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Nodular rheumatoid disease of the lung with cavitation.

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Nodular Rheumatoid Disease of the Lung with Cavitation
By C. D. NOONAN,
FLETCHER
B. TAYLOR,
JR. AND EPHRAIM
P. ENGLEMAN
Nodular rheumatoid disease of the lung
is an established clinical entity, having
been recorded previously in 14 cases.
The 14 cases were reviewed, with emphasis on their differentiation from
nodular lung disease associated with
pneumoconiosis and rheumatoid arthritis (Caplan’s syndrome). This report
adds a fifteenth case to the literature
with the characteristics of the second
recorded instance of roentgenologically
demonstrable cavitation of pulmonary
nodules, an associated high F-I1 titer,
and a silica-free biopsy specimen.
Nodular morbo rheumatoidee del pulmon es un establite entitate clinic in
tanto que ill0 ha previemente essite
reportate in 14 casos. Iste 14 casos es
revistate, con attention particular prestate a lor differentiation ab nodular
morbo pulmonari associate con pneumoconiosis e arthritis rheumatoidee (syndrome de Caplan). Le presente communication reporta un caso additional
que possede le characteristicas-jam
notate in le secunde caso in le litteratura-de roentgenologicamente demonstrabile cavitation del nodulos pulmonari, un associate alte titro de F-11, e un
specimen bioptic libere de silice.
N
UMEROUS AND VARIED pulmonary maniiestations of rheumatoid
arthritis have been described often since Copeman, Ellman, and Kersley’s1 original report in 1947 and Ellman and Ball’s2 in 1948. Only 13 cases,
however, of microscopically proved discrete rheumatoid nodules of the lung
in patients without known occupational exposure to dust have been reported.
This report of a patient with rheumatoid arthritis and cavitating rheumatoid
nodules of the lung adds another case to the literature.
The characteristics in this case were roentgenologic evidence of cavitation
of the pulmonary nodules, an associated high F-I1 titer for the rheumatoid
factor, and biopsy proof that the rheumatoid nodules were free of silica compounds.
CASEREPORT
A 51 year old retired businessnian was admitted to the University of California Hospital
(San Francisco) on September 17, 1961, because of intermittent fever and arthralgia of
1 month‘s duration. He had been well until January 1957 at which time he was examined
at the Mayo Clinic for polyarthritis. At that time synovial reaction involved the second and
third metacarpophalangeal joints of the left hand, the second metacarpophalangeal joints
of the right hand, and the right wrist. Systemic manifestations were absent. Both shoulder
joints were slightly tender with slight limitation of motion. Urinalysis and complete blood
counts were normal and the results of a serologic blood test for syphilis, negative. A serum
uric acid value was 4 mg. per cent and one L.E. preparation failed to show L.E. cells,
The chest roentgenogram showed evidence of left pleural reaction without other abnormalities. Roentgenograms showed that the feet, shoulders, cervical spine, and the right wrist
and hand were normal except for narrowing of the fifth cervical interspace. A regimen of
rest, physical therapy, and salicylates was advised.
By September 1957, aspirin no longer relieved the patient’s symptoms. He was given two
From the Department of Radiology, and the Rheumatic Disease Group of the Department
of Medicine, University of California School of Medicine, San Francisco, Calif.
232
ARTHRITISAND RHEUMATISM,VOL.6, No. 3
(JUNE),
1963
NODULAR RHEUMATOID LUNG DISEASE
233
1-week courses of intensive prednisone therapy ( 4 0 to 60 mg. per day) for exacerbation
of pain, swelling, and limitation of motion of both knees and of the left shoulder. Thz
patient improved and was maintained on prednisone, 10 mg. per day, until December
1960 at which time severe low back pain developed.
During the period from 1957 to 1961, the patient’s hemogram and urinalysis had been
normal on two occasions. A coccidioidin skin test gave a negative result and a roentgenogram of the chest on May 5, 1958 showed that the previously described left pleural reaction
had cleared completely.
At the time of admission to the University of California Hospital, the patient’s complaints
were of 1 month‘s duration. They consisted of myalgia, weakness, afternoon fever up to
102 F., and swelling and tenderness of the right knee and both ankles. There had been no
weight loss. The patient was receiving prednisone, 10 mg. daily, and 9 Gm. of acetylsalicylic acid. No family history of rheumatoid arthritis was obtained. A review of systems
was noncontributory. In particular, the patient had had no symptoms referable to the
pulmonary or gastrointestinal systems.
The patient was of husky build, 5 feet 10 inches in height, and weighed 150 pounds.
His blood pressure measured 120/60 mm. Hg. He suffered moderate distress upon movement of shoulders, elbows, metacarpophalangeal joints, knees, and ankles. No evidence of
subcutaneous nodules or of uveoparotid disease was seen. The thyroid was not enlarged and
lymph nodes were not palpable. The heart and lungs were normal on examination. No
liver or spleen enlargement was noted. Localized bone tenderness or limitation of motion
of the back was not present. Erythema and swelling over the right knee and both ankle
joints, and slight limitation of motion due to pain were present. Joints were not deformed.
Laboratory data included the following: Urinalysis was normal. Packed cell volume
(P.C.V.) was 37 mm. with a sedimentation rate (Wintrobe) of 36 mm. per hour. The
white blood cell count was 11,200 with 86 per cent neutrophils.
On October 3, 1961 the chest roentgenogram showed evidence of multiple, well
circumscribed parenchymal nodules located in the lung periphery and subpleural area
(fig. 1 ) . At least two foci suggested definite central necrosis on the initial chest examination. No abnormal hilar adenopathy or pleural reaction was noted. The initial roentgenographic impression was that of multiple metastatic parenchymal nodules, some of
which were thought to show cavitation.
A skeletal survey on October 4, 1961 showed an over-all demineralization in the
medullary cavities of the long bones (i.e., femora, humeri) with spotty demineralization in
the cervical spine.
Follow-up roentgenograms of the chest on November 20, 1961 and January 15, 1962
showed progression of the lung lesions, indicated by enlargement of the initial solid lesions,
more foci apparent, and the excavating lesions more discernible (i.e., air fluid level) as if
“exploding” (fig. 2 ) .
The patient was followed for 3 months prior to thoracotomy and the radiologist’s impression was, “multiple parenchymal infiltrates, well circumscribed, some of which show
definite cavitation. . . . Rheumatoid nodules should be strongly considered in the differential
diagnosis.”
The initial roentgenographic findings in the chest led to efforts over a 3-month period
to exclude metastatic carcinoma. During this time the patient was admitted on September
17, 1961 and again on January 15, 1962. The studies included examination of aspirated
sternal bone marrow, a biopsy of iliac crest bone marrow, and determinations of serum
calcium, phosphorus, and alkaline acid phosphatase, all of which were normal. The results
of skin tests for coccidioidomycosis, histoplasmosis, and tuberculosis were negative. Examination of three preparations for L.E. cells showed none. The total chemical serum protein
was 5.4 with a reversed A/G ratio ( 2 . U 3 . 3 ) and the cephalin flocculation test gave a 3
plus result after 48 hours. The serum glutamic oxalacetic transaminase (SGOT) determination was 44 units. Electrophoresis revealed a low albumin peak ( 2 8 per cent total area;
normal is 41 to 63 per cent), a high alpha-2 peak (20.5 per cent total area; normal is 7.1
to 16 per cent), and a slightly elevated gamma peak (25.5 per cent total area; normal is
234
NOONAN, TAYLOR AND ENGLEMAN
Fig. 1.-Initial
chest examination. Frontal and lateral views.
Fig. B.--(Left) Frontal view of excavated lung lesion on January 15, 1962.
(Right) Lateral view. Note air fluid level (arrow) and subpleural solid lesion.
11 to 24 per cent). The F-I1 hemoagglutination test showed a positive reaction to ii
dilution of 1:1,764,000(tanned sheep cells with human Fraction 11).
A diagnostic thoracotomy was done on January 23, 1962 and two nodules measuring
approximately 2.5 x 1.5 x 0.5 cm. were removed from the anterior segment of the left upper
lobe. On sectioning, the lumen of each nodule was filled with a yellowish-green thick pasty
material mixed with anthracotic pigment. On microscopic examination, each nodule consisted of a central necrotic area of eosinophilic debris and fibrillar basophilic material,
necrotic cells, and inflammatory cells (predominantly neutrophils) The necrotic area was
rimmed by epithelioid or fibroblastic cells which showed peripheral palisading in (some)
areas. This zone of palisaded cells was in turn rimmed by a zone of connective tissue, the
interstices of which were filled with plasma cells, lymphocytes, and monocytes (fig. 3 ) .
No giant cells were noted. There was no evidence of vasculitis and no organism could be
.
NODULAR RHEUMATOID LUNG DISEASE
235
Fig. 3.-Photomicrographs of a nodule in the lung showing typical nodular granulomatous lesions with central necrosis, palisading, and peripheral cellular infiltration. Hematoxylin and eosin stain. (Left) Original magnification ( 1OX) ; (right)
(40X).
demonstrated using acid-fast or PAS stains. Cultures for acid-fast bacteria and fungi were
negative. The nodule was incinerated and the ash assayed for silica content, but none could
be seen. The final diagnosis was granuloma of the left upper lobe of lung with central
necrosis consistent with rheumatoid arthritis.394
The patient’s postoperative course was uneventful and the patient was discharged 7
days after the operation with instructions to continue 2.5 mg. of prednisone and 0.6 Gm.
of aspirin, four times a day. Follow-up roentgenologic findings on January 19 and February
14, 1962 showed progressive improvement in the left pleural reaction which had been
present at operation. Central cavitation developed in at least one additional focus of solid
infiltrate.
DISCUSSION
The 14 cases of nodular rheumatoid disease of the lung reported previously,5-13J7and the one in the present report are summarized in table 1. The ages
ranged from 30 to 65 years and 10 of the patients were men. Our case adds further weight to the incidence of nodular rheumatoid disease of the lung in
men. The occupational history, important in excluding associated pneumoconiosis (Caplan’s syndrome),le w a s reported in only 7 of the 15 patients.
One of these was a coal miner in whom the nodules were described as not
absolutely typical of the lesions in Caplan’s syndrome.*l Our patient h a d
worked as a clerk in Chicago since 1937, and prior to that time was in the
floral business. There was no history of exposure to irritant dusts.
Subcutaneous noduEes were reported i n four patients, and in three others
236
NOONAN, TAYLOR AND ENGLEMAN
Table 1.-Non-Pneumoconwtic, Necrobiotic Rheumatoid Lung Nodules (Reported)
Clinical Data
No.
Age
Sex Occupation
Year
Author
1954
Bevans e t al.'
Case 2
41
ChristieR
Case 1
59
SeCretaW
Subcut.
nodules
none
F
67
Ellman et al."
65
M
jeweler
(retired )
Mahe+
64
housewife
ext. occipital
protuberance:
left olecranon,
forearms,
ischium.
F
Skogrand*
6
41
43
F
8
62
M
9
57
M
over right
olecranon
1958
Greshamand
Kellaway'O
1959
Flatleyl?
10
62
M
factory
foreman:
no exposure to
coal. dusts
or vapors
Horlerand
Thompson"
Case 8
11
51
coalminer
forearm
Robertson and
Brinkma+
12
bricklayer
both wrists,
fingers, arms,
shoulders,
scapulae.
SaeNrn. ears.
scalp, knees.
ankles. feet
1961
M
54
M
Chest X-ray
Initial
thickened left
pleura
9 mo.
fibrosis LUL
Oct. 1943; gross
cardiac enlargement (? effusion)
19 yr..
1934 to
1953
cortisone,
71 days
cardiac enlargement: left pleural effusion
21 yr.
1932 to
1953
cortisone
nodular lesions
interpreted as
Tbc; globular
heart
7 mo.,
Apr. to
Nov. 1960
ACTH
parenchymal
densities both
bases, Aug. 1950
4 yr..
1950 to
1964
ACTR
Sept. 1963
cortisone
prolonged
therapy
10 yr.,
1938 to
1948
M
7
Steroid
therapy
cortisone:
corticotrophin
16 yr.,
1936 to
1962
34 Yr.,
1913 to
1947
F
1956
Duration
lung nod.
15 yr.
1934 to
1949
M
Case 3
Duration
R.A.
E e p t . 1960
21 yr.
10 yr.,
1948 to
1958
6 wk.
4yr.,
1948 to
1952
1952tm
1966.
unchanged
onset
Feb.
1959
cortisone
LLL infiltrate
with effusion
mass RML: auspect carcinoma
hydrocortisone
chronic lung
process; acute
pleural change;
pulm. nodules
suggest metast.
237
NODULAR RHEUMATOID LUNG DISEASE
Table 1.-(Continued
Laboratory Data
Follow-UP
Proof
F-II*
L.E.
left lower lobe
consolidation,
effusion
Oct. 1946.9 mo.
prior to death,
round dense
shadow 6 em.
RUL
right pleuuraleffusion: calcified
pleural adhesions, left base
1:6120
neg.
1966 nodular
shadows both
lung f i e .
&usion
1:128
neg.
Autopsy
Biopsy
several small circumscribed
yellow nodules; 3 mm. LUL
soft and necrotic
granulomas of the lungs,
not tuberculous
firm white subpleural
nodules 2 to 3 em.; two on
left, three on right
fibrinous pneumonia foci
undergoing central necrhsis. spreading edge
sharp
encapsulated necrotic mass
with fibrotic edges 7 em.
RUL
indistinguishable from a
subcutaneous PeriaJticular nodule of rheumatoid
arthritis
numerous spherical lesions
up to 1 to 2 cm. in periphery, and subpleural soft,
cheese-like centers
occasional rheumatoid
lesion in the lung
parenchymal nodules 1 cm.
in diameter
two large lung nodules
probably rheumatoid, not
to be attributed to
tuberculosis
microscopic lung granuloma
rheumatoid granulomae
in lungs
macroscopic grain-sized
nodules (lung)
rheumatoid granulomas
in lunm
macroscopic pinhead to
pea-sized nodules in placea
to coalesce
rheumatoid granulomas
in lungs; nodules grayishyellow coalesced into
larger yellow infiltrates
LUL nodules
Typical appearance of
rheumatoid disease;
rheumatoid granuloma
1968 thoraeotonw; pleura
thickened gray-white,
8 x 2 x 1 cm. dense white
fibrous firm lesion, surrounding atelectasis and
fibrosis
histologic appearance of
rheumatoid nodules; one
rheumatoid lung nodule,
several subpleural
1962 thoracotomy; inter-
identical with thoue found
in a rheumatoid nodule
lobar effusion; small nodule
right apex RLL
-
Pa.
Pathologic report
nodular areas easily palpated; one excised from
superior segment of lower
lobe
large granuloma; central
zone had undergone a
little cavitation
238
NOONAN, TAYLOR AND ENGLEMAN
Table 1.4Continued)
Clinical Data
Sex Occupation
No. Age
Year
Author
1962
Sieniewicz
et al.”
Case 1
13
Case 2
14
41
M
Subeut.
nodules
none
Duration
R.A.
1 yr.,
1959 to
1960
Duration
lung nod.
Steroid
therapy
1 mo.
several nodular
lesions at both
bases (peripheral), localized
pleural reactive
changeonthe
right
nodular lesions,
both uDper lobes,
with right pleural effusion
1 mo.
30
F
1963
Noonanetal.
15
51
M
husinessman
(retired)
none
5 yr.,
1957 to
1962
Chest X-ray
Initial
Oct. 3,
1961 to
present
( 3 mo.)
cortisone
well circnmscribed lung nodules (peripheral
and subpleural).
some with cavitation
nodules of other visceral organs were described.8*9This distribution reflects
the potential dissemination of nodules in rheumatoid arthritis. In our patient,
however, no evidence of subcutaneous nodules was present. The duration of
this patient’s rheumatoid arthritis was 4 years, whereas that in the reported
patients varied from 1 month to 21 years. Our patient and 6 of those reported
were receiving steroid medication. Although the reports of the other eight
did not mention steroid therapy, additional inquiry verified that it had not
been used in those patients.6.9-11J’In none of the literature is a relationship
suggested between this medication and the formation of pulmonary rheumatoid nodules.
Reports of chest roentgenograms were given in 10 of the 14 previously
reported cases. Five of these described discrete pulmonary nodules, and in
Sieniewicz’s case 1, cavitation was reported. To our knowledge, our patient
was the second whose roentgenograms gave evidence of cavitation. This finding is expected ordinarily in 9 per cent of primary lung tumor cases and in
4 per cent of metastatic lung tumors.14 Thus, seen initially in a patient with
rheumatoid arthritis, the possibility of malignant disease was considered. The
F-11 test* was done in four previous patients with pulmonary nodules, one with
a positive result at a dilution of 1:5120.12In our patient, the F-I1 test gave
positive results at a dilution of 1:1,764,000. A titer this highly concentrated
has been associated more frequently with rheumatoid arthritis in patients
who have subcutaneous nodule formation and associated pulmonary disease
(i.e., fibrosis, Caplan’s syndrome, pleural nodules) .15 The search for L.E.
cells was successful in one of five previously reported patients. In our patient,
‘See footnote to table, facing page.
239
NODULAR RHEUMATOID LUNG DISEASE
Table l..--(Continued)
Laboratory Data
Follow-up
Proof
F-11*
L.E.
Cavitation in one
nodule 1 mo. later
at right base; no
new lesions seen
atter operation
pos.
neg.
thoracotomy 2.6 em. nodule
RML, grayish pleural
exudate over lower lobe
with communication to
another necrotic nodule
behind visceral pleura
post-operative:
no new lesions;
reduction in size
to disappearance
1 year later
pos.
neg.
thoracotomy serous effusion on right, multiple
small nodules with exudate
over lung surface; at
wedge resection, escape
of thick grayish material
of toothpaste viscosity
Progression in
sire and foci
of cavitation
1:1.'764
neg.
thoracotomy nodules. left
upper lobe; lumen on section yellow-green thick
pasty material
x10"
Autopsy
Biopsy
Pathologic report
histoloaic flndinns nonspecific but have been
described as being typical of rheumatoid nodule;%gassociated nonnecrotizing vasculitis
granuloma of the lung
with central necrosis
consistent with rheumatoid arthritis; ash assay:
no silica content
*The reported rheumatoid factor tarts varied as to method. Flatley used the latex agglutination method; Horler
and Thompson used the Rose Waaler test; and Sieniewici used the Hyland Slide latex fixation method. Our method
used tanned sheep cells with human Fraction 11.
no L.E. cells were found in three preparations, Of the cases reported, nine
were proved at necropsy, and six, including the one reported here, were
proved by biopsy.
From the preceding review it will be noted that the presence of nonpneumoconiotic, nodular, necrobiotic rheumatoid lung disease must rest on
a firm clinical diagnosis of rheumatoid arthritis. Evidence must be obtained
by history, microscopic examination, and analysis for silica to exclude associated
pneumoconiosis or chronic pulmonary disease.
In terms of diagnosis, these criteria serve to distinguish nodular rheumatoid
disease of the lung from Caplan's syndrome.le This syndrome is characterized
by rheumatoid nodules of the lung with rheumatoid arthritis and pneumoconiosis. Moreover, the diagnosis must be differentiated from an associated
malignant tumor with cavitating pulmonary lesions, and from necrotizing
arteritis, tuberculosis, fungus, Wegner's granulomatosis and histiocytosis X,
among others
It is apparent that with roentgen evidence of lung nodules with cavitation
in patients with rhmeumatoid arthritis, the presence of rheumatoid nodules must
be given serious consideration.
SUMMARY
A patient with nodular rheumatoid lung disease characterized by cavitation
of the nodules and by an associated high F-I1 titer is described. A lung biopsy
confirmed the diagnosis. The relevant literature is reviewed.
240
NOONAN, TAYLOR AND ENGLEMAN
ACKNOWLEDGMENT
The authors are indebted to Dr. L. Emerson Ward for supplying data from the Mayo
Clinic regarding the patient reported here.
REFERENCES
1. Copeman, W. S. C., Ellman, P., and
Kersley, G. D.: Aetiology of chronic
rheumatism. Proc. Roy. SOC. Med.
40:329, 1947.
2. Ellman, P., and Ball, R. E.: “Rheumatoid disease” with joint and pulmonary manifestations. Brit. M. J. 2:
816, 1948.
3. Collins, D. H.: Subcutaneous nodule
of rheumatoid arthritis. J. Path. &
Bact. 45:97, 1937.
4. Bennett, G. A., Zeller, J. W., and
Bauer, W.: Subcutaneous nodules of
rheumatoid arthritis and rheumatic
fever; pathologic study. Arch. Path.
30:70, 1940.
5. Ellman, P., Cudkowicz, L., and Elwood, J. S.: Widespread serous membrane involvement by rheumatoid
nodules. J. Clin. Path. 7:239, 1954.
6. Christie, G . S.: Pulmonary lesions in
rheumatoid arthritis. Australasian
Ann. Med. 3:49, 1954.
7. Bevans, M., Nadell, J., Demartini, F.
E., and Ragan, C.: Systemic lesions
of malignant rheumatoid arthritis.
Am. ]. Med. 16:197, 1954.
8. Maher, J. A,: Dural nodules in rheumatoid arthritis; report of case. A.
M. A. Arch. Path. 58:354, 1954.
9. Skogrand, A,: Visceral lesions in rheumatoid arthritis. Acta rheumat. scandinav. 2:17, 1956.
10. Gresham, G. A., and Kellaway, T. D.:
Rheumatoid disease in the larynx and
lung. Ann. Rheumat. Dis. 17:286,
1958.
11. Horler, A. R., and Thompson, M.:
Pleural and pulmonary complications
of rheumatoid arthritis. Ann. Int.
Med. 51:1179, 1959.
12. Flatley, F. J.: Rheumatoid pulmonary
disease; report of a case. New England J. Med. 261:1105, 1959.
13. Robertson, J. L., and Brinkman, G.
L.: Nodular rheumatoid lung disease.
Am. J. Med. 31:483, 1961.
14. Dodd, G.D., and Boyle, J. J.: Excavating pulmonary metastases. Am. J.
Roentgenol. 85:277, 1961.
15. Jacobson, A. S., Kammerer, W. H.,
Wolf, J., Epstein, W. V., and Heller,
G.: Hemagglutination test for rheumatoid arthritis; clinical evaluation
of sheep erythrocyte agglutination
(S.E.A.) test and gamma globulin
( F 11) tests. Am. J. Med. 20:490,
1956.
16. Caplan, A.: Certain unusual radiological appearances in chest of coalminers suffering from rheumatoid
arthritis. Thorax 8:29, 1953.
17. Sieniewicz, D. J., Martin, J. R., Moore,
S., and Miller, A.: Rheumatoid
nodules in the lung. J. Canadian
Assoc. Radiol. 13:73, 1962.
D. Noonun, M.D., Assistant Professm of Radwlogy
and Assistant Radiologist, D t ? p a m of R d i o b g y , University of California School of Medicine, San Francisco, Calif.
Churbs
Fletcher B . Taylor, M.D., at present, Assistant Research Physician, Cardimascular Research Institute, University of califmnia School of Medicine, Son Francisco, Calif.
Ephraim P . Engleman, M .D., Associate Clinicd Professor of
Medicine, Rheumatic Disease Group, Department of Medkiw,
University of California School of Medicine, San Francisco,
Calif.
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