Cutaneous Botryomycosis Secondary to Trauma A Case Presentation and Review of Literature Jennifer David DO, MBA Department of Dermatology

ABSTRACT PHYSICAL EXAM MANAGEMENT / OUTCOME

CASE DESCRIPTION: A 52-year-old Caucasian male with a past medical history of hypertension presented • Tissue cultures were obtained and showed to be negative to our clinic complaining of growths on bilateral forearms that developed two months prior. The lesion on his • Left forearm: 4cm pink exophytic vegetative plaque with central ulceration, crusting and background of excoriations and pink for acid-fast mycobacteria and fungal/yeast elements right forearm began as small pink papules that grew over the course of a few weeks, ulcerated and developed a atrophic scars on left forearm. (Figure 1) however did grow heavy amounts of Staphylococcus crusted scab. His main concern was regarding the growing lesion on the right arm. He admitted to occasional aureus. mild pruritus but denied any associated pain, tenderness or burning sensation of involved skin. Of note, he • Right forearm : scattered excoriations, pink/tan papules and a 1 cm crusted ulceration (Figure 2) worked as a HVAC (heating, ventilation and air conditioning) repairman with a history of repeated trauma to • Deep shave biopsy of lesion on left forearm his forearms due to reaching through confined spaces of larger industrial units. On physical exam patient presented with 4cm pink exophytic vegetative plaque with central ulceration, • Patient was treated with oral cephalexin 500mg and crusting and background of excoriations and pink atrophic scars on left forearm. (Figure 1) On the right topical mupirocin ointment twice daily for two weeks. forearm were scattered excoriations, pink/tan papules and a 1 cm crusted ulceration (Figure 2) Tissue cultures were obtained and showed to be negative for acid-fast mycobacteria and fungal/yeast elements however did grow heavy amounts of . CLINICAL PRESENTATION Deep shave skin biopsy displayed pseudoepitheliomatous hyperplasia with sinus tract formation, focal supprative inflammation and focal granuloma formation with gram-positive cocci within the Figure 5: Patient’s lesions clearing, two weeks after treatment with oral cephalexin and topical pseudoepitheliomatous hyperplasia in henatoxylin-eosin staining. (Figure 3) PAS staining was negative. mupirocin On higher power radiating deposits of amorphous, eosinophilic, hyaline material around colonies of cocci were noted, characterizing the Splendore-Hoeppli phenomenon. (Figure 4) A diagnosis of localized cutaneous botryomycosis was established and the patient was treated with oral DICUSSION cephalexin 500mg and topical mupirocin ointment twice daily for two weeks. His follow up physical exam two weeks after treatment revealed a significant improvement in all skin lesions. (Figure 5) The term botryomycosis is derived from the Greek word botrys (meaning "bunch of grapes") and mycosis (a DISCUSSION: Botryomycosis is a rare, chronic, supprative, granulomatous infectious disease that affects the misnomer, due to the presumed fungal etiology in early descriptions) (1). Other terms used to describe skin and occasionally the viscera. Staphylococcus aureus (40%) is the most common causative organism botryomycosis include bacterial pseudomycosis, staphylococcal actinophytosis, granular bacteriosis, and however it can also be caused by Pseudonomas aeruginosa (20%), coagulase-negative staphylococci, actinobacillosis. The most frequent etiological agent is Staphylococcus aureus (40%), followed by Pseudomonas spp, Escherichia coli, and Proteus spp. (1-3) There are few cases reported in literature and it sp (20%). Other microorganisms reported include Escherichia coli, Proteus vulgaris, Bacillus spp, Actinobacillus occurs in areas of the skin that are exposed and subject to repeated trauma . The pathogenesis of the disease lignieresii (1,2). has not been well established. It is thought to be related to low virulence of agents, large local bacterial The pathogenesis of the disease has not been well established. It is thought to be related to low virulence of agents, inoculum, change in specific cellular immunity (decreased number of T lymphocytes, like in large local bacterial inoculum, change in specific cellular immunity (decreased number of T lymphocytes, like in agammaglobulinemia, aplastic anemia, agranulocytosis and AIDS), or in humoral immune response (reduced agammaglobulinemia, aplastic anemia, agranulocytosis and AIDS), or in humoral immune response (reduced IgA IgA or increased IgE levels) (2). or increased IgE levels) (2). Most patients will present with localized disease on the extremities that may be preceded by trauma (2,3). CONCLUSION: Cutaneous botryomycosis is a relatively rare infectious disease process . Most patients will Cutaneous botryomycosis is the most common form of botryomycosis and usually occurs following cutaneous present with localized disease on the extremities that may be preceded by trauma (2,3). Cutaneous inoculation of bacteria due to trauma, surgery, or in conjunction with the presence of a foreign body. Lesions botryomycosis is the most common form of botryomycosis and usually occurs following cutaneous characteristically develop slowly and may evolve and enlarge for several months and, rarely, even years. inoculation of bacteria due to trauma, surgery, or in conjunction with the presence of a foreign body. Lesions The histopathologic appearance of botryomycosis is characterized by a central focus of necrosis surrounded by a characteristically develop slowly and may evolve and enlarge for several months and, rarely, even years. chronic inflammatory reaction containing histiocytes, epithelioid cells, multinucleated giant cells, and fibrosis (4).

Unlike the sulfur granules seen in (which contain filamentous branching organisms), the granules Figure 1: Right forearm exhibiting a1cm crusted ulceration with Figure 2: Left forearm exhibiting a 1 cm crusted ulceration with seen in botryomycosis contain bacteria surrounded by an eosinophilic matrix containing club-like projections. This surrounding erythema surFigure 1: Right forearm exhibiting a1cm crusted ulceration with surrounding erythema histologic appearance is commonly referred to as the Splendore-Hoeppli phenomenon, although it may not always rounding erythema be present (4). INTRODUCTION Diagnosing botryomycosis includes clinical suspicion and microbiologic studies. In general, patients should receive antibiotic therapy until signs and symptoms of have resolved. Antibiotics for cutaneous disease PATHOLOGY include: oral trimethoprim-sulfamethoxazole (10-12 mg/kg /d), oral clindamycin (30-40mg/kg/d), cephalexin • Botryomycosis is a rare, chronic, supprative, granulomatous infectious disease that affects (500mg QID), minocycline (100mg BID), doxycycline (100mg BID) or erythromycin (500 mg QID). (5,6) the skin and occasionally the viscera. • Staphylococcus aureus (40%) is the most common causative organism however it can also be caused by Pseudonomas aeruginosa (20%), coagulase-negative staphylococci, Streptococcus spp, Escherichia coli, and Proteus spp. (1-3). BIBLIOGRAPHY • Skin lesions can be single or multiple and present as cysts, accesses, fistulas, nodules, ulcers or plaques. (3) 1. . D.A. Mehregan, W.P. Su, J.P. Anhalt Cutaneous botryomycosis J Am Acad Dermatol, 24 (1991), pp. 393–396 2. Bonifaz A, Carrasco E. Botryomycosis. Int J Dermatol. 1996;35:381-8 CASE DESCRIPTION 3. Mechow N, Göppner D, Quist S, et al. Cutaneous botryomycosis diagnosed long after an arm injury. Journal Of The American Academy Of Dermatology. October 2014;71(4):e155-6. • A healthy 52-year-old Caucasian male with a past medical history of hypertension presented to our clinic 4. Schlossberg D, Pandey M, Reddy R. The Splendore-Hoeppli phenomenon in hepatic complaining of growths on bilateral forearms that developed two months prior. botryomycosis. J Clin Pathol 1998; 51:399 • The lesions began as small pink papules that grew over the course of a few weeks, ulcerated and developed 5. Askari, K., Seyed Saadat, S., Seyed Saadat, S., Yousefi, N., Ghorbani, G., & Zargari, O. (2014). a crusted scab. Cutaneous botryomycosis caused by Staphylococcus aureus in a patient with diabetes. • He admitted to occasional mild pruritus but denied any associated pain, tenderness or burning sensation of International Journal Of Dermatology, 53(4), 413-415 involved skin. Figure 3: Deep shave biopsy (HE 10X) demonstrating Figure 4: On higher power (HE, 40X) radiating deposits of pseudoepitheliomatous hyperplasia with sinus tract formation, focal 6. Fernandes NC, Maceira JP, Knackfuss IG, Fernandes N. Botriomicose cutânea. An Bras • Of note, he worked as a HVAC (heating, ventilation and air conditioning) repairman with a history of amorphous, eosinophilic, hyaline material around colonies of cocci supprative inflammation and focal granuloma formation with gram ositive bacteria were noted, characterizing the Splendore-Hoeppli Dermatol. 2002;77:65-70 repeated trauma to his forearms due to reaching through confined spaces of larger industrial units. cocci within the pseudoepitheliomatous hypersplasia. phenomenon