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QUERESHY ET AL
0278-2391/09/6702-0028$36.00/0
doi:10.1016/j.joms.2008.07.017
Report of a Case
The following patient was a 52-year-old female with a
past medical history significant for diabetes, alcoholism,
tobacco smoking, hypertension, and status post-Whipple
procedure in 1993 for a benign pancreatic cyst. She was
found confused and lethargic in her home by her daughter, and was transported to a nearby outside hospital by
emergency medical services. A computed tomography
(CT) scan at that time showed significant subcutaneous
emphysema of the anterior neck and mediastinum tracking along the great vessels (Fig 1). Through discussion
with the family, it was found out that over the previous
week the patient had been complaining of some lower
jaw and tooth pain, and was started on amoxicillin by her
primary care physician. She eventually presented to a
dentist who noted that her lower right wisdom tooth
should be extracted secondary to decay, and she was to
remain on antibiotics. Due, at least in part, to her lack of
insurance, she delayed subsequent treatment. Initial laboratory data were significant for a normal white blood
cell count with substantial neutrophil bandemia of 39%.
There was a marked increase in creatine kinase of 1,454
U/L (cf. 0-215 U/L reference range). Her metabolic panel
showed that she was hyperglycemic (435 mg/dL), with
an elevated blood urea nitrogen of 50 mg/dL. She was
febrile, with 38.8C and tachycardic. By the time consultation to our service was made, the patient was intubated
and in the medical intensive care unit. She was on anti-
420
FIGURE 1. Composite of CT axial images with intravenous contrast. Note profound tissue emphysema from inferior spread of the infection.
Infection is also noted around the great vessels.
Quereshy et al. Unique Case of Cervicothoracic Necrotizing Fasciitis. J Oral Maxillofac Surg 2009.
FIGURE 2. Preoperative photograph of patients gross appearance. There is significant duskiness and erythema along the maxillofacial and cervicothoracic area.
Quereshy et al. Unique Case of Cervicothoracic Necrotizing Fasciitis. J Oral Maxillofac Surg 2009.
Quereshy et al. Unique Case of Cervicothoracic Necrotizing Fasciitis. J Oral Maxillofac Surg 2009.
421
QUERESHY ET AL
Demographics:
n
Mean age
Gender male:female
Comorbidities % (n):
Smoking
Alcoholism
Diabetes
HIV
Elderly (65 years)
Malignancy
Other
Etiology % (n):
Odontogenic
Surgery/trauma
Tonsillar
Tung-Yiu et al
(2000)1
Ali et al
(1997)2
Whitesides et al
(2000)3
Lin et al
(2001)4
Umeda et al
(2003)5
Bahu et al
(2001)6
11
60.5
7:4
ND
ND
36% (4)
0
18% (2)
9% (1)
36% (4)
3
56
1:2
ND
ND
33% (1)
0
33% (1)
11
47
7:4
ND
36% (4)
45% (5)
18% (2)
36% (4)
47
57
39:8
49% (23)
9
52.5
3:6
ND
ND
22% (2)
0
22% (2)
0
10
46
7:3
70% (7)
70% (7)
20% (2)
0
0
0
100% (11)*
33% (1)
66% (2)
100% (11)*
100% (9)*
80% (8)
20% (2)
26% (12)
72% (34)
0
47% (22)
13% (6)
11% (5)
36% (17)
Literature Review
The literature was searched for publications over
the last decade reporting encounters of CNF. Ninetyone cases from 6 different institutions were identified.1-6
Discussion
We reported a case of CNF that was secondary to
an odontogenic infection. Without any doubt, the
patients prognosis was worsened by the fact that she
presented with poorly controlled diabetes mellitus,
alcoholism, and tobacco abuse. The avoidance of definitive care until there was systemic involvement (ie,
422
Table 2. IDENTIFIED ORGANISMS AND PROGNOSIS IN REPORTED CASES OF CERVICAL NECROTIZING FASCIITIS
Cultures:
Streptococcus
-hemolytic strep
-hemolytic strep
gp D streptococcus
Peptostreptococcus
Staphylococcus sp.
Eikenella sp.
Prevotella sp.
Major complication(s):
Mediastinitis
Septic shock
DIC
Pleural effusion
Hospital stay (mean days):
Mortality
Tung-Yiu et al
(2000)1
Ali et al
(1997)2
Whitesides et al
(2000)3
82% (9)
27% (3)
100% (3)
33% (1)
4
2
2
Lin et al
(2001)4
13%
15%*
Umeda et al
(2003)5
Bahu et al
(2001)6
78% (7)
55% (5)
33% (3)
100% (10)
20% (2)
60% (6)
11% (1)
20% (2)
60% (6)
64% (7)
2
4
ND
36% (4)
18% (2)
9% (1)
18% (2)
31.6
18% (2)
33.3% (1)
42
33.3% (1)
0
13% (6)
36% (17)
31
0%
26.4
25.5%
59% (5)
33.3
0%
24
10%
she was found down) was also more likely than not to
have complicated the outcome.
The literature review substantiated the findings in our
patient (Table 1). Namely, that diabetes is a known
predisposing factor for CNF. Presumably this is due to
the defect in leukocyte function brought about by
chronic hyperglycemia. There is a clear association between alcoholism, tobacco abuse, and CNF. It is unclear
if this is a direct effect of these agents or a consequence
of the malnutrition and poorer general health often
observed in these patients. In terms of patients with
overt immunosuppression (eg, HIV-positive), it is clear
that these patients make up a small proportion of the
overall number of CNF cases. Whether this is from a
more acute awareness of HIV-infected patients toward
dental care, the placement of many of these patients on
prophylactic antibiotics, or a testament to the efficacy of
current HIV treatment strategies is unclear. In most of
the cases reviewed, there was a clear male predilection.1,3,4,6 This may be due to lifestyle and social factors
that predispose one to CNF.
This review suggests that the presence of septic
shock is associated with mortality from CNF (Table 2).
The presence of mediastinitis may also be a determinant
for survival for example, in the study by Lin et al,4 where
mediastinitis was associated with a 50% mortality. The
degree of thoracic involvement appeared to also correlate with the patients mortality.6 Typically, CNF will
spare the muscles because of their inherent vascularization. It was of interest, therefore, that in our patient the
muscle tissue was deeply necrotic at the time of initial
debridement. Whether depth of invasion of necrosis is a
clear prognostic factor would require further study.
423
References
1. Tung-Yiu W, Jehn-Shyun H, Ching-Hung C, et al: Cervical necrotizing fasciitis of odontogenic origin: A report of 11 cases.
J Oral Maxillofac Surg 58:1347, 2000
2. Ali MH, Zaued ME: Necrotizing fasciitis of the head and neck:
Report of 3 cases. Ann Saudi Med 17:641, 1997
3. Whitesides L, Cotto-Cumba C, Myers RAM: Cervical necrotizing
fasciitis of odontogenic origin: A case report and review of 12
cases. J Oral Maxillofac Surg 58:144, 2000
4. Lin C, Yeh FL, Lin JT, et al: Necrotizing fasciitis of the head and
neck: An analysis of 47 cases. Plast Reconstr Surg 107:1684,
2001
5. Umeda M, Minamikawa T, Komatsubara H, et al: Necrotizing
fasciitis caused by dental infection: A retrospective analysis of
9 cases and a review of the literature. Oral Surg Oral Med Oral
Pathol Oral Radiol Endod 95:283, 2003
6. Bahu SJ, Shibuya TY, Meleca RJ, et al: Craniocervical necrotizing fasciitis: An 11-year experience. Otolaryngol Head Neck
Surg 125:245, 2001
7. Korhonen K, Kuttila K, Niinikoski J: Tissue gas tensions in
patients with necrotizing fasciitis and healthy controls during
treatment with hyperbaric oxygen: A clinical study. Eur J Surg
166:530, 2000
8. Cawley MJ, Briggs M, Haith LR Jr, et al: Intravenous immunoglobulin as adjunctive treatment for streptococcal toxic shock
syndrome associated with necrotizing fasciitis: Case report and
review. Pharmacotherapy 19:1094, 1999
9. Nakamori Y, Fujimi S, Ogura H, et al: Conventional open
surgery versus percutaneous catheter drainage in the treatment
of cervical necrotizing fasciitis and descending necrotizing
mediastinitis. AJR 182:1443, 2004
10. Arslan A, Pierre-Jerome C, Borthne A: Necrotizing fasciitis:
Unreliable MRI findings in the preoperative diagnosis. Eur J
Radiol 36:139, 2000
J Oral Maxillofac Surg
67:423-427, 2009
Hyperparathyroidism-Jaw Tumor
Syndrome: A Case Report
Brian P. Schmidt, MS, DMD,* Jon P. Bradrick, DDS, and
Ali Gabali, MD, PhD
Hyperparathyroidism-jaw tumor (HPT-JT) syndrome is
a rare multitumor syndrome that includes primary
hyperparathyroidism, fibro-osseous lesions of the
mandible and maxilla, renal tumors and cysts, and
uterine tumors.1,2 It was first reported by Jackson3 in
1958. Since then, multiple cases have been identi-
0278-2391/09/6702-0029$36.00/0
doi:10.1016/j.joms.2008.07.015