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Fatal Pneumomediastinum and Subcutaneous Emphysema in a Patient with Dermatomyositis.

Jong Hyun PARK ; Sung Hwan PARK ; Paek Sun KIM ; Joon Wook LEE ; Wan Hee YOO ; Yeong Shil JOO ; Shin Seok LEE ; Wan Uk KIM ; Jun Ki MIN ; Yeon Sik HONG ; Sang Heon LEE ; Chul Soo CHO ; Ho Youn KIM

The Journal of the Korean Rheumatism Association.1999;6(3):287-287.

Dermatomyositis is a clinical entity characterized by a distinctive cutaneous rash and inflammatory myopathy. In this disorder, the pneumomediastinum is quite a rare complication and is assumed to result from air leakage due to vasculitis, lung fibrosis or rarely after bronchoscopic lung biopsy and pulmonary function test. We describe patient with dermatomyositis who developed pneumomediastinum, pneumothorax and massive subcutaneous emphysema after pulmonary function test. She died due to respiratory failure. We think that careful observation is required in performing PFT in dermatomyositis patients with presumed interstitial lung diseases.
Biopsy ; Dermatomyositis* ; Exanthema ; Fibrosis ; Humans ; Lung ; Lung Diseases, Interstitial ; Mediastinal Emphysema* ; Myositis ; Pneumothorax ; Respiratory Function Tests ; Respiratory Insufficiency ; Subcutaneous Emphysema* ; Vasculitis

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A Case of Polyarteritis Nodosa Successfully Treated with Deflazacort.

Jong Hyoung CHOI ; Dae Hoon KIM ; Sung Hoon PARK ; Woon Geon SHIN ; Jun Ho LEE ; In Soo KANG

The Journal of the Korean Rheumatism Association.1999;6(3):283-286.

Polyarteritis nodosa(PAN), a systemic vasculitis involving multiple organs including the nervous system, requires a long-term glucocorticoid therapy. Deflazacort is a synthetic glucocorticoid, which has been claimed to have less side effects, but its use for vasculitis has never been reported in the literature. We report a case of polyarteritis nodosa presenting with mononeuritis multiplex causing a left foot drop, which was successfully treated with deflazacort.
Foot ; Mononeuropathies ; Nervous System ; Polyarteritis Nodosa* ; Systemic Vasculitis ; Vasculitis

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Behcet's Disease with Multiple Ulcers in Esophagus and Terminal Ileum: Report of Two Cases.

Hyun Kyu CHANG ; Yeon Suk KIM ; Wan Soo KIM ; Haingsub R CHUNG ; Seung Mun JUNG

The Journal of the Korean Rheumatism Association.1999;6(3):277-282.

Behcet's disease is a chronic polysymptomatic disease of recurrent systemic vasculitis. The etiology remains unclear. This disease affects several organs of the body concurrently or consecutively and sometimes has a serious outcome, depending upon the system involved. Though gastrointestinal symptoms are relatively common in Behcet's disease, ulcerative change of the intestine is infrequent. The most common sites for the intestinal Behcet's disease are terminal ileum and cecum. Only a few literatures report the esophageal ulcers in Behcet's disease. We describe 2 cases of Behcet's disease who have multiple ulcers in esophagus and terminal ileum. Esophageal ulcers have improved with low-dose prednisolone, colchicine and sulfasalazine in these patients.
Cecum ; Colchicine ; Esophagus* ; Humans ; Ileum* ; Intestines ; Prednisolone ; Sulfasalazine ; Systemic Vasculitis ; Ulcer*

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A Case of Acute Lupus Hemophagocytic Syndrome: A Case Report.

Dong Ho OH ; Yong Ho SONG ; Chang Soo KIM ; Jea Hoon KIM ; Joon Hyung DOH ; Byung Reul CHOI ; Sang Gyung KIM ; Sung Guk CHANG ; Jung Yoon CHOE

The Journal of the Korean Rheumatism Association.1999;6(3):272-276.

A 45-year-old woman was admitted to our hospital because of a high fever, dyspnea, and myalgia. At the time of admission, a diagnosis of systemic lupus erythematosus(SLE) was made by fulfilling four of the 1982 American College of Rheumatology criteria with increasing levels of anti-nuclear antibody titer(speckled pattern). Prednisolone given orally in an initial dosage of 30mg/day was not effective and she was expired by respiratory failure due to disseminated intravascular coagulation. A diagnosis of hemophagocytic syndrome was made because of the increased number of unusual hemophagocytic cells in the bone marrow. High levels of serum ferritin which are known to reflect macrophage activition, supported the diagnosis of hemophagocytic syndrome. Hemophagocytic syndrome is characterized by activated phagocytosis presumably induced by hypersecretion of cytokines. Malignant lymphoma and infection are the two representative diseases which may cause hemophagocytic syndrome. Recently several acute lupus hemophagocytic syndromes were reported in patients with SLE. Here we report a case of acute lupus hemophagocytic syndrome observed in a patient with SLE with brief review of literatures.
Bone Marrow ; Cytokines ; Diagnosis ; Disseminated Intravascular Coagulation ; Dyspnea ; Female ; Ferritins ; Fever ; Humans ; Lymphohistiocytosis, Hemophagocytic* ; Lymphoma ; Macrophages ; Middle Aged ; Myalgia ; Phagocytosis ; Prednisolone ; Respiratory Insufficiency ; Rheumatology

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A Case of Cutaneous Panniculitis in Relapsing Polychondritis.

Hyun Chul JUNG ; Jun Hyeop AN ; Sang Heun SONG ; Sung Il KIM ; Ihm Soo KWAK ; Ha Yeon RHA ; Mee Young SOL

The Journal of the Korean Rheumatism Association.1999;6(3):265-271.

Relapsing polychondritis is a rare disease characterized by widespread destructive inflammatory lesions, involving cartilaginous tissue throughout the body. Commonly involved organs include the external ear, nose, joints, eyes, tracheobronchial tree, cardiovascular system and cutaneous tissues. Erythema nodosum or mesenteric panniculitis have sometimes been described in association with relapsing polychondritis, but cutaneous panniculitis is rarely reported in relapsing polychondritis. We report here a relapsing polychondritis patient who developed cutaneous panniculitis, which was resolved by corticosteroid therapy.
Cardiovascular System ; Ear, External ; Erythema Nodosum ; Humans ; Joints ; Nose ; Panniculitis* ; Panniculitis, Peritoneal ; Polychondritis, Relapsing* ; Rare Diseases

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A Case of Remitting Seronegative Symmetrical Synovitis with Pitting Edema(RS3PE) Syndrome.

Mi Jin KIM ; Yong Tai KIM ; Il Hwa JUNG ; Chang Ho SONG ; Chong In LEE ; Young Hak SHIM

The Journal of the Korean Rheumatism Association.1999;6(3):260-264.

RS3PE is used to describe patients who have peripheral seronegative polyarthritis and pitting edema, especially in a man older than 60. It is characterized by sudden onset, high sedimentation rate, lack of bony erosion, remission within 18 months and good prognosis. Whether the RS3PE is a unique disease or syndrome has long been discussed, but conclusion was not obtained. We describe a 72 year old man of RS3PE with a review of the literature.
Aged ; Arthritis ; Edema ; Humans ; Prognosis ; Synovitis*

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Two Cases of Aspergillosis in Systemic Lupus Erythematosus.

Hyun Su KIM ; Yeon Sik HONG ; Sang Su PAE ; Wan Uk KIM ; Jun Ki MIN ; Sang Heon LEE ; Seong Whan PARK ; Chul Soo CHO ; Ho Youn KIM

The Journal of the Korean Rheumatism Association.1999;6(3):253-259.

Invasive infections with Aspergillus species may occur in patients with severe immune deficits and have been described rarely in systemic lupus erythematosus. We present two cases of pulmonary aspergillosis in steroid-treated systemic lupus erythematosus(SLE). Both patients had active SLE treated with high dose corticosteroids and prescribed with broad spectrum antibiotics. One patient had combined infection with pulmonary tuberculosis and the other present granulocytopenia. The diagnosis was delayed because symptoms and radiologic findings were confused with lupus pneumonitis and bacterial infections. This was similar to those reported previously. Diagnosis was confirmed by identification of the typical septated hyphae within tissue. We prescribed high dose amphotericin B to both patients. But one died with sepsis. Aspergillosis should be suspected in patients with active SLE who are immunocompromised and sustain concomitant bacterial infections. More aggressive diagnostic investigation and treatment may be needed to improve poor prognosis.
Adrenal Cortex Hormones ; Agranulocytosis ; Amphotericin B ; Anti-Bacterial Agents ; Aspergillosis* ; Aspergillus ; Bacterial Infections ; Diagnosis ; Humans ; Hyphae ; Lupus Erythematosus, Systemic* ; Pneumonia ; Prognosis ; Pulmonary Aspergillosis ; Sepsis ; Tuberculosis, Pulmonary

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A Case of Intractable Diarrhea Associated with Early Onset Secondary Amyloidosis in Rheumatoid Arthritis.

Sung Min NOH ; Chae Kyu KIM ; Yong Ho SONG ; Ja Hun JUNG ; Hyun Kyu CHANG ; Tae Hwan KIM ; Jae Bum JUN ; Sung Soo JUNG ; In Hong LEE ; Sang Cheol BAE ; Dae Hyun YOO ; Seong Yoon KIM

The Journal of the Korean Rheumatism Association.1999;6(3):247-252.

Amyloidosis is a disease that characterized by accumulation of an amorphous, proteinaceous materials in the various tissues and organs, but its origin is unknown. Recent clinical study showed that incidence of amyloidosis in rheumatoid arthritis was near 10% to 21%. Secondary amyloidosis is caused by accumulation of serum amyloid. A which is doing the acute phase behavior faster than C reactive protein. Symptoms of amyloidosis are various according to the involved organ. Gastrointestinal symptoms are obstruction, ulcer, malabsorption, and bleeding. Diarrhea is very intractable with the conventional antidiarrheal agent, the mechanism of that is infiltration of amyloid material in the intestinal myenteric plexus, sympathetic nerve and ganglion. We report a case of 65 years old woman with rheumatoid arthritis presented with abdominal pain, intractable diarrhea which was not controlled by antidiarrheal agents. Duration of arthritis is only 2 years. Immunohistochemical stain showed AA type which meant secondary form. We treated with Octreotide analogue and total parenteral nutrition for chronic diarrhea.
Abdominal Pain ; Aged ; Amyloid ; Amyloidosis* ; Antidiarrheals ; Arthritis ; Arthritis, Rheumatoid* ; C-Reactive Protein ; Diarrhea* ; Female ; Ganglion Cysts ; Hemorrhage ; Humans ; Incidence ; Myenteric Plexus ; Octreotide ; Parenteral Nutrition, Total ; Ulcer

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Study on the Plasma Homocysteine and Serum Vitamin B12, Folate Levels in Patients with Rheumatoid Arthritis.

Hoon Suk CHA ; Chan Hong JEON ; Jay Hyun KOH ; Chang Keun LEE ; Jin Seok KIM ; Jong Won KIM ; Seong Wook KANG ; Yeong Wook SONG ; Eun Mi KOH

The Journal of the Korean Rheumatism Association.1999;6(3):238-246.

OBJECTIVE: Cardiovascular disease is the most frequent cause of death in rheumatoid arthritis(RA) patients and hyperhomocysteinemia is an independent risk factor for cardiovascular disease. We evaluated the status of homocysteine, vitamin B12 and folate in RA patients and the influence of the drugs used in RA on homocysteine and vitamin levels. METHODS: Fifty-six RA patients and 22 controls were studied. Plasma total homocysteine was measured by HPLC method and serum folate and vitamin B12 were measured by chemiluminescence immunoassay. In RA patients, age, sex, disease duration, medications and laboratory findings were analyzed. RESULTS: Serum vitamin B12 level was significantly lower in RA patients compared to controls(p=0.033). No significant difference in serum folate level was found between RA patients and controls but plasma total homocysteine level was significantly higher in RA patients. There was no difference in plasma total homocysteine level between patients taking MTX with folate and controls, but plasma total homocysteine level was significantly higher in patients not taking MTX compared with controls(p=0.028). In RA patients taking only hydroxychloroquine(HCQ) as a DMARD, there was significantly lower serum folate level(p=0.033) and higher plasma total homocysteine level(p=0.043) compared with controls. There was a significant negative correlation between plasma total homocysteine level and serum folate level in RA patients(r=-0.319, p=0.017). CONCLUSION: Plasma total homocysteine level was increased in RA patients but not in patients taking MTX and folate. These findings suggested that folate supplementation may be effective to prevent hyperhomocysteinemia in RA patients.
Antirheumatic Agents ; Arthritis, Rheumatoid* ; Cardiovascular Diseases ; Cause of Death ; Chromatography, High Pressure Liquid ; Folic Acid* ; Homocysteine* ; Humans ; Hyperhomocysteinemia ; Immunoassay ; Luminescence ; Plasma* ; Risk Factors ; Vitamin B 12* ; Vitamins*

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Coagulation Abnormalities in Behcet's Disease.

Seong Wook KANG ; Ki Chyul SHIN ; Yun Jong LEE ; Eun Bong LEE ; Han Joo BAEK ; Hyun Ah KIM ; Yeong Wook SONG

The Journal of the Korean Rheumatism Association.1999;6(3):227-237.

OBJECTIVE: The predominant histopathologic lesion in Behcet's disease is vasculitis. Thrombotic complications have been reported in approximately 10-40% of patients with Behcet's disease, but the precise mechanisms are not known. To investigate the coagualtion abnormalities in patients with Behcet's disease, coagulation and fibrinolytic activities were examined. METHODS: Thirty-two patients with Behcet's disease and thirty-two healthy individuals as a control group were included in the study. The presence of thrombosis and risk factors for hypercoagulability, and blood components concerning coagulation and fibrinolytic activites were evaluated. RESULTS: Of thirty-two patients with Behcet's disease, thrombosis was found in four patients(13%). No patient had risk factors for hypercoagulability except one with lymphoma. Levels of white blood cell count(mean+/-SD 8,362+/-2,893 vs 5,934+/-1,755/mm2, p<0.001), erythrocyte sedimentation rate(40.5+/-37.6 vs 3.3+/-2.73mm/hr, p<0.001), C reactive protein(2.26+/-3.99 vs 1.20+/-0.26mg/dl, p=0.008), fibrinogen(387.7+/-128.5 vs 240.6+/-49.5mg/dl, p<0.001) and von Willebrand factor antigen(131.9+/-46.6 vs 105.2+/-1.75%, p=0.008) were significantly higher in patients with Behcet's disease compared with controls. The level of fibrinogen correlated with erythrocyte sedimentation rate(r=0.721, p<0.001) and C reactive protein(r=0.454, p=0.018). High density lipoprotein(HDL) cholesterol(46.6+/-12.7 vs 65.5+/-16.1mg/dl, p<0.001), apolipoprotein A-1(118.8+/-24.7 vs 134.6+/-18.5mg/dl, p=0.018) and antithrombin III(92.8+/-16.7 vs 106.3+/-14.7%, p=0.004) were significantly lower in patients with Behcet's disease. No differences were observed in lipoprotein(a), plasminogen, protein C, and protein S activities. Activated protein C(APC) resistance was not observed in any patients with Behcet's disease. Lupus anticoagulant was positive in four patients(13%), one of whom had deep vein thrombosis. Antiphospholipid antibody was found in one patient(3%), but thrombosis was not found. CONCLUSIONS: Significantly higher level of von Willebrand factor antigen was observed in Behcet's disease, which suggested injury of vascular endothelium. Levels of HDL cholesterol, apolipoprotein A-1 and antithrombin III were decreased in Behcet's disease. APC resistance was not found.
Activated Protein C Resistance ; Antibodies, Antiphospholipid ; Antithrombin III ; Apolipoprotein A-I ; Apolipoproteins ; Blood Sedimentation ; Cholesterol, HDL ; Endothelium, Vascular ; Fibrinogen ; Fibrinolysis ; Humans ; Leukocytes ; Lipoprotein(a) ; Lupus Coagulation Inhibitor ; Lymphoma ; Plasminogen ; Protein C ; Protein S ; Risk Factors ; Thrombophilia ; Thrombosis ; Vasculitis ; Venous Thrombosis ; von Willebrand Factor

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