Bucillamine is a disease modifying anti-rheumatic drug, structurally similar to D-penicillamine. Although D-penicillamine-induced pemphigus has been not infrequently demonstrated, pemphigus associated with bucillamine was rarely reported. We describe a patient complicating pemphigus vulgaris after bucillamine treatment in rheumatoid arthritis (RA) and polymyositis (PM) overlap syndrome. PM and RA overlap syndrome was diagnosed three years ago and bucillamine was administrated for 20 months. Skin lesions including erythematous flaccid blisters on her chest, axillae, and back were occurred and were compatible with pemphigus vulgaris by typical pathology. Withdrawal from bucillamine and prednisolone treatment made rapid improvement of pemphigus lesions.
Syndrome ; Skin/pathology ; Polymyositis/*complications/*drug therapy ; Pemphigus/*chemically induced/*pathology ; Middle Aged ; Humans ; Female ; Cysteine/adverse effects/*analogs & derivatives ; Biopsy ; Arthritis, Rheumatoid/*complications/*drug therapy ; Arthritis ; Antioxidants/*adverse effects

Pneumatosis cystoides intestinalis (PCI), characterized by presence of intramural gas cyst in the intestinal wall is associated with various medical condition. Polymyosistis, however, is rarely associated with PCI. Few cases are reported in the world, and none has not been reported previously in Korea. A 67-year-old woman with polymyositis developed mild abdominal pain and abdominal distension during treatment with steroid and azathioprine. Radiographic findings including CT scan showed intraperitoneal free gas and intramural air, compatible with PCI. The patient's symptom and clinical findings improved after the treatment with antibiotics and high-dose oxygen therapy.
Aged ; Anti-Bacterial Agents/therapeutic use ; Anti-Inflammatory Agents/therapeutic use ; Azathioprine/therapeutic use ; Cefotaxime/therapeutic use ; Female ; Humans ; Oxygen Inhalation Therapy ; Pneumatosis Cystoides Intestinalis/complications/*diagnosis/drug therapy ; Polymyositis/complications/*diagnosis/drug therapy ; Prednisolone/therapeutic use ; Radiography, Abdominal ; Tomography, X-Ray Computed

Pneumatosis cystoides intestinalis (PCI), characterized by presence of intramural gas cyst in the intestinal wall is associated with various medical condition. Polymyosistis, however, is rarely associated with PCI. Few cases are reported in the world, and none has not been reported previously in Korea. A 67-year-old woman with polymyositis developed mild abdominal pain and abdominal distension during treatment with steroid and azathioprine. Radiographic findings including CT scan showed intraperitoneal free gas and intramural air, compatible with PCI. The patient's symptom and clinical findings improved after the treatment with antibiotics and high-dose oxygen therapy.
Aged ; Anti-Bacterial Agents/therapeutic use ; Anti-Inflammatory Agents/therapeutic use ; Azathioprine/therapeutic use ; Cefotaxime/therapeutic use ; Female ; Humans ; Oxygen Inhalation Therapy ; Pneumatosis Cystoides Intestinalis/complications/*diagnosis/drug therapy ; Polymyositis/complications/*diagnosis/drug therapy ; Prednisolone/therapeutic use ; Radiography, Abdominal ; Tomography, X-Ray Computed

Polymyositis is a rare complication of interferon alpha treatment as a result of immunemodulating role of the drug itself. In this case, interferon alpha induced polymyositis and cardiomyopathy is diagnosed in a 33-yr-old male patient with history of chronic hepatitis B. To treat hepatitis B, interferon alpha was administered until the proximal muscle weakness developed. Thereafter, sixteen cycles of immunoglobulin treatment (400 mg/kg) along with corticosteroids were instituted and led to an improvement in subjective symptoms with decreases in level of CPK and LDH. However, dilated cardiomyopathy has not improved in spite of the cessation of interferon treatment. Unlike the persistently elevated serum HBV DNA level, the serum ALT and AST levels have gradually decreased. Our case shows that clinical symptoms of polymyositis improved with steroid and immunoglobulin treatment without deterioration of the hepatitis B. To our knowledge, this is the first case of polymyositis associated with dilated cardiomyopathy after the administration of interferon in a patient with hepatitis B.
Adrenal Cortex Hormones/therapeutic use ; Adult ; Antigens, CD13/blood ; Antiviral Agents/*adverse effects/therapeutic use ; Aspartate Aminotransferases/blood ; Cardiomyopathy, Dilated/blood/*chemically induced/drug therapy/physiopathology ; Hepatitis B, Chronic/blood/complications/*drug therapy ; Humans ; Immunoglobulins, Intravenous/therapeutic use ; Interferon-alpha/*adverse effects/therapeutic use ; Male ; Polymyositis/blood/*chemically induced/drug therapy/physiopathology ; Treatment Outcome