r-n-jРоссийский неврологический журналRussian neurological journal2658-79472686-7192МИА10.30629/2658-7947-2023-28-5-67-70r-n-j-486Research ArticleИССЛЕДОВАНИЯ И КЛИНИЧЕСКИЕ НАБЛЮДЕНИЯCLINICAL RESEARCHES AND CASE REPORTSИдиопатический синдром АрлекинаIdiopathic Harlequin syndrome.https://orcid.org/0000-0003-0448-7402ВойтенковВ. Б.VoitenkovV. B.

Войтенков Владислав Борисович, 

Санкт-Петербург;

Москва.

 

Voitenkov Vladislav B.,

Saint Petersburg;

Moscow.

vlad203@inbox.ruhttps://orcid.org/0000-0003-2646-9790КоманцевВ. Н.KomantsevV. N.

Санкт-Петербург.

Saint Petersburg.

https://orcid.org/0000-0002-3638-6094ЕкушеваЕ. В.EkushevaE. V.

Москва;

Белгород.

Moscow;

Belgorod.

https://orcid.org/0000-0003-1872-6697РоманенкоВ. Ю.RomanenkoV. Yu.

Саратов.

Saratov.

https://orcid.org/0000-0001-8924-5300ИриковаМ. А.IrikovaM. A.

Санкт-Петербург.

Saint Petersburg.

Детский научно-клинический центр инфекционных болезней Федерального медико-биологического агентства; Академия постдипломного образования ФГБУ «Федеральный научно-клинический центр специализированных видов медицинской помощи и медицинских технологий Федерального медико-биологического агентства»РоссияPediatric Research and Clinical Center for Infectious Diseases; Academy of Postgraduate Education of Federal Scientific Research Center of the Federal Medical and Biological AgencyRussian FederationСанкт-Петербургский институт усовершенствования врачей-экспертовРоссияSaint Petersburg Postgraduate Institute of Medical experts Labor and Social ProtectionRussian FederationАкадемия постдипломного образования ФГБУ «Федеральный научно-клинический центр специализированных видов медицинской помощи и медицинских технологий Федерального медико-биологического агентства»; Белгородский государственный национальный исследовательский университетРоссияAcademy of Postgraduate Education of Federal Scientific Research Center of the Federal Medical and Biological Agency; Belgorod State National Research UniversityRussian FederationСаратовская городская клиническая больницаРоссияState Healthcare Institution Saratov City Clinical Hospital No. 9Russian FederationДетский научно-клинический центр инфекционных болезней Федерального медико-биологического агентстваРоссияPediatric Research and Clinical Center for Infectious DiseasesRussian Federation2023261120232856770Copyright © Войтенков В.Б., Команцев В.Н., Екушева Е.В., Романенко В.Ю., Ирикова М.А., 20232023Войтенков В.Б., Команцев В.Н., Екушева Е.В., Романенко В.Ю., Ирикова М.А.Voitenkov V.B., Komantsev V.N., Ekusheva E.V., Romanenko V.Y., Irikova M.A.This work is licensed under a Creative Commons Attribution 4.0 License.https://www.r-n-j.com/jour/article/view/486

Синдром Арлекина (СА) — редкое расстройство, проявляющиееся покраснением половины лица, реже — лица и руки, иногда с сопутствующими гипергидрозом и синдромом Горнера. Его причиной могут быть автономные нарушения и другие патологические состояния. В большей части случаев это идиопатическое расстройство с доброкачественным течением. Тем не менее СА может быть проявлением серьезных заболеваний, а также осложнением хирургических вмешательств на шейном и верхнегрудном уровнях позвоночника. Представлены описание случая идиопатического СА и краткий обзор литературы.

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.

синдром Арлекинаавтономная нервная системасимпатическая системаHarlequin syndromeautonomic nervous systemsympathetic systemReferencesБелопасов В.В., Губанова М.В., Белопасова А.В. Калашникова Л.А., Добрынина Л.А. Лицо Арлекина — редкая форма вегетативной дисфункции при диссекции внутренней сонной артерии. Клиническая практика. 2019;10(1):88–93. DOI: 10.17816/clinpract10188–93Belopasov 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/clinpract10188–93Lehman K., Kumar N., Vu Q., Wells R. Harlequin Syndrome in Cluster Headache. Headache. 2016;56(6):1053–1054. PMID: 27238298. DOI: 10.1111/head.12852Lehman K., Kumar N., Vu Q., Wells R. Harlequin Syndrome in Cluster Headache. Headache. 2016;56(6):1053–1054. PMID: 27238298. DOI: 10.1111/head.12852Elboukhari 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.2019.33.141.18102Elboukhari 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.2019.33.141.18102Lance 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.51.5.635Lance 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.51.5.635Guilloton 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.2013.01.628Guilloton 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.2013.01.628Persson 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/anr3.12144Persson 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/anr3.12144Chrzanowski 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.2021.10.002Chrzanowski 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.2021.10.002Tatebe M., Kawakami D. Harlequin syndrome after surgery for aortic dissection. Intensive Care Med. 2021;47(12):1485–1486. PMID: 34286361. DOI: 10.1007/s00134-021-06477-0Tatebe M., Kawakami D. Harlequin syndrome after surgery for aortic dissection. Intensive Care Med. 2021;47(12):1485–1486. PMID: 34286361. DOI: 10.1007/s00134-021-06477-0Kim 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.17338Kim 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.17338Miyata 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/s00381-020-04948-1Miyata 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/s00381-020-04948-1McKenna 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.2019.06.037McKenna 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.2019.06.037Halle 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 .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 .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.13665Naharro-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.13665Manhães 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.2015.11.030Manhães 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.2015.11.030Kim 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)00093-0Kim 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)00093-0Hans-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/abd1806-4841.20187549Hans-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/abd1806-4841.20187549Moon 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.2005.20.2.329Moon 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.2005.20.2.329Breunig 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/s0365-05962012000600015Breunig 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/s0365-05962012000600015Rovner 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.13852Rovner 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.13852Honore 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-yHonore 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-yPasrija 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.2019190031Pasrija 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

The authors declare that there are no conflicts of interest present.