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LE cells in intermittent hydrarthrosis.

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LE cells in intermittent hydrarthrosis
To the Editor:
In vivo synovial fluid LE cells have been demonstrated in systemic lupus erythematosus (SLE) and
rheumatoid arthritis (1,2). We report here a case of a
young woman with clinical and laboratory findings consistent with the diagnosis of intermittent hydrarthrosis
(IHA) in whom synovial fluid LE cells and positive tests
for synovial fluid antinuclear antibody were demonstrated in the absence of any further evidence for the diagnosis of SLE.
A 23-year-old woman first experienced painless
swelling of the right knee at age 11 coincident with
menarche. This episode heralded a predictable cycle of
recurrent attacks, occurring 6 to 7 times per year and
more prominent in winter months. The knee would
swell rapidly but without pain, local heat, erythema, or
morning stiffness and it resolved spontaneously within 3
to 4 days. There was no response to salicylates, nonsteroidal antiinflammatory agents, or repeated intraarticular corticosteroid injections. No other joints were
affected and there were no other symptoms suggestive
of an underlying connective tissue disorder. Repeated
serologic profiles, synovianalyses, and roentgenograms
failed to reveal any abnormalities.
At age 22, the patient began to experience identical complaints in the left knee as well. One year of persistent symptoms led to hospital admission for arthroscopy. Physical examination revealed only mild
bogginess and positive fluid waves in both knees. Range
of motion was preserved; there was no creptitus or specific signs of patellofemoral joint disease. Laboratory
data included a normal complete blood count, serum
uric acid, sedimentation rate, urinalysis, serum complements, and a negative VDRL. Tests for serum antinuclear antibody, rheumatoid factor, antibody to native
DNA, and extractable nuclear antigen all gave negative
results. Synovial fluid was clear and viscous with normal total protein and complements. The cell count was
2,200 with 85% mononuclear cells. Wright’s stain of synovial fluid revealed LE cells as well as numerous tart
cells in both knees. Followup testing revealed a positive
ANA in the right knee at a titer of 1 :40 and in the left
knee a titer of 1 : 100, both of homogeneous pattern.
AntiDNA testing of synovial fluid by Crithidia luciliae
immunofluorescence was negative in both knees. Agarose gel electrophoresis of synovial fluid revealed a normal pattern. Bilateral arthroscopic examination demon-
strai d i durated and thicken d synovium with
multiple villous projections. The patellae manifested
minimal fibrillation but the menisci were unremarkable
and no pannus was noted. Biopsies were not taken and
the patient was discharged without a definite diagnosis.
Antibodies to deoxyribonucleoprotein are responsible for the LE cell phenomenon (3). Although of
historical importance in diagnosing SLE (4) and included as a preliminary criterion for classification of
SLE ( 5 ) , LE cells have been demonstrated in a variety
of connective tissue disorders, immunopathic diseases,
chronic infections, and in the aged (6). These conditions
share the features of chronicity and inflammation. A recent textbook of rheumatology states that advances in
immunologic laboratory testing have made the LE cell test
obsolete and rarely helpful in differential diagnosis (7).
Intermittent hydrarthrosis describes a disease of
unknown etiology typified by recurrent joint effusions,
usually involving a single knee, and occurring with distinctive periodicity (8). Cardinal signs of inflammation
are minimal or absent and laboratory workup is usually
nonrevealing. The natural history of IHA remains obscure; it has a tendency toward spontaneous remission.
In some patients, however, accelerated degenerative arthritis or evolution to classic rheumatoid arthritis is the
rule. The case reported here has certain laboratory features suggestive of SLE although such a diagnosis is
clearly not warranted at this time. The synovial fluid
abnormalities may reflect only epiphenomena of
chronic inflammation. The rarity of IHA precludes accumulation of large numbers of patients. We would be
curious if other investigators could confirm our findings
in this single case.
University of Pittsburgh
School of Medicine
Department of Medicine
Pittsburgh, PA I5261
1. Hollander JL, Reginato A, Torralba TP: Examination of
synovial fluid as a diagnostic aid in arthritis. Med Clin N
Am 50:1281-1293, 1966
2. Hunder GG, Pierre RV: In vivo LE cell formation in synovial fluid. Arthritis Rheum 13:448-451, 1970
3. Tan EM: Systemic lupus erythematosus-immunologic aspects, Arthritis and Allied Conditions. Ninth edition. Edited by DJ McCarty. Philadelphia, Lea & Febiger, 1979, pp
7 15-722
4. Hargraves MM: The LE cell phenomenon. Adv Intern
Med 6:133-160, 1954
5. Cohen AS, Reynolds WE, Franklin EC, Kulka JP, Ropes
MW, Shulman LE, Wallace SL: Preliminary criteria for
the classification of systemic lupus erythematosus. Bull
Rheum Dis 2 1:643-648, 1971
6. Friou GJ: The L.E. cell phenomenon and antinuclear
antibodies, Arthritis and Allied Conditions. Eighth edition.
Edited by JL Hollander, DJ McCarty. Philadelphia, Lea &
Febiger, 1972 pp 172-194
7. McCarty DJ: Synovial fluid, Arthritis and Allied Conditions. Ninth edition. Edited by DJ McCarty. Philadelphia,
Lea & Febiger, 1979, pp 51-69
8. Ehrlich GE: Intermittent and periodic arthritic syndromes,
Arthritis and Allied Conditions. ~ i n edition.
~ h
Edited by
DJ McCarty. Philadelphia, Lea & Febiger, 1979, pp 663680
Retrocardiac mass: A clue to the tuberculous
nature of monarthritis
To the Editor:
The diagnosis of monarticular diseases often
defies the clinician. Clues to their diagnoses, when present, are welcomed with relief.
An asymptomatic retrocardiac mass found in the
routine chest x-ray of a patient with unilateral knee
swelling gave a clue to its tuberculous nature and evidenced a second typical manifestation of musculoskeletal tuberculosis: Pott’s disease (1).
Case report. This 20-year-old woman consulted
our service because of pain and swelling of the left knee
that had lasted 7 months. During this time she had lost
10 kg and had become increasingly weaker. She had occasionally noticed low back pain which increased when
she bent over. Physical examination revealed only the
swelling of the left knee with both synovitis and effusion. This was aspirated. The fluid obtained was pale
yellow, clear, with few leukocytes. A Singer-Plotz test
for rheumatoid factor in the synovial fluid was positive
at a titer of 1 :320; a mucin-clot was grade 11. Cultures
were negative.
Laboratory tests were essentially normal, including serum rheumatoid factor, antinuclear antibodies,
and LE clot test. Tuberculin and serologic tests for tuberculosis were positive.
The roentgenogram of the chest revealed a lobulated mass in the lower mediastinum posterior to the
heart, which seemed to extend below the diaphragm
(Figure 1).
Roentgenograms of the spine as well as tomograms (Figure 2) revealed loss of height of the tenth
thoracic vertebra with erosion of the lower and upper
epiphyseal plateaus of the tenth and eleventh thoracic
vertebrae respectively. The mass was shown to extend
from the ninth through the twelfth thoracic vertebrae
with a lobulated appearance which indented at the level
of the tenth vertebral space. This image was characteristic of Pott’s disease.
Svnovectomv of the left knee showed chronic
granulomatous inflammation with acid fast bacilli.
Treatment with rifampicin, ethambutol, and isoniazid
resulted in gradual diminution and disappearance of the
mediastinal mass. Other clues to the tuberculous nature
Of this abscess, despite its low thoracic level, Were the
lobulated appearance, its extension below the diaphragm, and the coincident indolent monarthritis. The
absence of pulmonary tuberculosis did not rule out this
diagnosis since approximately 50% of patients with skeletal involvement have no detectable pulmonary disease
Skeletal disease is probably the most common
form of extrapulmonary tuberculosis (1, 2). When it af-
Figure 1. Chest roentgenogram of patient with tuberculous
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intermittent, hydrarthrosis, cells
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