Congenital Frey’s syndrome associated with nontraumatic bilateral trifid mandibular condyle

Abstract

Congenital Frey’s syndrome and bilateral trifid mandibular condyle are two different entities. The occurrence of both together is rare and has not been reported in the literature. This article describes the case of a 17-year-old male patient who complained of bilateral warmness, flushing and sweating in the preauricular area after eating spicy and sour foods since childhood. He had no complaint related to the functions of the temporomandibular joint and had no history of facial trauma. The patient was treated with injections of botulinum toxin A and the signs and symptoms of Frey’s syndrome improved significantly.

Warmness, flushing and sweating in the preauricular area are signs and symptoms of Frey’s syndrome. This syndrome was first described in 1757, but its occurrence after auriculotemporal nerve damage was described in 1923 by the Polish neurologist, Łucja Frey. The syndrome can be associated with direct or indirect damage of the greater auricular nerve and/or the auriculotemporal nerve.

An anatomically abnormal mandibular condyle is also unusual and can be associated with several situations, especially with previous trauma to the temporomandibular joint (TMJ). One of the abnormalities of the mandibular condyle is related to the number of condylar heads. The first report about this was published in 1941 by Hrdlicka. Only one specific case of trifid mandibular condyle associated with a history of previous facial trauma has been reported and the congenital occurrence of a trifid condyle is uncommon.

The aim of this paper is to report a case of congenital Frey’s syndrome associated with bilateral trifid mandibular condyle and to describe the treatment for this patient.

Case report

A 17-year-old man was referred to the authors by his general dentist. His medical history was noncontributory. His complaints were bilateral warmness, flushing and sweating of the preauricular area after eating spicy and sour foods since childhood. His daily routine was particularly disturbed by this, mainly when eating in public. There was no history of the use of medication by his mother during pregnancy and his birth was reported as uneventful. There was no report of facial trauma. To support the diagnosis, orthopantomography was performed and trifid condyle heads were observed on both sides ( Fig. 1 ).

Fig. 1
Orthopantomography displays the trifid condyle heads on both sides (arrows).

Considering the radiographic findings, a computed tomography (CT) of the mandible was performed. Three dimensional (3D) reconstruction of this examination revealed the trifid anatomy of both condyle heads and their relationship with the joint cavities ( Fig. 2 ).

Fig. 2
3D reconstruction showing the trifid mandible condyle: right (A) and left (B) with medial, lateral and posterior condylar heads.

Following the diagnostic imaging, Minor’s test was performed. A solution containing iodine and ethanol was applied to the preauricular area on both sides. When it was dry, corn starch powder was applied to the same areas. The patient was asked to eat a sour candy and after 30 s the preauricular areas tested positive with a bluish-black colour change because of the iodine–starch reaction, sweat secretion also increased ( Fig. 3 ). For an exact determination of the hyperaesthetic regions, flushing and sweating areas were mapped and divided into 9 fields of 2 cm 2 on each side.

Fig. 3
Positive Minor’s test. Sweating on left (A) and right (B) preauricular areas.

Based on the patient’s history and clinical, radiographic, 3D CT reconstruction and Minor’s test, the diagnosis of Frey’s syndrome associated with nontraumatic bilateral trifid mandibular condyle was suggested. The patient was treated conservatively with subcutaneous injection of botulinum toxin A (BTA) 25 U/0.1 ml in the centre of each field for treatment of the Frey’s syndrome. After 3 days, Minor’s test was performed again to confirm the effect of BTA; some remaining areas of sweating were observed. The patient had no complaints after the application of BTA and has been followed-up for 2 years.

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Jan 24, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Congenital Frey’s syndrome associated with nontraumatic bilateral trifid mandibular condyle
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