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Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology xxx (2017) xxx–xxx

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Journal of Oral and Maxillofacial Surgery,

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

Case report

Cervical necrotizing fasciitis of odontogenic origin in a healthy young

patient without pre-systemic disorders
Masanobu Abe a,b,∗,1 , Takahiro Abe a,1 , Ritsuka Mogi a , Hiroyuki Kamimoto a ,
Noriko Hatano a , Asako Taniguchi a , Hideto Saijo a , Kazuto Hoshi a , Tsuyoshi Takato a
a
Department of Oral & Maxillofacial Surgery, University of Tokyo Hospital, Tokyo, Japan
b
Division for Health Service Promotion, University of Tokyo, Tokyo, Japan

a r t i c l e i n f o a b s t r a c t

Article history: Necrotizing fasciitis (NF) is a rapidly spreading soft-tissue infection characterized by diffuse necrosis of
Received 24 September 2016 fasciae and subcutaneous tissues. Cervical NF (CNF) can develop from odontogenic infections that spread
Received in revised form 1 February 2017 to the deep fascial planes of the neck. This polymicrobial infection is rapidly progressive, destructive,
Accepted 1 March 2017
and often fatal. The majority of CNF patients are known to present an immunosuppressed status or to
Available online xxx
have systemic disease. CNF is uncommon in healthy individuals. Here we describe the case of a patient
with CNF due to an odontogenic polymicrobial infection including Streptococcus milleri group (S. milleri)
Keywords:
after extraction of the mandibular third molar in a healthy 32-year-old Thai man. The CNF progressed to
Odontogenic infection
Streptococcus milleri
descending mediastinitis, which often leads to a poor prognosis and decreased survival. Prompt diagnosis,
Necrotizing fasciitis intensive antibiotic therapy and repeated surgical treatment successfully contributed to the survival of
this patient. His case suggests the importance of perioperative care for invasive dental treatments in not
only patients with pre-systemic disorders but also healthy young individuals.
© 2017 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

Introduction Here we describe a rare case of CNF due to an odontogenic

Cervical necrotizing fasciitis (CNF) patients commonly present
with fever, tachycardia, and dehydration, and sometimes with
swallowing difficulty and trismus. The skin frequently presents as
hard and erythematous, and in the most severe cases the skin can
present wide tissue necrosis. A polymicrobial infection of aerobes
and anaerobes is involved in CNF. The most frequent primary origin
of CNF is reported to be odontogenic infection [1–4].
The incidence of necrotizing fasciitis (NF) is known to be much
higher in immunosuppressed patients and individuals with dia-
betes mellitus, cancer, or cardiovascular insufficiency, transplanted
patients, etc. [5]. Surgical procedures such as extraction of the
teeth occasionally lead to CNF in immunosuppressed patients.
The outcomes of CNF patients are quite adversely affected by
comorbidities. Advanced age and a delay in surgery also affect the
outcomes of CNF patients. CNF in healthy young individuals with-
out pre-systemic disorders are quite uncommon [5].
∗ Corresponding author at: Division for Health Service Promotion, University of
Tokyo Hospital, 7-3-1 Hongo, Bunkyo-ku, Tokyo 113-8655, Japan.
E-mail address: abem-ora@h.u-tokyo.ac.jp (M. Abe).
1
These authors equally contributed to this work.
infection in a healthy young man, and we discuss this enigmatic
situation. We also emphasize the importance of perioperative
infectious control for oral surgical procedures.
Case report
A 32 year-old Thai male with no history of pre-systemic disor-
ders was initially treated in a dental office for a lower third molar
removal (Fig. 1a). Cefditoren pivoxil (CDTR-PI, 100 mg) every 8 h
was used after extraction of the third molar. The doctor in the den-
tal office said that the oral hygiene quality of this patient was poor
and the gingiva around the third molar was swelling. Two days
after the procedure, his body temperature increased to 40 ◦ C and he
experienced trismus and facial erythema at the right submandibu-
lar region (Fig. 1b). His social history was negative for tobacco and
alcohol.
At admission, he was febrile, hypertensive (161/80 mmHg), and
dehydrated because he had not taken any liquids for the past 36 h.
A hemogram revealed a white cell count of 8.3 × 103 /␮L, with 79.8%
neutrophils. The inflammatory response (C-reactive protein; CRP)
was increased (29 mg/dL). Enhanced computed tomography (CT)
scanning revealed extensive abscesses and/or accumulation of gas
in the right deep cervical region (Fig. 2a). Abscesses and/or gas were

http://dx.doi.org/10.1016/j.ajoms.2017.03.001
2212-5558/© 2017 Asian AOMS, ASOMP, JSOP, JSOMS, JSOM, and JAMI. Published by Elsevier Ltd. All rights reserved.

Please cite this article in press as: Abe M, et al. Cervical necrotizing fasciitis of odontogenic origin in a healthy young patient without
pre-systemic disorders. J Oral Maxillofac Surg Med Pathol (2017), http://dx.doi.org/10.1016/j.ajoms.2017.03.001
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Fig. 1. (a) The pantomography after extraction of the patient’s lower right wisdom tooth (arrowhead). (b) Clinical views of the cervical region of the patient before surgery.
Swelling from the submandibular to cervical region was observed (arrowhead).

Fig. 2. Enhanced CT revealed (a) obvious abscess formation and gas accumulation in the right cervical region and (b) the presence of mediastinal emphysema (arrowheads).

found to be located (i) from the right submandibular and sublingual
space to the para- and retropharyngeal space, (ii) from the pterygo-
mandibular space to the temporal fossa, (iii) and from the carotid
space to the mediastinum (Fig. 2b). Based on this examination, we
made the diagnosis of CNF with mediastinitis. An administration
of empiric large-spectrum antibiotics by the parenteral route (sul-
bactam sodium/ampicillin sodium [SBT/ABPC] 3.0 g every 6 h plus
clindamycin [CLDM] 600 mg every 8 h) (Fig. 3) was started.
Extensive surgical debridement was performed after the
antibiotics administration was established. In the nasal tra-
cheal intubation procedure, airway obstruction by the sinking
tongue root was observed, and an emergency tracheotomy was
thus performed. During the surgical procedure, the platysma
muscle of the cervical region was reversed and the forward
edge of the sternocledomastoid was exfoliated as much as
possible. A huge amount of pus was observed in the deep
cervical region. The pus was gray in color, viscous and foul-
smelling. The culture test of the pus showed the presence of
group F beta-hemolytic Streptococcus (S. milleri), Peptostreptococcus
asaccharolyti, Fusobacterium nucleatum, Bacteroidessp., Neisse-
ria sp., and alpha-hemolytic Streptococcus. All of the detected
bacteria were susceptible to the following antibiotics: benzylpeni-
cillin, ampicillin, SBT/ABPC, amoxicillin/clavulanate, cefotaxime,
imipenem/cilastatin, panipenem/betamipron, meropenem, ery-
thromycin, clarithromycin, CLDM, minocycline, chloramphenicol,
vancomycin, moxifloxacin, levofloxacin.
Extensive necrosis of submandibular suprahyoid muscles was
observed under the superficial layer of cervical fascia between the
submandibular grand and the lower edge of the mandible. Surgical
debridement of the extensive necrotic area was performed. After
penetration of the cervical area and the oral floor, irrigation by
saline solution and drainage were conducted for all involved spaces.
Five of drains were left in these spaces. The internal jugular vein and
carotid sheath were exfoliated to approach the deep cervical region.
Parapharyngeal tissue was carefully exfoliated, but an approach
to the retropharyngeal space was not performed. After this 1 st

Please cite this article in press as: Abe M, et al. Cervical necrotizing fasciitis of odontogenic origin in a healthy young patient without
pre-systemic disorders. J Oral Maxillofac Surg Med Pathol (2017), http://dx.doi.org/10.1016/j.ajoms.2017.03.001
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Fig. 3. The time course of treatments and inflammatory changes. The intravenous antibiotic treatment was started on day 0. WBC, white blood cell; CRP, C-reactive protein;
BT, body temperature.
Fig. 4. CT revealed that the abscess in the retropharyngeal space remained after the
1 st surgery.
surgery, the antibiotics were changed to tazobactam/piperacillin
(TAZ/PIPC) 4.5 g every 6 h.
Three days after the 1 st surgery, enhanced CT scanning showed
that the abscess in the retropharyngeal space was still there (Fig. 4).
Accordingly, another debridement was performed with the patient
under general anesthesia. Necrosis of the suprahyoid muscles was
observed to have expanded, and the tissues did not maintain their
normal structures. The necrotized tissues were removed and fascia
spaces were exfoliated as much as possible. An accumulation of pus
was observed in the parapharyngeal space. The middle constrictor
and inferior constrictor were exfoliated and we approached the
retropharyngeal space. Irrigation by saline solution and drainage
were conducted for the involved spaces, and five of the drains were
left in the spaces.
The antibiotic treatment was returned to SBT/ABPC 3.0 g every
6 h plus CLDM 600 mg every 8 h after the above-describe 2nd
surgery, based on a consultation with specialists in the department
of infectious disease (Fig. 3).
Please cite this article in press as: Abe M, et al. Cervical necrotizing fasciitis of odontogenic origin in a healthy young patient without
pre-systemic disorders. J Oral Maxillofac Surg Med Pathol (2017), http://dx.doi.org/10.1016/j.ajoms.2017.03.001
3
Due to the repeated operative debridement and intensive antibi-
otic therapy, the patient avoided the potentially fatal outcome of
CNF. His body temperature, CRP, and leukocytes returned to the
normal levels after the surgeries. The general condition of this
patient almost recovered two weeks after the surgeries, antibi-
otic treatment and irrigation by saline solution had to be kept
conducting for the involved neck spaces for four weeks to pre-
vent reinfection and to promote wound healing. For antibiotic
treatment, SBT/ABPC was selected based on a consultation with
specialists in the department of infectious, because it contains a
beta-lactamase inhibitor and capable of long-time use. The swal-
lowing difficulty and trismus remained, but through rehabilitation,
those functions were recovered. The patient was discharged from
the hospital on the 69th day after admission. The results of bio-
chemical test on the 60th day after admission indicated recovery of
general conditions and suggested the patient was basically healthy.
The data of the biochemical test were as follows. TP 7.1(g/dL),
Alb 4.1(g/dL), AST 30 (U/L), ALT 58(U/L), ␥-GTP 28 (U/L), T-Bil
0.8(mg/dL), Ca 9.1(mg/dL), BUN 6.4(mg/dL), Cre 0.79(mg/dL), Na
140(mEq/L), K 4.3(mEq/L), Cl 103 (mEq/L), CRP 0.12(mg/dL), eGFR
92.8.
Discussion
NF is a rare but potentially fatal condition. The annual incidence
of NF is reported to be from 0.2 to 1.7 per 100,000 person-years
[6], and CNF is even more uncommon [7]. CNF is characterized by a
rapid, progressive, and generalized necrosis of the superficial fas-
cia, with necrosis of subjacent skin [3,4]. It normally starts from an
odontogenic infection, and the patients sometimes present with
swallowing difficulty and trismus [1,2,6]. The mortality rate of CNF
has been reported to be from 7% to 20%, depending on the extent
of neck involvement. When the disease progresses into the thorax,
the mortality rate increases to 41% [1].
NF, including CNF (approximately 3–4% of all NF cases) [8],
is common in the elderly people suffering from chronic debili-
tated illnesses such as diabetes mellitus, renal failure, liver failure,
malignancy, infections, intravenous drug abuse and immunodefi-
ciency conditions. Individuals with diabetes mellitus are reported
to have an especially high incidence (71%). CNF cases without
comorbidities are known to be relatively uncommon (13.5%) [5].
Dental infection is the most common cause of CNF (43–78%) [8–10].
Considering all of the above information, we can say that CNF of
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odontogenic origin in a healthy young patient without pre-systemic
disorders is quite rare and estimated to be from 0.03 to 7.2 per
100,000,000 person-years [5].
Necrotizing soft tissue infections are comprised of two distinct
bacteriologic entities: type I (polymicrobial infection) and type II
(group A streptococcal infection). CNF is considered generally a
mixed synergistic infection (type I) involving both aerobes and
anaerobes. In our patient’s case, a mixed synergistic infection with
S. milleri group, Peptostreptococcus spp., Fusobacterium nucleatum,
Bacteroides, Neisseria, and alpha- hemolytic Streptococcus was iden-
tified in a culture test. In particular, S. milleri group (known to
have strong virulence) is considered to be a main pathogen even
though it is present as part of the normal flora in the oral cavity
[11–13]. Importantly, the S. milleri group is characterized by a ten-
dency for abscess formation. An active infection of this group in the
oral cavity predisposes the host to maxillofacial infections, which
might cause distant metastatic abscess formations involving the
lung, brain, liver, kidney or soft tissues, leading to a life-threatening
situation [4,12].
Here we observed that even healthy young individuals could
develop CNF. The clinical information of the patient at the tooth
removal could not be obtained, however it is quite unlikely that
he has been in immune-suppressed situation considering his past
history and perioperative clinical data. In the oral cavity, a local
infection sometimes leads to a severe systemic infectious disease
by an imbalance of the oral bacterial community. Dentists must
be most careful when they perform tooth extractions in which
they find acute inflammations in the gingiva or other soft tissues.
Thorough antibiotic treatment and oral care to improve the oral
hygienic status are recommended before a tooth extraction in order
to remove the risk of infectious disease.
The present case of CNF in a healthy young individual empha-
sizes the importance of perioperative infectious control for oral
surgical procedures.
Conflict of interest
The authors declare that they have no competing interests.
Funding
No funding.
Please cite this article in press as: Abe M, et al. Cervical necrotizing fasciitis of odontogenic origin in a healthy young patient without
pre-systemic disorders. J Oral Maxillofac Surg Med Pathol (2017), http://dx.doi.org/10.1016/j.ajoms.2017.03.001
Ethics approval and informed consent
This research was approved by the research ethics committee
of Graduate School of Medicine and Faculty of Medicine, The Uni-
versity of Tokyo. Written informed consent was obtained from the
patient.
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