YIJOM-3749; No of Pages 4

Int. J. Oral Maxillofac. Surg. 2017; xxx: xxx–xxx

http://dx.doi.org/10.1016/j.ijom.2017.07.006, available online at http://www.sciencedirect.com

Case Report
Orthognathic Surgery

Unilateral blindness after Á. Rodrı́guez-Navarro1,

F. M. Gonzalez-Valverde2
1
Department of Anaesthesia and Critical

orthognathic surgery: Care, Reina Sofia General University

Hospital, Murcia, Spain; 2Department of
Surgery, University of Murcia, Reina Sofia
General University Hospital, Murcia, Spain

hypotensive anaesthesia is not

the primary cause
Á. Rodrı́guez-Navarro, F.M. Gonzalez-Valverde: Unilateral blindness after
orthognathic surgery: hypotensive anaesthesia is not the primary cause. Int. J. Oral
Maxillofac. Surg. 2017; xxx: xxx–xxx. ã 2017 International Association of Oral and
Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Abstract. Perioperative vision loss in non-ocular surgery represents a rare but

devastating complication and multiple causes have been proposed. Any portion of
the visual system may be involved and several authors have tried to relate that
complication with deliberate hypotension anaesthetic technique, used to control
intraoperative bleeding. We report a patient operated for orthognathism who
suffered unilateral blindness. After review of similar cases, we can state that the
transmission of forces generated during Le fort I osteotomy is related to the
complication. This osteotomy technique is regularly performed in our hospital using
Key words: orthognathism surgery complica-
a curved osteotome to achieve the pterygomaxillary disjunction and the adverse
tions; orthognatic surgery ophthalmic complica-
transmission of forces via the sphenoid bone is the main reason for indirect damage tions; perioperative visual loss; anaesthesia
to the optic nerve and its vascular structures causing the neuropathy and blindness. and controlled hypotensive complications.
Hypotensive anaesthesia may certainly lead to transient ischaemia but only in
specific cases because of decreased ocular perfusion pressured. Accepted for publication

Vision loss in the perioperative period is a
severe complication that has motivated
many studies because the mechanism is
not always clear. In particular, the onset of
blindness in non-ophthalmic surgery has
led to the creation of a register by the
American Society of Anesthesiologist
(ASA). The ASA Postoperative Visual
Loss Registry1 mainly shows postoperative
blindness cases after spinal surgery and the
authors, using a multicentre case–control
design, recently published the risk factors
associated with this complication: obesity,
male sex, Wilson frame use, longer anaes-
thetic duration, greater estimated blood loss,
and percent colloid administration.
The objective of this paper is to criti-
cally review the literature on orthog-
nathic surgery and its relation to
vision loss. Since deliberated hypoten-
sion is involved in this procedure, it is
possible to discuss in more detail the
potential anaesthetic etiologic causes for
this complication.

0901-5027/000001+04 ã 2017 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Rodrı́guez-Navarro A, Gonzalez-Valverde FM. Unilateral blindness after orthognathic surgery:
hypotensive anaesthesia is not the primary cause, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.07.006
YIJOM-3749; No of Pages 4
2 Rodrı́guez-Navarro and Gonzalez-Valverde
Osteotomy Le Fort I is regularly per-
formed during orthognatic surgery.
According to different authors, the pter-
ygomaxillar separation using a curved
osteotome produces high transmission of
forces and pressures; uncontrolled propa-
gation through the skull base and adjacent
structures such as the orbit would result in
fractures and vision loss2,3. The maxillary
downfracture can also contribute to forces
being transmitted to the orbit and the optic
canal area, as well as to untoward fractures
that extend to the sphenoid bone and orbit.
Other methods of achieving the pterygo-
maxillary disjunction, such as the use of a
micro-oscillating saw, seem to be less apt
to cause untoward fractures4.
Blindness after orthognathic surgery is
usually not from a direct injury to the optic
nerve itself, but more commonly is the
result of an ischaemic injury to the blood
supply to the optic nerve, either directly
from a fracture extending through the orbit
to the optic canal or foramen, or indirectly
from swelling and oedema around the
nerve in the optic canal disrupting its
blood supply. Hypotensive anaesthesia
and/or anomalies of the blood supply to
the optic nerve could be possible contrib-
uting factors to worsening the ischaemia in
selective instances. This could include
those cases where there was excessive
blood loss and hypotensive anaesthesia
was not well controlled and where the
mean arterial pressure (MAP) dropped
below a certain level for extended periods
of time.
Case report
A 41-year-old woman was admitted for
orthognathic surgical correction of her
malocclusion and temporomandibular
chronic pain: body mass index of
23.3 kg/m2, no remarkable medical histo-
ry, preoperative haemoglobin of 11.4 g/
dL. No abnormalities were found in the
complementary tests. The surgery was
performed under general anaesthesia. A
premedication with midazolam 2 mg was
administered intravenously and vasocon-
strictor (oxymetazoline) was applied
through the nostrils. In the operating room,
the patient was monitored by pulse oxim-
etry, capnography, bispectral index analy-
sis (BIS), continuous electrocardiography,
and invasive blood pressure. Anaesthesia
was induced after preoxygenation (with
O2 at 60% applied during 5 min using a
facial mask), propofol 150 mg, fentanyl
100 mg, and rocuronium 40 mg were ad-
ministered.
The patient was intubated easily
through the right nostril. Anaesthesia
Please cite this article in press as: Rodrı́guez-Navarro A, Gonzalez-Valverde FM. Unilateral blindness after orthognathic surgery:
hypotensive anaesthesia is not the primary cause, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.07.006
was maintained with an inspired oxygen
fraction of 40% (oxygen/air) and sevo-
fluorane at 2% (according to BIS). Remi-
fentanyl and additional doses of fentanyl
and morphine were administered through-
out the process to maintain the patient at
mean arterial pressure (MAP) values
around 65–70 mmHg. The operation
lasted 300 min. Le Fort I osteotomy se-
quence with the pterygomaxillary disjunc-
tion was carried out between the maxillary
tuberosity and pterygoid plates using a
curved Obwegeser osteotome. An impor-
tant aspect of this technique is the angle
given by surgeon to chisel. The chisel
should remain in all time under the peri-
osteum, with its tip in the pterygomaxil-
lary suture, angled from lateral to medial
and top to bottom. The surgeon’s index
finger should palpate the palatal region of
the tuberosity and pterygoid hamulus to
feel their separation.
Intraoperative blood loss was estimated
at 900 mL. MAP values ranged around
70 mmHg. The hourly urine output was
greater than 80 mL/hour (>1 mL/kg/
hour). No intraoperative incidents were
registered and the patient was transferred
to an intensive care unit for postoperative
monitoring, conscious and with spontane-
ous ventilation. Three hours following
surgery the patient first complained to
the nurses of being unable to see anything
from her right eye. Oedema and ecchymo-
sis could be seen at the lower eyelid.
Ophthalmic examination checked that in
spite of normal ocular fundus (no papillo-
oedema) there was no light perception in
the right eye. An immediate computerized
tomography scan (CT) performed on the
brain, facial, and sinuses (Fig. 1) showed
Fig. 1. A CT cut showing a high-level pterygoid plate fracture.
the existence of a small infraorbital hae-
matoma and emphysema caused by sur-
gery in the right eye, as well as fractures in
the pterygoid apophysis suggestive of
traumatic pterygomaxillary disjunction
(not unusual fracture patterns following
Le Fort I osteotomies). Brain pathology
was declined. The patient was empirically
treated with steroids (intravenous dexa-
methasone 8 mg/8 hour). The patient
was discharged to the ward with complete
amaurosis of the right eye and the initial
diagnosis of retrobular optic neuropathy.
Visual evoked potentials that were per-
formed 4 days after surgery showed ab-
normalities in pattern reversal stimulation,
which were suggestive of right axonal
optic neuropathy. The patient was dis-
charged from hospital 7 days after surgery
with complete amaurosis of the right eye.
Discussion
Amaurosis or unilateral blindness caused
by facial trauma (iatrogenic or not) is a
complication attributed by some authors to
damage of the optic nerve in the orbital
osseous channel whose incidence ranges
from 3% to 5%2–5. On many occasions the
significance of the damage is not propor-
tional to the trauma as very severe injuries
are observed on negligible traumatisms.
There is a close anatomic correlation
among the optic canal, the palatine bones,
the pterygoid, and the sphenoid. Direct
injuries to the nerve secondary to unto-
ward fractures which extend to the base of
the skull, orbit, or pterygopalatine fossa,
associated with the pterygomaxillary sep-
aration and/or maxillary downfracture,
can be the aetiology of blindness. A sec-
YIJOM-3749; No of Pages 4
ond possibility is damage to neurovascular
structures from traction, shearing, com-
pression, or contre coup injuries via an
adverse transmission of forces during the
pterygomaxillary disjunction using an
osteotome, or the maxillary downfrac-
ture4.
A moderate oedema or haemorrhage in
this area can be devastating. The retinal
tissue and the optic nerve are very sensi-
tive to pressure and the damage occurs
within 60 min and certainly within 2
hours3. Because of these reasons and also
because there was no evidence of papillo-
oedema, we only used corticosteroids.
Lanigan et al.6 offer an excellent review
summarizing all the ophthalmic complica-
tions associated with orthognatic surgery,
which include reduction or loss of vision,
extraocular muscle dysfunction, keratitis,
and nasolacrimal problems. In the paper
by Laningan, several cases and plausible
causal mechanisms are presented. The
aetiology of all these problems is related
with direct or indirect damage on adjacent
neurovascular and osseous structures close
to the orbit and the skull base. Tension,
compression, and pressure provoked at the
pterygomaxillar separation necessary to
force the maxillar movement can be trans-
mitted in an uncontrolled manner. Girotto
et al.2, using a corpse, attempted to repro-
duce, the magnitude and propagation
mechanism of those forces and their im-
pact on the orbit. The authors recommend
a gradually stepped and tapered osteot-
omy. Most of the postoperative unilateral
blindness cited in the literature occurs in
middle-aged patients and curiously in the
right eye (like our patient), making the
authors think about how the surgical pro-
Fig. 2. Coronal and axial views showing a small infraorbital haematoma and emphysema in the
right eye.
Please cite this article in press as: Rodrı́guez-Navarro A, Gonzalez-Valverde FM. Unilateral blindness after orthognathic surgery:
hypotensive anaesthesia is not the primary cause, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.07.006
Unilateral blindness after orthognathic surgery
cedure is performed (some theories de-
scribe that the surgeon should revise
where the osteotome is placed) and about
the decrease in osseous plasticity with age.
Following basic principles of causality
and according to works cited in that paper,
postoperative blindness in a patient who
underwent orthognatic surgery was related
to surgery. However, Lanigan et al.6 and
other authors cite a 1953 paper (Gillan)7 in
which two cases of unilateral blindness
following anaesthesia with vascular hypo-
tension are reported, neither of them in
maxillofacial surgery. In that work the
complication is attributed to an occlusion
of the central retinal artery, motivated by
the fact that it is a terminal artery that
would be ischaemic when hypotension is
extreme and intraocular pressure is high.
Nevertheless, neither these situations oc-
curred in our patient.
Owing to the implications of this refer-
ence, we should know that, as pointed out
by the 2008 paper by Choi and Samman8,
deliberate hypotension in anaesthesia can
be justified as a routine procedure for
orthognathic surgery, especially bimaxil-
lary osteotomy, as it contributes to a de-
crease in surgical bleeding and the rate of
severe complications of this method is low
if the indications are correct. In our pa-
tient, a healthy 41-year-old woman, sur-
gery was uneventful, with haemodynamic
stability with MAP around 65 mmHg and
performed in the supine position.
As we saw, the attempt to relate our
complication with the deliberated hypo-
tension procedure is not new, and anaes-
thesiology journals since 1953 contain
multiple cases where perioperative vision
loss in non-ophthalmic surgery is
3
reported1,7,9. A register has been devel-
oped at the ASA that includes all of these
patients, who in most of cases have been
operated on the spine or in prone position.
There are also cases from digestive pro-
cedures or who have suffered severe hy-
potension.
Although in our patient, the CT image
the level of pterygoid apophysis plate
fractures is suggestive of a traumatic dis-
junction and also a small infraorbital hae-
matoma and emphysema (Fig. 2) served as
an evidence, prior to this case, we had
knowledge of another patient that experi-
enced abundant blood loss (>2,000 mL)
during bimaxillary osteotomy which con-
tinued during the immediate postoperative
period forcing multiple transfusions and
prolonged intubation (72 hours) due to
cervicofacial oedema. What would have
happened if blindness had been the com-
plication in that patient? Would haemo-
dynamic instability and hypotension have
been identified as the causes despite the
consideration of this particular surgical
procedure?
As it is explained in referenced
papers6,9, most of the time, structural dam-
age cannot be established on radiologic
examination. As a consequence, uncer-
tainty in the aetiology of the diagnosis
remains open to the implication of anaes-
thesia. We do not totally reject that impli-
cation for this surgery and we believe
there might be certain specific instances
where it could at least be a contributing
factor to causing ischaemia in the optic
nerve, even though the major cause is as a
result of the surgery itself. But, in our
opinion, hypotensive anaesthesia should
not be considered the most adequate an-
swer to decreased visual acuity after
orthognathic surgery, nor could it be the
sole cause in itself.
Some authors7–10 have investigated the
most significant risk factors for postoper-
ative visual alterations: the loss of more
than 1000 mL of blood, maintained anae-
mia and hypotension, surgery lasting more
than 6 hours, prone position, and male sex.
It occurs more frequently during spinal
surgery although it has been described
even on liposuction. Consequently, anaes-
thesiologists should aim to prevent it9.
To summarize, complications in orthog-
natic surgery could be clinically signifi-
cant and involve all the surgical team
during the intraoperative and resuscitation
periods. They should also know if other
cases have been reported in the literature
and how they were solved so that compli-
cations can be prevented. Once blindness
develops, the prognosis is poor. High Le
Fort I osteotomy should be performed with
YIJOM-3749; No of Pages 4
4 Rodrı́guez-Navarro and Gonzalez-Valverde
extreme care, and perhaps informed con- American Society of Anesthesiologists Post-
sent should include visual loss as a com- operative Visual Loss Registry: analysis of
plication of the procedure10. 93 spine surgery cases with postoperative
visual loss. Anesthesiology 2006;105:652–9.
2. Girotto JA, Dadvison J, Wealthy M, Redett
Funding R, Muelberger T, Robertson B, Zinreich J,
None. Iliff N, Miller N, Manson PN. Blindness as a
complication of Le Fort osteotomies: role of
atypical fracture patterns and distortion of
Competing interests the optic canal. Plas Reconstr Surg
1998;102:1409–21.
None. 3. Cruz AA, Santos AC. Blindness after Lefort
I osteotomy: a possible complication associ-
Ethical approval ated with pterygomaxillary separation. J
Cranio Maxillofac Surg 2006;34:210–6.
Not required. 4. Lanigan DT. the inherent risks of the pter-
ygomaxillary dysjunction and maxillary
downfracture. Jpn J Jaw Deformities
Patient consent
2000;10:158–62.
Not required. 5. Kim JW, Chin BR, Park HS, Lee SH, Kwon
TG. Cranial nerve injury after Le Fort I
osteotomy. Int J Oral Maxillofac Surg
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hypotensive anaesthesia is not the primary cause, Int J Oral Maxillofac Surg (2017), http://dx.doi.org/10.1016/j.ijom.2017.07.006
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Address:
Francisco Miguel González Valverde
C/Victorio n 3
2 C
30003
Murcia Spain
E-mail: migova67@gmail.com