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Blindness after Le Fort I osteotomy: A possible complication associated with pterygomaxillary separation
ARTICLE IN PRESS Journal of Cranio-Maxillofacial Surgery (2006) 34, 210–216 r 2006 European Association for Cranio-Maxillofacial Surgery doi:10.1016/j.jcms.2006.01.001, available online at http://www.sciencedirect.com
Case Report Blindness after Le Fort I osteotomy: A possible complication associated with pterygomaxillary separation Antonio Augusto V. CRUZ1, Antonio Carlos dos SANTOS2 1
Department of Ophthalmology, Otorhinolaryngology and Head and Neck Surgery, (Chair: Prof. M. de Lourdes Veronese Rodrigues); 2Department of Radiology, (Chair: Prof. M. Cesar Foss), School of Medicine of Ribeira˜o Preto, University of Sa˜o Paulo, Brazil
SUMMARY. Introduction: Visual loss after Le Fort I osteotomy is a devastating complication the mechanism of which is not always clear. Methods: A case report of blindness following Le Fort I osteotomy is presented. The literature on the various skull base complications associated with Le Fort I osteotomies is reviewed and the mechanisms of these complications discussed. Conclusion: The radiological findings in this case are similar to those previously reported. They strongly support the hypothesis that an adverse transmission of forces associated with pterygomaxillary separation via the sphenoid bone to the intra- and extracranial portions of the skull base is the main reason for injury to the optic and other cranial nerves as well as to the branches of the carotid artery. r 2006 European Association for Cranio-Maxillofacial Surgery
Keywords: blindness; Le Fort I osteotomy; skull base; complications
INTRODUCTION Le Fort I osteotomy is a standard technique for the surgical management of dentofacial anomalies and also for providing access for many other operations. One of the key steps in this type of osteotomy is pterygomaxillary dysjunction. The transection of this suture is not without risks and can lead to several intra- and extracranial complications (Lanigan et al., 1990, 1991, 1993; Ueki et al., 2004). Visual loss following Le Fort I osteotomy is a rare event, the mechanisms for which are still open to debate. We are aware of only seven such cases (Lanigan et al., 1993; Bendor-Samuel et al., 1995; Girotto et al., 1998; Wilson et al., 2000; Lo et al., 2002). In the present paper another case of blindness after Le Fort I osteotomy is reported and the literature on skull base complications associated with Le Fort I osteotomies is reviewed.
CASE REPORT A 22-year-old female presented with visual loss in the right eye. One week before consultation she had undergone a Le Fort I osteotomy for occlusal correction at another hospital. Immediately postoperatively her right upper and lower eyelids became swollen. The next morning, the patient had been unable to open the right eye and when the upper eyelid was manually lifted, there was no light
Fig. 1 – 22 year old female: Top: Chemosis and esodeviation of the right eye on primary position of gaze. Bottom: Sixth nerve palsy. The patient is trying to abduct the right eye. 210
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 211
perception. Magnetic resonance imaging was performed with inconclusive results. High-dose steroids were initiated but as the patient failed to recover vision she was referred to this hospital. On examination the right upper and lower eyelids were still markedly swollen. There was complete blepharoptosis on the right side. Manual upper eyelid elevation revealed intense chemosis, loss of abduction and pupillary dilatation (Fig. 1). In spite of a normal ocular fundus, there was no light perception in the right eye. A CT scan revealed complex fractures of the pterygoid plates on both sides. On the right side there was a comminuted fracture of the medial and lateral walls of the maxillary sinuses, greater wing of the sphenoid bone and lateral and anterior walls of the sphenoid sinus. The fracture involved the inferior and superior orbital fissures with a bone fragment extending from the superior orbital fissure to the orbital apex (Figs. 2 and 3).
DISCUSSION Pterygomaxillary dysjunction involves separation between the maxillary tuberosity, pyramidal process of the palatine bone, and the pterygoid plates of the
sphenoid bone. The complexity of the sutures between these three bones increases with age and in adults the region is characterized by heavily interdigitated osseous surfaces. These can be disarticulated easily only during childhood (Melsen and Ousterhout, 1987). In adolescents and adults, attempts to transect the pterygomaxillary junction often produce fractures of the pterygoid plates (Robinson and Hendy, 1986). A comprehensive literature review disclosed 25 reports of ophthalmic and neurologic complications associated with Le Fort I osteotomy (Table 1). Sporadic cases of epiphora (Demas and Sotereanos, 1989; Lanigan et al., 1993) and reports of damage to the vessels of the pterygopalatine fossa (Lanigan et al., 1990, 1991) are not listed. Two patients (cases # 19 and 20) described by Girotto et al. (1998) may have also been reported by Wilson et al. (2000) (cases # 22 and 23). Overall, the complications listed in Table 1 can be grouped into four categories: (a) loss of function of the lacrimal gland, (b) cranial nerve palsies, (c) damage to the internal carotid artery, and (d) loss of vision. Four cases of impairment of lacrimal gland function have been reported, one by Tomasetti et al.
Fig. 2 – Computed tomography of the skull base. Bone window with axial (left) and coronal (right) scans: (A) Comminuted fracture of the medial and lateral walls of the maxillary sinuses, greater wing of the sphenoid bone and lateral and anterior walls of the sphenoid sinus. Both inferior fissure (black arrows) and intracranial temporal fossa (white arrows) are involved. (B) Bone fragments can be seen in the inferior orbital fissure (black arrows) and inside the middle cranial fossa (white arrows).
ARTICLE IN PRESS 212 Journal of Cranio-Maxillofacial Surgery
Fig. 3 – Computed tomography of the skull base. Bone window with axial (left) and coronal (right) scans: (A) Bone fragment in the superior orbital fissure (arrow) and (B) bone fragment near the optic canal and fracture involving the anterior clinoid process (arrow).
(1976; case #1) and three by Lanigan et al. (1993; cases # 14, 15 and 16). Anatomically, the preganglionic parasympathetic fibres to the lacrimal gland initially travel with the facial nerve leaving at the geniculate ganglion to reach the sphenopalatine ganglion via the greater superficial petrosal and vidian nerves. The greater superficial petrosal nerve passes through the hiatus of the facial canal to enter the middle cranial fossa running forward below the dura-mater in a groove on the anterior surface of the petrous portion of the temporal bone. It then traverses the foramen lacerum where it is joined by the deep petrosal nerve to form the vidian nerve. The deep petrosal nerve is a sympathetic branch arising from the carotid plexus. The vidian nerve passes forward through the pterygoid or vidian canal to enter the pterygopalatine fossa. Postganglionic fibres join the maxillary nerve and reach the lacrimal gland via the zygomatico-temporal anastomoses with the lacrimal nerve. Any damage to the greater superficial petrosal and vidian nerves, sphenopalatine ganglion or maxillary nerve during its path to the orbit through the inferior orbital fissure can thus impair the lacrimal gland function, provoking a reduction of reflex lacrimal secretion. In all four cases of dry eye secondary to Le
Fort I osteotomy, a full recovery of the lacrimal secretion occurred slowly after surgery (8–24 months) without any serious consequences. Case #14 (Table 1) is interesting because, besides a bilateral reduction of lacrimal function, her left eye was also anaesthetized. As the corneal sensation is transmitted by the first division (ophthalmic) of the trigeminal nerve, it may be postulated that a second injury might have occurred in the region of the superior orbital fissure. Isolated cranial nerve palsies have been reported six times (cases 4, 6, 8, 10, 20, 23; Watts, 1984; Carr and Gilbert, 1986; Reiner and Willoughby, 1988; Sirikumara and Sugar, 1990; Girotto et al., 1998; Wilson et al., 2000). Case # 10 was described as Adie’s pupil, but the association of mydriasis and lack of accommodation could be the expression of a selective third nerve palsy. As already stated, cases # 20 and 23 might be the same due to the similarity between the two histories and clinical findings. Only these two (or this one patient) had a permanent sequel (third nerve paresis with aberrant regeneration), while all others recovered within 2–18 months after surgery. Seven patients had vascular accidents related to the internal carotid artery (cases # 2, 3, 5, 7, 9, 17 and 24) and one patient had an acute orbital haemorrhage from an unknown source (case # 12). Except for this
F
F
F
M
Newhouse et al. (1982)
Watts 1984
Habal (1986)
Carr and Gilbert (1986) Lanigan and Tubman (1987)
3
4
5
6
F
M
F
Reiner and Willoughby (1988)
Hes and Man (1988)
Sirikumara and Sugar (1990)
9
10
M
8
7
M
F
Tomasetti et al. (1976) Brady et al. (1981)
1
2
Sex
Authors
Case
24
19
27
23
17
28
18
32
20
24
Age (years)
Maxillary retrusion and vertical deficiency
Maxillary hypoplasia
Maxillary retrusion
Cleft lip and palate deformity Vertical maxillary excess
Maxillary retrusion
Maxillary retrusion
Vertical maxillary excess
Hemifacial microsomia
Maxillary retrusion
Diagnosis
Table 1 – Skull base complications associated with Le Fort I osteotomy
Mydriasis and accommodation paresis
Carotid cavernous sinus fistula
Sixth nerve palsy
Carotid cavernous sinus fistula
Carotid cavernous sinus fistula Third nerve palsy
Internal carotid artery haemorrhage. Traumatic arteriovenous fistula involving the right internal carotid and internal jugular vein Sixth nerve palsy
Internal carotid thrombosis
Dry eye
Complication
None
Carotid arteriography Cranial CT
CT of cavernous sinus region
Carotid arteriography
Cranial CT
Carotid angiography
CT
Angiography
Arteriography
None
Postop. imaging
Carotid cavernous fistula near the foramen lacerum Comminuted fracture through the body of the sphenoid sinus passing through the sella turcica, with lateral displacement of a fragment onto the medial surface of the right cavernous sinus Carotid cavernous fistula Fracture of the base of the skull near the middle cranial fossa –
Carotid cavernous fistula Normal
Normal
Filling defect at the bifurcation of the right internal carotid extending into the proximal middle cerebral artery. Suggestion of tear of the carotid at the level of its entry into the petrous bone Arteriovenous fistula. Absence of intracranial flow from the right internal carotid artery
–
Postop. image findings
R
R
R
L
R
R
R
R
R
L
Side
Recovery after 18 months
Recovery after balloon occlusion
Recovery after 5 months
Recovery after 7 weeks Recovery after balloon occlusion Recovery after 8 weeks Recovery after balloon occlusion
Left hemiparesis, vagus and hypoglossal nerve palsies
Recovery after 8 months Left hemiparesis
Outcome
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 213
Sex
F
F
F
F
F
F
M
M
M
Authors
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Bendor-Samuel et al. (1995)
Bendor-Samuel et al. (1995)
Girotto et al. (1998)
Case
11
12
13
14
15
16
17
18
19
Table 1 (continued)
21
30
14
20
39
42
17
16
33
Age (years)
Congenital hemifacial hypertrophy
Maxillary hypoplasia secondary to bilateral complete cleft lip and palate
Maxillary hypoplasia secondary to bilateral complete cleft lip and palate
Vertical maxillary excess Vertical maxillary excess
Vertical maxillary excess, apertognathia Apertognathia
Midfacial retrusion
Malocclusion
Diagnosis
Blindness. Sixth nerve palsy
Blindness
Dry eye, ipsilateral hypoaesthesia, dryness of the turbinates Carotid cavernous sinus fistula. Intraand extra-cavernous carotid artery aneurysm. Third nerve palsy
Dry eye
Bilateral dry eye. Left corneal anaesthesia
Blindness
Orbital haemorrhage with proptosis, increased intraocular pressure
III nerve palsy. Blindness
Complication
Cranial CT and MRI
Cranial CT
Cranial CT Angiography
Cranial CT
None
CT of orbits and skull base CT
None
CT of orbits and head
Postop. imaging
No evidence of basal skull fractures. Subarachnoid haemorrhage. Carotid cavernous sinus fistula. Carotid artery intra- and extra-cavernous aneurysm Extradural intracranial air, discontinuity in the sphenoid bone involving the anterior and posterior tables of the sphenoid sinus Normal
Normal
Fractures of the pterygoid plates and posterior lateral wall of right maxillary sinus –
Normal
Fractures of the pterygoid plates, posterior wall of the maxillary sinus, sphenoid bone, extending through the floor of the middle cranial fossa. A small fragment of bone was close to the optic nerve near the superior orbital fissure –
Postop. image findings
L
R
L
R
R
R/L
L
L
R
Side
Blindness
Low vision (optic nerve atrophy)
Partial recovery after balloon occlusion
Recovery after 14 months Recovery after 13 months
Recovery in the right eye after 2 years
Recovery after lateral cantholysis, orbital drainage and medication for lowering the intraocular pressure Optic atrophy
Recovery of the third nerve palsy . Blindness
Outcome
ARTICLE IN PRESS
214 Journal of Cranio-Maxillofacial Surgery
M
M
M
M
F
F
Girotto et al. (1998)
Girotto et al. (1998)
Wilson et al. (2000)
Wilson et al. (2000)
Lo et al. (2002)
Lo et al. (2002)
20
21
22
23
24
25
11
11
20
20
Not stated
20
Cleft of lip, alveolus and palate
Maxillary retrusion secondary to cleft lip and palate with medial facial dysplasia
Maxillary hypoplasia
Unilateral cleft lip and palate Maxillary hypoplasia
Maxillary hypoplasia
Aneurysm at the junction of the basilar artery and the right posterior cerebral artery, aneurysm in the left internal carotid artery. Blindness Mydriasis and sixth nerve paresis (right eye). Blindness in the left eye
Blindness. Sixth nerve palsy The same as case # 20
Blindness
Cerebrospinal fluid leak. Superior orbital fissure syndrome
CT and MRI
Cranial CT Angiography
CT
CT of orbit
CT
CT
No evidence of skull base fractures
No evidence of skull base fracture. Subarachnoid haemorrhage in the basal and prepontine cisterns
The same as case # 20
Normal
Comminuted fracture of the posteromedial orbital wall and left pterygoid, bone fragments into the superior orbital fissure, linear fracture along the lateral wall of the sphenoid sinus, extending upward and including the anterior clinoid process Normal
R/L
R
L
L
Not stated
L
Left eye blindness
Third nerve paresis with aberrant regeneration Blindness
Blindness
Blindness
Superior orbital fissure syndrome
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 215
ARTICLE IN PRESS 216 Journal of Cranio-Maxillofacial Surgery
latter case whose orbital haemorrhage was controlled with decompression (cantholysis and drainage), all other cases had to be corrected by balloon occlusion. Four patients had permanent neural deficits. There are 8 reports of blindness (cases # 11, 13, 18, 19, 21, 22, 24 and 25). Of these, four cases also presented with either third (# 11) or sixth (# 19, 22 and 25) nerve palsies, with intracranial vascular aneurysms in one (# 24) or with blindness appearing as an isolated complication in the last three. From this review of the literature it is possible to draw some conclusions regarding the cases of blindness following Le Fort I osteotomy. The data summarized in Table 1 show that different types of complications may appear in one and the same patient and may thus may be linked to a common mechanism. Postoperative computed tomography of the head, orbits or skull base was performed in 17 cases. In 10 of these, the imaging failed to detect fractures of the skull base (cases # 4, 6, 13, 16, 17, 19, 21, 22, 24 and 25). These negative findings led some authors to speculate that the cause of blindness after Le Fort I osteotomy could be related to hypotension and hypoperfusion of the optic nerve (Lo et al., 2002). However, in the other 7 cases CT scans clearly demonstrated atypical fractures involving the sphenoid bone, some with bone fragments close to the (orbital) apex in the superior orbital fissure region (case #11 and 20). The radiological findings of the case described in this paper follow this pattern. As is depicted in Fig. 2, complex fractures could be seen in the skull base and a bone fragment was seen near the orbital apex. However, negative radiological findings do not necessarily exclude a mechanical factor as the mechanism of skull base complications linked to Le Fort I osteotomy because concussion or contrecouptype injuries may occur (Polley, 2002). Here osteotomy of the pterygoid plates might help (Ueki et al., 2004). CONCLUSION Adverse transmission of forces via the sphenoid bone to the base of the skull during separation of the pterygomaxillary junction may explain all the cases reported in the literature (listed in Table 1), including those of blindness with negative radiological findings. Transection of the pterygomaxillary junction is the most critical step in Le Fort osteotomies and must be performed with great care. Surgeons must be aware of skull base complications and avoid any mobilization of the maxilla until being sure that a complete separation of this bone from the pterygoid plates has been achieved. References Bendor-Samuel R, Chen YR, Chen PKT: Unusual complications of the Le Fort I osteotomy. Plast Reconstr Surg 96: 1289–1296, 1995 Brady S, Coutermanche A, Steinbok P: Carotidy artery thrombosis after elective mandibular and maxillary osteotomies. Ann Plast Surg 6: 121, 1981
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: 206–211, 1986 Demas PN, Sotereanos GC: Incidence of nasolacrimal injury and turbinectomy-associated atrophic rhinitis with Le Fort I osteotomies. J Craniomaxfac Surg 17: 116–118, 1989 Girotto JA, Davidson J, Wheatly M, Redett R, Muehlberger T, Robertson B, Zinreich J, Iliff N, Miller N, Manson PN: Blindness as a complication of Le Fort osteotomies: role of atypical fracture patterns and distortion of the optic canal. Plast Reconstr Surg 102: 1409–1421, 1998 Habal MB: A carotid cavernous sinus fistula after maxillary osteotomy. Plast Reconstr Surg 77: 981–985, 1986 Hes J, Man K: Carotid-cavernous sinus fistula following maxillofacial trauma and orthognathic surgery. Int J Oral Maxillofac Surg 17: 295–297, 1988 Lanigan DT, Tumban DE: Carotid-cavernous sinus fistula following Le Fort I osteotomy. J Oral Maxillofac Surg 45: 969–975, 1987 Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: hemorrhage associated with Le Fort I osteotomies. J Oral Maxillofac Surg 48: 561–573, 1990 Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: false aneurysms and arteriovenous fistulas following orthognathic surgery. J Oral Maxillofac Surg 49: 571–575, 1991 Lanigan DT, Romanchuk K, Olson CK: Ophthalmic complications associated with orthognathic surgery. J Oral Maxillofac Surg 51: 480–494, 1993 Lo LJ, Hung KF, Chen YR: Blindness as a complication of the Le Fort I osteotomy for maxillary distraction. Plast Reconstr Surg 109: 688–698, 2002 Melsen B, Ousterhout DK: Anatomy and development of the pterygopalatomaxillary region, studied in relation to Le Fort osteotomies. Ann Plast Surg 10: 16–28, 1987 Newhouse RF, Schow SR, Kraut RA, Price JC: Life-threatening hemorrhage from a Le Fort I osteotomy. J Oral Maxillofac Surg 40: 117–119, 1982 Polley JW: Blindness as complication of Le Fort I osteotomy for maxillary distraction—discussion. Plast Reconstr Surg 109: 699–700, 2002 Reiner S, Willoughby JH: Transient abducens nerve palsy following a Le Fort I maxillary osteotomy: report of a case. J Oral Maxillofac Surg 46: 699–701, 1988 Robinson PP, Hendy CW: Pterygoid plate fractures caused by the Le Fort I osteotomy. Br J Oral Maxillofac Surg 24: 198–202, 1986 Sirikumara M, Sugar AW: Adie’s pupil following Le Fort I maxillary osteotomy. A complication or coincidence? Br J Oral Maxillofac Surg 28: 306–307, 1990 Tomasetti BJ, Broutsas M, Gormley M, Jarret W: Lack of tearing after Le Fort I osteotomy. J Oral Surg 34: 1095–1097, 1976 Ueki K, Nakagawa K, Marukawa K, Yamamoto E: Le Fort I osteotomy using an ultrasonic bone curette to fracture the pterygoid plates. J Cranio Maxillofac Surg 32: 381–386, 2004 Watts PG: Unilateral abducent nerve palsy: a rare complication following a Le Fort I maxillary osteotomy. Br J Oral Maxillofac Surg 22: 212–215, 1984 Wilson M, Maheshwari P, Stokes K, Wheatley M, McLoughlin S, Talbot M, Shults WTMD, Dailey R, Wobig JL: Secondary fractures of Le Fort I osteotomy. Ophthalmic Plast Reconstr Surg 16: 258–270, 2000
Prof. Antonio Augusto V. CRUZ Hospital das Clı´ nicas-Campus Departamento de Oftalmologia, Otorrinolaringologia e Cirurgia de Cabec¸a e Pescoc¸o, Av. Bandeirantes 2900 14049-900, Ribeira˜o Preto, Sa˜o Paulo, Brazil Tel.: +55 16 602862 fax: +55 16 6022860 E-mail: [email protected] Paper received 18 March 2004 Accepted 17 January 2006
Case Report Blindness after Le Fort I osteotomy: A possible complication associated with pterygomaxillary separation Antonio Augusto V. CRUZ1, Antonio Carlos dos SANTOS2 1
Department of Ophthalmology, Otorhinolaryngology and Head and Neck Surgery, (Chair: Prof. M. de Lourdes Veronese Rodrigues); 2Department of Radiology, (Chair: Prof. M. Cesar Foss), School of Medicine of Ribeira˜o Preto, University of Sa˜o Paulo, Brazil
SUMMARY. Introduction: Visual loss after Le Fort I osteotomy is a devastating complication the mechanism of which is not always clear. Methods: A case report of blindness following Le Fort I osteotomy is presented. The literature on the various skull base complications associated with Le Fort I osteotomies is reviewed and the mechanisms of these complications discussed. Conclusion: The radiological findings in this case are similar to those previously reported. They strongly support the hypothesis that an adverse transmission of forces associated with pterygomaxillary separation via the sphenoid bone to the intra- and extracranial portions of the skull base is the main reason for injury to the optic and other cranial nerves as well as to the branches of the carotid artery. r 2006 European Association for Cranio-Maxillofacial Surgery
Keywords: blindness; Le Fort I osteotomy; skull base; complications
INTRODUCTION Le Fort I osteotomy is a standard technique for the surgical management of dentofacial anomalies and also for providing access for many other operations. One of the key steps in this type of osteotomy is pterygomaxillary dysjunction. The transection of this suture is not without risks and can lead to several intra- and extracranial complications (Lanigan et al., 1990, 1991, 1993; Ueki et al., 2004). Visual loss following Le Fort I osteotomy is a rare event, the mechanisms for which are still open to debate. We are aware of only seven such cases (Lanigan et al., 1993; Bendor-Samuel et al., 1995; Girotto et al., 1998; Wilson et al., 2000; Lo et al., 2002). In the present paper another case of blindness after Le Fort I osteotomy is reported and the literature on skull base complications associated with Le Fort I osteotomies is reviewed.
CASE REPORT A 22-year-old female presented with visual loss in the right eye. One week before consultation she had undergone a Le Fort I osteotomy for occlusal correction at another hospital. Immediately postoperatively her right upper and lower eyelids became swollen. The next morning, the patient had been unable to open the right eye and when the upper eyelid was manually lifted, there was no light
Fig. 1 – 22 year old female: Top: Chemosis and esodeviation of the right eye on primary position of gaze. Bottom: Sixth nerve palsy. The patient is trying to abduct the right eye. 210
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 211
perception. Magnetic resonance imaging was performed with inconclusive results. High-dose steroids were initiated but as the patient failed to recover vision she was referred to this hospital. On examination the right upper and lower eyelids were still markedly swollen. There was complete blepharoptosis on the right side. Manual upper eyelid elevation revealed intense chemosis, loss of abduction and pupillary dilatation (Fig. 1). In spite of a normal ocular fundus, there was no light perception in the right eye. A CT scan revealed complex fractures of the pterygoid plates on both sides. On the right side there was a comminuted fracture of the medial and lateral walls of the maxillary sinuses, greater wing of the sphenoid bone and lateral and anterior walls of the sphenoid sinus. The fracture involved the inferior and superior orbital fissures with a bone fragment extending from the superior orbital fissure to the orbital apex (Figs. 2 and 3).
DISCUSSION Pterygomaxillary dysjunction involves separation between the maxillary tuberosity, pyramidal process of the palatine bone, and the pterygoid plates of the
sphenoid bone. The complexity of the sutures between these three bones increases with age and in adults the region is characterized by heavily interdigitated osseous surfaces. These can be disarticulated easily only during childhood (Melsen and Ousterhout, 1987). In adolescents and adults, attempts to transect the pterygomaxillary junction often produce fractures of the pterygoid plates (Robinson and Hendy, 1986). A comprehensive literature review disclosed 25 reports of ophthalmic and neurologic complications associated with Le Fort I osteotomy (Table 1). Sporadic cases of epiphora (Demas and Sotereanos, 1989; Lanigan et al., 1993) and reports of damage to the vessels of the pterygopalatine fossa (Lanigan et al., 1990, 1991) are not listed. Two patients (cases # 19 and 20) described by Girotto et al. (1998) may have also been reported by Wilson et al. (2000) (cases # 22 and 23). Overall, the complications listed in Table 1 can be grouped into four categories: (a) loss of function of the lacrimal gland, (b) cranial nerve palsies, (c) damage to the internal carotid artery, and (d) loss of vision. Four cases of impairment of lacrimal gland function have been reported, one by Tomasetti et al.
Fig. 2 – Computed tomography of the skull base. Bone window with axial (left) and coronal (right) scans: (A) Comminuted fracture of the medial and lateral walls of the maxillary sinuses, greater wing of the sphenoid bone and lateral and anterior walls of the sphenoid sinus. Both inferior fissure (black arrows) and intracranial temporal fossa (white arrows) are involved. (B) Bone fragments can be seen in the inferior orbital fissure (black arrows) and inside the middle cranial fossa (white arrows).
ARTICLE IN PRESS 212 Journal of Cranio-Maxillofacial Surgery
Fig. 3 – Computed tomography of the skull base. Bone window with axial (left) and coronal (right) scans: (A) Bone fragment in the superior orbital fissure (arrow) and (B) bone fragment near the optic canal and fracture involving the anterior clinoid process (arrow).
(1976; case #1) and three by Lanigan et al. (1993; cases # 14, 15 and 16). Anatomically, the preganglionic parasympathetic fibres to the lacrimal gland initially travel with the facial nerve leaving at the geniculate ganglion to reach the sphenopalatine ganglion via the greater superficial petrosal and vidian nerves. The greater superficial petrosal nerve passes through the hiatus of the facial canal to enter the middle cranial fossa running forward below the dura-mater in a groove on the anterior surface of the petrous portion of the temporal bone. It then traverses the foramen lacerum where it is joined by the deep petrosal nerve to form the vidian nerve. The deep petrosal nerve is a sympathetic branch arising from the carotid plexus. The vidian nerve passes forward through the pterygoid or vidian canal to enter the pterygopalatine fossa. Postganglionic fibres join the maxillary nerve and reach the lacrimal gland via the zygomatico-temporal anastomoses with the lacrimal nerve. Any damage to the greater superficial petrosal and vidian nerves, sphenopalatine ganglion or maxillary nerve during its path to the orbit through the inferior orbital fissure can thus impair the lacrimal gland function, provoking a reduction of reflex lacrimal secretion. In all four cases of dry eye secondary to Le
Fort I osteotomy, a full recovery of the lacrimal secretion occurred slowly after surgery (8–24 months) without any serious consequences. Case #14 (Table 1) is interesting because, besides a bilateral reduction of lacrimal function, her left eye was also anaesthetized. As the corneal sensation is transmitted by the first division (ophthalmic) of the trigeminal nerve, it may be postulated that a second injury might have occurred in the region of the superior orbital fissure. Isolated cranial nerve palsies have been reported six times (cases 4, 6, 8, 10, 20, 23; Watts, 1984; Carr and Gilbert, 1986; Reiner and Willoughby, 1988; Sirikumara and Sugar, 1990; Girotto et al., 1998; Wilson et al., 2000). Case # 10 was described as Adie’s pupil, but the association of mydriasis and lack of accommodation could be the expression of a selective third nerve palsy. As already stated, cases # 20 and 23 might be the same due to the similarity between the two histories and clinical findings. Only these two (or this one patient) had a permanent sequel (third nerve paresis with aberrant regeneration), while all others recovered within 2–18 months after surgery. Seven patients had vascular accidents related to the internal carotid artery (cases # 2, 3, 5, 7, 9, 17 and 24) and one patient had an acute orbital haemorrhage from an unknown source (case # 12). Except for this
F
F
F
M
Newhouse et al. (1982)
Watts 1984
Habal (1986)
Carr and Gilbert (1986) Lanigan and Tubman (1987)
3
4
5
6
F
M
F
Reiner and Willoughby (1988)
Hes and Man (1988)
Sirikumara and Sugar (1990)
9
10
M
8
7
M
F
Tomasetti et al. (1976) Brady et al. (1981)
1
2
Sex
Authors
Case
24
19
27
23
17
28
18
32
20
24
Age (years)
Maxillary retrusion and vertical deficiency
Maxillary hypoplasia
Maxillary retrusion
Cleft lip and palate deformity Vertical maxillary excess
Maxillary retrusion
Maxillary retrusion
Vertical maxillary excess
Hemifacial microsomia
Maxillary retrusion
Diagnosis
Table 1 – Skull base complications associated with Le Fort I osteotomy
Mydriasis and accommodation paresis
Carotid cavernous sinus fistula
Sixth nerve palsy
Carotid cavernous sinus fistula
Carotid cavernous sinus fistula Third nerve palsy
Internal carotid artery haemorrhage. Traumatic arteriovenous fistula involving the right internal carotid and internal jugular vein Sixth nerve palsy
Internal carotid thrombosis
Dry eye
Complication
None
Carotid arteriography Cranial CT
CT of cavernous sinus region
Carotid arteriography
Cranial CT
Carotid angiography
CT
Angiography
Arteriography
None
Postop. imaging
Carotid cavernous fistula near the foramen lacerum Comminuted fracture through the body of the sphenoid sinus passing through the sella turcica, with lateral displacement of a fragment onto the medial surface of the right cavernous sinus Carotid cavernous fistula Fracture of the base of the skull near the middle cranial fossa –
Carotid cavernous fistula Normal
Normal
Filling defect at the bifurcation of the right internal carotid extending into the proximal middle cerebral artery. Suggestion of tear of the carotid at the level of its entry into the petrous bone Arteriovenous fistula. Absence of intracranial flow from the right internal carotid artery
–
Postop. image findings
R
R
R
L
R
R
R
R
R
L
Side
Recovery after 18 months
Recovery after balloon occlusion
Recovery after 5 months
Recovery after 7 weeks Recovery after balloon occlusion Recovery after 8 weeks Recovery after balloon occlusion
Left hemiparesis, vagus and hypoglossal nerve palsies
Recovery after 8 months Left hemiparesis
Outcome
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 213
Sex
F
F
F
F
F
F
M
M
M
Authors
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Lanigan et al. (1993)
Bendor-Samuel et al. (1995)
Bendor-Samuel et al. (1995)
Girotto et al. (1998)
Case
11
12
13
14
15
16
17
18
19
Table 1 (continued)
21
30
14
20
39
42
17
16
33
Age (years)
Congenital hemifacial hypertrophy
Maxillary hypoplasia secondary to bilateral complete cleft lip and palate
Maxillary hypoplasia secondary to bilateral complete cleft lip and palate
Vertical maxillary excess Vertical maxillary excess
Vertical maxillary excess, apertognathia Apertognathia
Midfacial retrusion
Malocclusion
Diagnosis
Blindness. Sixth nerve palsy
Blindness
Dry eye, ipsilateral hypoaesthesia, dryness of the turbinates Carotid cavernous sinus fistula. Intraand extra-cavernous carotid artery aneurysm. Third nerve palsy
Dry eye
Bilateral dry eye. Left corneal anaesthesia
Blindness
Orbital haemorrhage with proptosis, increased intraocular pressure
III nerve palsy. Blindness
Complication
Cranial CT and MRI
Cranial CT
Cranial CT Angiography
Cranial CT
None
CT of orbits and skull base CT
None
CT of orbits and head
Postop. imaging
No evidence of basal skull fractures. Subarachnoid haemorrhage. Carotid cavernous sinus fistula. Carotid artery intra- and extra-cavernous aneurysm Extradural intracranial air, discontinuity in the sphenoid bone involving the anterior and posterior tables of the sphenoid sinus Normal
Normal
Fractures of the pterygoid plates and posterior lateral wall of right maxillary sinus –
Normal
Fractures of the pterygoid plates, posterior wall of the maxillary sinus, sphenoid bone, extending through the floor of the middle cranial fossa. A small fragment of bone was close to the optic nerve near the superior orbital fissure –
Postop. image findings
L
R
L
R
R
R/L
L
L
R
Side
Blindness
Low vision (optic nerve atrophy)
Partial recovery after balloon occlusion
Recovery after 14 months Recovery after 13 months
Recovery in the right eye after 2 years
Recovery after lateral cantholysis, orbital drainage and medication for lowering the intraocular pressure Optic atrophy
Recovery of the third nerve palsy . Blindness
Outcome
ARTICLE IN PRESS
214 Journal of Cranio-Maxillofacial Surgery
M
M
M
M
F
F
Girotto et al. (1998)
Girotto et al. (1998)
Wilson et al. (2000)
Wilson et al. (2000)
Lo et al. (2002)
Lo et al. (2002)
20
21
22
23
24
25
11
11
20
20
Not stated
20
Cleft of lip, alveolus and palate
Maxillary retrusion secondary to cleft lip and palate with medial facial dysplasia
Maxillary hypoplasia
Unilateral cleft lip and palate Maxillary hypoplasia
Maxillary hypoplasia
Aneurysm at the junction of the basilar artery and the right posterior cerebral artery, aneurysm in the left internal carotid artery. Blindness Mydriasis and sixth nerve paresis (right eye). Blindness in the left eye
Blindness. Sixth nerve palsy The same as case # 20
Blindness
Cerebrospinal fluid leak. Superior orbital fissure syndrome
CT and MRI
Cranial CT Angiography
CT
CT of orbit
CT
CT
No evidence of skull base fractures
No evidence of skull base fracture. Subarachnoid haemorrhage in the basal and prepontine cisterns
The same as case # 20
Normal
Comminuted fracture of the posteromedial orbital wall and left pterygoid, bone fragments into the superior orbital fissure, linear fracture along the lateral wall of the sphenoid sinus, extending upward and including the anterior clinoid process Normal
R/L
R
L
L
Not stated
L
Left eye blindness
Third nerve paresis with aberrant regeneration Blindness
Blindness
Blindness
Superior orbital fissure syndrome
ARTICLE IN PRESS Blindness after Le Fort I osteotomy 215
ARTICLE IN PRESS 216 Journal of Cranio-Maxillofacial Surgery
latter case whose orbital haemorrhage was controlled with decompression (cantholysis and drainage), all other cases had to be corrected by balloon occlusion. Four patients had permanent neural deficits. There are 8 reports of blindness (cases # 11, 13, 18, 19, 21, 22, 24 and 25). Of these, four cases also presented with either third (# 11) or sixth (# 19, 22 and 25) nerve palsies, with intracranial vascular aneurysms in one (# 24) or with blindness appearing as an isolated complication in the last three. From this review of the literature it is possible to draw some conclusions regarding the cases of blindness following Le Fort I osteotomy. The data summarized in Table 1 show that different types of complications may appear in one and the same patient and may thus may be linked to a common mechanism. Postoperative computed tomography of the head, orbits or skull base was performed in 17 cases. In 10 of these, the imaging failed to detect fractures of the skull base (cases # 4, 6, 13, 16, 17, 19, 21, 22, 24 and 25). These negative findings led some authors to speculate that the cause of blindness after Le Fort I osteotomy could be related to hypotension and hypoperfusion of the optic nerve (Lo et al., 2002). However, in the other 7 cases CT scans clearly demonstrated atypical fractures involving the sphenoid bone, some with bone fragments close to the (orbital) apex in the superior orbital fissure region (case #11 and 20). The radiological findings of the case described in this paper follow this pattern. As is depicted in Fig. 2, complex fractures could be seen in the skull base and a bone fragment was seen near the orbital apex. However, negative radiological findings do not necessarily exclude a mechanical factor as the mechanism of skull base complications linked to Le Fort I osteotomy because concussion or contrecouptype injuries may occur (Polley, 2002). Here osteotomy of the pterygoid plates might help (Ueki et al., 2004). CONCLUSION Adverse transmission of forces via the sphenoid bone to the base of the skull during separation of the pterygomaxillary junction may explain all the cases reported in the literature (listed in Table 1), including those of blindness with negative radiological findings. Transection of the pterygomaxillary junction is the most critical step in Le Fort osteotomies and must be performed with great care. Surgeons must be aware of skull base complications and avoid any mobilization of the maxilla until being sure that a complete separation of this bone from the pterygoid plates has been achieved. References Bendor-Samuel R, Chen YR, Chen PKT: Unusual complications of the Le Fort I osteotomy. Plast Reconstr Surg 96: 1289–1296, 1995 Brady S, Coutermanche A, Steinbok P: Carotidy artery thrombosis after elective mandibular and maxillary osteotomies. Ann Plast Surg 6: 121, 1981
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: 206–211, 1986 Demas PN, Sotereanos GC: Incidence of nasolacrimal injury and turbinectomy-associated atrophic rhinitis with Le Fort I osteotomies. J Craniomaxfac Surg 17: 116–118, 1989 Girotto JA, Davidson J, Wheatly M, Redett R, Muehlberger T, Robertson B, Zinreich J, Iliff N, Miller N, Manson PN: Blindness as a complication of Le Fort osteotomies: role of atypical fracture patterns and distortion of the optic canal. Plast Reconstr Surg 102: 1409–1421, 1998 Habal MB: A carotid cavernous sinus fistula after maxillary osteotomy. Plast Reconstr Surg 77: 981–985, 1986 Hes J, Man K: Carotid-cavernous sinus fistula following maxillofacial trauma and orthognathic surgery. Int J Oral Maxillofac Surg 17: 295–297, 1988 Lanigan DT, Tumban DE: Carotid-cavernous sinus fistula following Le Fort I osteotomy. J Oral Maxillofac Surg 45: 969–975, 1987 Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: hemorrhage associated with Le Fort I osteotomies. J Oral Maxillofac Surg 48: 561–573, 1990 Lanigan DT, Hey JH, West RA: Major vascular complications of orthognathic surgery: false aneurysms and arteriovenous fistulas following orthognathic surgery. J Oral Maxillofac Surg 49: 571–575, 1991 Lanigan DT, Romanchuk K, Olson CK: Ophthalmic complications associated with orthognathic surgery. J Oral Maxillofac Surg 51: 480–494, 1993 Lo LJ, Hung KF, Chen YR: Blindness as a complication of the Le Fort I osteotomy for maxillary distraction. Plast Reconstr Surg 109: 688–698, 2002 Melsen B, Ousterhout DK: Anatomy and development of the pterygopalatomaxillary region, studied in relation to Le Fort osteotomies. Ann Plast Surg 10: 16–28, 1987 Newhouse RF, Schow SR, Kraut RA, Price JC: Life-threatening hemorrhage from a Le Fort I osteotomy. J Oral Maxillofac Surg 40: 117–119, 1982 Polley JW: Blindness as complication of Le Fort I osteotomy for maxillary distraction—discussion. Plast Reconstr Surg 109: 699–700, 2002 Reiner S, Willoughby JH: Transient abducens nerve palsy following a Le Fort I maxillary osteotomy: report of a case. J Oral Maxillofac Surg 46: 699–701, 1988 Robinson PP, Hendy CW: Pterygoid plate fractures caused by the Le Fort I osteotomy. Br J Oral Maxillofac Surg 24: 198–202, 1986 Sirikumara M, Sugar AW: Adie’s pupil following Le Fort I maxillary osteotomy. A complication or coincidence? Br J Oral Maxillofac Surg 28: 306–307, 1990 Tomasetti BJ, Broutsas M, Gormley M, Jarret W: Lack of tearing after Le Fort I osteotomy. J Oral Surg 34: 1095–1097, 1976 Ueki K, Nakagawa K, Marukawa K, Yamamoto E: Le Fort I osteotomy using an ultrasonic bone curette to fracture the pterygoid plates. J Cranio Maxillofac Surg 32: 381–386, 2004 Watts PG: Unilateral abducent nerve palsy: a rare complication following a Le Fort I maxillary osteotomy. Br J Oral Maxillofac Surg 22: 212–215, 1984 Wilson M, Maheshwari P, Stokes K, Wheatley M, McLoughlin S, Talbot M, Shults WTMD, Dailey R, Wobig JL: Secondary fractures of Le Fort I osteotomy. Ophthalmic Plast Reconstr Surg 16: 258–270, 2000
Prof. Antonio Augusto V. CRUZ Hospital das Clı´ nicas-Campus Departamento de Oftalmologia, Otorrinolaringologia e Cirurgia de Cabec¸a e Pescoc¸o, Av. Bandeirantes 2900 14049-900, Ribeira˜o Preto, Sa˜o Paulo, Brazil Tel.: +55 16 602862 fax: +55 16 6022860 E-mail: [email protected] Paper received 18 March 2004 Accepted 17 January 2006