We describe a case of the isolation of Bartonella quintana from the parotid gland of an apparently healthy man. Pathological examination showed intraparotid granulomatous abscessual lymphadenitis. Diagnosis was made on the basis of high titers of immunoglobulin G (IgG) and IgM antibodies and of culture isolation of a causative agent from parotid aspirate

INCANDELA, Cinzia

INCANDELA, Salvatore

MANSUETO, Pasquale

Micalizzi, A

Vitale, G

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JOURNAL OF CLINICAL MICROBIOLOGY, Mar. 2009, p. 862–864 Vol. 47, No. 30095-1137/09/$08.000 doi:10.1128/JCM.01012-08Copyright © 2009, American Society for Microbiology. All Rights Reserved.Isolation and Characterization of Bartonella quintana from the ParotidGland of an Immunocompetent ManGiustina Vitale,1 Salvatore Incandela,2 Cinzia Incandela,2 Anna Micalizzi,1 and Pasquale Mansueto1*Dipartimento di Medicina Clinica e delle Patologie Emergenti, University of Palermo, Palermo, Italy,1 and Dipartimento diScienze Otorinolaringoiatriche, University of Palermo, Palermo, Italy2Received 28 May 2008/Returned for modification 1 October 2008/Accepted 22 December 2008We describe a case of the isolation of Bartonella quintana from the parotid gland of an apparently healthyman. Pathological examination showed intraparotid granulomatous abscessual lymphadenitis. Diagnosis wasmade on the basis of high titers of immunoglobulin G (IgG) and IgM antibodies and of culture isolation of acausative agent from parotid aspirate.CASE REPORTAn apparently healthy 58-year-old man presented in April2001 to the emergency room of our Dipartimento di ScienzeOtorinolaringoiatriche of the University of Palermo with aswollen, indolent mass in the right retromandibular region thathad appeared 3 days earlier. He did not report other relevantclinical data and had no cats or other pets at home.His general condition was satisfactory, and the patient wasapyrexial. Clinical investigation revealed a palpable, not pul-sating, indolent mass (approximately 3 by 2 cm) behind themandibular angle in the region of the right parotid, not adher-ing with the overlying erythematous skin. The cervical lymphnodes were enlarged only on the right side. The physical ex-aminations of the other systems were essentially normal. Inaddition, 2 days after admission, the patient reported the ap-pearance of a little (0.4- by 0.4-cm), painful phlogistic ulcer-ation on the skin of the right cheek.For differential diagnosis, we considered infectious causes,collagen vascular diseases, sarcoidosis, neoplasms, sialolithia-sis, alcoholism, and congenital masses. Particularly, we consid-ered viral (i.e., cytomegalovirus, Epstein-Barr virus, and hu-man immunodeficiency virus infection), bacterial (Brucella),tuberculosis, and protozoan (Leishmania infantum and Toxo-plasma gondii) etiology, using both in vivo (i.e., Mantoux test)and in vitro (i.e., serologic assays) tests and direct examinationof parotid mass aspirate (see below).Table 1 shows the results of the routine hematochemistryand serologic assays performed during hospitalization. Ultra-sonography and computed tomography of the neck showed amass (3.5 by 3 cm), attributable to the right parotid gland, withswelling of the intraparotid lymph nodes. Chest X ray, trans-thoracic echocardiography, and ultrasonography of the abdo-men were normal.Aspirate from the right parotid mass showed an inflamma-tory infiltration of lymphocytes in different phases of follicularnodulation together with polymorphonuclear leukocyte andepithelioid histiocyte populations—pathological findings thatare compatible with the diagnosis of intraparotid granuloma-tous abscessual lymphadenitis. The approximate relative per-centages of cells in the parotid aspirate were as follows: lym-phocytes, 30%; polymorphonuclear leukocytes, 30%; andepithelioid histiocytes, 40%.The serum sample was examined by the indirect fluores-cent antibody test for the presence of immunoglobulin G(IgG) and IgM class antibodies to Bartonella quintana andBartonella henselae. For antigens, we used either our pre-pared in-house slides or commercial slides (Focus Technol-ogies, Cypress, California, distributed in Italy by Alifax). Weprepared the in-house slides by using B. henselae strainHouston-1 and B. quintana strain Oklahoma as antigens,obtained from the collection of the National ReferenceCenter of Rickettsiosis, Marseille, France, and grown inVero cells for 2 weeks. An IgG titer of 1:64 and IgM titerof 1:20 were used as cutoffs (18).The final diagnosis of B. quintana infection was first sug-gested by the finding of high titers of IgG and IgM antibod-ies for this agent, 1:256 and 1:40, respectively (serology forB. henselae was negative), and then was confirmed by theisolation and characterization of B. quintana from the pa-rotid aspirate.In particular, the isolation of B. quintana was performedon Columbia 5% sheep blood agar plates (BioMerieux,Marcy-l’Etoile, France). Inoculated plates were incubated at37°C under 5% CO2 for 30 to 40 days and controlled weeklyfor evidence of bacterial growth. Four weeks after some Bartonel-la-like colonies became visible, and 2 weeks after that, theywere inoculated in a Vero cell monolayer and incubated at37°C under 5% CO2 for 2 weeks. Then, infected Vero cellswere examined by the indirect fluorescent antibody test, usinghuman B. henselae and B. quintana hyperimmune serum fromour collection, and B. quintana, the only demonstrated micro-organism, was characterized by using PCR as previously re-ported (17). The amplicon size was 619 bp, and the sequencewas 100% identical to that of the B. quintana intergenic tran-scribed sequence (GenBank accession no. 897700).Meanwhile, antibiotics (clarithromycin, 500 mg orallytwice a day [b.i.d.], and ceftazidime, 1 g intramuscularlyb.i.d.), together with nonsteroidal anti-inflammatory drugs* Corresponding author. Mailing address: Dipartimento di Medi-cina Clinica e delle Patologie Emergenti, University of Palermo, Viadel Vespro, 141-90127 Palermo, Italy. Phone: 39 (091) 6552970. Fax:39 (091) 6555995. E-mail: pamansu@unipa.it. Published ahead of print on 7 January 2009.862(nimesulide, 100 mg orally three times a day) and steroids(deflazacort, 6 mg orally b.i.d., and betamethasone, 4 mgintramuscularly once daily), were administered for 14 days.The mass completely disappeared within 3 months, and the6-, 12-, and 24-month and 3-year follow-up visits were com-pletely normal. Serology for B. quintana, performed at the6-month follow-up visit, demonstrated an IgG titer of 1:256and an IgM titer of 1:20. At 24 months, serology for IgM wasnegative, whereas serology for IgG was at the cutoff titer(1:64). After 3 years, serology was repeated, and the resultswere negative for both IgM and IgG.Since its first descriptions during World War I, trench feverhas appeared on every continent except Australia and Antarc-tica. In Europe and North America, infection with B. quintanais nowadays associated with poverty, alcoholism, and home-lessness. Body lice seem to be the principal vector of B. quin-tana infection, although other ectoparasites, such as mites orcat fleas, might conceivably play a role in disease transmission(3, 8, 10, 13, 16). Unlike for most other Bartonella species,there is currently no clear evidence to support the existence ofan animal reservoir for B. quintana (4, 11, 15).Descriptions of the clinical manifestations of B. quintanainfection in the immunocompetent host vary tremendously.Features of “classic” trench fever include fever, sometimesoccurring at 5-day intervals with asymptomatic intervening pe-riods (so-called quintan fever); debilitating and persistenttyphoidal illness, often lasting many months; transient maculo-papular rash; conjunctivitis; severe headache; myalgias; and, inchronic cases, splenomegaly. Progression to death is extremelyrare (12).B. quintana, together with B. henselae, is also associated withbacillary angiomatosis, a bacterial infection characterized bythe proliferation of blood vessels, resulting in the appearanceof tumorlike masses in the skin and other organs (liver, spleen,bone marrow). It most commonly manifests in people affectedby acquired immune deficiency syndrome, rarely appearing inimmunocompetent individuals. While curable, it is potentiallyfatal if not treated (5, 9, 19).The recent descriptions of B. quintana bacteremia in urbanhomeless persons also reveal heterogeneous patterns of illness,especially chronic bacteremia and endocarditis, sometimeswith severe and progressive valvular damage requiring valvereplacement for the cure. These findings suggest that, althoughB. quintana clearly may cause acute and severe illness in home-less persons, it often produces a chronic and nonspecific illnessthat does not arouse clinical suspicion for Bartonella infection(2, 6).Finally, B. quintana has also been reported to cause isolatedchronic lymphadenopathy in both immunodepressed and im-munocompetent individuals, with the pathological features of agranulomatous reaction and without fever and/or other symp-tom/signs (7).Confirming B. quintana infection requires serological assays(including indirect immunofluorescence-based techniques),specific cultures, and nucleic acid amplification procedures (1).For therapy, treatment of uncomplicated B. quintana bacte-remia with a 4- to 6-week course of doxycycline (100 mg orallyb.i.d.), erythromycin (500 mg orally four times a day), orazithromycin (500 mg orally once daily) is recommended. Pa-tients with endocarditis should receive 4 to 6 months of ther-apy, with close monitoring for indications of the need for valvereplacement. Some authorities would recommend the additionof a bactericidal agent, such as a third-generation cephalospo-rin or an aminoglycoside, in the initial 2 to 3 weeks of therapyfor treating endocarditis (7, 14).To our knowledge, this is the first case of B. quintana infec-tion characterized by parotid gland localization, without feveror other symptoms/signs suggesting trench fever or other B.quintana infection features, in an immunocompetent individ-ual. We think that the phlogistic ulceration on the skin of theright cheek is the site of the body louse bite, even if the patientdid not report anything about it. In addition, the pathologicalexamination of the mass showed an intraparotid granuloma-TABLE 1. Laboratory findingsLaboratory testing Result Normal range or resultRed blood cell count 5,440,000 cells/l 4,000,000–6,000,000cells/lHemoglobin 15.2 g/dl 12–17 g/dlHematocrit 46.3% 40.7–50.3%Mean corpuscular volume 83.6 m3 80–99 m3Mean corpuscular hemoglobin 27.4 pg/cell 26–38 pg/cellPlatelet count 319,000 cells/l 150,000–450,000cells/lWhite blood cell count 9,470 cells/l 5,000–9,000 cells/lDifferential leukocyte countNeutrophils 57.4%Lymphocytes 27.6%Monocytes 9.0%Eosinophils 5.5%Prothrombin activity 100% 70–120%International normalized ratio 1.18 0.90–1.20Activated partialthromboplastin time31.0 s 18–35 sSerum fibrinogen 450 mg/dl 200–400 mg/dlGlycemia 98.0 mg/dl 70–105 mg/dlSerum urea nitrogen 23.0 mg/dl 10–50 mg/dlSerum creatinine 1.22 mg/dl 0.60–1.20 mg/dlSerum sodium 140 meq/liter 136–146 meq/literSerum potassium 4.90 meq/liter 3.50–5.10 meq/literTotal serum protein 6.8 g/liter 6.6–8.3 g/literSerum protein fractionsAlbumin 3.09 g/dl 3.48–5.39 g/dl1-Globulin 0.25 g/dl 0.20–0.50 g/dl2-Globulin 0.84 g/dl 0.45–0.97 g/dl1-Globulin 0.63 g/dl 0.32–0.51 g/dl2-Globulin 0.29 g/dl 0.20–0.53 g/dlGamma globulin 1.00 g/dl 0.67–1.56 g/dlAspartate transaminase 22 units/liter 40 units/literAlanine transaminase 12 units/liter 40 units/literPseudocholinesterase 8,890 units/liter 5,400–13,200 units/literTotal bilirubin 0.50 mg/dl 1.0 mg/dlDirect bilirubin 0.15 mg/dl 0.25 mg/dlErythrocyte sedimentation rate 15 mm/h 14 mm/hUrinary analysis Normal NormalCytomegalovirus serology test Negative NegativeEpstein-Barr virus serology test Negative NegativeHepatitis B virus serology test Negative NegativeHepatitis C virus serology test Negative NegativeHuman immunodeficiency virusserology testNegative NegativeBrucella serology test Negative NegativeMantoux test Negative NegativeMycobacterium tuberculosisserology testNegative NegativeLeishmania infantumserology testNegative NegativeDirect examination of parotidmass aspirateNegative NegativeToxoplasma gondii serology test Negative NegativeVOL. 47, 2009 CASE REPORTS 863tous abscessual lymphadenitis more characteristic of B.henselae infection, i.e., cat scratch disease, than of B. quintanainfection. It is thus possible to hypothesize a different specificB. quintana strain infection and/or different genetic character-istics for our patient, which is not easily demonstrated. Withthis subject, diagnosis was made on the basis of physical exam-ination, high titers of the IgG and IgM antibodies against thisagent, and especially, culture isolation from the mass. Therapywas successfully conducted by following the guidelines forcomplicated cases of B. quintana infection, i.e., endocarditis,with antibiotic association (macrolides plus cephalosporins)(7, 14).We thank Didier Raoult of the Faculte´ de Me´decine, Unite´ desRickettsies, WHO Collaborative Center for Rickettsial Reference andResearch, Marseille, France, for his contribution in the characteriza-tion of B. quintana by using PCR.REFERENCES1. Agan, B. K., and M. J. Dolan. 2002. Laboratory diagnosis of Bartonellainfections. Clin. Lab. Med. 22:937–962.2. Breathnach, A. S., J. M. Hoare, and S. J. Eykyn. 1997. 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Isolation and characterization of Bartonella quintana from parotid gland of an immunocompetent man.

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Isolation and characterization of Bartonella quintana from parotid gland of an immunocompetent man.

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