Favorable Outcome of Fournier Gangrene in Two Patients with

Advances in Peritoneal Dialysis, Vol. 30, 2014
Darlene Vigil,1 Anil Regmi,1,2 Reuben Last,3 Brenda Wiggins,1
Yijuan Sun,1,2 Karen S. Servilla,1,2 Joanna R. Fair,4 Larry
Massie,5 Antonios H. Tzamaloukas1,2
Fournier gangrene (FG), a form of necrotizing
fasciitis of the perineum and genitals, with high
morbidity and mortality in the general population,
carries the additional risk of involvement of the
peritoneal catheter tunnel and peritoneal cavity
in patients on chronic peritoneal dialysis (PD). We
describe two men with diabetes who developed FG
in the course of PD. Computed tomography showed
no extension of FG to the abdominal wall, and spent
peritoneal dialysate was clear in both patients.
Broad-spectrum antibiotic therapy with anaerobic
coverage and early aggressive debridement followed
by negative-pressure wound therapy and repeated
debridement led to improvements in clinical status
in both cases. Surgical closure and healing of the
wound was achieved in one patient; the wound of the
second patient is healing, but remains open. Both
patients experienced prolonged hospitalization,
with a serious decline in nutrition status. In patients
on PD, FG can be treated successfully. However,
additional measures are required to evaluate for
potential involvement of the PD apparatus and the
peritoneal cavity in the infectious process; and
prolonged hospitalization, worsening nutrition, and
multiple surgical interventions can result.
From: 1Renal Section, Raymond G. Murphy VA Medical
Center; 2Department of Medicine, University of New Mexico
School of Medicine; 3Surgery Service, Raymond G. Murphy
VA Medical Center, and Department of Surgery, University
of New Mexico School of Medicine; 4Radiology Service,
Raymond G. Murphy VA Medical Center, and Department of
Radiology, University of New Mexico School of Medicine;
5Pathology Service, Raymond G. Murphy VA Medical Center,
and Department of Pathology, University of New Mexico
School of Medicine, Albuquerque, New Mexico, U.S.A.
Favorable Outcome of
Fournier Gangrene in
Two Patients with Diabetes
Mellitus on Continuous
Peritoneal Dialysis
Key words
Fournier gangrene, diabetes mellitus, malnutrition,
negative-pressure vacuum dressings
Introduction
Fournier gangrene (FG) is a synergistic polymicrobial necrotizing fasciitis of the perineum and external genital organs primarily affecting men (1–3). Its
histologic picture is characterized by obliterative
endarteritis of the subcutaneous arteries and gangrene of the subcutaneous tissue and the overlying
skin (1–3). An acute surgical emergency, FG has a
mortality rate of 24% – 45% (4). Extension of FG to
the abdominal wall, chest, axillae, and thighs has
been reported (3). Gangrene of the abdominal wall
in patients on chronic peritoneal dialysis (PD) carries the added risk of extension of the necrotizing
infectious process into the tunnel of the PD catheter
and the peritoneal cavity.
Only 1 case of FG in a patient on PD (5)—with a
fatal outcome—was found in a bibliographic search.
We report 2 men with diabetes who developed FG
in the course of PD and survived. Their cases illustrate the diagnostic interventions, management,
and follow-up of FG in PD patients.
Case descriptions
Both patients reported here were receiving PD
for end-stage renal disease secondary to diabetic
nephropathy. The PD schedule, which was exactly
the same in the 2 patients, consisted of automated
nocturnal 9-hour PD with 4 exchanges (3.0-L fill
volume), plus two 2.5-L daytime exchanges, one
with 7.5% icodextrin and one with dextrose. Peritoneal transport was of the high-average type in
both patients.
Vigil et al.
Patient 1
A 68-year-old man on PD for 41 months presented
with excruciating pain and swelling of the left groin
and scrotum of 5 days’ duration. He had experienced
no episodes of peritonitis or exit-site or tunnel infection in the past, but had experienced an episode of
urinary tract infection 1 year before the development
of scrotal swelling and pain. Weekly Kt/V urea varied
between 1.82 and 2.43.
At presentation, the patient’s temperature was
38°C, his heart rate was 113 bpm, and his respiratory
rate was 23/min. His body mass index was 29.0 kg/
m2. The left side of the scrotum was swollen and
indurated, with extreme tenderness along the course
of the left epididymis, and crepitus in the perineum
and left hemiscrotum. Pertinent laboratory values (6)
included serum sodium 133 mmol/L (glucose 286 mg/
dL); potassium 4.5 mmol/L; creatinine 11.86 mg/dL;
total CO2 20 mmol/L; hematocrit 33.5%; and white
cell count 15.2×103/mm3, with 87% neutrophils.
Computed tomography (CT) showed scrotal wall
thickening, prominent soft-tissue thickening, and
subcutaneous gas posterior to the left inguinal canal,
consistent with necrotizing fasciitis involving the left
inguinal and intertriginous regions. The process did
not involve the abdominal wall.
The patient was treated with aggressive hydration,
parenteral vancomycin and piperacillin–tazobactam,
and urgent wound debridement. Histologic examination of the excised tissue revealed subcutaneous
tissue necrosis and inflammation. Tissue cultures
revealed mixed urogenital flora. Anaerobic and
fungal cultures of the excised tissue were negative.
A urinary tract infection was also present. Spent
peritoneal dialysate contained 56 cells per cubic
milliliter, with negative cultures.
The patient’s hospital course was complicated
by severe hypotension that lasted 6 days, and atrial
fibrillation with a rapid ventricular rate requiring
cardioversion. Three surgical explorations with
debridement were performed, and the wound was
treated with negative-pressure wound therapy. During this patient’s hospitalization, PD was continued
using the established schedule, and spent PD fluid was
monitored. Repeated effluent cell counts and cultures
were negative.
The patient was discharged after 17 days of hospitalization. During hospitalization, he lost 6.2 kg of
body weight. By 6 weeks after presentation, his wound
121
was healed, and he had regained his lost weight. His
serum albumin was 4.2 g/dL before hospitalization,
declined to 3.3 g/dL during hospitalization, and increased to 4.4 g/dL at 6 months after discharge.
Patient 2
A 54-year-old man on PD for 30 months was admitted
with generalized weakness, fever, and scrotal swelling and pain for 1 day. In the preceding months, his
weekly Kt/V urea had fluctuated between 1.66 and
1.92. He had experienced 1 episode of peritonitis with
coagulase-negative Staphylococcus 14 months before
the current admission, but no exit-site or tunnel infection. A perineal abscess had been drained 5 months
before admission, and 2 months before admission, he
had experienced the onset of persistent diarrhea which,
after admission, proved to be secondary to Clostridium
difficile infection.
On admission, the patient’s temperature was
38.6°C, his heart rate was 93 bpm, and his respiratory
rate was 21/min. His body mass index was 34.3 kg/
m2. Examination of the scrotum revealed erythema,
exquisite tenderness, and crepitus, particularly of the
left hemiscrotum. Pertinent laboratory studies (6) included serum sodium 136 mmol/L (glucose 268 mg/
dL); potassium 4.0 mmol/L; creatinine 15.78 mg/dL;
total CO2 24 mmol/L; hematocrit 26.9%; and white
cell count 20.7×103/mm3, with 92% neutrophils.
Blood cultures were negative, and spent dialysate was
clear. Imaging with CT revealed diffuse edema and
subcutaneous emphysema in the left scrotum, extending into the perineum and the penis (Figure 1), but not
involving the abdominal wall.
On the day of admission, the patient underwent
excision of the dead skin and debridement of the
scrotum and was placed on a combination of vancomycin, piperacillin–tazobactam, and metronidazole.
Histologic examination of the excised scrotal tissue
revealed extensive inflammation and necrosis involving the soft tissue under the skin and the adipose tissue
(Figure 2).
After the surgical intervention, the patient’s
clinical status improved, but he required two more
sessions of debridement of the surgical wound,
which was treated with negative-pressure wound
therapy. During his hospitalization, PD was continued on the established schedule. Physical debility
and a need for systematic physical therapy delayed
the patient’s discharge, which occurred after 38 days
122
figure 1 Computed tomography images of Fournier gangrene
(patient 2). Axial views in (A) soft-tissue and (B) bone windows
depict diffuse edema along the left aspect of the scrotum and penis
(arrowheads) and extensive scrotal and penile subcutaneous gas
(arrows).
of hospitalization. His wound was granulating, but
remained partially open on discharge.
During hospitalization, the patient lost 6.5 kg of
body weight. His serum albumin, which was 4.1 g/dL
before hospitalization, declined to a nadir of 2.8 g/dL,
but had improved to 3.4 g/dL after 1 month.
Summary
The histologic features and pathogenesis of FG and
of necrotizing fasciitis are indistinguishable (7). The
patients presented in this report illustrate several of the
cardinal features of FG. The disease more frequently
affects men than women, but it has been encountered
in all age groups. Diabetes mellitus and alcoholism are
the two conditions most commonly associated with FG
(2–4). Immunosuppression for malignancy or organ
transplantation (2), leukemia (3), intravenous drug
use, malnutrition (4), and HIV infection (8) are less
frequently associated with FG. Calciphylaxis, which
Fournier Gangrene in PD
figure 2 Histology of Fournier gangrene (patient 2). Low-power
image of hematoxylin and eosin skin section showing extensive
necrosis of the deep dermis and subcutaneous tissues, with gas
formation. Inset: High-power image of necrotic fibrous connective
tissue with acute inflammatory infiltrates.
could be relevant in PD patients, was associated with
1 case of FG (9).
The disease evolves from a combination of tissue
ischemia (diabetic small-vessel disease being a major
predisposing factor) and overgrowth of bacteria (anaerobes often having a major role) (2,3). A portal of
entry through the colorectal region, the genitourinary
tract, or the skin is usually identified (3,4). Multiple
microbial organisms are usually recovered from the
infected tissue. In one series of 236 patients, the most
common organisms recovered were, in descending
frequency, various species of Escherichia coli, Bacteroides, Staphylococcus, Proteus, Streptococcus,
Pseudomonas, Enterococcus, and Klebsiella (4).
In addition to identification of the portal of entry,
which could require endoscopic procedures such as
proctoscopy or retrograde pyelography, evaluation
of the FG margins is critical to management and, as
noted earlier, acquires even greater importance in PD
Vigil et al.
patients. Imaging, which also assists in making the
diagnosis in difficult cases, is used to determine the
extent of FG (10). Plain radiography, ultrasonography,
CT, and magnetic resonance imaging have all been
applied for this purpose (10). Imaging by CT was
reported to be superior to that by plain radiography
or ultrasonography (11). Magnetic resonance imaging is accurate in identifying the extent of disease
and the point of origin (12). Given those reports, CT
or magnetic resonance imaging, whichever is readily
available, should be considered the imaging procedure
of choice in FG.
The high mortality in FG has led to efforts to
develop prognostic indicators that might be used
to evaluate various therapeutic modalities. The FG
severity index is calculated using stratified (high and
low) values of body temperature; heart and respiratory rates; serum sodium, potassium, creatinine, and
bicarbonate; and hematocrit and white cell count (6).
High index values were found in several studies to
predict mortality with reasonable accuracy (13–15).
Other clinical factors that were found to be associated
with mortality were hepatic dysfunction, older age,
colorectal origin (3), extent of the disease (2), and
delays in surgical intervention (16).
Early recognition of FG is imperative (17). Management has two prongs that should both be applied
as soon as the diagnosis is made. First, parenteral
antibiotics with broad coverage, including anaerobic
agents, should be initiated, and hemodynamic stabilization should be pursued. At the same time, aggressive
surgical debridement should also be performed (17).
The wound should initially be treated with topical
antiseptics and frequent dressing changes, and the
edges of the wound should be carefully monitored for
signs that the FG has advanced. After the first 2 days,
treatment with negative-pressure wound therapy to
promote granulation and closure has been found to be
effective (18). Hyperbaric oxygen treatment has also
been applied (2,4). Repeated debridement is usually
necessary, until FG is found to be no longer advancing
and causing tissue necrosis.
Perineal and scrotal soft-tissue defects can be
extensive; they frequently lead to chronic wounds
that can require reconstructive surgery. If fecal contamination of the wound becomes problematic, an
enterocutaneous stoma might become necessary to
divert stool from the wound (19). Colectomy has also
been performed in patients with severe colonic disease
123
(19). Physical and mental debility might persist for a
long period after discharge (19). In patients on PD
who develop FG, imaging should address the potential
for involvement of the abdominal wall proximally to
the PD catheter tunnel and of the catheter exit site or
tunnel. Spent dialysate should be monitored for signs
of infection. In the absence of signs of peritoneal
involvement, PD can continue. As has been reported
with other severe infectious processes affecting PD
patients (20), FG has a large effect on nutrition.
Nutrition and physical rehabilitation require special
attention in patients with FG.
Acknowledgment
This work was supported by the Raymond G. Murphy
VA Medical Center.
Disclosures
The authors of this report declare that they have no
financial conflicts of interest.
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Corresponding author:
Antonios H. Tzamaloukas, md, Renal Section (111C),
Raymond G. Murphy VA Medical Center, 1501 San
Pedro SE, Albuquerque, New Mexico 87108 U.S.A.
E-mail:
Antonios.Tzamaloukas@va.gov