CASE HISTORY

A 55-year-old caucasian male, with a history of chronic HCV infection and ongoing injection drug use, was admitted due to severe bilateral wrist infection. The patient presented due to purulent drainage from several open wounds and multiple skin soft-tissue abscesses that emerged from prior sites of skin-popping with heroin in both wrists. Superficial wound culture grew methicillin-resistant Staphylococcus aureus (MRSA) and Pseudomonas putida for which the patient received two weeks of IV vancomycin and oral ciprofloxacin.

Three weeks after ceasing therapy, he re-presented for progressive bilateral wrist wound drainage, swelling, and wrist motion restriction. He denied any systemic signs of infection. Following admission, the patient underwent bilateral wound debridement at the OR. He had extensive skin soft-tissue compromise that extended deep into the radial bone and R-wrist joint. Wrist arthrotomy and radial bone biopsy was performed. MRSA grew from multiple tissue cultures, while acid fast and fungal staining was negative. Radial bone histopathology examination revealed acute osteomyelitis with surrounding suppurative soft-tissue inflammation. Periodic acid-Schiff (PAS) and Grocott-Gomori’s methenamine silver (GMS) stains were negative for fungal elements. The patient was started on IV vancomycin and piperacillin-tazobactam empirically, but the latter was discontinued when culture results showed only MRSA. He was transferred to the long-term rehabilitation unit of our institution to continue IV vancomycin therapy for six weeks.

PAST MEDICAL HISTORY

His medical conditions include gout, chronic HCV infection, alcohol abuse, history of injection drug use, methamphetamine abuse, smoking dependency, and a history of septic arthritis of the left knee for which he underwent arthrotomy and washout two years prior to presentation. One year prior to the current admission, the patient developed right-wrist swelling, motion restriction, synovitis, and bone erosion changes at X-rays. A rheumatoid factor (RF) test was positive and the patient was started on low-dose prednisone (5 mg daily) for presumed rheumatoid arthritis (RA). Nonetheless, his symptoms progressed and he was evaluated by Rheumatology, who decided to initiate anti-TNF blocking therapy with etanercept (Enbrel) and later with adalimumab (Humira), five and two months prior to the initial presentation, respectively. His family history was contributory for cardiovascular disease. The patient lived in a trailer house in central California. He worked as a mechanic for many years. He denies any sick contacts, animal exposures, gardening, or agricultural work. His home medication included colchicine, indomethacin, trazodone, and omeprazole.

PHYSICAL EXAMINATION

Upon admission, the patient seemed in good general condition, appeared uncomfortable, slightly disheveled, and hyperactive. His vital signs were normal. He had swelling and erythema over both wrist areas and active purulent discharge from several ventral and lateral wounds.

LABORATORY TESTING AND IMAGING

Laboratory investigation revealed a WBC (13700/mm3, 75% neutrophils); hemoglobin was 8.3 g/d. Sedimentation rate (ESR) was 70 mm/h and C-reactive protein (CRP) was 1.65 mg/dL. Two sets of peripheral blood culture drawn upon admission were negative. X-rays of the right wrist revealed interval development of severe erosive change with extensive fragmentation and disorganization of the distal radius, ulna, and proximal/distal carpal rows. Severe soft-tissue swelling bilaterally with likely joint effusions was present.

CLINICAL COURSE

The patient completed six weeks of IV vancomycin therapy as an inpatient in the rehab ward. Close to the antibiotic therapy termination, his left-wrist wounds were almost healed, with significant interval decrease of inflammatory changes and no limitations for range of motion. However, over the right wrist he developed a sinus tract that emerged from the wrist joint toward the skin. This was associated with chronic sero-purulent discharge and restricted range of motion. This finding triggered the investigation of the results of the fungal tissue culture from the surgical specimens taken upon admission. This culture was retrieved from an external reference laboratory and it showed the growth of the dimorphic fungus, Sporothrix schenckii. Unfortunately, the external reference laboratory failed in communicating to our institution, in a timely manner, the results of the fungal culture. A new fungal culture was sent from the R-wrist sinus tract, and confirmed, in less than a week, the presence of Sporothrix schenckii. A repeated chest X-ray was clear. At examination, there were no signs of extra-articular disease.

DIAGNOSIS

Wrist septic arthritis and radial osteomyelitis caused by Sporothrix schenckii.

DISCUSSION

Sporotrichosis — or “rose gardener disease” — is primarily an infection that affects the skin and subcutaneous tissue from the extremities. It is caused by the traumatic inoculation of the dimorphic fungi Sporothrix schenckii. Sporotrichosis has global distribution, but most cases are reported in tropical and subtropical areas in the Americas.1In the environment, Sporothrix schenckii exist in hyphal form at temperatures < 37°C, whereas in-vivo adopts the forms of oval or cigar-shaped budding yeast. The acquisition occurs through traumatic contact with a variety of environmental sources, including soil, plants, plant products such as timber, straw, sphagnum moss, hay, thorny plants (e.g., roses and barberry bushes); and from animal contact (armadillos, cats, and squirrels). Sporotrichosis manifests primarily as lymphocutaneous disease: an ulcerated, verrucous or erythematous papule-nodular lesion arises initially at the site of inoculation, which can be followed by multiple secondary lesions through lymphangitic spread. Extra-cutaneous forms of sporotrichosis occur infrequently and generally affect immunocompromised patients. The most common forms of extra-cutaneous disease are osteoarticular, followed by pulmonary (usually cavitary), sinusitis, CNS disease, and endophthalmitis. The diagnosis of sporotrichosis relies on culture methods or through direct visualization by histopathology, but the yeast may remain difficult to detect unless multiple sections are examined. After 5 to 7 days of incubation at 25°C, filamentous hyaline colonies start to grow in Sabouraud dextrose agar. Isolation of the Sporothrix schenckii from any site is considered diagnostic of infection. To date, no standard method of serologic testing is available.

Osteoarticular sporotrichosis occurs by contiguous spread from cutaneous foci or hematogenous dissemination. The most common joints involved by Sporothrix schenckii include (in descending order): knee, wrist, elbow, and ankle. Tenosynovitis, effusion, bursitis, and synovial cyst formation with or without sinus tract may develop in the affected joint.2 In one case series, failure to consider the diagnosis resulted in an average delay of 25 months, resulting in joint damage and increased need for arthrodesis.3

In a case review series that include 84 cases of osteoarticular sporotrichosis since 1970, 49% of the patients had ≥ 1 comorbidities (diabetes, hematological malignancy, AIDS, alcohol abuse, or long-term corticosteroid use).4 In comparison with other causes of dimorphic-fungi osteoarticular infection (e.g. C. immitisB. dermatitidisH. capsulatumP. brasiliensisP. marneffei), osteoarticular sporotrichosis is less likely to be diagnosed by histopathology (< 10% of the cases) but it has the highest rate of isolation by fungal culture (90%) compared with other dimorphic fungi.4

The Infectious Disease Society of America (IDSA) updated the clinical practice guidelines for the management of sporotrichosis in 2007.5 Oral forms of itraconazole (usually 200 mg/ twice per day) are recommended for lympho-cutaneous disease, with a length of therapy of 3-6 months necessary for complete resolution. In localized osteoarticular disease, itraconazole remains the first line of therapy but it should be extended for 12 months.5 Lipid formulations of amphotericin B are recommended as an alternative to itraconazole in cases of disseminated disease or treatment failure with itraconazole.

After identification of Sporothrix schenckii in tissue-wound culture, our patient was started in oral itraconazole (oral suspension) 200 mg BID with a planned length of therapy for one year. Therapeutic drug levels were confirmed two weeks after initiation of anti-fungal therapy. After three weeks of therapy, the patient started noticing reduction in the amount of discharge form the R-wrist sinus tract. The most common mode of acquisition of Sporothrix schenckii in this case could have been environmental contamination of the needles used by the patient for skin-popping in the affected area.

REFERENCES

  1. Mahajan VK. Sporotrichosis: An overview and therapeutic options. Dermatology Research and Practice 2014;2014:272-376.
  2. Howell SJ, Toohey JS. Sporotrichal arthritis in south central Kansas. Clinical Orthopaedics and Related Research 1998;(346):207-214.
  3. Crout JE, Brewer NS, Tompkins RB. Sporotrichosis arthritis: Clinical features in seven patients. Ann Intern Med 1977;86(3):294-297.
  4. Rammaert B, Gamaletsou MN, Zeller V, et al. Dimorphic fungal osteoarticular infections. Eur J Clin Microbiol 2014;33(12):2131-2140.
  5. Kauffman CA, Bustamante B, Chapman SW, Pappas PG, Infectious Diseases Society of A. Clinical practice guidelines for the management of sporotrichosis: 2007 update by the Infectious Diseases Society of America.Clin Infect Dis 2007;45(10):1255-1265.