Literature DB >> 33515404

Harlequin syndrome in a pediatric population: a case series.

N Beullens1,2, G Tourlamain3,4, L Vallaeys5, F Roelens4, M D'Hooghe6.   

Abstract

Harlequin syndrome is a rare condition, presenting with unilateral facial flushing and hyperhidrosis in response to physical exercise, heat or emotional stressors and has scarcely been reported in pediatric patients. It is caused by a dysfunction of vasomotor and sudomotor sympathetic fiber activity inhibiting the ability to flush on the affected side, causing the neurologically intact side to appear red. We present three pediatric cases of this uncommon syndrome, each of them of different origin and displaying distinct associated (neurological) symptoms, and review medical literature. Insight into the anatomical structure of the thoracocervical and facial sympathetic nervous system is pivotal as it dictates symptomatology. About half of Harlequin syndrome cases are complicated with ocular symptoms and a minority may be part of more extensive partial dysautonomias affecting facial sudomotor, vasomotor and pupillary responses, such as Holmes-Adie syndrome and Ross syndrome. Etiology is generally idiopathic, however, cases secondary to surgery, trauma or infection have been described. Considering its predominantly self-limiting nature, treatment is usually unnecessary and should be restricted to incapacitating cases.

Entities:  

Keywords:  Autonomic nervous system; Autonomic neuropathy; Facial flushing; Horner syndrome

Year:  2021        PMID: 33515404     DOI: 10.1007/s13760-021-01593-6

Source DB:  PubMed          Journal:  Acta Neurol Belg        ISSN: 0300-9009            Impact factor:   2.396


  3 in total

Review 1.  Harlequin syndrome: two new cases and a management proposal.

Authors:  W I M Willaert; M R M Scheltinga; S F Steenhuisen; J A P Hiel
Journal:  Acta Neurol Belg       Date:  2009-09       Impact factor: 2.396

2.  Unilateral facial and upper truncal anhidrosis and absence of physiological flushing: A case of idiopathic harlequin syndrome.

Authors:  Crystal Zhen Yu Phuan; Hong Liang Tey
Journal:  Indian J Dermatol Venereol Leprol       Date:  2017 Nov-Dec       Impact factor: 2.545

3.  Harlequin sign (hemifacial flushing and contralateral hypohidrosis) in a 4-year-old girl with Horner syndrome.

Authors:  Masatoshi Abe; Atsushi Tamura; Yoko Sogabe; Chie Hashimoto; Tomoko Shyuto; Osamu Ishikawa
Journal:  Pediatr Dermatol       Date:  2006 Jul-Aug       Impact factor: 1.588

  3 in total
  1 in total

1.  Harlequin syndrome: An asymmetric face.

Authors:  Mouna Korbi; Sirine Boumaiza; Asma Achour; Hichem Belhadjali; Jameleddine Zili
Journal:  Clin Case Rep       Date:  2022-05-12
  1 in total

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