Report of a case: Primary biliary cirrhosis (PBC) associated with polymyositis (PM)
A 58-year-old male suffered from PBC and PM is reported. He was admitted because of liver dysfunction chanced to be found by screening test asymptomatically. AMA and ANA (both homogeneous and speckled types) were positive. ALP and IgM levels were increased. Laparoscopic and histologic findings prove...
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| Published in | Kanzo Vol. 27; no. 6; pp. 822 - 828 |
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| Main Authors | , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
The Japan Society of Hepatology
1986
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| Online Access | Get full text |
| ISSN | 0451-4203 1881-3593 |
| DOI | 10.2957/kanzo.27.822 |
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| Abstract | A 58-year-old male suffered from PBC and PM is reported. He was admitted because of liver dysfunction chanced to be found by screening test asymptomatically. AMA and ANA (both homogeneous and speckled types) were positive. ALP and IgM levels were increased. Laparoscopic and histologic findings proved to be PBC (stage I). Two years after the diagnosis, he was re-admitted on account of muscle weakness and of increased CPK level (1321mU). Muscle biopsy findings were compatible with PM. Oral administration of prednisolone (45 mg/day initially) brought him marked improvement of the symptoms and laboratory data derived from PM. The cases in association with both PBC and PM have been rarely reported. Those literaures are reviewed. Such cases may be present more latently without correct diagnosis. It is important to pay attention to myositic manifestations in PBC patients for early detection of PM association. |
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| AbstractList | A 58-year-old male suffered from PBC and PM is reported. He was admitted because of liver dysfunction chanced to be found by screening test asymptomatically. AMA and ANA (both homogeneous and speckled types) were positive. ALP and IgM levels were increased. Laparoscopic and histologic findings proved to be PBC (stage I). Two years after the diagnosis, he was re-admitted on account of muscle weakness and of increased CPK level (1321mU). Muscle biopsy findings were compatible with PM. Oral administration of prednisolone (45 mg/day initially) brought him marked improvement of the symptoms and laboratory data derived from PM. The cases in association with both PBC and PM have been rarely reported. Those literaures are reviewed. Such cases may be present more latently without correct diagnosis. It is important to pay attention to myositic manifestations in PBC patients for early detection of PM association. |
| Author | YAZAKI, Yasuyuki SATOH, Hitoshi NAMIKI, Masayoshi SUZUKI, Takahisa TAKAHASHI, Atsushi SEKIYA, Chihiro |
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| References | 15) Epstein O, Thomas HC, Sherlock S: Primary biliary cirrhosis is a dry gland syndrome with features of chronic graft-versus-host disease. Lancet I: 1166-1168, 1980 4) Willson RA: Therapeutic quarndary. Asymptomatic primary biliary cirrhosis associated with polymyositis. Dig Dis Sci 26: 372-375, 1981 1) 関谷千尋,矢崎康幸:腹腔鏡所見およびERC所見からみた原発性胆汁性肝硬変の病態とその進展様式.「原発性胆汁性肝硬変」,島田宜浩,粕川禮司編,日本医学館,東京,1985, p93-100 2) Uhl GS, Baldwin LB, Arnett FC: Primary biliary cirrhosis in systemic sclerosis (scleroderma) and polymyositis. Hopkins Med J 135: 191-198, 1974 5) Epstein O, Burroughs AK, Sherlock S: Polymyositis and acute onset systemic sclerosis in a patient with primary biliary cirrhosis: A clinical syndrome similar to the mixed connective tissue disease. J Royal Soc Med 74: 456-458, 1981 12) 西川邦寿,五十嵐良典,武安宣明,他:食道静脈瘤を示す肝硬変症を合併した皮膚筋炎の1症例.Gastroenterol Endosc 26: 2426-2431, 1984 11) 板垣佑司,台一泰,半田洋,他:多発性筋炎を伴ったPBCの1例(会).日消会誌82: 112, 1985 13) 森安隆夫,渡辺進,七宮実,他:膠原病における肝障害について.肝臓22: 422-429, 1981 20) Schraeder PL, Peters HA, Dahl DS: Polymyositis and penicillamine. Arch Neurol 27: 456-457, 1972 14) Thomas HC, Potter BJ, Sherlock S: Is primary biliary cirrhosis an immune complex disease? Lancet II: 1261-1263, 1977 16) Culp KS, Fleming CR, Duffy J,: Autoimmune associations in primary biliary cirrhosis. Mayo Clin Proc 57: 365-370, 1982 9) Fee HJ, Gewirtz H, Schiller J,: Sclerosing cholangitis and primary biliary cirrhosis-A disease spectrum? Ann Surg 186: 589-593, 1977 19) 安倍達,原まさ子,西海正彦:多発性筋炎のステロイド療法に関する研究.厚生省特定疾患強皮症,皮膚筋炎,および多発性筋炎調査研究班,昭和50年度研究業績,p216-221 7) Sherlock S, Scheuer PJ: The presentation and diagnosis of 100 patients with primary biliary cirrhosis. N Engl J Med 289: 674-678, 1973 6) 栗田幸子,松本一仁:多発性筋炎を合併した原発性胆汁性肝硬変の1例.医療,37: 603-607, 1983 10) 佐々木博,井上恭一,樋口清博,他:原発性胆汁性肝硬変の全国統計.肝胆膵4: 171-178, 1982 8) James O, Macklon AF, Watson AJ: Primary biliary cirrhosis-A revised clinical spectrum. Lancet I: 1278-1281, 1981 3) Benoist M, Henin D, Kahn MF,: Cirrhose biliaire primitive associée à une polyarthrite rheumatoïde et à une polymyosite aiguë. Neuv Presse Med 6: 2427-2429, 1977 17) Dawkins RL, Mastaglia FL: Cell-mediated cytotoxicity to muscle in polymyositis. N Engl J Med 288: 434-438, 1973 18) Remade JP, Pellissier JF, Chamlian A,: Progressive ophthalmoplegia associated with asymptomatic primary biliary cirrhosis. Human Pathol 11(Supol): 540-548. 1980 |
| References_xml | – reference: 5) Epstein O, Burroughs AK, Sherlock S: Polymyositis and acute onset systemic sclerosis in a patient with primary biliary cirrhosis: A clinical syndrome similar to the mixed connective tissue disease. J Royal Soc Med 74: 456-458, 1981 – reference: 1) 関谷千尋,矢崎康幸:腹腔鏡所見およびERC所見からみた原発性胆汁性肝硬変の病態とその進展様式.「原発性胆汁性肝硬変」,島田宜浩,粕川禮司編,日本医学館,東京,1985, p93-100 – reference: 3) Benoist M, Henin D, Kahn MF,: Cirrhose biliaire primitive associée à une polyarthrite rheumatoïde et à une polymyosite aiguë. Neuv Presse Med 6: 2427-2429, 1977 – reference: 20) Schraeder PL, Peters HA, Dahl DS: Polymyositis and penicillamine. Arch Neurol 27: 456-457, 1972 – reference: 11) 板垣佑司,台一泰,半田洋,他:多発性筋炎を伴ったPBCの1例(会).日消会誌82: 112, 1985 – reference: 15) Epstein O, Thomas HC, Sherlock S: Primary biliary cirrhosis is a dry gland syndrome with features of chronic graft-versus-host disease. Lancet I: 1166-1168, 1980 – reference: 7) Sherlock S, Scheuer PJ: The presentation and diagnosis of 100 patients with primary biliary cirrhosis. N Engl J Med 289: 674-678, 1973 – reference: 12) 西川邦寿,五十嵐良典,武安宣明,他:食道静脈瘤を示す肝硬変症を合併した皮膚筋炎の1症例.Gastroenterol Endosc 26: 2426-2431, 1984 – reference: 14) Thomas HC, Potter BJ, Sherlock S: Is primary biliary cirrhosis an immune complex disease? Lancet II: 1261-1263, 1977 – reference: 4) Willson RA: Therapeutic quarndary. Asymptomatic primary biliary cirrhosis associated with polymyositis. Dig Dis Sci 26: 372-375, 1981 – reference: 9) Fee HJ, Gewirtz H, Schiller J,: Sclerosing cholangitis and primary biliary cirrhosis-A disease spectrum? Ann Surg 186: 589-593, 1977 – reference: 18) Remade JP, Pellissier JF, Chamlian A,: Progressive ophthalmoplegia associated with asymptomatic primary biliary cirrhosis. Human Pathol 11(Supol): 540-548. 1980 – reference: 2) Uhl GS, Baldwin LB, Arnett FC: Primary biliary cirrhosis in systemic sclerosis (scleroderma) and polymyositis. Hopkins Med J 135: 191-198, 1974 – reference: 13) 森安隆夫,渡辺進,七宮実,他:膠原病における肝障害について.肝臓22: 422-429, 1981 – reference: 17) Dawkins RL, Mastaglia FL: Cell-mediated cytotoxicity to muscle in polymyositis. N Engl J Med 288: 434-438, 1973 – reference: 6) 栗田幸子,松本一仁:多発性筋炎を合併した原発性胆汁性肝硬変の1例.医療,37: 603-607, 1983 – reference: 10) 佐々木博,井上恭一,樋口清博,他:原発性胆汁性肝硬変の全国統計.肝胆膵4: 171-178, 1982 – reference: 16) Culp KS, Fleming CR, Duffy J,: Autoimmune associations in primary biliary cirrhosis. Mayo Clin Proc 57: 365-370, 1982 – reference: 19) 安倍達,原まさ子,西海正彦:多発性筋炎のステロイド療法に関する研究.厚生省特定疾患強皮症,皮膚筋炎,および多発性筋炎調査研究班,昭和50年度研究業績,p216-221 – reference: 8) James O, Macklon AF, Watson AJ: Primary biliary cirrhosis-A revised clinical spectrum. Lancet I: 1278-1281, 1981 |
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| Title | Report of a case: Primary biliary cirrhosis (PBC) associated with polymyositis (PM) |
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