Acta Oto-Laryngologica

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Cervical necrotizing fasciitis: an overlooked

diagnosis of a fatal disease

Mohamed A. Al-Ali, Ashraf F. Hefny, Kamal M. Idris & Fikri M. Abu-Zidan

To cite this article: Mohamed A. Al-Ali, Ashraf F. Hefny, Kamal M. Idris & Fikri M. Abu-Zidan
(2017): Cervical necrotizing fasciitis: an overlooked diagnosis of a fatal disease, Acta Oto-
Laryngologica, DOI: 10.1080/00016489.2017.1393841

To link to this article: http://dx.doi.org/10.1080/00016489.2017.1393841

Published online: 06 Nov 2017.

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 ACTA OTO-LARYNGOLOGICA, 2017
https://doi.org/10.1080/00016489.2017.1393841

RESEARCH ARTICLE

Cervical necrotizing fasciitis: an overlooked diagnosis of a fatal disease

Mohamed A. Al-Alia,b, Ashraf F. Hefnya,b, Kamal M. Idrisc and Fikri M. Abu-Zidana,b
a
Department of Surgery, College of Medicine and Health Sciences, UAE University, Al Ain, Abu Dhabi, United Arab Emirates; bDepartment of
Surgery, Al Ain Hospital, Al Ain, Abu Dhabi, United Arab Emirates; cDepartment of Critical Care, Al Ain Hospital, Al Ain, Abu Dhabi, United
Arab Emirates

ABSTRACT ARTICLE HISTORY

Background: Necrotizing fasciitis of the neck is a rare potentially lethal condition if not early diag- Received 24 August 2017
nosed and managed. We aimed to study the clinical presentation, radiological and microbiological Revised 3 October 2017
diagnosis, management, and surgical outcome of patients having cervical necrotizing fasciitis (CNF). Accepted 8 October 2017
Materials and methods: We retrospectively studied patients having a final diagnosis of CNF who were
treated at Al Ain Hospital during the period of January 2000 to December 2016. KEYWORDS
Downloaded by [University of Florida] at 22:28 08 November 2017

Results: Six patients with CNF were studied. Diabetes mellitus was the most common predisposing fac- Fasciitis; infection; neck;
tor (83.3%). All patients presented with a painful neck swelling. The most common source of infection necrotizing; surgery
was odontogenic. Mixed microbiological flora was present in five patients. Five patients underwent CT
scan of the head and neck with a positive finding of gas in all of them. Repeated aggressive surgical
debridement in combination with antibiotic therapy was adopted. Four patients (66.7%) developed
superior mediastinitis, two had septicemia, and one patient had a perforated duodenal ulcer. One
patient died (overall mortality 16.7%).
Conclusion: Maintaining a high index of suspicion is crucially important for diagnosing CNF. Early diag-
nosis, timely resuscitation, and aggressive surgical debridement are the key to a successful clinical
outcome.

Introduction
Necrotizing fasciitis is a potentially fatal bacterial infection
characterized by progressive necrosis of the fascia and sub-
cutaneous tissue. It mainly affects the abdominal wall, peri-
neum, and extremities. It rarely involves the cervical region.
The reported incidence of cervical necrotizing fasciitis
(CNF) in Denmark was two per million per year [1].
CNF is predominantly odontogenic or pharyngeal in ori-
gin. It commonly occurs in elderly immunocompromised
patients. Diabetes mellitus, malnutrition, and burns are com-
mon predisposing factors [2–4].
The diagnosis of necrotizing fasciitis is usually delayed.
This is because of its rarity and its benign initial presenta-
tion. The delay in diagnosis may have significant morbidity
and mortality because of septic shock, disseminated intravas-
cular coagulation, and organ failure. The reported death
rates have been as high as 73% [1,5,6].
Diagnosis of CNF is based on a combination of clinical
presentation, microbiology, imaging, and ultimately surgical
exploration. It usually involves polymicrobial infection of
both aerobes and anaerobes [1,3,7]. Computed tomography
(CT) is very useful for early diagnosis because it can easily
detect the presence of fluid collection and presence of gas
along the fascial planes [5,8,9].
We aimed to study the clinical presentation, radiological
and microbiological diagnosis, management, and surgical
outcome of patients who had CNF and who were treated at
our hospital during the last 16 years.
Material and methods
This study was approved by Al-Ain Medical District Human
Research Ethics Committee, Al Ain, Abu Dhabi, United Arab
Emirates. (Ethical Approval Number: AAH/EC-06-15-010).
All patients who had CNF and were admitted to Al Ain
Hospital during the period of January 2000 to December
2016 were retrospectively studied.
Al Ain Hospital is located in the center of Al Ain city,
the largest city in the eastern district of Abu Dhabi Emirate
of the United Arab Emirates, with a population of 738,000
inhabitants [10]. It is a university affiliated secondary care
hospital which is specialized in acute and emergency care.
The diagnosis of CNF was made based on the clinical
presentation, CT scan findings, and intraoperative presence
of fascial and tissue necrosis.
A study protocol was designed to collect the required
data. Data collected from patients’ records included demog-
raphy, clinical features, diagnosis, the possible source of
infection, and outcome.
The collected data were entered into a Microsoft Excel
spreadsheet (Microsoft Corporation, Seattle, WA).
Descriptive statistical analysis was performed.

CONTACT Ashraf F Hefny ahefny@uaeu.ac.ae Assistant Professor, Department of Surgery, College of Medicine and Health Sciences, UAE University, P O
Box 18532 Main Building of Post Office, Al Ain, Abu Dhabi, 1006 UAE
ß 2017 Acta Oto-Laryngologica AB (Ltd)
 2 M. A. AL-ALI ET AL.

Table 1. Patients with cervical necrotizing fasciitis who were treated at Al Ain Hospital during the period of January 2000 to December 2016.
Case Age Gender DS (days) Initial diagnosis Origin Microbiology NOP Outcome
1 36 M 3 Cervical lymphadenitis Pharyngeal Gram () bacilli, coagulase () 2 Survived
2 40 M 8 Cervical cellulitis Odontogenic Gram () bacilli, Gram (þ) cocci 1 Survived
3 54 M 5 Neck abscess Odontogenic Gram () bacilli 3 Survived
4 43 M 3 Peritonsillar abscess Odontogenic Coagulase () staphylococcus 1 Survived
5 50 M 7 Ludwig angina Idiopathic No Growth 1 Survived
6 55 M 3 Neck abscess Odontogenic Klebsiella Pneumoniae, B hemolytic group G 3 Died
DS: duration of symptoms.
NOP: number of operation.

Table 2. Symptoms and signs of patients having cervical necrotizing fasciitis Table 3. Laboratory findings in patients having cervical necrotizing fasciitis
at presentation who were treated at Al Ain Hospital during the period of who were treated at Al Ain hospital during the period of January 2000 to
January 2000 to December 2016. December 2016.
Symptoms/signs Number (%) Variable Median Range
Symptoms WBC (10 9/L) 14.7 8–27
Neck swelling 6 (100) S. sodium (mmol/L) 134 127–137
Neck pain 5 (83.3) S. creatinine (mg/dL) 1.025 0.7–1.8
Sore throat 3 (50) S. albumin (g/dL) 2.5 2–3.6
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Dysphagia 2 (33.3) Random blood sugar (mmol/L) 19.35 6.5–37.6

Fever 1 (16.6)
Signs
Edema 5 (83.3)
Tenderness 5 (83.3)
Erythema 4 (66.6)
Skin necrosis 1 (16.6)
Trismus 1 (16.6)

Results
Presentations
Six patients were retrieved and studied; all were males. The
patients had a median (range) age of 46.5 years (36-55).
Diabetes mellitus was the most common associated comor-
bidity (five patients, 83.3%).
The median (range) time duration of symptoms was four
days (3-8) (Table 1). All patients presented initially with a
painful, red neck swelling and minimal distress. Two
patients (33.3%) had difficulty in swallowing (Table 2).
On examination, all patients had stable vital signs. The
median (range) mean arterial pressure was 82 mmHg (75-
105). The median (range) heart rate was 97 beats per minute
(80-106). One patient had a temperature of 38.7
C. All
patients had a tender erythematous swelling (Table 2). The
submandibular triangle of the neck was involved in all
patients. The right side of the neck was affected in five
(83.3%) patients. None of the patients were initially diag-
nosed to have CNF (Table 1). The possible source of infec-
tion was odontogenic in four patients, pharyngeal in one,
and idiopathic in another patient.

Investigations
Laboratory investigations showed leukocytosis (more than
13000  109/L) in four patients. One patient had high serum
creatinine level (more than 1.5 mg/dl) (Table 3).
Five patients underwent CT scan of the head and neck
within 24 h of admission. Air pockets were found in all of Figure 1. Cervical necrotizing fasciitis following tooth extraction in a 54-year-
them, two had areas of low attenuation consistent with a old diabetic man. Sagittal CT scan (A) shows air in the cervical spaces (arrows)
and at both the anterior and posterior mediastinum (arrow heads).
fluid collection, and four had air pockets detected in the Intraoperative findings of the same patient (B) showing necrotic fascial planes
mediastinum (Figure 1(A)). Multi-microbial bacterial species in the cervical region.

were identified on culture in five patients. One specimen
yielded no growth, probably due to previous antibiotic ther-
apy or improper collection of specimen (Table 1).
Management and outcome
All patients received broad-spectrum antibiotics on admis-
sion. They underwent surgical exploration and aggressive
debridement of the affected area (Figure 1(B)). Surgery was
carried out within 24 h of admission in two patients
(33.3%). Repeated surgical debridement was performed in
three patients (50%) (Table 1). Histopathological examin-
ation of the excised tissues was performed in two patients
and confirmed the diagnosis of CNF.
Four patients (66.7%) developed superior mediastinitis,
two (33.3%) had septicemia, and one (16.7%) had perforated
duodenum and multi-organ failure. The mediastinum was
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explored and debrided through the transcervical approach at
the time of initial surgery in three patients and through a
thoracotomy in one patient.
Four patients (66.7%) were admitted to the intensive care
unit. Data regarding the length of hospital stay was missing
in two patients, but the other four patients had a median
(range) hospital stay of 41 days (11-103). One patient died
(overall mortality was 16.7%). This patient had a perforated
duodenal ulcer, septicemia, and multi-organ failure.
Discussion
Necrotizing fasciitis of the cervical region is uncommon.
Y-M Liu et al. [11] reported a series of 87 cases of necrotiz-
ing fasciitis and found only five involving the head and neck
(6%). We have a higher percentage of CNF. Six out of 55
patients (10.9%) of necrotizing fasciitis who were treated at
our hospital during the same period were in the neck
(Hefny et al., unpublished data). CNF predominantly affects
men in their fourth decade of life. The median age of our
patients was 47 years which is similar to other studies
[11–15].
Although necrotizing fasciitis can affect healthy individu-
als, several predisposing factors like obesity, diabetes melli-
tus, chronic renal failure, and immunosuppression have
been implicated in its occurrence [12,13]. Similar to others,
we have shown that diabetes mellitus is the most common
disease associated with CNF [4,11,14]. Although the exact
cause of CNF is not always clear, the present study has
shown that it is commonly odontogenic, followed by pha-
ryngeal origin. This finding is like other studies [5,9,15].
The submandibular triangle, which is the major lymphatic
drainage area from the surrounding source, was affected in
all our patients.
Because of the nonspecific initial signs and symptoms of
CNF, misdiagnosis is common [14]. Patient’s presentation
ranges from mild localized soft tissue infection to severe
deep neck infection and thoracic extension. The diagnosis of
CNF was initially missed in all our patients. Majority, at
presentation, were diagnosed to have a localized infectious
ACTA OTO-LARYNGOLOGICA 3
process although four of them (66.7%) had mediastinitis
(Table 1).
It is essential to have a low threshold of suspicion to
diagnose CNF in its early stages. Patients usually complain
of pain that is out of proportion to the clinical finding
which can be attributed to associated neuropathy of the
infected area [5].
Wang et al. [16] have staged the disease progression into
three stages based on the cutaneous signs. Stage I has ten-
derness, erythema, swelling, and hotness. Stage II includes
blister or bullae formation. Stage III involves crepitus, skin
anesthesia and necrosis. Clinical data in this retrospective
study were not enough to classify our patients according to
the former proposed staging system.
Although the median time of duration of symptoms in
our study was four days, crepitus was not palpated in any of
our patients. This is probably due to gas formation in areas
inaccessible to accurate palpation or due to difficulty in
examining the patient because of severe tenderness. Systemic
signs of fever, tachycardia, tachypnea, and confusion were
uncommon. This is possibly because of the widespread use
of broad-spectrum antibiotics and the weak immunologic
response to infection in diabetic patients [16].
The use of CT scan may help in early diagnosis of CNF
and its complications such as mediastinitis. Although a nor-
mal CT scan cannot completely rule out necrotizing fasciitis,
certain alerting findings increase the likelihood of necrotiz-
ing fasciitis such as fat stranding and the presence of fluid
and gas tracking through the fascial planes [14,17]. CT scan
played a major role in making the correct diagnosis in our
patients. It was superior to digital palpation in detecting
subcutaneous emphysema, and it gave clear indications for
acute surgical intervention. With recent emerging evidence,
magnetic resonance imaging (MRI) is the most effective
imaging modality in the diagnosis of necrotizing fasciitis
[18,19]. It is useful for differentiating necrotizing fasciitis
from non-necrotizing fasciitis and for avoiding unnecessary
surgical interventions. The presence of deep fascial thicken-
ing of more than 3 mm on fat-suppressed T2 weighted
images associated with multiple musculofascial compart-
ments involvement are important MRI findings for early
diagnosis of necrotizing fasciitis [19]. None of our patients
had a MRI.
Similar to others, the microbiology of CNF in our
patients involved multiple organisms, including anaerobic
bacteria [3,7,12].
The mainstay of CNF treatment involves early surgical
exploration with aggressive debridement of necrotic tissue
and empiric broad-spectrum antibiotics followed by sensitiv-
ity directed antibiotics [7,13,14]. Bahu et al. [5] advocated
repeat wound exploration and debridement for a better out-
come. Repeated debridement was performed in 50% of our
patients, which is lower than other studies [5,17]. This could
be attributed to early and radical debridement of necrotic
tissues at the primary operation.
Hyperbaric oxygen treatment has been recently used as
an adjunctive therapy in reducing the mortality rate among
patients suffering from CNF [1,3,20]. We do not have yet
 4 M. A. AL-ALI ET AL.

this facility at our hospital although it is planned to be [4]
available.
Similar to others, complications of CNF included media- [5]
stinitis, septicemia, and multiple organ failures [1,7]. One
patient developed multi-organ failure and perforated duode-
num related to severe sepsis. The death rate of necrotizing [6]
fasciitis, if not treated, is 100%. Factors associated with
increased mortality include comorbidity, mediastinitis, and [7]
vascular complications [1,7]. The overall death rate was
16.7% in our study which is similar to others [5,20]. The
key to a successful clinical outcome consists of timely diag- [8]
nosis, early resuscitation, broad-spectrum antimicrobial ther-
apy, and aggressive surgical debridement [12–15]. [9]
We have to acknowledge that our study has certain limi-
tations. This study is a single institution study with a small
[10]
sample size. Given the rarity of the condition, it will be diffi-
cult to conduct a prospective study within a specified time
frame. Nevertheless, developing an international specific
Downloaded by [University of Florida] at 22:28 08 November 2017
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neck: 34 cases of a single institution experience. Eur Arch
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Surg. 2001;125:245–252.
Kihiczak GG, Schwartz RA, Kapila R. Necrotizing fasciitis: a
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Mohammedi I, Ceruse P, Duperret S, et al. Cervical necrotizing
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Available from: https://www.scad.ae/Release%20Documents/SYB-
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registry and prospectively collecting data on these patients
can be useful. Due to the retrospective design of this study,
some of the required data were missing like the staging of
the disease.
In conclusion, CNF is a rare lethal condition.
Maintaining a high index of suspicion is crucially important.
Early diagnosis and resuscitation, and timely aggressive sur-
gical debridement are key elements to a successful clinical
outcome for managing CNF.
Disclosure statement
The authors have no conflicts of interest. The authors alone are
responsible for the content and writing of the paper.
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