Preview

Russian neurological journal

Advanced search

Idiopathic Harlequin syndrome.

https://doi.org/10.30629/2658-7947-2023-28-5-67-70

Abstract

Harlequin syndrome (HA) is a rare pathology. It is manifested by isolated unilateral facial flushing (less often the face and limb), sometimes with concomitant hyperhidrosis and Horner’s syndrome. In most cases, this is an idiopathic condition with a benign course. However, HA can be a manifestation of serious diseases, as well as a complication of surgical interventions at the cervical and upper thoracic levels. We present clinical case of a female patient with Harlequin syndrome as well as a short review of scientific literature.

About the Authors

V. B. Voitenkov
Pediatric Research and Clinical Center for Infectious Diseases; Academy of Postgraduate Education of Federal Scientific Research Center of the Federal Medical and Biological Agency
Russian Federation

 

Voitenkov Vladislav B.,

Saint Petersburg;

Moscow.



V. N. Komantsev
Saint Petersburg Postgraduate Institute of Medical experts Labor and Social Protection
Russian Federation

Saint Petersburg.



E. V. Ekusheva
Academy of Postgraduate Education of Federal Scientific Research Center of the Federal Medical and Biological Agency; Belgorod State National Research University
Russian Federation

Moscow;

Belgorod.



V. Yu. Romanenko
State Healthcare Institution Saratov City Clinical Hospital No. 9
Russian Federation

Saratov.



M. A. Irikova
Pediatric Research and Clinical Center for Infectious Diseases
Russian Federation

Saint Petersburg.



References

1. Belopasov V.V., Gubanova M.V., Belopasova A.V. Kalashnikova L.A., Dobrinina L.A. Harlequin’s face is a rare form of autonomic dysfunction in dissection of the internal carotid artery. Clinical practice. 2019;10(1):88–93. (In Russ.). DOI: 10.17816/clinpract10188–93

2. Lehman K., Kumar N., Vu Q., Wells R. Harlequin Syndrome in Cluster Headache. Headache. 2016;56(6):1053–1054. PMID: 27238298. DOI: 10.1111/head.12852

3. Elboukhari K., Baybay H., Elloudi S. Idiopathic harlequin syndrome: a case report and literature review. Pan Afr Med J. 2019;33:141. PMID: 31558939. DOI: 10.11604/pamj.2019.33.141.18102

4. Lance J.W., Drummond P.D., Gandevia S.C., Morris J.G. Harlequin syndrome: the sudden onset of unilateral flushing and sweating. J Neurol Neurosurg Psychiatry. 1988;51(5):635–642. PMID: 3155385. DOI: 10.1136/jnnp.51.5.635

5. Guilloton L., Demarquay G., Quesnel L. Dysautonomic syndrome of the face with harlequin sign and syndrome: three new cases and a review of the literature. Rev Neurol (Paris). 2013;169(11):884–891. PMID: 23726525. 10.1016/j.neurol.2013.01.628

6. Persson R.M., Tellnes K., Hoven H. Harlequin syndrome associated with thoracic epidural anaesthesia. Anaesth Rep. 2022;10(1):10.1002/anr3.12144. PMID: 35118419. DOI: 10.1002/anr3.12144

7. Chrzanowski S., Baker A., Hayes L. Harlequin Syndrome, Apnea, and Acute on Chronic Hemiparesis: An Atypical Pediatric Case of Chiari I With Holocord Syringohydromyelia. Pediatr Neurol. 2022;126:108–109. PMID: 34788683. DOI: 10.1016/j.pediatrneurol.2021.10.002

8. Tatebe M., Kawakami D. Harlequin syndrome after surgery for aortic dissection. Intensive Care Med. 2021;47(12):1485–1486. PMID: 34286361. DOI: 10.1007/s00134-021-06477-0

9. Kim H.O., Kim J.C., Chung B.Y. Harlequin syndrome with petechiae caused by a pituitary adenoma. J Eur Acad Dermatol Venerol. 2021;35(9):e605–e607. PMID: 33974308. DOI: 10.1111/jdv.17338

10. Miyata K., Akaihata M., Shimomura Y. Harlequin syndrome associated with ganglioneuroblastoma-induced Horner syndrome. Childs Nerv Syst. 2021;37(8):2683–2686. PMID: 33125512. DOI: 10.1007/s00381-020-04948-1

11. McKenna M.C., Menon P., Smyth S., Murphy S. Harlequin Syndrome in Acute Thalamic Hemorrhage. J Stroke Cerebrovasc Dis. 2019;28(9):e127–e128. PMID: 31301985. DOI: 10.1016/j.jstrokecerebrovasdis.2019.06.037

12. Halle A., De Becdelievre A., Funalot B. SCN10A variants associated with congenital Harlequin Syndrome. Br J Dermatol. 2022;186(6):1039–1041. PMID: 35007332. DOI: 10.1111/bjd.21011 .

13. Naharro-Fernández C., de Quintana-Sancho A., LópezSundh A.E. Successful treatment of idiopathic Harlequin Syndrome with oxybutynin and propranolol. Australas J Dermatol. 2021;62(4):504–505. PMID: 34314021. DOI: 10.1111/ajd.13665

14. Manhães R.K., Spitz M., Vasconcellos L.F. Botulinum toxin for treatment of Harlequin syndrome. Parkinsonism Relat Disord. 2016;23:112–113. PMID: 26750113. DOI: 10.1016/j.parkreldis.2015.11.030

15. Kim H.J., Seo K., Yum K.W., Oh Y.S., Yoon T.G., Yoon S.M. Effects of botulinum toxin type a on the superior cervical ganglia in rabbits. Auton. Neurosci. 2002;102:8–12. doi: 10.1016/S1566-0702(02)00093-0

16. Hans-Bittner N.R., Bittner G.C., Hans Filho G. Do you know this syndrome? Harlequin syndrome. An Bras Dermatol. 2018;93(4):585–586. PMID: 30066772. DOI: 10.1590/abd1806-4841.20187549

17. Moon S.Y., Shin D.I., Park S.H., Kim J.S. Harlequin syndrome with crossed sympathetic deficit of the face and arm. J Korean Med Sci. 2005;20(2):329–330. PMID: 15832011. DOI: 10.3346/jkms.2005.20.2.329

18. Breunig J.de A., Hartmann M., Freire C.F., de Almeida H.L. Jr. Harlequin syndrome in childhood—case report. An Bras Dermatol. 2012;87(6):907–909. PMID: 23197213. DOI: 10.1590/s0365-05962012000600015

19. Rovner M.S., Redding A.T., Wolf B.J. Wharton J.A., Risely C.J., Furse C.M. Detection of subclinical Harlequin syndrome in pediatric patients. Paediatr Anaesth. 2020;30(5):592–598. PMID: 32160375. DOI: 10.1111/pan.13852

20. Honore P.M., Barreto Gutierrez L., Kugener L. Redant S, Attou R, Gallerani A. Risk of harlequin syndrome during bi-femoral peripheral VA-ECMO: should we pay more attention to the watershed or try to change the venous cannulation site? Crit Care. 2020;24(1):450. PMID: 32690070. DOI: 10.1186/s13054-020-03168-y

21. Pasrija C., Bedeir K., Jeudy J., Kon Z.N. Harlequin Syndrome during Venoarterial Extracorporeal Membrane Oxygenation. Radiol Cardiothorac Imaging. 2019;1(2):e190031. PMID: 33778505. DOI: 10.1148/ryct.2019190031


Review

For citations:


Voitenkov V.B., Komantsev V.N., Ekusheva E.V., Romanenko V.Yu., Irikova M.A. Idiopathic Harlequin syndrome. Russian neurological journal. 2023;28(5):67-70. (In Russ.) https://doi.org/10.30629/2658-7947-2023-28-5-67-70

Views: 3328


Creative Commons License
This work is licensed under a Creative Commons Attribution 4.0 License.


ISSN 2658-7947 (Print)
ISSN 2686-7192 (Online)