Harlequin syndrome in a young patient with osteosarcoma after pleural metastasectomy: A case report and a mini review of the literature

Moutafi,Myrto; Kyriazoglou,Anastasios; Kotsantis,Ioannis; Economopoulou,Panagiota; Efthymiou,Evgenia; Kyrkasiadou,Maria; Boulouta,Anna; Gavrielatou,Niki; Zazas,Evangelos; Markakis,Konstantinos; Psyrri,Amanda

doi:10.3892/etm.2025.12812

Harlequin syndrome in a young patient with osteosarcoma after pleural metastasectomy: A case report and a mini review of the literature

Authors:
- Myrto Moutafi
- Anastasios Kyriazoglou
- Ioannis Kotsantis
- Panagiota Economopoulou
- Evgenia Efthymiou
- Maria Kyrkasiadou
- Anna Boulouta
- Niki Gavrielatou
- Evangelos Zazas
- Konstantinos Markakis
- Amanda Psyrri
View Affiliations

Affiliations: Section of Medical Oncology, Second Department of Internal Medicine, School of Medicine, National and Kapodistrian University of Athens, Attikon University Hospital, Athens 12462, Greece, Second Department of Radiology, Attikon University Hospital, Athens 12462, Greece, Section of Internal Medicine, Second Department of Internal Medicine, School of Medicine, National and Kapodistrian University of Athens, Attikon University Hospital, Athens 12462, Greece
Published online on: January 30, 2025 https://doi.org/10.3892/etm.2025.12812
Article Number: 62

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Abstract

Harlequin syndrome is a rare autonomic disorder with at least 83 reported cases in literature, 6% of which are congenital. When the fibers responsible for sudomotor and vasomotor supply to the face at the T2‑T3 level are unilaterally blocked, it leads to hemifacial discoloration. This results in one‑half of the face appearing flushed and hyperemic, sharply contrasting with the pale appearance of the other half. The cause of this syndrome is unknown; however, it appears to involve an autonomic nervous system dysfunction. It can be caused by an injury, compression, or blockade of sympathetic fibers along the pathway. The present study was a case of a metastatic osteosarcoma patient with a history of video‑assisted thoracic surgery pleurectomy that presented in Attikon University Hospital (Athens, Greece) with worsening dyspnea and right‑sided facial flushing.

Introduction

Harlequin syndrome (HS), initially reported by Lance et al in 1988, is a rare autonomic disorder caused by hemifacial cutaneous sympathetic denervation (1,2,3). It is characterized by semi-facial sweating and discoloration, sharply demarcated at the midline with normal ocular sympathetic innervation (3). It is mainly idiopathic in nature, often triggered by exercise, emotions or exposure to heat. HS is used to describe the transient hemi body flushing often observed in premature neonates due to immaturity of the hypothalamic centers and vasomotor instability (5). Congenital cases represent ~6% of all the patients with HS (3,6). However, it can also be a manifestation of underlying cervical and upper thoracic pathologies resulting in the compression of these sympathetic fibers or iatrogenic from the surgical or anesthetic procedures around the neck. The present study revealed, to the best of the authors' knowledge, the first case of an oncologic patient with HS as a result of pleural metastasectomy with video-assisted thoracic surgery (VATS).

Case report

A 28-year-old woman with a history of metastatic osteosarcoma was presented in Attikon University Hospital (Athens, Greece) with 5 days of worsening dyspnea, in September 2023. Computed tomography (CT) revealed new metastatic lung lesions. She had received neoadjuvant methotrexate, doxorubicin and cisplatin (MAP) (7) in October 2020 and undergone surgical removal of the tumoral mass of the right proximal humerus in February 2021. Histopathological examination from the lesion was suggestive of low-grade osteosarcoma (mdm2 amplified). Afterwards the female patient completed adjuvant treatment with MAP and Mifamurtide. In July 2022 contrast-enhanced CT thorax revealed new pleural nodules located mainly on left upper lobe. Pleural biopsy confirmed disease progression. After receiving three cycles of high dose ifosfamide (8) in September 2022, progression was reported with pleural metastatic lesions and clinical worsening. VATS metastasectomy was decided by the multidisciplinary team and the female patient underwent partial left pneumonectomy, pericardiectomy and pleurectomy in January 2023. The female patient was then enrolled in phase IV clinical trial and had already received three cycles of cabozatinib when presented in Attikon University Hospital (Athens, Greece) with new metastatic lung lesions and new onset dyspnea in September 2023.

Following emotional distress, on the third day of admission, the female patient presented with right-sided facial flushing and profuse sweating with the left side of the face remaining pale and dry (Fig. 1). The symptoms were resolved spontaneously after 15 min. The physical examination did not reveal any neurological alterations. No ophthalmological abnormalities were observed. Dermatological examination at rest revealed no abnormalities. Routine laboratory studies and carotid artery ultrasonography did not reveal any pathological findings. Pre-operative and post-operative contrast-enhanced CT images illustrated the anatomical region where the iatrogenic injury may have occurred and the diagnosis of iatrogenic HS was considered (Fig. 2A-C) (9). The patient succumbed after 2 days and did not experience any further episodes of Harlequin syndrome prior to their passing.

Figure 1

Right-sided facial flushing and profuse sweating with the left side of the face remaining pale and dry.

Figure 2

(A) Pre-operative contrast-enhanced CT image, axial section soft tissue window, demonstrates circumferential and nodular pleural thickening (white arrow) predominantly along the left lateral pleural space including mediastinum pleural surface along with extensive calcifications, representing osteosarcoma pleural metastasis; blue arrows reveal sympathetic chain. (B) Post-operative contrast-enhanced CT image, axial section soft tissue window, demonstrates post-operative changes in the subcutaneous fat of the left hemithorax (arrow) in 8th to 9th intercostal space. (C) Anatomic image of sympathetic track in 8th intercostal place; Courtesy of IMAIOS ©️ ‘Micheau A, Hoa D, e-Anatomy, www.imaios.com, DOI: 10.37019/e-anatomy’. CT, computed tomography.

Discussion

The 28-year-old female patient represented an example of probable iatrogenic HS. Localization of the lesion in HS must be based on both the patient's clinical history and her constellation of symptoms.

The pathology can manifest at any point along the sympathetic outflow to the face. The second neuron (preganglionic fibers) leaves the spinal cord at T2-T3 and synapse with the third neuron (post-ganglionic fibers) in the superior cervical ganglion. Post-ganglionic fibers that supply the medial forehead and nose travel with the internal carotid artery, while fibers for other facial areas and the neck travel with the external carotid artery (10).

HS is caused by the unilateral blockade of the T2-T3 fibers carrying sudomotor and vasomotor supply to the face. VATS pleurectomy can rarely cause damage to the thoracic sympathetic chain (11). However, the possible mechanism of damage in the present case might be the utilization of diathermy or mechanical disruption (12). This method has been historically employed for treating palmar and axillary hyperhidrosis by interrupting the upper thoracic sympathetic chain (13).

Regarding causes of HS, ~54.6% of reported cases have idiopathic etiology, while in 45.4% of cases, the HS-associated symptomatology occurred secondarily, concurring with other autonomic nervous system impairments or iatrogenic (14). In adults, a majority of HS cases lack a discernible medical cause, with one-third attributed to expanding neoplasms or iatrogenic damage (15).

The iatrogenic origin of HS has been documented in several cases. Fringeli et al (16) reported HS in a 55-year-old female patient with large median and right paramedian disc hernia-induced right C7 radiculopathy following C6-C7 fusion discectomy and additional anterior spondylodesis. Visible HS-suggestive symptoms on the right side of the face (flushing and excessive sweating), and contralateral facial anhidrosis, miosis and ptosis, were noted similar to the first case reported (3). Similarly, Sullivan et al (17) noted HS symptoms in a female patient post-T3 erector spinae plane block for radical mastectomy and axillary dissection, while Burlacu and Buggy described HS and Horner syndrome coexisting after left mastectomy and immediate latissimus dorsi reconstruction (18). Post-operative malignancies have also been linked to HS, such as in a case involving a 72-year-old man with squamous non-small cell lung cancer, where axillary and supraclavicular lymph node enlargement exerted compressive actions on nearby vascular structures, including carotid artery, the internal jugular vein and subclavian artery (19,20). Another possible iatrogenic cause of HS has been reported by Van Slycke et al (21) in a 74-year-old woman who underwent compressive retrosternal goiter thyroidectomy. Post-operation, patients reported recurring sudden left-sided facial flushing and sweating triggered by emotional changes, physical exertion, or heat. In some cases, HS could fully remit with no further relapses.

Treatment of HS typically depends on the severity of symptoms and the underlying cause. For most patients, HS is benign and does not require intervention. In cases where HS persists, treatment strategies are often tailored to the underlying condition or predisposing factor, especially if symptoms cause significant distress or social embarrassment. Stellate ganglion block (22), botulinum toxin injections to regulate sweating and other symptoms (23), surgery or radiation to remove a tumor or lesion (24) have been employed to manage symptoms of HS. The 28-year-old female patient of the present case report did not have oculomotor changes (ptosis or miosis), or upper extremity sudomotor or vasomotor changes. This indicates that T1 and T4 remained unaffected, thereby locating the sympathetic injury to the second or third thoracic segments of the spinal cord. Since this incident happened 8 months post left pleurectomy this indicates an iatrogenic HS. The present study reports, to the best of the authors' knowledge, the first case of a patient with HS as a result of pleural metastasectomy. HS case reports published in PubMed (https://pubmed.ncbi.nlm.nih.gov/) are summarized in Table I.

Table I

HS case reports in PubMed database.

Table I

HS case reports in PubMed database.

First author/s, year	Title	(Refs.)
Sharma et al, 2023	HS and autonomic seizures - a rare association	(25)
Navickaitė et al, 2023	Sarcoidosis-associated sensory ganglionopathy and HS: A case report	(26)
Karam et al, 2023	HS during peripheral cardiopulmonary bypass in a patient with an obstructing tracheal schwannoma: A case report	(27)
Melka et al, 2023	Idiopathic HS in a patient from Ethiopia: A case report	(28)
Giunta et al, 2023	Management of HS Under ECPELLA support: A report of two cases and a proposed approach	(29)
Hylton et al, 2022	HS as a complication of epidural anesthesia in an infant: Do adjunct medications play a role?	(30)
Dalldorf et al, 2022	HS following regional liposomal bupivacaine use in a partial sternectomy	(31)
Yan et al, 2022	HS induced by intraspinal analgesia in patients with advanced cancer: a case report	(32)
Korbi et al, 2022	Harlequin syndrome: An asymmetric face	(33)
Persson et al, 2022	HS associated with thoracic epidural anesthesia	(34)
Schultz et al, 2020	HS following microwave ablation in a child with a symptomatic paraspinal mass	(35)
Wagner et al, 2019	HS after thoracoscopic repair of a child with tracheoesophageal fistula	(12)
Tanaka et al, 2019	Cardiac sympathetic hyperactivity of lung cancer-associated HS	(36)
Elboukhar et al, 2019	Idiopathic HS: A case report and literature review	(37)
Pasrija et al, 2019	HS during venoarterial extracorporeal membrane oxygenation	(38)
Hans-Bittner et al, 2018	Do you know this syndrome? HS	(39)
Lee et al, 2017	HS and Horner syndrome after neck schwannoma excision in a pediatric patient	(40)
Lefevre et al, 2017	Development of HS following placement of thoracic epidural anesthesia in a pediatric patient undergoing Nuss procedure	(41)
Jeon et al, 2017	HS following resection of mediastinal ganglioneuroma	(42)
Algahtani et al, 2017	Idiopathic HS manifesting during exercise: A case report and review of the literature	(43)
Al Hanshi et al, 2017	A case study of HS in VA-ECMO	(44)
Kim et al, 2016	A pediatric case of idiopathic HS	(45)
Naqvi et al, 2016	HS with contralateral anhidrosis after an upper chest gunshot wound	(46)
Jung et al, 2015	Iatrogenic HS: A new case	(47)
Emsley et al, 2013	Post-exertional HS with spontaneous improvement	(48)
Breunig et al, 2012	HS in childhood - Case report	(49)
Pradeep et al, 2011	HS in a case of toxic goitre: A rare association	(50)
Moon et al, 2005	HS with crossed sympathetic deficit of the face and arm	(51)
Fallon et al, 2005	HS in two athletes	(52)
Lombardi et al, 2004	HS: An association with overlap parasomnia	(53)
Swan et al, 2003	Iatrogenic HS	(54)
Corbett et al, 1999	HS	(55)
Noda et al, 1991	HS due to superior mediastinal neurinoma	(56)
Lance et al, 1988	HS: The sudden onset of unilateral flushing and sweating	(3)

[i] HS, harlequin syndrome.

Acknowledgements

Not applicable.

Funding

Funding: No funding was received.

Availability of data and materials

The data generated in the present study may be requested from the corresponding author.

Authors' contributions

MM and AK conceptualized the present study, performed the methodology, and wrote/prepared the original draft. AP, IK, PE, EE contributed to interpretation of the data. MK, AB, NG, EZ, KM contributed to acquisition of data. AP supervised the present study. AP, IK, PE, EE, MK, AB, NG, EZ and KM wrote, reviewed and edited the manuscript. All authors read and approved the final version of the manuscript. MM and AK confirm the authenticity of all the raw data.

Ethics approval and consent to participate

Written informed consent was obtained from the legal guardian for patient participation in the present study.

Patient consent for publication

Patient characteristics have been anonymized in compliance with ethical standards. Written informed consent was obtained from the next of kin for the publication of any associated images or patient data.

Competing interests

The authors declare that they have no competing interests.

References

1	Cheshire WP Jr and Low PA: Harlequin syndrome: Still only half understood. J Neuroophthalmol. 28:169–170. 2008.PubMed/NCBI View Article : Google Scholar
2	Bremner F and Smith S: Pupillographic findings in 39 consecutive cases of harlequin syndrome. J Neuroophthalmol. 28:171–177. 2008.PubMed/NCBI View Article : Google Scholar
3	Lance JW, Drummond PD, Gandevia SC and Morris JG: Harlequin syndrome: The sudden onset of unilateral flushing and sweating. J Neurol Neurosurg Psychiatry. 51:635–642. 1988.PubMed/NCBI View Article : Google Scholar
4	Tascilar N, Tekin NS, Erdem Z, Alpay A and Emre U: Unnoticed dysautonomic syndrome of the face: Harlequin syndrome. Auton Neurosci. 137:1–9. 2007.PubMed/NCBI View Article : Google Scholar
5	Drummond PD and Finch PM: Reflex control of facial flushing during body heating in man. Brain. 112 (Pt 5):1351–1358. 1989.PubMed/NCBI View Article : Google Scholar
6	Drummond PD and Edis RH: Loss of facial sweating and flushing in Holmes-Adie syndrome. Neurology. 40:847–849. 1990.PubMed/NCBI View Article : Google Scholar
7	Yu D, Zhang S, Feng A, Xu D, Zhu Q, Mao Y, Zhao Y, Lv Y, Han C, Liu R and Tian Y: Methotrexate, doxorubicin, and cisplatinum regimen is still the preferred option for osteosarcoma chemotherapy: A meta-analysis and clinical observation. Medicine (Baltimore). 98(e15582)2019.PubMed/NCBI View Article : Google Scholar
8	Sanfilippo R, Bertulli R, Marrari A, Fumagalli E, Pilotti S, Morosi C, Messina A, Dei Tos AP, Gronchi A and Casali PG: High-dose continuous-infusion ifosfamide in advanced well-differentiated/dedifferentiated liposarcoma. Clin Sarcoma Res. 4(16)2014.PubMed/NCBI View Article : Google Scholar
9	Micheau A HD. e-Anatomy.
10	Vidal Esteban A, Natera-de Benito D, Martínez Sánchez D, Reche Sainz A, Rodríguez Díaz MR, Alfaro Iznaola CM and de Santos Moreno MT: Congenital Harlequin syndrome as an isolated phenomenon: A case report and review of the literature. Eur J Paediatr Neurol. 20:426–430. 2016.PubMed/NCBI View Article : Google Scholar
11	van Brakel TJ and Barendregt WB: Wet and dry hands after video-assisted thoracoscopic pleurectomy. Interact Cardiovasc Thorac Surg. 16:563–564. 2013.PubMed/NCBI View Article : Google Scholar
12	Wagner R, Lacher M, Merkenschlager A and Markel M: Harlequin syndrome after thoracoscopic repair of a child with tracheoesophageal fistula (TEF). European J Pediatr Surg Rep. 7:e63–e65. 2019.PubMed/NCBI View Article : Google Scholar
13	Sugimura H, Spratt EH, Compeau CG, Kattail D and Shargall Y: Thoracoscopic sympathetic clipping for hyperhidrosis: long-term results and reversibility. J Thorac Cardiovasc Surg. 137:1370–6; discussion 1376-7. 2009.PubMed/NCBI View Article : Google Scholar
14	Mavroudis I, Balmus IM, Ciobica A, Luca AC, Chowdhury R, Iordache AC, Gorgan DL and Radu I: Mini-review on the harlequin syndrome-a rare dysautonomic manifestation requiring attention. Medicina (Kaunas). 58(938)2022.PubMed/NCBI View Article : Google Scholar
15	Willaert WI, Scheltinga MR, Steenhuisen SF and Hiel JA: Harlequin syndrome: Two new cases and a management proposal. Acta Neurol Belg. 109:214–220. 2009.PubMed/NCBI
16	Fringeli Y, Humm AM, Ansorge A and Maestretti G: Harlequin sign concomitant with Horner syndrome after anterior cervical discectomy: A case of intrusion into the cervical sympathetic system. J Neurosurg Spine. 26:684–687. 2017.PubMed/NCBI View Article : Google Scholar
17	Sullivan TR, Kanda P, Gagne S and Costache I: Harlequin Syndrome associated with erector spinae plane block. Anesthesiology. 131(665)2019.PubMed/NCBI View Article : Google Scholar
18	Burlacu CL and Buggy DJ: Coexisting harlequin and Horner syndromes after high thoracic paravertebral anaesthesia. Br J Anaesth. 95:822–824. 2005.PubMed/NCBI View Article : Google Scholar
19	Biondi A, Persiani R, Zoccali M, Rausei S, Cananzi F and D'Ugo D: Harlequin syndrome. Ann Thorac Surg. 88(304)2009.PubMed/NCBI View Article : Google Scholar
20	Tubbs RS, Salter G, Wellons JC III and Oakes WJ: Blood supply of the human cervical sympathetic chain and ganglia. Eur J Morphol. 40:283–288. 2002.PubMed/NCBI View Article : Google Scholar
21	Van Slycke S, Stockman A, Dionigi G, Carette R, Gillardin JP, Brusselaers N and Vermeersch H: Harlequin syndrome after thyroidectomy for compressive retrosternal goiter. case report and review of the literature. Acta Chir Belg. 114:212–214. 2014.PubMed/NCBI View Article : Google Scholar
22	Reddy H, Fatah S, Gulve A and Carmichael AJ: Novel management of harlequin syndrome with stellate ganglion block. Br J Dermatol. 169:954–956. 2013.PubMed/NCBI View Article : Google Scholar
23	Manhães RK, Spitz M and Vasconcellos LF: Botulinum toxin for treatment of Harlequin syndrome. Parkinsonism Relat Disord. 23:112–113. 2016.PubMed/NCBI View Article : Google Scholar
24	Kirk F, Crathern K and Stroebel A: Video-assisted thoracoscopic sympathectomy for Harlequin syndrome. Eur J Cardiothorac Surg. 63(ezac577)2023.PubMed/NCBI View Article : Google Scholar
25	Sharma B, Sharma P, Ahuja I and Pemawat A: Harlequin syndrome and autonomic seizures-A rare association. Ann Indian Acad Neurol. 26:839–841. 2023.PubMed/NCBI View Article : Google Scholar
26	Navickaitė I, Ališauskienė M, Petrauskienė S and Žemgulytė G: Sarcoidosis-associated sensory ganglionopathy and harlequin syndrome: A case report. Medicina (Kaunas). 59(1495)2023.PubMed/NCBI View Article : Google Scholar
27	Karam C, Abou Nafeh N, Aouad MT, Siddik-Sayyid S, Kaddoum R, Zeeni C, Anka S, Shaya B and Khalili A: Harlequin syndrome during peripheral cardiopulmonary bypass in a patient with an obstructing tracheal schwannoma: A case report. Clin Case Rep. 11(e7509)2023.PubMed/NCBI View Article : Google Scholar
28	Melka D and Zebenigus M: Idiopathic harlequin syndrome in a patient from Ethiopia: A case report. Ethiop J Health Sci. 33:383–386. 2023.PubMed/NCBI View Article : Google Scholar
29	Giunta M, Recchia EG, Capuano P, Toscano A, Attisani M, Rinaldi M and Brazzi L: Management of harlequin syndrome under ECPELLA support: A report of two cases and a proposed approach. Ann Card Anaesth. 26:97–101. 2023.PubMed/NCBI View Article : Google Scholar
30	Hylton JRE: Harlequin syndrome as a complication of epidural anaesthesia in an infant: Do adjunct medications play a role? Indian J Anaesth. 66:669–672. 2022.PubMed/NCBI View Article : Google Scholar
31	Dalldorf DA, Hart A, Grant SA and Teeter EG: Harlequin syndrome following regional liposomal bupivacaine use in a partial sternectomy. Cureus. 14(e28005)2022.PubMed/NCBI View Article : Google Scholar
32	Yan R, Dang C and Yuan W: Harlequin syndrome induced by intraspinal analgesia in patients with advanced cancer: A case report. Transl Cancer Res. 11:2457–2461. 2022.PubMed/NCBI View Article : Google Scholar
33	Korbi M, Boumaiza S, Achour A, Belhadjali H and Zili J: Harlequin syndrome: An asymmetric face. Clin Case Rep. 10(e05833)2022.PubMed/NCBI View Article : Google Scholar
34	Persson RM, Tellnes K, Hoven H, Haaverstad R and Svendsen ØS: Harlequin syndrome associated with thoracic epidural anaesthesia. Anaesth Rep. 10(10.1002/anr3.12144)2022.PubMed/NCBI View Article : Google Scholar
35	Schultz L, Mackarey A, Oh C and Kent P: Harlequin syndrome following microwave ablation in a child with a symptomatic paraspinal mass. BMJ Case Rep. 13(e232700)2020.PubMed/NCBI View Article : Google Scholar
36	Tanaka Y and Satomi K: Cardiac sympathetic hyperactivity of lung cancer-associated harlequin syndrome. JMA J. 2:190–191. 2019.PubMed/NCBI View Article : Google Scholar
37	Elboukhari K, Baybay H, Elloudi S, Douhi Z and Mernissi FZ: Idiopathic harlequin syndrome: A case report and literature review. Pan Afr Med J. 33(141)2019.PubMed/NCBI View Article : Google Scholar
38	Pasrija C, Bedeir K, Jeudy J and Kon ZN: Harlequin syndrome during venoarterial extracorporeal membrane oxygenation. Radiol Cardiothorac Imaging. 1(e190031)2019.PubMed/NCBI View Article : Google Scholar
39	Hans-Bittner NR, Bittner GC and Hans Filho G: Do you know this syndrome? Harlequin syndrome. An Bras Dermatol. 93:585–586. 2018.PubMed/NCBI View Article : Google Scholar
40	Lee DH, Seong JY, Yoon TM, Lee JK and Lim SC: Harlequin syndrome and Horner syndrome after neck schwannoma excision in a pediatric patient: A case report. Medicine (Baltimore). 96(e8548)2017.PubMed/NCBI View Article : Google Scholar
41	Lefevre A and Schnepper G: Development of harlequin syndrome following placement of thoracic epidural anesthesia in a pediatric patient undergoing Nuss procedure. Clin Case Rep. 5:1523–1525. 2017.PubMed/NCBI View Article : Google Scholar
42	Jeon YJ, Son J and Cho JH: Harlequin syndrome following resection of mediastinal ganglioneuroma. Korean J Thorac Cardiovasc Surg. 50:130–132. 2017.PubMed/NCBI View Article : Google Scholar
43	Algahtani H, Shirah B, Algahtani R and Alkahtani A: Idiopathic harlequin syndrome manifesting during exercise: A case report and review of the literature. Case Rep Med. 2017(5342593)2017.PubMed/NCBI View Article : Google Scholar
44	Al Hanshi SAM and Al Othmani F: A case study of Harlequin syndrome in VA-ECMO. Qatar Med J. 2017(39)2017.
45	Kim JY, Lee MS, Kim SY, Kim HJ, Lee SJ, You CW, Kim JS and Kang JH: A pediatric case of idiopathic Harlequin syndrome. Korean J Pediatr. 59 (Suppl 1):S125–S128. 2016.PubMed/NCBI View Article : Google Scholar
46	Naqvi SY, Zainal A, Flood SP and Kinniry P: Harlequin syndrome with contralateral anhidrosis after an upper chest gunshot wound. BMJ Case Rep. 2016(bcr2016216931)2016.PubMed/NCBI View Article : Google Scholar
47	Jung JM, Lee MH, Won CH, Chang SE, Lee MW, Choi JH and Moon KC: Iatrogenic harlequin syndrome: A new case. Ann Dermatol. 27:101–102. 2015.PubMed/NCBI View Article : Google Scholar
48	Emsley HC: Postexertional harlequin syndrome with spontaneous improvement. BMJ Case Rep. 2013(bcr2013200516)2013.PubMed/NCBI View Article : Google Scholar
49	Breunig Jde A, Hartmann M, Freire CF and de Almeida HL Jr: Harlequin syndrome in childhood-case report. An Bras Dermatol. 87:907–909. 2012.PubMed/NCBI View Article : Google Scholar
50	Pradeep PV, Benede AK, Harshita SS and Jayashree B: Harlequin syndrome in a case of toxic goitre: A rare association. Case Rep Med. 2011(293076)2011.PubMed/NCBI View Article : Google Scholar
51	Moon SY, Shin DI, Park SH and Kim JS: Harlequin syndrome with crossed sympathetic deficit of the face and arm. J Korean Med Sci. 20:329–330. 2005.PubMed/NCBI View Article : Google Scholar
52	Fallon KE and May JJ: Harlequin syndrome in two athletes. Br J Sports Med. 39(e1)2005.PubMed/NCBI View Article : Google Scholar
53	Lombardi C, Vetrugno R, Provini F, Plazzi G, Pierangeli G, Coccagna G, Lugaresi E, Montagna P and Cortelli P: Harlequin syndrome: An association with overlap parasomnia. J Neurol Neurosurg Psychiatry. 75:341–342. 2004.PubMed/NCBI
54	Swan MC, Nicolaou M and Paes TR: Iatrogenic harlequin syndrome. Postgrad Med J. 79(278)2003.PubMed/NCBI View Article : Google Scholar
55	Corbett M and Abernethy DA: Harlequin syndrome. J Neurol Neurosurg Psychiatry. 66(544)1999.PubMed/NCBI View Article : Google Scholar
56	Noda S: Harlequin syndrome due to superior mediastinal neurinoma. J Neurol Neurosurg Psychiatry. 54(744)1991.PubMed/NCBI View Article : Google Scholar