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Legionnaires' disease in a patient with systemic lupus erythematosus.

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975
CASE REPORT
LEGIONNAIRES’ DISEASE IN A
PATIENT WITH SYSTEMIC LUPUS
ERYTHEMATOSUS
RALPH F. JACOX and I . DONALD STUARD
A young woman with systemic lupus erythematosus
developed a rapidly fatal pneumonia from which no visible
or culturable organisms were found. Subsequent stains
disclosed typical findings of Legionnaires’ disease. A cutaneous portal of entry was suspected and a fulminant lung
abscess developed, neither of which has been previously
reported in Legionnaires’ disease.
Pneumonias of unknown etiology represent a
perplexing and frustrating clinical problem. With the
application of silver impregnation stains ( I ) which are
normally used to identify spirochetes in tissues, a search
can be made for the bacillus of Legionnaires’ disease in
such cases of obscure pneumonia. Legionnaires’ pneumonia occurs in epidemic form (2) and in the immune
suppressed host (3).
This report describes a patient with systemic
lupus erythematosus who developed a severe uncontrolled pneumonia and died, illustrating a need to be
aware of the sporadic occurrence of Legionnaires’ disease i n patients who are on a chronic regimen of antimetabolic or immunosuppressive therapy.
From the Departments of Medicine and Pathology, University of Rochester School of Medicine and Dentistry, Rochester, New
York.
Ralph F. Jacox, M.D.: Department of Medicine; I. Donald
Stuard, M.D.: Department of Pathology.
Address reprint requests to Ralph F. Jacox, M.D., University
of Rochester Medical Center, 601 Elmwood Avenue, Rochester, New
York 14642.
Submitted for publication May 31, 1978; accepted July 6,
1978.
Arthritis and Rheumatism, Vol. 21, No. 8 (November-December 1978)
CASE REPORT
J.A.V., a 20-year-old female, developed pleurisy and
polyarthritis in July of 1968. All symptoms abated until March
of 1969 when a recurrent polyarthritis, fever, and pericarditis
associated with positive LE and ANA tests made a diagnosis
of systemic lupus erythematosus possible. Initial control of the
disease was effected with 50 mg of prednisone, which was later
reduced to 25 mg daily. In August of 1970 the patient again
developed recurring fever, facial rash, polyarthritis, and a right
pleural effusion. Azathioprine, 150 mg, was added to a daily
regimen of 25 mg of prednisone. Mild leukopenia occurred in
December, and the azathioprine was reduced to 100 mg daily.
During the next 27 months she was maintained on this therapy
until a mild leukopenia of 2,000 white blood cells with 50%
granulocytes occurred, and azathioprine was discontinued.
On July 25, 1973, the patient experienced acute swelling and pain of the right elbow that had been preceded by the
appearance of small vesicles on the skin of the inner aspect of
the elbow. Examination at this time disclosed a dusky erythema and edema of the lateral right elbow area. The elbow
joint was normal and there was no associated lymphadenitis or
axillary adenopathy. The leukocyte count was 3,100 with a
normal differential and hematocrit. Oral penicillin V was administered; however, fever and the skin lesion persisted, and 4
days later she was admitted to the hospital.
On admission she was febrile, toxic, and apprehensive.
Several pustules were present over the lateral right elbow;
culture revealed a few coagulase negative staphylococci. A
chest x-ray disclosed an infiltrative lesion of the right upper
lobe, although the patient had no respiratory symptoms and
physical examination had not disclosed signs of a pulmonic
infiltration. Twelve grams of Keflin were administered on the
day of admission. The following day, chest radiographs revealed an extensive consolidation in the left upper lobe and
involvement of the posterior basilar segment of the left lower
lobe. The patient produced bloody, tenacious sputum which,
976
JACOX AND STUARD
on culture, revealed no pathogens. The leukocyte count was
8,200 with 92% granulocytes and blood cultures were negative.
Bronchial lavage of the left upper lobe disclosed a few granulocytes; no bacteria were found on smear and culture. Gentamicin, clindamycin, and nafcillin were administered in an
attempt to control the fulminant pneumonia. Three days after
admission, a patchy infiltrate developed in the middle and
right lower lobes of the lung. Because the patient was in severe
respiratory distress, extracorporeal membrane oxygenation
procedure was begun. I t became difficult to maintain an adequate blood pressure. Acidosis occurred and was treated by
dialysis, but the patient developed an uncontrollable auricular
fibrillation and died on August 9, 1973.
Postmortem examination disclosed extensive bilateral
bronchopneumonia with acute inflammatory exudate filling
alveolar lumina (Figure 1). The left upper lobe had a large
abscess filled with necrotic debris. Cultures of the abscess and
consolidated lung tissue produced no bacterial growth by
aerobic or anaerobic techniques. Lung tissues examined for
organisms with Braun Bren, Giemsa, and Gomori methenamine silver stains revealed no bacteria or fungi. Figure 2 shows
the appearance of the silver impregnation stain which reveals
short rods in the exudate compatible with Legionnaires’ disease.
Figure 1. Pneumonia in the acute exudative phase ( X 350).
DISCUSSION
Patients treated with chemotherapy for malignancy or by immune suppression are vulnerable to unusual types of infectious agents. T h e Legionnaires’ bacillus is the most recent addition to the list of unusual
causes of pneumonia. This patient with systemic lupus
erythematosus illustrates a typical, rapidly developing
type of pneumonia which progressed to lobar consolidation. T h e inflammatory reaction in Legionnaires’
pneumonia is nonspecific and identical to the more common types of bacterial pneumonia.
T h e Warthin-Starry a n d the Dieterle silver impregnation stains are nonspecific. Silver is deposited on
the bacterial membrane surface of all types of bacteria.
Consequently, the identification of short, plump bacilli
must be supported by negative culture data and the nonstainability of the organisms for the Braun Bren and
Giemsa stains.
LEGIONNAIRES’ DISEASE IN SLE
977
Figure 2. Warthin-Starry silver stain of pneumonia with short plump rods outlined both within cells and free in the alveolar space
1500).
(X
This patient illustrates that pneumonia due to
Legionnaires’ bacillus may be necrotizing with abscess
formation, a pathological effect not heretofore described. The possibility must also be raised that the skin
lesion described in this patient might have been a portal
of entry for the bacillus, even though no significant
pathogen was isolated from the pustules overlying the
elbow joint. There have been no reports of cutaneous
lesions in Legionnaires’ disease and all epidemics, so far
reported, probably have a pulmonary portal of entry.
The epidemiology of sporadic cases is unknown. It is
evident that the bacillus of Legionnaires’ disease, which
is probably of low virulence, will be found in increasing
numbers i n the immunologically suppressed patient.
Thus, a recognition of this possibility will provide a
better rationale of therapy with the chance that erythromycin therapy would be much more effective than any
of the antibiotics administered to this patient.
REFERENCES
I . Chandler FW, Hicklin MD, Blackman JA: Demonstration
of the agent of Legionnaires’ disease in tissue. N Engl J
Med 297:1218-1220, 1977
2. Frazer DW, Tsai T R , Orenstein W, et al: Legionnaires‘
disease, description of an epidemic. N Engl J Med
297:1189-1197, 1977
3. Bock BU, Kirby BD, Edelstein PH, et al: Legionnaires‘
disease in renal transplant recipients. Lancet 1:410-413,
1978
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