Harlequin syndrome and Horner syndrome after neck schwannoma excision in a pediatric patient

Lee, Dong Hoon; Seong, Jong Yuap; Yoon, Tae Mi; Lee, Joon Kyoo; Lim, Sang Chul

doi:10.1097/md.0000000000008548

Cited by 10 publications

(6 citation statements)

References 6 publications

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“…Among the secondary causes, it is necessary to rule out space‐occupying lesions, especially neuroblastoma in children, as well as ischemic causes . Complications of surgery and interventions are the main iatrogenic causes, in which erythema and hemifacial hyperhidrosis are temporary, disappearing in hours or days . Although less common, other dysautonomic disorders can be associated with HS.…”

Section: Discussionsupporting

confidence: 68%

“…5 Complications of surgery and interventions are the main iatrogenic causes, in which erythema and hemifacial hyperhidrosis are temporary, disappearing in hours or days. 6 Although less common, other dysautonomic disorders can be associated with HS. In addition to Horner's syndrome, other disorders include the following: Adie syndrome, which consists of the presence of tonic pupils and areflexia, and Ross syndrome, where a patchy hypohidrosis is added to the clinical picture.…”

Section: Diagnosis: Congenital Harlequin and Horner Syndromes Discussionmentioning

confidence: 99%

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Hemifacial erythema and sweating after exercise in a child

Sánchez‐Martínez

Moneva-Leniz

Gegúndez-Hernández

et al. 2020

Pediatric Dermatology

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A 9-year-old boy presented with a one-year history of several episodes of erythema and increased sweating on the right side of his face that appeared immediately after intense physical exercise. The skin findings were not present at the time of consultation. A photograph of one of the episodes was provided by the parent showing right hemifacial erythema (Figure 1). Physical examination, however, revealed contralateral anisocoria, miosis, mild hypochromia of the iris, ptosis measuring approximately 2 mm, as well as slight enophthalmos of the left eye ( Figure 2). These ocular features were permanent and had been present since birth. What is the diagnosis?

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Section: Discussionsupporting

confidence: 68%

Section: Diagnosis: Congenital Harlequin and Horner Syndromes Discussionmentioning

confidence: 99%

Hemifacial erythema and sweating after exercise in a child

Sánchez‐Martínez

Moneva-Leniz

Gegúndez-Hernández

et al. 2020

Pediatric Dermatology

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“…In reported cases of iatrogenic damage, the sympathetic structures were injured in the apex of the lung or even further cranial. 2 4 15…”

Section: Discussionmentioning

confidence: 99%

Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF)

Wagner

Lacher

Merkenschlager

et al. 2019

European J Pediatr Surg Rep

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Harlequin syndrome (HS) is a rare dysautonomia of the sympathetic nervous system leading to asymmetric facial flushing and sweating. In the literature, only a few cases of HS after thoracoscopic tracheoesophageal fistula (TEF) repair are reported. We report on a newborn with TEF who developed HS after thoracoscopic repair. On the first day of life, the girl (3,480 g, gestation age: 41 week) underwent thoracoscopic repair of a type C esophageal atresia (TEF; OR time 105 minute) without complications. The postoperative course was uneventful, the patient swallowed and thrived well and did not require esophageal dilatations. At 2 years of age, missing facial flushing, transpiration, and warming on the right side of her face during agitation were noticed. As no further intervention was required, the girl and her parents adapted well to the symptoms. Our report shows that the late onset of HS after the surgical procedure is unlikely a direct causal relation to the thoracoscopic operation but rather a shared embryological pathogenesis, like a neurocristopathy.

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“…In this context, it was shown that the stellate—superior cervical ganglion—sympathetic chain neurovascular compression was caused by the elongated inferior thyroid artery and led to HS symptoms development in a 55-year-old female patient [ 19 ]. HS was also reported as a possible consequence to neck Schwannoma excision in a pediatric patient [ 25 ]. A recent case report of a healthy 52-year-old man with sudden onset HS also suggested Schwannomas in the cervical chain ganglion as a possible secondary cause of HS [ 26 ].…”

Section: Etiopathologymentioning

confidence: 99%

Mini-Review on the Harlequin Syndrome—A Rare Dysautonomic Manifestation Requiring Attention

et al. 2022

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Harlequin syndrome (HS) is a rare autonomic disorder. The causes and risk factors of the disease are not fully understood. Some cases of HS are associated with traumatic injuries, tumors, or vascular impairments of the head. Symptoms of HS can also occur in some autoimmune disorders, ophthalmic disorders, sleep disorders, and with certain organic lesions. In this context, a thorough review of the pathophysiology of HS in relation to neurological, ophthalmological, and dermatological conditions is necessary. In this mini-review, we aim to review the pathophysiological changes and underlying mechanisms in primary and secondary HS. Additionally, we discuss possible management approaches for patients with HS in light of the discussed pathological mechanisms. The main symptoms of HS that are correlated with autonomic nervous system impairments include sudden unilateral flushing of the face, neck, chest, and rarely arm, with concurrent contralateral anhidrosis. Despite reported co-occurring syndromes (such as cluster headaches), several studies have shown that HS could frequently overlap with other syndromes that are disruptive to the idiopathic nerve pathways. HS usually does not require any medical treatment. In some severe cases, symptomatic treatments could be needed. However, total symptomatic relief may not be achieved in many cases of HS. We therefore suggest an approach to comprehensive management of HS, which may lead to better long-term control of HS.

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Harlequin syndrome and Horner syndrome after neck schwannoma excision in a pediatric patient

Cited by 10 publications

References 6 publications

Hemifacial erythema and sweating after exercise in a child

Hemifacial erythema and sweating after exercise in a child

Harlequin Syndrome after Thoracoscopic Repair of a Child with Tracheoesophageal Fistula (TEF)

Mini-Review on the Harlequin Syndrome—A Rare Dysautonomic Manifestation Requiring Attention

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