Gustatory rhinorrhea after maxillectomy

Int. ,L Oral Maxillo/ac. Surg. 1997; 26:124-126 Printed in Denmark. All rights reserved

Copyright 9 Munksgaard 1997 [nteroationalJoumalof

Oral& MaxillofacialSurgery [SSN 0901-5027

Gustatory rhinorrhea after maxillectomy

Hassan Mir Mohammad Sadeghi, Sergio Siciliano, Herv4 Reychler Oral and MaxillofacialSurgery Unit, St Luc University Hospital, Catholic University of Louvain (UCL), School of Medicine, Brussels, Belgium

Two case reports and considerations on etiology and pathophysiology H. M. M. Sadeghi, S. Siciliano. H. Reychler. Gustatory rhinorrhea @ e r rnaxillectomy. Two case reports' and considerations on etiology and pathophysiology. Int. J. Oral Maxillofac. Surg. 1997; 26. 126126. 9 M u n k s g a a r d , 1997 Abstract. G u s t a t o r y r h i n o r r h e a consists of free discharge of thin m u c u s f r o m the nose d u r i n g ingestion or after other g u s t a t o r y stimulus. A t maxillectomy, the nerve fibers going b o t h to the salivary glands of the palate a n d to the secretory glands o f the nasal m u c o s a m a y be damaged. A misdirection between the regenerating fibers of these two groups produces g u s t a t o r y r h i n o r r h e a . R#sumd. L a rhinorrh6e gustative (RG) consiste en u n 6coulement de m u c u s clair du nez lors d ' u n stimulus salivaire. L o t s d ' u n e maxillectomie, les fibres nerveuses allant aux glandes salivaires palatines et responsables de leur sdcr6tion, ainsi que les fibres allant aux glandes m u q u e u s e s nasales risquent une 16sion. U n e r6g6n&escence a n a r c h i q u e de ces deux groupes de fibres nerveuses p o u r r a avoir c o m m e cons6quence une r h i n o r r h 6 e gustative si les fibres d u premier g r o u p e vont innerver p a r erreur les glandes nasales.

G u s t a t o r y r h i n o r r h e a ( G R ) consists of secretion of thin m u c u s from the nose d u r i n g ingestion or after o t h e r gustatory stimulation, w i t h o u t any a p p a r e n t cause such as i n f l a m m a t i o n , allergy, or salivary fistulas 2. As a side-effect of surgery, G R has n o t been well described in the literature 1,4. This is p r o b a b l y due to lack of awareness by b o t h the p a t i e n t a n d the clinician. Patients tend not to link G R to t h e i r p r e v i o u s surgery, while surgeons do not specifically ask a b o u t s y m p t o m s o f G R after surgery. This p a p e r draws a t t e n t i o n to this p h e n o m e n o n a n d discusses its origin a n d pathophysiology.

Material and methods Anatomy Some detailed anatomic study is necessary to understand the mechanism of the GR 3,6,8.

The maxillary nerve (V2) is the second branch of the trigeminal nerve and is strictly a sensory nerve. Immediately distal to the trigeminal ganglion, where it originates, it exits the skull base via the foramen rotundum, and passes through the pterygopalatine fossa, in a lateral to anterior direction (Fig. 1). It then enters the orbit via the inferior orbital fissure and passes through the infraorbital foramen as the infraorbital nerve, after which it branches out into the lip and the lateral part of the nose (Fig. 2). There are six collateral branches of the maxillary nerve: 1) the meningeal nerve 2) the zygomatic nerve with some orbital fibers 3) the sphenopalatine nerve 4) the posterior superior alveolar nerve 5) the middle superior alveolar nerve 6) the anterior superior alveolar nerve. The sphenopalatine nerve originates from the first fold of the maxillary nerve in the pterygomaxillary fossa. It then curves medially, and passes in front of the sphenopalat-

Key words: rhinorrhea; nerve regeneration; maxillectomy. Accepted for publication 3 October 1996

ine ganglion, giving rise to four groups of collateral nervous fascicles (Fig. 3): t) some orbital branches 2) the pterygopalatine nerve 3) the nasal nerves 4) the palatine nerves. The orbital branches bear no relevance to the subject of this study. The pterygopalatine nerve enters the canal of the same name and furnishes sensory innervation to the mucous membrane lining the rhinopharynx. The nasal nerves travel through the sphenopalatine foramen to arrive in the nasal cavity. There, they divide into superior nasal nerves and the nasopalatine nerve. The superior nasal nerves innervate the mucous membrane lining the superior and the middle meatus. The nasopalatine nerve crosses the nasal septum obliquely to the lower anterior side, and it innervates the septal mucosa before entering the anterior palatine canal to innervate the mucous membrane lining the anterior palate (Fig. 3). The palatine nerves pass first through the

Rhinorrhea after maxillectomy

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Fig. 1. Course of right maxillary nerve V2 (superior view) (reproduced with permission of GI~GO~m~ & OBeRHN3). A. sous-orbit.: infraorbital artery; N. orbit, lacr: =orbitolacrimal nerve; N. temp. mal.: zygomatic nerve; Fil, orb.: orbital fibers; N. has. s.: superior nasal nerve; N. nas. pal.: nasopalatine nerve; N. pal. p.: posterior palatine nerve; N. pt6r. p.: pterygopalatine nerve; Gang. sph. p.: sphenopalatine ganglion; R. m6ning6: meningeal nerve.

Fig. 2. Course of right maxillary nerve V2 (lateral view) (reproduced with permission of Gg,~GO~r,~ & O~RL~Ya). G1. lacrym.: lacrimal gland; L.: lacrimal nerve; N. temp. m.: zygomatic nerve; N. orbit.: orbital nerve; R. mdning6: meningeaI nerve; N. dent. p.: posterior superior alveolar nerve; N. dent. m.: middle superior alveolar nerve; N. dent. a.: anterior superior alveolar nerve.

also the nasal glands and the minor salivary glands of the palate through the sympathetic and parasympathetic autonomic nerves, which accompany these nerves. Parasympathetic stimulation increases and accelerates the secretion, while sympathetic stimulation deactivates and slows down secretion. At its origin, the maxillary nerve (V2) is strictly a sensory nerve. The nervous fibers responsible for glandular secretion rejoin the sphenopalatine nerve via the sphenopalatine (or pterygopalatine) ganglion. This parasympathetic ganglion is located on the medial side of the pterygomaxillary fossa. The vidian (or pterygopalatine) nerve is the main afferent nerve, and is formed by three groups of branches (Fig. 4): 1) the greater superficial petrosal nerve including parasympathetic fibers from the facial nerve (VII) 2) the greater deep petrosal nerve including sympathetic fibers from the glossopharyngeal nerve (IX) 3) some sympathetic fibers coming from the carotid plexus. Thus formed, the pterygopalatine nerve courses through the membrane closing the anterior foramen lacerum, and passes the pterygopalatine canal before entering the sphenopalatine ganglion. In this ganglion, the synapses between pre- and postganglionic parasympathetic fibers are to be found. These parasympathetic axons and the nonsynaptic sympathetic fibers join the sphenopalatine nerve and the branches of its division. Thus, the sphenopalatine nerve is the only branch of V2 which contains sensitive, sympathetic, and parasympathetic fibers as well (Fig. 5).

ine ganglion (reproduced with permission of GR~GOIr,Z & OBERLIN3).Max. s.: maxillary bone; N. max. s.: maxillary nerve (V2); Palatin: palatine bone; R. orbit.: orbital branch; N. sph. pal.: sphenopalatine nerve; N. aft.: afferent nerve; N. palat, p.: posterior palatine nerve; N. eft.: efferent nerve; Ganglion sph. pal.: sphenopalatine ganglion; N. has.: nasal nerve; N. ptdr. pal.: pterygopalatine nerve; Apo. sph6n, du pal.: sphenoidal process of palatine bone; Rac. ptdryg.: origin of pterygoide muscle; N. vidien: vidian nerve; N. de Jacobson: Jacobson's nerve; C. I.: internal carotid artery; Rac. sympa.: sympathetic fibers; N. gd. p6tr. pr.: greater deep petrosal nerve; N. gd. p6tr. sup.: greater superficial petrosal nerve.

A wide resection of the maxilla, including the entire lateral nasal mucosa, the inferior turbinate, and the right pterygoid process, was performed. Although the excision was large, it was not radical, and external postsurgical radiotherapy was indicated. The patient received a total dose of 60 Gy from a combination of cobalt-60 and 8 MeV photons. This therapy was delivered in 30 treatments over 48 days. According to the patient, the gustatory rhinorrhea started about 4 months after the operation. It was always unilateral (right side) and was present during every meal. Some foods did not stimulate this phenomenon at all, while others had a greater effect on secretion. Rhinorrhea was present even when the patient only saw or smelt some favorite food. Case 2

posterior palatine canal, and then through the major and minor palatine canals. They innervate the nasal mucosa lining the inferior turbinate and also the mucosa lining the entire posterior hard and soft palates. The palatine nerves and the nasal nerves innervate not only the palatine and nasal mucosa, but

Case reports Case 1 An 81-year-old patient underwent right complete hemimaxillectomy for adenoid-cystic carcinoma. He had an ulcerative lesion on the palatal mucosa, next to the premolar region, which was classified as T4NOM0.

A 50-year-old female patient underwent partial right hemimaxillectomy for a squamous cell carcinoma on the hard palate. The lesion was classified as TINOM0. The excision, including ostectomy of right premolar and molar region with part of the maxillary wall: was radical and no further treatment was necessary.

126

Sadeghi et al. Neff lacrymal (V1) Noyau du nerf trijumeau

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Fig. 5. Nerve fibers which stimulate secretion of nasal secretory glands, salivary glands of palate, and lacrimal glands (reproduced with permission of LEBLANC6).Neff lacrymal: lacrimal nerve; Glande iacrymale: lacrimal gland; Arcade orbito-lacrymal: orbitolacrimal circle; Nerf grand p6treux: greater petrosal nerve; Rameau orbitaire: orbital branch; Ganglion pt6rygo-palatin: sphenopalatine ganglion; Nerf maxillaire: maxillary nerve; Glandes palatines: salivary glands of palate; Noyau du neff trijumeau: trigeminal nerve origin; Noyau parasympathique du nerf facial: parasympathetic origin of facial nerve; Ganglion trig6minal: trigeminal (gasserian) ganglion; Ganglion g6nicul6: geniculate ganglion; Racine sympathique: sympathetic fibers; Nerf grand p6treux profond: greater deep petrosal nerve; Nerfs palatins: palatine nerves; Palais mou: soft palate.

It was not possible to determine when the GR began. According to the patient, it started approximately 2 months after the surgery. She had never suffered from any nasal problems before. The GR was always unilateral (right side), and was present at every meal. The patient was obliged to blow her nose three or four times during a meal. In contrast to the first case, the patient did not experience GR when she looked at or smelt some particular food, but rhinorrhea was more abundant when she had a bad cold.

Discussion G R has been been r e p o r t e d as a complication after s e p t o r h i n o p l a s t y 4. D u r i n g this operation, there is a n increased risk of d a m a g e to the n a s o p a l a t i n e nerve 7. Once this nerve is sectioned, the fibers try again to find their direction a n d to reach the target cells. D u r i n g the nervous regeneration, axons could take the w r o n g route; l b r example, fibers normally directed to palatal m i n o r glands a n d m u c o s a could i n n e r v a t e the nasal

glands a n d mucosa. T h e result would be nasal secretion each time there is a stimulation of salivary glands, as d u r i n g ingestion. T r a u m a to the s p h e n o p a l a t i n e nerve or some of its b r a n c h e s at the level of the p t e r y g o m a x i l l a r y fossa is comm o n d u r i n g maxillectomy. This could also result in anomalies of the nervous regeneration a n d could explain the GR. G R is n o t r e p o r t e d as a complication of Le Fort I osteotomies because the cuts of the maxillary osteotomy are positioned too low to d a m a g e the fibers directed to the nasal mucosa. Only the palatal b r a n c h e s are sectioned; therefore, the nervous regeneration c a n n o t cause G R . P h e n o m e n a caused by faulty nerve regeneration are n o t rare in the maxillofacial region 5,9,1~ G R is only one of the possible effects. A certain degree of alertness to this disorder o u g h t to be present a m o n g clinicians w h o are dealing with this type of surgery.

References 1. BODDIE AW, GUILLAMONDEGUIOM, BYERS RM. Gustatory rhinorrhea developing after radical parotidectomy a new syndrome? Arch Otolaryngol 1976: 102: 248 50. 2. FAUSSAT JM, GHIASSI B, PRINC O. Une rhinorrh6e d'origine parotidienne (~ propos d'un cas). Rev Stomatol Chir Maxillofac 1993: 94:363 5. 3. GRI~GOIRER, OBERLIN S. Pr6cis d'anatomie. 9th ed. Vol. 1. Paris: Baillibre, 1981. 4. GUYURON B, MICHELOW B, THOMAS T. Gustatory rhinorrhea a complication of septoplasty. Plast Reconstr Surg 1994: 94: 454-6. 5. LATERREECH, PIERREPH. Pathologies algique et neuromusculaire crgmio-faciomaxillaires. In: PIETTE E, REYCHLER H. Trait6 de pathologies buccale et maxillofaciale, Brussels: De Boeck Universit6, 1991: 15834. 6. LEBLANC A. Imagerie anatomique des nerfs crfiniens: m6thode d'investigation pour l'imagerie par r6sonance magn6tique (IRM) et la tomodensitom6trie (CT). Berlin: Springer-Verlag, 1989. 7. MARSHALL DR, SLATTERY PG. Intracranial complications of rhinoplasty. Br J Plast Surg 1983: 36: 342-4. 8. ROUVI~RE H, DELMAS A. Anatomie humaine (descriptive, topographique et fonctionnelle). 13th ed. Paris: Masson, 1991. 9. SUNDERLAND as. Cranial nerve injury, structural andphysiological considerations and a classification of nerve injury. In: SAMn M, JANETTAPJ, eds.: The cranial nerves. Berlin: Springer, 1981:16 26. 10. SUNDERLANDSS. Nerve injuries and their repair. A critical appraisal, London: Churchill, Livingstone, 1991: 79-128.

Address: Hassan Mir Mohammad Sadeghi, MD, DDS Unit of Oral and Maxillofacial Surgery St Luc University Hospital Catholic University of Louvain ( UCL), School of Medicine 15, Av. Hippoerate B-1200 Brussels Belgium