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A Case of Systemic Sclerosis Sine Scleroderma.

Chae Gi KIM ; Hun Mo RHOO ; Joong Goo KWON ; Chang Hyeong LEE ; Yong Ho SONG ; Jung Yoon CHOE

The Journal of the Korean Rheumatism Association.2000;7(3):313-322.

Systemic sclerosis (SSc) is a generalized connective tissue disorder of unknown etiology. Clinically, there is a broad spectrum of disease ranging from widespread severe skin thickening to skin thickening limited to the distal extremities. In rare cases of systemic sclerosis, no cutaneous change only with internal organ involvement has been reported, which is called ?ystemic sclerosis sine scleroderma (ssSSc)?. We describe a patient with Raynaud? phenomenon, who showed intestinal pseudoobstruction as a presenting symptom but did not show any skin change. She had also an esophageal motility disorder, but other organ involvement was not evident. Antinuclear antibody was positive. Her obstruction symptoms were improved by decompression by nasogastric tube and pharmaceutical treatment with erythromycin and octreotide.
Antibodies, Antinuclear ; Connective Tissue ; Decompression ; Erythromycin ; Esophageal Motility Disorders ; Extremities ; Humans ; Intestinal Pseudo-Obstruction ; Octreotide ; Scleroderma, Systemic* ; Sclerosis ; Skin

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A Case of Periosteal New Bone Formation Associated with Behcet? Disease.

Dong Joo KEUM ; Doo Kyoung YANG ; Kwang Jin KIM ; Sam Ryong JEE ; Chin Hyuk YUN ; Sung Won LEE ; Won Tae CHUNG

The Journal of the Korean Rheumatism Association.2000;7(3):308-312.

Behcet? disease is a recurring illness characterized by the triple symptom complex of aphthous stomatitis, genital ulceration, and uveitis. The disease is multisystemic disorder with involvement of skin, gastrointestinal tract, blood vessels, central nervous system, joints, and epididymis. Vascular involvement of Behcet? disease affects both arteries and veins and blood vessels of all sizes. Periosteal new bone formation is one feature of hypertrophic osteoarthropathy. Other features are clubbing and arthritis. Periosteal new bone formation on the bones of the lower extremities without other features of hypertrophic osteoarthropathy has been reported in patients with varicose veins, vitamin A intoxication, infantile cortical hyperostosis, tuberous sclerosis, congenital syphilis and chronic renal failure with hyperparathyroidism, and chronic venous insufficiency. However, periosteal new bone formation has not, to our knowledge, been described in patients with Behcet? disease yet although similar findings are occasionally noted in polyarteritis nodosa. We report a case of Behcet? disease complicated by leg swelling with periosteal new bone formation of right lower leg.
Arteries ; Arthritis ; Behcet Syndrome ; Blood Vessels ; Central Nervous System ; Epididymis ; Gastrointestinal Tract ; Humans ; Hyperostosis, Cortical, Congenital ; Hyperparathyroidism ; Joints ; Kidney Failure, Chronic ; Leg ; Lower Extremity ; Male ; Osteogenesis* ; Polyarteritis Nodosa ; Skin ; Stomatitis, Aphthous ; Syphilis, Congenital ; Tuberous Sclerosis ; Ulcer ; Uveitis ; Varicose Veins ; Veins ; Venous Insufficiency ; Vitamin A

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A Case of Abdominal Aortic Aneurysm Treated with Immunosuppressants in Behcet's Disease.

Wan Sik UHM ; Jae Bum JUN ; Yong Wook PARK ; Dae Kook CHANG ; Tae Hwan KIM ; Sung Soo JUNG ; In Hong LEE ; Sang Cheol BAE ; Dae Hyun YOO ; Seong Yoon KIM

The Journal of the Korean Rheumatism Association.2000;7(3):303-307.

Behcet's disease can elicit serious vascular complications such as thrombophlebitis, arterial occlusion, and arterial aneurysm. The most common cause of death in Behcet's disease is secondary to vascular complication. The treatment for abdominal aortic aneurysm which was frequently complicated by fatal rupture (60%) is mainly a surgical management. If surgical treatment is not possible due to various causes, alternative medical therapy such as immunosuppressives and steroid would be recommended. We report here the case of medically controlled abdominal aortic aneurysm in a 47-year-old patient with Behcet's disease who refused to undergo surgical treatment. Immunosuppressive therapy and steroid could be an alternative treatment of aneurysmal manifestations in Behcet's disease.
Aneurysm ; Aortic Aneurysm ; Aortic Aneurysm, Abdominal* ; Cause of Death ; Humans ; Immunosuppressive Agents* ; Middle Aged ; Rupture ; Thrombophlebitis

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Systemic Lupus Erythematosus and Thymoma: A Case Report.

Moo Suk PARK ; Min Chan PARK ; Jung sik SONG ; Yong Beom PARK ; Soo Kon LEE ; Se Hoon KIM ; Woo Ick YANG

The Journal of the Korean Rheumatism Association.2000;7(3):297-302.

Systemic lupus erythematosus (SLE) is an autoimmune disease with a loss of the control over B lymphocytes which is normally exerted by T-suppressor cell. The thymus play a central role in regulating and controlling immune reaction through its effects on T cell differentiation of both T helper and T suppressor cells. Tumors of the thymus are rare but have been reported in concurrence with a variety of autoimmune diseases, including most frequently myasthenia gravis and rarely SLE and other disorders. The association of thymoma and SLE is very rare, fewer than 10 case reports in the world literature. The woman had been thymectomized because of incidental thymoma. After two years, she was diagnosed as SLE with features of arthritis, leukopenia, high titer of ANA, positivity of anti-dsDNA and hypocomplementemia. We report here one case of concurrent SLE and thymoma.
Arthritis ; Autoimmune Diseases ; B-Lymphocytes ; Cell Differentiation ; Female ; Humans ; Leukopenia ; Lupus Erythematosus, Systemic* ; Myasthenia Gravis ; Thymectomy ; Thymoma* ; Thymus Gland

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Secondary Amyloidosis Involving Liver in Rheumatoid Arthritis Treated with Methotrexate and Low dose Prednisolone.

Yoon Ju HAN ; Chan Ho SONG ; Dong Hyuk SHEEN ; Sang Seok YANG ; Jee Youn LEE ; Yong Seong LIM ; Yeong Wook SONG

The Journal of the Korean Rheumatism Association.2000;7(3):291-296.

Amyloidosis is a heterogenous group of often fatal disorders characterized by extracellular deposition of a proteinaceous material with a unique fibrillar form in various tissues and organs. Presenting with severe hepatomegaly, a 46 year-old man who has suffered with rheumatoid arthritis lasting more than 12 years was confirmed to have secondary amyloidosis by liver biopsy. After treatment with methotrexate and low dose prednisolone, we have observed clinical improvement in which hepatomegaly was resolved remarkably. This is the first published case report of a patient with rhuematoid arthritis complicated by liver amyloidosis which partially regressed after treatment with methotrexate and prednisolone.
Amyloidosis* ; Arthritis ; Arthritis, Rheumatoid* ; Biopsy ; Hepatomegaly ; Humans ; Liver* ; Methotrexate* ; Middle Aged ; Prednisolone*

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Facial Palsy as a Presenting Symptom of Wegener's Granulomatosis.

Yun Jong LEE ; Young Joo CHOE ; Eun Bong LEE ; Hyun Ah KIM ; Yeong Wook SONG

The Journal of the Korean Rheumatism Association.2000;7(3):286-290.

Wegener's granulomatosis is a serious multi-systemic disease pathologically characterized by necrotizing vasculitis and granuloma. Although nervous system is one of major target organs in Wegener's granulomatosis, the involvement of facial nerve was rarely reported as an initial symptom. We experienced a female patient with Wegener's granulomatosis presenting otitis media, sinusitis and right facial palsy. Nasal mucosal biopsy revealed necrotizing vasculitis with granuloma formation, which was consistent with Wegener's granulomatosis. During steroid and cyclophosphamide therapy, she developed paralysis of abducens nerve. Methyprednisolone pulse therapy was administrated with symptomatic improvement. We describe a case of Wegener's granulomatosis presenting facial nerve palsy.
Abducens Nerve ; Biopsy ; Cyclophosphamide ; Facial Nerve ; Facial Paralysis* ; Female ; Granuloma ; Humans ; Nervous System ; Otitis Media ; Paralysis ; Sinusitis ; Vasculitis ; Wegener Granulomatosis*

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A Case of Pulmonary Hypertension and Left Vocal Cord Palsy in a Patient with Systemic Lupus Erythematosus.

Do Young KIM ; Jung Hoon SUH ; Shin Myung KANG ; Chang Oh KIM ; Ho Guun NA ; Joong Sun KIM ; Jung sik SONG ; Yong Beom PARK ; Won Ki LEE ; Hong Sik CHOI ; Soo Kon LEE

The Journal of the Korean Rheumatism Association.2000;7(3):280-285.

There have been few case reports on the association of vocal cord palsy and pulmonary hypertension in a systemic lupus erythematosus (SLE) patient. Most cases had left vocal cord palsy caused by compression of left recurrent laryngeal nerve secondary to pulmonary hypertension, and only two cases in the literature were caused by vasculitis or inflammation of the right recurrent laryngeal nerve not related to pulmonary hypertension. Recently, we have experienced a 23-year-old female patient who presented hoarseness and multiple joint pain. She was diagnozed as SLE and left vocal cord palsy, and echocardiographic examination demonstrated enlargement of right atrium, right ventricle, and elevated right ventricular pressure (systolic pressure 47mmHg). She took high-dose glucocorticoid and calcium channel blocker. About one month later, there was significant improvement of hoarseness, arthritis, and cardiomegaly, and showed improvement of the left vocal cord palsy on the laryngoscopic examination. We report a case of left vocal cord palsy and pulmonary hypertension accompanied by SLE who experienced improvement of hoarseness after steroid treatment.
Arthralgia ; Arthritis ; Calcium Channels ; Cardiomegaly ; Echocardiography ; Female ; Heart Atria ; Heart Ventricles ; Hoarseness ; Humans ; Hypertension, Pulmonary* ; Inflammation ; Lupus Erythematosus, Systemic* ; Recurrent Laryngeal Nerve ; Vasculitis ; Ventricular Pressure ; Vocal Cord Paralysis* ; Vocal Cords* ; Young Adult

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A Case with Systemic Lupus Erythematosus Complicated with Multifocal Osteoarticular Tuberculosis.

Seong Ho YOON ; Yong Eun KWON ; Dong Gyu KIM ; Gwang Sik HEO ; Sang Yong KIM ; Hee Kwan KOH ; Seung Myung LEE

The Journal of the Korean Rheumatism Association.2000;7(3):274-279.

Infection is a frequent problem in patients with systemic lupus erythematosus (SLE). Infections contribute greatly to the morbidity of patients and are one of the commonest causes of death. The high frequency and unusual spectrum of infections can be attributed to the multiple disturbances of immune function in SLE in combination with the effects of immunosuppressive therapy. There is increasing evidence to indicate that opportunistic infections including tuberculosis make a large contribution to the infectious mortality in SLE. Tuberculosis is a major cause of morbidity and mortality, particularly in our country where tuberculosis is still endemic. The indolent nature of tuberculous bone and joint disease often leads to delayed or missed diagnosis, sometimes with devastating consequences for the patient. We report a case of multifocal and complicated osteoarticular tuberculosis developing in the spines and knee joint due to delayed diagnosis, with review of literatures.
Cause of Death ; Delayed Diagnosis ; Diagnosis ; Humans ; Joint Diseases ; Knee Joint ; Lupus Erythematosus, Systemic* ; Mortality ; Opportunistic Infections ; Spine ; Tuberculosis ; Tuberculosis, Osteoarticular*

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A Case of Inflammatory Arthritis in the Myelodysplastic Syndrome.

Ji Hyun LEE ; Dae Hyun YOO ; Yoon Ho CHUNG ; Wan Sik UHM ; Tae Hwan KIM ; Jae Bum JUN ; Sung Soo JUNG ; In Hong LEE ; Sang Cheol BAE ; In Soon KIM ; Hee Kwan KOH ; Kyung Bin JOO ; Seong Yoon KIM

The Journal of the Korean Rheumatism Association.2000;7(3):268-273.

The myelodysplastic syndrome (MDS) is a heterogeneous group of refractory anemia resulting from a clonal abnormality in the pleuripotent stem cell. A variety of immunologic abnormalities have been reported in patients with MDS, and various rheumatic diseases, including rheumatoid arthritis, lupus, Sjogren? syndrome, vasculitis, have been reported in association with MDS. Inflammatory arthritis is a rare complication of MDS. Here we report a patient with refractory anemia with excess blasts with unusual manifestation of inflammatory arthritis, with a review of the literature.
Anemia, Refractory ; Arthritis* ; Arthritis, Rheumatoid ; Humans ; Myelodysplastic Syndromes* ; Rheumatic Diseases ; Stem Cells ; Vasculitis

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A Case of Transverse Myelitis as a First Manifestation of Systemic Lupus Erythematosus.

Byung Kwan CHO ; Jeong Soo SONG ; Sung Kwon BAE ; Seung Jae YOON ; Won PARK

The Journal of the Korean Rheumatism Association.2000;7(3):263-267.

This is a case report of systemic lupus erythematosus presented as a transverse myelitis. Central nervous system involvement in systemic lupus erythematosus is a relatively common complication. But the transverse myelitis as a central nervous system involvement in systemic lupus erythematosus is rare and the transverse myelitis as the first manifestation of systemic lupus erythematosus has not been reported in Korea. Thus we report a case of the transverse myelitis as a first manifestation of systemic lupus erythematosus with the literature review.
Central Nervous System ; Korea ; Lupus Erythematosus, Systemic* ; Myelitis, Transverse*

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