The coexistence of Behçet's disease and ankylosing spondylitis

Eur J Gen Med 2013;10(2):112-114 Case Report Coexistence of Behçet’s Disease and Ankylosing Spondylitis Sami Küçükşen1, Sinan Bağçacı1, A. Yavuz Karahan2, Muhammed Şahin1, Hatice Uğurlu1 ABSTRACT Behçet’s disease (BD) is a disease which has effects on different systems. Genital ulcer, aphthous stomatitis and iritis are characterized by triple symptom complex of BD. BD is considered to be a systemic vasculitis. BD was previously accepted in spondyloarthropathy (SSpA) group, but there are many reasons for not classifying BD as one of SSpA group. Ankylosing spondylitis (AS) is a prototype of seronegative spondyloarthropathy, and mainly axial skeleton is affected. In this paper, we aimed to present a-33-year old female patient with coexistence of BD and AS. In addition, the coexistence was aimed to be discussed. Key words: Behçet's disease, ankylosing spondylitis, coexistence Behçet Hastalığı ve Ankilozan Spondilit Birlikteliği ÖZET Behçet hastalığı (BH) farklı sistemler üzerine etkileri olan bir hastalıktır. Genital ülser, aftöz stomatit ve iritis, üçlü semptom kompleksi ile karakterizedir. BH sistemik bir vaskülit olarak kabul edilir. BH daha önceleri spondiloartropati (SSPA) grubu içerisinde kabul edilirdi, ancak BH’ yi SSPA grubu içerisinde sınıflandırmamak için pek çok neden vardır. Ankilozan spondilit (AS) özellikle aksiyal iskelet tutulumu ile seyreden seronegatif spondilartropati grubunun bir prototipidir. Bu yazıda BH’ na eşlik eden AS tablosuyla izlenen 33 yaşındaki bir kadın hastayı sunmayı ve bu iki hastalığın birlikteliğini tartışmayı amaçladık. Anahtar kelimeler: Behçet hastalığı, ankilozan spondilit, birliktelik INTRODUCTION Behçet’s disease (BD) is a disease which has effects on different systems. BD is characterized with oral or orogenital ulcers and various systemic (eye, skin, joint, central nervous system, and blood vessels) symptoms. The basic anatomical lesion is vasculitis (1,2). Ankylosing spondylitis (AS), a prototype of seronegative spondiloartropathy (SSpA) group, is a chronic inflammatory disease of the axial skeleton primarily involving the sacroiliac joint and vertebra. The coexistence of BD and AS has been rarely reported. Whether BD is one of SSpA group and whether BD progresses with sacroiliitis development have been subjects of debate (2). In this paper, we aimed 1 Department of Physical Medicine and Rehabilitation of the Medical Faculty of Konya University, 2Department of Physical Medicine and Rehabilitation of the State Hospital of Karaman Received: 13.02.2012, Accepted: 28.04.2012 European Journal of General Medicine to present a-33-year old female patient with coexistence of BD and AS. In addition, the coexistence was aimed to be discussed. CASE A-33-year old woman was admitted to our clinic with low back pain lasting for three months. She was suffering from oral ulcers frequently (4 times a month). BD had been diagnosed seven years ago. She had a history of erythema nodosum. A pathergy test was performed, and its finding was positive. Morning stiffness continuCorrespondence: Ali Yavuz Karahan Yunusemre mh Nurani Sk No:1 Meram/Konya, Türkiye E-mail: ayk222@hotmail.com Coexisting Behçet’s disease and ankylosing spondylitis (4) reported only a single case defined AS among 114 patients with BD. This study showed no relationship between AS and BD. In our case, both HLA B51 and B27 were positive. In another study, Yazıcı et al. (5) mentioned that the inter-observer variation may be the major cause for discrepancies in the evaluation of pelvic radiography for sacroiliitis. Figure 1. Sacroiliac MRI showing bilaterally sacroiliitis ing over 30 minutes was accompanied by back pain. On physical examination, spinal extension was limited and painful, and other spinal motions were open and painless. Tenderness on bilateral sacroiliac joints was present with compression. Schober test was:14,5 cm, and modified Schober test was 20cm. The erythrocyte sedimentation rate (ESR) was 25 mm/h, and the C-reactive protein level was 4 mg/dl (0-8). Other biochemical routine test results were normal. RF was negative. HLA-B27 and HLA-B51 were both positive. Sacroiliac Magnetic resonance imaging (MRI) showed bilaterally sacroiliitis (Figure 1). Ankylosing spondylitis was diagnosed under the new Assessments in Spondyloarthritis International Society (ASAS) classification criteria (1), and the case met the Internal Study Group’s (ISG) diagnostic criteria (2) for BD. The patient was administered colchicine 1 mg / day and started on sulfosalazine 2 g/day and indomethacin 150 mg/day. Two months later, patient’s complaints significantly faded away. DISCUSSION There is a discussion on whether Behçet’s disease is in the seronegative spondyloarthropathy group. BD is accepted as a vasculitic syndrome. AS is considered to be in SspA group. Dilsen et al. (3) carried out a study including 334 Turkish patients with BD. Among this study population, 10 % was reported to be AS, and 34% of the patients had sacroiliitis. HLA B51 and B27 were more frequently found to be positive in patients with coexisting BD and AS than normal population. Yazıcı et al. In 2004, ASAS developed a new diagnostic criteria for SSpA to be used in the early stage of disease (6). Before the development of the criteria, New York criteria mainly depending on radiographic sacroiliitis was being commonly used for the diagnosis. Radiographic evaluation for sacroiliitis often reflects no changes in early period (7). While utilizing the criteria, the observer could fail to detect the inflammation on radiographies at early stages. Radiography shows structural changes occuring due to inflammation in sacroiliac joints rather than the signs of inflammation. Inflammation in sacroiliac joints can be detected using MRI earlier than radiographies (811). In our case, we diagnosed sacroiliitis via sacroiliac MRI. AS was diagnosed under ASAS classification criteria about 3 months earlier than the beginning of symptoms. In other words, the diagnosis of AS was at an early stage in our case. The coexistence of AS and BD is a rare entity. On the other hand, the number of studies reporting this coexistence is increasing (12). Some medications like NSAIDs may be effective in the treatment of BD, and this may mask the symptoms of AS. As the number of studies related to the coexistence between AS and BD increases, the relationship between the conditions will become more understandable. Some medications like NSAIDs may be used in BD, and this may mask symptoms of AS. 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