ABSTRACT: Although most fistulae are not problematic, (1) surgeons
occasionally encounter recurrent and/or refractory fistulae in the field
of oral and maxillofacial surgery. In this case report, the authors
describe a case in which a patient experienced a recurrent and
refractory fistula or perforation at his oral floor through the
submandible, with heterotopic bone formation arising on both sides of
the mylohyoid line. These heterotopic bones were connected to each
other, forming a bone bridge at the center of the oral floor. A
fistulectomy and wound closure with a tongue flap was successful. The
perforation has not recurred after over four years of follow-up, and the
bone bridge is still present.
Fistulae that cause inflammation and infections after surgery are
often encountered by surgeons, but their occurrence can usually be
controlled by adequate treatment. If incomplete treatment for
inflammation is performed, they often recur. Conservative fistula
closing strategies include wound drainage and minimal debridement. When
fistulae do not close after conservative interventions, debridement and
surgical approaches such as a flap closure should be performed. (2)
Several techniques that are performed to close oral fistulae, including
the use of grafts and flaps, have been reported, and the results are
generally good. (3) In particular, tongue flaps have been reported to be
very useful for repairing defects because of the volume and excellent
blood supply of this tissue. (4)
Infections sometimes induce heterotopic ossifications. Sclerosing
osteomyelitis such as Garre disease and diffuse sclerosing osteomyelitis
are well known to form new bone due to a periosteal reaction. (5) Here,
the authors report a case of refractory perforation with ectopic bone
formation, successfully treated with a tongue flap.
A 76-year-old male Japanese patient presented with a sensation of
swelling and pain in his left mandibular angle. He suffered from
hypertension and cataracts, but had no history of allergy and no
significant family medical history. He was diagnosed with osteomyelitis
of the posterior mandible and was admitted to the hospital for
treatment. An incision into the patient's skin to his mandibular
bone was made to drain the affected region, and the patient was given
antibiotics for osteomyelitis of the left mandible. Following that
treatment, the patient's swelling and pain disappeared, and he was
discharged from the hospital. The authors followed up with the patient
once a month.
After six months, the patient complained of oral floor submental
wound dehiscence and that saliva and food leaked through the perforation
to the outside of the oral cavity [Figure I(A)]. Therefore, the
perforation was closed and a sequestrectomy performed at the lingual
side of the anterior body of the mandible while the patient was under
general anesthesia. The surgical approach was via a submandible incision
in which the mandible bone was exposed and the mandible's marginal
and lingual bones were removed as blocks, along with the granular
tissue. Finally, the wound was closed after lengthy irrigation with
saline. At this time, heterotopic bone formation was found around both
ends of the mylohyoid line, and the bones were connected to each other
at the center of the oral floor like a bone bridge, as seen on an
occlusal x-ray film [Figure 1 (X-Ps)]. Because the patient's wound
dehiscence occurred three weeks after the first operation, the authors
tried to close the opened wound in the same region under general
anesthesia (during the second operation). Pathology of the excise
specimen was osteomyelitis and no bone sequestrum was found. During this
time, both intraoral and submental approaches were used. The intraoral
wound was tightly closed, and the submental wound remained open.
Finally, the submental wound was completely closed at one month after
surgery, and the patient was discharged from the hospital. We again
followed up with him once a month. After one year, there was no
recurrence of the perforation; however, the oral floor-submental wound
had reopened. The authors closed the perforation the same way as in the
second operation (third operation). Because the wound reopened again so
soon after the surgery [Figure 1(B)], a fourth operation was performed,
and the perforation was closed using a bone bridge. The tissue around
the perforation was fibrous, granulation tissue. Briefly, the bone
bridge was cut at the center and both fragments (still connected at the
mylohyoid line of the mandible) were pulled forward with wire and
ligated to the mental region of the mandible. However, wound dehiscence
soon recurred. Using a tongue flap, the authors performed a fifth
operation to close the perforation (Figure 2) on the basis of a previous
report. (5) Briefly, the anteriorly-based dorsal tongue flap was
separated from donor site. The flap was designed on the raw undersurface
to fit the defect at the oral floor and sutured with submental skin.
Because there was no sign of wound rupture, the flap was cut, and the
tongue was released two weeks after the surgery.
[FIGURE 1 OMITTED]
There was no evidence of recurrence of the perforation, and this
patient still has the bone bridge on his oral floor at his four-year
follow-up. The treatment process is shown in Figure 3.
[FIGURE 2 OMITTED]
In the present case, the size of the fistula was small after the
first fistulectomy. The authors thought the wound would close
spontaneously, but perforation recurred. Because remaining infection is
the most critical reason for wound dehiscence, (1) insufficient
treatment of osteomyelitis is considered to be the most critical cause
for a first recurrence of wound dehiscence. However, the authors believe
that the repeated wound dehiscence after the second operation was caused
by scar contraction and a lack of blood supply. The patient's oral
floor had a strong tension, and the skin was very thin, resulting in
ischemia. This may have caused the wound to open repeatedly. In
addition, continuous leaking of saliva may have prevented the
perforation from closing spontaneously. The oral floor consists of thin
elastic skin and a bone bridge that may lead to more tension on the
skin. Moreover, chronic inflammation results in thinner skin, which may
have caused increased tension and ischemia in the patient's oral
floor. The authors were finally able to close the perforation using a
tongue flap; tissue that is reported to provide enough volume to supply
sufficient blood flow. (4)
[FIGURE 3 OMITTED]
The authors experienced a rare case in which heterotopic bone had
formed on both sides of the mylohyoid line of the mandible, and these
bones were connected to each other, creating a bone bridge. The authors
believe that myositis ossificans (MO), which is characterized by
non-neoplastic bone formation in soft tissue and skeletal muscle and is
induced by trauma, burns, or other events, (6) may have triggered
formation of the ectopic bone in the current case. Although several MO
cases of the masseter and temporalis have been reported, there have been
no reports of MO in the sublingual region. Chronic inflammation may be
the most important cause of ectopic bone formation in this case, based
on a previous report that described the relationship between
inflammation and a periosteal reaction. (7) However, according to
Seung-Jun, et al., (8) static tension force induces bone absorption and
formation, which depends on the force direction. Thus, it is possible
that continuous tension led to heterotopic bone formation at the oral
floor in this case.
The authors' conclusions in this case are as follows: 1.
shortage of the surrounding tissue caused by infection resulted in
ischemia and tension of the tissue, leading to repeated perforations; 2.
spontaneous stimulation by chronic infection caused ectopic bone
formation in the sublingual region; and 3. the tongue is a good donor
tissue for use as a flap to close the perforation at the oral floor.
(1.) Demir Z, Velidedeoglu H, Celebioglu S: Repair of
pharyngocutaneous fistulas with the submental artery island flap. Plast
Reconstr Surg 2005; 115:38-44.
(2.) Papazoglou G, Doundoulakis G, Terzakis G, Dokianakis G:
Oharyngocutaneous fistula after total laryngectomy, incidence, case. and
Otol Rhinol Laryngol 1994: 103:801-805.
(3.) Oswald TM, Stover SA, Gerzenstein J, Lei MP, Zhang F, Muskett
A, et al.: Immediate and delayed use of arteriovenous fistulae in
microsurgical flap procedures: a clinical series and review of published
cases. Ann Plast Surg 2007; 58:6-13.
(4.) Agrawal K, Panda KN: Management of a detached tongue flap.
Plast Reconstr Surg 2007; 120:151-156.
(5.) Francisco JD, Alicia D, Francisco JA, Alamillos MD, Luis N.
Jacino Florencio M: Tongue flaps for reconstruction of the oral cavity.
Head Neck 1994; 16:550-554.
(6.) McCarthy EF, Sundaram M: Heterotopic ossification: a review.
Skeletal Radiol 2005; 34: 609-619.
(7.) Ida M, Tetsumura A, Kurabayashi T, Sasaki T: Periosteal new
bone formation in the jaws. A computed tomographic study.
Dentomaxillofac Radiol 1997; 26: 169-176.
(8.) Ku SJ, Chang YI, Chae CH, Kim SG, Park YW, Jung YK, Choi JY:
Static tensional forces increase osteogenic gene expression in
three-dimensional periodontal ligament cell culture. BMB Rep 2009;
Seigo Ohba, D.D.S., Ph.D.; Joji Sekine, D.D.S., Ph.D.; Takayoshi
Tobita, D.D.S., Ph.D.; Hideyoshi Ikeda, D.D.S.; Izumi Asahina, D.D.S.,
Manuscript received June 10, 2010; revised manuscript received
December 16, 2010; accepted December 17, 2010
Address for correspondence:
Dr. Seigo Ohba
Dr. Seigo Ohba is a lecturer at the Department of Regenerative Oral
Surgery at Nagasaki University Graduate School of Medical Sciences,
Japan. He received his D.D.S. degree at Nagasaki University in 1999 and
his Ph.D. degree from the same university in 2003. During 20072009, he
studied the relationship between chondrocyte and leptin at fire
Department of Rheumatology, Internal Medicine, University of Michigan,
Ann Arbor as a visiting researcher.
Dr. Joji Sekine is a professor at the Department of Oral and
Maxillofacial Surgery, Shimane University Faculty of Medicine, Japan. He
received a D.D.S. degree at Fukuoka Dental College in 1989 and his Ph.D.
degree from Nagasaki University in 1996. He worked for the Department of
Oral and Maxillofacial Surgery, Nagasaki University during 1989-2006,
and also worked in the Department of Oral & Maxillofacial Surgery,
Umea University as a visiting professor during 2006-2007.
Dr. Takayoshi Tobita is a lecturer in the Department of Dentistry
and Oral Surgery at the University of Fukui Hospital in Japan since
April, 2010. He received his D.D.S. degree at Nagasaki University School
of Dentistry in 1997 and his Ph.D. degree at the same university in
2001. He worked as an assistant professor in the Division of
Regenerative Oral Surgery at the Nagasaki University Graduate School of
Biomedical Sciences from 2001 to 2010. During 2002-2004, he worked for
the Department of Oral and Maxillofacial Surgery at the University of
Michigan as the visiting researcher.
Dr. Hideyoshi Ikeda is a guest researcher at the Department of
Regenerative Oral Surgery at Nagasaki University Graduate School of
Biomedical Sciences, Japan. In 2006, he received his D.D.S. degree at
Nagasaki University, and his Ph.D. degree from the same university in
Dr. Izumi Asahina is a chairman at the Department of Regenerative
Oral Surgery at Nagasaki University Graduate School of Medical Sciences,
and a Vice President of Nagasaki University Hospital, Japan. He received
his D.D.S. degree at Tokyo Medical and Dental University in 1983 and his
Ph.D. degree from the same university in 1987. During 1991-1993, he
studied bone cell biology at Harvard University and Children's
Hospital, Boston, and engaged in clinical research for bone regeneration
at the Institute of Medical Sciences, University of Tokyo, as an
associate professor from 2004 to 2006.