Melioidotic Necrotizing Fasciitis Presenting As a Supraclavicular Mass

Jpn. J. Infect. Dis., 61, 151-153, 2008

Short Communication Melioidotic Necrotizing Fasciitis Presenting as a Supraclavicular Mass Tsung-Liang Ma, Guan-Cheng Huang, Hung-Jen Tang and Chia-Ming Chang1,2* Department of Internal Medicine, Chi Mei Medical Center, and 1Division of Infectious Diseases and 2Division of Geriatrics and Gerontology, Department of Internal Medicine, National Cheng Kung University Hospital, Tainan, Taiwan (Received August 10, 2007. Accepted January 21, 2008) SUMMARY: Melioidosis is an infection caused by Burkholderia pseudomallei that usually involves the respi- ratory tract. It may manifest as pneumonia, septicemia, or localized infection. We present here a case of melioidosis initially manifesting as a mass over the supraclavicular area and subsequently progressing to necrotizing fasciitis. With appropriate antimicrobial treatment and adequate surgical debridement, localized melioidosis can be treated successfully. Melioidosis should be considered in the differential diagnosis of neck masses, especially in patients who have traveled to or stayed in an endemic area.

Melioidosis is an infectious disease caused by the Gram- hard nodule that enlarged gradually. He had no sore throat, negative bacilli Burkholderia pseudomallei. It is endemic in cough, or other discomfort. Physical examination revealed a Southeast Asia, northern Australia, Central and South America, hard, non-tender mass about 4 × 5 cm in diameter on the and China (1). Clinical manifestations are variable, and it has right supraclavicular region. The liver and spleen were not acute, sub-acute, chronic, and sub-clinical forms (1,2). In palpable. Computed tomography of the head and neck showed Taiwan, endemic cases of melioidosis have emerged in the a 3.7 × 3.3 cm ill-defined lesion with central necrosis over past decade (2). From July through September 2005, 40 cases the right lower neck to the supraclavicular region with ad- of melioidosis were reported in southern Taiwan after a jacent soft tissue infiltration (Figure 1). Multiple necrotic typhoon. Most of these patients presented as bacteremia lymphadenopathies over the right carotid space of the neck and pleuropulmonary infections (3). Diagnosis by clinical and mediastinal regions were also noted (Figure 2). presentations only, without microbiological information, is Echo-guided fine-needle aspiration was arranged for the difficult. The presentation usually involves the respiratory necrotic mass lesion. Ultrasonography disclosed multiple bi- tract and may manifest as pneumonia, septicemia, or localized lateral neck lymph nodes and a 7.9 cm multi-loculated mass infection (1). Although soft tissue infections are not un- with a heterogeneous hypo-echoic pattern in the right lower common, necrotizing fasciitis caused by B. pseudomallei has neck. The mass was aspirated, and a specimen was collected rarely been reported (4,5). We present here a case of melioido- for cytology and bacterial culture. For further pathology sis manifesting initially as a supraclavicular mass that sub- sequently progressed to necrotizing fasciitis. A 41-year-old man suffered from general malaise for about half a year with recent exacerbation. On June 19, 2006, he was referred to the hematology clinic due to leukocytosis found at a local clinic. He had had no chronic diseases such as diabetes mellitus or hypertension in the past. In the first two outpatient visits, only mild leukocytosis (leukocyte counts: 10,200/μL and 12,000/μL; neutrophils 70.8 and 80%, respectively) was found. No fever or lymphadenopathy was noted. Biochemical data were normal for glucose (86 mg/ dL) and for renal and liver functions (blood urea nitrogen 14 Fig. 1. Computed tomography of head and neck showed a 3.7 × 3.3 mg/dL, creatinine 1.0 mg/dL, aspartate aminotransferase 18 cm ill-defined mass lesion with central necrosis on the right lower IU/L, alanine aminotransferase 12 IU/L). Chest X-ray and neck (arrow) with multiple necrotic lymph nodes over the right carotid abdominal ultrasonography were unremarkable, while space of the lateral neck. esophago-gastroduodenoscopy showed only erythematous gastritis. However, on the third outpatient visit to the hematolology clinic (about 28 days after the first visit), a palpable mass was noted on his right supraclavicular region, which the pa- tient noted about 4 days earlier. It began as a small painless

*Corresponding author: Mailing address: Divisions of Infectious Diseases, Geriatrics and Gerontology, Department of Internal Medicine, National Cheng Kung University Hospital, 138, Sheng- Li Rd., Tainan City 70403, Taiwan. Tel: +886-6-2353535, ext. 3596, Fax: +886-6-2752038, E-mail: [email protected]. Fig. 2. Several mediastinal lymph nodes, some with calcification and tw, [email protected] central necrosis (arrow).

151 examination, the patient was then referred to the general sur- Skin and soft tissue infections are common manifestations gery clinic for incision biopsy of the mass. At that time, about of melioidosis, but progression to necrotizing fasciitis has a week after the mass was initially noted, it enlarged further rarely been reported (4), especially involving the head and and became painful. The biopsy specimen was composed of neck. Lim et al. reported 4 cases of melioidosis involving the muscle and soft tissue but no nodal tissue. Pathology showed head and neck region, with one case of parotid abscess that necrotizing inflammation with no malignancy. was drained but subsequently progressed to necrotizing Bacterial culture of the aspirated tissue later yielded growth fasciitis (5). Our patient had undergone needle aspiration and of B. pseudomallei, which was susceptible to ceftazidime, incision biopsy of the mass but finally underwent debride- cefpirome, imipenem, and piperacillin-tazobactam but was ment due to poor wound condition. Whether or not the previ- intermediately resistant to trimethoprim-sulfamethoxazole ous procedure on the lesion led to the progression of the and ciprofloxacin. The patient was then admitted for further disease to necrotizing fasciitis requires further investigation. antimicrobial therapy. Suppurative lymphadenitis caused by melioidosis has rarely On admission, purulent discharge from the previous inci- been encountered. In addition to the head and neck region, sion wound of the supraclavicular mass was noted. Hemogram inguinal and mediastinal lymph nodes could also be infected disclosed a hemoglobin level of 14.6 g/dL and a leukocyte (6). The imaging study in our case showed necrotic lym- count of 9,400/μL, with 72.3% neutrophils and 14.9% lympho- phadenopathies over the right carotid space of the neck and cytes. Other biochemical data, including liver and renal func- mediastinal regions. Melioidosis should be considered in the tions, were all within normal limits. No serological test was differential diagnosis of suppurative regional lymphadenitis, available at our hospital. especially among patients who have traveled to, or stayed in, The personal history of this patient was reviewed in ad- an endemic area (6). vance. He lives in the Che-Ting Country of Kaohsiung County, The modes of acquisition of melioidosis include ingestion, which is a seaside village in southern Taiwan. The patient inhalation, and inoculation (1). In the past, inhalation was also mentioned that an alcoholic man living in his neighbor- thought to be the primary mode of infection, but now in- hood died of melioidosis. The patient was a cement worker oculation is believed to be the primary mode (1). Based on in building construction and had the habit of cigarette smok- his occupational history of being a construction worker, the ing about more than one pack per day for 20 years. He also patient might have been infected through the inhalation of had the habit of social drinking and betel nut chewing for 10 contaminated dusts or inoculation with contaminated soil or years but had quit half a year earlier when he felt discomfort. water through wounds or skin abrasions. He had a pet dog for years and had never traveled abroad. Because of intrinsic resistance of B. pseudomallei to com- During admission, after two sets of blood culture were col- monly used first-line antimicrobial agents, only amoxicillin- lected, the patient was initially treated with ceftazidime 2 g clavulanate, ceftazidime, meropenem, or imipenem, alone intravenously every 8 h for 9 days. Blood culture later yielded or in combination with co-trimoxazole, have been reported no growth of pathogen. However, purulent discharge still to have in vitro and in vivo bactericidal effects (7,8). The oozed from the incision wound. On the 10th hospital day, a current recommended therapy includes two phases, an acute decision to perform surgical excision of the mass and wound phase with intravenous ceftazidime or carbapenem for at debridement was made. Intra-operatively, diffuse necrotic least 14 days, followed by an eradication phase with oral tissue was noted within the subcutaneous space, underlying amoxicillin-clavulanate or a combination of co-trimoxazole muscle, and scattered over the surrounding tissue and fascia and doxycycline for 12 to 20 weeks (1,9). Relapse can occur space. Loss of resistance of the normally adherent superfi- after a median of 6 to 8 months. An eradication therapy of cial fascia to blunt dissection was noted, consistent with less than 8 weeks is associated with a higher risk of relapse necrotizing fasciitis. Fasciectomy and debridement were per- (1). Our patient received only a 3-week eradication therapy. formed. The pathology of the debrided tissue showed necrotic Although he remained asymptomatic after cessation of the tissue, collection of pus cells, and edematous tissue infiltrated oral antimicrobial agent for 8 months, long-term follow-up by acute and chronic inflammatory cells. Several multinucle- is mandatory because of the potentially incomplete treatment ated giant cells were found in the necrotic focus. Acid fast and the fairly long period before relapse occurs. and Auramine-Rhodamine stains were negative for bacilli. As melioidosis becomes an emerging infectious disease, Aerobic culture of the debrided tissues did not yield any bac- clinicians should consider it in the differential diagnosis of terial growth. neck mass. It is possible to misidentify a supraclavicular soft Postoperatively, the antimicrobial agent was switched to tissue mass as simple lymphadenopathy. In addition to early imipenem 500 mg intravenously every 6 h for 3 weeks. After recognition, appropriate antimicrobial treatment and surgi- discharge, the patient received oral amoxicillin-clavulanate cal debridement are required to improve prognosis. for additional 3 weeks. At the end of the antimicrobial treat- ment, the value of the erythrocyte sedimentation rate was REFERENCES 17 mm/h (normal, 0-15 mm/h). The patient was followed 1. Cheng, A.C. and Currie, B.J. (2005): Melioidosis: epidemiology, up for 8 months after discharge during which he remained pathophysiology, and management. Clin. Microbiol. 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