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Contents lists available at ScienceDirect

Journal of Oral and Maxillofacial Surgery,


Medicine, and Pathology
journal homepage: www.elsevier.com/locate/jomsmp

Case Report

A massive osteonecrosis with Le Fort I-type pathological fracture,


caused by bisphosphonate-related osteonecrosis of the jaw (BRONJ):
A rare case report
Shintaro Sukegawa a, , Takahiro Kanno b , Naoki Katase c , Hidenobu Matsuzaki d ,
Akane Shibata a , Yuka Takahashi a , Yoshihiko Furuki a
a

Division of Oral and Maxillofacial Surgery, Kagawa Prefectural Central Hospital, Kagawa, Japan
Department of Oral and Maxillofacial Surgery, Shimane University Faculty of Medicine, Shimane, Japan
c
Department of Molecular and Developmental Biology, Kawasaki Medical School, Okayama, Japan
d
Department of Oral Diagnosis and Dentomaxillofacial Radiology, Okayama University Hospital, Okayama, Japan
b

a r t i c l e

i n f o

Article history:
Received 28 November 2014
Received in revised form 26 August 2015
Accepted 13 October 2015
Available online xxx
Keywords:
Osteonecrosis
Bisphosphonate
Pathological fracture

a b s t r a c t
Bisphosphonates (BPs) are useful drugs for the management of osteoporosis and metastatic bone disease,
and are used to prevent skeletal-related events. However, the side effects of BPs, bisphosphonate-related
osteonecrosis of the jaw (BRONJ), sometimes severely impairs patient quality of life. Here we report a case
of severe BRONJ in the maxilla, which caused Le Fort I-type fracture-like bone fracture due to formation
of a large sequestrum in the maxilla.
2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

1. Introduction
Bisphosphonates (BPs) are frequently prescribed for the treatment of patients with malignant bone diseases such as metastases
from other solid malignancies [1] and multiple myeloma [2] and for
the treatment of those with metabolic diseases such as osteoporosis [3]. However, as a complication of BP administration, BP-related
osteonecrosis of the jaw (BRONJ) has been reported in a large
number of patients in recent years. In the advanced stages, the presence of osteolysis extends to the jaw bone and forms sequestrum,
resulting in bone weakness and pathological fracture [4]. The definition of pathological fractures is controversial, but the term can
be described as fractures occurring as a result of normal function
or minimal trauma in a bone weakened by pathological conditions

Asian AOMS: Asian Association of Oral and Maxillofacial Surgeons; ASOMP: Asian
Society of Oral and Maxillofacial Pathology; JSOP: Japanese Society of Oral Pathology; JSOMS: Japanese Society of Oral and Maxillofacial Surgeons; JSOM: Japanese
Society of Oral Medicine; JAMI: Japanese Academy of Maxillofacial Implants.
Corresponding author at: Division of Oral and Maxillofacial Surgery, Kagawa
Prefectural Central Hospital, 1-2-1, Asahi-cho, Takamatsu, Kagawa 760-8557, Japan.
Tel.: +81 87 811 3333; fax: +81 87 802 1134.
E-mail address: gouwan19@gmail.com (S. Sukegawa).

including metabolic bone diseases, severe infection, cysts and bone


tumors. Diagnosis of the fracture itself is done based on physical
and radiological examination. Generally, pathological fractures in
the maxillofacial region occur in the mandible. To date, no advanced
cases of pathological fractures in the maxilla caused by BRONJ have
been reported. Here we report a case of BRONJ that progressed to
a Le Fort I-type pathological fracture. Here we report a rare case of
massive osteonecrosis caused by BRONJ with Le Fort I-type pathological fracture.
2. Case report
A 70s/M patient noticed swelling and discomfort in the right
upper canine to premolar area along with a suppurative stula, and
he was referred to a general dental practitioner. At the clinic, he was
treated by irrigation of the swollen area and a clinical diagnosis of
dental infection was made. However, he experienced absolutely no
improvement after the treatment. Two months later, he noticed
greatly increased mobility in the upper canine; thus, the canine
was extracted by dentist. During the following month, his bilateral rst premolars, right second premolar, and rst molar in the
maxilla spontaneously fell out. A partial upper denture was subsequently created. Soon afterward, the patient noticed swelling

http://dx.doi.org/10.1016/j.ajoms.2015.10.001
2212-5558/ 2015 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Sukegawa S, et al. A massive osteonecrosis with Le Fort I-type pathological fracture, caused
by bisphosphonate-related osteonecrosis of the jaw (BRONJ): A rare case report. J Oral Maxillofac Surg Med Pathol (2015),
http://dx.doi.org/10.1016/j.ajoms.2015.10.001

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S. Sukegawa et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxxxxx

and pain of the left upper premolar to molar area and was again
referred to a general dental practitioner. His left second premolar and rst molar in the maxilla were found to be highly mobile;
thus, they were extracted in a month later from denture creation.
After that, bilateral second molars in the maxilla spontaneously
fell out. Although the teeth extraction sockets failed to heal well,
a complete upper denture could be remanufactured as there were
no abnormalities of the oral mucosa, including the palate. However, several weeks later, the denture became unstable, so it was
lined using a soft denture-lining material. After the denture was
adjusted, it had to continuously remain in his oral cavity for few
days. At the end of this period, he was referred to a dental clinic for
removal of the denture lining, which proved to be difcult. When
attempting to remove the denture, the dentist noticed shaking of
the maxilla along with the denture. The patient was then referred
to our service of Oral and Maxillofacial Surgery Division, Kagawa
Prefectural Central Hospital for further evaluation and treatment
of the whole maxillary movement.
His medical history included prostate cancer. He had suffered
from pain due to bone metastasis, and he had been treated with
monthly therapy with zoledronate (4 mg), which was intravenously
administered, for approximately 27 months. However, he and his
family did not mention this to the dentist as they believed that there
would be no relationship between his dental problems and the use
of BPs for past several years.
Extraoral examination detected a cutaneous discharging stula
in the right side wall of the nose area (Fig. 1A). The stula was
in communication with the corresponding right apical maxillary
canine region. He had hypoesthesia of the bilateral infraorbital
nerve distributions. Symptomatic maxillary sinusitis was not
observed. His maxilla was mobile with no simultaneous movement of the nasal bones upon palpation. He was unable to bite
by maxillary movement, and the denture was unsuitable. Intraoral
examination revealed exposed necrotic bone coupled to his denture
at the site of total maxilla (Fig. 1B).
Computed tomography (CT) revealed fractures of the left pterygoid plate, bilateral anterior and lateral maxillary walls, and
posterior nasal septum in the axial bony window (Fig. 2A). Coronal views revealed bilateral fractures through the lateral walls of
the maxillary sinuses (Fig. 2B). A three-dimensional reconstruction
view allowed clear visualization of the fracture line at the Le Fort I
level (Fig. 2C). Although no lesions were observed in the skull base,
evidence of inammation was observed from the right part of the
sphenoid to the external surface of the skull base.

Under local anesthesia, sequestrectomy of the bilateral total


maxillary bone along with his denture was performed. The maxilla was easily removed as it was attached to a region of palatine
mucous membrane only. The necrotic bone with irregular ragged
margins of the cortex, was removed en block with the maxillary
denture rmly attached to the bone (Fig. 3). After removal of the
maxillary sequestrum, the discharging right cutaneous facial stula
gradually decrease, his stula disappeared in a month after surgery.
Histopathological evaluation of the resected maxilla conrmed
necrosis of the maxillary bone with bacterial overgrowth and no
evidence of metastatic disease.
On the basis of clinical, histological, and instrumental examinations, osteonecrosis of the jaw induced by the previous treatment
with zoledronate was diagnosed.
3. Discussion
BRONJ was rst reported as a serious side effect of long-term BP
treatment by Marx [5] in the United States in 2003. The mandible
is more commonly affected than the maxilla [68]. The exact reason for the characteristic distribution of osteonecrosis in the jaws
is unknown; several reasons have been postulated such as the
comprehensive blood supply, different bone architecture, and different embryological background [4,9]. This case demonstrated
that BRONJ can affect maxillary bone. In addition, this was a rare
case of osteonecrosis affecting such a large sequestrum in the maxilla.
BRONJ mainly occurs after dentoalveolar surgery, including procedures such as tooth extraction, periodontal surgery, and dental
implant placement [6,7]. BRONJ may also occur spontaneously
without any apparent dental disease, treatment, or trauma. In this
case, spontaneous loss of bilateral premolar and molar teeth in
the maxilla had previously occurred. Therefore, etiology of spontaneous BRONJ was suspected. When the maxillary denture was
tted, this may have caused the sequestrum to spread to the entire
maxilla.
After performing denture adjustment, the sequestrum had
become strongly adhered to the denture through the oral mucosa
and denture lining material. As a result of the weak downward force exerted on the molars around the anterior maxillary
portion to remove the denture, a pathological fracture resembling a Le Fort I-type fracture was caused. Maxillary fractures
most frequently occur as a result of high energy trauma caused by
assaults, sport injuries, or trafc accidents. In contrast, in this case,

Fig. 1. (A) The patient was unable to close his mouth by maxillary movement, and the denture was unsuitable. (B) Intraoral examination revealed exposed necrotic bone
connected to his denture at the site of total maxilla.

Please cite this article in press as: Sukegawa S, et al. A massive osteonecrosis with Le Fort I-type pathological fracture, caused
by bisphosphonate-related osteonecrosis of the jaw (BRONJ): A rare case report. J Oral Maxillofac Surg Med Pathol (2015),
http://dx.doi.org/10.1016/j.ajoms.2015.10.001

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Fig. 2. Computed tomography images. (A) Axial bone window image showing fractures of the left pterygoid plates, bilateral anterior and lateral maxillary walls, and posterior
nasal septum. (B) Coronal bone window image showing bilateral fractures through the lateral walls of the maxillary sinuses. (C) A three-dimensional reconstruction image
showing clear visualization of the fracture line at the Le Fort I level.

maxillary fracture was caused by the low energy trauma of denture


removal. This indicated that formation of the sequestrum separated the maxillary bone along a boundary of the Le Fort I fracture
line.
In this case, no abnormalities in the palate mucosa were
observed on the production of dentures by the patients dental
practitioner despite healing failure of the extraction socket. This
nding suggests spontaneous formation of the submucosal maxillary sequestration. Early BRONJ may be identied in cases of fossa
healing failure following tooth extraction despite normal mucosal
appearance and doubt regarding the formation of sequestration
under the mucous membrane. The maxillary sinus is found directly
superior to the maxilla. The clinical symptoms of maxilla sinusitis have been reported as an early sign of maxillary BRONJ [10]. In
this case, no typical maxillary sinusitis symptoms were observed as
the maxillary sinuses were open following obliteration by severe
inammation.

The facial stula in this case was formed by the upward spread
of inammation, likely due to infection of the sequestration as the
facial stula resolved following removal of the maxillary sequestration. In addition, hypoesthesia in bilateral infraorbital nerve
distributions was likely due to the spread of inammation to the
infraorbital foramen. Further, the patient had markedly reduced
QOL due to progression of the sequestration, including the formation of an oral maxillary sinus communication after sequestrum
removal. The use of local aps [11,12] or the creation of obturator
prostheses [13] may be used to treat small maxillary sequestrations
before they become more extensive or difcult to treat. Recognition of the clinical symptoms associated with the upward spread
of inammation from sequestra may allow early detection of maxillary sequestration.
Most BRONJ cases are related to treatment with intravenous
BPs, and only a few are related to oral BP administration [68].
Intravenous BPs have been conventionally used for the indication

Fig. 3. The resected specimen showed the presence of necrotic bone with irregular ragged margins of the cortex, which was removed en block with the maxillary denture
rmly attached to the bone.

Please cite this article in press as: Sukegawa S, et al. A massive osteonecrosis with Le Fort I-type pathological fracture, caused
by bisphosphonate-related osteonecrosis of the jaw (BRONJ): A rare case report. J Oral Maxillofac Surg Med Pathol (2015),
http://dx.doi.org/10.1016/j.ajoms.2015.10.001

G Model
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ARTICLE IN PRESS
S. Sukegawa et al. / Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2015) xxxxxx

of bone metastases from malignant tumors and multiple myeloma


in Japan. In contrast, oral BPs have been used only for the treatment
of osteoporosis. However, subcutaneous administration of BPs was
recently approved at about the same time in Japan, Europe, and the
United States. Because this is a novel method of administering BPs,
it will be necessary to pay more attention to such patients in future
with regard to dental treatment.
As soon as oncologists or physicians prescribe BP therapy, the
patient should be referred to an experienced dentist or oral and
maxillofacial surgeon for an urgent examination. Close and ongoing communication is crucial; there should be thorough sharing of
information pertaining to BP use between the doctor and patient.
In this case, in spite of the fact that the patient had been administered BPs for an extended period, this information was not shared
with the dentist. In such cases, there is a possibility that osteonecrosis will develop. In the future, dentists and oral and maxillofacial
surgeons must play an important role in further enlightening the
doctors who administer BPs, as well as patients who have been
treated with BPs. At the same time, follow-up of patients treated
with BPs is also required.
4. Conclusions
Here we report a case of advanced, spontaneous BRONJ that
resulted in severe osteolysis such that minor force caused a Le Fort
I-type fracture and loss of the entire width of the maxilla. In order
to prevent an onset and aggravation of BRONJ as possible, it is necessary to share the information of using BPs with physician and
patient.
Funding
The authors did not receive any support in the form of grants.

Conict of interest
The authors declare no conicts of interest.
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Please cite this article in press as: Sukegawa S, et al. A massive osteonecrosis with Le Fort I-type pathological fracture, caused
by bisphosphonate-related osteonecrosis of the jaw (BRONJ): A rare case report. J Oral Maxillofac Surg Med Pathol (2015),
http://dx.doi.org/10.1016/j.ajoms.2015.10.001

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