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Oculocardiac Reflex in an Adult with a Trapdoor Orbital Floor Fracture: Case Report, Literature Review, and Differential Diagnosis

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Oculocardiac Reflex in an Adult with a Trapdoor Orbital Floor Fracture: Case Report, Literature Review, and Differential Diagnosis https://doi.org/10.5125/jkaoms.2020.46.6.428 CASE REPORT pISSN 2234-7550 · eISSN 2234-5930 Oculocardiac reflex in an adult with a trapdoor orbital floor fracture: case report, literature review, and differential diagnosis Bernardo Ferreira Brasileiro1, Joseph E. Van Sickels2, Larry L. Cunningham, Jr.2 1Kentucky Clinic, 2Division of Oral and Maxillofacial Surgery, College of Dentistry, University of Kentucky, Lexington, KY, USA Abstract (J Korean Assoc Oral Maxillofac Surg 2020;46:428-434) Orbital floor blowout fractures can result in a variety of signs and symptoms depending on the severity of the bone defect. Large defects often result in enophthalmos and restriction of ocular movement; yet the timing of surgery can be delayed up to two weeks with good functional outcomes. In con- trast, an orbital trapdoor defect with entrapment of the inferior rectus muscle usually elicits pain with marked restriction of the upward gaze and activa- tion of the oculocardiac reflex without significant dystopia or enophthalmos. When autonomic cardiac derangement is diagnosed along with an orbital floor fracture, it has been suggested that the fracture should be treated immediately. Otherwise, it will result in continued hemodynamic instability and muscular injury and may require a second surgery. This article reports the management of an unusual presentation of a trapdoor blowout orbital floor fracture surgery with oculocardiac response in an adult, with emphasis on its pathophysiology, management, and differential diagnosis. Key words: Orbital fractures, Facial injuries, Oculocardiac reflex [paper submitted 2018. 10. 9 / revised 2018. 10. 31 / accepted 2018. 11. 17] I. Introduction who has softer, more elastic bones, an impact can result in a fracture that first hinges open, springing inferiorly, and then Orbital floor fractures can present in isolation or in com- returns to the original position, thus entrapping tissue. Small bination with other facial fractures. The etiology is usually floor fractures are more likely to entrap extraocular muscles, direct trauma to the orbital region resulting in fracture to the leading to a compartment syndrome, which if not promptly orbital floor1. The fracture pattern is usually an “open door” relieved can lead to prolonged and possibly permanent pa- defect and is typically seen in adults with herniation of the resis and muscular scarring. The usual presentation of a orbital contents into the maxillary sinus. Clinically, there may linear fracture of the orbital floor or medial wall can occur be periorbital bruising, subconjunctival hemorrhage, diplopia, as a “trapdoor” with incarceration of orbital soft tissue and altered sensation of the infraorbital nerve, and enophthalmos. muscles accompanied by little sign of soft tissue injury4. This Any limitation in ocular movement is usually due to pain or type of fracture has been called the white-eyed orbital blow- “relative entrapment” of the ocular muscles2. out fracture (WEOBF) and was first named in a report by Greater elasticity of the facial skeleton in children can Jordan et al.5. They described it occurring in a group of young result in a slightly different fracture pattern, thus changing patients (less than 16 years of age) with little or no clinical the clinical presentation3. Following orbital trauma in a child evidence of soft tissue trauma, diplopia with restriction of the vertical gaze, lack of exophthalmos, and radiographic signs of minimal bone displacement with or without tissue hernia- Bernardo Ferreira Brasileiro tion into the maxillary sinus. In an even more serious sce- Kentucky Clinic, University of Kentucky, Adult Dentistry, 740 South Limestone, Lexington, KY 40508, USA nario, these patients may have true entrapment of the inferior TEL: +1-859-323-8873 FAX: +1-859-257-3270 rectus, pain with restricted ocular movement, and autonomic E-mail: [email protected] 6 ORCID: https://orcid.org/0000-0002-5886-267X signs, which are secondary to the oculocardiac reflex (OR) . CC This is an open-access article distributed under the terms of the Creative The OR is a vagally-mediated slowing of the heart follow- Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits unrestricted non-commercial use, distribution, ing painful stimulation of the orbital contents through an arc and reproduction in any medium, provided the original work is properly cited. reflex response, which can result in a 20% or more reduction Copyright © 2020 The Korean Association of Oral and Maxillofacial Surgeons. All 7,8 rights reserved. in heart rate, the presence of arrhythmias, or both . The first 428 Oculocardiac reflex in an adult with a trapdoor orbital floor fracture description of this reflex was simultaneously but indepen- II. Case Report dently reported in 1908 by Aschner9 and Dagnini10. They described a pressure-induced neural reflex causing cardiac A 26-year-old white female was seen following a motor depression through vagal stimulation, and the phenomenon vehicle collision. She was an unrestrained backseat passen- was labeled “oculocardiac reflex”. For centuries before their ger who hit her face against the front seat backrest. Her vital reports, pressure on the globes had been used therapeutically signs showed stable blood pressure readings, normal heart to treat paroxysmal atrial tachycardia11. Nowadays, the OR and respiratory rates, and regular temperature. However, she is a well-known condition in ophthalmology, anesthesiology, had nausea, vomiting, and syncope with attempts to move the otolaryngology, neurology, and maxillofacial surgery. Typical right eye. Visual acuity was 20/20 OU. There was no loss of presenting signs and symptoms include bradycardia, altera- globe integrity, enophthalmos, or hypoglobus. No afferent pu- tions in blood pressure, faintness, and nausea, when any of pillary defect or infraorbital nerve paraesthesia was detected. the peripheral branches or the central component of the tri- Minimal subconjunctival and upper eyelid ecchymosis was geminal nerve are stimulated12. The OR has been documented observed on the affected side. Remarkably, eye pain, diplo- during temporomandibular joint surgery and mandibular pia, and dizziness were noted and increased with attempts at osteotomies, emphasizing that the maxillary and mandibu- a superior gaze. The patient was found to have severe impair- lar divisions of the trigeminal nerve can be involved as the ment of superior and limited inferior gaze of the right eye.(Fig. ophthalmic branch4. In 1988, Shelly and Church13 introduced 1) Coronal computed tomography (CT) scans with 1-mm the term “trigeminocardiac reflex” to describe the overall sections revealed a small disruption in the median aspect of mechanism, clarifying that the OR, although most commonly the inferior floor of the right orbit, which was compatible elicited, is a particular manifestation of a more general reflex with entrapment of periorbital soft tissue contents.(Fig. 2) phenomenon. Therefore, any surgical intervention in the dis- Cerebral and spinal injuries were ruled out after clinical and tribution of the trigeminal nerve poses a risk of precipitating tomographic evaluations. There were no chest or abdominal a cardiovascular incident in response to a trigeminal stimula- injuries noted. tion14. Based on the radiographic findings of orbital content en- The WEOBF in a pediatric patient is an often cited cause trapment and the clinical signs of impaired globe movement for inducing the OR. In this report, the authors describe an with uncontrolled nausea, a diagnosis of an orbital floor frac- unusual presentation of an orbital floor blowout fracture with ture with potential risk for an OR was established. A decision entrapment of orbital contents, minimal soft tissue trauma was made to take the patient immediately to the operating signs, and elicited OR during orbital floor fracture surgery in room to release the entrapped inferior rectus muscle and to an adult patient. The article also aims to discuss the patho- reconstruct the orbital floor. Early intervention was deemed physiology of the OR and its management in orbital floor necessary to prevent muscle ischemia and to improve ocular fractures with muscle entrapment, as well as reviewing a dif- motility. The risk of OR exacerbation was discussed with the ferential diagnosis in a trauma patient. anesthesiology team prior to surgery. The patient underwent an uneventful induction and oral intubation in the operating room within 4 hours after pre- Fig. 1. Preoperative clinical examination of the upward gaze, Fig. 2. Preoperative coronal computed tomography (coronal view) demonstrating vertical restriction in ocular motility after an orbital showing right floor linear discontinuity with entrapment of orbital floor blowout fracture on the right side. contents (white arrow) into the maxillary sinus (tear drop sign). Bernardo Ferreira Brasileiro et al: Oculocardiac reflex in an adult with a trapdoor Bernardo Ferreira Brasileiro et al: Oculocardiac reflex in an adult with a trapdoor orbital floor fracture: case report, literature review, and differential diagnosis. J Korean orbital floor fracture: case report, literature review, and differential diagnosis. J Korean Assoc Oral Maxillofac Surg 2020 Assoc Oral Maxillofac Surg 2020 429 J Korean Assoc Oral Maxillofac Surg 2020;46:428-434 sentation to the Emergency Department. Periorbital tissue III. Discussion infiltration with local anesthetics was administered. A forced duction
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