Gustatory sweating syndrome of the submandibular gland.

Abstract

Gustatory sweating syndrome involving the submandibular gland is rare. We present a case of a patient who experienced this syndrome 5 years after undergoing submandibular gland resection. Our patient was satisfied simply with an explanation of the disorder and reassurance. But in cases where further intervention is sought, medical and surgical options are available and should be individualized for the patient.

Introduction

Gustatory sweating syndrome (auriculotemporal syndrome) is a common sequela of parotid gland surgery. Gustatory sweating syndrome associated with the parotid gland was first reported in 1757 by Duphenix. [1] In 1923, Frey reported this syndrome as a complication of infection of the parotid gland. [1] As a result, gustatory sweating related to parotid surgery or injury is known as Frey's syndrome.

Reported cases of gustatory sweating syndrome involving the submandibular gland are extremely rare. We present a case of a patient who had undergone a left submandibular gland excision. At the initial and 3- and 6-month postoperative visits, no complications were noted. The patient presented again 5 years later with gustatory sweating involving the excision site.

Case report

A white, 17-year-old girl came to our institution with a complaint of hyperhidrosis that occurred during mastication. Five years earlier, she had been treated for a persistent left neck mass, which was presumed to be a reactive lymph node. At that time she received multiple antibiotics, but the condition did not resolve. She eventually underwent an excisional biopsy of the left submandibular gland. The procedure had been performed in the standard fashion. The facial artery and vein had been identified and ligated. The lingual nerve and submandibular ganglion had been identified, and a clamp had been placed just proximal to the submandibular gland, and this portion had been ligated. Overall, the surgical procedure was uneventful. Her biopsy specimen showed no histopathologic abnormalities, and her initial postoperative followup visits were unremarkable.

Discussion

We have briefly presented a rare case of gustatory sweating syndrome affecting the submandibular gland. Gustatory sweating syndrome has been associated with surgical excision of the submandibular gland during radical neck dissection, [2] but there have been only two previously reported cases in the otolaryngologic literature of this condition as a complication following a simple submandibular gland excision. The condition might be unrecognized by many patients. The onset of gustatory sweating symptoms associated with parotid gland surgery has been reported to range from 1 month to 5 years after the operation. [3]

Gustatory sweating syndrome occurs as a reaction to any stimulus of salivation, including the aroma, sight, taste, and mastication of food. The innervation of the parotid involves the preganglionic parasympathetic portion of the gland, beginning in the inferior salivatory nucleus and passing via the glossopharyngeal nerve, the tympanic plexus, and the lesser petrosal nerve to the oticganglion. The postganglionic fibers travel along the auriculotemporal nerve and into the parotid gland, where they control glandular secretions. Preganglionic sympathetic fibers originate in the first and second thoracic segments and synapse in the cervical sympathetic ganglia. Postganglionic sympathetic fibers travel along the external carotid artery to the superficial temporal artery and the transverse facial artery and terminate in the sweat glands. [4]

Frey's syndrome occurs when misdirected regenerating postganglionic parasympathetic nerve fibers invade the severed postganglionic sympathetic fibers that supply the sweat glands. The innervation of the submandibular gland is similar to that of the parotid gland; the difference involves preganglionic parasympathetic fibers that originate in the superior salivatory nucleus and travel along the facial nerve and the chorda tympani to the submandibular ganglion. Postganglionic parasympathetic fibers originate in the submandibular ganglion and travel to the submandibular gland. Preganglionic sympathetic fibers originate in the first and second thoracic spinal nerves, synapse in the superior cervical ganglion with postganglionic sympathetic fibers, and travel along the external carotid artery and the facial artery to the sweat glands. [2,4] During surgical excision of the submandibular gland, aberrant postganglionic parasympathetic fibers can subsequently innervate severed postganglionic sympathetic fibers, supply ing the sweat glands in a misdirected pattern. This results in a hyperhidrosis of the affected area, with or without concomitant erythema caused by parasympathetic vascular effects at the site.

Diagnosis. The diagnostic test of choice is the simple Minor's test. [2,5] A tincture of iodine is applied to the skin of the affected area and allowed to dry. Then a light layer of powdered cornstarch is applied to the area. As the patient masticates and sweating occurs, the cornstarch will turn black as it reacts to both the sweat and the iodine.

Therapeutic procedures. Three modalities for treating hyperhidrosis of the submandibular area have been proposed. [5] One is to prescribe drug therapy with systemic and topical anticholinergic agents to diminish sweat gland function. A second method is to elevate the skin flap at the surgical site and make a temporalis fascia graft. The third method involves lysis of the chorda tympani. Finally, it is always beneficial to provide reassurance and a clear explanation of the causes to the patient.

From the Department of Otolaryngology, Metropolitan Hospital, Grand Rapids, Mich. (Dr. Persaud), and the Department of Otolaryngology, Children's Hospital Medical Center, Cincinnati (Dr. Myer and Dr. Rutter).

Reprint requests: Neil A. Persaud, DO, Department of Otolaryngology, Metropolitan Hospital, 1919 Boston S.E., Grand Rapids, MI 49506.

References

(1.) Spiro RH, Martin H. Gustatory sweating following parotid surgery and radical neck dissection. Ann Surg 1967;165:118-27.

(2.) Myers EN, Conley J. Gustatory sweating after radical neck dissection. Arch Otolaryngol 1970;91:534-42.

(3.) Conley JJ. Gustatory Sweating Syndrome: Salivary Glands and the Facial Nerve. New York: Grune and Stratton, 1975:51-2.

(4.) Clemente CD. Gray's Anatomy. 13th ed. Philadelphia: Lea and Febiger, 1985:665-85, 1171-6.

(5.) Seifert B, Miehlke A, Haubrich J, Chilla R. Diseases of the Salivary Glands: Pathology, Diagnosis, Treatment, Facial Nerve Surgery. New York: Georg Thieme Verlag 1986:325-6.