Open Access

Severe Fournier’s gangrene in a patient with rectal cancer: case report and literature review

  • Yu Yoshino1,
  • Kimihiko Funahashi1Email author,
  • Rei Okada1,
  • Yasuyuki Miura1,
  • Takayuki Suzuki1,
  • Takamaru Koda1,
  • Kimihiko Yoshida1,
  • Junichi Koike1,
  • Hiroyuki Shiokawa1,
  • Mitsunori Ushigome1,
  • Tomoaki Kaneko1,
  • Yasuo Nagashima1,
  • Mayu Goto1,
  • Akiharu Kurihara1 and
  • Hironori Kaneko1
World Journal of Surgical Oncology201614:234

https://doi.org/10.1186/s12957-016-0989-z

Received: 5 February 2016

Accepted: 17 August 2016

Published: 1 September 2016

Abstract

Background

Fournier’s gangrene in the setting of rectal cancer is rare. Treatment for Fournier’s gangrene associated with rectal cancer is more complex than other cases of Fournier’s gangrene. We report on a patient with severe Fournier’s gangrene in the setting of locally advanced rectal cancer who was treated with a combined modality therapy.

Case presentation

A 65-year-old man presented with general fatigue and anal pain. The medical and surgical histories were unremarkable. A black spot on the perineal skin surrounded by erythema was found on physical examination, suspicious for Fournier’s gangrene. Computed tomography scan showed a rectal tumor invading into the bladder (clinically T4bN2M0) and abscess formation with emphysema around the rectum. He was thus diagnosed with locally advanced rectal cancer and Fournier’s gangrene with a severity index score of 12 points. We created a diverting loop colostomy of the transverse colon and performed extensive debridement of the perineum and perianal area. Fifty days later, the patient underwent radical total pelvic exenteration with sacrectomy. In addition, reconstruction of the soft tissue defect was performed using the rectus muscle, the gluteus maximus muscle, and the femoral muscle. Histopathological findings of the specimen were as follows: the tumor was a moderately adenocarcinoma with invasion to the bladder and the prostate (T4b), metastases to four resected lymph nodes (N2), and lymphovascular invasion. There were no major postoperative complications, and the patient was discharged 108 days postoperatively.

Conclusions

We report a rare case of locally invasive rectal cancer associated with Fournier’s gangrene. This case highlights a usual cause of Fournier’s gangrene. Physicians should be cognizant not only of the more common condition but also of the rare presentations including those associated with rectal cancer.

Keywords

Fournier’s gangrene Rectal cancer Surgical treatment Reconstructive surgery

Background

Fournier’s gangrene (FG) is necrotizing fasciitis and commonly begins without trauma or urinary tract disease. FG in the setting of rectal cancer is rare. A specific risk factor is rectal cancer perforation. Treatment includes extensive debridement of the areas of necrosis and the administration of broad-spectrum intravenous antibiotics. In the setting of rectal cancer, the causative rectal tumor should be removed, but the timing of this is a complex clinical decision.

We report on a patient with severe FG in the setting of locally advanced rectal cancer who was treated with a combined modality therapy.

Case presentation

A 65-year-old man was brought to our outpatient hospital in an ambulance complaining of general fatigue and anal pain. The medical and surgical histories were unremarkable. His initial body temperature was 95.7 °F, blood pressure was 125/58 mmHg, heart rate was 92 beats/min, and oxygen saturation was 98 % on room air. On physical examination, a black spot on the perineal skin surrounded by erythema was found. Due to a clinical suspicion for FG, a computed tomography (CT) scan and blood tests were obtained urgently (Fig. 1). CT scan showed abscess formation with emphysema around the rectum, as well as a tumor invading the bladder (cT4b) with some lymph node metastases in the mesorectum (cN2); there was no evidence of distant metastasis (M0) (Fig. 2). Blood tests were remarkable for a hemoglobin of 3.2 g/dl and a leukocytosis of 63,200/μl. The patient was thus diagnosed with locally advanced rectal cancer and FG with a severity index score of 12 points [1]. Urgent diverting colostomy of the transverse colon and extensive debridement of the perineal area were performed (Fig. 3). Biopsies taken previously revealed a moderately differentiated adenocarcinoma.
Fig. 1

The appearance of the patient’s perineum. A black spot and emphysema were found in the perineal skin

Fig. 2

Computed tomography scan showing a rectal tumor invading the bladder (a) and abscess formation with emphysema in the pelvis and perineum (b)

Fig. 3

Extensive debridement and diverting colostomy were performed on the day of admission (a). Fifty days later, a more extensive debridement was performed (b)

The patient received nutritional support after surgery. Follow-up CT scan after the surgery showed no new lesions, so we performed a radical total pelvic exenteration (TPE) with sacrectomy 50 days after the initial surgery. For the urinary diversion, Bricker’s operation was performed (Fig. 4). In addition, reconstruction of the soft tissue defect was performed using the rectus muscle, the gluteus maximus muscle, and the femoral muscle (Fig. 5). Histopathological findings of the specimen were as follows: the tumor was a moderate adenocarcinoma with invasion to the bladder and the prostate (T4b), metastases to four resected lymph nodes (N2), and lymphovascular invasion. There were no major postoperative complications, and the patient was discharged on postoperative day 108.
Fig. 4

TPE with sacrectomy was performed to obtain a negative resection margin. Histopathological findings of the specimen revealed a moderate adenocarcinoma invading into the bladder and the prostate (T4b), metastasis to four perirectal lymph nodes (N2), and lymphovascular invasion

Fig. 5

Reconstruction was performed using the rectus muscle, the gluteus maximus muscle, and the femoral muscle

Discussion

Although perforation of rectal cancer after treatment with bevacizumab or radiation therapy has been well documented, reports of spontaneous perforation of rectal cancer presenting as FG are rare. There were 23 cases evaluated in a review by Bruketa et al. [2]; in Japan, 17 cases including the present case have been reported (Table 1). The median age in the Japanese group was 58 (range, 30–80) years with a male to female ratio of 15:2. Six (35 %) of 17 patients had diabetes mellitus (DM) as a comorbidity. DM was considered a risk factor for FG in the Japanese group as well. Colostomy was created in 9 of 17 patients. Treatment for rectal cancer with FG was abdominoperineal resection (APR) for six patients, TPE for three patients, and chemotherapy alone for three patients. Four patients were followed conservatively without treatment. Regarding long-term prognosis of the patients, survival of 4 years and 7 months was obtained in one patient who underwent APR, and the survival of the other 16 patients was not given. In this patient, we performed TPE with sacrectomy to obtain a negative resection margin, resulting in a good oncological outcome. In addition, reconstruction was performed using the rectus muscle, the gluteus maximus muscles, and the femoral muscle. There were no major postoperative complications, and the patient was discharged on postoperative day 108.
Table 1

Case reports of Fournier’s gangrene in the setting of rectal cancer in Japan

No.

Author

 

Gender

Age

Location

Comorbidity

Operation

Outcome

1

Futamura et al. [6]

1995

M

56

Rb

None

APR

4 years and 7 months, alive

2

Fujisawa et al. [7]

1999

M

75

Rb

None

None

Death at 6 days

3

Nakao et al. [8]

1999

M

51

Rb

DM

None

Unknown

4

Saito et al. [9]

2000

M

60

Rb

None

TPE

1 year, alive

5

Noriyuki et al. [10]

2003

M

58

Ra

DM

5-FU + LV

1 year and 2 months, alive

6

Enomoto at al. [11]

2006

M

35

Rb

None

APR

3 months, alive

7

Moriwaki et al. [12]

2007

M

30

Rb-P

Unknown

APR

Death at 11 months

8

Kojima et al. [13]

2007

M

56

Rb

DM

TPE

4 months, alive

9

Morohashi et al. [14]

2008

M

60

Rb

DM

None

Death at 2 months

10

Ishibashi et al. [15]

2009

F

80

Rb

HT

APR

2 months, alive

11

Yamazaki et al. [16]

2010

M

50s

Rb

DM

APR

1 months, alive

12

Onizuka et al. [17]

2010

M

55

Rs

None

Chemo

1 year and 2 months, alive

13

Tanaka et al. [18]

2010

F

52

Rb

DM

APR

5 months, alive

14

Watabe et al. [19]

2013

M

77

Rb-P

Unknown

None

2 months, alive

15

Monma et al. [20]

2013

M

79

P

HT

CRT

1 year, alive

16

Kawagoe et al. [21]

2014

M

72

Rb

None

FOLFOX + bev

6 months, alive

17

Our case

2016

M

65

Rb

None

TPE

Death at 1 year

M male, F female, Rb lower rectum, Rs rectosigmoid colon, P proctos, APR abdominoperineal resection, TPE total pelvic exenteration, Chemo chemotherapy, CRT chemoradiation therapy, Bev bevacizumab, DM diabetes mellitus, HT hypertension

The mortality of FG is high, frequently due to a delay in the diagnosis and management. The interesting report about the metabolic evaluation of FG was reported by Surucu et al. [3]. They showed the significance of 18F-fluorodeoxyglucose positron emission tomography for FG in metabolic levels before the visual changes occurred. The mortality of FG in the setting of rectal cancer is not clear. The most common causes of death in FG are sepsis and multiple organ failure. Prognostic predictions can be based on Fournier’s gangrene severity index (FGSI), comprised of the following: body temperature, heart rate, respiratory rate, and serum level of sodium, potassium, creatinine, and bicarbonate, as well as hematocrit value and leukocyte count [1]. Laor et al. [1] stated that a score of >9 was associated with a 75 % mortality, while a score of ≤9 corresponded to a 78 % probability of survival. While our patient presented with an FGSI score of 12 points, we managed to treat the patient without a major complication. Recently, hyperbaric oxygen (HBO) has been recommended as an additional therapy for patients with FG because HBO inhibits the growth of anaerobic bacteria in the affected tissues, prevents further extension of tissue necrosis, and reduces systemic toxicity [4, 5]. Actually, for this patient creating colostomy as an initial procedure, early radical debridement of necrotic tissues, drainage, and antibiotic therapy were effective to manage severe FG in the setting of rectal cancer. Although TPE with sacrectomy was required to obtain a negative resection margin, we could get a good outcome. Although adjuvant therapy was required because of locally advanced rectal cancer of pathologically T4b N2 M0, the patient was followed up at the outpatient section of our institution without adjuvant therapy until all wound had healed completely. Unfortunately, we suspected that local recurrence developed in the perineal wound because of an abdominal 18F-fluorodeoxyglucose positron emission tomography in 5 months after curative TPE (Fig. 6). The patient died of primary disease 1 year after prior surgery.
Fig. 6

18F-fluorodeoxyglucose positron emission tomography scan demonstrated increased fluorodeoxyglucose uptake lesions in the perineal wound. The maximum standardized uptake values (SUV max) of the lesions were 6.36 and 13.45, respectively. Local recurrence was suspected because of an abnormal 18F-fluorodeoxyglucose positron emission tomography scan

Conclusions

Aggressive debridement, broad-spectrum antibiotics, and intensive supportive care are critical for the management of FG. Although colorectal cancer has previously been reported as a cause of FG, it remains an extremely rare phenomenon.

Abbreviations

FG: 

Fournier’s gangrene

CT: 

Computed tomography

TPE: 

Total pelvic exenteration

APR: 

Abdominoperineal resection

FGSI: 

Fournier’s gangrene severity index

Declarations

Acknowledgements

We thank for the help of the plastic surgeons at Toho University Omori Medical Center. Without their efforts, this operation would not be possible.

Funding

The authors have no support or funding to report.

Availability of data and materials

All relevant data are within the paper.

Authors’ contributions

YY, KF, RO, MY, TS, TK, JK, HS, MU, TK, and YN performed the surgery. YY and KF were the major contributors in writing the manuscript. MG, KY, and AK helped YY and KF to collect references. HK has been involved in drafting the manuscript. All authors read and approved the final manuscript.

Competing interests

The authors declare that they have no competing interests.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Ethics approval and consent to participate

Because this report involves no experiment, ethics approval is waived.

Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Authors’ Affiliations

(1)
Department of General and Gastroenterological Surgery, Toho University Omori Medical Center

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Copyright

© The Author(s). 2016

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