Necrotizing fasciitis secondary to peritonsillar abscess: A new case and review of eight earlier cases. (Original Article).

Abstract

Necrotizing fasciitis is a potentially fatal soft-tissue infection that occurs only rarely in the head and neck region. Broad-spectrum parenteral antibiotics and surgical debridement are the mainstays of treatment. Until now, only eight cases of necrotizing fasciitis secondary to peritonsillar abscess have been described in the English-language literature. In this article, we report a new case that occurred in an otherwise healthy 43-year-old woman. In addition to standard treatment, the patient underwent a hot tonsillectomy. After 23 months of follow-up, she is in good health.

Introduction

Only eight previous cases of necrotizing fasciitis secondary to peritonsillar abscess have been reported in the literature (table 1). (1-7) Necrotizing fasciitis is a soft-tissue infection that causes extensive necrosis of the fascia and subcutaneous tissue, as well as gas formation. The skin and muscle are spared initially.

In 1871, Joseph Jones published the first description of this disease, which he called hospital gangrene, and he referenced more than 2,600 cases. (8) In 1952, Wilson first used the term necrotizing fasciitis, which is the most accurate description of this disease. (9) The most common cause is dental infection; other causes are maxillofacial trauma, peritonsillar and pharyngeal abscess, and osteoradionecrosis. Peritonsillar abscesses can spread to parapharyngeal and retropharyngeal areas, as well as to the mediastinum, and they can lead to airway obstruction, sepsis, thrombophlebitis, and rupture of the great vessels. (6)

Necrotizing fasciitis of the face or neck is extremely rare; the high degree of vascularity in this region means that these tissues are not very susceptible to ischemia and infection. (10) Until now, only eight cases of necrotizing fasciitis resulting from a peritonsillar abscess have been reported in the English-language literature; only five of these patients survived (table 1). None of these cases occurred in our country.

In this article, we report a new case of necrotizing fasciitis of the head and neck that developed as a sequela to peritonsillar abscess. We also discuss the use of intravenous broad-spectrum antibiotic therapy, prompt surgical debridement, and abscess tonsillectomy, and we review the previously published cases.

Case report

A 43-year-old woman sought treatment from a general practitioner for a sore throat. She was given a diagnosis of tonsillitis and an intramuscular injection of penicillin. Three days later, she developed right cervical swelling, and the physician referred her for hospitalization. She was admitted under the care of the ENT department with a diagnosis of a right peritonsillar abscess and a deep cervical infection.

Physical examination revealed that the patient had a noncrepitant, nonfluctuant, erythematous submandibular and facial edema, along with a spontaneous fistula through the upper pole of the tonsillar area on the right. Her body temperature, blood pressure, and pulse rate were normal. She was started on parenteral antibiotics (intravenous penicillin, 4 MU six times daily, and intramuscular chloramphenicol, 500 mg four times daily), and her peritonsillar abscess was drained. The spontaneous fistula had widened, and between 3 and 4 ml of purulent material was drained. Povidone-iodine gargle was added to the treatment regimen. Initially, needle aspiration of the neck did not detect any abscess.

The results of the hemogram, biochemical liver and kidney function tests, and electrolyte measurements revealed that most parameters were normal. Her serum glucose and urea levels were slightly beyond the upper limit, but her white blood cell count was 20,700/[mm.sup.3], almost double the upper limit of normal (normal range: 4,800 to 10,800). Results of the first culture analysis of the drainage from the spontaneous fistula revealed the presence of group A beta-hemolytic streptococci, which were sensitive to penicillin. Her leukocyte count remained high during the first 3 days of hospitalization.

On the 3rd day of hospitalization, a fluctuation developed over the submandibular region, and approximately 25 ml of brownish pus was drained through a 2-cm submandibular neck incision. The drained material was negative on culture. On the 4th day, the patient's white cell count fell to 11,400/[mm.sup.3]. The drainage area in the tonsillar plica remained open. The patient also experienced a hemorrhage in the right tonsillar bed, which was initially controlled with a tampon. When the hemorrhage recurred, the patient underwent an urgent abscess tonsillectomy under general anesthesia.

The right tonsil was excised, thin purulent material was drained, and coagula, thrombotic vessels, and necrotic tissue were removed from the parapharyngeal space through the tonsillar bed. The major vessels remained intact. Another incision was made in the submental region, and approximately 50 ml of foul-smelling pus was drained. Culture analysis detected the presence of coagulase-negative staphylococci and enterococci, both of which were sensitive to chloramphenicol and vancomycin. The patient was switched from intravenous penicillin to vancomycin at 500 mg four times daily.

Despite the tonsillectomy, the continuous submandibular drainage, and the intravenous antibiotic treatment, the patient's condition deteriorated. The infection had spread to the lower neckandpectoral region. A massive, erythematous, brawny, and crepitant edema developed on the 7th day of hospitalization. Consultation with both the departments of general surgery and infectious disease led to a diagnosis of necrotizing fasciitis.

The patient was taken back to the operating room for exploration of the neck under general anesthesia. The submandibular incision was extended over the clavicle and down to the level of the nipple vertically and the axilla horizontally. The incisions caused only minimal bleeding, and the skin flaps were easily elevated. A relatively large amount of thin, brown, foul-smelling pus was drained. Some portions of the platysma and the stemocleidomastoid and strap muscles were black, and the external jugular vein was thrombotic. Dead tissue extended from the mandible and mastoid tip to the level of the fourth rib and 3 to 4 cm back to the retrostemal region; necrotic tissue also extended deep to the parapharyngeal space and was connected to the right tonsillar bed. All necrotic issue--including muscle, numerous thrombotic vessels, adipose tissue, fascia, the tail of the parotid, and the submandibular gland--were removed. The surgical area was washed with saline solution and rifamycin. The open wound was packed with gauze soaked with 10% povidone-iodine, rifamycin, and nitrofurazone.

Postoperatively, nutrition was delivered via a nasogastric route. Complete blood count, glucose, urea, albumin, and electrolyte levels were measured daily. When required, albumin and electrolytes were supplied intravenously. When the hemoglobin level fell below 8 g/dl, 2 units of blood were administered. The wound dressings were changed twice daily, but on the sixth day of hospitalization, granulation tissue was observed. The patient was again taken to the operating room, and the pectoral and lower neck flaps were closed. During this procedure, the patient spontaneously began to bleed profusely from the oral cavity and nose. The oral cavity was immediately explored, and the source of the bleeding was determined to be a branch of the external carotid artery (probably the lingual artery) and the tonsillar bed. The hemorrhage was wadded, and the bleeding vessel was clamped and ligated.

Open-wound dressings, intravenous antibiotics, and supportive treatment were continued. The skin flaps were closed gradually (primarily by suturing) until the entire wound was closed. The patient was discharged on the 28th hospitalization day with no other complication. At the 23-month follow-up, the patient remains in good health.

Discussion

The cornerstone of the proper management of necrotizing fasciitis includes prompt diagnosis, broad-spectrum antibiotics, aggressive surgical debridement, and intense supportive care. (6,11,12)

Among the nine patients (mean age: 4l [+ or -] 6 yr) with necrotizing fasciitis secondary to peritonsillar abscess who have now been described in the English-language literature, there were eight men and one woman (table 1). Infection arose from the left tonsil in five patients and from the right in four. All nine cases involved the neck, five involved the anterior chest wall, five involved the parapharyngeal space, four involved the mediastinum (one of these cases was very advanced(1)), and three involved the skull base.

Clinical characteristics. The course of necrotizing fasciitis is usually rapidly progressive. The disease can spread into the chest wall and mediastinum, as it did in our patient, and put the patient at risk of death. (13) The onset of symptoms usually occurs 2 to 4 days following tonsillar abscess. The presence of sudden pain and erythematous and edematous skin can be misdiagnosed as cellulitis or erysipelas. Clinically, it is not unusual to find a low-grade fever, anemia, jaundice from bacterial hemolysis, and tissue crepitation as a result of gas formation. Hyponatremia, hypoproteinemia, hypocalcemia, and dehydration can also be detected in severe cases. Evaluation should involve routine blood tests--including daily measurements of serum glucose, urea, and electrolytes--in order to detect the metabolic abnormalities previously mentioned. Computed tomography is the most valuable study for detecting gas in areas that are inaccessible to palpation, for identifying any extension of infection preoperatively, an d for detecting vascular thrombosis, erosion of vessels, and mediastinitis. Plain soft-tissue films of the chest are also helpful in detecting mediastinitis.

It is also important to realize that this devastating infection can occur spontaneously, and it should at least be considered in patients who have unexplained softtissue pain and tenderness. When treatment is delayed or inadequate, many patients with necrotizing fasciitis do not survive. (7) To prevent the high risk of mortality and morbidity associated with cervicofacial necrotizing fasciitis, early recognition and treatment are essential. (14) In the nine patients with necrotizing fasciitis discussed in this article, the mean time (when known) between diagnosis and surgical therapy was 10 days ([+ or -]4). Much of this time had passed before these patients had been admitted to a fully equipped hospital. Three of the nine patients died (mortality rate: 33%), a mortality rate that is much lower than the reported 60% rate among patients with necrotizing fasciitis caused by other etiologies. (14)

Causative organisms. Necrotizing fasciitis was once thought to be the result of a purely streptococcal infection, but improvements in culture techniques eventually led to the discovery that it is caused by a polymicrobial infection. Anaerobes account for approximately 30% of the bacteriology; the most common are Bacteroides and Peptostreptococcus spp. (11,12) Other frequently cultured organisms are Staphylococcus spp., non-group A streptococci, Escherichia coil, Kiebsiella pneumoniae, Pseudomonas aeruginosa, and Citrobacter and Enterobacter spp. Specimens of both blood and the wound should be sent for routine and anaerobic cultures. In our patient, group A streptococci were initially cultured from the peritonsillar abscess; after the neck incisions were made for surgical debridement, coagulase-negative staphylococci and enterococci were cultured from the necrotic tissues. Among the nine cases overall, culture detected aerobes in eight patients (89%), streptococci in seven (78%), gram-negative micro-organisms in six (67%), Bacteroides spp. in five (56%), and staphylococci in three (33%) (table 2).

Antibiotic therapy. Antibiotic therapy must be directed against gram-positive and gram-negative aerobic and anaerobic organisms, and the selection of the specific antibiotic should be based on the results of cultures and sensitivity tests. Penicillin is the drug of choice for group A, B, C, G, and H streptococci, alpha-hemolytic streptococci, and most Clostridium spp. Clindamycin is adequate for patients with penicillin-resistant anaerobes, and an aminoglycoside should be used to cover gram-negative bacilli. Our review of the nine cases suggests that antibiotic therapy against gram-positive and -negative aerobes and anaerobes should be started empirically and continued until the results of a culture antibiogram are available.

Surgical debridement. The need for antibiotics notwithstanding, the most important aspect of the treatment of necrotizing fasciitis is debridement of all necrotic tissue. Immediate surgical exploration is indicated in the presence of subcutaneous emphysema (the presence of gas or air in subcutaneous tissues), rapidly advancing infection despite 24 to 48 hours of medical therapy, obvious fluctuation, or skin necrosis in an area of cellulitis. The most important point to remember is that fascia necrosis usually extends further than cutaneous involvement.

In our patient, tonsillectomy was performed because of the tonsillar hemorrhage and the suspicion of a parapharyngeal abscess. Surgical debridement was then performed on the neck and chest wall down to the level of the bottom of the breasts. Retrosternal transcervical mediastinotomy was also performed because of the upper mediastinal involvement. Neck debridement was performed on all nine patients (100%), drainage of the peritonsillar abscess on eight (89%), and upper mediastinal debridement on three (33%). In one of the nine cases, hyperbaric oxygen therapy was administered as an addition to extensive surgical debridement and antibiotic treatment. (5) In the nine cases, ours was the only one in which tonsillectomy was performed to treat the peritonsillar abscess.

Acknowledgments

The authors extend special thanks to Rahmi Kilic, Erdal Samim, and Cavit Ozeri of the ENT Clinic for their assistance in managing our patient and to Nilgun Acar of the Microbiology Laboratory for classifying the microorganisms listed in table 2.

References

(1.) Wills PI, Vernon RP, Jr. Complications of space infections of the head and neck, Laryngoscope 1981;91:1129-36.

(2.) Wenig BL, Shikowitz MJ, Abramson AL. Necrotizing fasciitis as a lethal complication of peritonsillar abscess. Laryngoscope 1984;94:1576-9.

(3.) Tovi F, Fliss DM, Zirkin HJ. Necrotizing soft-tissue infections in the head and neck: A clinicopathological study. Laryngoscope 1991;101:619-25.

(4.) Scott PM, Dhillon RS, McDonald PJ. Cervical necrotizing fasciitis and tonsillitis. J Laryngol Otol 1994;108:435-7.

(5.) Jackson BS, Sproat JE. Necrotizing fasciitis of the head and neck with intrathoracic extension. I Otolaryngol 1995;24:60-3.

(6.) Greinwald JH, Jr., Wilson JF, Haggerty PG. Peritonsillar abscess: An unlikely cause of necrotizing fasciitis. Ann Otol Rhinol Laryngol 1995;104:133-7.

(7.) Hadfield PJ, Motamed M, Glover GW. Synergistic necrotizing cellulitis resulting from peri-tonsillar abscess. J Laryngol Otol 1996;110:887-90.

(8.) Balcerak RJ, Sisto JM, Bosack RC. Cervicofacial necrotizing fasciitis: Report of three cases and literature review. J Oral Maxillofac Surg 1988;46:450-9.

(9.) Wilson B. Necrotizing fasciitis. Am Surg 1952;18:416-31.

(10.) Sepulveda A, Sastre N. Necrotizing fasciitis of the face and neck. Plast Reconstr Surg 1998;102:814-7.

(11.) Kantu S. Har-El G. Cervical necrotizing fasciitis. Ann Otol Rhinol Laryngol 1997; 106:965-70.

(12.) Raboso E, Llavero MT, Rosell A, Martinez-Vidal A. Craniofacial necrotizing fasciitis secondary to sinusitis. J Laryngol Otol 1998;112:371-2.

(13.) Klabacha ME, Stankiewicz JA, Clift SE. Severe soft tissue infection of the face and neck: A classification. Laryngoscope 1982;92:1135-9.

(14.) Roberson JB, Harper JL, Jauch EC. Mortality associated with cervicofacial necrotizing fasciitis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 199 6;82:264-7.

Table 1.

Clinical summary of the nine reported cases of necrotizing fasciitis
secondary to peritonsillar abscess

          Patient/                          Age,
           author                        sex, side

1. Wills and Vernon, 1981 (1)              35/M/R









2. Wenig et al. 1984 (2)                   50/M/L








3. Wenig et al. 1984 (2)                   50/M/L








4. Tovi et al, 1994 (3)                    20/M/L




5. Scott et al, 1994 (4)                   51/M/R














6. Jackson and Sproat, 1995                63/M/R
 (5)





7. Greinwald et al, 1997 (6)               65/M/L








8. Hadfield et al, 1996 (7)                53/M/L




9. Safak et al, 2001 (ss)                  43/F/R








          Patient/
           author                        Infection            Days (*)

1. Wills and Vernon, 1981 (1)  R pharyngomaxillary space; R      11
                               skull base; R neck;
                               suprasternal bilateral
                               empyema; pericarditis; chest
                               wall abscess





2. Wenig et al. 1984 (2)       L neck; L anterior chest wall     ?
                               to the level of the nipple







3. Wenig et al. 1984 (2)       L parotid region; L neck to       14
                               the level of the clavicle;
                               anterior chest wall






4. Tovi et al, 1994 (3)        L neckl mediastinitis; lung       ?
                               abscess; meningitis
                               meningitis


5. Scott et al, 1994 (4)       R neck from the temple to the     12
                               clavicle and deep to the
                               pharynx; R parotid; R
                               pectoral level of the nipple











6. Jackson and Sproat, 1995    Neck; superior mediastinum to     ?
 (5)                           the level of the aortic arch
                               and aorticopulmonary window




7. Greinwald et al, 1997 (6)   L facial; L skull base;           4
                               parapharyngeal space;
                               belateral cervical; anterior
                               chest wall to the level of
                               the second rib; external
                               jugular vein thrombosis; L
                               sternocleidomastoid and
                               strap-muscle necrosis

8. Hadfield et al, 1996 (7)    L facial, L skull base, and       ?
                               bilateral upper cervical
                               regions from the mandible to
                               the retropharngeal space

9. Safak et al, 2001 (ss)      R neck from the mandible and      7
                               mastoid tip to the level of
                               the nipple on the chest wall
                               and to the axilla, back to
                               the retrosternal region 3 to
                               4 cm; R parapharyngeal space



          Patient/
           author                  Wound culture results

1. Wills and Vernon, 1981 (1)  Bacteroides melaninogenicus;
                               Stenotrophomonas maltophilla;
                               alpha-hemolytic streptococci







2. Wenig et al. 1984 (2)       Hemolytic streptococci;
                               Fusobacterium; nucleatum;
                               B melaninogenicus;
                               Eubacterium; Enterobacter
                               cloace; hemolytic
                               Staphylococcus epidermidis;
                               Klebsiella spp.; Pseudomonas
                               aeruginosa

3. Wenig et al. 1984 (2)       Alpha-hemolytic streptococci;
                               Escherichia coli;
                               Streptococcus mitis;
                               Streptococcus sanguis;
                               Streptococcus faecalis; B
                               melaninogenicus; Bacteroides
                               fragilis; Corynebacterium
                               spp.

4. Tovi et al, 1994 (3)        Enterobacter spp.; Proteus
                               mirabilis; klebsiella
                               pneumoniae


5. Scott et al, 1994 (4)       Streptococcus milleri;
                               nonhemolytic streptococci;
                               unknown anaerobes












6. Jackson and Sproat, 1995    E lentum; P anaerobius; B
 (5)                           fragilis





7. Greinwald et al, 1997 (6)   Group A beta-hemolytic
                               streptococci initially,
                               then S epidermidis






8. Hadfield et al, 1996 (7)    S milleri; two Bacteroides
                               spp.



9. Safak et al, 2001 (ss)      Group A beta-hemolytic
                               streptococci; coagulase-
                               negative staphylococci and
                               enterococci





          Patient/                       Other
           author                       disease

1. Wills and Vernon, 1981 (1)  None reported









2. Wenig et al. 1984 (2)       Diabetes








3. Wenig et al. 1984 (2)       Diabetes; hypertension;
                               angina pectoris







4. Tovi et al, 1994 (3)        None reported




5. Scott et al, 1994 (4)       None reported














6. Jackson and Sproat, 1995    None reported
 (5)





7. Greinwald et al, 1997 (6)   Diabetes; hypercnolester-
                               olemia hypertension;
                               coronary artery disease






8. Hadfield et al, 1996 (7)    None reported




9. Safak et al, 2001 (ss)      None








          Patient/
           author                   Antibiotic treatment

1. Wills and Vernon, 1981 (1)  [BC.sup.+]: penicillin (oral,
                               IM, IV); [AC.sup.+]: IV
                               chioramphenicol; IM
                               gentamicin






2. Wenig et al. 1984 (2)       BC: penicillin (oral, IV);
                               gentamicin; clindamycin; AC:
                               same as BC






3. Wenig et al. 1984 (2)       BC: cefazolin initially,
                               then switch to penicillin;
                               gentamicin; chloram-
                               phenicol; AC: penicillin;
                               amikacin; metronidazole;
                               cefotaxime



4. Tovi et al, 1994 (3)        ?




5. Scott et al, 1994 (4)       BC: IV cefotaxime,
                               flucloxacillin, and
                               metronidazole; AC: IV
                               penicillin and
                               metronidazole










6. Jackson and Sproat, 1995    BC: penicillin G; ceftazidime;
 (5)                           metronidazole; AC: imipenem;
                               metronidazole




7. Greinwald et al, 1997 (6)   BC: IV ampicillin; AC:
                               initially IV clindamycin,
                               ampicillin/sulbactam, and
                               tobramycin, then IV
                               imipenem and clindamycin;
                               discharged on oral
                               ciprofloxacin


8. Hadfield et al, 1996 (7)    BC: IV cefotaxime and
                               metronidazole; AC: imipenem;
                               imipenem; penicillin
                               metronidazole; gentamicin

9. Safak et al, 2001 (ss)      BC: penicillin (IM, IV); IM
                               chloram-phenicol; AC: IV
                               vancomycin; IM
                               chloramphenicol





          Patient/             Surgical approach and type of
           author                      reconstruction

1. Wills and Vernon, 1981 (1)  Peritonsillar abscess (PTA)
                               drainage via R
                               submandibular and cervical
                               approach; closed R tube
                               thoracotomy;
                               pericardiocentesis;
                               pericardiectomy; L chest
                               drainage via open mediastinal
                               approach

2. Wenig et al. 1984 (2)       PTA drainage; L neck and
                               chest wall debridement via
                               multiple stepladder incisions
                               to the level of the nipples
                               (twice)




3. Wenig et al. 1984 (2)       PTA drainage; L neck and
                               anterior chest wall
                               debridement (twice)






4. Tovi et al, 1994 (3)        PTA drainage; local excision
                               and debridement;
                               transcervical mediasti-
                               notomy

5. Scott et al, 1994 (4)       PTA drainage; excision of
                               necrotic skin, parotid, facia
                               facial nerve,
                               submandibular gland, and angle
                               of the mandible; debridment
                               of necrotic muscles
                               and infratemporal fossa;
                               crossed facial nerve grafting
                               with sural nerve; mandibular
                               reconstruction
                               with nonvascularized rib
                               graft; free latissimus dorsi
                               myocutaneous grafting and
                               split-thickness skin grafting

6. Jackson and Sproat, 1995    PTA drainage; neck exploration
 (5)                           from the mandible to the
                               manubrium; super mediastinal
                               approach through the
                               retrosternal region;
                               hyperbaric oxygen the therapy

7. Greinwald et al, 1997 (6)   PTA drainage; surgical
                               debridement (twice); split-
                               thickness skin grafting






8. Hadfield et al, 1996 (7)    Tonsillectomy; surgical
                               debridment (five times); skin
                               grafting after 2 wk


9. Safak et al, 2001 (ss)      PTA drainage; R submandibular
                               drainage; R tonsillectomy;
                               intraoral R parapharyngeal
                               debridment; extended
                               cervical debridement
                               to the level of
                               the nipple; transcervical
                               mediastinotomy

          Patient/              Duration
           author              of therapy  Outcome

1. Wills and Vernon, 1981 (1)  >2mo        Survived









2. Wenig et al. 1984 (2)       9 days      Died








3. Wenig et al. 1984 (2)       11 days     Died








4. Tovi et al, 1994 (3)        ?           Died




5. Scott et al, 1994 (4)       >3mo        Survived














6. Jackson and Sproat, 1995    5 wk        Survived
 (5)





7. Greinwald et al, 1997 (6)   31 days     Survived
                                           with
                                           dysphonia






8. Hadfield et al, 1996 (7)    2 to 3 wk   Survived




9. Safak et al, 2001 (ss)      28 days     Survived








(*)Duration of disease prior to surgery.

(+)BC=before culture;

AC=after culture.

(ss)Present study.
Table 2. Bacteriology in the nine cases

Aerobes

Gram-positive                   Gram-negative

Streptococci

Alpha-hemolytic streptococci    Enterobacter cloacae (pt. 2)
(pts. 1 and 3)                  Enterobacter spp. (pt. 4)
Group A beta-hemolytic          Escherichia coli (pt. 3)
streptococci (pts. 7 and 9)     Klebsiella pneumoniae (pt. 4)
Hemolytic streptococci (pt. 2)  Klebsiella spp. (pt. 2)
Streptococcus faecalis (pt. 3)  Proteus mirabilis (pt. 4)
Streptococcus milleri           Pseudomonas aeruginosa (pt. 2)
(pts. 5 and 8)                  Stenotrophomonas maltophilia
Streptococcus mitis (pt. 3)     (pt. 1)
Streptococcus sanguis (pt. 3)
Nonhemolytic streptococci
(pt. 5)

Staphylococci

Coagulase-negative
staphylococci (pt. 9)
Hemolytic Staphylococcus
epidermidis (pt. 2)
Staphylococcus epidermidis
(pt. 7)

Other

Corynebacterium spp. (pt. 3)
Enterococcus spp. (pt. 9)

Anaerobes

Gram-positive                   Gram-negative

Eubacterium lentum              Bacteroides fragilis
(pts. 2 and 6)                  (pts. 3 and 6)
Peptostreptococcus anaerobius   Bacteroides melaninogenicus
(pts. 2 and 6)                  (pts. 1, 2, and 3)
                                Bacteroides spp. (pt. 8)
                                Fusobacterium nucleatum
                                (pt. 2)