- Email: [email protected]
An early account of gustatory sweating (Frey's syndrome): a chance observation 250 years ago
Brirish Journul ojPlas?ic Surqrry (1985) 38, 122-123 Q 1985 The Trustees of British Association of Plastic Surgeons
An early account of gustatory sweating (Frey’s syndrome): a chance observation 250 years ago J.-P. A. NICOLAI Plastic and Reconstructive
Surgery Centre, The Municipal
Hospital,
Arnhem,
The Netherlands
Summary-Quite by chance, when reading an old manuscript dealing with fistulae of the parotid duct, the author came across a clear description of what is now well known as gustatory sweating (Frey’s syndrome). The incident which gave rise to this observation is recalled and the account is reproduced in the original text. In a paper entitled Observations sur les$stules du salivaire de Stenon published in 1757, a
canal
certain M. Duphenix describes the management of a complicated facial wound in which the parotid duct and the facial nerve were damaged. Tucked away in the original text is a remarkably astute observation of the phenomenon that we now universally recognise as gustatory sweating (Frey’s syndrome). Case report In November 1726, a riding master with the deer-hunting team of the Duke of Chantilly was lifted off his horse by the antlers of the hunted deer and thrown to the ground some 10 yards away where he lay unconscious. He had sustained two penetrating wounds, one affecting the left upper arm, the other the left side of his face. He was seen a few hours later by Dr Duphenix who found that the facial wound extended through the masseter muscle, beneath the malar bone where a comminuted fracture of the infra-orbital margin could be clearly felt with the finger tip. The parotid duct had also been torn across. The fractured bone segments were repositioned as accurately as possible and to keep them in place the wound was packed with pieces of fine linen supported by compresses and a bandage. The patient remained deeply unconscious and oliguric and was “bled” on no less than eight occasions. After the last attempt, he regained consciousness, passed urine in adequate amounts and “his belly opened”! Feeding now became a problem because of his inability to open his jaws, but with the help of a nursing bottle, a decoction of herbs and chicken broth could be administered to the patient who steadily regained strength. The facial wound was dressed repeatedly and after several fragments of bone were spontaneously discharged between the 39th and 41st day, the wound rapidly became smaller leaving only a tiny fistulous opening. It was at this time that Dr Duphenix noted that whenever the patient moved his jaw, great quantities of saliva escaped through the fistula: he measured the
amount on several occasions and recorded, for example, as much as 68 grams of saliva in 1.5 minutes, 88 grams of saliva in 18 minutes, 106 grams of saliva in 23 minutes and 132 grams of saliva in 28 minutes. Dr Duphenix also observed that when the patient was eating there was also a “transudation of a clear and transparent fluid through the skin covering the parotid gland.. .“. These clear drops of liquid coalesced and formed a stream that ran down the patient’s neck, making it necessary to place a cloth round the neck to collect the fluid. At the time Dr Duphenix believed the liquid was no different from the saliva that he had observed escaping from the fistulous opening itself. At the end of January 1727, the riding master was well enough to return to work but he could not blow his hunting horn as he could not close his lips properly on the left side. He had loss of sensation over the lips, curious twitching movements of some of the facial muscles and lack of control of air and saliva “. . . as in all those affected by paralysis in those parts”. In May 1727, Dr Duphenix successfully closed the external opening of the fistula by introducing a lead cannula into the opening after freeing the external scar and bringing the fistulous track into the oral cavity. The operative details are meticulously described in the original case report, but no further comments are made anywhere in the text of the gustatory sweating that had been noticed previously. The riding master’s inability to blow his horn remained. However, it seems that the Duke of Chantilly had an interest in mechanical devices and designed a gadget which he had made “on the spot”. It consisted of a horn mouth-piece fitted with a kind of bowl or plate to which a spring was fitted to keep the mouth-piece pressed against the malfunctioning lip. Duphenix christened this device an “obturateur des IPvres”and the gadget worked SO well that the riding master was able to blow the hunting horn almost as well as he could before the accident. Dr Duphenix, with incredible prescience, ended his short contribution by pointing out that the same principle might well be adapted for other people with lip injuries to provide a good lip seal-in particular the design of an efficient embouchure for those musicians who play wind instruments such as the oboe, bassoon, flute, bugle etc. 122
AN EARLY
ACCOUNT
OF GUSTATORY
SWEATING
(FREY’S
t
Discussion
OBSERVATIONS SUR LES
FISTULES
DU DE
CANAL
SALIVAIRE
STENON.
1.
S u R ant PIaye comp2igue’e ri la Joue , ou Zecanal faliuaire fat de’cbirel.
A
123
SYNDROME)
U
mois de Novembre 1726 , un Piqueur dk l’Equi age du cerf de S. A. S. M. le DUC, chap. fant i trois Pieucs de Chantiliy , fut enlevt de defTus Ton cheval par le cerf qu’on pourfiivoit , & jettt H une diftance de pri?s de fix pas, oh il refia fans aucune connoiffince : on me I’amena peu d’heures aprb ; je lui uouvai deux playes fakes par les andouillets du cerf, l’une H la partie moyenne fiptrieurc & pofl&ieure du bras gauthe , & l’auue au vifage du meme c&t&.
. . . . . . . . . . . . . . . . . . . . . . . . ie voalus @tre prtfent loifque le blefft? preadroit des alimens ; je remarquai qu’H mefure qu’il mkhoit, il fe faifoit au rravers de la peau qui couvre la parotide, nne sranfudation d’une liqueur Claire & tranfparente, qui formoit un nombre in&i de petites goutelettes ,~lefquelles, en fe Aniffant , en formoient de plus confidtrables, & celles-ci fe joignant les unes aux autres , faifoient une ou plufieurs trainees de liqueur qui couloit le long du co1 , de faGon qu’on Ptoit obligt de mettle un linge au-deffous pour la recevoir. Cette tvacuation diminua un peu la douleur con-
. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . II eR tvident que la liqueur qui fortoit i travers la pem, n’ttoit autre chore que la falive donr Ie tours nature1 avoit td intcrrompu par la comprefion.
This remarkable case report of a complicated facial injury involving a facial fracture, division of the parotid duct and facial paralysis is interesting because of the astute observation of the phenomenon of gustatory sweating that we now associate with the title of “Frey’s syndrome”. Its true significance was not appreciated at the time, but its description could not be bettered. We are not told whether the gustatory sweating persisted or ceased, as it may do in about 20% of the patients (Gorlin et al., 1976). Quite apart from the successful treatment of a persistent salivary fistula, Duphenix also presents us with a fascinating description of an early external appliance designed to help those with labial incompetence and its possible value in providing a sound embouchure for players of wind instruments.
References Duphenix, M. (1757). Observations
SLIT les fistules du canal salivaire de Stenon. Memoires de I’Academie Ro,vale de Chirurgie, 3, 431. Frey, L. (1923). Le syndrome du nerf auriculo-temporal. Revue Neurologique, 2. 97.
Gorlin, R. J., Pindborg, J. J. and Cohen, M. M. (1976). Syndromes of the Head and Neck. 2nd Edition, McGraw Hill.
New York:
The Author J.-P. A. Nicolai, MD, Netherlands.
PhD,
Plastic
Surgeon,
Arnhem,
The
Requests for reprints to: J.-P. A. Nicolai, MD, PhD. Centrum voor Plastische en Reconstructieve Chirurgie, Gemeente Ziekenhuis, Wagnerlaan 55, 6815 AD Arnhem. The Netherlands.
An early account of gustatory sweating (Frey’s syndrome): a chance observation 250 years ago J.-P. A. NICOLAI Plastic and Reconstructive
Surgery Centre, The Municipal
Hospital,
Arnhem,
The Netherlands
Summary-Quite by chance, when reading an old manuscript dealing with fistulae of the parotid duct, the author came across a clear description of what is now well known as gustatory sweating (Frey’s syndrome). The incident which gave rise to this observation is recalled and the account is reproduced in the original text. In a paper entitled Observations sur les$stules du salivaire de Stenon published in 1757, a
canal
certain M. Duphenix describes the management of a complicated facial wound in which the parotid duct and the facial nerve were damaged. Tucked away in the original text is a remarkably astute observation of the phenomenon that we now universally recognise as gustatory sweating (Frey’s syndrome). Case report In November 1726, a riding master with the deer-hunting team of the Duke of Chantilly was lifted off his horse by the antlers of the hunted deer and thrown to the ground some 10 yards away where he lay unconscious. He had sustained two penetrating wounds, one affecting the left upper arm, the other the left side of his face. He was seen a few hours later by Dr Duphenix who found that the facial wound extended through the masseter muscle, beneath the malar bone where a comminuted fracture of the infra-orbital margin could be clearly felt with the finger tip. The parotid duct had also been torn across. The fractured bone segments were repositioned as accurately as possible and to keep them in place the wound was packed with pieces of fine linen supported by compresses and a bandage. The patient remained deeply unconscious and oliguric and was “bled” on no less than eight occasions. After the last attempt, he regained consciousness, passed urine in adequate amounts and “his belly opened”! Feeding now became a problem because of his inability to open his jaws, but with the help of a nursing bottle, a decoction of herbs and chicken broth could be administered to the patient who steadily regained strength. The facial wound was dressed repeatedly and after several fragments of bone were spontaneously discharged between the 39th and 41st day, the wound rapidly became smaller leaving only a tiny fistulous opening. It was at this time that Dr Duphenix noted that whenever the patient moved his jaw, great quantities of saliva escaped through the fistula: he measured the
amount on several occasions and recorded, for example, as much as 68 grams of saliva in 1.5 minutes, 88 grams of saliva in 18 minutes, 106 grams of saliva in 23 minutes and 132 grams of saliva in 28 minutes. Dr Duphenix also observed that when the patient was eating there was also a “transudation of a clear and transparent fluid through the skin covering the parotid gland.. .“. These clear drops of liquid coalesced and formed a stream that ran down the patient’s neck, making it necessary to place a cloth round the neck to collect the fluid. At the time Dr Duphenix believed the liquid was no different from the saliva that he had observed escaping from the fistulous opening itself. At the end of January 1727, the riding master was well enough to return to work but he could not blow his hunting horn as he could not close his lips properly on the left side. He had loss of sensation over the lips, curious twitching movements of some of the facial muscles and lack of control of air and saliva “. . . as in all those affected by paralysis in those parts”. In May 1727, Dr Duphenix successfully closed the external opening of the fistula by introducing a lead cannula into the opening after freeing the external scar and bringing the fistulous track into the oral cavity. The operative details are meticulously described in the original case report, but no further comments are made anywhere in the text of the gustatory sweating that had been noticed previously. The riding master’s inability to blow his horn remained. However, it seems that the Duke of Chantilly had an interest in mechanical devices and designed a gadget which he had made “on the spot”. It consisted of a horn mouth-piece fitted with a kind of bowl or plate to which a spring was fitted to keep the mouth-piece pressed against the malfunctioning lip. Duphenix christened this device an “obturateur des IPvres”and the gadget worked SO well that the riding master was able to blow the hunting horn almost as well as he could before the accident. Dr Duphenix, with incredible prescience, ended his short contribution by pointing out that the same principle might well be adapted for other people with lip injuries to provide a good lip seal-in particular the design of an efficient embouchure for those musicians who play wind instruments such as the oboe, bassoon, flute, bugle etc. 122
AN EARLY
ACCOUNT
OF GUSTATORY
SWEATING
(FREY’S
t
Discussion
OBSERVATIONS SUR LES
FISTULES
DU DE
CANAL
SALIVAIRE
STENON.
1.
S u R ant PIaye comp2igue’e ri la Joue , ou Zecanal faliuaire fat de’cbirel.
A
123
SYNDROME)
U
mois de Novembre 1726 , un Piqueur dk l’Equi age du cerf de S. A. S. M. le DUC, chap. fant i trois Pieucs de Chantiliy , fut enlevt de defTus Ton cheval par le cerf qu’on pourfiivoit , & jettt H une diftance de pri?s de fix pas, oh il refia fans aucune connoiffince : on me I’amena peu d’heures aprb ; je lui uouvai deux playes fakes par les andouillets du cerf, l’une H la partie moyenne fiptrieurc & pofl&ieure du bras gauthe , & l’auue au vifage du meme c&t&.
. . . . . . . . . . . . . . . . . . . . . . . . ie voalus @tre prtfent loifque le blefft? preadroit des alimens ; je remarquai qu’H mefure qu’il mkhoit, il fe faifoit au rravers de la peau qui couvre la parotide, nne sranfudation d’une liqueur Claire & tranfparente, qui formoit un nombre in&i de petites goutelettes ,~lefquelles, en fe Aniffant , en formoient de plus confidtrables, & celles-ci fe joignant les unes aux autres , faifoient une ou plufieurs trainees de liqueur qui couloit le long du co1 , de faGon qu’on Ptoit obligt de mettle un linge au-deffous pour la recevoir. Cette tvacuation diminua un peu la douleur con-
. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . II eR tvident que la liqueur qui fortoit i travers la pem, n’ttoit autre chore que la falive donr Ie tours nature1 avoit td intcrrompu par la comprefion.
This remarkable case report of a complicated facial injury involving a facial fracture, division of the parotid duct and facial paralysis is interesting because of the astute observation of the phenomenon of gustatory sweating that we now associate with the title of “Frey’s syndrome”. Its true significance was not appreciated at the time, but its description could not be bettered. We are not told whether the gustatory sweating persisted or ceased, as it may do in about 20% of the patients (Gorlin et al., 1976). Quite apart from the successful treatment of a persistent salivary fistula, Duphenix also presents us with a fascinating description of an early external appliance designed to help those with labial incompetence and its possible value in providing a sound embouchure for players of wind instruments.
References Duphenix, M. (1757). Observations
SLIT les fistules du canal salivaire de Stenon. Memoires de I’Academie Ro,vale de Chirurgie, 3, 431. Frey, L. (1923). Le syndrome du nerf auriculo-temporal. Revue Neurologique, 2. 97.
Gorlin, R. J., Pindborg, J. J. and Cohen, M. M. (1976). Syndromes of the Head and Neck. 2nd Edition, McGraw Hill.
New York:
The Author J.-P. A. Nicolai, MD, Netherlands.
PhD,
Plastic
Surgeon,
Arnhem,
The
Requests for reprints to: J.-P. A. Nicolai, MD, PhD. Centrum voor Plastische en Reconstructieve Chirurgie, Gemeente Ziekenhuis, Wagnerlaan 55, 6815 AD Arnhem. The Netherlands.