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LETTER TO EDITOR
Year : 2021  |  Volume : 18  |  Issue : 2  |  Page : 148

Harlequin syndrome secondary to thoracic spinal tuberculosis


Department of Neurosciences, Apollomedics Hospital, Lucknow, Uttar Pradesh, India

Date of Submission18-Jan-2021
Date of Acceptance06-Feb-2021
Date of Web Publication11-Mar-2021

Correspondence Address:
Raghav Gopal Poduval
Department of Neurosciences, Apollomedics Hospital, Lucknow - 226 012, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/am.am_6_21

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How to cite this article:
Poduval RG. Harlequin syndrome secondary to thoracic spinal tuberculosis. Apollo Med 2021;18:148

How to cite this URL:
Poduval RG. Harlequin syndrome secondary to thoracic spinal tuberculosis. Apollo Med [serial online] 2021 [cited 2021 Jun 21];18:148. Available from: https://www.apollomedicine.org/text.asp?2021/18/2/148/311112

Sir,

Harlequin syndrome is a disorder of the sympathetic supply to the face and was first described by Lance and Drummond in 1988.[1] It manifests as anhidrosis on the affected side with compensatory flushing and sweating on the contralateral side. A recently seen patient of Harlequin syndrome is reported below.

A 47-year-old female diabetic patient presented with intractable mid-thoracic pain of 2 months' duration. The pain was associated with the absence of sweating on the left half of face and excessive sweating and flushing on the right half. There was no history of other neurological or constitutional symptoms. On examination, the patient was afebrile and had no focal neurological deficit. Moderate severe spinal tenderness was present over the fifth and sixth thoracic spine. Anhidrosis was seen on the left half of the face on testing with starch iodine. There was no evidence of Horner's syndrome clinically. A magnetic resonance imaging of the dorsal spine, plain and contrast, revealed a destructive lesion of T5 and T6 thoracic vertebrae and a paravertebral abscess extending up to the body of the third thoracic vertebra [Figure 1]. Mycobacterium tuberculosis was isolated from the pus of the paravertebral abscess. The sudomotor response on the left arm showed increased latency. The patient symptoms regressed within 1 month of starting antitubercular treatment.
Figure 1: Magnetic resonance imaging contrast thoracic spine showing destructive lesion of T5 and T6 vertebrae with paravertebral abscess extending up to T3

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The vasomotor and sudomotor sympathetic supply to the face is via the ipsilateral third thoracic root and the superior cervical ganglion.[2] Lesions in the sympathetic outflow anywhere in this course can result in Harlequin syndrome. Higher lesions produce Horner's syndrome. Harlequin syndrome is rare and most cases are idiopathic. Secondary causes of Harlequin syndrome are usually following surgery of the neck and cervical spine.[3] This particular case was unique that it was of a lesion extending up from the mid-thoracic spine and of infectious etiology.

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  References Top

1.
Lance JW, Drummond P, Gandevia SC, Morris JG. Harlequin syndrome: The sudden onset of unilateral flushing and sweating. JNNP 1988;51:635-42.  Back to cited text no. 1
    
2.
The Autonomic Nervous System. In: Kandel ER, Schwartz JH, Jessel TM, editors. Principles of Neuroscience. 4th ed. New York: McGraw Hill; 2000. p. 962-63.  Back to cited text no. 2
    
3.
Fringeli Y, Humm AM, Ansorge A, Maestretti G. Harlequin sign concomitant with Horner syndrome after anterior cervical discectomy: A case of intrusion into the cervical sympathetic system. J Neurosurg Spine 2017;26:684-7.  Back to cited text no. 3
    


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