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J Oral Maxillofac Surg

70:1678-1691, 2012

Unusual and Rare Complications of


Orthognathic Surgery: A Literature Review
Ben J. Steel, BDS, MFDS RCSEd,* and Martin R. Cope, FRCS†

An old surgical adage states: if you have not had any that the clinician is fully informed and able to answer
complications, you have not done enough surgery questions, should they arise. The scope of this review
yet. The diverse range of procedures that collectively includes all those orthognathic procedures performed
come under the umbrella term of orthognathic sur- on the mandible or maxilla, with the exclusion of
gery are widely recognized to be safe,1-10 although it distraction techniques and more complex Le Fort II
has been consistently stated that clinicians must re- and III procedures. For the purposes of this article,
main vigilant to the unexpected.11-14 Numerous ex- the term “complication” refers to an unintended con-
amples of unusual or rare complications of such pro- sequence of the surgery that causes harm to the pa-
cedures have been published. The purpose of this tient, occurring either intraoperatively or early or late
literature review is to gather reported complications postoperatively. A few of these complications were
of orthognathic surgery, and provide a discussion not necessarily related to the orthognathic procedure
relating to those classified as unusual or rare. per se but occurred during general anesthesia for that
It is accepted that there may be no clear boundary purpose.
between complications that are more usual versus The relative safety of these procedures has been
unusual. For the purposes of this review, unusual is confirmed by a number of reviews of orthognathic
considered to be a complication that may not nor- surgery patients. A large series of 2,049 patients con-
mally be considered as part of the consent or surgical ducted by Van de Perre et al2 identified 1 patient
planning process. It is accepted that there would be death and 39 patients with primary and 5 with sec-
inherent inaccuracies in attempting to quantify the ondary severe (life-threatening) complications. The
incidence of many of these complications by a review others comprised 2 severe bradycardias, 2 tracheos-
of this kind, except where already published figures tomies, 4 prolonged intubations or reintubations, 4
are available, because publication bias favors the intermaxillary fixation (IMF) releases due to respira-
more unusual. This may lead to some complications tory distress, and 26 major bleeds. The secondary
being under-represented in the literature and the true complications comprised 3 cases of deep vein throm-
incidence not being appreciated. Therefore it is not bosis (heparinization not used) and 2 of aspiration
the intention of this review to provide incidences; atelectasis. A review of 1,294 consecutive patients
rather, our intent is to bring together all reported undergoing orthognathic surgery by Chow et al3 re-
unusual complications in a comprehensive review to ported a complication rate of 9.7%, which included
remind the clinician of the possible unusual risks of 7.4% of patients who had postoperative infections.
this surgery. No serious or rare events attributable to the surgery
During the consent process, it is clearly inappropri- itself were recorded. Panula et al4 reviewed 655 of
ate to routinely discuss all of these rare complications their osteotomy patients, finding only 1 serious com-
with the patient; however, individual circumstances plication (an intraoperative bleed), with altered sen-
may impart relevance to some of them. It is important sation in the inferior alveolar nerve distribution being
the most common (32%). A prospective study of
1,000 patients by Kramer et al5 found the Le Fort I
*Senior House Officer, Oral and Maxillofacial Surgery Depart- operation to be safe, with intraoperative or perioper-
ment, James Cook University Hospital, Middlesbrough, United ative complications of some kind occurring in 6.4% of
Kingdom. cases, including extensive bleeding in 1.1%. In a study
†Consultant, Humberside Maxillofacial Unit, Hull Royal Infir- of 821 patients by Ayoub et al,15 only 12 patients
mary, Hull, United Kingdom. needed surgical intervention for early postoperative
Address correspondence and reprint requests to Dr Steel: Oral complications.
and Maxillofacial Surgery Department, James Cook University Hos-
pital, Middlesbrough TS4 3BW, United Kingdom; e-mail: b_steel_
Materials and Methods
1_the@yahoo.co.uk
© 2012 American Association of Oral and Maxillofacial Surgeons Studies and case reports published up to August
0278-2391/12/7007-0$36.00/0 2010 were located by use of systematic searches in
doi:10.1016/j.joms.2011.05.010 the PubMed and Embase electronic databases, with

1678
STEEL AND COPE 1679

an emphasis on those published after 1980 and Table 1. COMMON COMPLICATIONS OF


limited to the English-language literature. Keywords ORTHOGNATHIC SURGERY
included complications, rare, unusual, orthognathic, os-
teotomy, Le Fort I, sagittal split, ramus, genioplasty, Postoperative nausea and vomiting
Temporary tympanomimetic changes
maxillary, and mandibular. In addition, the databases of Acute infection
individual key journals including the Journal of Oral Chronic infection
and Maxillofacial Surgery, British Journal of Oral and Sinusitis
Maxillofacial Surgery, and Journal of Craniomaxillo- Excessive bleeding
facial Surgery were searched by use of the same terms. Soft-tissue damage
Localized skin burns
The full texts of relevant articles were obtained Tooth injury
and the references checked against those already Loss of pulpal vitality
identified. Periodontal disease
Gingival recession
Nerve exposure
Results Inferior alveolar nerve disturbance
Lingual nerve disturbance
The volume of material published on the complica- Temporary taste disturbance
tions of orthognathic surgery is very large, which Instrument fracture
relates to both more common and more unusual Instrument/screw loss
events. In the same vein, a significant number of case Foreign body
Buccal/lingual plate fracture
reports or case series pertaining to the unusual were Bad split
identified, overall showing a long list of complications Incomplete/undesirable fracturing
of a wide range of forms and severities. A vast major- Malunion
ity of studies located refer to the Le Fort I osteotomy, Condylar resorption
bilateral sagittal split osteotomy (BSSO), or vertical Temporomandibular joint effects
Relapse—skeletal and dental
subsigmoid ramus osteotomy. An overview of the more Screw loosening
common complications is shown in Table 1, with other Instrument fracture
rarer complications listed in Table 2. These are dis- Respiratory difficulty
cussed in detail in the main text. Neck pain
The unusual and rare complications identified by Malocclusion
the search process will now be explored in more Steel and Cope. Complications of Orthognathic Surgery. J Oral
detail. Maxillofac Surg 2012.

Neurologic Complications
tion of the inferior alveolar nerve has been described
Neurosensory disturbance is a well-recognized by Vaughan Thomas and Cronin23 after a BSSO pro-
complication of orthognathic surgery. Effects on the cedure, which required treatment with a spinal cord
inferior alveolar nerve are common after mandibular stimulator and then a right motor cortex stimulator.
procedures.1,16-18 The effect of the BSSO on this nerve Reports of injuries to other nerves appear to be
was subject to a systematic review by Collela et al.16 restricted mainly to the facial nerve, mainly during
This review pooled data from 7 eligible studies and mandibular procedures. A retrospective study of
found the incidence of objective neurosensory im- 1,747 BSSO patients by de Vries et al24 found an
pairment to be 63.3% 7 days postoperatively, 49.2% incidence of cranial nerve VII palsy of 0.26% per side
after 14 days, 42.5% after 1 month, 33% after 6 (total of 9 patients). Almost 95% of the osteotomies
months, and 12.8% after 1 year. Surprisingly, the in- included were setbacks, representing a much higher
cidence of lingual nerve damage meanwhile has been risk of this complication when compared with man-
quoted in various reports as 9.3%,17 18%,19 and dibular advancements. This is in agreement with
19.4%20 after sagittal split osteotomy, although these other published studies, because a majority of reports
data were collected through patient questionnaires. are related to setbacks.24-26 A review by Choi et al27 of
The true incidence of sensory disturbance to this 3,105 patients who underwent a BSSO at a hospital in
nerve, as tested objectively, is believed to be much Taiwan reported an incidence of facial nerve palsy of
lower. Hegtvedt and Zuniga21 reported an isolated 0.1%, representing the 6 patients who had unilateral
case caused by direct nerve injury by the fixation facial weakness. The conclusion from that review was
plate placed on the superior aspect of the mandible. that complete recovery is expected in most cases (5
An increase in tactile threshold on the palate and of 6). A review by Lanigan and Hohn28 found that a
maxillary gingivae after Le Fort I osteotomy has been majority of these palsies recover within a period of a
noted in 64% of patients.22 Allodynia in the distribu- few weeks to a year after injury, although 3 of the 9
1680 COMPLICATIONS OF ORTHOGNATHIC SURGERY

Table 2. SUMMARY OF UNUSUAL/RARE


nerve ischemia through reflex sympathetic vaso-
COMPLICATIONS OF ORTHOGNATHIC SURGERY spasm, fracture and posterior displacement of the
styloid process, and compression by postoperative
Injury to cranial nerves II, III, IV, V, VI, VII, VIII, X, XI, edema.26,29,31-33 This complication has also been re-
and XII
Ophthalmoplegia ported after intraoral subcondylar ramus osteotomy.34
Neurosensory deafness The only report of facial nerve effects related to max-
Secretomotor rhinopathy illary surgery is a single report after combined Le Fort
Frey syndrome I osteotomy and BSSO.31 In this case it was thought
Altered tear secretion likely that the cause of the palsy was related to the
Hemolacria
Blindness mandibular procedure, through the pressure pack
Adie pupil used on the medial aspect of the ascending ramus
Retrobulbar hemorrhage pressuring the facial nerve, rather than the maxillary
Brain abscess procedure. The rarity of facial nerve damage is at-
Actinomycosis tested to by the lack of any instances located in a
Bone graft donor-site infection
Osteonecrosis of maxilla/mandible review of 2,049 patients by Van de Perre et al.2
Avulsion of maxilla Frey syndrome was reported by Guerissi and Stoy-
Condylar dislocation anoff35 presenting in the cheek of a patient 6 months
Condylar resorption (progressive, idiopathic, condylar after Le Fort I osteotomy by use of the Obwegeser
atrophy) technique. This was postulated to be due to aberrant
Vomero-sphenoidal disarticulation
CSF leak regeneration of secretomotor fibers from the auricu-
Cerebrovascular accident (stroke, subarachnoid lotemporal nerve entering the long buccal nerve as a
hemorrhage) result of direct surgical trauma to the former.
Severe hemorrhage Bilateral hypoesthesia in the dermatome of the my-
Arteriovenous fistula/false aneurysm/carotid-cavernous lohyoid nerve has been reported after genioplasty,36
sinus fistula
Cavernous sinus thrombosis with direct trauma from the bone saws used sug-
Malignant hyperpyrexia/herniation of tube cuff/tube gested as a mechanism. Normal sensation returned in
sectioning the reported case within 6 months.
Conversion disorder Palsy of cranial nerves X, XI, and XII was described
Severe clinical depression by Baddour et al37 in a patient who also had a life-
Acute pulmonary edema/apnea
Pneumomediastinum/pneumothorax threatening intraoperative bleed. It was thought that
Surgical ciliated cyst these palsies were caused by either the maxillary
Traumatic neuroma downfracture or the insertion of pressure packs to
Dysphagia control bleeding. Newhouse et al38 also described
Compartment syndrome palsies of cranial nerves X and XII in a 32-year-old
Perforated lateral nasal wall/septum
Oroantral/oronasal fistula female patient who underwent a Le Fort I osteotomy.
Loss of orthodontic bracket into airway There was a significant intraoperative bleed, and fur-
Severe eustachian tube malfunction ther surgery was undertaken to repair an arterio-
Ischemic finger injury venous fistula between the internal carotid artery and
Death internal jugular vein close to the base of the skull.
Steel and Cope. Complications of Orthognathic Surgery. J Oral Postulated causes for these palsies included trauma
Maxillofac Surg 2012. during maxillary downfracture, during placement of
pressure packs, or during later exposure for treatment
of the fistula.
patients in the study of de Vries et al24 showed in- Traumatic neuroma of the inferior alveolar nerve
complete recovery. Sammartino et al29 concluded has been described in patients who have had mandib-
that a majority of such injuries are transient, and ular osteotomies.39,40 One was found 11 years after a
reported 2 cases with delayed onset and spontaneous BSSO was performed: an incidental finding in the
recovery. Lanigan and Hohn reported 2 cases related ascending ramus on a side that exhibited complete
to mandibular advancement, as did Macintosh.30 In 1 mental anesthesia.40
case occult high-level subcondylar fracture reportedly Stroke was reported by Newhouse et al38 in a pa-
allowed the condylar neck to be positioned more tient who underwent a Le Fort I osteotomy. A signif-
posteriorly, thus applying traction to the main trunk icant intraoperative bleed occurred when the sepa-
of the nerve, and in the other, traction on the nerve rated maxilla was manipulated downward. Control
was thought to have been caused by placement of a was effected with pressure packs and wiring of the
pressure pack in the retromolar area.28 Other possible maxilla, although the next day during helicopter
causes include direct trauma by retractor placement, transfer to another unit for angiography, the nasotra-
STEEL AND COPE 1681

cheal tube became disconnected, leading to cyanosis tracheal intubation. Reversal to preoperative values
and respiratory distress. At this point, it was noted was typical.
that the patient had a left hemiparesis. Angiography
showed a traumatic arteriovenous fistula between the
internal carotid artery and internal jugular vein on the Ophthalmic Complications
right side. The patient subsequently underwent ex-
ploration of this area and obliteration of the right Lack of tearing has been reported after Le Fort I
jugular foramen with a muscle graft. It is reported that osteotomy.8,45,46 Various explanations have been pro-
the patient retained a dense left hemiparesis with posed, including damage to the greater petrosal or
some improvement and also had cranial nerve X and vidian nerves, which could interrupt parasympathetic
cranial nerve XII palsies. It was suggested that with supply to the lacrimal gland.46 In the case reported by
Tomasetti et al,45 recovery was complete by 8 months
pterygomaxillary dysjunction, the right pterygoid
postoperatively. Conversely, excessive tearing has
complex was also detached, forcing a sharp piece of
been reported,38,45,47 in some cases identified as re-
bone posteriorly, thus lacerating the vessels and caus-
lating to nasolacrimal duct damage.45,48 Hemolacria
ing the vascular injury. Another case of left-sided
(bleeding from the lacrimal puncta) was reported by
hemiparesis was reported by Brady et al41 after com-
Humber et al49 in 2 patients, both after Le Fort I
bined Le Fort I osteotomy and BSSO, in which internal
advancements. This was thought to be a result of
carotid artery thrombosis occurred, thought to result
minor surgical trauma to the vessels in the nasal wall
from a neck flexion–induced intimal tear.
accompanied by a small tear in the nasolacrimal duct.
Subarachnoid hemorrhage was reported by Bendor-
The bleeding presented soon after surgery in 1 pa-
Samuel et al42 in a 14-year-old male patient undergo-
tient, which was treated with nasal packing, and 8
ing Le Fort I osteotomy. A presumptive diagnosis of
days postoperatively in another, which was treated
cavernous sinus thrombosis, with cranial nerve III
conservatively.
palsy, was made on the second postoperative day. On
A review by Newlands et al50 identified 5 cases of abdu-
the eighth postoperative day, the patient had a sud- cens palsy,51-54 3 cases of oculomotor palsy,46,54,55 and 1
den massive headache develop and a computed to- case where both palsies were present together.56 Other
mography (CT) scan showed subarachnoid hemor- cases of abducens palsy have been reported.52,54,57-59
rhage, with a carotico-cavernous fistula and internal Most presented immediately after surgery, although
carotid artery aneurysm visible on angiography. The cases have been reported up to 5 days postoperatively.59
postulated cause was a possible skull base fracture, Bendor-Samuel et al42 reported a case of oculomo-
although none was visible on the CT scans. tor palsy after a Le Fort I osteotomy in a cleft patient
Secretomotor rhinopathy was reported by Marais with CT evidence of cavernous sinus thrombosis and
and Brookes.43 This seems to be the only reported subarachnoid hemorrhage. Recovery was almost com-
case of this complication. A 33-year-old skeletal Class plete at 12 months, with residual ophthalmoplegia on
II woman underwent a Le Fort I procedure, and clear upward gaze in the affected eye. The other reported
rhinorrhea and lacrimal hypersecretion developed at cases of oculomotor palsy have been attributed to
3 days postoperatively. Investigations ruled out a ce- transmitted force or hematoma.46,55
rebrospinal fluid (CSF) leak, hypersensitivity, and in- Complete isolated abducens palsy was reported by
fection, with bulky inferior turbinates the only struc- Hanu-Cernat and Hall59 occurring 5 days after a Le
tural feature of note. Resolution occurred with Fort I osteotomy. This was unilateral on the right side,
regular use of an anticholinergic inhaler, although the with CT showing a fracture of the right pterygoid
lacrimal hypersecretion persisted. The cause was plate and blood in the right side of the sphenoid
thought to have related to sphenopalatine ganglion sinus. Over a period of 6 weeks, with no intervention,
dysfunction by a local hematoma or its fibrous orga- a complete recovery was made. It was postulated that
nization, shifting the autonomic balance toward a the cause may have been compression or distraction
parasympathetic predominance. of cranial nerve VI due to a suspected hairline fracture
Reduced hearing after Le Fort I osteotomy in cleft in the sphenoid sinus extending toward the orbit and
patients was reported by Gotzfried and Thumfart.44 causing temporary displacement. Watts57 described a
Twenty-six cleft lip and palate patients had preoper- case of an 18-year-old female patient in whom a right-
ative and postoperative audiometric testing, and in sided abducens nerve palsy developed on the first post-
nine who had normal preoperative hearing, a deteri- operative day after a standard Le Fort I osteotomy,
oration was observed; conversely, others had an im- which took 7 weeks to resolve. Postulated causes of
provement in hearing. The deteriorations were likely abducens palsy in other Le Fort I cases include direct
caused by edema and/or hematoma formation in the trauma to the medial aspect of the cavernous sinus51 and
eustachian tubes resulting from the surgery or naso- a pre-existing carotid aneurysm.42
1682 COMPLICATIONS OF ORTHOGNATHIC SURGERY

The review of Newlands et al50 also presented a cedure. Complete blindness of the right eye was
case of concurrent oculomotor and abducens palsy. noted postoperatively with a Marcus Gunn pupil. CT
In a 33-year-old female patient who underwent Le showed evidence of swelling of the optic nerve and
Fort I maxillary impaction, a complete left-sided pto- skull base fracture. One case reported by Cheng et
sis and almost complete left-sided ophthalmoplegia al63 in 2007 occurred in a patient with hypoplasia of
developed, with sparing of pupillary reflexes, over the left internal carotid artery. Light perception and
the first 24 hours postoperatively. Oculomotor func- pupillary reflexes were absent immediately after Le
tion recovered by 1 week and abducens by 10 weeks. Fort I and intraoral vertical subsigmoid ramus osteot-
Fracture at the superior orbital fissure was thought to omies. Insomnia and anxiety with uncooperative and
have been causative. This appears to be only the regressive behavior developed in this patient. Ex-
second report of such palsies occurring simultane- trapyramidal motor effects also occurred. Magnetic
ously after this surgery. resonance imaging showed hypoxia of the basal gan-
Nine cases of blindness can be found in the litera- glia, and no signal from the internal carotid artery
ture.42,45,58,60-63 Lanigan et al46 conducted a survey of could be detected. A diagnosis of hypoxic damage to
oral and maxillofacial surgeons (OMSs) in North the basal ganglia and optic nerve was made, which
America, receiving nearly 800 responses. Among resolved gradually.
these, 2 cases of blindness were identified. One in- Of the 9 reported cases of blindness, 5 were of
volved a 33-year-old female patient who, the morning unknown cause, whereas 1 was due to arterial aneu-
after a Le Fort I osteotomy and BSSO, complained of rysm, 2 were attributed to propagation of the ptery-
right-sided blindness. She was found to have no light gomaxillary dysjunction fracture through the skull
perception in that eye, a fixed dilated pupil with base (as investigated by Girotto et al60), and 1 was due
reduced consensual light reflex, and partial cranial to hypoperfusion of the optic nerve.45 There was no
nerve III palsy. It was thought that the cause was a recovery in 3 cases, light perception returned in 3,
fractured skull base related to high pterygoid dysjunc- hand movements were visible in 1, fingers could be
tion due to thick bone from a previous osteotomy. CT counted in 1, and visual acuity showed much im-
showed a bone fragment either very close to or in the provement in 1.
canalicular optic nerve. High-dose steroids failed to Adie pupil was reported on the first postoperative
give a resolution, and the blindness persisted. An- day after a Le Fort I procedure by Sirikumara and
other patient had vision limited to counting fingers Sugar64; however, it was not clear whether there was
after Le Fort I osteotomy and genioplasty. A group of any causal link or whether it was just coincidence.
surgeons in Taiwan described 2 cases of blindness, The review of Lanigan et al46 described a case of
both related to Le Fort I osteotomy.58 An 11-year-old retrobulbar hemorrhage whose signs were noticed
girl with bilateral cleft lip and palate and midface when the surgical drapes were removed at the end of
hypoplasia underwent Le Fort I osteotomy with fitting a Le Fort I advancement procedure. An ophthalmo-
of an external distractor device. On the second post- logic opinion was sought and lateral canthotomy per-
operative day, the patient complained of total right- formed. Li et al65 published a further case of acute
sided blindness. There was no light perception, and orbital compartment syndrome due to retrobulbar
the direct pupillary reflex was absent. The clinical hemorrhage in a 34-year-old woman undergoing bilat-
impression was of right traumatic optic neuropathy. eral posterior segmental maxillary osteotomies. Eyelid
CT showed subarachnoid hemorrhage in the basal edema and proptosis of the left eye were noted upon
and subpontine cisterns. Angiography showed a rup- removal of the drapes at the end of the operation, and
tured aneurysm at the junction of the basilar and right despite megadose steroid therapy, mannitol, and lat-
posterior cerebral arteries and another in the ophthal- eral canthotomy, the patient reportedly never re-
mic segment of the internal carotid artery. A traumatic gained useful acuity in the eye.
origin for these aneurysms could not be confirmed,
but they were thought to be responsible for the blind-
Infective Complications
ness in this patient, who had only light perception 3
months later. The second case involved a 12-year-old Baker et al66 reported a case of brain abscess in a
boy with a left-sided cleft lip and palate who had a Le 15-year-old male patient who had a Le Fort I osteot-
Fort I osteotomy. He complained of vision loss in his omy, BSSO, and genioplasty. The medical history of
left eye. He also had a right abducens palsy, although the patient is not mentioned besides hemifacial mi-
CT and magnetic resonance imaging failed to offer crosomia, but he was diagnosed with a right frontal
any explanation. Recovery was limited to seeing hand lobe abscess about 4 weeks postoperatively, having
movements at 50 cm after 2 years. Bendor-Samuel et shown acute mental status deterioration after chronic
al42 reported blindness with partial recovery in a low-grade pyrexia. Craniotomy and intravenous anti-
30-year-old male patient after a second Le Fort I pro- biotic administration successfully treated the infec-
STEEL AND COPE 1683

tion. Purported etiologies were spread of bacteria majority of the alveolar ridge and teeth had to be
from the paranasal air sinuses, especially the eth- removed, with treatment involving hyperbaric oxy-
moidal air cells, or retrograde spread of bacteria gen therapy, iliac crest graft, and implant-supported
through seeding into the pterygoid venous plexus. prosthodontics.
Iliac abscess occurring 4 years after orthognathic sur-
gery for which an iliac graft was harvested has been AVULSION OF MAXILLA
described by De Riu et al.67 A 4 ⫻ 3– cm monocortical Bendor-Samuel et al42 described an instance of avul-
corticocancellous block had been taken from the inner sion of the left hemi-maxilla and palate in a 20-year-old
table of the left iliac crest to provide a graft for a max- male patient with repaired bilateral cleft lip and palate
illary advancement. The abscess was thought to have undergoing intended Le Fort I osteotomy with iliac
been due to a foreign-body granuloma related to bone crest bone graft. Overzealous undermining of the mu-
wax placed in the area. This appears to be the only case cosa through what was presumably a horseshoe inci-
of this complication being reported. sion allowed the entire left bony segment complete
Six cases of actinomycosis after orthognathic sur- with dentoalveolar segment and attached gingiva to
gery can be identified.68-70 Ozaki et al68 reported a be completely detached and avulsed. After stripping
case of actinomycosis occurring in the left subman- of this attached gingiva, the segment was replaced in
dibular area, which presented as a 2 ⫻ 3– cm swelling the pre-osteotomy position, with the right side being
6 months after BSSO with advancement. The infection advanced as planned. The avulsed segment survived,
resolved with 2 months of antibiotic therapy. This and further surgery was not undertaken.
pattern of infection conforms to general principles of
cervicofacial actinomycosis because surgery is a OSTEONECROSIS OF MANDIBLE
known predisposing factor. Six case reports were located by a review of man-
dibular aseptic necrosis in 1990.75 Two of these were
published as case histories, one after BSSO and the
Skeletal/Bony Complications other after BSSO with genioplasty. In the former the
patient was a 38-year-old woman with no predispos-
OSTEONECROSIS OF MAXILLA ing medical factors. A technetium bone scan showed
Various anecdotal reports exist, but contrary to a large area on 1 side from the premolars posteriorly
popular myth, a published report of necrosis of a including all of the ramus except the coronoid and
whole maxilla does not appear to exist in the English- condylar neck areas to be avascular. Hyperbaric oxy-
language language. A survey of OMSs in the United gen treatment and antibiotic therapy led to only a
States by Sher,71 from whom 35 responses were re- 5-mm piece of bone eventually being sequestered.
ceived, identified 1 case of “sloughed maxilla (entire)” The cause was thought to have been overzealous
although no details are given. There are various pub- stripping of the pterygomasseteric sling. In the latter
lished cases of partial necrosis. A review by Lanigan et case a 14-mm advancement of the genial fragment
al72 identified 51 such cases, of which 36 were de- was undertaken while still attached to its lingual mus-
tailed in the article. The extent of the necrosis ranged culature. During removal of the fixation wires at a
from just the maxillary central incisor pulps to the later date, the whole of this piece of bone was
whole of the alveolar ridge and all of the premaxilla. avulsed. It was replaced and effectively served as a
Various other patterns were seen between these ex- free bone graft. The size of the advancement was said
tremes, including just the premolar segment and the to have led to the loss of the muscular attachments. A
alveolar ridge on 1 side only. Pedeira et al73 reported further case restricted to the anterior mandible was
a case of maxillary aseptic necrosis in a middle-aged reported in 1972.76
female smoker who underwent a Le Fort I osteotomy Dislocation of the condyles was reported by Wein-
and BSSO, which was successfully treated with hyper- berg et al77 as a case report of an 18-year-old woman
baric oxygen. The exact extent of the necrosis is not who presented after a fall 1 week after bilateral ex-
described, but the mucosa overlying the maxilla was traoral subcondylar osteotomies. The patient’s symp-
described as ischemic, with generalized maxillary gin- toms are not described, but radiographs showed ante-
gival recession and an ulcer on the left side of the rior dislocation of the right condyle despite the dental
hard palate on the seventh postoperative day, with occlusion reportedly being the same as that of the im-
complete resolution by the 22nd day. mediate postoperative period. This condylar position
Singh et al74 reported a case of near-total necrosis persisted, although extensive remodeling was noted. It
of the maxillary alveolus after Le Fort I osteotomy and was thought that this dislocation may have been intro-
rib graft augmentation, which was seen and treated duced in an iatrogenic manner during surgery.
some 8 years after the procedure, which had been Condylar resorption is a known uncommon com-
undertaken by another specialty. It is said that the plication of orthognathic surgery, leading to relapse.
1684 COMPLICATIONS OF ORTHOGNATHIC SURGERY

The terminology is confusing, with the condition var- arm movement after Le Fort I osteotomy and BSSO,
iously described as progressive or idiopathic condylar developing into hemiparesis. The cause was thought
resorption or as condylar atrophy. The term “idio- to have been an intimal tear, which may have oc-
pathic” does not apply in this case because surgery curred because of neck hyperextension and lateral
can be identified as a precipitating factor. Differenti- flexion.91 A similar presentation was noted by Sanni et
ation between these separate terms is unclear. The al91 after BSSO, which may have been due to a blunt
condition is well-represented in the literature, but few blow from a channel retractor placed lingual to the
incidence figures relating to after orthognathic sur- mandible or indirect trauma from malleting during
gery are available. Scheerlinck et al78 reported an the sagittal split. A good review of the possible
incidence of 7.5% after BSSO in 106 patients. A larger mechanisms of injury to the internal carotid artery
sample of 222 BSSO patients studied by Bortslap et in relation to orthognathic surgery is provided by
al,79 using rigid diagnostic criteria, quoted 4% at 2 Baddour et al.90
years postoperatively. All of the patients in the resorp- Numerous reviews of the complications of orthog-
tion group showed clinically measurable relapse. A nathic surgery have concluded that serious bleeds are
review of previously published studies by Gill et al80 rare in these patients.2,4,5 A prospective study of 35 Le
identified risk factors for this, comprising female gender, Fort I patients by O’Regan and Bharadwaj92 found no
mandibular retrognathia associated with an increased instances of serious bleeding. A survey of OMSs in the
maxillary-mandibular plane angle, pre-existing condylar United States by Lanigan et al93 identified 18 cases of
resorption, and surgery including posterior displace- serious intraoperative bleeding and 21 cases of post-
ment of the condyles or upward and forward rotation of operative bleeding among around 800 responses. All
the mandible. Purported causes of this complication are were related to Le Fort I procedures, and a majority
remodeling of the condyle-fossa complex in response to were said to be from the descending palatine and
altered loading patterns or compromise of the temporo-
sphenopalatine arteries, with fewer from the ptery-
mandibular joint vasculature inducing avascular necro-
goid venous plexus. All of these patients required
sis, thus leading to condylar resorption.81 Cases de-
transfusions, and all of the postoperative bleeds oc-
scribed as condylar atrophy have been reported,82-84
curred within 2 weeks, except 1 that occurred after 5
including 1 associated with temporomandibular joint
weeks and was brought on by heavy lifting. The
osteoarthrosis after bimaxillary surgery.85 It may be a
sphenopalatine and maxillary arteries are thought to
difficult condition to manage, with various approaches
be particularly important sources of significant arte-
described comprising nonsurgical treatment with
rial bleeding.
splints, orthodontics, or restorative dentistry or repeat
Baddour et al37 reported a case of a life-threatening
surgical procedures, including joint replacement.86
CSF leak after orthognathic surgery is a very rare intraoperative bleed localized only to the right pos-
event, with only 2 cases reported in the literature.87,88 terior region that occurred after pterygoid plate sep-
Gruber et al87 reported a case in a 19-year-old woman aration and did not respond to external carotid artery
who had Le Fort I impaction and BSSO and showed a ligation. The patient in the same case also showed
CSF leak from the nostril at 3 days postoperatively, palsies of cranial nerves X through XII. Figures per-
which resolved after placement of a lumbar drain. taining to significant bleeding during or after BSSO
Vomero-sphenoidal disarticulation has been re- have been quoted at 0.38%94 and 1.2%.95 At least 13
ported once, by Smith and Heggie.89 In a 20-year-old cases of serious bleeding related to a vertical subsig-
healthy female patient undergoing bimaxillary surgery moid osteotomy have been reported,96,97 and 2 for
to correct a Class III skeletal relationship, the vomer genioplasties, including 1 where an ooze from that
remained articulated with the maxillae after downfrac- site led to floor-of-mouth hematoma and tongue ele-
ture. Subsequently, during use of rongeurs to reduce an vation, necessitating tracheostomy.97
irregular piece of nasal septum, the vomer was unex- Severe epistaxis is a distinct clinical presentation of
pectedly delivered. It was not replaced, and no adverse what may be the same cause of bleeding as the other
consequences were reported, with the cause of the hemorrhages described previously and is known to be
complication suggested as slippage of the osteotome rare after Le Fort I osteotomy, Lanigan and West97
during sectioning of the cartilaginous septum. quoted an incidence of 0.75%. Solomons and Blumgart98
reported the case of a 20-year-old male patient in whom
2 significant epistaxes developed, one 4 weeks postop-
Bleeding Complications eratively and the second about 10 weeks postopera-
Various injuries to the internal carotid arteries have tively. The latter episode led to life-threatening blood
been reported in the literature: false aneurysms, arte- loss and hypovolemic shock and was found to be caused
riovenous fistulae, hemorrhage, and thrombosis.90 by a false aneurysm of the left internal maxillary artery,
One case of thrombosis presented as decreased left which was then successfully embolized. Other reported
STEEL AND COPE 1685

cases of postoperative epistaxis have been caused by and hyperthermia upon exposure to volatile anes-
similar vascular phenomena.99 thetic agents or succinylcholine chloride. It is a seri-
A review in 1991 examined the vascular complica- ous condition, requiring aggressive management. In
tions of orthognathic surgery.99 Three cases of false the case reported by Monaghan and Hindle,118 sur-
aneurysm were described. All occurred after Le Fort I gery was terminated 70 minutes into the operation,
osteotomy, presenting between 6 days and 6 weeks and malignant hyperpyrexia diagnosed by a combina-
postoperatively with epistaxis; they were massive in 2 tion of tachycardia of 160 beats/min, cyanosed ve-
cases. Two were thought to be of the sphenopalatine nous ooze, and noticeably warm skin. Another case
artery and one of the maxillary artery itself. A surgical was reported by Laureano et al.120
trauma etiology is implicated by partial occlusion and Herniation of the airway tube cuff leading to occlu-
extravasation of an artery, forming a hematoma that sion of the tube lumen has been reported during an
then organizes and undergoes endothelialization. A osteotomy procedure, although the operation was
case of post-osteotomy arteriovenous fistula, in the not required to be terminated.121
form of carotico-cavernous fistula, was also described. Sectioning of the endotracheal tube has been re-
Its signs were noted in the recovery room immedi- ported,122,123 an event obviously capable of produc-
ately after a Le Fort I advancement. The fistula was ing severe intraoperative ventilatory difficulties. Pagar
successfully embolized, but its possible causes were et al122 reported 2 cases of such an event, during
not alluded to. Other authors have reported false which the tubes to the cuff and the endotracheal
aneurysms61,100 (including after a mandibular proce- tubes themselves were incompletely cut, although
dure101), carotico-cavernous fistulae,102-104 and other not to the extent that reintubation was required.
arteriovenous fistulae.105-108 Albernaz and Tomsick109 A case of contact granuloma of the vocal cords was
reported 2 cases of arteriovenous fistulae of the max- described by Chua et al,124 which was related to
illary artery after Le Fort I osteotomies. In both cases pressure necrosis of the mucosa overlying the vocal
the fistulae were successfully embolized, leading to processes of the arytenoid cartilages caused by an
complete cessation of symptoms. The cause was be- endotracheal tube in situ for 4 hours during an or-
lieved likely to have been untoward fractures from thognathic procedure. There is only 1 other report of
pterygomaxillary dysjunction extending to the ptery- this complication, which is itself rare, during orthog-
gomandibular fossa. A false aneurysm of the facial nathic procedures.125 Bilateral nodules on the aryte-
artery was reported by Pappa et al,110 presenting with noids in 1 patient were diagnosed as contact granu-
a severe bleed 1 week after BSSO, and another case by lomas after a 3.5-hour intubation for an orthognathic
Madani et al111 involving trauma 4 weeks after a Le procedure.126
Fort I osteotomy. Hemmig et al112 reported a patient Acute intraoperative pulmonary edema in a 16-year-
with a ruptured pseudoaneurysm of the sphenopala- old female patient undergoing BSSO was reported by
tine artery, presenting as extreme recurrent epistaxis. Kademani et al.127 Induction with propofol was
As well as these cases, other instances of abnormal achieved after diazepam, vecuronium, cefazolin, and
vascular lesions occurring postoperatively and suc- dexamethasone were administered. Ten minutes into
cessfully embolized have been reported.113 Silva et the procedure, acute hypertension developed, later
al114 provided a good review of pseudoaneurysms followed by pink frothy exudate within the endotra-
arising in relation to orthognathic surgery. Cavernous cheal tube. An intraoperative chest radiograph showed
sinus thrombosis has also been reported after orthog- fulminant pulmonary edema, and an electrocardiogram
nathic surgery.115 showed sinus tachycardia. Pharmacologic attempts
Deep vein thrombosis is known to be rare among were made to resolve this, and normotension was
orthognathic surgery patients, with an incidence of re-established over the next 30 minutes. A full recov-
0.00035% published.116 In a review of 129 such pa- ery was made. It was suggested that the initial hyper-
tients treated in Liverpool, 2 cases of deep vein throm- tension was related to adrenaline in the local anes-
bosis were identified, neither of which progressed to thetic, with the pulmonary edema precipitated by an
pulmonary embolism.117 intravenous infusion of esmolol given in an attempt to
reduce the blood pressure. Such an event occurring
under anesthesia is very rare. Acute postoperative
Anesthetic Complications
dyspnea was reported by Aziz et al128 in an 18-year-old
Malignant hyperpyrexia has been reported during a female patient who had undergone a Le Fort I and
segmental osteotomy procedure.118 It is known that vertical subsigmoid ramus osteotomy (VSRO), which
dental procedures can act as a trigger for such an presented immediately postoperatively. The mecha-
event,119 which involves an abnormality of muscle nism was thought to have been right middle and
metabolism, of genetic origin, manifesting as wide- lower lobe atelectasis due to obstruction by aspirated
spread skeletal muscle contraction, hypercatabolism, material.
1686 COMPLICATIONS OF ORTHOGNATHIC SURGERY

Apnea of 9 hours’ duration was reported by O’Ryan true clinical depression; its incidence, however, ap-
and Ebker129 in a healthy female patient after an pears to be underappreciated. A study by Kiyak et
orthognathic procedure, which was found to be due al142 into the emotional impact of orthognathic sur-
to a qualitative and quantitative defect in the enzyme gery showed significantly higher scores for tension
cholinesterase, of genetic origin. Previous general an- and fatigue immediately after surgery than nonsurgi-
esthetics had been uneventful. cal controls and raised anger-hostility that took up to
Pneumomediastinum and pneumothorax have been 5 months to decline. Postsurgical discomfort, pain,
reported rarely as complications.130-135 Edwards et al130 and neurologic disturbance were found to correlate
detailed 2 cases occurring after orthognathic surgery. with a postsurgical altered emotional state.
Both involved young women (aged 16 and 21 years)
having Le Fort I osteotomies (1 also had a BSSO and
Other Complications
genioplasty). In the first patient respiratory distress de-
veloped 14 hours postoperatively, and investigations Death is recognized as an exceedingly rare compli-
showed minimal collapse of the left lung. Alveolar rup- cation relating to orthognathic surgery. Of numerous
ture caused by high intra-alveolar pressure resulting large case series published, only 1 death has been
from mucus plugs and ventilation with an Ambubag described. Van de Perre et al,2 from a series of 2,049
(Ambu A/S, Baltorpbakken, Denmark) was suggested to patients, reported the death of a 17-year-old male
be explanatory. The second case presented similarly, patient who was described as being slightly mentally
with a left apical pneumothorax and a degree of subcu- retarded. He underwent a staged bimaxillary osteot-
taneous emphysema, which has been reported else- omy to address gross vertical and transverse maxillary
where.136,137 In both cases this resolved spontaneously. hyperplasia. The maxillary procedure was completed
Complete left lung collapse due to spontaneous pneu- without any problem, followed by BSSO with genio-
mothorax was reported by Goodson et al,134 and bilat- plasty 3 months later. Six hours postoperatively, a
eral pneumothorax and pneumomediastinum after pos- cardiac arrest developed in the patient, and attempts
itive pressure ventilation were reported in 2010.135 A to resuscitate him failed. The cause was thought to
case of mediastinal emphysema after Le Fort I osteotomy have been a pre-existing cardiomyopathy. Another
was reported in 1986.133 In a literature review on the death was reported by Waack143 as part of a series of
subject, McKenzie and Rosenberg137 located 2 cases of 63 consecutive Le Fort I patients. A healthy 15-year-
surgical subcutaneous emphysema arising from orthog- old female patient died on the first postoperative day
nathic surgery. from unknown causes, with a postmortem examina-
tion failing to identify a cause of death.
Nagler et al144 reported a case of prolonged dys-
Psychological Complications
phagia in a 29-year-old female patient who underwent
Orthognathic surgery is capable of having a dra- a Le Fort I maxillary intrusion and BSSO with advance-
matic improvement on various psychological param- ment and rotation. After nasogastric tube removal 1
eters but conversely has been reported to sometimes week postoperatively, profound dysphagia occurred,
have a detrimental impact.138,139 A review by Cun- which persisted for 7 weeks. Pharyngeal manometry
ningham et al139 presented a case of a 26-year-old showed spasm of the upper esophageal sphincter,
female patient who underwent a bimaxillary proce- and barium swallow fluoroscopy showed no bolus
dure to treat a retrusive mandible. After surgery, the passage beyond the pharynx despite seemingly nor-
patient and her mother described problems adapting mal esophageal peristalsis on endoscopy. A speech
to the new situation. The patient’s parents divorced therapist provided swallowing retraining for 5 weeks,
several months later, reportedly because of their feel- followed by sudden resolution and resumption of
ing of unpreparedness for the outcomes of surgery, in normal swallowing 2 weeks later. Psychiatric assess-
terms of both the healing period and the personality ment found the patient to be regressive, obsessive,
changes of their daughter. Conversion disorder (a and compulsive with a limited intellectual perfor-
disorder whereby neurologic symptoms occur mance. The authors suggested the mechanism in this
without organic neurologic cause, with stress being case was a globus sensation due to central inhibition
held responsible) has been reported after orthog- caused by a compromised psychological condition.
nathic surgery.140 Gaukroger145 reported complete immediate postop-
Depression is said to be relatively frequent among erative dysphagia in a 22-year-old male patient who
orthognathic patients postoperatively, in common underwent Le Fort I maxillary advancement and BSSO
with many other surgical procedures,141 especially setback with fitting of IMF. He was unable to even
when IMF is used.125 The “4-day blues” is a well- swallow his own saliva for 4 weeks, despite periodical
recognized and common phenomenon, although a release of the IMF, hence being fed by nasogastric
majority of such cases are not thought to represent tube. Esophagoscopy showed a tightly closed upper
STEEL AND COPE 1687

esophageal sphincter, balloon dilatation of which was of the tube was said to have possibly been caused
only successful for a few hours, which necessitated by the surgery or subsequent scarring, although
myotomy of the muscle leading to complete resolu- there was no direct evidence of this and coinci-
tion. A hypothesis for this was a change in the anat- dence could not be excluded.
omy of the hyoid region, which may have led to The entity of a “surgical ciliated cyst” has been
reduced tension in the suprahyoid musculature and reported after elective orthognathic surgery.154-160 Al-
hence a reduced dilator effect on the sphincter. though studies in the Japanese literature have re-
Spontaneous leg compartment syndrome has been ported a prevalence of 20%,161 there is very little
reported as a complication after orthognathic surgery. published material in the English-language literature.
Strickland and Westrich146 described 2 cases, 1 with Sugar et al155 described a case of this pathology pre-
mandibular osteotomy for deformity and another with senting in the maxillary buccal sulcus 3 years after a
Le Fort I osteotomy, where leg pain indicated develop- Le Fort I osteotomy. As the name suggests, they are
ment of this syndrome on the second and first postop- typically lined by respiratory epithelium and the
erative days, respectively. Both patients needed 4-com- pathogenesis is hypothesized to involve antral mu-
partment fasciotomy. Beadnell et al147 also described a cosa trapped in the surgical wound, or closure of the
case and listed possible causes as incorrect positioning ostium.151 Shakib et al156 reported an unusual case in
of the patient or leg straps and postoperative muscular the midline of the palate 7 years after a Le Fort I
trauma. A case reported by Teeples et al148 was thought osteotomy. Koutlas et al157 reported a case in the left
to have been due to a drug interaction between the mandibular ramus diagnosed 13 years after maxillary
anesthetic or postoperative medications, or the effect of and mandibular osteotomy procedures, thought to
antidepressant drugs superimposed on chronic, mild, relate to respiratory epithelium traumatically admit-
exercise-induced compartment syndrome. ted to the area. Two other mandibular cases resulted
Perforation of the lateral nasal mucosa by fixation from the use of nasal bone and cartilage grafts in the
screws has been reported by Levine and Super149 in area.158,159
the case of a patient who had postoperative nasal Dental malocclusion after orthognathic surgery
congestion and pain after a Le Fort I maxillary ad- may be encountered relatively frequently due to skel-
vancement with fixation using 7-mm screws. Surgical etal and/or dental relapse. It is, however, rare for
removal and replacement with shorter screws were failure of fixation to be responsible. Such an event is
necessary. Perforation of the nasal septum has also likely to require reoperation. Three cases were re-
been reported.150 ported by Ellis and Esmail,162 all undergoing BSSO
Oroantral and oronasal fistulae have been re- with advancement. In 2 of these patients anterior
ported as a complication by El Deeb et al12 should open bites developed, 1 of which was successfully
slight tears in the oral and antral or nasal mucosa rectified orthodontically after about 5 weeks, and the
correspond. These authors state that oronasal fistu- third case had a 4-mm left midline shift. Radiographs
lae are more common when the maxillae are seg- showed that these malocclusions had resulted from
mentalized or expanded. bending of the single miniplate used to fixate the
Laureano Filho et al151 reported loss of an orth- osteotomized segments, thought to be part of a rota-
odontic bracket into the airway during an orthog- tional movement. Such an event has not been previ-
nathic procedure. ously reported, although mechanical failure of osteot-
Grundig et al152 reported a case of postoperative omy miniplates was described by Fujioka et al.163
recurrent eustachian tube malfunction with middle One of the more bizarre complications in the liter-
ear effusion in a 22-year-old female patient after Le ature is that of an ischemic finger injury caused by a
Fort I advancement in Germany. This did not respond pulse oximetry probe being in the same place for too
to grommet insertion, and investigations showed a long during an orthognathic procedure during which
forward dislocation of the pterygoid hamulus on the hypotensive anesthesia was used.164
same side as well as damage to the tensor veli palatini Rarer complications for which further details are
muscle, purported to be causative. Klemm et al105 not available are as follows:3 herpes labialis, corneal
reported a case of tinnitus and hearing loss in a 36- ulcer, recurrent laryngeal nerve palsy, and arytenoid
year-old who underwent the same operation, as cartilage dislocation.
caused by an arteriovenous fistula of the left maxillary
artery, embolization of which led to resolution of the
Discussion
problem. Wong et al153 reported a cleft palate pa-
tient who was found to have a patulous eustachian It is clear that orthognathic surgery is a safe area of
tube by investigations undertaken in response to oral and maxillofacial surgery, although it carries a
the patient’s complaint of autophony 3 months number of rare risks depending on the exact tech-
after a Le Fort I procedure. This abnormal patency niques used. This review has identified reports of the
1688 COMPLICATIONS OF ORTHOGNATHIC SURGERY

plethora of complications, some with only 1 reported Acknowledgments


case, ranging across all possibilities from neurologic,
The authors offer many thanks to S. T. Crank and C. Mannion for
infective, bony, and bleeding to psychological, anes- their valuable advice on this article.
thetic, and other complications. We feel satisfied that
this review has achieved its goal to describe the re- References
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