Case Report Fournier's Gangrene

Bone Marrow Transplantation, (1998) 22, 1023–1026 1998 Stockton Press All rights reserved 0268–3369/98 $12.00 http://www.stockton-press.co.uk/bmt Case report Fournier’s gangrene: a clinical presentation of necrotizing fasciitis after bone marrow transplantation G Martinelli1, EP Alessandrino1, P Bernasconi1, D Caldera1, A Colombo1, L Malcovati1, MR Gaviglio2, GP Vignoli2, G Borroni2 and C Bernasconi1 1Centro Trapianti di Midollo Osseo, Istituto di Ematologia, and 2Clinica Dermatologica, IRCCS Policlinico S Matteo, Pavia, Italy Summary: BMT (allo-BMT); the third an auto-BMT; all were suffer- ing from acute non-lymphocytic leukemia (ANLL). Three patients with ANLL developed Fournier’s gangrene as an early complication after allo-BMT (two cases) and auto-BMT (one case); two patients were in Case reports first CR, the third had resistant disease. Patients developed fever, perineal pain, swelling and blistering Case No. 1 of the genital area. Pseudomonas aeruginosa was iso- A 41-year-old male, with ANLL in first hematological com- lated from the lesions and patients received systemic antibiotic therapy, surgical debridement and medication plete remission (CR), underwent allo-BMT from an HLA identical sibling donor. Pre-transplant tests showed normal with potassium permanganate solution. Two patients renal and hepatic function; chest X-ray revealed evidence made a complete recovery although one died of sepsis. The third had progressive involvement of the abdominal of a previous right pleuritis; ECG and echocardiography were normal. Performance status was good and clinical wall and later died of leukemia. Early diagnosis of this disorder and prompt initiation of appropriate therapy examination was negative. Conditioning consisted of busul- can prevent progression of this acute necrotizing phan (BU) and cyclophosphamide (CY). The patient received 2 × 108/kg donor bone marrow cells; take was infection. documented on day +11 from transplant. On day +4, he Keywords: Fournier’s gangrene; necrotizing fasciitis; Ͼ ° bone marrow transplantation complained of chills and fever 38 C; physical examination showed genital erythema, pain, swelling and crepi- tation. Broad-spectrum systemic antibiotic therapy was started; white blood cell (WBC) and platelets counts were Fournier’s gangrene (FG) is an acute severe necrotizing dis- respectively 0.5 × 109/l and 0.1 × 109/l. Cultures from the ease of the fascia, subcutaneous fat and skin caused by a central venous catheter (CVC) were positive thereafter for combination of aerobic and anaerobic bacteria, and Staphylococcus aureus. On day +10 the cutaneous genital involves the lower parts of the genitourinary tract, anorectal lesions worsened with blistering and ulceration. The patient soft tissue and genital skin.1–5 Fournier’s gangrene usually developed scrotal gangrene (Figure 1) involving the peri- involves male genitalia, but it has also been described in females.2,3 Schultz et al6 suggest that Fournier’s gangrene may be related to a form of localized vasculitis with histo- logical evidence of hemorrhagic necrosis. A mortality rate of 30–50% has been reported;2,3,6 predisposing factors include diabetes mellitus, perineal trauma or infection, chronic alcoholism, malignancies and an immunocompromised status.3 Despite the severe immunodeficiency that occurs in patients who undergo bone marrow transplantation (BMT),7 Fournier’s gangrene has been described in only one case of autologous BMT (auto-BMT).8 We report three further cases who developed FG in the early cytopenic post-transplant phase. Two had received an allogeneic Correspondence: Dr P Alessandrino, Istituto di Ematologia, IRCCS Poli- clinico S Matteo, 27100 Pavia, Italy Figure 1 Fournier’s gangrene of scrotum with skin blistering and Received 18 February 1998; accepted 20 June 1998 redness. Fournier’s gangrene after BMT G Martinelli et al 1024 neal region and upper thighs and FG was diagnosed. His abdominis (Figure 2). Surgical debridement and local ther- clinical parameters at this time are shown in Table 1: apy with potassium permanganate solution 0.01% were Pseudomonas aeruginosa was isolated from blood, perineal given and systemic antibiotic therapy was continued. On areas, scrotal and abdominal skin. On day +11, systemic day +40, microbiological examination of a swab from the antibiotic therapy with imipenem at a dose of 1 g three cutaneous lesions and echography of the abdominal wall times daily was started together with local potassium per- were negative. Despite topical antiseptic treatment and manganate solution 0.01% and surgical debridement of the intravenous antibiotic therapy the skin lesions did not lesions. The fever disappeared within 24 h and his general resolve completely. The patient died of leukemia on day condition improved. Systemic and topical treatment was +64 from BMT. continued for 4 weeks. At discharge from hospital skin and blood cultural tests were negative. Two weeks later, the Case No. 3 patient entered another hospital because of fever and died from sepsis on day +90 from BMT, in complete hematolog- A 25-year-old female with ANLL in first CR underwent ical remission. auto-BMT. Conditioning consisted of BCNU, etoposide and Ara-C from day −5today−2. On day 0, she received × 8 Case No. 2 2.2 10 /kg unmanipulated cryopreserved autologous bone A 26-year-old female with ANLL in early relapse after allo- BMT from an HLA-identical sibling donor, received per- ipheral blood stem cells (PBSC) collected from the same donor as a rescue after one cycle of standard chemotherapy. At the time of the procedure, her performance status was poor, although hepatic and renal parameters were normal. On day +7, fever of Ͼ38°C occurred and clinical examination showed redness and swelling of the right labium majorum. Two days later, the lesions reached the pubis and dusky discoloration of the skin was observed associated with ulceration and pain. Hematological and biochemical characteristics of the patient at this time are shown in Table 1. Pseudomonas aeruginosa was isolated from the lesions and Fournier’s gangrene was diagnosed. WBC count and platelets count were respectively 0.16 × 109/l and 0.09 × 109/l. Systemic therapy with amikacin 500 mg/12 h was started. The fever disappeared 2 days later but the pain and swelling spread to the anterior abdominal wall. Echo- Figure 2 Acoustic imaging of an abscess of the abdominal wall: cross graphy of the area showed an abscess of the right rectus section of right rectus abdominis. Table 1 Clinical parameters and FG severity index according to Laor9 Case No. 1 Case No. 2 Case No. 3 Age (years) 41 26 24 Sex male female female Timing (day from BMT) +10 +13 +10 Temperature (°C) 38 39 38 Heart rate 120 108 100 Respiratory rate 25 22 18 Serum sodium (mmol/l) 129 128 136 Serum potassium (mmol/l) 2 3.5 2.8 Serum creatinine (mg/dl) 0.8 0.54 0.74 Serum bicarbonate mm/l 35 28 20 Hct (%) 31 25 28 WBC (×109/l) 0.5 0.16 0.6 Hb (g/dl) 11.1 8.7 9.6 Plts (×109/l) 0.1 0.09 0.23 Total bilirubine (mg/dl) 7.5 2.7 0.3 AST/ALT (mg/dl) 8/11 37/39 6/7 Albumin (g/dl) 2.53 2.16 1.93 FG Severity indexa Ͼ13 8 9 aFrom Laor et al.9 1995. AST/ALT = aspartate aminotransferase/alanine aminotransferase. Fournier’s gangrene after BMT G Martinelli et al 1025 marrow cells. Fever Ͼ38°C appeared on day +2. Systemic and by GVHD itself.13 Fournier’s gangrene, however, has antibiotic therapy with ceftazidime, gentamicin and teicho- never been described in allo-BMT patients. planin was started without any improvement. On day +10, In our experience, Fournier’s gangrene was observed in the WBC count and platelets count were respectively three severely immunocompromised patients in the cyto- 0.6 × 109/l and 0.23 × 109. She complained of pain, edema, penic phase after BMT. Two were females and one a male: erythema and swelling of the perineal area. The skin lesions Fournier’s gangrene has usually been described in males; rapidly spread to the suprapubic region and became a nec- in a series of 449 patients, only 63 were females.2 Two of rotic ulcer. Cultures of the cutaneous lesions were positive our patients had undergone allo-BMT from an HLA-ident- for Pseudomonas aeruginosa. Therefore antibiotics were ical related donor and the third an auto-BMT. In all our changed to imipenem and amikacin, and daily local potass- cases Pseudomonas aeruginosa was isolated from the skin, ium permanganate solution 0.01% was used, along with and in one case from the blood also. We promptly started surgical debridement. Complete resolution of skin lesions systemic antibiotics together with surgical debridement and occurred by day +27 and the patient was discharged. She topical treatment. In two cases we observed complete resol- is still well, in complete hematological remission at day ution within 3 weeks (Table 2); one died later of sepsis. +1230. We suggest that in this last case the terminal event was related to the previous infection; antibiotic therapy and local medication may have improved the cutaneous lesions, Discussion but the infection is likely to have spread into the pelvis, producing a late fatal sepsis. The third patient (case No. 2) Fournier’s gangrene has been described as an infectious who had active hematological disease after transplantation, complication in genito-urinary disorders, hematological dis- did not respond to therapy and developed an abscess of the eases and acquired immunodeficiency syndrome. Diabetes anterior abdominal wall (Figure 2). In this case the disease mellitus, advancing age, malignancy and alcoholism are status at transplant could have impaired the healing frequently reported as contributing factors.1–6,8,10–12 processes. The first case of Fournier’s gangrene associated with a In this report, Pseudomonas aeruginosa was isolated in hematological disease was described in 1983 by Patrizi et all three cases.

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