Intracranial Hemorrhage Resulting From
Skull Base Fracture As a Complication of Le
Fort III Osteotomy
Kazuya Matsumoto, MD,* Hideki Nakanishi, MD,* Takuya Seike, MD,* Yoshio Koizumi, MD,*
Shinichi Hirabayashi, MD†
Tokushima, Japan
Tokyo, Japan
Various complications of Le Fort osteotomies have
been reported. We describe a lethal complication of
Le Fort III osteotomy we encountered in a 9-year-
old boy with Crouzon syndrome. A standard Le
Fort III osteotomy, including pterygomaxillary dys-
junction with a curved osteotome and down-
fracture manipulation, was performed unevent-
fully. When the intraoral buccal wound was closed
after fixation of the external midface distraction de-
vices, we discovered hemorrhage originating from
the right posterior maxillary region. Although it
was stopped with pressure on the osteotomized
maxilla, the volume of intraoperative blood loss
was nearly 2,000 ml. During the observation period
in the intensive care unit, the patient suffered brain
death, and he died 3 months later. A computed to-
mography scan obtained the day after surgery re-
vealed vigorous subarachnoid and intraventricular
hemorrhage and transverse fracture of the middle
cranial fossa. This skull base fracture was believed
to result from intraoperative maneuvers, including
the pterygomaxillary dysjunction and down-
fracture manipulation. We emphasize the risk of
intracranial hemorrhage with Le Fort osteotomy
and advise discussing this risk with patients and
family members during preoperative consultations.
Key Words: Lethal complication, Le Fort osteotomy,
subarachnoid hemorrhage
From the *Section of Plastic and Reconstructive Surgery, Uni-
versity Hospital, University of Tokushima, Tokushima; and †De-
partment of Plastic and Reconstructive Surgery, School of Medi-
cine, Teikyo University, Tokyo, Japan.
Address correspondence to Dr Matsumoto, Section of Plastic
and Reconstructive Surgery, University Hospital, University of
Tokushima, 2-50-1, Kuramoto-cho, Tokushima 770–8503, Japan;
e-mail: kazuya@clin.med.tokushima-u.ac.jp
L
e Fort osteotomy, especially Le Fort I osteot-
omy, is a widely accepted technique for cor-
recting certain facial deformities. Various
complications associated with Le Fort I oste-
otomy have been described, but the incidences are
low.1,2 Complications result primarily from the
pterygomaxillary dysjunction and down-fracture
maneuvers that are used in all three types of Le Fort
osteotomy; thus, the possibility of complications ex-
ists for type II and III osteotomies as well as for type
I osteotomies.3,4 We encountered a patient with
Crouzon syndrome who suffered intracranial hem-
orrhage after a Le Fort III osteotomy.
CASE REPORT
9-year-old boy presented with exophthalmos
A and midface retrusion (Fig 1). At the age of 7
years, he was diagnosed with Crouzon syndrome
and underwent fronto-orbital advancement. We
planned a midface distraction with rigid external dis-
traction devices (RED System; Martine, Germany)
(Fig 2). We performed a standard Le Fort III osteot-
omy, including pterygomaxillary dysjunction by
curved osteotome and down-fracture manipulation.
Maxillary mobilization was uneventful, and the ex-
ternal distraction devices were fixed with six pins
inserted with a torque-controlled driver. We ob-
served hemorrhage originating from the right poste-
rior maxillary region when the intraoral buccal
wound was closed, however. Pressure on the os-
teotomized maxilla stopped the bleeding, but intra-
operative blood loss was nearly 2,000 ml. To avoid
respiratory obstruction by postoperative edema and
to allow the surgical region to rest, the patient was
intubated under sedation in the intensive care unit.
The postoperative course was generally uneventful.
We began to decrease sedation on the morning after
surgery, but the patient’s level of consciousness did
not improve. Both pupils were fixed and dilated. A
computed tomography scan revealed subarachnoid
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THE JOURNAL OF CRANIOFACIAL SURGERY / VOLUME 14, NUMBER 4 July 2003
Fig 1 Preoperative photographs of our patient at the age
of 9 years. Note the mild maxillary retrusion (A) and ex-
ophthalmos (B).
hemorrhage and transverse fracture of the middle
cranial fossa (Fig 3). Mannitol was administered, but
total brain function was lost, and the large volume of
urine indicated diabetes insipidus. Brain death was
diagnosed on the basis of neurological findings. Al-
though we continued general treatment, the patient’s
condition did not improve, and he died 3 months
after surgery. Autopsy revealed a transverse fracture
of the middle cranial fossa (Fig 4).
DISCUSSION
e Fort osteotomy is a standard maxillofacial sur-
L gery procedure with a low incidence of associ-
ated complications. With Le Fort I osteotomy, com-
Fig 2 Three-dimensional model of the patient’s head con-
structed from the preoperative computed tomography
data includes marks made in preparation for the osteoto-
mies. Rigid external distraction devices are attached.
546
Fig 3 (A) Postoperative computed tomography scan
shows pointed bony shadows suggesting a fracture in the
middle cranial fossa. (B) There are linear shadows along
the subarachnoid space that suggest subarachnoid hemor-
rhage.
plications reportedly occur in 6% to 9% of cases.1
Complications reported previously include hemor-
rhage, arteriovenous fistula, and ophthalmic symp-
toms, of which blindness is one of the most severe.1–6
Whereas most reported complications have been as-
sociated with Le Fort I osteotomy, there have been a
few associated with Le Fort II and III osteotomies.7,8
The investigative focus on complications of Le Fort I
osteotomy may be a result of the popularity of this
type of osteotomy. In our case, the patient suffered a
lethal subarachnoid hemorrhage after Le Fort III os-
teotomy; our case is unusual in terms of the degree of
complication and the type of osteotomy involved.
Cranial base fracture was the most likely cause
Fig 4 (A) At autopsy, a fracture line in the middle cranial
fossa was apparent across the hypophyseal fossa. (B)
Schema of the photograph obtained at autopsy indicates
the fracture line.
 INTRACRANIAL HEMORRHAGE RESULTING FROM SKULL BASE FRACTURE / Matsumoto et al

of subarachnoid hemorrhage in our patient, consid-
ering the computed tomography and autopsy find-
ings. To date, six cases of cranial base fracture asso-
ciated with Le Fort osteotomy have been reported
(Table 1).1,3,5,9–11 In all six cases, the basal fracture
was believed to occur in association with the ptery-
gomaxillary dysjunction and maxillary down-
fracture. A curved osteotome is generally used for
pterygomaxillary dysjunction. Dysjunction with this
osteotome as well as down-fracture manipulation es-
sentially raises the vector force that pushes the ptery-
goid plate posteriorly. This force is transferred to the
skull base through the sphenoid bone and can frac-
ture the skull base,5 as we believe it did in our pa-
tient. Extradural hemorrhage could easily result
from such a fracture; however, penetration of the
dura mater by the spicula of the fractured bone can
cause subarachnoid hemorrhage. This explains the
subarachnoid hemorrhage in our patient.
Aneurysm is the other possible cause of hemor-
rhage. Although an aneurysm in an infant with
Crouzon syndrome has been reported,12 this condi-
tion is believed to be rare. Aneurysm in our case is
unlikely; the cranial base fracture was visualized
during autopsy.
The pterygomaxillary separation procedure
used in Le Fort osteotomies can cause an atypical
fracture that extends to the skull base, orbit, or ptery-
goid plate.1,5,13 Untoward fractures such as ptery-
goid plate fractures reportedly occur at a much
higher frequency than previously thought.13,14 To
minimize the risk of such fractures, researchers are
investigating alternate pterygomaxillary separation
procedures. Reiner and Willoughby9 recommended
that the curved osteotome be directed anteriorly, me-
dially, and inferiorly at the pterygoid plate. Trimble
et al15 suggested that the incision be made with a
straight osteotome through the tuberosity, ending at
the posterior aspect of the hard palate, medial and
anterior to the pterygomaxillary fissure. Wikkeling
and Tacoma16 designed an osteotome resembling a
swan’s neck to permit the cutting force to be directed
from behind. Hiranuma et al17 determined and com-
pared the strain distribution over the surrounding
bone structures during pterygomaxillary separation
with the Obwegeser and swan’s neck osteotomes
and found no significant advantage in using the
swan’s neck osteotome. We use a thin curved os-
teotome with the blade facing obliquely in the an-
teromedial direction to avoid posterior-superior
compression of the pterygoid process. A curve at the
end of the osteotome does not eliminate the posterior
vector of force applied along the axis of the os-
teotome handle.18 Several recent studies suggest the
use of an oscillating saw for separation instead of an
osteotome.18–20 Chen and Fisher18 reported absence
of postoperative visual disturbance in Le Fort oste-
otomy patients since they began using a right-angled
oscillating saw for separation. A new method of en-
doscopic pterygomaxillary dysjunction has been re-
ported,21 but the safest method remains controver-
sial.
Skull base fracture is so rare that the possibility
is not mentioned by most maxillofacial surgeons dur-
ing preoperative counseling. It may be necessary to
discuss this risk when obtaining preoperative in-

Table 1. Reported cases of skull base fracture after Le Fort osteotomy

Patient Type of Surger in addition
Authors Age (y)/sex Clinical diagnosis osteotomy to Le Fort Osteotomy Complication(s)

Girotto et al1 20/male Congenital maxillary Le Fort I None Cerebrospinal fluid rhinorrhea and left
hypoplasia superior orbital fissure syndrome
Lanigan et al3 23/male Vertical maxillary excess and Le Fort I Bilateral sagittal mandibular Carotid-cavernous sinus fistula
mandibular retrognathia ramus osteotomies and
genioplasty
Lanigan et al5 33/female Open-bite malocclusion and Le Fort I Bilateral sagittal mandibular Blindness in the right eye
complex facial asymmetry ramus osteotomies,
genioplasty, and bone graft
Reiner et al9 27/female Facial deformity Le Fort I Bilateral vertical ramal Transient abducens nerve palsy
osteotomies
Bendor-Sameul 30/male Left cleft palate Le Fort I Iliac bone graft augmentation of Impaired visual acuity
et al10 the maxilla
Lanigan et al11 27/female Left cleft lip and palate Le Fort I Bone graft to the alveolar cleft Carotid-cavernous fistula and
site transient abducens nerve palsy
Matsumoto et al* 9/male Crouzon syndrome Le Fort III None Lethal subarachnoid hemorrhage

*Present case.

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formed consent, however. Patients with syndromic
craniosynostosis in which the cranial base is abnor-
mal may be particularly vulnerable to unexpected
fracture.22,23 We should exercise care in the perfor-
mance of pterygomaxillary dysjunction, especially in
patients with syndromic craniosynostosis. At the
same time, we should develop surgical procedures
and instruments that ensure the safety of this ma-
neuver.
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