Oxybutynin and propranolol combo successfully resolves symptoms in Harlequin syndrome: Case report
Oxybutynin and propranolol combination successful in Harlequin syndrome- 1st case report- Harlequin syndrome (HS) is a rare disorder characterised by unilateral facial flushing and sweating induced by exercise, heat and emotion. It is mostly idiopathic. In some cases, it can lead to social embarrassment. It is difficult to treat and can have a significant impact on quality...
Oxybutynin and propranolol combination successful in Harlequin syndrome- 1st case report- Harlequin syndrome (HS) is a rare disorder characterised by unilateral facial flushing and sweating induced by exercise, heat and emotion. It is mostly idiopathic. In some cases, it can lead to social embarrassment. It is difficult to treat and can have a significant impact on quality of life.
Recently a case describing successful treatment of idiopathic HS with a combined therapy of oxybutynin and propranolol was published in Australasian Journal of Dermatology.
A 47-year-old woman presented with hyperhidrosis and erythema episodes limited to the right side of the face, triggered by physical exercise. She was receiving prophylactic metoprolol with suboptimal control of her migraine episodes. Magnetic resonance imaging of brain and cervical spine and chest radiograph were normal. Treatment with oxybutynin (5 mg/daily orally) resulted in optimal control of hyperhidrosis but facial flushing persisted so she was switched over from metoprolol to propranolol (10 mg/8 h orally). The patient experienced a marked improvement in frequency, intensity and duration of the facial flushing, and better control of her migraine episodes.
Treatment of HS has been unsatisfactory with medical line of treatment and contralateral sympathectomy is reserved for severe cases. Oxybutynin is the most commonly used anticholinergic drug in hyperhidrosis. The frequent adverse effects of this drug are xerostomia, mydriasis, cycloplegia, urinary retention, tachycardia and palpitations which should be kept in mind before prescribing this drug.
Scientific evidence regarding the control of flushing symptoms in HS is scarce. Activation of beta-2 receptors located in the vessels may play a role in the flushing phenomena observed in HS patients. These receptors are antagonised by nonselective beta blocker, inducing vasoconstriction and minimising the resulting erythema thus explaining improvement with switching over from metoprolol to propranolol. Propranolol should not be prescribed to patients with heart failure or asthma. No interactions between oxybutynin and propranolol have been documented.
To conclude this is the first report of a patient with idiopathic HS successfully treated with oxybutynin and propranolol in whom both hyperhidrosis and facial erythema resolved. Thus the combination of oxybutynin and propranolol should be considered in patients with HS not having any contraindications to these medications.
Source- Naharro-Fernández C, de Quintana-Sancho A, López-Sundh AE, Reguero-Del Cura L, Gónzalez-López MA. Successful treatment of idiopathic Harlequin Syndrome with oxybutynin and propranolol. Australas J Dermatol. 2021 Nov;62(4):504-505. doi: 10.1111/ajd.13665. Epub 2021 Jul 27. PMID: 34314021.a
