Journal of Neuro-Ophthalmology, June 1992, Volume 12, Issue 2

Horner's syndrome caused by intra-oral trauma.

A 7-year-old boy developed a Horner's syndrome after falling on a stick that penetrated his peritonsillar soft palate. He did not suffer from any major vascular injury, and pharmacologic testing indicated a preganglionic lesion. We review previously reported cases of oculosympathetic paresis caused by surgical and nonsurgical intra-oral trauma. Because of the proximity between sympathetic and vascular structures in the lateral and parapharyngeal space, Horner's syndrome in the setting of intra-oral trauma should prompt evaluation of the internal carotid artery. Magnetic resonance imaging may be a reasonable noninvasive method for this investigation.

' 9 1992 Raven Press, Ltd., New York Horner's Syndrome Caused by Intra- oral Trauma Grant T, Liu, M. D" Ronald W, Deskin, M. D" and Don C Bienfang, M, D. A 7- year- old boy developed a Horner's syndrome after falling on a stick that penetrated his peritonsillar soft palate. He did not suffer from any major vascular injury, and pharmacologic testing indicated a preganglionic le­sion. We review previously reported cases of oculosym­pathetic paresis caused by surgical and nonsurgical in­tra- oral trauma. Because of the proximity between sym­pathetic and vascular structures in the lateral and parapharyngeal space, Horner's syndrome in the setting of intra- oral trauma should prompt evaluation of the in­ternal carotid artery. Magnetic resonance imaging may be a reasonable noninvasive method for this investiga­tion. Key Words: Horner's syndrome- Intra- oral trauma­Internal carotid artery- Magnetic resonance imaging. From the Divisions of Neurology ( G. T. L.) and Ophthalmol­ogy ( D. C. B.), Brigham and Women's HospitaL and the Depart­ments of Neurology ( G. T. L.) and Otolaryngology ( R. W. D.), Children's HospitaL Harvard- Longwood Neurology Program, Harvard Medical School, Boston, Massachusetts, U. S. A. Address correspondence and reprint requests to Dr. Don C. Bienfang, Division of Ophthalmology, Brigham and Women's HospitaL 75 Francis Street, Boston MA 02115, USA. 110 Horner's syndrome, characterized by unilateral ptosis, miosis, and facial anhydrosis, indicates an interruption in the sympathetic innervation of the eye ( 1). First- order hypothalamic neurons descend through the brainstem and then synapse with pre­ganglionic neurons that exit the spinal cord ipsilat­erally at C8 through T2. These second- order fibers then travel rostrally via the sympathetic chain and terminate in the superior cervical ganglion, Post­ganglionic neurons ascend within the internal ca­rotid plexus and then through the cavernous sinus to reach the eyelids and iris dilator muscle ( 2), Su­domotor fibers to the lower face follow the external carotid artery ( 3). In this article we describe a rare patient who developed a Horner's syndrome as a result of in­tra- oral trauma. We then review the literature of other cases of surgical and nonsurgical intra- oral trauma resulting in oculosympathetic paresis and discuss the implication of Horner's syndrome in the evaluation of this type of injury, CASE REPORT A previously healthy 7- year- old boy was re­ferred to Children's Hospital for evaluation of a new Horner's syndrome after a peritonsillar in­jury, He had slipped and fallen on the ice, and a stick pointing up from the ground grazed the mid­dle of his tongue and then punctured his soft pal­ate. Immediately he spit up approximately 100 m1 of blood, and over the next several hours the bleeding continued insidiously, eventually causing him to vomit coffee- ground materiaL However, aside from a sore throat and right- sided neck pain, he felt well enough to stay at home, The next day his mother brought him to his pe­diatrician for a tetanus shot. The pediatrician no­ticed the child's peritonsillar puncture site, and af- HORNER'S SYNDROME 111 ter observing anisocoria and a right droopy eyelid, he diagnosed a Horner's syndrome. He then re­ferred the child to the hospital. Initial examination revealed an alert boy with right upper lid ptosis and lower lid inverse ptosis that the father stated was new. The right pupil measured 3 ml and the left 5 ml, but both were equally reactive to light. The irises were the same color. Eye movements were normal. The right half of the face was dry compared to the left. The tongue and palate moved normally, and the re­mainder of his neurological examination was un­remarkable. On the right anterior tonsillar pillar there was a 0.5 cm healing laceration ( Fig. 1). There was soft- tissue swelling and crepitus over the right side of the neck and superior chest wall. There were no carotid bruits. A chest x- ray demonstrated pneumomediasti­num, pneumopericardium, and subcutaneous em­physema in the neck and shoulders. Magnetic res­onance imaging ( MRI) of the neck ( Fig. 2) revealed air within the soft tissues in the retropharyngeal space. No significant hematoma or injury to the internal or common carotid artery was detected, and no thrombus or dissection was seen. The pa­tient received antibiotics and then left the hospital with his eye findings unchanged. Sixteen days after his initial injury, the right pto­sis and miosis were still present ( Fig. 3A). After the lights were dimmed, the right pupil dilated more slowly than the left, so the anisocoria was more prominent at 5 seconds than at 15 seconds. Two FIG. 1. View of the oral cavity. The arrow points to the area where a stick punctured our patient's right peri­tonsillar soft palate. FIG. 2. Axial T1- weighted " fast" ( TR = 33, TE = 13) MRI of the neck. The high signal ( large white arrow) indicates normal blood flow within the right internal carotid artery ( RICA). The low signal intensity ( small arrow) represents air surrounding the RICA intro­duced by the peritonsillar injury. The air also tracks behind the pharynx and surrounds the left internal ca­rotid artery, but to a lesser extent. drops of 1% hydroxyamphetamine ( Paredrine) in each eye dilated both pupils; the right dilated rel­atively more than the left ( Fig. 3B). Examinations 1 and 2 months later were basically unchanged. DISCUSSION In our patient the diagnosis of a new Horner's syndrome seems certain based on clinical criteria of ipsilateral facial anhydrosis, upper lid and " up­side- down" ( lower lid) ptosis ( 4), miosis, and pu­pillary dilation lag ( 5). Testing with cocaine, which blocks norepinephrine reuptake and relies on a completely intact oculosympathetic pathway, was not performed. Thus, a " pseudo- Horner's" syn­drome, characterized by ptosis and miosis not caused by oculosympathetic paralysis ( 6), could not be excluded though it seems unlikely. Iris het­erochromia, typical of a congenital Horner's syn­drome ( 7), was not present. Penetrating intra- oral trauma may cause a Horner's syndrome by damaging sympathetic structures adjacent to the peritonsillar area. In an adult, the superior cervical ganglion, which is of- JClin Neuro- ophthalmol. Vol. 12, No. 2, 1992 112 ( A) G. T. LIU ET AL. ( S) FIG. 3. A: Horner" s syndrome, characterized by right upper eyelid ptosis, lower lid inverse ptosis, and miosis. B: After two drops of 1% hydroxyamphetamlne ( parednn~) in each eye, the right pupil dilates relatively more than the left, Implying a pregangli­onic oculosympathetic lesion. ten 2 to 3 cm long ( 2), lies approximately 30--- 40 degrees lateral and only 1.5 cm behind the palatine tonsil ( 8) ( Fig. 4). At this level the internal carotid artery ( lCA), surrounded by the sympathetic plexus, is situated immediately lateral to the supe­rior cervical ganglion. Other neighboring struc­tures include the internal jugular vein, external ca­rotid artery, and the glossopharyngeal ( IX), vagus ( X), and hypoglossal ( XII) nerves. The cervical sympathetic chain lies caudally, below the angle of the mandible ( 2). Hydroxyamphetamine, which causes release of norepinephrine at sympathetic nerve endings and depends on an intact postganglionic neuron, di­lated our patient's miotic pupil more than the my­driatic one, suggesting a preganglionic lesion ( 9,10). Because of its proximity to the tonsil, we suspect that direct but partial injury to the right superior cervical ganglion caused our patient's Horner's syndrome. Partial injury to the superior cervical ganglion, by sparing some cell bodies of third- order neurons, would allow some pupillary dilation during hydroxyamphetamine testing ( 11). Hemifacial anhidrosis is consistent with pregangli­onic injury. Other explanations for a preganglionic lesion, such as direct trauma to the cervical sympathetic JClin Neuro- nphthalmol, Vol. 12, No. 2, 1992 chain, subcutaneous air directly or indirectly com­pressing on second- order axons near the medias­tinum or in the lower part of the neck, or injury to central neurons, cannot be excluded. Although le­sions of second- order and central neurons are in­distinguishable pharmacologically, a central defect seems improbable because there were no other findings referable to the brainstem or spinal cord. MRI ruled out any compressing hematoma or mass lesion in the neck. Horner's syndrome following intra- oral trauma may also result from direct blunt injury to the ICA plexus. Any ICA intimal disruption subsequently could result in thrombus formation ( 12) or dissec­tion ( 13,14) and then plexus ischemia ( 15). Plexus injury or ischemia, however, would by definition produce a postganglionic lesion, and sweating in the lower half of the face would be intact ( 3). Pos­sible ischemic damage to the superior cervical gan­glion, which derives its blood supply from the ICA, is more complicated. Complete compromise would produce a postganglionic lesion with hy­droxyamphetamine testing; incomplete lesions might be preganglionic ( 11). Nevertheless, our pa­tient's ICA as visualized by MRI was intact. It is possible that the pharmacological localiza­tion was incorrect. Although cocaine will poorly HORNER'S SYNDROME 113 20m lateral anterior L ramus of mandi ble medial J pterygoid and styloglossus muscles external ca rotida....". (-\.. fj posterior bell y su:::: jzfQ;::~~ riC muscle cervicol ganglion '-..,..~ o ...; n, ernal ~... ICA C)~ Jugularv. vertebral " ~ ~--. a. CNX oral cavlfy posterior' pharyngeal \\ Iall FIG. 4. Vascular and nervous structures posterior and lateral to the palatine tonsil ( axial view). a, artery; CN, cranial nerve; ICA, internal carotid artery; v, vein. Adapted, with permission from Schnitzlein and Murtagh ( 8). dilate and therefore identify all Horner's pupils ( 7), the hydroxyamphetamine test to differentiate pre­from postganglionic injury may sometimes be mis­leading and inaccurate ( 16). A miotic pupil result­ing from an incomplete carotid plexus lesion might still dilate after instillation of hydroxyamphet­amine, thereby leading to the incorrect diagnosis of a preganglionic lesion ( 10). It is also conceivable, though unlikely, that days or weeks must pass be­fore the supply of presynaptic norepinephrine is exhausted after postganglionic injury. In addition, hydroxyamphetamine acts variably from person to person ( 17,18). Horner's syndrome resulting from intra- oral trauma is rare and is not mentioned in many of the large series of patients with oculosympathetic paresis ( 4,16,19,20). However, several isolated cases have been reported ( 21- 32). The reports are summarized in Table 1. We are unable to explain why intra- oral trauma, which is a common child­hood injury ( 12), so infrequently leads to Horner's syndrome. As with our patient, two of the patients reported with nonsurgical trauma developed Horner's syn­drome as their sole neurological manifestation ( 26,32). Pruett ( 26), whose patient fell on a dowel stick, localized his patient's lesion to the carotid plexus, whereas Bazak et al. ( 32) attributed their patient's Horner's syndrome to sympathetic chain injury resulting from accidental ingestion of a sew­ing needle that embedded in the retrostyloid space. In both cases the Horner's syndrome re­solved within a few months, presumably after reinnervation. Unfortunately, neither author local­ized the damage pharmacologically. Acquaviva et al. ( 25) described an unfortunate individual who suffered a traumatic fracture of his dental plate and an " intra- buccal" hematoma on the left. Two days later he became lethargic and aphasic and developed a left miosis and a right hemiparesis. Angiography demonstrated throm­bus within the left ICA. The pupils normalized af­ter 2 days, but the aphasia and hemiparesis per­sisted and the patient died 17 days after the initial trauma. ICA occlusion and stroke following intra­oral trauma have been well described, and several authors have reviewed this topic ( 12,33- 35). Pitner ( 12) stressed that there may be a 3- to 24- hour " lu­cid interval" between the actual injury and neuro­logic symptoms. Horner's syndrome may also occur after intra­oral surgery or anesthesia ( see Table 1). Oculosym­pathetic paresis after tonsillectomy ( 21,23,28,31) may be long lasting; in contrast a Horner's syn­drome due to peritonsillar anesthesia, caused by accidental injection around sympathetic struc- JCli" Neuro- ophthalmol. Vol. 12, No. 2, 1992 114 TABLE 1. G. T. LIU ET AL. Reported cases of Horner's syndrome due to nonsurgical and surgical intra- oral trauma Author and reference Acquaviva et al. ( 25) Pruett ( 26) Bazak et al. ( 32) Liu et al. Year 1961 1967 1987 1992 Mode of injury NON- SURGICAL Traumatic fracture of dental plate leading to ICA thrombosis and miosis of ipsilateral pupil Dowel- stick impaled tonsillar fossa Swallowed sewing needle that lodged next to internal carotid artery Stick penetrated peritonsillar soft palate SURGICAL Author's proposed location of sympathetic injury ? Carotid plexus Sympathetic chain Partial injury of superior cervical ganglion Time to resolution 2 days; patient died after 17 days 4 months 3 months At least 14 weeks Hammer ( 21) 1933 Meurman ( 22) 1934 Frishman ( 23) 1938 Hald & Godtfredsen ( 24) 1942 Strumien ( 27) 1969 Zollner & Herrmann ( 28) 1971 Campbell et al. ( 29) 1979 Novoselitskii ( 30) 1979 Gariuk ( 31) 1982 Tonsillectomy Ten out of " hundreds" of cases of peritonsillar anesthesia prior to tonsillectomy Tonsillectomy- 4 cases 11 out of 22 cases of peritonsillar anesthesia prior to tonsillectomy Dental extraction Abcess following tonsillectomy leading to Horner's syndrome and hypoglossal and laryngeal nerve paralysis Hematoma of tonsillar pillars and posterior pharyngeal wall after attempted inferior alveolar nerve anesthesia Tonsillectomy Following removal of branchiogenic cyst of palatine tonsil Sympathetic trunk Sympathetic trunk Carotid plexus vs. stellate ganglion block from extravasated anesthesia B 1 hour B 1- 2 hours B 2 hours ICA, internal carotid artery; ?, author( s) did not give this information B Further details unavailable in these foreign reports. tures, is transient ( 22,24,30). Trauma to the lCA may occur during tonsillectomy ( 36,37), but was not implicated in any of these cases. A Horner's syndrome following tonsillectomy would reflect unintentional injury to the sympathetic chain, su­perior cervical ganglion, or carotid plexus. Because of the proximity between the superior cervical ganglion, sympathetic plexus, and the lCA ( 8), a Horner's syndrome may herald lCA throm­bus formation or dissection after trauma to the peritonsillar region ( 26). It may be the only neuro­logical abnormality during the " lucid interval." As in our patient, MRI with axial images through the neck may be the preferred modality to exclude ex­tracranial lCA occlusion ( 38) or dissection ( 14,39). Because of the 30% morbidity associated with ca­rotid injury caused by intra- oral trauma ( 33) and because of the safety of MRI, we suggest that all lOin Neuro- ophth"' mol. "" 01. 1~. 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