Harlequin Syndrome in a Case of Toxic Goitre: A Rare Association

Abstract

Harlequin syndrome (HS) is known to be associated with conditions like brain stem infarcts and superior mediastinal neurinoma. However, it has not been reported in association with autoimmune hyperthyroidism. We report a case of exacerbation of unilateral sweating in a patient with HS following the onset of toxic goitre. Previous reports have suggested that a tortuous inferior thyroid artery can produce neurovascular compression of the sympathetic chain which was not observed in our patient. Autoimmune aetiology for HS needs to be explored. Increased sweating in hyperthyroid patients needs to be assessed properly so as to prognosticate appropriately.

Harlequin Syndrome Following Implantation of Intrathecal Pumps: A Case Series

“…). Literature has described HS followed by brainstem infarction , thoracic root compression from lung cancer , thoracic mass , thoracic surgery , enlarged thyroid gland , elongated blood vessel , injury or surgery of the neck , and pituitary surgery (Table ). However, many patients with HS have not been associated with aforementioned pathology or procedures, and etiologies are still unknown.…”

“…Even though the etiology is not well defined, the condition has been associated with one‐sided sympathetic denervation of the face. Case reports associate HS with certain thoracic tumors , thoracic surgeries , neck masses , or neck surgery , and neuraxial anesthesia . However, to the best of our knowledge, this is the first case series of HS following implantation of an intrathecal drug delivery system.…”

Harlequin Syndrome: A new observation after thyroidectomy

“…However, it is preferable to reserve the term Harlequin syndrome for patients that show the paroxysmal signs of hemifacial flushing and sweating, without other neurological symptoms. The Harlequin sign can be used to denote flushing and sweating in patients that also exhibit other associated autonomic signs or syndromes, such as Horner's syndrome, Adie's syndrome and Ross Syndrome [5].Harlequin syndrome is also known to be associated with brain stem infarcts, superior mediastinal neurinoma, internal jugular vein catherisation and carotid artery dissection [6].…”

Harlequin Syndrome post-transsphenoidal pituitary macroadenoma surgery

“…It has been associated, inter alia, with brain stem infarcts, superior mediastinal neurinoma, internal jugular vein catheterization and internal carotid artery dissection resulting from damage of the vasodilator sympathetic fibers. 2 …”