Acta Oto-Laryngologica

ISSN: 0001-6489 (Print) 1651-2251 (Online) Journal homepage: http://www.tandfonline.com/loi/ioto20

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

were identified on culture in five patients. One specimen process although four of them (66.7%) had mediastinitis
yielded no growth, probably due to previous antibiotic ther- (Table 1).
apy or improper collection of specimen (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
Management and outcome which can be attributed to associated neuropathy of the
All patients received broad-spectrum antibiotics on admis- infected area [5].
sion. They underwent surgical exploration and aggressive Wang et al. [16] have staged the disease progression into
debridement of the affected area (Figure 1(B)). Surgery was three stages based on the cutaneous signs. Stage I has ten-
carried out within 24 h of admission in two patients derness, erythema, swelling, and hotness. Stage II includes
(33.3%). Repeated surgical debridement was performed in blister or bullae formation. Stage III involves crepitus, skin
three patients (50%) (Table 1). Histopathological examin- anesthesia and necrosis. Clinical data in this retrospective
ation of the excised tissues was performed in two patients study were not enough to classify our patients according to
and confirmed the diagnosis of CNF. the former proposed staging system.
Four patients (66.7%) developed superior mediastinitis, Although the median time of duration of symptoms in
two (33.3%) had septicemia, and one (16.7%) had perforated our study was four days, crepitus was not palpated in any of
duodenum and multi-organ failure. The mediastinum was our patients. This is probably due to gas formation in areas
inaccessible to accurate palpation or due to difficulty in
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explored and debrided through the transcervical approach at

the time of initial surgery in three patients and through a examining the patient because of severe tenderness. Systemic
thoracotomy in one patient. signs of fever, tachycardia, tachypnea, and confusion were
Four patients (66.7%) were admitted to the intensive care uncommon. This is possibly because of the widespread use
unit. Data regarding the length of hospital stay was missing of broad-spectrum antibiotics and the weak immunologic
in two patients, but the other four patients had a median response to infection in diabetic patients [16].
The use of CT scan may help in early diagnosis of CNF
(range) hospital stay of 41 days (11-103). One patient died
and its complications such as mediastinitis. Although a nor-
(overall mortality was 16.7%). This patient had a perforated
mal CT scan cannot completely rule out necrotizing fasciitis,
duodenal ulcer, septicemia, and multi-organ failure.
certain alerting findings increase the likelihood of necrotiz-
ing fasciitis such as fat stranding and the presence of fluid
Discussion and gas tracking through the fascial planes [14,17]. CT scan
played a major role in making the correct diagnosis in our
Necrotizing fasciitis of the cervical region is uncommon.
patients. It was superior to digital palpation in detecting
Y-M Liu et al. [11] reported a series of 87 cases of necrotiz-
subcutaneous emphysema, and it gave clear indications for
ing fasciitis and found only five involving the head and neck acute surgical intervention. With recent emerging evidence,
(6%). We have a higher percentage of CNF. Six out of 55 magnetic resonance imaging (MRI) is the most effective
patients (10.9%) of necrotizing fasciitis who were treated at imaging modality in the diagnosis of necrotizing fasciitis
our hospital during the same period were in the neck [18,19]. It is useful for differentiating necrotizing fasciitis
(Hefny et al., unpublished data). CNF predominantly affects from non-necrotizing fasciitis and for avoiding unnecessary
men in their fourth decade of life. The median age of our surgical interventions. The presence of deep fascial thicken-
patients was 47 years which is similar to other studies ing of more than 3 mm on fat-suppressed T2 weighted
[11–15]. images associated with multiple musculofascial compart-
Although necrotizing fasciitis can affect healthy individu- ments involvement are important MRI findings for early
als, several predisposing factors like obesity, diabetes melli- diagnosis of necrotizing fasciitis [19]. None of our patients
tus, chronic renal failure, and immunosuppression have had a MRI.
been implicated in its occurrence [12,13]. Similar to others, Similar to others, the microbiology of CNF in our
we have shown that diabetes mellitus is the most common patients involved multiple organisms, including anaerobic
disease associated with CNF [4,11,14]. Although the exact bacteria [3,7,12].
cause of CNF is not always clear, the present study has The mainstay of CNF treatment involves early surgical
shown that it is commonly odontogenic, followed by pha- exploration with aggressive debridement of necrotic tissue
ryngeal origin. This finding is like other studies [5,9,15]. and empiric broad-spectrum antibiotics followed by sensitiv-
The submandibular triangle, which is the major lymphatic ity directed antibiotics [7,13,14]. Bahu et al. [5] advocated
drainage area from the surrounding source, was affected in repeat wound exploration and debridement for a better out-
all our patients. come. Repeated debridement was performed in 50% of our
Because of the nonspecific initial signs and symptoms of patients, which is lower than other studies [5,17]. This could
CNF, misdiagnosis is common [14]. Patient’s presentation be attributed to early and radical debridement of necrotic
ranges from mild localized soft tissue infection to severe tissues at the primary operation.
deep neck infection and thoracic extension. The diagnosis of Hyperbaric oxygen treatment has been recently used as
CNF was initially missed in all our patients. Majority, at an adjunctive therapy in reducing the mortality rate among
presentation, were diagnosed to have a localized infectious patients suffering from CNF [1,3,20]. We do not have yet
 4 M. A. AL-ALI ET AL.

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