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Harlequin syndrome in a case of toxic goitre: a rare association.

Pradeep PV, Benede AK, Harshita SS, Jayashree B - Case Rep Med (2011)

Bottom Line: Harlequin syndrome (HS) is known to be associated with conditions like brain stem infarcts and superior mediastinal neurinoma.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.Increased sweating in hyperthyroid patients needs to be assessed properly so as to prognosticate appropriately.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrine Surgery, Narayana Medical College & Superspeciality Hospital, Chinthareddypalem, Nellore, Andhra Pradesh 524002, India.

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.

No MeSH data available.


Related in: MedlinePlus

It depicts the long dilated inferior thyroid artery of the left side.
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fig4: It depicts the long dilated inferior thyroid artery of the left side.

Mentions: It has been suggested that since the inferior thyroid artery crosses the sympathetic chain in the neck between the stellate and superior cervical ganglion, a tortuous inferior thyroid artery can produce neurovascular compression of the sympathetic chain [5]. Wasner et al. [5] had suggested tortuous inferior thyroid artery as a possible mechanism for Harlequin syndrome in one of their euthyroid patients. In patients with hyperthyroidism, the inferior and superior thyroid arteries enlarge in calibre and become tortuous. Since our patient developed the symptoms of Harlequin six months after developing hyperthyroidism, we explored the possibility of this theory as the cause of Harlequin. Even though during surgery, it was noticed that the inferior thyroid artery of the left side was larger in diameter and had a tortuous course (Figure 4), the Harlequin syndrome has persisted after the total thyroidectomy (1-year postoperative followup). During the thyroidectomy, the main trunk of the inferior thyroid artery was ligated and divided. After thyroidectomy, in cases of hyperthyroidism, the remaining part of the inferior thyroid artery will decrease in calibre and size and therefore may not compress the sympathetic trunk any more. Hence, we feel that dilated tortuous inferior thyroid artery as a possible mechanism may not hold true. However, it is also possible that the HS has persisted even after surgery in our case because of the severe irreversible axonal injury secondary to long-standing compression by the inferior thyroid artery (5 years).


Harlequin syndrome in a case of toxic goitre: a rare association.

Pradeep PV, Benede AK, Harshita SS, Jayashree B - Case Rep Med (2011)

It depicts the long dilated inferior thyroid artery of the left side.
© Copyright Policy - open-access
Related In: Results  -  Collection

Show All Figures
getmorefigures.php?uid=PMC3139896&req=5

fig4: It depicts the long dilated inferior thyroid artery of the left side.
Mentions: It has been suggested that since the inferior thyroid artery crosses the sympathetic chain in the neck between the stellate and superior cervical ganglion, a tortuous inferior thyroid artery can produce neurovascular compression of the sympathetic chain [5]. Wasner et al. [5] had suggested tortuous inferior thyroid artery as a possible mechanism for Harlequin syndrome in one of their euthyroid patients. In patients with hyperthyroidism, the inferior and superior thyroid arteries enlarge in calibre and become tortuous. Since our patient developed the symptoms of Harlequin six months after developing hyperthyroidism, we explored the possibility of this theory as the cause of Harlequin. Even though during surgery, it was noticed that the inferior thyroid artery of the left side was larger in diameter and had a tortuous course (Figure 4), the Harlequin syndrome has persisted after the total thyroidectomy (1-year postoperative followup). During the thyroidectomy, the main trunk of the inferior thyroid artery was ligated and divided. After thyroidectomy, in cases of hyperthyroidism, the remaining part of the inferior thyroid artery will decrease in calibre and size and therefore may not compress the sympathetic trunk any more. Hence, we feel that dilated tortuous inferior thyroid artery as a possible mechanism may not hold true. However, it is also possible that the HS has persisted even after surgery in our case because of the severe irreversible axonal injury secondary to long-standing compression by the inferior thyroid artery (5 years).

Bottom Line: Harlequin syndrome (HS) is known to be associated with conditions like brain stem infarcts and superior mediastinal neurinoma.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.Increased sweating in hyperthyroid patients needs to be assessed properly so as to prognosticate appropriately.

View Article: PubMed Central - PubMed

Affiliation: Department of Endocrine Surgery, Narayana Medical College & Superspeciality Hospital, Chinthareddypalem, Nellore, Andhra Pradesh 524002, India.

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.

No MeSH data available.


Related in: MedlinePlus