PA13 Unilateral facial flushing in a 3-year-old girl

Abstract

Abstract A 3-year-old girl presented with an 18-month history of left-sided facial erythema involving her left cheek. This occurred within seconds to minutes after eating food and lasted between 5 and 20 min before complete spontaneous resolution without any associated symptoms. The flushing was not confined to a particular food type. She was born at 37 weeks via forceps-assisted delivery, which had resulted in a small laceration to her left upper cheek. She had no significant past medical history and was not on any medications. Her twin sister did not suffer from the same phenomenon. Examination revealed no abnormalities to her facial skin. Ingestion of a chocolate bar provoked immediate subtle erythema extending from her left preauricular skin to her left mid-cheek. Overlying skin texture and temperature was unchanged, and there was no associated hyperhidrosis. Examination of the cranial nerves was unremarkable at baseline and after provocation. The erythema completely resolved after 5 min. This little girl had Frey syndrome (FS) secondary to traumatic forceps-assisted delivery at birth. Frey syndrome typically consists of gustatory hyperhidrosis and flushing in response to chewing or the smell, taste or thought of food in the auriculotemporal distribution. It is due to aberrant regeneration of parasympathetic fibres instead of sympathetic fibres along the auricolotemporal branch of the mandibular nerve following trauma or infection to the parotid gland (O’Neill JP. Lucja Frey—historical relevance and syndrome review. Surgeon 2008; 6:178–81). Instead of causing the production of saliva in response to food, the aberrant fibres cause flushing and hyperhidrosis. In adults, FS is associated with previous surgery to the parotid gland. There are case reports of it arising as a consequence of forceps-assisted delivery in infants. One study reported eight cases of FS in infants in response to a food stimulus. Six of the cases had a history of forceps-assisted delivery. None of the infants experienced any associated hyperhidrosis. It was concluded that FS arose from trauma to the parotid gland following forceps-assisted delivery at birth. All cases resolved spontaneously without treatment (Dizon MV. Localized facial flushing in infancy. Auriculotemporal nerve (Frey) syndrome. Arch Dermatol 1997; 133:1143–5). The FS is often erroneously attributed to a food allergy. A thorough history for previous trauma to the parotid region or forceps-assisted delivery is recommended in infants with unilateral facial flushing. As FS runs a benign, self-limiting course in children, our patient was reassured and discharged without treatment.