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Cutaneous Infectious Granuloma Caused by Phenylobacterium in an Adult with Myelodysplastic Syndrome A First Case Report Xiao-hua Zhu, Feng Li, Jin-hua Xu, Lei-hong Xiang and Ke-fei Kang Department of Dermatology,This Huashan Hospital, Fudan material University, Shanghai, China is Abstract Painful granulomatous lesions appeared on the face of a 36-year-old man with myelodysplastic syn- thedrome. Skincopyright biopsy revealed chronic inflammatory granuloma. of Bacterial the cultures of the lesions and blood indicated the same unknown Gram-negative rod bacterium. The 16S ribosomal RNA sequence of the unknown bacterium yielded Phenylobacterium. Thus, we diagnosed cutaneous infectious granuloma caused by Phenylobacterium and myelodysplastic syndrome/refractory cytopenia with multi-lineage dysplasia. originalAfter treatment with combined antibacterials publisher. that were selected based on the tests for drug sensitivity, the lesions disappeared with only scars remaining and without any signs of relapse after 1 year. This is the first case report of cutaneous infectious granuloma caused by Phenylobacterium. InfectiousUnauthorised granulomatous lesions may present as skin hemoglobin copying 8.3 g/dL (reference value 12–16 g/dL), and granulomas infected by Mycobacterium, Trypanosoma cruzi, platelets 32.9 · 109/L (reference value 100–300 · 109/L). Bone and Leishmania species. We report a case of a patient with marrow aspirate indicated myelodysplastic syndrome/refractory myelodysplastic syndrome complicated with Phenylobacterium- cytopenia with multi-lineage dysplasia. Cytogenetic analysis infected granulomatous lesionsand on his face. distributionwas abnormal: 46, XY, trp (1) (q21q32) [20]. Erythrocyte sedimentation rate was 28 mm/h (reference value 0–10 mm/h). Liver and renal function, electrolytes, blood sugar, and tumor markers such as prostate-specific antigen, carcino-embryonic 1. Case Report is prohibited.antigen, a-fetoprotein, and carbohydrate antigen 50 were nor- A 36-year-old Chinese man visited our department in mal. Hepatitis B surface antigen (HBsAg), hepatitis B envelope January 2007, presenting with painful pyogenic granulomatous antigen (HBeAg), antibodies to HBeAg, hepatitis B core anti- lesions on his face. The lesions were tender and had been pre- gen, hepatitis A virus IgM, hepatitis C virus, HIV, the rapid sent for 15 months. He had been treated with systemic peni- plasma reagin test, and rheumatoid factor were all negative. cillins and mupirocin ointment at primary-care centers, with no Lesion biopsy showed epithelial proliferation, and dermal improvement, and his lesions had subsequently worsened. He inflammation characterized by diffuse infiltration of primary had a history of trauma to the face. A diagnosis of myelodys- lymphocytes, histiocytes, plasmocytes, with occasional neutro- plastic syndrome was confirmed by an internal medicine spe- phils, and multinucleated giant cells (figure 2a) indicating chronic cialist 2 months prior to the lesions occurring. inflammatory granuloma. Physical examination showed many nodules ranging from Bacterial cultures of the lesional surface indicated the size of a pinhead to 2–3 cm in diameter distributed around Staphylococcus epidermidis. Fungal and Mycobacterium cul- the cheek, nasal, and submaxilla regions. Most of the lesions tures of the lesional surface and blood were negative. However, were ulcerated and covered with crusts (figure 1a). bacterial cultures of the lesions and blood both indicated the Laboratory tests showed peripheral blood pancytopenia: same unknown Gram-negative bacterium, with a rod shape and white blood cell count 2.3 · 109/L (reference value 4.5–11 · 109/L), varying in size from 0.3–0.5 mm in diameter and 0.5–2 mmin 364 Zhu et al. a b This material is the copyright of the cd original publisher. Unauthorised copying and distribution is prohibited. Fig 1. (a) Lesions before treatment; (b) lesions after 1 month of treatment; (c) lesions after 3 months of treatment; and (d) lesions 1 year later. length (figure 2b).The cultures were plated onto blood agar (5% gene (primers: 23F, AGAGTTTGATC(C/A)TGGCTCAG; sheep erythrocytes) and incubated in a humidified incubator at 1492R, TACGG(C/T)TAC CTTGTTACGACTT). The PCR 37°C for 4 days. The colonies were grayish, circular, and convex product was 1504 bp (figure 2d), and was followed by sequen- with smooth edges and a smooth surface (figure 2c). The un- cing using the ABI PrismÒ 377 DNA Sequencer (Shanghai known bacterium was further studied with the APIÒ 20NE test Invitrogen Biotech Co. Ltd, Shanghai, China). The 16S rRNA kit (for identification of Pseudomonas alcaligenes or Coma- sequence of the unknown bacterium was compared with the monas testosteroni) [bioMe´rieux, Marcy l’Etoile, France] with known sequences in the GenBank/EMBL/DDBJ databases. negative results. Drug sensitivity tests showed that the bacter- The unknown bacterium shares 99% identity of the 16S rRNA ium was sensitive to erythromycin, gentamicin, ciprofloxacin, sequences with Phenylobacterium (gene bank accession number cefoperazone, ceftazidime, aztreonam, and meropenem, and AY628697). Even though Phenylobacterium, P. alcaligenes, resistant to piperacillin and piperacillin/tazobactam. and C. testosteroni all belong to the Proteobacteria group, since Purified DNA 0.5 mg was subjected to polymerase chain re- Phenylobacterium is not identified by the APIÒ 20NE test kit, action (PCR) amplification for the 16S ribosomal RNA (rRNA) the unknown bacterium was then convincingly categorized into ª 2010 Adis Data Information BV. All rights reserved. Am J Clin Dermatol 2010; 11 (5) Cutaneous Infectious Granuloma caused by Phenylobacterium 365 the genus Phenylobacterium. Thus, the patient was diagnosed bacteriology, and molecular biology to confirm the diagnosis of with cutaneous infectious granuloma caused by Phenylo- cutaneous infectious granuloma caused by Phenylobacterium. bacterium and myelodysplastic syndrome/refractory cytopenia To our knowledge, this is the first case in a human that has with multi-lineage dysplasia. been reported in the literature. The genus Phenylobacterium at We commenced treatment with oral erythromycin (1 g/day), present comprises four species,[1-4] which have been isolated levofloxacin (500 mg/day), testosterone undecanoate (andriol; from a subsurface aquifer, alkaline groundwater, soil, and 80 mg/day), leucogen (60 mg/day), folic acid (30 mg/day), and activated sludge from a wastewater treatment plant, respect- vitamin B12 (cyanocobalamin; 75 mg/day) simultaneously. One ively. In our case, it appears that the skin wound on the month later, the lesions had shrunk in size and reduced in patient’s face caused this opportunistic infection. It has been number and no new lesions had occurred (figure 1b). The peri- recently documented that a new strain of Phenylobacterium pheral blood cell count had increased. Three months later, all was isolated from a human erythroleukemia cell line,[5] so the lesions had disappeared,This with only the scars material remaining clinicians need to be awareis of the pathogenic relevance to (figure 1c). The antibacterial treatment was then withdrawn. human disease. The patient remained in remission at the 1-year follow-up Myelodysplastic syndrome is characterized by one or more without any signs of clinical relapse (figure 1d). cytopenias despite a relatively hypercellular bone marrow, and the copyrighthas a tendency of to transform the into acute myeloid leukemia. Op- portunistic infections have been reportedly associated with 2. Discussion myelodysplastic syndrome; Mycobacterium avium,[6] Myco- We present an unusual case with multiple tumor-like granu- bacterium kansasii,[7] and Bordetella hinzii[8] have been isolated lomas on the face. Theoriginal lesions were studied by histopathology, publisher.from patients with myelodysplastic syndrome. Unauthoriseda b copying and distribution is prohibited. cd bp 2000 1504 1000 Fig 2. (a) Histopathology showing chronic inflammatory granuloma (hematoxylin and eosin stain; original magnification