A Case of Bronchiectasis Accompanied by Ulcerative Colitis (UC) and HTLV-1 Associated Myelopathy (HAM)

We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, ches...

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Published inNihon Kyōbu Shikkan Gakkai zasshi Vol. 32; no. 4; pp. 358 - 363
Main Authors Suzuki, Isamu, Munakata, Mitsuru, Kawakami, Yoshikazu, Watanabe, Naomi, Suzuki, Junichi, Yamaguchi, Etsurou, Fujita, Miri
Format Journal Article
LanguageJapanese
Published Japan The Japanese Respiratory Society 01.04.1994
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ISSN0301-1542
1883-471X
DOI10.11389/jjrs1963.32.358

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Abstract We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, chest X-ray films, tomography and CT-scan revealed dilated lumens and thickened airway walls extending from the trachea to subsegmental bronchi. Pulmonary function tests showed hypoxemia and mixed ventilatory disturbance with a predominantly obstructive component. HAM was diagnosed on the basis of neurological examination and cerebrospinal fluid analysis. A biopsy specimen from the carinal mucosa showed marked T cell infiltration. In these T cells, we detected polyclonal integration of HTLV-1 proviral DNA. Some of the infiltrating T cells showed atypia. In recent times, respiratory diseases other than infiltration of adult T-cell leukemia cells or opportunistic infection have been reported in HTLV-1 carriers and new clinical entities designated as HABA (HTLV-1 associated bronchiolo-alveolar disorder) and HBA (HTLV-1 associated bronchopneumonopathy) have been proposed. This case is classified among these new entities, in a broad sense, and is a rare case in that the respiratory disorder is apparently related to UC.
AbstractList We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, chest X-ray films, tomography and CT-scan revealed dilated lumens and thickened airway walls extending from the trachea to subsegmental bronchi. Pulmonary function tests showed hypoxemia and mixed ventilatory disturbance with a predominantly obstructive component. HAM was diagnosed on the basis of neurological examination and cerebrospinal fluid analysis. A biopsy specimen from the carinal mucosa showed marked T cell infiltration. In these T cells, we detected polyclonal integration of HTLV-1 proviral DNA. Some of the infiltrating T cells showed atypia. In recent times, respiratory diseases other than infiltration of adult T-cell leukemia cells or opportunistic infection have been reported in HTLV-1 carriers and new clinical entities designated as HABA (HTLV-1 associated bronchiolo-alveolar disorder) and HBA (HTLV-1 associated bronchopneumopathy) have been proposed. This case is classified among these new entities, in a broad sense, and is a rare case in that the respiratory disorder is apparently related to UC.We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, chest X-ray films, tomography and CT-scan revealed dilated lumens and thickened airway walls extending from the trachea to subsegmental bronchi. Pulmonary function tests showed hypoxemia and mixed ventilatory disturbance with a predominantly obstructive component. HAM was diagnosed on the basis of neurological examination and cerebrospinal fluid analysis. A biopsy specimen from the carinal mucosa showed marked T cell infiltration. In these T cells, we detected polyclonal integration of HTLV-1 proviral DNA. Some of the infiltrating T cells showed atypia. In recent times, respiratory diseases other than infiltration of adult T-cell leukemia cells or opportunistic infection have been reported in HTLV-1 carriers and new clinical entities designated as HABA (HTLV-1 associated bronchiolo-alveolar disorder) and HBA (HTLV-1 associated bronchopneumopathy) have been proposed. This case is classified among these new entities, in a broad sense, and is a rare case in that the respiratory disorder is apparently related to UC.
We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, chest X-ray films, tomography and CT-scan revealed dilated lumens and thickened airway walls extending from the trachea to subsegmental bronchi. Pulmonary function tests showed hypoxemia and mixed ventilatory disturbance with a predominantly obstructive component. HAM was diagnosed on the basis of neurological examination and cerebrospinal fluid analysis. A biopsy specimen from the carinal mucosa showed marked T cell infiltration. In these T cells, we detected polyclonal integration of HTLV-1 proviral DNA. Some of the infiltrating T cells showed atypia. In recent times, respiratory diseases other than infiltration of adult T-cell leukemia cells or opportunistic infection have been reported in HTLV-1 carriers and new clinical entities designated as HABA (HTLV-1 associated bronchiolo-alveolar disorder) and HBA (HTLV-1 associated bronchopneumopathy) have been proposed. This case is classified among these new entities, in a broad sense, and is a rare case in that the respiratory disorder is apparently related to UC.
We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC at age 20. HTLV-1 was presumably transmitted to this patient by means of a blood transfusion he received at around age 30. On admission, chest X-ray films, tomography and CT-scan revealed dilated lumens and thickened airway walls extending from the trachea to subsegmental bronchi. Pulmonary function tests showed hypoxemia and mixed ventilatory disturbance with a predominantly obstructive component. HAM was diagnosed on the basis of neurological examination and cerebrospinal fluid analysis. A biopsy specimen from the carinal mucosa showed marked T cell infiltration. In these T cells, we detected polyclonal integration of HTLV-1 proviral DNA. Some of the infiltrating T cells showed atypia. In recent times, respiratory diseases other than infiltration of adult T-cell leukemia cells or opportunistic infection have been reported in HTLV-1 carriers and new clinical entities designated as HABA (HTLV-1 associated bronchiolo-alveolar disorder) and HBA (HTLV-1 associated bronchopneumonopathy) have been proposed. This case is classified among these new entities, in a broad sense, and is a rare case in that the respiratory disorder is apparently related to UC.
Author Munakata, Mitsuru
Kawakami, Yoshikazu
Watanabe, Naomi
Fujita, Miri
Suzuki, Isamu
Yamaguchi, Etsurou
Suzuki, Junichi
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References 6) 丸山征郎: HTLV-1 associated bronchopneumonopathy (HAB) とその免疫学的側面. 臨床免疫, 21: 570, 1989.
11) Kraft, S. C., Earle, R. H., Roseler, M. & Esterly, J. R.: Unexplained bronchopulmonary disease with inflammatory bowel disease. Arch. Intern. Med., 136: 454, 1976.
2) Maruyama, I., Chihara, J., Sakashita, I., Mizoguchi, A., Mori, S., Usuku, K., Jonosono, M., Tara, M., Matsumoto, M., Niina, S., Sonoda, S., Yashiki, S. & Osame, M.: HTLV-1 associated broncho-pneumonopathy—A new clinical entity? Am. Rev. Respir. Dis., 137: 4, 46, 1988.
9) Isenberg, J. I., Goldstein, H., Korn, A. R., Ozeran, R. S. & Rosen, V.: Pulmonary vasculitis—An uncommon complications of ulcerative colitis. N. Eng. J. Med., 279: 1376, 1968.
13) Desai, S. J., Gephardt, G. N. & Stoller, J. K.: Diffuse panbronchiolitis preceding ulcerative colitis. Chest, 45: 1342, 1989.
1) 木村郁郎: HTLV-1 関連細気管支・肺胞異常症, HTLV-1 associated bronchiolo-alveolar disorder (HABA). 日胸, 47: 283, 1988.
14) Wilcox, P., Miller, R., Miller, G., Heath, J., Nelems, B., Muller, N. & Ostrow, D.: Airway involvement in ulcerative colitis. Chest, 92: 18, 1987.
4) Yoshioka, R., Yamaguchi, K., Yoshinaga, T. & Takatuki, K.: Pulmonary complications in patients with adult T-cell leukemia. Cancer, 55: 2491, 1985.
5) 木村郁郎: HABA (HTLV-1 Associated bronchiolo-alveolar disorder), Med. Immunol., 18: 769, 1989.
16) Higenbottam, T., Cochrane, G. M., Clark, T. J. H., Turner, D., Millis, R. & Seymour, W.: Bronchial disease in ulcerative colitis. Thorax, 35: 581, 1980.
3) Rogers, B. H. G., Clark, L. M. & Kirsner, J. B.: The epidemiologic and demographic characteristics of a computerized file of 1,400 patients. J. Chron. Dis., 24: 743, 1971.
15) 荒木潤, 増本英男, 須山尚史, 原口増穂, 浅井貞宏, 広瀬清人, 早田宏, 原耕平: 潰瘍性大腸炎に対する全結腸切除施行後に気管・気管支病変を呈した1症例. 気管支学, 14: 64, 1992.
10) Mckee, A. L., Rajapaksa, A., Kalish, P. E. & Pitchmoni, C. S.: Severe interstitial fibrosis in a patient with chronic ulcerative colitis. Am. J. Gastroenterol., 78: 86, 1983.
12) Swinburn, C. R., Jackson, G. J., Cobden, I., Ashcroft, T., Morritt, G. N. & Corris, P. A.: Bronchiolitis obliterans organizing pneumonia in a patient with ulcerative colitis. Thorax, 43: 735, 1988.
7) 杉本峯晴, 安藤正幸, 荒木淑郎: HTLV-1 感染における間質性病変. 呼吸, 11: 238-241, 1992.
8) Osame, M., Janssen, R., Kubota, H., Nishitani, H., Igata, A., Nagataki, S., Mori, M., Goto, I., Shimabukuro, H., Khabbaz, R. & Kaplan, J.: Nationwide survey of HTLV-1 associated myelopathy in Japan. Ann. Neurol., 28: 50, 1990.
References_xml – reference: 15) 荒木潤, 増本英男, 須山尚史, 原口増穂, 浅井貞宏, 広瀬清人, 早田宏, 原耕平: 潰瘍性大腸炎に対する全結腸切除施行後に気管・気管支病変を呈した1症例. 気管支学, 14: 64, 1992.
– reference: 8) Osame, M., Janssen, R., Kubota, H., Nishitani, H., Igata, A., Nagataki, S., Mori, M., Goto, I., Shimabukuro, H., Khabbaz, R. & Kaplan, J.: Nationwide survey of HTLV-1 associated myelopathy in Japan. Ann. Neurol., 28: 50, 1990.
– reference: 10) Mckee, A. L., Rajapaksa, A., Kalish, P. E. & Pitchmoni, C. S.: Severe interstitial fibrosis in a patient with chronic ulcerative colitis. Am. J. Gastroenterol., 78: 86, 1983.
– reference: 5) 木村郁郎: HABA (HTLV-1 Associated bronchiolo-alveolar disorder), Med. Immunol., 18: 769, 1989.
– reference: 9) Isenberg, J. I., Goldstein, H., Korn, A. R., Ozeran, R. S. & Rosen, V.: Pulmonary vasculitis—An uncommon complications of ulcerative colitis. N. Eng. J. Med., 279: 1376, 1968.
– reference: 4) Yoshioka, R., Yamaguchi, K., Yoshinaga, T. & Takatuki, K.: Pulmonary complications in patients with adult T-cell leukemia. Cancer, 55: 2491, 1985.
– reference: 16) Higenbottam, T., Cochrane, G. M., Clark, T. J. H., Turner, D., Millis, R. & Seymour, W.: Bronchial disease in ulcerative colitis. Thorax, 35: 581, 1980.
– reference: 2) Maruyama, I., Chihara, J., Sakashita, I., Mizoguchi, A., Mori, S., Usuku, K., Jonosono, M., Tara, M., Matsumoto, M., Niina, S., Sonoda, S., Yashiki, S. & Osame, M.: HTLV-1 associated broncho-pneumonopathy—A new clinical entity? Am. Rev. Respir. Dis., 137: 4, 46, 1988.
– reference: 11) Kraft, S. C., Earle, R. H., Roseler, M. & Esterly, J. R.: Unexplained bronchopulmonary disease with inflammatory bowel disease. Arch. Intern. Med., 136: 454, 1976.
– reference: 14) Wilcox, P., Miller, R., Miller, G., Heath, J., Nelems, B., Muller, N. & Ostrow, D.: Airway involvement in ulcerative colitis. Chest, 92: 18, 1987.
– reference: 7) 杉本峯晴, 安藤正幸, 荒木淑郎: HTLV-1 感染における間質性病変. 呼吸, 11: 238-241, 1992.
– reference: 13) Desai, S. J., Gephardt, G. N. & Stoller, J. K.: Diffuse panbronchiolitis preceding ulcerative colitis. Chest, 45: 1342, 1989.
– reference: 1) 木村郁郎: HTLV-1 関連細気管支・肺胞異常症, HTLV-1 associated bronchiolo-alveolar disorder (HABA). 日胸, 47: 283, 1988.
– reference: 3) Rogers, B. H. G., Clark, L. M. & Kirsner, J. B.: The epidemiologic and demographic characteristics of a computerized file of 1,400 patients. J. Chron. Dis., 24: 743, 1971.
– reference: 12) Swinburn, C. R., Jackson, G. J., Cobden, I., Ashcroft, T., Morritt, G. N. & Corris, P. A.: Bronchiolitis obliterans organizing pneumonia in a patient with ulcerative colitis. Thorax, 43: 735, 1988.
– reference: 6) 丸山征郎: HTLV-1 associated bronchopneumonopathy (HAB) とその免疫学的側面. 臨床免疫, 21: 570, 1989.
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Snippet We report a case of bronchiectasis with marked thickening of the respiratory tract wall occurring in a 37-year-old man with UC and HAM. He was diagnosed as UC...
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SubjectTerms Adult
Bronchiectasis
Bronchiectasis - etiology
Colitis, Ulcerative - complications
HTLV-1 associated bronchiolo-alveolar disorder
HTLV-1 associated bronchopneumonopathy
HTLV-1 associated myelopathy
Humans
Male
Paraparesis, Tropical Spastic - complications
Ulcerative colitis
Title A Case of Bronchiectasis Accompanied by Ulcerative Colitis (UC) and HTLV-1 Associated Myelopathy (HAM)
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