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Ecthyma Gangrenosum: a Rare Cutaneous Manifestation Caused by Stenotrophomonas Maltophilia in a Leukemic Patient

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Ecthyma Gangrenosum: a Rare Cutaneous Manifestation Caused by Stenotrophomonas Maltophilia in a Leukemic Patient Ann Dermatol Vol. 21, No. 4, 2009 CASE REPORT Ecthyma Gangrenosum: A Rare Cutaneous Manifestation Caused by Stenotrophomonas maltophilia in a Leukemic Patient Young Min Son, M.D., So Young Na, M.D., Hye Young Lee, M.D., Jin Ok Baek, M.D., Jong Rok Lee, M.D., Joo Young Roh, M.D. Department of Dermatology, Gachon University of Medicine and Science, Gil Medical Center, Incheon, Korea Ecthyma gangrenosum (EG) is a well-recognized cutaneous pressed and burn patients1,2. It usually occurs as a result of infection that most commonly affects immunocompromised bacteremia or, rarely, as a primary cutaneous lesion. The patients. It typically occurs on the extremities, or in gluteal lesions are present in the gluteal and perineal regions or and perineal regions. Although Pseudomonas aeruginosa is on the extremities, but they can occur anywhere on the the most well-known pathogen causing EG, other organisms body1,3,4. Most cases of ecthyma gangrenosum are asso- have been reported to cause EG. Herein we report a rare case ciated with Pseudomonas aeruginosa bacteremia5,6. But of ecthyma gangrenosum presenting as aggressive necrotic numerous other organisms have been reported to cause skin lesions in perioral and infraorbital areas in a 47-year-old EG. patient with acute myelocytic leukemia after allogeneic Stenotrophomonas maltophilia is an aerobic gram-neg- bone marrow transplantation. It was caused by Stenotropho- ative bacillus that is a frequent colonizer of fluids used in monas maltophilia, which is an aerobic, gram-negative hospital settings. The incidence of S. maltophilia infection pathogen that has been associated only rarely with cuta- has been increasing. Until now, one case of S. maltophilia neous disease. Blood culture and tissue culture were positive infection, paronychia, have been reported in Korea7, and for S. maltophilia. Histological examination revealed numer- several cases of mucocutaneous, skin and soft tissue in- ous tiny bacilli in the dermis and perivascular area. Early rec- fection have been reported abroad8-10. ognition of skin lesions caused by S. maltophilia is important It is important to identify early the skin lesions caused by to decrease associated mortality in immunosuppressed S. maltophilia because it is associated with a high mortal- patients. (Ann Dermatol 21(4) 389∼392, 2009) ity in immunocompromised hosts. -Keywords- CASE REPORT Ecthyma gangrenosum, Stenotrophomonas maltophilia A 47-year-old man with a history of AML stage M2 was admitted to the hematology-oncology department due to INTRODUCTION high fever. Nine days after an allogeneic bone marrow transplantation, he developed a 38.8oC-high fever. Intrave- Ecthyma gangrenosum (EG) is a well known cutaneous nous cefazolin, teicoplanin, metronidazole, and mer- manifestation that most commonly affects immunosup- openem were maintained after admission. The next day, he developed rapid, progressive, severe, edematous, er- ythematous, and necrotic plaques with bullae formation Received January 19, 2009, Revised February 9, 2009, Accepted for around the lips (Fig. 1) and was referred to the dermatol- publication February 10, 2009 ogy department for evaluation of the skin lesions. A skin Reprint request to: Joo Young Roh, M.D., Department of Dermatology, Gachon University of Medicine and Science, Gil Medical Center, biopsy from a perioral necrotic hemorrhagic vesicle with 1198, Guwol-dong, Namdong-gu, Incheon 405-760, Korea. Tel: 82- blood culture, fungus culture, and open pus culture was 32-460-2000, Fax: 82-32-460-2001, E-mail: [email protected] performed. Two days later, he suffered from bradycardia, Vol. 21, No. 4, 2009 389 YM Son, et al hypotension, and respiratory distress and expired. Blood tions of gram-negative bacilli which were subsequently and tissue culture results revealed S. maltophilia which identified as S. maltophilia on blood culture (Fig. 3). was resistant to the antibiotics that he had been treated with; this pathogen was sensitive to trimethoprim-sulfame- DISCUSSION thoxazole. Although an autopsy was not done, the cause of death was probably sepsis caused by S. maltophilia. EG is a rare necrotizing vasculitis3 and a well recognized The histopathologic findings included a sub-epidermal manifestation that occurs in debilitated persons who are blister with hemorrhage and dermal edema with vascular suffering from leukemia, in severely burned patients, in congestion (Fig. 2A). There was perivascular cuffing with pancytopenia or neutropenia, and in patients with a func- basophilic deposits accompanied by a mixed inflam- tional neutrophilic defect, terminal carcinoma, or other se- matory cell infiltrate of lymphocytes and histiocytes in the vere chronic diseases1. It most commonly affects im- dermis. Basophilic deposits suggestive of bacterial colo- munocompromised and burn patients but it also occurs in nies surrounded the affected vessels and were scattered in the perineal area of healthy infants after antibiotic therapy the interstitial dermis. In addition, intravascular thrombo- in conjunction with maceration of the diaper area1,3. The sis was present with endothelial swelling (Fig. 2B). A tis- skin lesions of ecthyma gangrenosum are the manifes- sue Gram stain revealed basophilic deposits to be collec- Fig. 1. Severe edematous erythematous necrotic plaques with Fig. 3. Numerous gram-negative bacilli surround papillary perioral bullae formation. dermal vessels and infiltrate the tissue (Gram stain, ×1,000). Fig. 2. (A) Skin biopsy specimen showing subepidermal blister with hemorrhage and dermal edema with vascular congestion (H&E, ×100). (B) Perivascular cuffing with basophilic deposits accompanied by a mixed inflammatory infiltrate of lymphocytes and histiocytes in dermis. Intravascular thrombosis was present with endothelial swelling (H&E, ×200). 390 Ann Dermatol Ecthyma Gangrenosum: A Rare Cutaneous Manifestation Caused by Stenotrophomonas maltophilia in a Leukemic Patient tation of a necrotizing vasculitis3. The lesion characteristi- relatively low mortality, ranging from 7% to 15% in a re- cally begins as an erythematous nodule, macule, vesicle, cent series, but the mortality rate in patients with bacter- or bullae and evolves into gangrenous ulcerations with emia ranges from 38% to 96%3. The physical finding of black eschar and a surrounding rim of erythema6,11,12. The bullous cellulitis is associated with higher mortality, and vesicles, initially filled with serous fluid, appear on the the number of skin lesions is positively correlated with the surface of the edematous skin, and then coalesce to form severity of the disease3. Other prognostic factors include large bullae. The bullae slough away, leaving ulcerated, the time of onset of antibiotic therapy and the patient’s re- necrotic centers with erythematous halos3,13. The matura- sponse to therapy11. tion of the lesion is very rapid and often occurs in less Stenotrophomonas maltophilia, formerly named Xantho- than 24 hours as in our patient11. Fifty seven percent of monas maltophilia and Pseudomonas maltophilia, is an skin lesions were located in the gluteal region, 30% on aerobic, gram-negative bacillus and has emerged as an im- the extremities including the axillae, and 6% on the trunk. portant opportunistic nosocomial infection often asso- Facial involvement is unusual, occurring only in 6% of ciated with central venous catheter-related bacteremia16,17. cases3. It is a frequent colonizer of fluids used in the hospital set- The pathogenic mechanism of EG is unknown, but several ting, such as nebulizers, dialysis machines, water baths possible mechanisms have been suggested6. Two mecha- and intravenous fluids8. Colonization with S. maltophilia nism of EG have been well described. In classic bactere- is seen to a greater degree in patients treated with broad- mic EG, the skin lesions are considered to represent spectrum antibiotics as in our patient10. It grows slowly in blood-borne metastatic seeding of an organism into the disinfectant agents and has a high rate of mutation, which skin5,6,14. Our patient also revealed a positive blood cul- can confer resistance to antibiotics16. Resistance to be- ture result. In nonbacteremic EG, the lesion is located at ta-lactams, carbapenems, aminoglycosides, and quino- the site of entry or inoculation of the organism into the lones is well known16. skin5,6. Some investigators have suggested that non- Skin and soft tissue manifestations of S. maltophilia in- bacteremic EG may be an early type of EG6. fection are becoming increasingly well recognized8,9, and Although Pseudomonas aeruginosa is the most common are most frequently associated with posttraumatic, pathogen of EG-associated bacteremia, other organisms post-surgical, or burn-related wounds and chronic cuta- have been reported. The most commonly reported one are neous ulcers16. Clinical manifestations include cellulitis, Citrobacter freundii, Aeromonas hydrophila, and Escheri- cellulitis-like skin lesions, infected mucocutaneous ulcers, chia coli3,5,15. Our case is unique in that the patient’s EG EG and paronychia7-10,18. The route of transmission is lesion was caused by a rarely isolated gram-negative ba- mainly unknown but it seems likely that invasion takes cillus, S. maltophilia. place via defects in mucous membranes and by colo- Histopathologically, EG is characterized by epidermal ne- nization of central venous catheters10. crosis with hemorrhage and dermal infarction, usually ac- Infections with S. maltophilia can be life-threatening be- companied by a mixed inflammatory
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