ONLINE
CHRONIC INVASIVE FUNGAL SINUSITIS CAUSING A EXCLUSIVE
PATHOLOGIC LE FORT I FRACTURE IN AN IMMUNOCOMPETENT PATIENT
Chronic invasive fungal sinusitis
causing a pathologic Le Fort I fracture
in an immunocompetent patient
Amy L. Richter, MD; K. Kelly Gallagher, MD
Abstract
We describe the case of a 77-year-old immunocompetent
woman with a history of chronic rhinosinusitis who pre-
sented with a pathologic Le Fort I fracture after a forceful
sneeze. Imaging revealed diffuse sinus opacification and
a Le Fort type I complex fracture involving the maxilla,
pterygoid plates, clivus, and right nasal bridge. The pa-
tient underwent endoscopic debridement of her sinuses,
which revealed mucosal dehiscence and otherwise normal
healthy bleeding tissue. Anatomic pathology identified
necrotic bone with invasive fungal hyphae. Cultures
demonstrated Burkholderia cepacia, diphtheroid organ-
isms, and Enterococcus and Serratia spp. The patient was
administered an intravenous antibiotic and antifungal for
several months, but interval imaging found no significant
improvement in bone healing although the stability of her
palate had improved on clinical examination. Chronic
rhinosinusitis has been found to be a complication of soft-
tissue, orbital, and intracranial infections but, to the best
of our knowledge, a pathologic facial fracture secondary
to chronic invasive fungal and bacterial rhinosinusitis has
not been previously reported in the literature.
Introduction
Chronic invasive fungal sinusitis has been found to be a
complication of soft-tissue, orbital, and intracranial in-
fections in immunocompromised patients, but advanced
From the Department of Otolaryngology, University of Colorado School
of Medicine, Denver (Dr. Richter); and the Department of Otolar-
yngology–Head and Neck Surgery, Baylor College of Medicine,
Houston (Dr. Gallagher). The case described in this article occurred
at the Baylor College of Medicine.
Corresponding author: K. Kelly Gallagher, MD, Department of Otolaryn-
gology–Head and Neck Surgery, Baylor College of Medicine, One
Baylor Plaza, NA 102, Houston, TX 77030. Email: kkgallag@bcm.edu
Volume 95, Number 9
complications infrequently affect immunocompetent
patients.1-3 Osteomyelitis is a rare complication of chronic
rhinosinusitis that typically occurs in the frontal bone
in the setting of Pott puffy tumor, but osteomyelitis of
the other sinuses is rare.1
Pathologic fractures are a rare complication of os-
teomyelitis. Pathologic mandibular fractures have been
related to mandibular osteomyelitis.4 Other causes of
pathologic fractures of the mandible include surgical
interventions, osteoradionecrosis, and bisphosphonate-
related osteonecrosis.4 To the best of our knowledge, no
case of a pathologic midface fracture related to chronic
invasive fungal sinusitis and facial osteomyelitis has been
previously reported in the literature. We describe such
a case in an immunocompetent patient with chronic
invasive fungal sinusitis who experienced a pathologic
Le Fort I fracture.
Case report
A 77-year-old woman with a history of chronic rhinosi-
nusitis and poor nutrition presented to the emergency
department after an episode of syncope secondary to
long-standing aortic stenosis. She was admitted for
further workup. She also noted a 2-month history of
maxillary tooth discomfort and loose maxillary teeth fol-
lowing an episode of forceful sneezing. The ENT service
was consulted after computed tomography (CT) revealed
extensive sinus opacification and a complete complex
Le Fort I fracture (figure 1). The fracture involved the
maxilla, pterygoid plates, basisphenoid skull base, clivus,
hard palate, and right nasal bridge.
The patient complained of dry nasal crusting, but
she denied nasal congestion, rhinorrhea, facial pain
and pressure, and facial swelling. Her medical history
included hypertension, aortic stenosis, anemia, diabetes,
and chronic rhinosinusitis. She had undergone endo-
scopic sinus surgery 4 years earlier, and she exhibited
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 RICHTER, GALLAGHER

A B C

Figure 1. A: Coronal noncontrast CT shows opacification of the maxillary sinus and nondisplaced fractures of the lateral and medial
maxillary walls bilaterally (arrowheads). Also seen are poor mineralization of the maxillary bone and extensive air trapping between
the hard palate bone and mucosa (arrow). B: Second noncontrast CT demonstrates opacification of the sphenoid sinus along with the
same fractures (arrowheads). Extremely poor mineralization is seen in the skull base, with fractures extending through the sella turcica,
petrous apices, and basisphenoid area (arrows). C: Sagittal noncontrast CT shows opacification of the frontal sinus, a Le Fort I fracture
through the maxilla (arrowhead), a transverse clival fracture (white arrow), and poor mineralization of the skull base (red arrow).

no evidence of fungal disease. On physical examination,
all of her maxillary teeth were loose, and the hard palate
was edematous and spongy on palpation, with trapped
air pockets. Her palate was freely mobile relative to her
midface. Her cranial nerve function was normal.
Nasal endoscopy identified a thick, green discharge
and nasal crusting with evidence of a previous maxillary
antrostomy and ethmoidectomy. Sensation was intact
throughout the nasal cavity. The patient’s blood glucose
level ranged from 110 to 140 mg/dl during her inpatient
stay. Serology demonstrated hypoalbuminemia (2.2 g/
dl), a severely low prealbumin level (6 mg/dl), and anemia
(hemoglobin: 9.3 g/dl); other measurements were within
normal limits, including a normal neutrophil count.
After obtaining written consent from the patient, we
performed an endonasal endoscopic biopsy and debrided
both sinuses. Endoscopy of the right nasal cavity revealed
mucosal dehiscence of the cartilaginous septum, as well
as exposed bone in the posterior maxillary and ethmoid
sinuses, the anterior face of the sphenoid sinus, and the
skull base (figure 2). Frozen-section pathology of an ul-
ceration at the anterior septum was negative for invasive
fungal disease. Purulence was expressed from the right
maxillary, ethmoid, and sphenoid sinuses. Endoscopic
examination of the left nasal cavity revealed the presence
of mucoid and purulent secretions from the maxillary,
ethmoid, and sphenoid sinuses (figure 3).
Irrigation and debridement of all the sinuses revealed
evidence of normal healthy bleeding mucosa. The ex-
posed bone at the sphenoid sinus and skull base was
irrigated and left in place. The final pathology review
identified invasive fungal hyphae in the bone fragments,

A B

Figure 2. A: Endoscopic view of the middle meatus of the right nasal cavity shows necrotic mucosa and exposed bone (arrow) in the
posterior ethmoid sinus. B: Image shows a close-up view of the exposed bone.

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 CHRONIC INVASIVE FUNGAL SINUSITIS CAUSING A PATHOLOGIC LE FORT I FRACTURE IN AN IMMUNOCOMPETENT PATIENT

further consideration was being given to hyperbaric

oxygen therapy. Unfortunately, her midface fractures
could not be repaired because her bone quality was poor;
plates, screws, and wires cannot be secured to unhealthy
bone such as hers to achieve fixation.
As far as we know, this report represents the first
published case of a pathologic fracture related to osteo-
myelitis secondary to chronic invasive fungal sinusitis.

References
1. Epstein VA, Kern RC. Invasive fungal sinusitis and complications
of rhinosinusitis. Otolaryngol Clin North Am 2008;41(3):497-524.
2. deShazo RD, O’Brien M, Chapin K, et al. A new classification and
diagnostic criteria for invasive fungal sinusitis. Arch Otolaryngol
Head Neck Surg 1997;123(11):1181-8.
Figure 3. Endoscopic view of the middle meatus of the left nasal
3. Jackson LL, Kountakis SE. Classification and management of
cavity shows the purulent drainage (arrow) from the ethmoid sinus. rhinosinusitis and its complications. Otolaryngol Clin North Am
2005;38(6):1143-53.
4. Boffano P, Roccia F, Gallesio C, Berrone S. Pathological mandibular
and cultures grew Burkholderia cepacia, diphtheroid fractures: A review of the literature of the last two decades. Dent
organisms, and Enterococcus and Serratia spp. No fungi Traumatol 2013;29(3):185-96.
were seen in the final fungal cultures. 5. Capener N, Pierce KC. Pathological fractures in osteomyelitis. J
Bone Joint Surg Am 1932;14(3):501-10.
The patient was placed on an intravenous antibiotic
and antifungal, and a nasogastric tube was placed for
nutritional supplementation. She was switched to an
oral antibiotic for insurance purposes and followed as
an outpatient for 5 months. At the most recent evalua-
tion, the stability of her maxilla had improved, but her
nasal crusting persisted and she developed ulcerations
of her maxillary alveolus. Repeat CT revealed no im-
provement in her Le Fort fracture pattern, along with
persistent sinus opacification and osteomyelitis of the
skull base and maxilla. Repeat endoscopic sinus surgery
and debridement were planned, and hyperbaric oxygen
was being considered if her signs and symptoms failed
to improve.

Discussion
Pathologic fractures of the mandible account for only
2% of all mandible fractures; osteomyelitis is a rare
cause of fracture.4 While there have been no previously
reported cases of pathologic midface fracture, the dental
and orthopedic literature does include discussions of
osteomyelitis as a possible cause of pathologic fracture.4,5
Our patient presented with a 2-month history of maxil-
lary mobility. The extensive structural changes that were
seen on imaging despite normal healthy bleeding tissue
suggested a chronic invasive fungal process.2 Manage-
ment of chronic invasive fungal disease involves long-
term antifungal medications and appropriate surgical
debridement in addition to antibiotics, routine sinus
hygiene, and serial endoscopic debridement.3
Since our patient failed to improve with treatment,

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