CRANIOMAXILLOFACIAL DEFORMITIES/COSMETIC SURGERY

Intracranial Subdural Hygroma After Le Fort I Osteotomy William L. Thompson, DDS, MD,* Michael Lee, BS,y and Robert Bruce MacIntosh, DDSz Various intra- and postoperative complications have been well-documented after Le Fort I osteotomies; however, an intracranial subdural hygroma has not yet been reported in oral and maxillofacial studies. We report a unique case of an intracranial subdural hygroma requiring neurosurgical intervention after Le Fort I advancement. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 73:727-731, 2015

Case Report

The day after discharge from the hospital, the patient telephoned complaining of facial and head pain; however, he denied nausea, vomiting, or changes in vision or hearing. The patient was advised to return to the office for observation the next day. When seen in the office on postoperative day 5, the elastics holding the intermaxillary fixation were changed, and the patient was ambulating without difficulty. However, he continued to complain of a headache and facial pain. On postoperative day 8, the patient telephoned still complaining of head pain not relieved by the prescribed oral medications (opioids). He also reported vertigo. He was instructed to go to the emergency room for evaluation. The emergency staff ordered a computed tomography scan (Fig 1) and magnetic resonance imaging (MRI) studies (Figs 2, 3). The results indicated a right-to-left midline shift of the cerebral hemispheres, with fluid in the subdural space. The neurosurgery service immediately took the patient to the operating room. After burr hole evacuation in the frontotemporal area, an immediate escape of fluid under pressure ensued. The neurosurgical team collected the fluid for culture. After irrigation of the subdural space, they placed a drain and exteriorized it through their surgical incision. The laboratory tests confirmed that the fluid was cerebrospinal, and a new MRI study suggested a diagnosis of cerebrospinal fluid (CSF) hygroma. The headache resolved, the drain was removed, and the

A 30-year-old man presented for routine orthognathic evaluation and treatment. The patient’s chief complaints were unhappiness with his occlusion and a poor outcome from his earlier chin implant. His medical history was unremarkable, and his surgical history included only the chin implant. The orthognathic evaluation revealed an asymmetric mandible (3.5 mm to the patient’s left), a midline discrepancy of the maxilla to the left (1.0 mm), a retruded mandible, and a deep mentolabial fold. The patient’s occlusion was Class II, with a deep impinging anterior occlusion, and the mandibular left first molar was missing. The cephalometric measurements revealed an SNA of 77, SNB of 72, and a convex facial profile. After completion of preoperative orthodontics, the patient underwent Le Fort I osteotomy with 8-mm advancement, bilateral sagittal split ramus osteotomy, removal of the chin implant, advancement genioplasty, and an osseointegrated implant for the mandibular left first molar. The operative procedure was uneventful. On postoperative days 1 and 2, the patient had the expected postoperative facial edema. He was tolerating oral fluids, and the pain was well-controlled with intravenous opioids. On postoperative day 2, the patient was ambulating, voiding, and taking fluids; he was discharged on postoperative day 3. *Surgeon, Huntington Group, Huntington Woods, MI.

Received September 3 2014

ySenior Student, University of Detroit Mercy School of Dentistry, Detroit, MI.

Accepted October 21 2014 Ó 2015 American Association of Oral and Maxillofacial Surgeons

zClinical Professor, Department of Oral and Maxillofacial Surgery,

0278-2391/14/01622-X

University of Detroit Mercy School of Dentistry, Detroit, MI.

http://dx.doi.org/10.1016/j.joms.2014.10.023

Address correspondence and reprint requests to Dr Thompson: Huntington Group, 26111 Woodward Ave, Huntington Woods, MI 48070-1367; e-mail: [email protected]

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FIGURE 1. Computed tomography image demonstrating right-sided cerebrospinal fluid density extra-axial fluid collection. The cerebrospinal fluid collection on the right side of the skull (arrow). Thompson, Lee, and MacIntosh. Intracranial Subdural Hygroma. J Oral Maxillofac Surg 2015.

patient was discharged on postoperative day 4. The patient continued his postoperative evaluation with the oral and maxillofacial and neurosurgical services and had no return of symptoms.

Discussion CSF is produced by the choroid plexus of the brain and acts as a mechanical cushion between the brain and the hard skull. The CSF flows within the subarachnoid space to lubricate the layers of the meninges and the other components of the brain. Although commonly and safely used, the published data have indicated that Le Fort I osteotomies are not free of intra- and postoperative complications. Steel and Cope1 have offered a most recent exhaustive overview, describing postoperative nausea, vomiting, infection, sinusitis, malunion, and relapse of skeletal and dental occlusion. Ophthalmic complications, including a lack of tearing,2,3 excessive tearing,4 vision loss,5,6 abducens palsy,7,8 and oculomotor palsy,9-11 have been reported in published studies. However, very few cases have been reported regarding postoperative intracranial complications.

Baker et al12 reported a brain abscess after Le Fort I osteotomy, bilateral sagittal ramus osteotomy, and sliding genioplasty. Bendor-Samuel et al11 reported initial cavernous sinus thrombosis and oculomotor nerve palsy, with eventual subarachnoid hemorrhage, carotid cavernous sinus fistula, and internal carotid artery aneurysm. Finally, Bhaskaran et al13 reported CSF rhinorrhea, which was attributed to an unusually thick pterygomaxillary junction that resulted in an unfavorable separation. However, the patient did not develop any of the previously reported complications. Subdural hygroma (SDG) is a nonhemorrhagic, intracranial, subdural, CSF collection. SDG occurs most often in the extremes of life,14 with it most commonly occurring in patients older than 50 and younger than 5 years of age.15 The most common etiology has been head trauma.15,16 Lee et al15 reported 61 patients with SDG, of whom 85.2% had head trauma, with 40.9% of all reported patients having radiographic signs of skull fracture. Zanini et al16 reported 34 patients who presented with SDG, with the main cause of head trauma being road traffic accidents. Our patient fit neither the age group nor, apparently, the amount of trauma required for presentation of traumatic SDG.

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FIGURE 2. Magnetic resonance image demonstrating right-sided cerebrospinal fluid density extra-axial fluid collection. The cerebrospinal fluid collection on the right side of the skull (arrow). Thompson, Lee, and MacIntosh. Intracranial Subdural Hygroma. J Oral Maxillofac Surg 2015.

However, 1 rare etiology of acute SDG is rupture of pre-existing arachnoid cysts, with only 46 cases with this etiology reported in published studies. Our report would seem the first in oral and maxillofacial studies. Arachnoid cysts are usually asymptomatic and only incidentally found on intracranial imaging, with a frequency of 1.4% in adults. They can be congenital or result from minor trauma, and their long-term behavior is not well understood. Reports have described the cysts becoming both larger and smaller and even undergoing spontaneous resolution. Furthermore, arachnoid cysts occur most commonly in the middle cranial fossa, on the left side, and in males.17 We believe that our patient presented preoperatively with an undisclosed asymptomatic arachnoid cyst. The cyst could have conceivably ruptured, gradually seeping CSF into the subdural space through a ‘‘flap-valve’’18 effect, causing the increase in intracranial pressure. We postulate this rupture could have been caused in 1 of 4 ways: 1) routine trauma from the procedure itself; 2) a hypertensive episode intraoperatively; 3) a postoperative Valsalva maneuver19; or 4) spontaneous and coincidental rupture of the cyst.

First, the procedure itself, with pterygomaxillary separation and maxillary down fracture, might have caused routine trauma to the cranial base, rupturing the arachnoid cyst. However, we believe that although the forces from the procedure were distributed throughout the cranial base, such forces would not be enough to have had an effect on the intracranial entities (ie, the arachnoid cyst). In addition, intraoperatively, the separation and down fracture was of normal difficulty, and postoperatively, the CT scan showed no evidence of a cranial base fracture. In contrast to the study by Bhaskaran et al13 of CSF rhinorrhea, our patient showed no signs of such a phenomenon, signifying no violation to the cranial base. Second, the anesthesia records did not show evidence of a hypertensive episode that would have increased the intracranial pressure, rupturing the cyst. However, postoperative evaluation of our patient revealed normal postoperative nasal congestion. Also, although the patient was instructed not to evacuate his nasal secretions, he might not have been compliant. This behavior could have increased the intracranial pressure enough to rupture the arachnoid cyst. Finally, spontaneous rupture of arachnoid cysts have been

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FIGURE 3. Magnetic resonance imaging slice demonstrating right-sided cerebrospinal fluid density extra-axial fluid collection. The cerebrospinal fluid collection on the right side of the skull (arrow). Thompson, Lee, and MacIntosh. Intracranial Subdural Hygroma. J Oral Maxillofac Surg 2015.

reported in published studies.20-22 We believe that the most probable theory for this complication was the Valsalva maneuver, which constantly challenged the integrity of the cyst, or a result of a spontaneous phenomenon that coincidentally presented after surgery. The ruptured cyst then allowed the CSF to collect in the subdural space, causing a slight rightto-left midline shift and resulting in neurologic symptoms requiring emergency neurosurgical intervention. Maher et al21 in 2013 reported the most recent exhaustive data review of arachnoid cyst rupture resulting in a SDG. With the cases listed in their overview, including 2 additional cases,23,24 47 cases (including ours) have now been reported. The average patient age was 12 years (range 5 to 25); our patient, age 30, was the oldest. Also, 89% of the reported patients were male, 74% of the cases were due to head trauma, and 95% of the patients presented with 1 or more of the following symptoms: headache, nausea and vomiting, papilledema, or diplopia. The location of the hygroma was nearly even, with the left side having a slight preponderance (21 and 20 on the left and right side, respectively, excluding bilateral presentations and other intracranial locations). All the hygromas

occurred in the middle cranial fossa, except for 1 in the quadrigeminal cistern22 and 1 in the retrocerebellar area.25 Three hygromas presented bilaterally.21,22,26 In conclusion, most published studies have recommended surgical treatment as the standard of care. Some have even suggested that nonoperative management is inappropriate.27 It has been recently noted, however, that the natural course of SDG is generally benign, with ultimate spontaneous resolution.21 That, with our patient, the neurosurgery service believed it was necessary to operate suggests that more standardization and discussion of the treatment modalities to manage this entity are required. Acknowledgments Special thanks to David M. Rombach, DDS, MD, John V. Gaul, DDS, and Michael E. Heath, DDS for reviewing and contributing to our report.

References 1. Steel BJ, Cope MR: Unusual and rare complications of orthognathic surgery: A literature review. J Maxillofac Oral Surg 70: 1678, 2012

THOMPSON, LEE, AND MACINTOSH 2. Lanigan DT, Romanchuk K, Olson CK: Ophthalmic complications associated with orthognathic surgery. J Maxillofac Oral Surg 51:480, 1993 3. Tomasetti BJ, Broutas M, Gormley M, et al: Lack of tearing after Le Fort I osteotomy. J Oral Surg 34:1095, 1976 4. Keller EE, Sather AH: Quadrangular Le Fort I osteotomy: Surgical technique and review of 54 patients. J Oral Maxillofac Surg 48:2, 1990 5. Cruz AA, dos Santos AC: Blindness after Le Fort I osteotomy: A possible complication associated with pterygomaxillary separation. J Craniomaxillofac Surg 34:210, 2006 6. Cheng HC, Chi LH, Wu JY, et al: Blindness and basal ganglia hypoxia as a complication of Le Fort I osteotomy attributable to hypoplasia of the internal carotid artery: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104:e27, 2007 7. Hanu-Cernat LM, Hall T: Late onset of abducens palsy after Le Fort I maxillary osteotomy. Br J Oral Maxillofac Surg 47:414, 2009 8. Reiner S, Willoughby JH: Transient abducens palsy following Le Fort I maxillary osteotomy: Report of a case. J Oral Maxillofac Surg 46:699, 1988 9. Herold J, Falworth M: Sub total unilateral oculomotor nerve palsy in a Le Fort I osteotomy. Br J Oral Maxillofac Surg 34:104, 1996 10. Carr RJ, Gilbert P: Isolated partial third nerve palsy following Le Fort I maxillary osteotomy in a patient with cleft lip and palate. Br J Oral Maxillofac Surg 24:206, 1986 11. Bendor-Samuel R, Chen YR, Chen PK: Unusual complications of the Le Fort I osteotomy. Plast Reconstr Surg 96:1289, 1995 12. Baker SB, Weinzweig J, Bartlett SP, et al: Brain abscess as a complication of orthognathic surgery: Diagnosis, management, and pathophysiology. Plast Reconstr Surg 104:480, 1999 13. Bhaskaran AA, Courtney DJ, Anand P, et al: A complication of Le Fort I osteotomy. Int J Oral Maxillofac Surg 39:292, 2010 14. Lee KS: The pathogenesis and clinical significance of traumatic subdural hygroma. Brain Inj 12:595, 1998

731 15. Lee KS, Bae WK, Park YT, et al: The pathogenesis and fate of traumatic subdural hygroma. Br J Neurosurg 8:551, 1994 16. Zanini MA, De Lima Resende LA, De Souza Faleiros AT, et al: Traumatic subdural hygromas: Proposed pathogenesis based classification. J Trauma 64:705, 2000 17. Al-Holou WN, Terman S, Kilburg C, et al: Prevalence and natural history of arachnoid cysts in adults. J Neurosurg 118:222, 2013 18. Naffziger HC: Subdural fluid accumulations following head injury. JAMA 82:1751, 1924 19. Cullis PA, Gilroy J: Arachnoid cyst with rupture into the subdural space. J Neurol Neurosurg Psychiatry 46:454, 1983 20. Cayli SR: Arachnoid cyst with spontaneous rupture into the subdural space. Br J Neurosurg 14:568, 2000 21. Maher CO, Garton HJ, Al-Holou WN, et al: Management of subdural hygromas associated with arachnoid cysts. J Neurosurg Pediatr 12:434, 2013 22. Albuquerque FC, Giannotta SL: Arachnoid cyst rupture producing subdural hygroma and intracranial hypertension: Case reports. Neurosurgery 41:951, 1997 23. Galassi E, Piazza G, Gaist G, et al: Arachnoid cysts of the middle cranial fossa: A clinical and radiological study of 25 cases treated surgically. Surg Neurol 14:211, 1980 24. Gasegawa Y, Tanaka T, Kato N, et al: Arachnoidplasty for traumatic subdural hygroma associated with arachnoid cyst in the middle fossa. Neurol Med Chir (Tokyo) 50:698, 2010 25. Herman TE, Siegel MJ: Autosomal dominant polycystic disease with associated arachnoid cysts and subudral cystic hygroma requiring shunting. J Perinatol 30:566, 2010 26. Offiah C: Non-haemorrhagic subdural collection complicating rupture of a middle cranial fossa arachnoid cyst. Br J Radiol 79:79, 2006 27. Donaldson JW, Edwards-Brown M, Luerssen TG: Arachnoid cyst rupture with concurrent subdural hygroma. Pediatr Neurosurg 32:137, 2000

Intracranial subdural hygroma after Le Fort I osteotomy.

Various intra- and postoperative complications have been well-documented after Le Fort I osteotomies; however, an intracranial subdural hygroma has no...
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