265
Views
0
CrossRef citations to date
0
Altmetric
Case Report

Recognition of a patient with neck autonomic dysfunction: findings from a rare case report of harlequin syndrome in direct access physiotherapy

ORCID Icon, , ORCID Icon, , , ORCID Icon, ORCID Icon, ORCID Icon, ORCID Icon & ORCID Icon show all
Received 16 Sep 2023, Accepted 24 Apr 2024, Published online: 17 May 2024

References

  • Ritter N, Bittner H, Bittner GC, et al. Do you know this syndrome? Harlequin syndrome. An Bras Dermatol. 2018;93(4):585–586. doi: 10.1590/abd1806-4841.20187549
  • Mavroudis I, Balmus IM, Ciobica A, et al. Mini-review on the harlequin syndrome-A rare dysautonomic manifestation requiring attention. Medicina (Kaunas). 15 2022;58(7):938. PMID: 35888657; PMCID: PMC9324885. doi:10.3390/medicina58070938
  • Willaert WI, Scheltinga MR, Steenhuisen SF, et al. Harlequin syndrome: two new cases and a management proposal. Acta Neurol Belg. 2009 Sep;109(3):214–220. PMID: 19902816.
  • Mourad F, Giudice A, Maritati G, et al. A guide to identify cervical autonomic dysfunctions (and associated conditions) in patients with musculoskeletal disorders in physical therapy practice. Braz J. 2023;27(2):100495. doi: 10.1016/j.bjpt.2023.100495
  • Wasner G, Maag R, Ludwig J, et al. Harlequin syndrome—one face of many etiologies. Nat Clin Pract Neurol. 2005;1(1):54–59. doi: 10.1038/ncpneuro0040
  • Nida T, Nilgun ST, Zuhal E, et al. Unnoticed dysautonomic syndrome of the face: Harlequin syndrome. Auton Neurosci Basic Clin. 2007;137(1–2):1–9. doi: 10.1016/j.autneu.2007.05.004
  • Drummond PD, Lance JW. Facial flushing and sweating mediated by the sympathetic nervous system. Brain. 1987;110(3):793–803. doi: 10.1093/brain/110.3.793
  • Sarikaya H, Georgiadis D, Baumgartner RW. Harlequin syndrome in spontaneous dissection of the cervical carotid artery. Neurology. 2008;71(18):1459. doi: 10.1212/01.wnl.0000327877.74795.d7
  • Pradeep PV, Benede AK, Harshita SS, et al. Harlequin syndrome in a case of toxic goitre: a rare association. Case RepMed. 2011;2011:1–4. doi: 10.1155/2011/293076
  • Kilinc¸er C, Oztürk L, Hamamcioglu MK, et al. An upper thoracic spinal cord tumor presenting as hemifacial hyperhidrosis. Surg Neurol. 2007;68(4):461–463. doi: 10.1016/j.surneu.2006.10.067
  • Duddy ME, Baker MR. Images in clinical medicine. Harlequin’s Darker Side N Engl J Med. 2007 Nov 15;357(20):e22. doi: 10.1056/NEJMicm067851
  • Jiménez PE. Síndrome del arlequín secundario a condrosarcoma torácico. Rev Neurol. 2008;46(4):252–253. doi: 10.33588/rn.4604.2007603
  • Carroll CB, Zajicek JP. The ‘‘harlequin’’ sign in association with multiple sclerosis. J Neurol. 2004;25(9):1145–1146. doi: 10.1007/s00415-004-0486-0
  • Esterly NB, Spraker MK. Neonatal skin problems. In: Moschella SL, and Hurley HJ, editors. Dermatology. Philadelphia, Saunders: W.B. Saunders Company; 1985. Vol. 2. p. 1882.
  • Coleman PJ, Goddard JM. Harlequin syndrome following internal jugular vein catheterisation in an adult under general anesthetic. Anesthesiology. 2002;97(4):1041. doi: 10.1097/00000542-200210000-00070
  • Burlacu CL, Buggy DJ. Coexisting harlequin and Horner syndromes after high thoracic paravertebral anaesthesia. Br J Anaesth. 2005;95(6):822–824. doi: 10.1093/bja/aei258
  • Turco GR, Farber NE. Postoperative autonomic deficit: a case of harlequin syndrome. Anesthesiology. 1996;85(5):1197–1199. doi: 10.1097/00000542-199611000-00031
  • Bremner F, Smith S. Pupillographic findings in 39 consecutive cases of harlequin syndrome. J Neuro-Ophthalmol. 2008;17(3):171–177. doi: 10.1097/WNO.0b013e318183c885
  • Taylor A, Mourad F, Kerry R, et al. A guide to cranial nerve testing for musculoskeletal clinicians. J Man Manip Ther. 2021;29(6):376–389. doi: 10.1080/10669817.2021.1937813
  • Mourad F, Lopez G, Cataldi F, et al. Assessing cranial nerves in physical therapy practice: findings from a cross-sectional survey and implication for clinical practice. Healthcare (Basel). 2021;9(10):1262. doi: 10.3390/healthcare9101262
  • Adeboye KA, Emerton DG, Hughes T. Cervical sympathetic chain dysfunction after whiplash injury. J R Soc Med. 2000;93(7):378–379. doi: 10.1177/014107680009300713
  • Maselli F, Piano L, Cecchetto S, et al. Reply to Moretti et al. Would moving forward mean going back? Comment on “Maselli et al. Direct access to physical therapy: should Italy move forward? int. J. Environ. Res. Public health 2022, 19, 555”. Int J Environ Res Public Health. 2022;19(8):4620. doi: 10.3390/ijerph19084620
  • Maselli F, Piano L, Cecchetto S, et al. Direct access to physical therapy: should Italy move forward? Int J Environ Res Public Health. 2022;19(1):555. doi: 10.3390/ijerph19010555
  • Piscitelli D, Furmanek MP, Meroni R, et al. Direct access in physical therapy: a systematic review. Clin Ter. 2018;169(5):e249–e260. doi: 10.7417/CT.2018.2087
  • Faletra A, Bellin G, Dunning J, et al. Assessing cardiovascular parameters and risk factors in physical therapy practice: findings from a cross-sectional national survey and implication for clinical practice. BMC Musculoskelet Disord. 2022;23(1):749. doi: 10.1186/s12891-022-05696-w
  • Gagnier JJ, Kienle G, Altman DG, et al. CARE Group*. The CARE guidelines: consensus-based clinical case reporting guideline development. Glob Adv Health Med. 2013;2(5):38–43. doi: 10.7453/gahmj.2013.008
  • ) Whelton PK; Carey RM; Aronow WS; Casey DE Jr; Collins KJ; Dennison Himmelfarb C; et al. 2017ACC/AHA/AAPA/ABC/ACPM/AGS/APhA/ASH/ASPC/NMA/PCNA guideline for the prevention, detection, evaluation, and management of high blood pressure in adults: executive summary: a report of the American college of cardiology/American heart association task force on clinical practice guidelines. Hypertension. 2018 Jun;71(6): 1269-1324. 10.1161/HYP.0000000000000066. Epub 2017 Nov 13. Erratum in: hypertension. 2018 Jun;71(6): e136-e139. Erratum in: hypertension. 2018 Sep;72(3): e33. PMID: 29133354.
  • Currie KD, Floras JS, La Gerche A, et al. Exercise blood pressure guidelines: time to Re-evaluate what is normal and exaggerated? Sports Med. 2018 Aug;48(8):1763–1771. doi: 10.1007/s40279-018-0900-x. PMID: 29574665.
  • Shin RK, Galetta SL, Ting TY, et al. Ross syndrome plus: beyond Horner, Holmes-Adie, and harlequin. Neurology. 2000;55(12):1841–1846. doi: 10.1212/wnl.55.12.1841
  • Martinelli P. Holmes-adie syndrome. Lancet. 2000;356(9243):1760–1761. Erratum in: Lancet 2001, 357(9255),562. Erratum in: Lancet 2002, 359(9300), 84. doi: 10.1016/S0140-6736(00)03216-5
  • Bremner F, Smith S. Pupillographic findings in 39 consecutive cases of harlequin syndrome. J Neuro-Ophthalmol. 2008;28(3):171–177. doi: 10.1097/WNO.0b013e318183c885
  • Kerry R, Taylor A. Cervical spine pre-treatment screening for arterial dysfunction: out with the old, in with the new. Touch. 2014;147(Summer):10–14. doi: 10.13140/2.1.1509.9526
  • Müller M:, Jürgens J, Redaèlli M, et al. Impact of the communication and patient hand-off tool SBAR on patient safety: a systematic review. BMJ Open. 2018;8(8):e022202. doi: 10.1136/bmjopen-2018-022202
  • Mourad F, Lopez G, Cataldi F, et al. Assessing cranial nerves in physical therapy practice: findings from a cross-sectional survey and implication for clinical practice. Healthc. 2021;9(10):1262. doi: 10.3390/healthcare9101262
  • Lemeunier N, da Silva-Oolup S, Chow N, et al. Reliability and validity of clinical tests to assess the anatomical integrity of the cervical spine in adults with neck pain and its associated disorders: part 1-A systematic review from the Cervical Assessment and Diagnosis Research Evaluation (CADRE) collaboration. Eur Spine J. 2017;26(9):2225–2241. doi: 10.1007/s00586-017-5153-0
  • Schmid AB, Brunner F, Luomajoki H, et al. Reliability of clin- ical tests to evaluate nerve function and mechanosensitivity of the upper limb peripheral nervous system. BMC Musculos- Kelet Disord. 2009;10(1):11. doi: 10.1186/1471-2474-10-11
  • El-Badawy MA, El Mikkawy DM. Sympathetic dysfunction in patients with chronic low back pain and failed back surgery syndrome. Clin J Pain. 2016;32(3):226–231. doi: 10.1097/AJP.0000000000000250
  • Meroni R, Piscitelli D, Ravasio C, et al. Evidence for managing chronic low back pain in primary care: a review of recommendations from high-quality clinical practice guidelines. Disabil Rehabil. 2021;43(7):1029–1043. doi: 10.1080/09638288.2019.1645888
  • Gianola S, Bargeri S, Cinquini M, et al. More than one third of clinical practice guidelines on low back pain overlap in AGREE II appraisals. Research Wasted? BMC Med Res Methodol. 2022;22(1):184. doi: 10.1186/s12874-022-01621-w
  • Maselli F, Storari L, Barbari V, et al. Prevalence and incidence of low back pain among runners: a systematic review. BMC Musculoskelet Disord. 2020 Jun 3;21(1):343. doi: 10.1186/s12891-020-03357-4. PMID: 32493481; PMCID: PMC7271446.
  • Martinez-Lavin M, Biology, Therapy of Fibromyalgia. Stress, the stress response system, and fibromyalgia. Arthritis Res Ther. 2007;9(4):216. doi: 10.1186/ar2146
  • Zaproudina N, Ming Z, Närhi M. Sensory and sympathetic disorders in chronic non-specific neck pain. Funct Neurol. 2015;30(3):165–171. doi: 10.11138/fneur/2015.30.3.165
  • Mani R, Cooper C, Kidd BL, et al. Use of laser Doppler flowmetry and transcutaneous oxygen tension electrodes to assess local autonomic dysfunction in patients with frozen shoulder. J R Soc Med. 1989;82(9):536–538. doi: 10.1177/014107688908200910
  • Courties A, Sellam J, Berenbaum F. Role of the autonomic nervous system in osteoarthritis. Best pract res Clin Rheumatol. 2017;31(5):661–675.
  • Suri S, Gill SE, Massena de Camin S, et al. Neurovascular invasion at the osteochondral junction and in osteophytes in osteoarthritis. Ann Rheum Dis. 2007;66(11):1423–1428. doi: 10.1136/ard.2006.063354
  • Lorenz J, Schäfer N, Bauer R, et al. Norepinephrine modulates osteoarthritic chondrocyte metabolism and inflammatory responses. Osteoarthritis Cartilage. 2016;24(2):325–334. doi: 10.1016/j.joca.2015.08.007
  • Zabalza Estévez RJ, Unanue López F. Harlequin syndrome, a rare neurological disease. Neurologia. 2015;30(3):185–187 English, Spanish. 10.1016/j.nrl.2013.04.007
  • Walsh FB, Kerrison JB, Newman NJ, et al. Anatomy and physiology of the autonomic nervous system. In: Neil RM, Nancy JN, Valerie B, John BK, editors. Walsh and Hoyt’s clinical neuro-ophthalmology. 6th ed. Vol. 2. Baltimore (MD), USA: Lippincott-Williams & Wilkins; 2005. pp. 649–714. ISBN 9781975118921.
  • Drummond PD, Finch PM. Reflex control of facial flushing during body heating in man. Brain. 1989 Oct;112(Pt 5):1351–1358. doi: 10.1093/brain/112.5.1351. PMID: 2804616.Maselli, F.; Testa, M.; Superficial peroneal nerve schwannoma presenting as lumbar radicular syndrome in a non-competitive runner. Journal of Back and Musculoskeletal Rehabilitation, 2019, 32(2), pp. 361–365. Doi: 10.3233/BMR-181164.
  • Sterling M. Whiplash-associated disorder: musculoskeletal pain and related clinical findings. J Man Manip Ther. 2011;19(4):194–200. doi: 10.1179/106698111X13129729551949
  • Sterling M, Kenardy J. The relationship between sensory and sympathetic nervous system changes and posttraumatic stress reaction following whiplash injury—a prospective study. J Psychosom Res. 2006;60(4):387–393. doi: 10.1016/j.jpsychores.2005.08.016
  • Munglani R. Neurobiological mechanisms underlying chronic whiplash associated pain. J Musculoskelet Pain. 2000;8(1–2):169–178. doi: 10.1300/J094v08n01_14
  • Pauling JD, Hughes M, Pope JE. Raynaud’s phenomenon-an update on diagnosis, classification and management. Clin Rheumatol. 2019;38(12):3317–3330. doi: 10.1007/s10067-019-04745-5
  • Devgire V, Hughes M. Raynaud’s phenomenon. Br J Hosp Med. 2019;80(11):658–664. doi: 10.12968/hmed.2019.80.11.658
  • Sterling M, Jull G, Vicenzino B, et al. Sensory hypersensitivity occurs soon after whiplash injury and is associated with poor recovery. Pain. 2003;104(3):509–517. doi: 10.1016/S0304-3959(03)00078-2
  • Sterling M, Jull G, Vicenzino B, et al. Physical and psychological factors predict outcome following whiplash injury. Pain. 2005;114(1–2):141–148. doi: 10.1016/j.pain.2004.12.005
  • Sansone VC, Meroni R, Boria P, et al. Are occupational repetitive movements of the upper arm associated with rotator cuff calcific tendinopathies? Rheumatol Int. 2015;35(2):273–280. doi: 10.1007/s00296-014-3086-z
  • Sansone V, Bonora C, Boria P, et al. Women performing repetitive work: is there a difference in the prevalence of shoulder pain and pathology in supermarket cashiers compared to the general female population? Int J Occup Med Environ Health. 2014 Oct;27(5):722–735. doi: 10.2478/s13382-014-0292-6
  • Hutting N, Mourad F, Kranenburg R, et al. What to look out for, what to do, and when: 3 key messages for safely treating neck pain, headache, and/or orofacial symptoms in musculoskeletal rehabilitation settings. J. 2023;53(2):59–63. doi: 10.2519/jospt.2022.11568
  • Hutting N, Kerry R, Kranenburg R, et al. Assessing vascular function in patients with neck pain, headache, and/or orofacial pain: part of the job description of all physical therapists. J Orthop Sports Phys Ther. 2021;51(9):418–421. doi: 10.2519/jospt.2021.10408
  • Taylor A, Kerry R, Mourad F, et al. Vascular flow limitations affecting the cervico-cranial region: understanding ischaemia. Braz J Phys Ther. 2023;27(3):100493. doi: 10.1016/j.bjpt.2023.100493
  • Maselli F, Testa M. Superficial peroneal nerve schwannoma presenting as lumbar radicular syndrome in a non-competitive runner. J Back Musculoskelet Rehabil. 2019;32(2):361–365. doi: 10.3233/BMR-181164
  • Maselli F, Rossettini G, Viceconti A, et al. Importance of screening in physical therapy: vertebral fracture of thoracolumbar junction in a recreational runner. BMJ Case Rep. 2019;12(8):e229987. doi: 10.1136/bcr-2019-229987
  • Pennella D, Giagio S, Maselli F, et al. Red flags useful to screen for gastrointestinal and hepatic diseases in patients with shoulder pain: a scoping review. Musculoskelet Care. 2022;20(4):721–730. doi: 10.1002/msc.1628
  • Mourad F, Cataldi F, Patuzzo A, et al. Craniopharyngioma in a young woman with symptoms presenting as mechanical neck pain associated with cervicogenic headache: a case report. Physiother Theory Pract. 2019;37(4):549–558. doi: 10.1080/09593985.2019.1636433
  • Mourad F, Giovannico G, Maselli F, et al. Basilar impression presenting as intermittent mechanical neck pain: a rare case report. BMC Musculoskelet Disord. 2016;17(1):7. doi: 10.1186/s12891-015-0847-0
  • Wehrwein EA, Orer HS, Barman SM. Overview of the anatomy, physiology, and pharmacology of the autonomic nervous system. Compr Physiol. 2016 Jun 13;6(3):1239–1278. doi: 10.1002/cphy.c150037. PMID: 27347892.
  • Finucane LM, Downie A, Mercer C, et al. International framework for red flags for potential serious spinal pathologies. J Orthop Sports Phys Ther. 2020 Jul;50(7):350–372. doi: 10.2519/jospt.2020.9971. Epub 2020 May 21. PMID: 32438853.

Reprints and Corporate Permissions

Please note: Selecting permissions does not provide access to the full text of the article, please see our help page How do I view content?

To request a reprint or corporate permissions for this article, please click on the relevant link below:

Academic Permissions

Please note: Selecting permissions does not provide access to the full text of the article, please see our help page How do I view content?

Obtain permissions instantly via Rightslink by clicking on the button below:

If you are unable to obtain permissions via Rightslink, please complete and submit this Permissions form. For more information, please visit our Permissions help page.