CASE CORRESPONDENCE

Isolated cervical lympadenopathy: unusual presentation of localisedbrucellosis

Introduction

Neck masses are critical diagnostic and thera-peutic challenges in the practise of otolaryn-gology. Although there are numerouscongenital, infectious, benign and malignantconditions that can cause a neck mass, themost common cause is infection.

Brucellosis is a zoonosis transmitted fromanimals to humans. The portal of entry isusually the gastrointestinal tract throughingestion of unpasteurised milk. Brucellosis isa protean disease (1). That is, it can mimicany other disease. This condition leads to adiagnostic difficulty. Isolated neck massbecause of brucellar infection is rare, and thisis the second case reported to date.

Case report

A 63-year-old farmer admitted to theDepartment of Otolaryngology with a swell-ing on the right side of his neck for

6 weeks. His systemic and otolaryngologicexaminations were normal except for a3 · 4-cm painful mass on the right side ofthe neck (Fig. 1).

Computed tomography of the neck showedthe mass in the right submandibular region,which had hypodense centre and thin andcontrast enhanced wall. These radiologic find-ings were suggestive of an abscess or a lymph-adenopathy with central necrosis.

Full blood count and blood chemistry werenormal. Fine needle aspiration biopsy of themass was consistent with an infectious orinflammatory process. Purified protein deriv-ative (PPD) or Mantoux test was negative,and chest X-ray was normal.

According to his history, he was living in arural area, and there were some other peoplein his neighbourhood who had similar com-plaints including neck mass. Although theyhad been operated on for their neck masses,their complaints recurred or persisted afteroperations. Therefore, brucella agglutinationtest was performed, and a very high titre of

1 ⁄ 320 was found. Becasue of that high anti-body titre suggestive for brucellar infection,blood or bone marrow culture was not per-formed. With a presumptive diagnosis of cer-vical brucellar lymphadenopathy, antibrucellartreatment was started with doxicyline2 · 100 mg and rifampicin 1 · 600 mg perday for 6 weeks.

Close follow-up of the patient was per-formed. After initiation of the treatment, thesize of the mass decreased progressively. Theantibody titre decreased to 1 ⁄ 40, and therewas no mass in the neck at the end of 2months follow-up.

Discussion

Brucellar infection is protean in its manifesta-tions. Therefore, it can mimic any other dis-ease. In general, the symptoms are non-specific and include fever, sweats, malaise,anorexia, headache and back pain. Brucellosiscan cause disease in any system or organ inthe body as well (2). These symptoms orother organ or disease manifestations wereabsent in our patient. Thus, the cervicallymphadenopathy was considered to be alocalised brucellar disease rather than a sys-temic disease.

Brucellar infections can mimic malignancy.On admission, the patient was considered tohave a malignant neck mass because of hisadvanced age and size of the mass. However,presence of people with similar complaints inhis neighbourhood and short duration of hissymptoms was suggestive for an infectiousprocess. This presumption was supportedwith the results of fine need aspiration cytol-ogy, which also revealed an infectious process.Considering the increase in the mycobacterialinfections in the worldwide, PPD ⁄ Mantouxand chest X-ray were obtained, which wereunremarkable. However, the Grubbel-Widaltest revealed a high antibody titre suggestingbrucellar infection. Blood and bone marrowcultures were not performed because of theabsence of systemic sings and symptoms ofbrucellosis as well as presence of very highantibody titres. An antigen titre >1 ⁄ 160 is92% sensitive and 100% specific for brucello-sis (3). Despite this fact, coincidence of amalignancy and brucellar disease might bepossible, and close follow-up of the patientwas necessary, as biopsy might have beenneeded. However, the entire neck mass disap-peared in a short time after antibrucellar

Figure 1 Postcontrast computerised tomography reveals an enhancing thick-walled cystic mass (asterisk)consistent with the enlarged and necrotic right anterior jugular lymph node. The skin is thickened (whitearrow), the subcutaneous fat is infiltrated and the platysma muscle is thickened (arrowhead) because ofinflammation. There is also right retropharyngeal lymph node with effacement of the right pyriform sinus(black arrow)

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ª 2009 The AuthorsJournal compilation ª 2009 Blackwell Publishing Ltd Int J Clin Pract, April 2009, 63, 4, 672–675

treatment. This suggested that there was nocoexisting malignancy, and the mass was alocalised form of brucellar infection manifest-ing with cervical lymphadenopathy.

Involvement of neck because of brucellarinfection is uncommon. Three cases of bru-cella infection of the thyroid gland werereported who presented with fever, chills andpainful swelling in the neck (4). Cervicallymphadenopathy because of brucellar infec-tion is also rare, and only one case has beenreported to date who had a chronic drainingbrucellar neck mass in the supraclavicularregion as well as systemic symptoms ofbrucellosis such as fever, erythema and pain(5).

In conclusion, brucellar cervical adenopa-thy should be considered in differential diag-nosis of neck masses in patients who liveor come from regions where brucellosis isendemic.

M. Yilmaz,1 N. Bayazit,2 L. Gurbuzler,1

N. Ercan,3 A. Ceylan,1 Y. Bayazit1

1Department of Otolaryngology,Faculty of Medicine, Gazi University,

Besevler, Ankara, Turkey2Department of Infectious Diseases and

Clinical Microbiology,Siteler Outpatient Clinic Ankara

Numune Hospital, Altindag,Ankara, Turkey

3Department of Radiology,Gazi University, Besevler,

Ankara, Turkey

Correspondence to:Metin Yilmaz

Gazi Universitesi Tip Fakultesi,Kulak Burun Bogaz Hastaliklari AD,

14. kat, Besevler, Ankara, TurkeyFax: + 90 312 2150589

Email: drmyilmaz@gazi.edu.tr

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JE. Dolin R, eds. Principles and Practice of Infectious

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2000: 2386–93.

2 Bayazit YA, Namiduru M, Bayazit N, Ozer E,

Kanlikama M. Hearing status in brucellosis.

Otolaryngol Head Neck Surg 2002; 127: 97–100.

3 Sauret JM, Vilissova N. Human brucellosis. Am

Board Fam Pract 2002; 5: 401–6.

4 Azizi F, Katchoui A. Brucella infection of the thyroid

gland. Thyroid 1996; 6: 461–3.

5 Varona JF, Guerra JM, Guillen V, Guillen S, Menassa

A, Palenque E. Isolated cervical lymphadenopathy as

unique manifestation of brucellosis. Scand J Infect Dis

2002; 34: 538–40.

Paper received March 2005, accepted December 2005

doi: 10.1111/j.1742-1241.2005.00812.x

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ª 2009 The AuthorsJournal compilation ª 2009 Blackwell Publishing Ltd Int J Clin Pract, April 2009, 63, 4, 672–675