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A Case of Relapsing Polychondritis Associated with Ankylosing Spondylitis.

Nam Hee YI ; Seung Woon PARK ; In Seong PARK ; Chi Hwan PARK ; Choong Won LEE

Journal of Rheumatic Diseases.2015;22(1):56-60. doi:10.4078/jrd.2015.22.1.56

Relapsing polychondritis is an uncommon disease associated with inflammation in cartilaginous tissues throughout the body, particularly affecting the cartilaginous structures of ears, nose, joints, and respiratory tract. Several autoimmune diseases, including vasculitis, are associated with the concurrent relapsing polychondritis. However, ankylosing spondylitis primarily affecting the sacroiliac joints and spine is rare in patients with relapsing polychondritis. We report on a 54-year-old man with concurrently relapsing polychondritis and ankylosing spondylitis.
Autoimmune Diseases ; Ear ; Humans ; Inflammation ; Joints ; Middle Aged ; Nose ; Polychondritis, Relapsing* ; Respiratory System ; Sacroiliac Joint ; Spine ; Spondylitis, Ankylosing* ; Vasculitis

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A Case of Essential Thrombocythemia in a Patient with Ankylosing Spondylitis Concomitantly Treated with Adalimumab.

Dae Sung LEE ; Seung Geun LEE ; Ho Jin SHIN ; Sun Hee LEE ; Eun Kyoung PARK ; Hae Jung NA ; Chul Hong PARK ; Ji Heh PARK ; In Sub HAN ; Geun Tae KIM

Journal of Rheumatic Diseases.2015;22(1):51-55. doi:10.4078/jrd.2015.22.1.51

Extreme thrombocytosis in patients with ankylosing spondylitis (AS) is rarely reported. Because the relationship between high disease activity and increased platelet counts is somewhat contradictory, severe thrombocytosis in AS patients can be secondary to infection, iron deficiency anemia, drug administration, and hematologic malignancies. Essential thrombocythemia (ET) is a rare acquired stem cell neoplasm characterized by overproduction of platelets by megakaryocytes in the bone marrow in the absence of other causes of thrombocytosis. There is no report in the literature regarding the association between AS and ET. We report on a case of a 34-year-old Korean man with active AS diagnosed as JAK2V617F mutation negative ET during adalimumab treatment.
Adult ; Anemia, Iron-Deficiency ; Bone Marrow ; Hematologic Neoplasms ; Humans ; Megakaryocytes ; Platelet Count ; Spondylitis, Ankylosing* ; Stem Cells ; Thrombocythemia, Essential* ; Thrombocytosis ; Tumor Necrosis Factor-alpha ; Adalimumab

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Psoriatic Onycho-pachydermo-periostitis of the Fingertips: A Report of Two Cases.

Myung Il PARK ; Bo Yeon KIM ; In PARK ; Ki Tae KWON ; Dong Joo SHIN ; Gun Woo KIM ; Sung Ho KIM ; Seung Woo HAN

Journal of Rheumatic Diseases.2015;22(1):45-50. doi:10.4078/jrd.2015.22.1.45

Psoriatic onycho-pachydermo-periostitis (POPP) causes severe nail dystrophy, painful soft tissue swelling, and marked periosteal reaction of the involved distal phalanx. There are few reports of POPP involving the great toe. We report on 2 cases of POPP involving the fingertips. A 60-year-old woman presented with fusiform swelling of her right 4th fingertip with severe tenderness, and her fingernails and toenails had varying degrees of onycholysis. She had mixed multiple erosions and meta-epiphyseal periostitis at the distal phalanx of the right 4th finger but was treated successfully with methotrexate and cyclosporine. A 39-year-old woman presented with painful swelling of the left 2nd and 5th fingertip, psoriatic lesions on the knees and soles of the feet, and onycholysis without reactive periostitis of the left 2nd and 5th fingers. She was treated successfully with cyclosporine. Despite its rarity, POPP should be considered when diagnosing arthritic or infectious conditions affecting the distal interphalangeal joint.
Adult ; Arthritis ; Cyclosporine ; Female ; Fingers ; Foot ; Humans ; Joints ; Knee ; Methotrexate ; Middle Aged ; Nails ; Onycholysis ; Periostitis ; Psoriasis ; Toes

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Knee Synovitis Mimicking a Septic Arthritis.

Jeong Eun PARK ; Hyun Sik KIM ; Jae Ho SEONG ; Sung Sam HA ; Seoung Wan NAM ; Hyang Sun LEE ; Jae Seok KIM ; Jae Won YANG ; Taeyoung KANG

Journal of Rheumatic Diseases.2015;22(1):39-44. doi:10.4078/jrd.2015.22.1.39

Synovitis is the inflammation of the synovial membrane with unknown etiology which occurs in association with auto-immune inflammatory arthritis, mainly in rheumatoid arthritis. Synovitis manifesting as rapidly progressing monoarticular or pauciarticualr symptoms could make early diagnosis difficult, thus it could be misdiagnosed as other forms of arthritic diseases. We experienced a rare case of knee joint synovitis which initially manifested as mimicking a septic arthritis. A 58-year-old-male patient underwent renovascular embolization due to retroperitoneal hemorrhage which was developed after renal biopsy. Suddenly, the patient's left knee joint became swollen rapidly with redness and tenderness. Moreover, his right knee also became inflamed. Surgical irrigation and intravenous antibiotics had never worked on his knee joint inflammation, however administration of intermediate dose of steroid could decrease inflammatory signs dramatically. Synovitis in a large joint could be mistaken as a septic arthritis, delaying the right diagnosis. Thus, we report this case with literature review.
Anti-Bacterial Agents ; Arthritis ; Arthritis, Infectious* ; Arthritis, Rheumatoid ; Biopsy ; Diagnosis ; Early Diagnosis ; Hemorrhage ; Humans ; Inflammation ; Joints ; Knee Joint ; Knee* ; Synovial Membrane ; Synovitis*

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A Case of Sudden Sensorineural Hearing Loss in a Patient with Axial Spondyloarthritis.

Hyon Joung CHO ; Inku YO ; Heejung RYU ; Juuhyoung LEE ; Han Joo BEAK

Journal of Rheumatic Diseases.2013;20(2):132-135. doi:10.4078/jrd.2013.20.2.132

Sudden sensorineural hearing loss (SNHL) is rarely presented in patients with several immune-mediated inflammatory diseases. We report a case of sudden SNHL in a patient with axial spondyloarthritis (aSpA). A 29-year-old male with aSpA was admitted for sudden unilateral hearing loss. His aSpA symptom was stable; however, pure tone audiometry revealed that he had SNHL in the left ear at low frequency. His hearing was fully recovered with systemic and local steroid therapy. This case suggests that sudden SNHL may be an extra-articular manifestation of aSpA and thus need prompt steroid therapy in order to restore hearing.
Audiometry ; Ear ; Hearing ; Hearing Loss, Sensorineural ; Hearing Loss, Unilateral ; Humans ; Male ; Spondylitis, Ankylosing

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A Case of ANA-negative, Anti-dsDNA Negative, and Anti-Ro/SSA Positive Membranous Lupus Nephropathy.

Hee Chan YANG ; Myong Joo HONG ; Jin Han LIM ; Seol A JANG ; So Yeon JEON ; Wan Hee YOO

Journal of Rheumatic Diseases.2013;20(2):127-131. doi:10.4078/jrd.2013.20.2.127

Systemic lupus erythematosus (SLE) is an autoimmune disease characterized by the production of diverse autoantibodies with various systemic organ involvements. In patients with SLE, autoantibodies, such as antinuclear antibody (ANA) and anti-dsDNA antibody, play an important role not only in diagnosing the disease, but also representing the pathogenesis of the disease. ANA is the main screening tool in diagnosis and serum complement levels and anti-dsDNA antibody level are closely related to the disease activities. Nevertheless, exceptionally, some patients represent with negative ANA and/or anti-dsDNA antibody leading to difficulties in diagnosing the disease. Here, we report a case of 37-year old female SLE patient with negative ANA, negative anti-dsDNA antibody, and positive anti-Ro/SSA antibody, which manifested with nephrotic syndrome.
Antibodies, Antinuclear ; Autoantibodies ; Autoimmune Diseases ; Complement System Proteins ; Female ; Glomerulonephritis, Membranous ; Humans ; Lupus Erythematosus, Systemic ; Mass Screening ; Nephrotic Syndrome

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A Case of Refractory Behcet's Uveitis Improving after Insertion of Fluocinolone Acetonide Implant.

Yun Sung KIM ; Jung Ran CHOI ; Seok Won KIM ; Hyun Sook KIM

Journal of Rheumatic Diseases.2013;20(2):123-126. doi:10.4078/jrd.2013.20.2.123

Behcet's disease (BD) is systemic vasculitis that can manifest severely debilitating. Despite the understanding mechanisms of overall BD, there are remains many questions in various critical manifestations and treatments. The ocular manifestation is characterized by a prototype of chronic relapsing and persistent uveitis. The main treatment is topical corticosteroid, and topical nonsteroidal anti-inflammatory drugs in mild uveitis. The recurrent and severe uveitis could be treated with ocular corticosteroid injections, and systemic corticosteroid for inducing long-lasting suppression of the inflammation. Systemic corticosteroids should rapidly be tapered within weeks for avoiding side effects. Recent advances have led to the development of sustained-release corticosteroid devices using different corticosteroids. We present a case of 67-year-old woman who received a fluocinolone acetonide implant for recurrent Behcet's uveitis. She was successfully treated with implant and the uveitis became quiescent within a month.
Adrenal Cortex Hormones ; Female ; Fluocinolone Acetonide ; Humans ; Inflammation ; Systemic Vasculitis ; Uveitis

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Remission of Lymphocytic Interstitial Pneumonia in Sjogren's Syndrome after Autologous Peripheral Blood Stem Cell Transplantation.

Han Na CHOI ; Jung Yoon CHOE ; Si Hye KIM ; Seong Kyu KIM ; Hun Mo RYOO ; Sung Hoon PARK

Journal of Rheumatic Diseases.2013;20(2):118-122. doi:10.4078/jrd.2013.20.2.118

Interstitial pneumonia occurs in approximately 25% of patients with primary Sjogren's syndrome. Interstitial pneumonia combined with primary Sjogren's syndrome usually responds well to systemic steroids, and fatal cases are rare. Lymphocytic interstitial pneumonia shows diffuse infiltration of polyclonal B and T cells. Autologous stem cell transplantation is performed in cases of primary Sjogren's syndrome as an optional treatment when the condition responds poorly to conventional treatment. The hypothesis that primary Sjogren's syndrome improves after transplantation relies on the role of B-cell abnormalities in pathogenesis or the strong effects of immunosuppressive therapy. We experienced the case of a patient diagnosed with primary Sjogren's syndrome and lymphocytic interstitial pneumonia progression refractory to conventional treatment (steroid and immunosuppressive drugs) and cyclophosphamide pulse therapy. Our patient demonstrated improvement of lung manifestations and autoimmune disease activity after autologous stem cell transplantation.
Autoimmune Diseases ; B-Lymphocytes ; Cyclophosphamide ; Humans ; Lung ; Lung Diseases, Interstitial ; Peripheral Blood Stem Cell Transplantation ; Sjogren's Syndrome ; Stem Cell Transplantation ; Steroids ; T-Lymphocytes ; Transplants

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Typical 18-FDG-PET/CT Findings of Polymyalgia Rheumatica: A Case Report.

Jin Su PARK ; Jung Yoon PYO ; Hee Jin PARK ; Hyang Sun LEE ; Yoon KANG ; Mi Il KANG ; Jungsik SONG ; Yong Beom PARK ; Soo Kon LEE ; Sang Won LEE

Journal of Rheumatic Diseases.2013;20(2):113-117. doi:10.4078/jrd.2013.20.2.113

Polymyalgia rheumatica (PMR) is an inflammatory rheumatic condition characterized by generalized pain and morning stiffness in the shoulders, hip girdle, and neck. Since the pathogenesis of PMR is still uncertain, the diagnosis of PMR depends on clinical features. There have been several studies regarding radiological tools for the diagnosis of PMR. Recent studies using 18-FDG-PET showed bursitis, synovitis, uptake in the spinous process and asymptomatic large-vessel vasculitis in PMR patients. However, there was no report on the efficacy of 18-FDG-PET for diagnosis of PMR in Korea. Here, we are first reporting a case of a Korean patient with PMR, who had radiological findings including bursitis, synovitis, uptake in the spinous process and asymptomatic large-vessel vasculitis on 18-FDG-PET/CT.
Bursitis ; Hip ; Humans ; Korea ; Neck ; Polymyalgia Rheumatica ; Positron-Emission Tomography and Computed Tomography ; Shoulder ; Synovitis ; Vasculitis

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Rituximab for Rheumatoid Arthritis Following TNF-alpha Inhibitor Associated Splenic Tuberculosis.

Jin Su KIM ; Jung Ran CHOI ; Jung Soo SONG ; Kyung Joon KIM ; Youn Su PARK ; Jun Hwan CHO ; Min Jee HAN ; Sang Tae CHOI

Journal of Rheumatic Diseases.2013;20(2):108-112. doi:10.4078/jrd.2013.20.2.108

One of the most important adverse effects of a tumor necrosis factor (TNF)-alpha inhibitor is the reactivation of tuberculosis. Most of them occur in the lung, but sometimes they can be found in other organs. Moreover, the proper management of active rheumatoid arthritis (RA) in patients with anti-TNF-alpha associated tuberculosis is still in debate. We present the case of a seropositive RA patient who showed good response with rituximab, an anti-CD20 monoclonal antibody, after developing splenic tuberuculosis, following treatment with TNF-alpha inhibitor. Confirming a diagnosis of splenic tuberculosis is difficult and can be delayed due to its nonspecific symptoms and rare occurrence. This case suggests that splenic tuberculosis should be doubted in RA patients treated with TNF-alpha inhibitor, and that rituximab may be considered as an alternative treatment option in RA patients with anti-TNF-alpha associated tuberculosis.
Antibodies, Monoclonal, Murine-Derived ; Arthritis, Rheumatoid ; Humans ; Lung ; Tuberculosis ; Tuberculosis, Splenic ; Tumor Necrosis Factor-alpha ; Rituximab

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