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Letter
Granulomatous Lymphadenitis
as a Manifestation of Q Fever
Pierre Tattevin,* Cédric Arvieux,* Mathieu Dupont,* Pascal
Guggenbuhl,† Alexandre Lemeur,† and Christian Michelet*
* Hôpital Pontchaillou, Rennes, France; and †Hôpital Sud, Rennes, France
Suggested citation for this article: Tattevin
P, Arvieux C, Dupont M, Guggenbuhl P, Lemeur A, Michelet C. Granulomatous
lymphadenitis as a manifestation of Q fever. Emerg Infect Dis [serial
online] 2003 Jan [date cited]. Available from: URL: http://www.cdc.gov/ncidod/EID/vol9no1/02-0211.htm
To the Editor: Q fever is a worldwide zoonosis caused by the obligate
intracellular pathogen Coxiella burnetii (1).
Human infection is usually the result of exposure to infected cattle,
sheep or goats. Acute Q fever may be asymptomatic or manifest as a self-limiting
febrile illness, pneumonia, hepatitis, or meningoencephalitis. Most cases
of acute Q fever will resolve without sequelae, but endocarditis, granulomatous
hepatitis, osteomyelitis, and endovascular infections are well-documented
manifestations of chronic C. burnetii infection (1).
Recently, various atypical manifestations of acute (2),
and chronic (3) Q fever have been reported as well as
changing clinical presentation of Q fever endocarditis (4)
and changing epidemiology of Q fever (5). Researchers
have suggested that heightened awareness of Q fever among doctors, coupled
with improved diagnostic methods, could increase the medical knowledge
about this difficult-to-diagnose and difficult-to-treat infection (4).
We report two cases of granulomatous lymphadenitis associated with C.
burnetii infection.
A 70-year-old man was admitted to the hospital because of weight loss,
night sweats, and a continuous high-grade fever of 2 months’ duration.
His past medical history was unremarkable, except for pulmonary tuberculosis
treated 55 years earlier and chronic glaucoma. He lived in a rural area
and had rare contact with cattle. On admission, his body temperature was
39.5°C; his right laterocervical lymph nodes were enlarged (3 cm x 4 cm)
and inflamed. Blood values were unremarkable except for an elevated C-reactive
protein level of 150 mg/L (normal<6). A computed tomography scan of
the chest showed hilar calcifications and enlarged mediastinal lymph nodes.
A biopsy of cervical lymph nodes indicated granulomatous lymphadenitis
with foci of necrosis. C. burnetii DNA was detected on the lymph
nodes with a C. burnetii–specific pair of primers that amplified
an htpAB-associated repetitive element (6). Results
of serologic testing by indirect immunofluorescence (IF) were positive
for C. burnetii with immunoglobulin (Ig) G antibody titer to phase
1 and phase 2 antigen of 800 and 1,600, respectively, and IgM antibody
titer to phase 2 antigen of 50.
A 44-year-old man was admitted to the hospital because of a continuous
low-grade fever of 3 months’ duration. He had worked as a farmer for 15
years and assisted in the birth of sheep and cattle. On admission, his
body temperature was 38°C, and right inguinal lymph nodes were inflamed,
measuring 4 x 4 cm. A lymph node biopsy showed granulomatous lymphadenitis
with stellate abscesses surrounded by palisading epithelioid cells. Serologic
testing by indirect IF was positive for C. burnetii with an IgG
antibody titer to phase 1 antigen of 320.
For both patients, results of Ziehl staining and Lowenstein (Bio-Rad,
Marne-La-Coquette, France) cultures of gastric aspirates (x 3) and lymph
node specimens were negative for mycobaceria, as were the results of tuberculin
skin tests. Other diseases were ruled out, including brucellosis, yersiniosis,
bartonellosis, and chlamydial infections (by serologic testing) and fungal
infections (parasitologic studies on lymph node tissue). Antinuclear antibodies
were absent, and angiotensin-converting-enzyme values were normal. Both
patients received doxycycline, 200 mg once a day, and rifampin, 600 mg
twice a day, for 1 year, and the symptoms resolved (follow-up at 18 months
for patient 1 and 9 months for patient 2, respectively). For patient 1,
serologic testing after 1 year of treatment showed an IgG antibody titer
to phase 1 antigen of 320.
Granulomatous lymphadenitis has been described during mycobacterial infections,
tularemia, cat scratch disease, yersiniosis, lymphogranuloma venereum,
histoplasmosis, coccidioidomycosis, and chronic granulomatous diseases
(7). One well-documented case of acute Q fever with necrotic
cervical lymphadenitis has been recently reported (8);
to our knowledge, granulomatous lymphadenitis has never been reported
during Q fever. In both cases reported here, C. burnetii was the
likely etiologic agent, given the results of polymerase chain reaction
and serologic studies (patient 1) or the patient’s occupation and results
of the serologic testing (patient 2). Moreover, for both, no other potential
cause could be identified, and the response to doxycycline-rifampin regimen
was favorable. We suggest that granulomatous lymphadenitis be added to
the list of atypical presentations of Q fever.
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