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Sarcoidosis accompanied by pulmonary tuberculosis and complicated by sacroiliitis.

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We report the unique occurrence of a unilateral
sacroiliitis in a patient with active sarcoidosis accompanied by pulmonary tuberculosis. Convincing (clinical)
evidence of sarcoidosis as the extremely rare cause of
this articular involvement is presented. Discussion is
focused on comparison of sarcoidosis and tuberculosis,
particularly with respect to their articular involvement,
and the literature of previously reported cases of sarcoid
sacroiliitis is briefly reviewed.
The granulomatous diseases of sarcoidosis and
tuberculosis can both lead to arthritis (1-7). Since both
disorders may occur simultaneously (8), an apparent
articular involvement can cause confusion. In tuberculosis, the vertebral column, including the sacroiliac
(SI) joints, is involved relatively frequently (1,2); such
involvement is very rare in sarcoidosis (3-7).
To our knowledge, this report describes the first
patient with sarcoidosis accompanied by pulmonary
tuberculosis with concomitant evidence of a complicating sarcoid sacroiliitis.
In January 1990, a 34-year-old Moroccan man
presented in our emergency unit with severe asthma
From the Departments of Rheumatology and Pulmonology ,
Slotervaart Hospital, Amsterdam, The Netherlands.
Eduard N. Griep, MD: Department of Rheumatology,
Slotervaart Hospital; Paul I. van Spiegel, MD: Department of
Pulmonology, Slotervaart Hospital; Rende M. van Soesbergen, MD,
PhD: Department of Rheumatology, Slotervaart Hospital.
Address reprint requests to Eduard N. Griep, MD, Department of Rheumatology, Jan van Breemen Institute, Dr Jan van
Breemenstraat 2, 1056 AB Amsterdam, The Netherlands.
Submitted for publication August 18, 1992; accepted in
revised form November 19, 1992.
Arthritis and Rheumatism, Vol. 36, No. 5 (May 1993)
provoked by a recent upper respiratory tract infection.
His family history suggested a familial tendency for
this asthma. His medical history was otherwise unremarkable. Physical examination revealed no further
The chest radiograph revealed a bilateral hilar
and right-sided paratracheal polycyclic lymphadenopathy with a diffuse reticulonodular pattern in both
upper lung fields (Figure 1A). Laboratory investigations showed an erythrocyte sedimentation rate (ESR)
of 16 mm/hour (Westergren), and normal values on
hematologic, serum creatinine, and liver function
tests. The angiotensin-converting enzyme (ACE) level
was 58.7 unitsfliter (normal 16-51). The tuberculin skin
reaction was 0 x 0 mm. Sputum showed no acid-fast
bacilli (AFB) on Ziehl-Neelsen (ZN) stain, and cultures for (myco)bacteria were negative. Pulmonary
function data were normal (Table 1). A fiberoptic
bronchoscopy revealed no visible abnormalities, but
peripheral transbronchial biopsies from the right upper
lobe revealed tissue with noncaseating epithelioid cell
granulomas without AFB ; cultures for (myco)bacteria
were also negative.
A diagnosis of sarcoidosis was made, and the
condition was managed accordingly. His asthma responded well to a short course of oral prednisolone,
followed by a regimen of inhaled bronchodilators and
He presented in March 1991 because of a
3-month history of slowly progressive, exerciseinduced fatigue and dyspnea. Results of the physical
examination were unremarkable. The ESR and ACE
values were unchanged. Radiographically, the upper
lung fields showed alveolar shadowing. Pulmonary
function data had worsened (Table 1). Fiberoptic
Figure 1. Radiographs of the patient’s chest. A, On initial admission (January 1%), films showed bilateral hilar and right-sided paratracheal
polycyclic lymphadenopathy accompanied by a diffuse reticulonodular pattern in both upper lung fields. B, Recent films (January 1992) show
regression of both the lymphadenopathy and the parenchymal abnormalities.
bronchoscopy , with bronchoalveolar lavage (BAL) in
the right upper lobe, was repeated because of a suspected increase in the activity of the sarcoidosis.
Surprisingly, the BAL fluid showed many AFB and
suggested a chronic inflammatory process, and not the
high-intensity alveolitis one would expect in active
pulmonary sarcoidosis. Cultures of the BAL fluid for
(myco)bacteria were negative. Nevertheless, the high
suspicion of pulmonary tuberculosis, accompanying
the sarcoidosis, urged us to initiate a 9-month course
of antituberculous therapy (isoniazid, rifampicin, pyrazinamide). The patient experienced prompt relief of
his symptoms and restoration of his general health. A
chest radiograph obtained 2 months later demonstrated partial resorption of the upper lung-field abnor-
Table 1. Pulmonary function test results at various intervals during the course of the disease(s)*
Jan. Feb. Dec. Mar. Aug. Nov. 1992
VC (liter)
RV (liter)
Kco (%predicted) ND
* VC = vital capacity (predicted value 5.0); RV = residual volume
(predicted value 1.8); Kco = carbon monoxide transfer capacity
(carbon monoxide diffusing capacity/alveolar volume); ND = not
malities. Thus, the diagnosis of an accompanying
pulmonary tuberculosis was supported.
Progressive, left-sided, low back pain developed in August 1991. The left SI joint was painful to
local pressure and with strain. Physical examination
also revealed generalized lymphadenopathy, slight
hepatomegaly, and a subcutaneous nodule near the
right nipple. A left-sided sacroiliitis was suspected
(Figure 2A). The ESR had risen to 31 mmhour, the
ACE level to 102 unitsfliter, and the serum uric acid
was increased at 0.86 mmolesfliter (normal 0.17-0.45).
All other laboratory test values were (still) normal, and
the tuberculin skin reaction was negative, as before.
Pulmonary function data were essentially unchanged
(Table 1). The subcutaneous nodule was biopsied, and
histologic examination showed noncaseating epithelioid cell granulomas, without any signs of AFB.
Detailed examination of the SI joints with conventional and computed-tomography (CT) scans supported the suspected diagnosis of a left-sided sacroiliitis, without extension into the juxtaarticular soft
tissues (Figures 2B and C). All available evidence
suggested a diagnosis of sarcoid sacroiliitis. Biopsy of
the left SI joint confirmed this diagnosis. The tissues
showed evidence of granulomatous disease, without
signs of (myco)bacteria on ZN stain and culture (Figure 3).
Prednisone was begun at a dosage of 40 mg
daily, which resulted in prompt recovery, with com-
Figure 2. Radiograph of the patient’s lumbosacral spine. A, Film taken in August 1991 suggests abnormal sclerosis of the left sacroiliac (SI)
joint. B and C, Plain tomography and computed tomography scans taken a short time later show left-sided sacroiliitis with sclerosis, blurred
margins, and cysts, possibly even erosions (iliac more so than sacral region), without signs of extension into juxtaarticular soft tissues. D,
Radiograph taken in January 1992 shows marked improvement of the left SI joint.
plete relief of pain within days. In fact, this response
ultimately supported our diagnosis of sarcoid sacroiliitis. During the following months, his condition returned to almost normal, clinically, biochemically, as
well as radiographically (Figures 1B and 2D). The
tuberculostatic therapy was recently discontinued. At
present, he remains free of symptoms and signs while
on a regimen of minimum doses of corticosteroids.
Sarcoidosis and tuberculosis are multisystem disorders characterized by the presence of epithelioid cell
granulomas (1-7). In tuberculosis these granulomas are
usually caseating and contain AFB, while in sarcoidosis
these features must be absent to justify the diagnosis
(1,47). Nevertheless, some suggest a common (myco-
Figure 3. Biopsy material from the patient’s left sacroiliac joint
shows a cluster of epithelioid cells, with partial fusion, indicating
granulomatous disease (original magnification x 250).
bacterial) etiology (8), even though corticosteroids,
which are usually beneficial in sarcoidosis, are known t+
escalate activity in mycobacterial-mediated disease.
The pattern of organ involvement in both disorders differs (1-7). Sarcoidosis involves the lungs,
lymph nodes, skin, liver, and eyes, in declining order
of frequency. The reported incidences of bone and
joint involvement vary, ranging from 3% to 14% and
from 5% to 39%, respectively (3,4,6,7). In tuberculosis
the lungs are most commonly affected, but involvement of other organs, notably, the lymph nodes,
kidneys, long bones (about 5%), genital tract, brain,
and meninges, or dissemination may also occur. Skin
and eye involvement is rare. The joints are involved in
about 15% of cases (1,2).
The pulmonary manifestations of tuberculosis
and sarcoidosis may mimic each other and are often
indistinguishable (1). However, there are some features which help in differentiation. A radiographic
pattern of bilateral hilar and right-sided pulmonary
lymphadenopathy, as in our patient, is quite characteristic of sarcoidosis (4,6,7). In tuberculosis, hilar
lymphadenopathy is mostly unilateral, or can even be
radiologically undetectable (1,4). Histologic evidence
of noncaseating granulomas, without signs of (myco)bacteria, or other microorganisms, and a raised ACE
level (4,6,7,9) also favor a diagnosis of sarcoidosis. In
every respect, the diagnosis of sarcoidosis in our
patient was therefore justified. Later, there was evidence of an accompanying pulmonary tuberculosis,
with BAL fluid showing AFB and a non-high-intensity
alveolitis; retrospectively, the alveolar shadowing was
compatible with tuberculosis as well (1). Negative
cultures do not exclude this diagnosis, since (myco)-
bacterial growth can be inhibited by the bacteriostatic
properties of lidocaine when used as an endobronchial
anesthetic (lo), as was the case in our patient.
With regard to articular involvement, tuberculosis usually displays a monarticular, chronic destructive form of septic arthritis (1,2). Any joint may be
affected, but there is a definite predilection for the
spine, including the SI joints, and the larger, weightbearing joints (hips more so than the knees more so
than the ankles). Joint infection is due to hematogenous spread or to direct extension from adjacent sites
of bone involvement. The combination of arthritis and
osteomyelitis often occurs. In peripheral joints, subchondral erosions, followed by necrosis, often precede
cartilaginous destruction. Infection may therefore be
advanced, without significant joint space narrowing,
producing a quite characteristic radiographic picture.
In the spine (Pott’s disease), there is early
involvement of the vertebral margins and adjacent
discs, with disc space narrowing and eventual vertebral collapse, which results in kyphosis (gibbus). Tuberculous sacroiliitis has no specific features, but
extension to the juxtaarticular soft tissues (cold abscess) sometimes suggests the diagnosis on appropriate imaging (CT scan) (1,2). Histologically, the presence of granulomas-caseating or noncaseating-in
synovial tissue, is variable. Ultimately, a diagnosis of
tuberculous arthritis rests upon the demonstration of
tubercle bacilli in synovial fluid or tissue by smear or
by culture.
An arthritis is the most frequent rheumatic
manifestation of sarcoidosis (5-7). Two main clinical
patterns of sarcoid arthritis have been recognized
both of which are often accompanied by elevated ESR and ACE levels, while hypercalcemia and
hyperuricemia are less common. Histologically, noncaseating granulomas may be found in synovial specimens, but nonspecific inflammatory patterns are also
encountered (4-7). The most common form occurs
acutely as a symmetric migratory polyarthritis, usually
with minimal swelling, most frequently involving the
knees, ankles, proximal interphalangealjoints, wrists,
and elbows. This form is often the initial manifestation
of sarcoidosis, sometimes preceding other symptoms
by many years (4-7). Frequently, there is an association with bilateral hilar lymphadenopathy (>90%)
and/or erythema nodosum (Lofgren’s triad) (6). Monarticular acute sarcoid arthritis is unusual. The acute
form probably reflects a circulating immune complex
reaction, is generally self-limiting, and seldom causes
joint deformity or destruction (4,6,7). Chronic sarcoid
arthritis is less common, usually polyarticular, and
produces a picture of recurrent episodes of joint pain
in patients with sarcoidosis of several months or
several years duration (4-7). Target joints include the
shoulders, wrists, and small joints of hands and feet,
but most commonly, the knees and ankles are involved
(5-7). Associated pulmonary and skin abnormalities
are frequent (5,7).Joint deformities and occasionally
even joint destruction may occur; radiographs lack
specific signs (5).
While chronic sarcoid monarthritis is rare, SI
involvement is extremely unusual (5,7). Obviously,
considering the pulmonary tuberculosis in our patient,
an infectious arthritis had to be excluded. Our diagnosis of sarcoid sacroiliitis was based on the presence of
a cluster of epithelioid cells, without tubercle bacilli or
other pathogens, in biopsy material from the SI joint,
given that the histologic appearance and culture findings of synovial tissue biopsies are diagnostic in up to
90% of patients with tuberculous arthritis (1,2). It
should be noted that the sacroiliitis in our patient
developed long after tuberculostatic therapy was
started. Clinically, the prompt (and persistent) recovery with corticosteroid therapy delivered ultimate
“proof’ of the diagnosis.
Thus far, only 4 cases of sarcoid sacroiliitis
have been reported, all in the French literature (1114). Three of them occurred in combination with
(osseous) spinal involvement (1 1,12,14),which is itself
uncommon (5,6,15).The sacroiliitis was bilateral in 1
patient (12). Neither plain radiographs (in all 4 patients) nor additional tomographic evaluation (in l
patient) showed distinct features (13).Biopsy of the SI
joint was performed in 3 cases (11,13,14),and was
conclusive in only 1 (1 1). The other 2 were nonspecific
and the diagnosis was based on sufficient circumstantial evidence (13,14),as was the case with the remaining patient (12).The literature also reports 1 case of a
presumed sarcoid sacroiliitis treated with corticosteroids; unfortunately, this condition turned out to be of
tuberculous origin and deteriorated (16). A review in 1
report (14)mentions 2 other possible cases (17,18),but
on reflection, those 2 patients merely displayed osteosclerotic lesions in the pelvic bones and elsewhere
(17), and not sacroiliitis. From all available data, it
would seem that our patient’s case is the first reported
with sarcoidosis and concomitant pulmonary tuberculosis, accompanied by a sarcoid sacroiliitis.
In conclusion, we suggest that in patients with
simultaneous sarcoidosis and tuberculosis who de-
velop a sacroiliitis (or a monarthritis generally), a
tuberculous origin should not be assumed. The rare
possibility of sarcoidosis as the etiology should be kept
in mind. A probable diagnosis can be made using
noninvasive procedures, but articular biopsy is necessary for confirmation.
We thank Dr. David A. Batchelor (Head, Department of Radiology, Slotervaart Hospital, Amsterdam, The
Netherlands), for his evaluation of the radiographs and his
comments on the manuscript.
1. Stead WW, Bates JH: Mycobacterial diseases: tubercu-
losis, Harrison’s Principles of Internal Medicine. Tenth
edition. Edited by RG Petersdorf, RD Adams, E Braunwald, KJ Isselbacher, JB Martin, JP Wilson. Auckland,
McGraw-Hill, 1983
Hoffman GS, Sentochnik DE: Mycobacterial and fungal
infections, Textbook of Rheumatology. Third edition.
Edited by WN Kelley, ED Harris Jr, S Ruddy, CB
Sledge. Philadelphia, WB Saunders, 1989
Mayock RL, Bertrand P, Morrison CE, Scott JH: Manifestations of sarcoidosis: analysis of 145 patients, with a
review of nine series selected from the literature. Am J
Med 35:67-89, 1963
James DG, Neville E, Carstairs IS: Bone and joint
sarcoidosis. Semin Arthritis Rheum 653-81, 1976
Sartons D, Resnick D, Resnick C: Musculoskeletal
manifestations of sarcoidosis. Semin Roentgen01 20:
37&386, 1985
Arnold WJ: Saxcoidosis, Textbook of Rheumatology.
Third edition. Edited by WN Kelley, ED Harris Jr, S
Ruddy, CB Sledge. Philadelphia, WB Saunders, 1989
Sharma OP, Izumi T: Sarcoidosis, The Lung in Rheumatic Diseases. Edited by GW Cannon, GA Zimmerman. New York, Dekker, 1990
Lyons DJ, Fielding JF: What’s in a relationship? Sarcoidosis and tuberculosis. Ir Med J 83:76-79, 1990
Sequiera W, Stinar D: Serum angiotensin converting
enzyme levels in sarcoid arthritis. Arch Intern Med
Strange C, Barbarash RA, Heffner JE: Lidocaine concentrations in bronchoscopic specimens. Chest 93547549, 1988
Brun J, Pozzetto H, Buffat JJ, Soustelle J, Vauzelle JL,
Patin R: Sarcoidose verttbrale et sacro-iliaque avec
image de pseudo-abces pottique: guerison par corticothkrapie. Presse Med 7451 1-516, 1966
Cabanel G, Jacquot F, Phelip X, Gras JP, Mouries D:
Les formes articulaires de la sarcoidose. Semin HBp
Paris 49:3051-3060, 1973
13. Blanchon P, Paillas J, Lauriat H, Tominez G: Localisations pelvirachidiennes et rachidiennes de la sarcoidose
de BBS. Ann Med Interne (Paris) 127:843-848, 1976
14. Mijiyawa M, Fereres M, Deutsch JP, Awada H, Dougados M, Amor B: Atteinte pelvirachidienne de la sarcoidose: a propos d'une observation: revue de la littkrature.
Rev Rhum 56529-532, 1989
15. Perlman SG, Damergis J, Witorsch P, Cooney FD,
Gunther SF, Barth WF: Vertebral sarcoidosis with paravertebral ossification. Arthritis Rheum 21:271-276, 1978
16. Treves R, Rosey T, Dupenloup P, Dry11 A, Ryckewaart
A: Arthrite sacroiliaque au cours de la sarcoidose. Rev
Rhum Mal Osteoartic 45:733-736, 1978
17. Lin SR, Levy W, Go EB, Lee I, Wong WK: Unusual
osteosclerotic changes in sarcoidosis, simulating osteoblastic metastases. Radiology 106:311-312, 1973
18. Marcove R, Rooney R, Weis L: Osteosclerotic lesions in
sarcoidosis. Clin Orthop 129:248-250, 1977
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tuberculosis, pulmonaria, complicated, accompanied, sacroiliitis, sarcoidosis
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