Cutaneous botryomycosis diagnosed long after an arm injury (2014)

To the Editor: We report a 44-year-old Turkish man who presented with an expanding, brownish-reddish to hypopigmented, nodular, scarred skin lesion with multiple ulcerations on his right arm, primarily at the elbow and fingers (Fig 1). The fingers of the right hand showed flexion contractures, and the ulcerated nodules were located above the proximal interphalangeal joints (Fig 1). The patient reported that his skin problems were initiated by a work-related welding accident 25 years ago, wherein the lower aspect of his arm was burned by iron filings. Thereafter, the patient was seen at various dermatologic and surgical clinics. Tuberculostatic treatments were administered repeatedly because of the patient's history of lung tuberculosis 7 years before his arm injury, although tests were consistently negative for Mycobacterium tuberculosis. In addition, sarcoidosis, atypical mycobacteriosis, lupus vulgaris, Wegener granulomatosis, leprosy, and skin neoplasms were excluded by prior histopathological and microbiological examinations; the correct diagnosis remained unknown. Despite continuous medical care, the skin never properly healed. Treatment with alprostadil (20 μg intravenously, 20 infusions over 2 months, 3 times repeated within 6 years) temporarily improved his skin condition by decreasing the number of skin ulcers. He presented at our department approximately 13 years after his skin condition initially developed. Histopathological examination revealed the Splendore-Hoeppli phenomenon; eosinophilic material adjacent to the inciting grains was surrounded by inflammatory cells consisting of eosinophils, histiocytes, epithelioid cells, and giant cells as described by Hussein.1Hussein M.R. Mucocutaneous Splendore-Hoeppli phenomenon.J Cutan Pathol. 2008; 35: 979-988Crossref PubMed Scopus (118) Google Scholar More importantly, clusters of gram-positive cocci (Fig 2), confirmed as Staphylococcus aureus, were observed. Fungi and acid-fast rods were not detected. A complete blood cell count showed hypochromic, normocytic anemia. Angiotensin-converting enzyme, glycosylated hemoglobin, and α1-antitrypsin levels were within normal ranges. Blood cultures produced negative results. Tuberculosis was not detected in skin samples (polymerase chain reaction) or gastric fluids or by puncture test (tine test) or enzyme-Linked Immunospot. Infectious diseases, including HIV, syphilis, and hepatitis (A, B, and C), were excluded. No signs of the following were detected: thrombophilia, cold agglutinins, cryoglobulins and cryofibrinogens, antinuclear antibodies, and neutrophil cytoplasmic antigens. The phagocytosis and chemotaxis of immune cells such as neutrophils was normal. Magnetic resonance imaging of the right arm showed inflammation of the cutis and subcutis and early signs of osteomyelitis. Chest x-rays and abdominal ultrasonography revealed normal findings. A psychosomatic examination revealed an auto-aggressive personality. Detection of S aureus in the skin lesions confirmed the diagnosis. The lesion had been partially maintained through the patient's self-manipulation to attain his pension claim. Clindamycin matched the antibiogram, and a fusidic acid lotion was applied topically. Iloprost (5 μg) was administered intravenously to enhance blood flow to the arm. Physiotherapy included manual lymphatic drainage of the right arm. In addition, compression bandages were applied to prevent further self-manipulation. Altogether, these treatments improved his skin condition.Fig 2Histopathology of a biopsy specimen of the lower ventral aspect of the arm. Glandlike masses of gram-positive cocci (arrows) typical of staphylococcal botryomycosis were detected by Accustain Gram stain (Sigma, Steinheim, Germany). (Original magnification: ×100.)View Large Image Figure ViewerDownload Hi-res image Download (PPT) Cutaneous botryomycosis is a chronic granulomatous bacterial skin infection characterized by nodules, abscesses, and ulcers on the hands, feet, genitals, and head.2Mehregan D.A. Su W.P. Anhalt J.P. Cutaneous botryomycosis.J Am Acad Dermatol. 1991; 24: 393-396Abstract Full Text PDF PubMed Scopus (47) Google Scholar It primarily results from the inoculation and persistent infection of injured skin3Karthikeyan K. Thappa D.M. Jeevankumar B. Cutaneous botryomycosis in an agricultural worker.Clin Exp Dermatol. 2001; 26: 456-457Crossref PubMed Scopus (9) Google Scholar by bacteria such as S aureus4Akiyama H. Kanzaki H. Tada J. Arata J. Staphylococcus aureus infection on cut wounds in the mouse skin: experimental staphylococcal botryomycosis.J Dermatol Sci. 1996; 11: 234-238Abstract Full Text PDF PubMed Scopus (57) Google Scholar and Pseudomonas aeruginosa. T cell–related immune deficiency and diabetes can promote the disease.2Mehregan D.A. Su W.P. Anhalt J.P. Cutaneous botryomycosis.J Am Acad Dermatol. 1991; 24: 393-396Abstract Full Text PDF PubMed Scopus (47) Google Scholar