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Progressive synergistic bacterial gangrene arising from abscesses of the vulva and Bartholin's gland duct
L'OlU7TZe
114 October
American
3
?2Un2be7
1,
1972
of Obstetrics and Gynecology
Journal
Transactions Annual
Meeting
Gynecological
Progressive synergistic
of
the of
Ninety-fifth the
American
Society
bacterial gangrene
arising from abscesses of the vulva and Bartholin’s DAVID
B.
LAWRENCE
ROBERTS, L.
Charleston,
Six cases
gland duct
South
M.D.*
HESTER,
JR.,
M.D.
Carolina
diabetes mellitus, diabetic ketoacidosis, and progressive synergistic bacterial in Bartholin’s gland duct abscesses and zulvar abscesses are presented. Four of the cases were managed in a conservative fashion with antibiotic therapy and incision and drainage. Three patients died, and one patient is totally disabled as a result of the disease. Two cases were managed with wide excision of the necrotic and infected tissues. These patients had good end results. Brewer and Meleneyi reported 2 cases of progressive synergistic bacterial gangrene of the skin and subcutaneous tissues following appendectomy. They demonstrated that successful treatment requires wide excision beyond the extent of induration and necrosis. The cases reported in this article demonstrate that patients with diabeter mellitus and an abscess of Bartholin’s duct or of the vulva must be considered to have a necrotiting fascitis secondary to a synergistic infection until pror#ed otherwire. Such patients should have the vulva explored under anesthesia as soon as the ketoacidosis has been corrected, and, if necrosis is found, radical excision to include removal of all necrotic and indurated tissues should be performed. of
gangrene arising
S I N c E C A s P A R Bartholin in 1677, as noted by Word,‘” described the location and function of the perineal vestibular glands, physicians have been aware of the fact that infections and cysts may occur in the re,cion of these glands. These infections and cysts have been the subject of numerous articles
Presented at the Ninety-fifth Annual Meeting of the American Gynecological Society, Hot Springs, Virginia, May 18-20, 1972. Reprint requests: 07. Lawrence L. Hester, Jr., 80 Barre St., Charleston, South Carolina 29401. *Present Columbus,
address: 2400 Georgia.
13th
St..
285
286
Roberts
Table
ond
I. Patients
Patient No.
Octobel 1, 1972 Am. J. Obstet. Gynccol.
Hester
with
Age (years)
necrotizing
fascitis
Blood pressure
Blood glucase on admission
Previous hypoglycemic treatment
Gnsproducing organisms
Culture
Admission until operation (days) 1
1
63
181)/80
648
17 U. NPH insulin
Bacteroides, Meleney’s streptococcus (micro-aerophilir streptococcus), Proteus
Yes
2
55
138,'80
618
Oral
agents
Beta hemolytic cm, E. coli
Yes
3
45
1 10/60
760
Oral
agent
Proteus,
4
60
170/100
291
Oral
agent
Proteus, Klebsiella, Alpha hemolytic enterococcus
No
1
5
47
100/70
250
Oral
agent
E. coli,
Yes
2
6
42
160/100
508
30 U. NPH insulin
NO
0
describing
techniques
of treatment.
reviewing the more guage medical literature, in
tion
that
infections
of
recent there the
gland
However,
English lanis no indicaand
of
duct can be of any serious consequence. particular, there were no reports of death sulting of the
from vulva
synergistic arising
bacterial
in abscesses
its
In re-
gangrene of Rartholin’s
gland duct. Patients with diabetes mellitus, regardless of severity and prior treatment, and an abscess of the Bartholin’s gland duct or of the vulva should be considered as having a potentially lethal disease. These patients need immediate hospitalization and aggressive surgical management. Incision and drainage, insertion of a self-retaining catheter,” and antibiotic
therapy-’
Historical
are
insufficient
therapy.
review
In 1926 Brewer and Meleney”. 7 reported 2 cases of progressive gangrenous infections of the skin and subcutaneous tissues which occurred in and around the abdominal wounds following operation for acute perforative appendicitis. Characteristic infections developed
around
sutures
and
did
not
resolve
streptococ-
Yes
enterococcus
enterococcus
Staphylococcus, enterococcus
citrobacter,
following suture removal. Wound infections were stated to resemble small carbuncles; the skin edges had early necrosis, and the skin was undermined with rather extensive necrosis. The first patient was treated by creating a deep circumferential incision which enclosed the disease area and by packing the new wound. The enclosed area sloughed, but the infection did not extend beyond the created barrier. The second patient was cured by widely excising the area of infection and necrosis. Cultures from the slough produced hemolytic Staphylococcus aureus and a diphtheroid species. Cultures from the zone outside the gangrenous margin grew out an apparent anaerobic nonhemolytic streptococcus. Animal inoculation studies showed that the streptococcus prepared the way, but that for the actual production of gangrene a second organism was necessary, in this case. Staphylococcus aureus. Arndtl adds other well-known infectious processes to that of Meleney and Brewer’s progressive synergistic bacterial
gangrene.
Impetigo
depends
upon
presence of both the hemolytic streptococcus and a staphylococcus. Vincent’s angina and human bite infections are synerthe
Synergistic
Extent Encircled to both
Admission until wide excision (days)
Initial procedure
of necrosis abdomen axillae
upward
to umbilicus flanks
bacterial
gangrene
Extent of wide excision
287
outcome
Incision and drainage and dkbridement
None
Extensive dbbridement of subcutaneous tissues of lower abdomen
Died second hospital day
and
Incision age
and drain-
None
Extensive dkbridement of subcutaneous tissues of lower abdomen
Died fifty-first hospital day
Thigh muscles, left femur, symphysis pubis
Incision aw
and drain-
131
12 procedures including aboveknee amputation, hemipelvectomy
Died
179 days
Pubic bones and symphysis, subcutaneous tissues of lower abdomen
Incision and drainage and debridement
39
Wide debridement of lower abdomen, excision of inferior and superior rami symphysis pubis
Total
disability
2
Wide excision of subcutaneous tissues to right rib cage and of femoral triangle
Cured
Wide excision of subcutaneous tissues of right lahium majus
Cured
Upward hoth
* Subcutaneous right thigh, up to right Labium
tissues of mons veneris, rib cage
majus
only
Wide
excision
Wide
excision
<1
gistic, both infections resulting from a spirochete, Borrelia vincentii, and a fusiform bacillus. In 1931, Meleney’ described a pelvic abscess lvhich did not respond to incision and drainage and which had a carbuncular appearance. Wide excision resulted in rapid disappearance of the infection. Other synergistic infections have been reported with Proteus bacillus and Pseudomonas being cultured.;’ In 1952, Wilson” reported 22 cases of necrotizing fascitis at Dallas’ Parkland Hospital and emphasized that even though gangrene develops its late occurrence detracts from its diagnostic value. The pathognomonic feature in his cases was subcutaneous and fascial necrosis which was manifested by extensive undermining of the skin. The diagnostic skin change, when present, was widespread ecchymosis. Wilson stated that when underimmediate mining was found by probing surgical
treatment
postpone
surgery
antibiotics lerj
noted
been patients
was
indicated
and
use
is ineffective.” that
even
called chronic died within
massive Meade
though
and
these
“. . . to doses
lesions
gangrene both two weeks.
of
Muel-
and
had
of their
Meleney” stated, “. . before 1925 treatment was notoriously unsuccessful. The lesion progressed relentlessly and irresistibly in spite of all kinds of local and systemic therapeutic measures. In 1925, Brewer demonstrated that the lesion could be cured by wide excision outside the zone of erythema, followed by a 1 per cent formalin dressing applied to thf denuded surface. Subsequently, other surgeons found that wide excision alone either with the knife or cautery controlled the infection, but there were frequent recurrences particularly if the line of excision was in the erythematous zone where the streptococci had already become established.” Predisposing factors Meade and Mueller,” in 1967, reported two cases of subcutaneous infections of the leg which occurred in diabetes and pointed out that synergistic infections the presence of systemic
ly controlled
diabetes
tive
in
patients,
siderable been Wyrick,’
weight,
subjected
generally occur in diseases such as poor-
mellitus,
patients
and
who
in postoperahave
in patients
to protracted
in 1970, presented
stress.
lost
who Kea
44 patients
con-
have and
with
288
Roberts
and
Hester
necrotizing infections of the abdomen and estremities. Eight patients were diabetic. and fi\re of those died, gi\.ing a 63 per cent mortality rate in their series. Of the 6 patients presented in this article, 3 died. liea and Wyrick also felt arteriosclerosis contributed to thr mortality rate and pointed out that of the 18 patients in their series who \vc’rc 50 years of age or over 12 died. In the series reported here (Table I ) , all of the patients had adultonset diabetes mellitus, and 3 of the 6 were 50 or older. T\vo of those over 50 years of age died. Three of the 6 patients had evidence of hypertensive vascular disease, and one died. Patient
review
As it is apparent from reviewing other references on the topic of necrotizing fascitis and from reviewing the present series of patients, the lessons learned by Brewer and Meleney’ in 1926 have not been sufficiently appreciated. The first 4 patients had procedures which did not initially involve wide excision beyond the area of necrosis; 3 died. and the fourth subsequently required rathe extensive and disabling operations. In C&e No. 1, operation consisted of dkbridement but not excision beyond the extent of the involved area as recommended by Meleney. In Case No. 2, 2 days in the hospital rlapsed before operation. and, in this case, also, incision and drainage and dPbridement were performed on ‘r occasions, but radical excision was not performed and this patient died. In C:asc No. 3, operation was performed on the day of admission, but again the procedure was incision and draina,qe alone. Ten surgical procedures were subsequently performed. consisting of debridement, skin grafts, and above-the-knee amputation. Eventually, wide excision consisting of a hemipelvectomy \tas done on the patient’s one hundred and thirtyfirst hospital day. However, she died after 179 days of hospitalization. In Case No. 4. there was a one-day delay prior to operation, and again operation consisted only of incision and drainage. Wide excision was not pcrforrncd initially. and this patient had recurrence of the infection. Wide excision of the soft tissues down to the bladder ant1 the,
removal of the superior and inferior rarni of the symphysis pubis hverc required due to osteomyelitis. She is presently totally disabled as a result of the disease. In case No. 5. incision and drainage were prrformed 2 days prior to admission. Two days following nclmission, crepitation was present, and \vicle c+sion of the superficial tissues of the right \ulvar region was performed, Subcutaneous tissues from the lnons pubis to the right superior iliac spine cstending upward to the right rib cage \cere removed. Subcutaneous tissues to thr left of the midline were removed to the level of the unlbilicus, and subcutaneous tissues over the femoral triangle were clscised. On the twenty-ninth hospital day. split-thickness skin grafts were applied. She was discharged in good condition on thr forty-eighth hospital day. In case No. 6. wide cxrision was performed on the day of admission with an excellent result. Melenc)‘f pointed out that \\-ide excision extending hepond the area of tissue necrosis and induration is required to effect a cure in patirnts fascitis. Others”, !’ 1lai.r with necrotizing pointed out that although some cures may be effected with antibiotic therapy alone, in general. complete excision is required. Bacteriology
Grcpitation has been thought to be pathognomonic of clostridial infections, but it is known that other infections may be gas producimg. as with E. coli in diabetes. Four of the 6 patients in the series presented had crepitation, and 3 died (Table I), Each patient had an infection involving more than one organism (Table I). The most commonly present organism was Enterococcus which was prrsent in 5 of the 6 patients. Proteus was present in 3 of the 6. E. c,oli was cultured in 2 of the 6, Pseudomonas, Klebsiclla, Batteroides. Clostridium perfringens. Staph+(~OC(~USaureus, Meleney’s streptococcus inlicropaerophilic streptococcus J, and Clitrobatter were each found in one patient only. Comment
Six ca~rs of diabetes mcllitus, diabetic ketoacidosis. and progressive synergistic bacterial gangrene arising in Bartholin’s gland
Synergistic
duct
abscesses
presented. managed
vulvar of
abscesses
the
cases
in a conservative
biotic.
therapy
Three
died,
nursing
and and
home
from
the
were
managed
crotic
tissues.
Patients
and
Four
one
disease.
with
of
with diabetes
last wide
been
initially with
and
patient
The
These
fashion
incision
because
have
were
anti-
is confined disability 2 cases
abscesses
gland
duct
a
synergistic
to a
Such
presented
found,
radical
of good
the
ne-
results.
regardless
of
all
necrotic
excision and as
sufficiently
corrected.
as
or
secondary proved the
if
289
Bar-
considered
vulva
tissue
to include
indurated
soon
vulva
be
have and,
performed
the
until
should
anesthesia,
gangrene
fascitis
infection
patients
of must
necrotizing
under
had
mellitus,
and
tholin’s
resulting
excision
patients
severity, have
drainage.
bacterial
tissues ketoacidosis
to to
a
otherwise. explored necrosis
is
removal
of
should has
be been
REFERENCES
1.
2. 3. 4. 5.
Amdt, W. F., Jr.: Synergistic infections, in Mailbach, H. I., and Hildick-Smith, G., editors: Skin Bacteria and Their Role in Infections, New York, 1929, chap. 10, McGrawHill Book Company, Inc. Brewer, G. E., and Meleney, F. L.: Ann. Surg. 84: 438. 1926. Goldberg. J, E.: Obstet. Gynecol. 35: 109, 1970. Langmade, C.: GP 28: 135, 1963. Meade, J, W., and Mueller, C. B.: Ann. Surg. 168: 274. 1968.
Discussion DR. LY. C. KEETTEL, Iowa City, Iowa. Bacterial synergistic gangrene as originally defined by Brewer and Meleney involves the subcutaneous tissue of the abdominal wall; it is characterized by a wide peripheral zone of erythema with central necrosis. This develops slowly with few signs of toxicity and is caused by micro-aerophilic streptococcus and hemolytic Staphylococcus uureus. Necrotizing fascitis is a rapidly progressive, highly toxic, and often fatal synergistic bacterial infection, caused hy minor trauma. This involves deeper structures and produces marked tissue destruction often without cutaneous manifestation except for cellulitis and evidence of gas formation. .i variety of bacteria have been identified in such infections including Bacteroides, Escherichia coli. or hemolytic Proteus, anaerobic streptococci, Staphylococcus aureus. Since most of Drs. Roberts and Hester’s patients were acutely ill with extensive tissue destruction, I feel the title of the paper should IX changed to “Necrotizing fascitis.” The term Meleney’s streptococcus is incorrect; the current terminology for this organism is microaerophilic streptococcus. It is important to re-emphasize the significance of early diagnosis, proper antibiotic therapy, and aggressive surgical management, including wide
6.
7.
8. 9. 10.
Meleney, F. L.: Clinical Aspects and Treatment of Surgical Infections, Philadelphia, 1949, chap. 2, W. B. Saunders Company, p. 66. Meleney, F. L.: Treatise on Surgical Infections, 1948, New York, 1948, chap. 14, Oxford University Press, p. 449. Rea, W. J., and Wyrick, W. J., Jr.: Ann. Surg. 172: 957, 1970. Wilson, B.: Ann. Surg. 18: 416, 1952. Word, B. J.: J. Med. Assoc. State Ala. 35: 515. 1965.
excision and drainage of all necrotic tissue as well as peripheral tissue which appears to be undergoing bacterial invasion. In 3 of the cases, the wounds were packed with iodoform gauze; it would seem better to pack the wounds lightly with a fine mesh gauze to prevent the formation of anaerobic pockets. There is a disagreement as to whether diahete\ increases susceptibility to infections, hut there is uniform agreement that once an infection has started it is more severe in the diabetic patient. .4lthough results of various investigations arc contradictory, there is no clear evidence of a defect in antibody synthesis or plasma hactcritidal substances. It has been reported that polymorphonuclear leukocytes from acidotic diabetic patients are defective in their capacity to phagocytize bacteria. The initial treatment of the infection with the proper antibiotic is essential since this can prcvent thr serious complications. A bactericidal antibiotic or a combination of bactericidal antihiotics which will work synergistically is essential. Penicillin or cephalosporin combined with either streptomycin or gentamicin bvould he the antibiotic of choice. In the last 6 months, we have seen on referral various types of necrotizing fascitis: a posthys-
290
Roberts
and
October 1, 1972 Am. J. Obstet. Gynccol.
Hester
terectomy diabetic patient with an extensive abdominal wound infection extending to the vulva, 2 patients with post-cesarean section uterine necrosis resulting in a spreading peritonitis, a Bartholin’s gland abscess in an arteriosclerotic diabetic patient with extensive necrosis of thr rectovaginal septum producing a rectovaginal hstula, and 2 obstetric patients with extensive subgluteal and retropsoal infections. Dr. Lester Hibhard and co-authors1 have recently described 8 obstetric patients who developed extensive infections following paracervical or pudendal blocks. These infections have had all the serious characteristics of the patients described hy Dr. Hester including deaths and significant morbidity. The only difference seems to br the locations of the tissue destruction, the initial symptoms, and the fact that they were young, healthy patients. Thus. any postpartum patient who becomes fehrile and has hip or buttock pain should be suspect. They should be treated promptly with appropriate antibiotic therapy, to arrest the infection before tissue destruction results; if there is no response and there is evidence of abscess fornration, wide debridement and drainage are required. In closing, I wonder if Drs. Roberts and Hester have any explanation for the marked increase in synergistic bacterial infection related to Bacteroides, Escherichia coli, and anaerobic streptococci. REFERENCE
1. Hibbard, E.
M.:
L. T., McVann, Obstet. Gynecol.
R. M., and Snyder, 39: 137, 1972.
DR. JULIAN S~IITH, Houston, Texas. I will present a patient from the M. D. Anderson HO>pita1 with this condition. Some of the features of this patient’s illess are worthwhile to review,. So few cases of progressive synergistic bacterial gangrene have been reported, it is advantageous to the profession from time to time to review the features of this disease. The condition is so rare that Dr. Meleney m New York said that the busy surgeon operating on patients with complications could expect to see one or two patients with progressive postoperative synergistic gangrene in his lifetime. In other words, it is important that this condition be recognized. This patient’s illness was not recognized, and she died from the infection. This patient demonstrates the typical features of this condition. Most patients who have been described have had either drains in an appenditeal abscess or drainage of an empyema. Our
patient had an adenoma-carcinoma of the endometrium. She was treated with radiation, external x-ray therapy, and one radium insertion followed 6 weeks later by a hysterectomy. The typical features, then, of this case are the use of retention sutures, the onset of the infection about a week after operation, and intense pain. The patient required narcotics to be moved in bed or for any care of the wound. Severe pain has been a constant feature of all reported cases. She had everything necessary to make the diagnosis. Streptococcus, a nonhemolytic microaerophilic type, was cultured from the periphery of the wound, and staphylococcus aureus was cultured from the center of the wound. Another finding which is probably diagnostic of this COIIdition without bacterial cultures is the sevrrc, red erythema around the periphery of the wound, the raised purple-red central portion of the infection, and the black-yellow necrosis around the retention sutures and in the middle of the infection where the wound edges are gangrenous. This was a very slow, progressive entity. This patient survived 125 days while the abdominal wall rotted away. She developed 4 enterocutaneous fistulas and a vesicocutaneous fistula. Although consultations were obtained from every medical service in the hospital, none could offer help. Finally, after the patient died, as the resident was ready to start the autopsy, the staff pathologist walked in the room and exclaimed, “This is a typical case of postoperative progressive synergistic bacterial gangrene.” The treatment for this infection had, in the world’s literature, been wide excision of the wound. Meleney described the successful trentment of several patients with bacitracin in l%D, and, in 5 consecutive patients, he leas able to treat this infection successfully without extensive operation. DR. SEY~~OUR L. ROMNEY, Bronx, New York. This is a very impressive and distressing condition which has been described. What occurs to me are questions rather than points of discussion. Do these patients demonstrate septic shock?
In therapy, was metabolic management in all detail? In terms of synergistic when one obtains cultures of organisms, demonstrate any particular sensitivity?
pursued infections, do they
DR. GRAY H. TWOXBLY, New York, Neu York. I knew Dr. Meleney personally. Unfortunately, years ago, we did have a case or two which I can’t illustrate with sensational slides. 1 would suggest that the discussers and Dr. Smith
Volume 114 1Number 3
are dealing with two different conditions. The ones presented in the paper are not the ones that Dr. Meleney made known to us by his writings. I think the one presented by Julian Smith is. Dr. Meleney had two methods of management that we have forgotten. One was the use of zinc peroxide as a local agent in the wounds which we used very extensively in radiation ulcers of the mouth and other places where it worked wonders. It cleared up the necrotizing bacteria very quickly. The other was a combination of neomycin and bacitracin which we found a very useful agent. The formula is 50 C.C. of distilled water, v2 Gm. of neomycin, and 50,000 U. of bacitracin. These are mixed together and used in amounts of 5 to 10 C.C. soaked on gauze and packed into the wound. Sometimes this combination is very effective in these very difficult and terrifying infections. DR. ROBERTS (Closing). First, mention has been made by some of the discussants that synergistic bacterial gangrene and necrotizing fascitis are probably variants of the same disease entity but should be recognized as being distinct variants. Amdt has pointed out there are other synergistic infections. Impetigo is stated to be a synergistic infection dependent on the presence of hemolytic staphylococcus and a streptococcus. Vincent’s angina and the human bite infection
Synergistic
bacterial
gangrene
291
are both synergistic infections which depend upon the presence of a spirochete, Borrelia vincentii, and a fusiform bacillus. I would also like to point out again that the patient with diabetes mellitus and ketoacidosis who has a vulvar abscess must be considered as having a surgical emergency. Furthermore, the extent of the necrosis of the disease cannot be determined by the apparent skin changes. And, if undermining is present, then necrotizing fascitis should be presumptively diagnosed, and wide excision beyond the extending necrosis should be carried out as soon as possible. I have no explanation for the marked increase in synergistic infection. Unfortunately, since we did not recognize the disease process we were dealing with, no photographs were made of these patients when they initially presented. It was asked if any of these patients presented evidence of septic shock. One patient was admitted in shock. She was seen by the staff of Internal Medicine and was placed on appropriate therapy. But she died rather quickly. Dr. Twombly correctly pointed out the fact that the disease entity that we have presented is probably somewhat different from the progressive synergistic bacterial gangrene that Dr. Meleney described.
114 October
American
3
?2Un2be7
1,
1972
of Obstetrics and Gynecology
Journal
Transactions Annual
Meeting
Gynecological
Progressive synergistic
of
the of
Ninety-fifth the
American
Society
bacterial gangrene
arising from abscesses of the vulva and Bartholin’s DAVID
B.
LAWRENCE
ROBERTS, L.
Charleston,
Six cases
gland duct
South
M.D.*
HESTER,
JR.,
M.D.
Carolina
diabetes mellitus, diabetic ketoacidosis, and progressive synergistic bacterial in Bartholin’s gland duct abscesses and zulvar abscesses are presented. Four of the cases were managed in a conservative fashion with antibiotic therapy and incision and drainage. Three patients died, and one patient is totally disabled as a result of the disease. Two cases were managed with wide excision of the necrotic and infected tissues. These patients had good end results. Brewer and Meleneyi reported 2 cases of progressive synergistic bacterial gangrene of the skin and subcutaneous tissues following appendectomy. They demonstrated that successful treatment requires wide excision beyond the extent of induration and necrosis. The cases reported in this article demonstrate that patients with diabeter mellitus and an abscess of Bartholin’s duct or of the vulva must be considered to have a necrotiting fascitis secondary to a synergistic infection until pror#ed otherwire. Such patients should have the vulva explored under anesthesia as soon as the ketoacidosis has been corrected, and, if necrosis is found, radical excision to include removal of all necrotic and indurated tissues should be performed. of
gangrene arising
S I N c E C A s P A R Bartholin in 1677, as noted by Word,‘” described the location and function of the perineal vestibular glands, physicians have been aware of the fact that infections and cysts may occur in the re,cion of these glands. These infections and cysts have been the subject of numerous articles
Presented at the Ninety-fifth Annual Meeting of the American Gynecological Society, Hot Springs, Virginia, May 18-20, 1972. Reprint requests: 07. Lawrence L. Hester, Jr., 80 Barre St., Charleston, South Carolina 29401. *Present Columbus,
address: 2400 Georgia.
13th
St..
285
286
Roberts
Table
ond
I. Patients
Patient No.
Octobel 1, 1972 Am. J. Obstet. Gynccol.
Hester
with
Age (years)
necrotizing
fascitis
Blood pressure
Blood glucase on admission
Previous hypoglycemic treatment
Gnsproducing organisms
Culture
Admission until operation (days) 1
1
63
181)/80
648
17 U. NPH insulin
Bacteroides, Meleney’s streptococcus (micro-aerophilir streptococcus), Proteus
Yes
2
55
138,'80
618
Oral
agents
Beta hemolytic cm, E. coli
Yes
3
45
1 10/60
760
Oral
agent
Proteus,
4
60
170/100
291
Oral
agent
Proteus, Klebsiella, Alpha hemolytic enterococcus
No
1
5
47
100/70
250
Oral
agent
E. coli,
Yes
2
6
42
160/100
508
30 U. NPH insulin
NO
0
describing
techniques
of treatment.
reviewing the more guage medical literature, in
tion
that
infections
of
recent there the
gland
However,
English lanis no indicaand
of
duct can be of any serious consequence. particular, there were no reports of death sulting of the
from vulva
synergistic arising
bacterial
in abscesses
its
In re-
gangrene of Rartholin’s
gland duct. Patients with diabetes mellitus, regardless of severity and prior treatment, and an abscess of the Bartholin’s gland duct or of the vulva should be considered as having a potentially lethal disease. These patients need immediate hospitalization and aggressive surgical management. Incision and drainage, insertion of a self-retaining catheter,” and antibiotic
therapy-’
Historical
are
insufficient
therapy.
review
In 1926 Brewer and Meleney”. 7 reported 2 cases of progressive gangrenous infections of the skin and subcutaneous tissues which occurred in and around the abdominal wounds following operation for acute perforative appendicitis. Characteristic infections developed
around
sutures
and
did
not
resolve
streptococ-
Yes
enterococcus
enterococcus
Staphylococcus, enterococcus
citrobacter,
following suture removal. Wound infections were stated to resemble small carbuncles; the skin edges had early necrosis, and the skin was undermined with rather extensive necrosis. The first patient was treated by creating a deep circumferential incision which enclosed the disease area and by packing the new wound. The enclosed area sloughed, but the infection did not extend beyond the created barrier. The second patient was cured by widely excising the area of infection and necrosis. Cultures from the slough produced hemolytic Staphylococcus aureus and a diphtheroid species. Cultures from the zone outside the gangrenous margin grew out an apparent anaerobic nonhemolytic streptococcus. Animal inoculation studies showed that the streptococcus prepared the way, but that for the actual production of gangrene a second organism was necessary, in this case. Staphylococcus aureus. Arndtl adds other well-known infectious processes to that of Meleney and Brewer’s progressive synergistic bacterial
gangrene.
Impetigo
depends
upon
presence of both the hemolytic streptococcus and a staphylococcus. Vincent’s angina and human bite infections are synerthe
Synergistic
Extent Encircled to both
Admission until wide excision (days)
Initial procedure
of necrosis abdomen axillae
upward
to umbilicus flanks
bacterial
gangrene
Extent of wide excision
287
outcome
Incision and drainage and dkbridement
None
Extensive dbbridement of subcutaneous tissues of lower abdomen
Died second hospital day
and
Incision age
and drain-
None
Extensive dkbridement of subcutaneous tissues of lower abdomen
Died fifty-first hospital day
Thigh muscles, left femur, symphysis pubis
Incision aw
and drain-
131
12 procedures including aboveknee amputation, hemipelvectomy
Died
179 days
Pubic bones and symphysis, subcutaneous tissues of lower abdomen
Incision and drainage and debridement
39
Wide debridement of lower abdomen, excision of inferior and superior rami symphysis pubis
Total
disability
2
Wide excision of subcutaneous tissues to right rib cage and of femoral triangle
Cured
Wide excision of subcutaneous tissues of right lahium majus
Cured
Upward hoth
* Subcutaneous right thigh, up to right Labium
tissues of mons veneris, rib cage
majus
only
Wide
excision
Wide
excision
<1
gistic, both infections resulting from a spirochete, Borrelia vincentii, and a fusiform bacillus. In 1931, Meleney’ described a pelvic abscess lvhich did not respond to incision and drainage and which had a carbuncular appearance. Wide excision resulted in rapid disappearance of the infection. Other synergistic infections have been reported with Proteus bacillus and Pseudomonas being cultured.;’ In 1952, Wilson” reported 22 cases of necrotizing fascitis at Dallas’ Parkland Hospital and emphasized that even though gangrene develops its late occurrence detracts from its diagnostic value. The pathognomonic feature in his cases was subcutaneous and fascial necrosis which was manifested by extensive undermining of the skin. The diagnostic skin change, when present, was widespread ecchymosis. Wilson stated that when underimmediate mining was found by probing surgical
treatment
postpone
surgery
antibiotics lerj
noted
been patients
was
indicated
and
use
is ineffective.” that
even
called chronic died within
massive Meade
though
and
these
“. . . to doses
lesions
gangrene both two weeks.
of
Muel-
and
had
of their
Meleney” stated, “. . before 1925 treatment was notoriously unsuccessful. The lesion progressed relentlessly and irresistibly in spite of all kinds of local and systemic therapeutic measures. In 1925, Brewer demonstrated that the lesion could be cured by wide excision outside the zone of erythema, followed by a 1 per cent formalin dressing applied to thf denuded surface. Subsequently, other surgeons found that wide excision alone either with the knife or cautery controlled the infection, but there were frequent recurrences particularly if the line of excision was in the erythematous zone where the streptococci had already become established.” Predisposing factors Meade and Mueller,” in 1967, reported two cases of subcutaneous infections of the leg which occurred in diabetes and pointed out that synergistic infections the presence of systemic
ly controlled
diabetes
tive
in
patients,
siderable been Wyrick,’
weight,
subjected
generally occur in diseases such as poor-
mellitus,
patients
and
who
in postoperahave
in patients
to protracted
in 1970, presented
stress.
lost
who Kea
44 patients
con-
have and
with
288
Roberts
and
Hester
necrotizing infections of the abdomen and estremities. Eight patients were diabetic. and fi\re of those died, gi\.ing a 63 per cent mortality rate in their series. Of the 6 patients presented in this article, 3 died. liea and Wyrick also felt arteriosclerosis contributed to thr mortality rate and pointed out that of the 18 patients in their series who \vc’rc 50 years of age or over 12 died. In the series reported here (Table I ) , all of the patients had adultonset diabetes mellitus, and 3 of the 6 were 50 or older. T\vo of those over 50 years of age died. Three of the 6 patients had evidence of hypertensive vascular disease, and one died. Patient
review
As it is apparent from reviewing other references on the topic of necrotizing fascitis and from reviewing the present series of patients, the lessons learned by Brewer and Meleney’ in 1926 have not been sufficiently appreciated. The first 4 patients had procedures which did not initially involve wide excision beyond the area of necrosis; 3 died. and the fourth subsequently required rathe extensive and disabling operations. In C&e No. 1, operation consisted of dkbridement but not excision beyond the extent of the involved area as recommended by Meleney. In Case No. 2, 2 days in the hospital rlapsed before operation. and, in this case, also, incision and drainage and dPbridement were performed on ‘r occasions, but radical excision was not performed and this patient died. In C:asc No. 3, operation was performed on the day of admission, but again the procedure was incision and draina,qe alone. Ten surgical procedures were subsequently performed. consisting of debridement, skin grafts, and above-the-knee amputation. Eventually, wide excision consisting of a hemipelvectomy \tas done on the patient’s one hundred and thirtyfirst hospital day. However, she died after 179 days of hospitalization. In Case No. 4. there was a one-day delay prior to operation, and again operation consisted only of incision and drainage. Wide excision was not pcrforrncd initially. and this patient had recurrence of the infection. Wide excision of the soft tissues down to the bladder ant1 the,
removal of the superior and inferior rarni of the symphysis pubis hverc required due to osteomyelitis. She is presently totally disabled as a result of the disease. In case No. 5. incision and drainage were prrformed 2 days prior to admission. Two days following nclmission, crepitation was present, and \vicle c+sion of the superficial tissues of the right \ulvar region was performed, Subcutaneous tissues from the lnons pubis to the right superior iliac spine cstending upward to the right rib cage \cere removed. Subcutaneous tissues to thr left of the midline were removed to the level of the unlbilicus, and subcutaneous tissues over the femoral triangle were clscised. On the twenty-ninth hospital day. split-thickness skin grafts were applied. She was discharged in good condition on thr forty-eighth hospital day. In case No. 6. wide cxrision was performed on the day of admission with an excellent result. Melenc)‘f pointed out that \\-ide excision extending hepond the area of tissue necrosis and induration is required to effect a cure in patirnts fascitis. Others”, !’ 1lai.r with necrotizing pointed out that although some cures may be effected with antibiotic therapy alone, in general. complete excision is required. Bacteriology
Grcpitation has been thought to be pathognomonic of clostridial infections, but it is known that other infections may be gas producimg. as with E. coli in diabetes. Four of the 6 patients in the series presented had crepitation, and 3 died (Table I), Each patient had an infection involving more than one organism (Table I). The most commonly present organism was Enterococcus which was prrsent in 5 of the 6 patients. Proteus was present in 3 of the 6. E. c,oli was cultured in 2 of the 6, Pseudomonas, Klebsiclla, Batteroides. Clostridium perfringens. Staph+(~OC(~USaureus, Meleney’s streptococcus inlicropaerophilic streptococcus J, and Clitrobatter were each found in one patient only. Comment
Six ca~rs of diabetes mcllitus, diabetic ketoacidosis. and progressive synergistic bacterial gangrene arising in Bartholin’s gland
Synergistic
duct
abscesses
presented. managed
vulvar of
abscesses
the
cases
in a conservative
biotic.
therapy
Three
died,
nursing
and and
home
from
the
were
managed
crotic
tissues.
Patients
and
Four
one
disease.
with
of
with diabetes
last wide
been
initially with
and
patient
The
These
fashion
incision
because
have
were
anti-
is confined disability 2 cases
abscesses
gland
duct
a
synergistic
to a
Such
presented
found,
radical
of good
the
ne-
results.
regardless
of
all
necrotic
excision and as
sufficiently
corrected.
as
or
secondary proved the
if
289
Bar-
considered
vulva
tissue
to include
indurated
soon
vulva
be
have and,
performed
the
until
should
anesthesia,
gangrene
fascitis
infection
patients
of must
necrotizing
under
had
mellitus,
and
tholin’s
resulting
excision
patients
severity, have
drainage.
bacterial
tissues ketoacidosis
to to
a
otherwise. explored necrosis
is
removal
of
should has
be been
REFERENCES
1.
2. 3. 4. 5.
Amdt, W. F., Jr.: Synergistic infections, in Mailbach, H. I., and Hildick-Smith, G., editors: Skin Bacteria and Their Role in Infections, New York, 1929, chap. 10, McGrawHill Book Company, Inc. Brewer, G. E., and Meleney, F. L.: Ann. Surg. 84: 438. 1926. Goldberg. J, E.: Obstet. Gynecol. 35: 109, 1970. Langmade, C.: GP 28: 135, 1963. Meade, J, W., and Mueller, C. B.: Ann. Surg. 168: 274. 1968.
Discussion DR. LY. C. KEETTEL, Iowa City, Iowa. Bacterial synergistic gangrene as originally defined by Brewer and Meleney involves the subcutaneous tissue of the abdominal wall; it is characterized by a wide peripheral zone of erythema with central necrosis. This develops slowly with few signs of toxicity and is caused by micro-aerophilic streptococcus and hemolytic Staphylococcus uureus. Necrotizing fascitis is a rapidly progressive, highly toxic, and often fatal synergistic bacterial infection, caused hy minor trauma. This involves deeper structures and produces marked tissue destruction often without cutaneous manifestation except for cellulitis and evidence of gas formation. .i variety of bacteria have been identified in such infections including Bacteroides, Escherichia coli. or hemolytic Proteus, anaerobic streptococci, Staphylococcus aureus. Since most of Drs. Roberts and Hester’s patients were acutely ill with extensive tissue destruction, I feel the title of the paper should IX changed to “Necrotizing fascitis.” The term Meleney’s streptococcus is incorrect; the current terminology for this organism is microaerophilic streptococcus. It is important to re-emphasize the significance of early diagnosis, proper antibiotic therapy, and aggressive surgical management, including wide
6.
7.
8. 9. 10.
Meleney, F. L.: Clinical Aspects and Treatment of Surgical Infections, Philadelphia, 1949, chap. 2, W. B. Saunders Company, p. 66. Meleney, F. L.: Treatise on Surgical Infections, 1948, New York, 1948, chap. 14, Oxford University Press, p. 449. Rea, W. J., and Wyrick, W. J., Jr.: Ann. Surg. 172: 957, 1970. Wilson, B.: Ann. Surg. 18: 416, 1952. Word, B. J.: J. Med. Assoc. State Ala. 35: 515. 1965.
excision and drainage of all necrotic tissue as well as peripheral tissue which appears to be undergoing bacterial invasion. In 3 of the cases, the wounds were packed with iodoform gauze; it would seem better to pack the wounds lightly with a fine mesh gauze to prevent the formation of anaerobic pockets. There is a disagreement as to whether diahete\ increases susceptibility to infections, hut there is uniform agreement that once an infection has started it is more severe in the diabetic patient. .4lthough results of various investigations arc contradictory, there is no clear evidence of a defect in antibody synthesis or plasma hactcritidal substances. It has been reported that polymorphonuclear leukocytes from acidotic diabetic patients are defective in their capacity to phagocytize bacteria. The initial treatment of the infection with the proper antibiotic is essential since this can prcvent thr serious complications. A bactericidal antibiotic or a combination of bactericidal antihiotics which will work synergistically is essential. Penicillin or cephalosporin combined with either streptomycin or gentamicin bvould he the antibiotic of choice. In the last 6 months, we have seen on referral various types of necrotizing fascitis: a posthys-
290
Roberts
and
October 1, 1972 Am. J. Obstet. Gynccol.
Hester
terectomy diabetic patient with an extensive abdominal wound infection extending to the vulva, 2 patients with post-cesarean section uterine necrosis resulting in a spreading peritonitis, a Bartholin’s gland abscess in an arteriosclerotic diabetic patient with extensive necrosis of thr rectovaginal septum producing a rectovaginal hstula, and 2 obstetric patients with extensive subgluteal and retropsoal infections. Dr. Lester Hibhard and co-authors1 have recently described 8 obstetric patients who developed extensive infections following paracervical or pudendal blocks. These infections have had all the serious characteristics of the patients described hy Dr. Hester including deaths and significant morbidity. The only difference seems to br the locations of the tissue destruction, the initial symptoms, and the fact that they were young, healthy patients. Thus. any postpartum patient who becomes fehrile and has hip or buttock pain should be suspect. They should be treated promptly with appropriate antibiotic therapy, to arrest the infection before tissue destruction results; if there is no response and there is evidence of abscess fornration, wide debridement and drainage are required. In closing, I wonder if Drs. Roberts and Hester have any explanation for the marked increase in synergistic bacterial infection related to Bacteroides, Escherichia coli, and anaerobic streptococci. REFERENCE
1. Hibbard, E.
M.:
L. T., McVann, Obstet. Gynecol.
R. M., and Snyder, 39: 137, 1972.
DR. JULIAN S~IITH, Houston, Texas. I will present a patient from the M. D. Anderson HO>pita1 with this condition. Some of the features of this patient’s illess are worthwhile to review,. So few cases of progressive synergistic bacterial gangrene have been reported, it is advantageous to the profession from time to time to review the features of this disease. The condition is so rare that Dr. Meleney m New York said that the busy surgeon operating on patients with complications could expect to see one or two patients with progressive postoperative synergistic gangrene in his lifetime. In other words, it is important that this condition be recognized. This patient’s illness was not recognized, and she died from the infection. This patient demonstrates the typical features of this condition. Most patients who have been described have had either drains in an appenditeal abscess or drainage of an empyema. Our
patient had an adenoma-carcinoma of the endometrium. She was treated with radiation, external x-ray therapy, and one radium insertion followed 6 weeks later by a hysterectomy. The typical features, then, of this case are the use of retention sutures, the onset of the infection about a week after operation, and intense pain. The patient required narcotics to be moved in bed or for any care of the wound. Severe pain has been a constant feature of all reported cases. She had everything necessary to make the diagnosis. Streptococcus, a nonhemolytic microaerophilic type, was cultured from the periphery of the wound, and staphylococcus aureus was cultured from the center of the wound. Another finding which is probably diagnostic of this COIIdition without bacterial cultures is the sevrrc, red erythema around the periphery of the wound, the raised purple-red central portion of the infection, and the black-yellow necrosis around the retention sutures and in the middle of the infection where the wound edges are gangrenous. This was a very slow, progressive entity. This patient survived 125 days while the abdominal wall rotted away. She developed 4 enterocutaneous fistulas and a vesicocutaneous fistula. Although consultations were obtained from every medical service in the hospital, none could offer help. Finally, after the patient died, as the resident was ready to start the autopsy, the staff pathologist walked in the room and exclaimed, “This is a typical case of postoperative progressive synergistic bacterial gangrene.” The treatment for this infection had, in the world’s literature, been wide excision of the wound. Meleney described the successful trentment of several patients with bacitracin in l%D, and, in 5 consecutive patients, he leas able to treat this infection successfully without extensive operation. DR. SEY~~OUR L. ROMNEY, Bronx, New York. This is a very impressive and distressing condition which has been described. What occurs to me are questions rather than points of discussion. Do these patients demonstrate septic shock?
In therapy, was metabolic management in all detail? In terms of synergistic when one obtains cultures of organisms, demonstrate any particular sensitivity?
pursued infections, do they
DR. GRAY H. TWOXBLY, New York, Neu York. I knew Dr. Meleney personally. Unfortunately, years ago, we did have a case or two which I can’t illustrate with sensational slides. 1 would suggest that the discussers and Dr. Smith
Volume 114 1Number 3
are dealing with two different conditions. The ones presented in the paper are not the ones that Dr. Meleney made known to us by his writings. I think the one presented by Julian Smith is. Dr. Meleney had two methods of management that we have forgotten. One was the use of zinc peroxide as a local agent in the wounds which we used very extensively in radiation ulcers of the mouth and other places where it worked wonders. It cleared up the necrotizing bacteria very quickly. The other was a combination of neomycin and bacitracin which we found a very useful agent. The formula is 50 C.C. of distilled water, v2 Gm. of neomycin, and 50,000 U. of bacitracin. These are mixed together and used in amounts of 5 to 10 C.C. soaked on gauze and packed into the wound. Sometimes this combination is very effective in these very difficult and terrifying infections. DR. ROBERTS (Closing). First, mention has been made by some of the discussants that synergistic bacterial gangrene and necrotizing fascitis are probably variants of the same disease entity but should be recognized as being distinct variants. Amdt has pointed out there are other synergistic infections. Impetigo is stated to be a synergistic infection dependent on the presence of hemolytic staphylococcus and a streptococcus. Vincent’s angina and the human bite infection
Synergistic
bacterial
gangrene
291
are both synergistic infections which depend upon the presence of a spirochete, Borrelia vincentii, and a fusiform bacillus. I would also like to point out again that the patient with diabetes mellitus and ketoacidosis who has a vulvar abscess must be considered as having a surgical emergency. Furthermore, the extent of the necrosis of the disease cannot be determined by the apparent skin changes. And, if undermining is present, then necrotizing fascitis should be presumptively diagnosed, and wide excision beyond the extending necrosis should be carried out as soon as possible. I have no explanation for the marked increase in synergistic infection. Unfortunately, since we did not recognize the disease process we were dealing with, no photographs were made of these patients when they initially presented. It was asked if any of these patients presented evidence of septic shock. One patient was admitted in shock. She was seen by the staff of Internal Medicine and was placed on appropriate therapy. But she died rather quickly. Dr. Twombly correctly pointed out the fact that the disease entity that we have presented is probably somewhat different from the progressive synergistic bacterial gangrene that Dr. Meleney described.