436-439
Clinicalrheurnatology, 1992, 11, N ~ 3
Case report
Ankylosing Spondylitis and Sarcoidosis Coincidence or Association ? Case Report and Review of the Literature G. S T U C K I ,
A. V O N
FELTEN*,
R. S P E I C H * ,
B.A.
MICHEL
Summary
We report a 25-year-old woman presenting with sarcoidosis and bilateral sacroiliitis. Her sarcoidosis related symptoms (malaise, cough and dyspnoea) improved dramatically under treatment with steroids but severe back pain persisted. Only seven similar cases have been described over the last 40 years and the question of a possible association between the two diseases has been raised. However, prevalence data from the literature and the apparent lack of genetic links are better arguments for coincidence than for association.
Key words Ankyiosing Spondylitis, Sacroiliitis, Sarcoidosis, HLA-B27, HLA-B8, HLA-DR3.
INTRODUCTION Since 1951, when Verstraeten (1) reported a first case with both sacroiliitis and sarcoidosis, six similar cases have been described in the literature, including only one female patient. Already Verstraeten and later Martin (2), Deshayes (3), Cabanel (4), Blanchon (5) and Gerster (6) raised the question about a possible association between the two diseases. We report another case of a 25-year-old woman presenting typical findings of sarcoidosis and a six-month history of bilateral iliosacral pain. The probability of an association between the two diseases is discussed. CASE R E P O R T The personal history of the 25-year-old female patient revealed a single episode of lumbar pain on motion irradiating to both legs in 1985; X-rays showed a spondylolisthesis of L5 on S1. Being in good health until February 1990, the patient then noted pain in her midback and in the area of the right sacroiliac joint irradiating to the right buttock. Maximum pain regularly interrupted her sleep between one and three o'clock in the
Department of Rheumatologyand PhysicalTherapy, *Department of Internal Medicine, UniversityHospital, Zurich, Switzerland.
morning. And low back stiffness was present for about two hours after rising. Further symptoms included recurrent fever up to 38~ poor appetite, and weight loss of 10 kilos within a six-month period. In March 1990 ankylosing spondylitis was diagnosed by a practicing rheumatologist based on symptoms, clinical findings and inflammatory erosive X-ray changes of both sacroiliac joints (Fig. 1). HLA-B27 was positive, an ileocolonoscopy including biopsy and histology examinations of both the ileum and colon were normal, the erythrocyte sedimentation rate (ESR) was 16 mm after 1 hour (Westergren). There was no evidence of presence of another form among the seronegative spondyloarthropathies. In June 1990 the patient was referred to our inpatient clinic for evaluation because of severe general malaise and increasing pain resistent to NSAID's (indomethacin, diclofenac, ibuprofen, phenylbutazone). Upon admission the patient reported severe low back pain irradiating to the buttocks, dyspnoea and palpitation when climbing two flights of stairs, and a nonproductive cough. The clinical examination revealed limited painful motion of both lumbar (Schober Index 10/11 cm) and thoracic spine in all directions. The thoracic excursion was limited to 80/83 cm. The sacroiliac provocation tests were strongly positive on both sides. No other significant findings were present. Laboratory screening showed an ESR of 34 mm after 1 hour, a C-reactive protein of 36.1 mg/1 (upper limit 10 mg/1), a slight thrombocytosis of 443 x
Ankylosing spondylitis and sarcoidosis
437
Fig. 1 : Bilateral sacroiliitis on ap view.
Table I : Case reports with both ankylosing spondylitis and sarcoidosis Author (year of publication)
Sex
Age
Musculoskeletal symptoms (age at onset)
Manifestations of sarcoidosis (age at onset)
Time interval (months)
Therapy
Course
Verstraeten (1951) Martin (1953) Deshayes (1%5)
m
36
none
?
58
84(?)
none
m
48
bihilar lymphadenopathy (36) bihilar lyrnphadenopathy (38) bihilar lymphadenopathy, pulmonary fibrosis
72
m
ankylosing spondylitis (30) ankylosing spondylitis (45 ?) ankylosingspondyfitis (40)
?
prednisolone
subsequent pulmonary fibrosis less back pain under steroids
Brun (1966)
f
34
left sacroiliitis (34)
8
steroids, chemotherapy
Cabanel (1973)
m
40
bilateral sacroiliitis (24)
bihilar lymphadenopathy, pulmonary fibrosis (33) bihilar iymphadenopathy, pulmonary fibrosis
0 (?)
chemotherapy, phenylbutazone
Blanchon (1976)
m
24
prednisone
m
27
pulmonary fibrosis, parotitis, peripheral iymphadenopathy (18) bihilar lymphadenopathy, erythema nodosum, arthritis left ankle (25)
12
Gerster (1981) "
left sacroiliitis (21) bilateral sacroiliitis, relapsing uveitis (24)
6
phenylbutazone
(4O)
(24)
103/~tl and normocalcaemia (the reported incidence of hypercalcaemia in sarcoidosis varies from 2 to 63%, (7)). The angiotensin converting enzyme level was also within normal limits (44% of patients with bihilar lymphadenopathy but normal lung parenchyma have angiotensin converting enzyme levels within normal limits, (8)). On
subsequent oligoarthritis (knee, hip) and ankylosing spondylitis subsequent bony cysts (hand, feet), skin ulceration, epididymitis subsequent bihilar lymphadenopathy; left ankle arthritis and erythema nodosum disappear; low back pain perSlSts.
chest X-ray (Fig. 2) a bihilar lymphadenopathy and a normal lung parenchyma were found and confirmed by computerized tomography. Pulmonary function tests revealed a moderate restrictive lung disease with a mild obstructive component, and a slightly decreased carbon monoxide diffusing capacity. Transbronchial biopsy
438
G. Stucki, A. von Felten, R. Speich, B.A. Michel
showed multiple noncaseating granulomas with epitheloid and multinucleated giant cells (Fig. 3), and bronchioalveolar lavage yielded an increased helper T-cell to supressor T-cell ratio of 4.5:1 (a ratio of over 3.5:1 yielded a sensitivity of 53.2% and a specificity of 92.8% in accord with reference 9). The ratio in the blood was 2.1:1. All acid-fast and fungal stains and cultures were negative. Electrocardiogram, echocardiography, and ophthalmologic examination were normal. Under treatment with prednisone starting at 50 mg per day, general malaise, dyspnoea and cough disappeared, and ESR (6 mm/1 hour), C-reactive protein (16 rag/l), pulmonary function tests and bihilar lymphadenopathy returned to normal. After a follow-up of one year, however, the patient still suffers from severe low back pain exacerbating at night despite high doses of indomethacin. A tissue typing for the HLA antigens revealed the following resuits: A1, - ; B8, 27; DR3, 4 (DRw52, 53; DQw2, 3).
DISCUSSION
2: Chest roentgenogram showing bilateral hilar lymphadenopathy.
Fig.
This is a report of a 25-year-old woman with ankylosing spondylitis who developed symptomatic pulmonary sarcoidosis. Despite prompt improvement of the sarcoidosis related malaise, dyspnoea and cough as well as the ESR under treatment with prednisone, persistent in-
Fig. 3: Transbronchialbiopsy showingnoncaseatinggranulomawith epitheloid cells including a multinucleargiant cell.
Ankylosing spondylitis and sarcoidosis
f l a m m a t o r y b a c k p a i n was n o t e d d e s p i t e a d d i t i o n a l N S A I D t h e r a p y . This d i f f e r e n c e b e t w e e n t h e t h e r a p e u tic r e s p o n s e s o f p u l m o n a r y a n d m u s c u l o s k e l e t a l symptoms suggests the p r e s e n c e o f d i f f e r e n t p a t h o g e n e t i c m e c h a n i s m s involved in t h e two d i s o r d e r s . B a s e d on e p i d e m i o l o g i c a l d a t a t h e r e is n o r e a s o n to a s s u m e a n association o f t h e two e n t i t i e s e a c h having a r e l a t i v e l y high p r e v a l e n c e . T h e p r e v a l e n c e o f ankylosing s p o n d y l i t i s is r e p o r t e d as 1 p e r 1000 p o p u l a t i o n (10), a n d t h e preva l e n c e of s a r c o i d o s i s varies f r o m 0.04 in S p a i n to 64 p e r 1000 p o p u l a t i o n in S w e d e n (11). T h e r e f o r e , b a s e d on p u r e c o i n c i d e n c e , p r e v a l e n c e d a t a for t h e two d i s o r d e r s a r e to b e e x p e c t e d in t h e o r d e r o f 0.00004 to 0.064 p e r 1000, the n u m b e r s b e i n g even h i g h e r if a s s o c i a t i o n o f t h e two d i s e a s e s is a s s u m e d . In t h e l i t e r a t u r e , h o w e v e r , only seven similar cases w e r e r e p o r t e d o v e r a p e r i o d o f 39 y e a r s i n d i c a t i n g t h e l a c k of an a s s o c i a t i o n b e t w e e n sarcoidosis a n d ankylosing spondylitis ((1-6,12), T a b l e I). A g e n e t i c a l l y - d e t e r m i n e d p r e d i s p o s i t i o n to d e v e l o p ei-
439
t h e r s a r c o i d o s i s o r a n k y l o s i n g spondylitis has b e e n n o t e d in c e r t a i n g r o u p s o f p a t i e n t s . H L A - D R 5 was significantly i n c r e a s e d in a n e n d e m i c c l u s t e r o f p a t i e n t s with sarcoidosis in G e r m a n y (13,14), a n d H L A - B 8 has b e e n ass o c i a t e d with a c u t e s a r c o i d o s i s - r e l a t e d a r t h r i t i s (15) a n d with s p o n t a n e o u s r e s o l u t i o n (16). H L A - B 8 a n d H L A - D R 3 have b e e n a s s o c i a t e d with a c u t e o n s e t o f sarc o i d o s i s with a r t h r i t i s (17,18). O n t h e o t h e r h a n d , t h e r e is a c l e a r a s s o c i a t i o n o f a n k y l o s i n g s p o n d y l i t i s with H L A B27 (19). H o w e v e r , n o c o m m o n g e n e t i c links have b e e n i d e n t i f i e d for t h e two d i s o r d e r s . I n t e r e s t i n g l y , in o u r p a t i e n t w e f o u n d H L A - B 2 7 as well as H L A - B 8 a n d H L A D R 3 to b e positive. T h e p r e s e n c e o f t h e typical H L A specificities for b o t h d i s e a s e s m a y i n d i c a t e t h e coexiste n c e o f two i n d e p e n d e n t d i s e a s e p r o c e s s e s . I n conclusion, r e s p o n s e to t h e r a p y , p r e v a l e n c e d a t a f r o m t h e l i t e r a t u r e a n d t h e a p p a r e n t lack o f g e n e t i c links a r e b e t t e r a r g u m e n t s for c o i n c i d e n c e t h a n for association.
REFERENCES
1.
Verstraeten, J.M., Bekaert, J. Association de spondylite ankylosante et de sarcoidose. Acta Tuberc Belg 1951, 42, 149-151. 2. Martin, E., Fallet, G.H. Pneumopathies chroniques et rhumatisme. Schweiz Med Wochenschr 1953, 83, 776-782. 3. Deshayes, P., Desseauve, J., Hubert, J., Lemercier, J.P., Geoffroy, Y. Un cas de polyarthrite au cours d'une sarcoidose. Un cas de spondylarthrite ankylosante au cours d'une sarcoidose. Rev Rhum 1965, 32, 671-674. 4. Cabanel, G., Jacquot, F., Phelip, X., Gras, J.P., Mouries, D. Les formes articulaires de la sarcoidose. Sem H6p Paris 1973, 49, 3051-3060. 5. Blanchon, P., Paillas, J., Lauriat, H., Tominez, G. Localisations pelvi-rachidiennes et rachidiennes de la sarcoidose de B.B.S.. Ann Med Interne 1976, 127, 843-848. 6. Gerster, J.C., Chappuis, P. Association d'une sarcoidose agu~ et d'une spondylarthrite ankylosante. Schweiz Rundschau Med 1981, 70, 2356-2359. 7. Goldstein, R.A., Israel, H.L., Becker, K.L., Moore, C.F. The infrequency of hypercaleemia in sarcoidosis. Am J Med 1971, 51, 21-30. 8. Studdy, P.R., Lapworth, R., Bird, R. Angiotensin converting enzyme and its clinical significance-a review. J Clin Pathol 1983, 36, 938-947. 9. Costabel, U., Teschler, H., Guzman, J., Kroegel, C. Bronchoalveol~ire Lavage : Zwischenbilanz nach zehn Jahren ldinischer Anwendung. Med Klin 1990, 85, 367-387. 10. Carter, E.T. Epidemiology of ankylosing spondylitis in Rochester, Minnesota, 1935-1973. Arthritis Rheum 1979, 22, 365-370. 11. Teirstein, A.S., Lesser, M. Worldwide distribution and epidemiology of sarcoidosis. In : Sarcoidosis and Other Granulomatous Diseases of the Lung Vol 20. Ed.: Fanburg, B.L., Marcel Dekker, Inc 1983, 101-134.
12. Brun, J., Pozzetto, H., Buffat, J.J., Soustelle, J., Vauzelle, J.L., Patin, R. Sarcoidose vertebrale et sacro-iliaque avec image de pseudo-absc6s pottique. Presse Med 1966, 74, 511-515. 13. Nowack, D., Goebel, K.M. Genetische und diagnostische Bedeutung der HLA-Merkmale bei der Sarkoidose. Schweiz Med Wschr 1986, 116, 1479-1480. 14. Nowack, D., Goebel, K.M. Genetic aspects of sarcoidosis. Arch Intern Med 1987, 147, 481. 15. Brewerton, D.A., Cockburn, C., James, D.C., James, D.G., Neville, E. HLA antigens in sarcoidosis. Clin Exp Immunol 1977, 27, 227-229. 16. Smith, M.J., Turton, C.W.G., Turner-Warwick, M., Morris, L.M., Lawler, S.D. Association of HLA B8 with spontaneus resolution in sarcoidosis. Thorax 1981, 36, 296-298. 17. Hedfors, E., Lindstr6m, F. HLA-B8/DR3 in sarcoidosis. Correlation to acute onset disease with arthritis. Tissue Antigens 1983, 22, 200-203. 18. Kremer, J.M. Histologicfindings in siblingswith acute sarcoid arthritis: Association with the B8, DR3 phenotype. J Rheumatol 1986, 13, 593-597. 19. Khan, M.A. HLA and ankylosing spondylitis. In: Ankylosing Spondylitis - New Clinical Applications Vol. I. Editor : Calabro, J.J., Dick C, Lancaster (England) MTP Press Limited 1987, 23.
Received : 2 December 1991 ; Revision-accepted : 24 March 1992 Correspondence to: G. STUCKI, M.D., Department of Rheumatology, University Hospital, Gloriastr. 25, CH-8091 Zurich.
Clinicalrheurnatology, 1992, 11, N ~ 3
Case report
Ankylosing Spondylitis and Sarcoidosis Coincidence or Association ? Case Report and Review of the Literature G. S T U C K I ,
A. V O N
FELTEN*,
R. S P E I C H * ,
B.A.
MICHEL
Summary
We report a 25-year-old woman presenting with sarcoidosis and bilateral sacroiliitis. Her sarcoidosis related symptoms (malaise, cough and dyspnoea) improved dramatically under treatment with steroids but severe back pain persisted. Only seven similar cases have been described over the last 40 years and the question of a possible association between the two diseases has been raised. However, prevalence data from the literature and the apparent lack of genetic links are better arguments for coincidence than for association.
Key words Ankyiosing Spondylitis, Sacroiliitis, Sarcoidosis, HLA-B27, HLA-B8, HLA-DR3.
INTRODUCTION Since 1951, when Verstraeten (1) reported a first case with both sacroiliitis and sarcoidosis, six similar cases have been described in the literature, including only one female patient. Already Verstraeten and later Martin (2), Deshayes (3), Cabanel (4), Blanchon (5) and Gerster (6) raised the question about a possible association between the two diseases. We report another case of a 25-year-old woman presenting typical findings of sarcoidosis and a six-month history of bilateral iliosacral pain. The probability of an association between the two diseases is discussed. CASE R E P O R T The personal history of the 25-year-old female patient revealed a single episode of lumbar pain on motion irradiating to both legs in 1985; X-rays showed a spondylolisthesis of L5 on S1. Being in good health until February 1990, the patient then noted pain in her midback and in the area of the right sacroiliac joint irradiating to the right buttock. Maximum pain regularly interrupted her sleep between one and three o'clock in the
Department of Rheumatologyand PhysicalTherapy, *Department of Internal Medicine, UniversityHospital, Zurich, Switzerland.
morning. And low back stiffness was present for about two hours after rising. Further symptoms included recurrent fever up to 38~ poor appetite, and weight loss of 10 kilos within a six-month period. In March 1990 ankylosing spondylitis was diagnosed by a practicing rheumatologist based on symptoms, clinical findings and inflammatory erosive X-ray changes of both sacroiliac joints (Fig. 1). HLA-B27 was positive, an ileocolonoscopy including biopsy and histology examinations of both the ileum and colon were normal, the erythrocyte sedimentation rate (ESR) was 16 mm after 1 hour (Westergren). There was no evidence of presence of another form among the seronegative spondyloarthropathies. In June 1990 the patient was referred to our inpatient clinic for evaluation because of severe general malaise and increasing pain resistent to NSAID's (indomethacin, diclofenac, ibuprofen, phenylbutazone). Upon admission the patient reported severe low back pain irradiating to the buttocks, dyspnoea and palpitation when climbing two flights of stairs, and a nonproductive cough. The clinical examination revealed limited painful motion of both lumbar (Schober Index 10/11 cm) and thoracic spine in all directions. The thoracic excursion was limited to 80/83 cm. The sacroiliac provocation tests were strongly positive on both sides. No other significant findings were present. Laboratory screening showed an ESR of 34 mm after 1 hour, a C-reactive protein of 36.1 mg/1 (upper limit 10 mg/1), a slight thrombocytosis of 443 x
Ankylosing spondylitis and sarcoidosis
437
Fig. 1 : Bilateral sacroiliitis on ap view.
Table I : Case reports with both ankylosing spondylitis and sarcoidosis Author (year of publication)
Sex
Age
Musculoskeletal symptoms (age at onset)
Manifestations of sarcoidosis (age at onset)
Time interval (months)
Therapy
Course
Verstraeten (1951) Martin (1953) Deshayes (1%5)
m
36
none
?
58
84(?)
none
m
48
bihilar lymphadenopathy (36) bihilar lyrnphadenopathy (38) bihilar lymphadenopathy, pulmonary fibrosis
72
m
ankylosing spondylitis (30) ankylosing spondylitis (45 ?) ankylosingspondyfitis (40)
?
prednisolone
subsequent pulmonary fibrosis less back pain under steroids
Brun (1966)
f
34
left sacroiliitis (34)
8
steroids, chemotherapy
Cabanel (1973)
m
40
bilateral sacroiliitis (24)
bihilar lymphadenopathy, pulmonary fibrosis (33) bihilar iymphadenopathy, pulmonary fibrosis
0 (?)
chemotherapy, phenylbutazone
Blanchon (1976)
m
24
prednisone
m
27
pulmonary fibrosis, parotitis, peripheral iymphadenopathy (18) bihilar lymphadenopathy, erythema nodosum, arthritis left ankle (25)
12
Gerster (1981) "
left sacroiliitis (21) bilateral sacroiliitis, relapsing uveitis (24)
6
phenylbutazone
(4O)
(24)
103/~tl and normocalcaemia (the reported incidence of hypercalcaemia in sarcoidosis varies from 2 to 63%, (7)). The angiotensin converting enzyme level was also within normal limits (44% of patients with bihilar lymphadenopathy but normal lung parenchyma have angiotensin converting enzyme levels within normal limits, (8)). On
subsequent oligoarthritis (knee, hip) and ankylosing spondylitis subsequent bony cysts (hand, feet), skin ulceration, epididymitis subsequent bihilar lymphadenopathy; left ankle arthritis and erythema nodosum disappear; low back pain perSlSts.
chest X-ray (Fig. 2) a bihilar lymphadenopathy and a normal lung parenchyma were found and confirmed by computerized tomography. Pulmonary function tests revealed a moderate restrictive lung disease with a mild obstructive component, and a slightly decreased carbon monoxide diffusing capacity. Transbronchial biopsy
438
G. Stucki, A. von Felten, R. Speich, B.A. Michel
showed multiple noncaseating granulomas with epitheloid and multinucleated giant cells (Fig. 3), and bronchioalveolar lavage yielded an increased helper T-cell to supressor T-cell ratio of 4.5:1 (a ratio of over 3.5:1 yielded a sensitivity of 53.2% and a specificity of 92.8% in accord with reference 9). The ratio in the blood was 2.1:1. All acid-fast and fungal stains and cultures were negative. Electrocardiogram, echocardiography, and ophthalmologic examination were normal. Under treatment with prednisone starting at 50 mg per day, general malaise, dyspnoea and cough disappeared, and ESR (6 mm/1 hour), C-reactive protein (16 rag/l), pulmonary function tests and bihilar lymphadenopathy returned to normal. After a follow-up of one year, however, the patient still suffers from severe low back pain exacerbating at night despite high doses of indomethacin. A tissue typing for the HLA antigens revealed the following resuits: A1, - ; B8, 27; DR3, 4 (DRw52, 53; DQw2, 3).
DISCUSSION
2: Chest roentgenogram showing bilateral hilar lymphadenopathy.
Fig.
This is a report of a 25-year-old woman with ankylosing spondylitis who developed symptomatic pulmonary sarcoidosis. Despite prompt improvement of the sarcoidosis related malaise, dyspnoea and cough as well as the ESR under treatment with prednisone, persistent in-
Fig. 3: Transbronchialbiopsy showingnoncaseatinggranulomawith epitheloid cells including a multinucleargiant cell.
Ankylosing spondylitis and sarcoidosis
f l a m m a t o r y b a c k p a i n was n o t e d d e s p i t e a d d i t i o n a l N S A I D t h e r a p y . This d i f f e r e n c e b e t w e e n t h e t h e r a p e u tic r e s p o n s e s o f p u l m o n a r y a n d m u s c u l o s k e l e t a l symptoms suggests the p r e s e n c e o f d i f f e r e n t p a t h o g e n e t i c m e c h a n i s m s involved in t h e two d i s o r d e r s . B a s e d on e p i d e m i o l o g i c a l d a t a t h e r e is n o r e a s o n to a s s u m e a n association o f t h e two e n t i t i e s e a c h having a r e l a t i v e l y high p r e v a l e n c e . T h e p r e v a l e n c e o f ankylosing s p o n d y l i t i s is r e p o r t e d as 1 p e r 1000 p o p u l a t i o n (10), a n d t h e preva l e n c e of s a r c o i d o s i s varies f r o m 0.04 in S p a i n to 64 p e r 1000 p o p u l a t i o n in S w e d e n (11). T h e r e f o r e , b a s e d on p u r e c o i n c i d e n c e , p r e v a l e n c e d a t a for t h e two d i s o r d e r s a r e to b e e x p e c t e d in t h e o r d e r o f 0.00004 to 0.064 p e r 1000, the n u m b e r s b e i n g even h i g h e r if a s s o c i a t i o n o f t h e two d i s e a s e s is a s s u m e d . In t h e l i t e r a t u r e , h o w e v e r , only seven similar cases w e r e r e p o r t e d o v e r a p e r i o d o f 39 y e a r s i n d i c a t i n g t h e l a c k of an a s s o c i a t i o n b e t w e e n sarcoidosis a n d ankylosing spondylitis ((1-6,12), T a b l e I). A g e n e t i c a l l y - d e t e r m i n e d p r e d i s p o s i t i o n to d e v e l o p ei-
439
t h e r s a r c o i d o s i s o r a n k y l o s i n g spondylitis has b e e n n o t e d in c e r t a i n g r o u p s o f p a t i e n t s . H L A - D R 5 was significantly i n c r e a s e d in a n e n d e m i c c l u s t e r o f p a t i e n t s with sarcoidosis in G e r m a n y (13,14), a n d H L A - B 8 has b e e n ass o c i a t e d with a c u t e s a r c o i d o s i s - r e l a t e d a r t h r i t i s (15) a n d with s p o n t a n e o u s r e s o l u t i o n (16). H L A - B 8 a n d H L A - D R 3 have b e e n a s s o c i a t e d with a c u t e o n s e t o f sarc o i d o s i s with a r t h r i t i s (17,18). O n t h e o t h e r h a n d , t h e r e is a c l e a r a s s o c i a t i o n o f a n k y l o s i n g s p o n d y l i t i s with H L A B27 (19). H o w e v e r , n o c o m m o n g e n e t i c links have b e e n i d e n t i f i e d for t h e two d i s o r d e r s . I n t e r e s t i n g l y , in o u r p a t i e n t w e f o u n d H L A - B 2 7 as well as H L A - B 8 a n d H L A D R 3 to b e positive. T h e p r e s e n c e o f t h e typical H L A specificities for b o t h d i s e a s e s m a y i n d i c a t e t h e coexiste n c e o f two i n d e p e n d e n t d i s e a s e p r o c e s s e s . I n conclusion, r e s p o n s e to t h e r a p y , p r e v a l e n c e d a t a f r o m t h e l i t e r a t u r e a n d t h e a p p a r e n t lack o f g e n e t i c links a r e b e t t e r a r g u m e n t s for c o i n c i d e n c e t h a n for association.
REFERENCES
1.
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Received : 2 December 1991 ; Revision-accepted : 24 March 1992 Correspondence to: G. STUCKI, M.D., Department of Rheumatology, University Hospital, Gloriastr. 25, CH-8091 Zurich.
