Le Infezioni in Medicina, n. 3, 184-187, 2009 Casi clinici hydrophila gangrenosum Case reports without bacteraemia in a diabetic man: the first case report in Italy Echtyma gangrenosum da in uomo diabetico in assenza di batteriemia: il primo caso segnalato in Italia

Manuela Avolio1, Claudio La Spisa2, Francesco Moscariello2, Rita De Rosa1, Alessandro Camporese1 1Microbiologia e Virologia, Dipartimento di Medicina di Laboratorio; 2Medicina d’Urgenza, Dipartimento di Emergenza, Azienda Ospedaliera S. Maria degli Angeli, Pordenone, Italy

n INTRODUCTION and suitable antibiotic therapy are important for the management of . eromonas hydrophila is a motile Gram-nega- In this report we present a case of A. hydrophila tive rod-shaped bacterium found in water ecthyma gangrenosum without bactaeremia in Asources that is typically pathogenic for fish. a diabetic male. Although human beings are atypical hosts, A. hy- drophila has been reported to cause a broad spec- trum of disease syndromes (acute , n CASE REPORT soft tissue infections, meningitis, peritonitis, sep- sis) [1]. A previously healthy 63-year-old with a chief While initially believed to be an opportunistic or- complaint of ulcer, painful swelling of the low- ganism only capable of infecting immunocom- er left leg was admitted at the Emergency De- promised individuals, a body of evidence now in- partment of S. Maria degli Angeli Regional dicates that A. hydrophila causes a range of ex- Hospital in Pordenone, Italy. traintestinal illness in immunocompetent hosts On admission he was febrile (temperature of [2-7]. 39.4°C), with BP 160/90, CR 107 for atrial fibril- In the literature A. hydrophila wound infections lation. The patient was obese, with a BMI of 30. reports are increasing and are represented by His anamnesis was positive for diabetes melli- three types in order of increasing severity: cel- tus and hypertensive cardiopathy. Examined lulitis, myonecrosis and ecthyma gangrenosum dermatologically he was treated for leg lesions [8-10]. with local (sulfadiazine argentic cream) and Ecthyma gangrenosum is a cutaneous necrotic systemic (amoxicillin/clavulanic acid 1 g x 2) or gangrenous pustule that occurs secondary to antibiotics. sepsis known to be caused by Pseudomonas The following day the pain became unbearable, aeruginosa sepsis usually in immunocompro- and the patient had two lipothymic episodes; mised people [11, 12]. he received liquid infusion and the antibiotic Lesions have an erythematous border sur- therapy started the previous day before was rounding a vesicle which can progress to necro- changed to amoxicillin/clavulanic acid 4g x 4 sis of the soft tissue within 24 hours. This type and clindamycin 600 mg x 3. After 24 hours the of infection is usually fatal [13]. Early diagnosis leg lesions became worse, appearing like

184 2009 ered to be pathognomonic of pseudomonas sepsis until it was described in cases of infec- tions by Group A , Aeromonas hy- drophila, Staphylococcus aureus, Serratia marcescens, and (15]. Ecthyma gangrenosum lesions charac- teristically begin as painless red macules that evolve into papules and later into haemorrhag- ic bullae (Figure 1). These ruptures produce gangrenous ulcers with a grey-black eschar. In classical bacter- aemic ecthyma gangrenosum, the lesions are a blood-borne metastatic seeding of the pathogens to the skin. Figure 1 - LEG lesions appearing like necrotic-hae- However, there are several reports that de- morrhagic bullae. scribe ecthyma gangrenosum unaccompanied by bacteraemia or systemic infection [16-18]. necrotic-haemorrhagic bullae (Figure 1), as- The absence of bacteraemia is associated with suming the appearance of pseudomonal ecthy- the best outcome [19]. ma gangrenosum lesions. Negative blood culture suggests that ecthyma The patient’s haemodynamic parameters re- gangrenosum occurred as a primary lesion at mained normal despite a temperature rise to the site of prior skin trauma. In our report, im- 38.8°C. Chest X-rays were normal but a blood pairment of local cutaneous mechanisms due to test showed a mild increase in WBC (12,800 µl) diabetes associated with exposure to contami- and CRP (27.7 mg/dl); indexes of hepatic and nated water is likely to explain the rapid wors- renal function were normal. ening of the infection, and the delayed response Given suspected pseudomonal ecthyma gan- to antibiotics, albeit in a patient with non-com- grenosum, and in the presence of negative promised immune systems and in the absence blood culture, a bloody exudate was collected of bacteraemia. by aspiration from the necrotic bullae for cul- Early diagnosis and adequate antibiotic therapy ture, and the antibiotic therapy was adjusted to are important for the management of ecthyma meropenem 3 g/die plus tigecycline 100 mg x 2 gangrenosum. on first day, then 50 mg/die. In case of lesions where neither Gram-positive The day after, the response of the microbiologi- nor anaerobic are detected, A. hydrophila cal culture showed evidence of Aeromonas hy- should be suspected and considered when se- drophila, only resistant to ampicillin, compatible lecting antibiotics, since Aeromonas strains are with ecthyma gangrenosum. mostly penicillinase-producers, hence resis- The patient, questioned once again, then re- tant to penicillin, ampicillin, carbenicillin and vealed that a few days prior to admission he piperacillin. had worked in a well near his house without More than half are resistant to cephalothin [20]. taking due precautions. Moreover, the typical presentation of A. hy- He was then transferred to another hospital for drophila soft tissue infection may well mimic a hyperbaric oxygen chamber therapy. Two Gram-positive infection, which may result in months later he was healthy and waiting for ad- delay in administration of appropriate antibi- mission to another hospital for skin grafting otics. Therefore it is important to stress the need surgery. to deliver prompt and appropriate empiric an- tibiotic therapy, to consider the presence of multidrug-resistant organisms, and to use the n DISCUSSION shortest duration of antibiotic therapy possible through antibiotic therapy de-escalation that is Ecthyma gangrenosum is a well-recognised driven both by available culture and sensitivity manifestation of pseudomonal sepsis in im- data and by the clinical status of the patient. munocompromised hosts. It occurs in only-1-6% of patients with Key words: Aeromonas hydrophila, ecthyma gan- pseudomonal sepsis [14]. It had been consid- grenosum, bacteraemia.

185 2009 SUMMARY

Ecthyma gangrenosum is a well recognized cuta- leg pseudomonal-ecthyma gangrenosum lesions neous manifestation of severe, invasive infection developed after admission. The patient, ques- by usually in immunocom- tioned again, stated that a few days before he had promised and critically ill patients. This type of in- worked in a well near his house without taking fection is usually fatal. Aeromonas infection is infre- precautions. quently reported as the cause of ecthyma gan- We conclude that early diagnosis and suitable an- grenosum. Here we show the first case described tibiotic therapy are important for the management in Italy of Aeromonas hydrophila ecthyma gangreno- of ecthyma gangrenosum. The typical presentation sum in the lower extremities in an immunocompe- of soft tissue infection of A. hydrophila should mim- tent diabetic without bacteraemia. ic a Gram-positive infection, which may result in a A 63-year-old obese diabetic male was admitted delay in administration of appropriate antibiotics. with an ulcer on his left leg, oedema, pain and Moreover, A. hydrophila should be considered a fever. Throughout his hospitalization blood cul- possible agent for non-pseudomonal ecthyma gan- tures remained sterile, but a culture of A. hydrophi- grenosum in a diabetic man with negative blood la was isolated following punctures from typical cultures, in presence of anamnestical risk factors.

RIASSUNTO

Introduzione: Ecthyma gangrenosum è un’infezione ma gangrenosum da Pseudomonas aeruginosa, e in cutanea tipicamente secondaria a sepsi da Pseudomonas mancanza di positivizzazione delle emocolture prelevate aeruginosa in pazienti immunocompromessi. Raramen- all’ingresso, viene aspirato del materiale direttamente te A. hydrophila è stata ritenuta causa di ecthyma gan- dalle lesioni da cui verrà isolato A. hydrophila. Il pa- grenosum. In questo report viene segnalato per la prima ziente interrogato nuovamente riferisce di avere lavora- volta in Italia un caso di ecthyma gangrenosum causa- to senza precauzioni in un pozzo qualche giorno prima to da A. hydrophila, in un soggetto diabetico e in assen- della comparsa dei sintomi. za di batteriemia. Conclusioni: Diagnosi precoce e terapia antibiotica mira- Presentazione del caso: Un uomo di 63 anni si pre- ta sono essenziali nel corretto management dell’ecthyma senta al pronto soccorso con un quadro di cellulite simil- gangrenosum. Infatti nelle fasi iniziali l’aspetto dell’ecthy- streptococcica localizzata alla gamba sinistra. Il pazien- ma gangrenosum può mimare un’infezione da Gram posi- te, diabetico ed obeso, è febbrile e lamenta dolore alla tivi dei tessuti molli. In questi casi A. hydrophila dovrebbe gamba. Viene impostata la terapia antibiotica del caso, essere preso in considerazione come agente eziologico, an- ma nei giorni successivi il quadro clinico non migliora, che in assenza di batteriemia e in soggetti immunocompe- le lesioni aumentano ed acquisiscono un aspetto bolloso tenti, così da indagare su una pregressa esposizione ad am- e confluente con odore sgradevole. Nel sospetto di ecthy- bienti potenzialmente contaminati.

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