Fluctuations in serum cytokine levels in the patient with cyclic neutropenia.

The fluctuations of the levels of serum cytokines in a patient with cyclic neutropenia were studied. The greatest fluctuation was found in granulocyte colony-stimulating factor (G-CSF). Tumor necrosis factor-alpha level fluctuated inversely with fluctuation of G-CSF, and oscillation of interleukin (IL)-6 level preceded that in G-CSF level. It is likely that the variations of the levels of cytokines play a significant role in regulating hematopoiesis in cyclic neutropenia. Our results suggested that the stage of the granulocyte-committed stem cell is the major step of the defect, and the defect at the stage of the multipotent stem cell or bone marrow microenvironment was also suggested.     

Pseudopseudarthrosis in a patient with ankylosing spondylitis

There is a growing consensus that the mechanism leading to extensive discovertebral destruction (type III) in most patients with ankylosing spondylitis relates to fracture and subsequent pseudarthrosis. We introduced the term "pseudopseudarthrosis" to describe (in our case) the occurrence of abnormal motion between 2 fused spinal segments, resulting in extensive discovertebral destruction without fracture or pseudarthrosis.     

Oligo-secretory myeloma in a patient with ankylosing spondylitis

Non-secretory myeloma is a plasma cell dyscrasia characterized by the absence of serum and urinary monoclonal immunoglobulins on electrophoretic tests. Because of the lack of monoclonal protein, the identification of the disease is more difficult than for secretory myelomas. The coexistence of ankylosing spondylitis and multiple myeloma has been reported occasionally. We report a rare case of oligo-secretory myeloma coexistent with ankylosing spondylitis.     

Regional migratory osteoporosis in a patient with ankylosing spondylitis

We report the first case of regional migratory osteoporosis (RMO) in a patient with ankylosing spondylitis (AS). This middle-aged man suffered from an acute onset of knee pain that increased on weight bearing, followed by ankle pain. The diagnosis of RMO was confirmed using magnetic resonance imaging (MRI), after exclusion of other causes of knee pain. MRI revealed a large area of bone marrow oedema without a zone of demarcation or subchondral fracture with a demonstration of shifting marrow oedema on the follow-up MRI scan from the medial femur condyl to the tibia plateau lateral and then to the distal tibia epiphysis. Treatment with the bisphosphonate ibandronate, however, was unsuccessful. RMO is characterized clinically by migrating arthralgia of the weight-bearing joints of the lower limbs, mainly in middle-aged males. Although the aetiology is unknown, the pathophysiology of RMO seems to be closely related to transient osteoporosis of the hip (TOH), which has been considered a reversible stage of avascular necrosis of the hip (AVN). There is no causal treatment for RMO. Avoidance of weight bearing and use of analgesics are effective in reducing symptoms. The combination of RMO and AS yielded diagnostic difficulties, as the clinical picture and the marrow oedema seen on MRI could be attributed to several AS-related causes such as enthesitis, early stadium of arthritis, osteonecrosis, or sterile osteomyelitis.     

Sensorineural hearing loss in a patient with ankylosing spondylitis

Rheumatology International -     

Infliximab-induced lupus-like syndrome in a patient with ankylosing spondylitis

Infliximab is a chimerical monoclonal antibody currently used in the treatment of various inflammatory diseases. Lupus-like syndrome is a rarely reported adverse event, and generally observed in rheumatoid arthritis cases. We hereby define and describe a case of a lupus-like syndrome, which developed following the 4th infliximab infusion in a 62-year-old patient with ankylosing spondylitis (AS). As far as we acknowledge, the present case is the third AS case with infliximab-induced lupus.     

Transverse myelitis in a patient with long-standing ankylosing spondylitis

Ankylosing spondylitis is reported to involve not only the joints but other organs as well. Among these extra-articular involvements, uncommon complications associated with nervous system such as single root lesions, compression of the myelum and cauda equina syndrome have also been documented. Here we present a patient with long-standing ankylosing spondylitis who developed spastic paraparesis. Extensive study to find the cause of a spastic paraparesis failed and therefore led to the conclusion that this patient was suffering from transverse myelitis. Similar reports in the past have been attributed to an association with multiple sclerosis; however, we suggest that the findings support the diagnosis of a rare complication of ankylosing spondylitis with an unknown etiology.     

Subacute bacterial endocarditis in a patient with ankylosing spondylitis.

A 57 year old white man with aortic insufficiency and previously undiagnosed ankylosing spondylitis, who developed subacute bacterial endocarditis (SBE), is described. Emergency aortic valve replacement was necessary, and the aortic valve pathology showed diffusely fibrosed and thickened valve leaflets with bacterial vegetations. This is the first recorded case of SBE in ankylosing spondylitis.     

Tuberculous myositis in a patient with ankylosing spondylitis treated with adalimumab

We present a rare case of tuberculous myositis in a 36-year-old man with long-standing ankylosing spondyltis treated with adalimumab. We review the association between antitumor necrosis factor therapy and tuberculous myositis. Our case illustrates that the index of suspicion of tuberculosis in these patients, even with atypical clinical features, must be very high and emphasizes that this rare infection may occur even with negative tuberculosis screening before therapy was started.     

Splenic tuberculosis in a patient with ankylosing spondylitis treated with adalimumab

We present a rare case of splenic tuberculosis in a 42-year old man with long-standing ankylosing spondylitis treated with adalimumab. We review the association between antitumor necrosis factor therapy and splenic tuberculosis. Our case, like many other reported cases, illustrates that the index of suspicion of tuberculosis in patients treated with anti TNF therapies must be high and emphasizes that this rare infection may occur even with negative tuberculosis screening before the initiation of therapy.     

Pulmonary sarcoidosis in a patient with ankylosing spondylitis treated with infliximab

We present a case of a 34 year-old male diagnosed with ankylosing spondylitis. After two years of treatment with infliximab, the patient developed a clinical picture compatible with stage II thoracic sarcoidosis. These findings resulted in the interruption of infliximab therapy. The patient was not administered new treatment since respiratory function testing did not confirm harmful repercussions. After a follow-up of 1 year, the patient is asymptomatic and radiologic tests show complete resolution of pulmonary infiltrates and mediastinal and bilateral hilar lymphad- enopathy.