Human infection due to CDC group IV c-2, a gram negative bacillous, are rare. We describe a case of nosocomial bacteremia caused by this organism in a neutropenic patient with acute lymphoblastic leukemia and include a literature review of CDC group IV c-2 infection in patients with hematologic malignancies

Carratalà, Jordi

González Barca, Eva

Grañena Batista, Alberto

Salar, Antonio

Zurita, Amado

Haematologica

English

Diposit Digital de la Universitat de Barcelona

Scientific letters670radiation therapy to the mediastinum (36 Gy). Thepatient achieved CR in February 1995. Over the next14 months she remained well and in complete hema-tologic remission. In May 1996 she noted extensiveecchymoses and petechiae. Her hematocrit was43.2%, leukocyte count 0.643109/L and the plateletcount 1.03109/L. The spleen was not palpable andthere was no evidence of HD relapse. A bone marrowbiopsy revealed normal erythroid and myeloid matu-ration. The diagnosis of ITP was suspected and a tri-al of corticosteroids was begun. After 30 days theplatelet count was normal (2243109/L) and corti-costeroids were stopped. To date, the patientremains in complete hematologic (ITP) and HDremission.  Although some studies have reported thatthe presence of ITP in HD patients could signify activelymphoma, especially with splenic involvement,2 oth-er reports have shown that ITP can occur in theabsence of active HD,6 and that the HD status isindependent of the onset of ITP. Our two patients did not have active HD when theydeveloped ITP, and the development of ITP did notindicate subsequent relapse, supporting the hypoth-esis of two independent diseases with differentonsets. Furthermore, as for primary ITP occurring inassociation with HD, so thrombocytopenic purpuraalso seems to respond to conventional therapy.2 Bothpatients responded with a single course of oral pred-nisolone, but one relapsed after 15 months andsplenectomy was necessary for CR.5The recognition of a picture of ITP as a complicationof HD could have important implications, asdescribed by Doan and Bouroncle.7 We suggest, there-fore, that an occult HD should be considered in anypatient who presents with thrombocytopenic purpu-ra of the ITP type, and particularly in a patient knownto have been previously treated for HD, where the ITPpicture may signal the recurrence of lymphoma. Itshould be noted that we have also observed throm-bocytopenic purpura in chronic myeloid leukemia(CML) during treatment with interferon-a.8In conclusion, ITP associated with HD responds totherapy in a similar manner to that of primary ITP. Itspresence seems to confer no prognostic significance,9although the possibility of localization of splenic HDdisease10 has to be considered.CorrespondenceGiovanni Martinelli, MD, Institute of Hematology and Med-ical Oncology “Seràgnoli”, University of Bologna, Policlinico S.Orsola, via Massarenti 9, 40138, Bologna, Italy. Phone: inter-national +39-051-6363680 • Fax: international +39-051-398973 • E-mail: seragnol@kaiser.alma.unibo.itReferences1. Jones SE. Autoimmune disorders and malignant lym-phoma. Cancer 1973; 31:1092-8.2. Rudders RA, Aisenberg AC, Schiller AL. Hodgkin’s dis-ease presenting as idiopathic thrombocytopenic pur-pura. Cancer 1972; 30:220-30.3. Santoro A, Bonadonna G, Valagussa P. Long termresults of combinated chemotherapy radiotherapyapproach in Hodgkin’s disease: superiority of ABVDplus radiotherapy versus MOPP plus radiotherapy. JClin Oncol 1987; 5:27-30.4. Mazza P, Poletti G, Zinzani PL, et al. Critical value oflaparatomy with splenectomy in stage III Hodgkin’sdisease as restaging procedure after chemotherapy.Eur J Haematol 1988; 41:6-11.5. Silvestri F, Russo D, Fanin R, et al. Laparoscopicsplenectomy in the management of hematologic dis-eases. Haematologica 1995; 80:47-9.6. Cohen JR. Idiopathic thrombocytopenic purpura inHodgkin’s disease. Cancer 1978; 41:733-6.7. Doan CA, Bouroncle BA, Wiseman BK. Idiopathic andsecondary thrombocytopenic purpura; clinical studyand evaluation of 381 cases over a period of 28 years.Ann Intern Med 1960; 53:861-76.8. Zuffa E, Vianelli N, Martinelli G, Tazzari P, Cavo M,Tura S. Autoimmune mediated thrombocytopeniaassociated with the use of interferon-a in chronicmyeloid leukemia. Haematologica 1996; 86:533-59. Bettini R, Tonolini M, Maccianti E. Verification andcomparison of two different predictive equations inHodgkin’s disease. Haematologica 1997; 82:324-7.10. Iannitto E, Accurso V, Federico M, et al. Hodgkin’sdisease presenting below diaphragm. The experienceof the Gruppo Italiano Studio Linfomi (GISL). Haema-tologica 1997; 82:676-82.Bacteremia caused by CDC Group IV c-2 in apatient with acute leukemiaANTONIO SALAR, JORDI CARRATALÀ,* AMADO ZURITA,EVA GONZÁLEZ-BARCA, ALBERTO GRAÑENADepartment of Clinical Hematology, Institut Català d’Oncologiaand *Department of Infectious Diseases, Hospital Príncipes deEspaña, Ciudad Sanitaria y Universitaria de Bellvitge, Hospitaletde Llobregat, Barcelona, SpainHuman infections due to CDC group IV c-2, a gram-neg-ative bacillus, are rare. We describe a case of nosoco-mial bacteremia caused by this organism in a neu-tropenic patient with acute lymphoblastic leukemia andinclude a literature review of CDC group IV c-2 infectionin patients with hematologic malignancies.Human infections due to CDC group IV c-2, a gram-negative bacillus, have been recently reported.1-8 Bac-teremia caused by this organism is rare in patientswith hematologic malignancies.2,5,6,8We report here a case of septicemia caused by CDCgroup IV c-2 in a neutropenic patient with acuteleukemia and review the literature on this entity(MEDLINE, National Library of Medicine, Bethesda,MD covering the years 1965-97).A 71-year-old previously healthy woman wasadmitted to our hospital with a temperature of37.4ºC, dyspnea and pleuritic chest pain. The patienthad a history of fatigue, weakness and weight loss of10 kg over the three previous months. Physical exam-ination revealed cutaneous pallor, shortness ofbreath and a few crackles at the left lung base. Whiteblood cell count was 1883109/L with 95% blasticforms, the hemoglobin level was 6.8 g/dL andplatelet count 443109/L. A chest radiograph showeda left lower lobe infiltrate and a small left pleural effu-sion. Empirical therapy with amoxicillin/clavulanatewas administered intravenously. On the second dayof admission the patient was afebrile. Sputum andblood cultures were negative.Microscopical examination of a bone marrow aspi-rate revealed acute lymphoblastic leukemia, L-2according to the criteria of the FAB classification.Immunophenotype was consistent with ALL-preB. Aninduction chemotherapy regimen consisting ofdaunorubicin, vincristine, L-asparaginase and pred-nisone was initiated.After two weeks of antibiotic treatment, the patien-t’s condition improved and a new chest radiographwas normal. Amoxicillin/clavulanate was stopped andoral prophylaxis with norfloxacin was then initiated.Twelve days later, while neutropenic, the patient’stemperature rose to 38.5ºC and her blood pressure fellto 80/50 mm Hg. After acquisition of two specimensfor blood culturing, fluid replacement was initiatedand, ceftazidime and amikacin were given intra-venously. Twenty-four hours after beginning empiri-cal antibiotic therapy she became afebrile with no clin-ical signs of infection. A gram-negative rod was iso-lated from all the blood cultures. Cultures from urineand indwelling line specimens were negative.The chemotherapy course was complicated by sev-eral episodes of intestinal bleeding due to severethrombocytopenia. Several days later, the patientbecame agitated and severe hypotension and oligoa-nuria developed despite fluid replacement andinotropic drug therapy. The patient died because ofa new bleeding episode. At that time blood cultureswere negative. The isolate was identified as CDC group IV c-2 byconventional methods from the blood cultures after72 hours. Its microbiological characteristics were con-sistent with those previously defined for the bacil-lus.9,10 With standard disk diffusion testing, the organ-ism was susceptible to trimethoprim-sulfametho-xazole, ciprofloxacin and ceftazidime, but was resis-tant to ampicillin, amoxicillin plus clavulanate, piper-acillin, ticarcillin, ticarcillin plus clavulanate, cephalo-thin, cefoxitin, cefotaxime, ceftriaxone, imipenem,aztreonam, gentamicin, tobramycin, amikacin andtetracycline.CDC group IV c-2 is a gram-negative environmen-tal bacillus rarely isolated from clinical specimens.However, several cases of well-documented infectionscaused by this organism have been reported, witheight cases of septicemia (five of them occurring in ahospital outbreak),2,5,6,8 two cases of peritonitis relat-ed to peritoneal catheter infection,1,4 one case ofplantar abscess complicated with bacteremia3 andone case of tenosynovitis.7 All but one of thesepatients had underlying disease. We found five pre-viously reported cases of infection caused by CDC IVc-2 in patients with hematologic malignancies in ourreview of the literature.2,5,6,8 These 5 cases with theaddition of our case are summarized in Table 1. Fourpatients had acute leukemia and two non-Hodgkin’slymphoma. Our review shows that most of the infec-tions were nosocomial-acquired and presented withbacteremia, mainly catheter-related, occurring duringchemotherapy-induced neutropenia.In spite of the well-known severity of gram-negativebacteremia in patients with cancer, all reportedpatients with bacteremia caused by CDC group IV c-2 were cured from infection following appropriateantimicrobial therapy, probably indicating a low lev-el of pathogenicity of this organism. It should be not-ed that catheter removal was only necessary in one ofthese cases.It has been shown that CDC group IV c-2 is sus-ceptible to a wide range of antimicrobial agents,including aminopenicillins, antipseudomonal peni-cillins, cephalosporins, carbapenems and fluoro-quinolones. However, this organism is usually resis-tant to aminoglycosides.1-8,10 In our case, the organ-ism showed an unusual pattern of antibiotic-suscep-tibility, showing resistance to a broad spectrum ofantimicrobial agents. To our knowledge, this is theScientific letters 671Table 1. CDC group IV c-2 infection in patients with hematologic malignancies.Reference Age/Sex Underlying condition Neutropenia Specimen cultured Source of infection Acquisition OutcomeDan et al.2 37/M Plasma cell leukemia No Blood Unknown Community RecoveryArduino et al.5 77/M Non Hodgkin’s lymphoma No? Blood Catheter Nosocomial RecoveryRamos et al.6 10/F Acute leukemia Yes Blood Catheter Nosocomial RecoveryMoissenet et al.8 11/NR Non Hodgkin’s lymphoma* Yes Blood Catheter Nosocomial RecoveryMoissenet et al.8 14/NR Acute leukemia* Yes Blood Catheter Nosocomial RecoverySalar et al.PR 77/F Acute leukemia Yes Blood Unknown Nosocomial RecoveryPR, present report; NR, not reported; *cases reported in a hospital outbreak of CDC group IV c-2 infection.Scientific letters672first report in which resistance of this bacillus toimipenem is noted.In summary, CDC group IV c-2 bacillus is anopportunistic pathogen that rarely causes bacteremiain patients with hematologic malignancies andindwelling intravenous lines.Key wordsCDC group IV c-2, bacteremia, acute leukemia, neu-tropenia.FundingThis work was supported by a grant (AG/97) from theJosé Carreras International Leukemia Foundation.CorrespondenceAntonio Salar, MD, Department of Clinical Hematology,Institut Català d’Oncologia, Autovía de Castelldefels km 2.7,08907 Hospitalet de Llobregat, Barcelona, Spain.  Fax: inter-national + 34-3-263-4579 • E-mail: 28609ass@comb.esReferences1. Hansen W, Glupczynski Y. Group IV c-2 associatedperitonitis. Clin Microbiol Newsl 1985; 7:43-4.2. Dan M, Berger SA, Aderka D, Levo Y. Septicemiacaused by the gram-negative CDC group IV c-2 in animmunocompromised human. J Clin Microbiol 1986;23:803.3. Crowe HM, Brecher SM. Nosocomial septicemia withCDC group IV c-2, an unusual gram-negative bacillus.J Clin Microbiol 1987; 25:2225-6.4. Zapardiel J, Blum G, Caramelo C, Fernández-RoblasR, Rodriguez-Tudela JL, Soriano F. Peritonitis withCDC group IV c-2 bacteria in a patient on continuousambulatory peritoneal dialysis. Eur J Clin MicrobiolInfect Dis 1991; 10:509-11.5. Arduino S, Villar H, Veron MT, Koziner B, Dictar M.CDC group IV c-2 as a cause of catheter-related sep-sis in an immunocompromised patient. Clin Infect Dis1993; 17:512-3.6. Ramos JM, Soriano F, Bernacer M, Esteban J, Zapar-diel J. Infection caused by the nonfermentative gram-negative bacillus CDC group IV c-2: Case report andliterature review. Eur J Clin Microbiol Infect Dis 1993;12:456-8.7. Musso D, Drancourt M, Bardot J, Legré R. Humaninfection due to the CDC group IV c-2 bacterium:Case report and review. Clin Infect Dis 1994; 18:482-4.8. Moissenet D, Tabone MD, Girardet JP, Leverger G,Garbarg-Chenon A, Vu-Thien H. Nosocomial CDCgroup IV c-2 bacteremia: epidemiological investiga-tion by randomly amplified polymorphic DNA analy-sis. J Clin Microbiol 1996; 34:1264-6.9. Gilardi GL. Identification of glucose-nonfermentinggram-negative bacilli. Clin Microbiol Newsl 1984;6:126-9.10. Gilardi GL. Antimicrobial susceptibility of glucose-nonfermenting gram-negative bacilli. Clin MicrobiolNewsl 1984; 6:149-52.

Bacteremia caused by CDC group IV c-2 in a patient with acute leukemia.

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Bacteremia caused by CDC group IV c-2 in a patient with acute leukemia.

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