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Journal of Clinical Microbiology, December 2001, p. 4568-4570, Vol. 39, No. 12
Department of Medicine, Stadtspital Triemli,
CH-8063 Zürich,1 Swiss National
Center for Legionella, Istituto Cantonale Batteriosierologico, CH-6904
Lugano,2 and Institute for Medical
Microbiology, University of Zürich, CH-8028
Zürich,3 Switzerland
Received 13 June 2001/Returned for modification 18 July
2001/Accepted 5 September 2001
Recurrent soft tissue abscesses of the jaw, wrist, and arm
developed in a 73-year-old housewife with nephrotic syndrome and immunoglobulin A( Infections due to
Legionella species have been described in numerous outbreaks
and case reports ever since the first isolation of the organism
following an outbreak at a convention of veterans in Philadelphia, Pa.,
in 1976 (9). Various Legionella species cause
infections in persons exposed to common water-related sources and have
been implicated in community and nosocomial outbreaks (5, 6, 8,
9, 12, 17-19). Patients with Legionella infections,
especially immunocompromised patients, have a poor prognosis; however,
Legionella infections only rarely affect organs outside of
the lung. Legionella cincinnatiensis, first isolated from a
dialysis patient with pneumonia in 1988 (25), has, until now, been implicated in clinical infections in only four patients, including three immunocompromised patients with pneumonia
(14) and one patient with fatal encephalomyelitis
following Pontiac fever (23). We report here on the first
case of recurrent soft tissue abscesses caused by L. cincinnatiensis, occurring in a patient with only presumed mild
immunosuppression due to nephritis and immunoglobulin A (IgA) gammopathy.
Case report.
The patient was a 73-year-old housewife with no
relevant medical history aside from treated arterial hypertension. In
April 1999, a swelling on her left jaw disappeared spontaneously within 2 weeks. In June 1999, a large right cervical abscess was incised and
drained. Gram-negative rods were seen on microscopy, but conventional aerobic and anaerobic cultures remained negative. Histology of the
abscess wall showed nonspecific necrotizing granulocytic inflammation by hematoxylin-eosin staining. The patient was treated with oral amoxicillin-clavulanic acid at 1 g twice a day for 10 days. The wound healed over 3 weeks. Hematuria, proteinuria of 2,400 mg/24 h, and
mild anemia (hemoglobin concentration, 10 g/dl) were noted at the time,
with a normal renal function (serum creatinine level, 95 µmol/liter).
Further evaluation revealed a gammopathy of undetermined etiology with
an IgA( Microbiology.
The specimens were centrifuged at 1,711 × g for 15 min. Conventional cultures were set up on 5% sheep
blood Columbia agar and on chocolate agar; and the cultures were
incubated in 5% CO2 at 35°C for 48 h, in
thioglycolate broth at 30°C for 5 days, as well as on Schaedler's
and Columbia-colistin nalidixic acid agar anaerobically at 35°C for
48 h. Following the positive result by broad-spectrum PCR, culture
on special Legionella medium (buffered charcoal yeast agar
[CYA; Difco] prepared in-house) was included. After 48 h,
grayish round colonies with an irregular internal structure were seen
on CYA. Gram staining showed thin gram-negative rods. For
identification, subcultures were grown on CYA for 48 h at 37°C,
and the cellular fatty acids were analyzed with the MIDI system
(Microbial ID, Inc., Newark, Del.) as described earlier (7).
0095-1137/01/$04.00+0 DOI: 10.1128/JCM.39.12.4568-4570.2001
Copyright © 2001, American Society for Microbiology. All rights reserved.
Recurrent Soft Tissue Abscesses Caused by
Legionella cincinnatiensis
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ABSTRACT
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Abstract
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References
) gammopathy of unknown etiology. Conventional cultures remained negative, despite visible gram-negative rods on
microscopy. Broad-spectrum PCR revealed Legionella
cincinnatiensis, which was confirmed by isolation of the
organism on special Legionella medium. Infections due to
Legionella species outside the lungs are rare. L.
cincinnatiensis has been implicated in only four cases of
clinical infection; these involved the lungs in three patients and the
central nervous system in one patient. We conclude that broad-spectrum
PCR can be a valuable tool for the evaluation of culture-negative
infections with a high probability of bacterial origin and that
Legionella might be an underdiagnosed cause of pyogenic
soft tissue infection.
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TEXT
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Abstract
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References
) paraprotein. One month later, her family physician excised
an abscess from her left wrist; no samples for culture were taken. In
November 1999, an abscess measuring 8 by 12 cm developed on her left
arm, with an axillary lymph node measuring 5 by 5 cm. Puncture of the
abscess yielded purulent fluid, with numerous gram-negative rods seen
on microscopy. Again, conventional cultures and a search for
mycobacteria by acid-fast staining and culture on solid and liquid
media (BACTEC 460; Becton Dickinson) showed no growth. The fluid was
examined by broad-spectrum PCR, and a positive result for bacterial DNA
was obtained. Sequence analysis revealed L. cincinnatiensis.
A large subfascial abscess extending to the humerus was surgically
debrided; the axillary lymph node was not excised. Following retrieval
of positive results by broad-spectrum PCR with the clinical sample from
the initial puncture, cultures of the abscess were set up for
conventional microorganisms as well as for
Legionella. An organism later identified as L. cincinnatiensis was isolated. The patient was treated orally with
clarithromycin (500 mg twice per day) combined with rifampin (600 mg
per day) for 6 weeks. Two weeks after the cessation of therapy, a
recurrent abscess at the same site had to be drained, and L. cincinnatiensis was again recovered from cultures of the abscess.
The same antibiotic treatment was given for 8 weeks, with complete
healing of the wound, disappearance of the axillary lymph node, and no
recurrence of abscesses at a follow-up visit 13 months later.
The patient did not report any travel outside Switzerland and denied
unusual exposures through hobbies or daily activities.
6c, 5.9%;
C15:0, 2.8%; Ci16:0, 22%;
C16:1
7c, 21.5%; C16:0,
6.5%; Cai17:0, 3.4%; and
Ccyc17:0, 5.5%. This fatty acid composition was
very similar to that described previously for L. cincinnatiensis (7), with differences only in
C15:1
6c (2.5% ± 0.2%) and
Ccyc17:0 (11.0% ± 2.5%). L. cincinnatiensis is not included in the MIDI system database.
Discussion. We describe a patient with recurrent soft tissue abscesses in whom, after numerous negative conventional cultures, the search for bacterial DNA by broad-spectrum PCR finally led to the isolation of L. cincinnatiensis.
Legionella species, well described as agents of waterborne and nosocomial outbreaks of respiratory and systemic infections, especially among immunocompromised patients, have only rarely been implicated in extrapulmonary infections such as wound infections (3, 15, 20), endocarditis (22, 26), myositis (28), pericarditis (21), and cutaneous or perirectal abscesses (1, 2, 16, 27). In our patient, although no other infections have occurred, the presence of a paraprotein and of significant proteinuria might have led to immunosuppression sufficient to make the patient susceptible to infection with this unusal organism. Failure of primary antibiotic therapy, despite in vitro susceptibility, was ascribed to insufficient surgical drainage, since further therapy with the same agents was successful. We have not been able to elucidate for our patient any special source of exposure that could have led to her infection. We conclude that Legionella species may be an underdiagnosed cause of extrapulmonary infections because they remain undetected unless special media are used. As in previous cases in our experience (4, 10, 11, 24), broad-spectrum PCR has been a valuable tool for the direction of investigations.| |
ACKNOWLEDGMENTS |
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We acknowledge the help of Richard Hanselman, who cared for the patient.
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ADDENDUM |
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In June 2001, a new large lymph node was excised from the patient's groin. Histology and further workup showed a disseminated diffuse large B-cell lymphoma. Following the second course of chemotherapy, the patient developed meningitis due to Cryptococcus neoformans. No recurrence of infection with Legionella was observed. The development of a high-grade malignant lymphoma might be due to transformation of a previously undiagnosed low-grade lymphoma. This would explain the gammopathy and therefore reflect some immunosuppression.
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FOOTNOTES |
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* Corresponding author. Mailing address: Department of Medicine, Stadtspital Triemli, CH-8063 Zürich, Switzerland. Phone: 41-1-466 11 11. Fax: 41-1-466 26 02. E-mail: jacques.gubler{at}triemli.stzh.ch.
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