinical signs in syringomyelia is caused by longitudinal cysts formed in the cervical and cervicothoracic regions of the spinal cord. Depending on the existence of the underlying disorder, the number and localization of the syrinxes, a range of symptoms, involving pain, sensation deficit, loss of motor function, and deep tendon reflex abnormality, emerge. The case is here described of a 68-year-old female patient with shoulder NA following syringomyelia, who partially responded to the rehabilitation program. Furthermore, the available case reports were comprehensively reviewed on Web of Science, Scopus, and PubMed/Medline. Furthermore, the available case reports were comprehensively evaluated on Web of Science, Scopus, and PubMed/Medline. Thus, we aimed to present the demographic characteristics, symptoms, physical examination signs, treatment, and follow-up parameters of syringomyelia-related shoulder NA cases.

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References

  1. Shinjo SK, de Carvalho JF (2010) Charcot’s arthropathy secondary to herpetic encephalitis sequelae: an unusual presentation. Rheumatol Int 30:973–975. https://doi.org/10.1007/s00296-009-1011-7

    Article  PubMed  Google Scholar 

  2. Mortada M, Ezzeldin N, Hammad M (2020) Ultrasonographic features of acute Charcot neuroarthropathy of the foot: a pilot study. Clin Rheumatol 39:3787–3793. https://doi.org/10.1007/s10067-020-05107-2

    Article  CAS  PubMed  Google Scholar 

  3. Lu V, Zhang J, Thahir A, Zhou A, Krkovic M (2021) Charcot knee—presentation, diagnosis, management—a scoping review. Clin Rheumatol 40:4445–4456. https://doi.org/10.1007/s10067-021-05775-8

    Article  PubMed  PubMed Central  Google Scholar 

  4. Zheng YC, Liu YT, Wei KC, Huang YC, Chen PY, Hsu YH, Lin CL (2022) Outcome predictors and clinical presentation of syringomyelia. Asian J Surg S1015–9584(22):00891. https://doi.org/10.1016/j.asjsur.2022.06.150

    Article  Google Scholar 

  5. Flint G (2021) Syringomyelia: diagnosis and management. Pract Neurol 21:403–411. https://doi.org/10.1136/practneurol-2021-002994

    Article  PubMed  Google Scholar 

  6. Baumhauer JF, O’Keefe RJ, Schon LC, Pinzur MS (2006) Cytokine induced osteoclastic bone resorption in Charcot arthropathy: an immunohistochemical study. Foot Ankle Int 27:797–780. https://doi.org/10.1177/107110070602701007

    Article  PubMed  Google Scholar 

  7. Guyton GP, Saltzman CL (2002) The diabetic foot: basic mechanisms of disease. Instr Course Lect 51:169–181

    PubMed  Google Scholar 

  8. Snoddy MC, Lee DH, Kuhn JE (2017) Charcot shoulder and elbow: a review of the literature and update on treatment. J Shoulder Elbow Surg 26(3):544–552. https://doi.org/10.1016/j.jse.2016.10.015

    Article  PubMed  Google Scholar 

  9. Gasparyan AY, Ayvazyan L, Blackmore H, Kitas GD (2011) Writing a narrative biomedical review: considerations for authors, peer reviewers, and editors. Rheumatol Int 31:1409–1417. https://doi.org/10.1007/s00296-011-1999-3

    Article  PubMed  Google Scholar 

  10. Adiyeke L, Durakbaşa MO, Duymuş TM (2017) Bilateral neuropathic osteoarthropathy of the shoulder due to syringomyelia. Clin Pract 7:952. https://doi.org/10.4081/cp.2017.952

    Article  PubMed  PubMed Central  Google Scholar 

  11. Alai A, Reddy CG, Amrami KK, Spinner RJ (2013) Charcot arthropathy of the shoulder associated with typical and atypical findings: charcot-induced brachial plexopathy. Clin Anat 26:1017–1023. https://doi.org/10.1002/ca.22110

    Article  PubMed  Google Scholar 

  12. Atalar AC (2010) Neuropathic arthropathy of the shoulder associated with syringomyelia: a report of six cases. Acta Orthop Traumatol Turc 44:328–336. https://doi.org/10.3944/AOTT.2010.2216

    Article  PubMed  Google Scholar 

  13. Bai YP, Yu Y, Qi W, Liu L, Chen H (2022) The shoulder charcot arthropathy and secondary to hemangioblastoma in conus medullari with syringomyelia. J Coll Physicians Surg Pak. https://doi.org/10.29271/jcpsp.2022.04.531

    Article  PubMed  Google Scholar 

  14. Barber DB, Janus RB, Wade WH (1996) Neuroarthropathy: an overuse injury of the shoulder in quadriplegia. J Spinal Cord Med 19:9–11. https://doi.org/10.1080/10790268.1996.11719411

    Article  CAS  PubMed  Google Scholar 

  15. Bocca LF, Tonholo Silva TY, Ejnisman B, Pedroso JL, Barsottini OG (2017) Teaching neuro images: the charcot shoulder: Chiari malformation with syringomyelia associated with arthropathy. Neurology 89:e38–e39. https://doi.org/10.1212/WNL.0000000000004154

    Article  PubMed  Google Scholar 

  16. Butala RR, Arora M, Rao AA, Samant PD, Mukherjee S (2014) A rare case of ipsilateral shoulder and thumb CMC joint neuropathic arthropathy. J Surg Case Rep. https://doi.org/10.1093/jscr/rju054

    Article  PubMed  PubMed Central  Google Scholar 

  17. Chakraborty PP, Datta S, Ray S, Bhattacharjee R, Chowdhury S (2015) Unilateral neuropathic arthropathy of the shoulder secondary to syringomyelia Diagnostic challenges. World J Clin Cases. https://doi.org/10.12998/wjcc.v3.i12.1017

    Article  PubMed  PubMed Central  Google Scholar 

  18. Chen K, Deng L, She H, Hu F, Li T (2019) MR neurography showed brachial plexus abnormalities in syringomyelia with shoulder Charcot arthropathy: a case report. Br J Neurosurg. https://doi.org/10.1080/02688697.2019

    Article  PubMed  Google Scholar 

  19. Choudhury P, Mohapatra A, Kharat A, Chauhan V (2019) Neuropathic arthropathy of the shoulder joint caused by syringomyelia: report of a rare case. BMJ Case Rep 12:e229494. https://doi.org/10.1136/bcr-2019-229494

    Article  PubMed  PubMed Central  Google Scholar 

  20. Crowther MAA, Bell SN (2007) Neuropathic shoulder in syringomyelia treated with resurfacing arthroplasty of humeral head and soft-tissue lining of glenoid: a case report. J Shoulder Elbow Surg 16:e38-40. https://doi.org/10.1016/j.jse.2006.11.006

    Article  PubMed  Google Scholar 

  21. Cullen AB, Ofluoglu O, Donthineni R (2005) Neuropathic arthropathy of the shoulder (Charcot shoulder). Medscape Gen Med 7:29

    Google Scholar 

  22. Deng X, Wu L, Yang C, Xu Y (2013) Neuropathic arthropathy caused by syringomyelia: clinical article. J Neurosurg Spine 18:303–309. https://doi.org/10.3171/2012.11.SPINE12860

    Article  PubMed  Google Scholar 

  23. Dharmshaktu GS (2021) Syringomyelia presenting as neuropathic arthropathy of basal and shoulder joints. Eur J Rheumatol 8:235–236. https://doi.org/10.5152/eurjrheum.2020.20053

    Article  PubMed  Google Scholar 

  24. Drake D, McKinley W (2000) Charcot shoulder joint associated with syringomyelia: a case report. J Spinal Cord Med 23:234–237. https://doi.org/10.1080/10790268.2000

    Article  CAS  PubMed  Google Scholar 

  25. Ekim A, Armağan O (2007) Neuropathic arthropathy caused by syringomyelia in different joints and lesion of brachial plexus at right upper extremity: a case report. Agri 19:54–58

    PubMed  Google Scholar 

  26. Frankart L, Nisolle JF, Ayoubi S, Boutsen Y, Donckier J (1997) Neuroarthropathy of the shoulder of unexpected origin. Clin Rheumatol 16:413–416. https://doi.org/10.1007/BF02242461

    Article  CAS  PubMed  Google Scholar 

  27. Frewin DB, Downey JA, Feldman F, Myers SJ (1973) Neuropathic arthropathy: a report of two cases. Aust N Z J Med 3:587–592. https://doi.org/10.1111/j.1445-5994.1973.tb04300.x

    Article  CAS  PubMed  Google Scholar 

  28. Garg RK, Kar AM (2008) Charcot shoulder in syringomyelia. Intern Med J 38:868–869. https://doi.org/10.1111/j.1445-5994.2008.01784.x

    Article  CAS  PubMed  Google Scholar 

  29. Gaskins RB III, Miller BJ, Scarborough MT (2011) Charcot arthropathy of shoulder: a case report: charcot arthropathy of shoulder. Orthop Surg 3:268–270. https://doi.org/10.1111/j.1757-7861.2011.00152.x

    Article  PubMed  Google Scholar 

  30. Grahovac G, Vilendecic M, Srdoc D (2011) Charcot shoulder caused by Chiari type I malformation with syringomyelia with 6-year follow-up. Wien Klin Wochenschr 123:512–514. https://doi.org/10.1007/s00508-011-0010-3

    Article  PubMed  Google Scholar 

  31. Gunay C, Atalar E, Ataoglu B (2014) An unusual presentation of charcot arthropathy caused by syringomyelia mimicking a soft tissue tumor. Case Rep Orthop 2014:760861. https://doi.org/10.1155/2014/760861

    Article  PubMed  PubMed Central  Google Scholar 

  32. Hatzis N, Kaar TK, Wirth MA, Toro F, Rockwood CA Jr (1998) Neuropathic arthropathy of the shoulder. J Bone Joint Surg Am 80:1314–1319. https://doi.org/10.2106/00004623-199809000-00010

    Article  CAS  PubMed  Google Scholar 

  33. Hernigou J, Boulares S, Delahaut O (2018) Spontaneous posterior shoulder dislocation as the first symptom of a Charcot arthropathy. Acta Orthop Belg 84:73–77

    PubMed  Google Scholar 

  34. Hirsch M, San Martin M, Krause D (2021) Neuropathic osteoarthropathy of the shoulder secondary to syringomyelia. Diagn Interv Imaging 102:193–194. https://doi.org/10.1016/j.diii.2020.09.010

    Article  CAS  PubMed  Google Scholar 

  35. Hwang G, Nakamura R, Lee SW (2019) Imaging of progressive neuropathic arthropathy of the shoulder. Am J Phys Med Rehabil 98:e15–e16. https://doi.org/10.1097/PHM.0000000000000994

    Article  PubMed  Google Scholar 

  36. Kirksey KM, Bockenek W (2006) Neuropathic arthropathy. Am J Phys Med Rehabil 85:862. https://doi.org/10.1097/01.phm.0000237874.66989.3c

    Article  PubMed  Google Scholar 

  37. Kisabay A, Zeybek S, Serpil SA, Tanyeli A, Selcuki D (2013) A pre-and postoperative evaluation of multiple joint involvement as the initial finding of cervical syringomyelia and neuropathic arthropathy. Arch Rheumatol 28(4):271–7

    Google Scholar 

  38. Kolawole T, Banna M, Hawass N, Khan F, Rahman N (1987) Neuropathic arthropathy as a complication of post-traumatic syringomyelia. Br J Radiol 60:702–704. https://doi.org/10.1259/0007-1285-60-715-702

    Article  CAS  PubMed  Google Scholar 

  39. Kumar S, Sharma V, Kumar S, Jain S (2011) Imaging findings in Chiari I malformation with syringomyelia in a case of charcot shoulder. J Clin Imaging Sci 1:46. https://doi.org/10.4103/2156-7514.85173

    Article  PubMed  PubMed Central  Google Scholar 

  40. Liu H, Wang Y, Yang Z, Wang K (2014) A case report of charcot arthropathy caused by syringomyelia and Chiari malformation complicated with scoliosis. BMC Res Notes 7:277. https://doi.org/10.1186/1756-0500-7-277

    Article  PubMed  PubMed Central  Google Scholar 

  41. Mahmoud I, Zarrouk Z, Ben Tekaya A, Ben Salah M, Bouden S, Rouached L, Tekaya R, Saidane O, Abdelmoula L (2022) Neuropathic arthropathy of the shoulder as a presenting feature of Chiari malformation with syringomyelia: a case report with a systematic literature review. Eur Spine J. https://doi.org/10.1007/s00586-022-07299-4

    Article  PubMed  Google Scholar 

  42. Matsuhashi T, Nagahama K, Suenaga N, Oizumi N, Minami A (2011) Midterm outcomes after humeral head replacement with rotator cuff repair in patients with syringomyelia shoulder neuroarthropathy: a report on three cases. J Shoulder Elbow Surg 20:e8-15. https://doi.org/10.1016/j.jse.2011.04.032

    Article  PubMed  Google Scholar 

  43. Mazlout O, Larnaout A, El Mufti T, Kammoun N, Ladeb MF (2005) Shoulder arthropathy (Charcot joint) and syringomyelia: report of two patients. Eur J Orthop Surg Traumatol 15:37–41

    Article  Google Scholar 

  44. Mortamais J, Ramain A, Vo Hoang V, Fouque-Aubert A, Ltaief-Boudrigua A, Pialat JB (2013) Neuropathic arthropathy. answer to the e-quid “A highly damaged shoulder.” Diagn Interv Imaging 94:350–354. https://doi.org/10.1016/j.diii.2012.05.016

    Article  CAS  PubMed  Google Scholar 

  45. Murray EC (2011) Painless arm swelling. Scott Med J 56:181. https://doi.org/10.1258/smj.2011.011130

    Article  CAS  PubMed  Google Scholar 

  46. Nacir B, Arslan Cebeci S, Cetinkaya E, Karagoz A, Erdem HR (2010) Neuropathic arthropathy progressing with multiple joint involvement in the upper extremity due to syringomyelia and type I Arnold-Chiari malformation. Rheumatol Int 30:979–983. https://doi.org/10.1007/s00296-009-1013-5

    Article  PubMed  Google Scholar 

  47. Nambiar M, Onggo JR, Pai V (2018) Neuropathic arthropathy of the shoulder joint secondary to a syringomyelia. BMJ Case Rep. https://doi.org/10.1136/bcr-2018-228228

    Article  PubMed  PubMed Central  Google Scholar 

  48. Nolkha N, Srivastava D, Wakhlu A, Dhakad U (2014) Neuropathic arthropathy and syringomyelia complicating a case of ankylosing spondylitis with peripheral arthritis. BMJ Case Rep. https://doi.org/10.1136/bcr-2014-205604

    Article  PubMed  PubMed Central  Google Scholar 

  49. Ozkan K, Yavuz U, Akman B, Ozkan NK, Eralp L (2008) Charcot joint of shoulder. Eur J Orthop Surg Traumatol 18:229–231. https://doi.org/10.1007/s00590-007-0291-4

    Article  Google Scholar 

  50. Panagariya A, Sharma AK (2012) Bilateral Charcot arthropathy of shoulder secondary to syringomyelia: an unusual case report. Ann Indian Acad Neurol 15:202–204. https://doi.org/10.4103/0972-2327.99718

    Article  PubMed  PubMed Central  Google Scholar 

  51. Panda S, Madan VS, Sud S (2011) Charcot’s shoulder in syringomyelia. Neurol India 59:771–772. https://doi.org/10.4103/0028-3886.86563

    Article  PubMed  Google Scholar 

  52. Regan M, Lloyd Jones JK, Snape J (1995) Destructive large joint arthritis. Ann Rheum Dis 54:626–627. https://doi.org/10.1136/ard.54.8.626

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  53. Richards RR, Delaney J (1992) Syringomyelia presenting as shoulder instability. J Shoulder Elbow Surg 1:155–161. https://doi.org/10.1016/1058-2746(92)90094-J

    Article  CAS  PubMed  Google Scholar 

  54. Riente L, Frigelli S, Sedie AD (2002) Neuropathic shoulder arthropathy associated with syringomyelia and Arnold-Chiari Malformation (Type I). J Rheumatol 29:638–639

    PubMed  Google Scholar 

  55. Ruette P, Stuyck J, Debeer P (2007) Neuropathic arthropathy of the shoulder and elbow associated with syringomyelia: a report of 3 cases. Acta Orthop Belg 73:525–529

    PubMed  Google Scholar 

  56. Sackellares JC, Swift TR (1976) Shoulder enlargement as the presenting sign in syringomyelia. report of two cases and review of the literature. JAMA 236:2878–2879

    Article  CAS  PubMed  Google Scholar 

  57. Sankhe A, Shah A, Deshpande S, Kshirsagar R (2021) Charcot joint in a case of syringomyelia with spinal dysraphism. BMJ Case Rep 14:e239497. https://doi.org/10.1136/bcr-2020-239497

    Article  PubMed  Google Scholar 

  58. Singer L, Brust JCM, Chailenor YB (1992) Syringomyelia presenting as shoulder dysfunction. Arch Phys Med Rehabil 73:285–288

    CAS  PubMed  Google Scholar 

  59. Su J, Al-Delfi F, Mills G, Peddi P (2016) Charcot’s osteoarthropathy mimicking an osteosarcoma of humerus. BMJ Case Rep. https://doi.org/10.1136/bcr-2015-212638

    Article  PubMed  PubMed Central  Google Scholar 

  60. Makihara T, Onishi S, Wadano Y, Fujii K, Nagata H, Sakane M (2015) Regrowth of the deteriorated glenoid in advanced Charcot shoulder after suboccipital decompression for syringomyelia: a case report. J Shoulder Elbow Surg 24:e223–e228. https://doi.org/10.1016/j.jse.2015.04.022

    Article  PubMed  Google Scholar 

  61. Tristano AG, Willson ML, Montes de Oca I (2005) Axillary vein thrombosis as a manifestation of rapidly progressive neuropathic arthropathy of the shoulder associated with syringomyelia. Mayo Clin Proc 80:416–418. https://doi.org/10.4065/80.3.416

    Article  PubMed  Google Scholar 

  62. Ueyama K, Okada A, Echigoya N, Yokoyama T, Harata S (2001) Neurotrophic arthropathy caused by spinal disorders. J Musculoskelet Res 5:65–72. https://doi.org/10.1142/S0218957701000362

    Article  Google Scholar 

  63. Wawrzyniak A, Lubiatowski P, Kordasiewicz B, Brzóska R, Laprus H (2022) Shoulder arthropathy secondary to syringomyelia: case series of 10 patients. Eur J Orthop Surg Traumatol 32:1275–1281. https://doi.org/10.1007/s00590-021-03102-0

    Article  PubMed  Google Scholar 

  64. Wakhlu A, Wakhlu A, Tandon V, Krishnani N (2017) “Smouldering conditions of the shoulder” lest we forget! Indian J Rheumatol 12:169–174. https://doi.org/10.4103/injr.injr_43_17

    Article  Google Scholar 

  65. Walbom-Jørgensen S (1966) Neuropathy of the shoulder joint primarily diagnosed as sarcoma. Clin Radiol 17:365–367. https://doi.org/10.1016/s0009-9260(66)80055-7

    Article  PubMed  Google Scholar 

  66. Wang X, Li Y, Gao J, Wang T, Li Z (2018) Charcot arthropathy of the shoulder joint as a presenting feature of basilar impression with syringomyelia: a case report and literature review. Medicine (Baltimore) 97:e11391. https://doi.org/10.1097/MD.0000000000011391

    Article  PubMed  Google Scholar 

  67. Yanık B, Tuncer S, Seçkin B (2004) Neuropathic arthropathy caused by Arnold-Chiari malformation with syringomyelia. Rheumatol Int 24:238–241. https://doi.org/10.1007/s00296-003-0389-x

    Article  PubMed  Google Scholar 

  68. Rickert MM, Cannon JG, Kirkpatrick JS (2019) Neuropathic arthropathy of the shoulder: a systematic review of classifications and treatments. JBJS Rev 7:e1. https://doi.org/10.2106/JBJS.RVW.18.00155

    Article  PubMed  Google Scholar 

  69. Ueblacker P, Ansah P, Vogt S, Imhoff AB (2007) Bilateral reverse shoulder prosthesis in a patient with severe syringomyelia. J Shoulder Elbow Surg 16:e48-51. https://doi.org/10.1016/j.jse.2006.12.003

    Article  PubMed  Google Scholar 

  70. Alpert SW, Koval KJ, Zuckerman JD (1996) Neuropathic arthropathy: review of current knowledge. J Am Acad Orthop Surg 4:100–108. https://doi.org/10.5435/00124635-199603000-00005

    Article  CAS  PubMed  Google Scholar 

  71. Ducreux D, Attal N, Parker F, Bouhassira D (2006) Mechanisms of central neuropathic pain: a combined psychophysical and fMRI study in syringomyelia. Brain 129:963–976. https://doi.org/10.1093/brain/awl016

    Article  PubMed  Google Scholar 

  72. Hatem SM, Attal N, Ducreux D, Gautron M, Parker F, Plaghki L, Bouhassira D (2010) Clinical, functional and structural determinants of central pain in syringomyelia. Brain 133:3409–3422. https://doi.org/10.1093/brain/awq244

    Article  PubMed  Google Scholar 

  73. Jones EA, Manaster BJ, May DA, Disler DG (2000) Neuropathic osteoarthropathy: diagnostic dilemmas and differential diagnosis. Radiographics. https://doi.org/10.1148/radiographics.20.suppl_1.g00oc22s279

    Article  PubMed  Google Scholar 

  74. Olsson L, Antepohl W, Ravn SL (2022) Active conservative management of primary spinal syringomyelia: a scoping review and perspectives for an activity-based clinical approach. J Rehabil Med. https://doi.org/10.2340/jrm.v54.2398

    Article  PubMed  PubMed Central  Google Scholar 

  75. Schoch B, Werthel JD, Sperling JW, Cofield RH, Sanchez-Sotelo J (2016) Is shoulder arthroplasty an option for charcot arthropathy? Int Orthop 40:2589–2595. https://doi.org/10.1007/s00264-016-3309-x

    Article  PubMed  Google Scholar 

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BFK and BK designed the study. BFK and BK reviewed the literature and collected the data. BFK analyzed the data. BFK and BK interpreted the data analyses. BFK and BK drafted the study and reviewed it critically for important intellectual content. BFK and BK prepared tables and figures. BFK and BK approved the final version of the manuscript and agree to be accountable for all aspects of the work.

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Kocyigit, B.F., Kızıldağ, B. Neuropathic arthropathy of the shoulder secondary to operated syringomyelia: a case-based review. Rheumatol Int 43, 777–790 (2023). https://doi.org/10.1007/s00296-022-05234-w

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