Ann Thorac Surg 2005;79:1051-1052
© 2005 The Society of Thoracic Surgeons
Case report
Living-Donor Lobar Lung Transplantation for Bronchiolitis Obliterans After Bone Marrow Transplantation
Yoshifumi Sano, MDa,*,
Hiroshi Date, MDa,
Itaru Nagahiro, MDa,
Motoi Aoe, MDa,
Nobuyoshi Shimizu, MDa
a Department of Cancer and Thoracic Surgery, Okayama University Graduate School of Medicine and Dentistry, Okayama, Japan
Accepted for publication September 10, 2003.
* Address reprint requests to Dr Sano, Department of Cancer and Thoracic Surgery, Okayama University Graduate School of Medicine and Dentistry, 2-5-1 Shikata-cho, Okayama 700-8558, Japan
ysano{at}md.okayama-u.ac.jp
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Abstract
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We report a successful case of living-donor lobar lung transplantation (LDLLT) for severe bronchiolitis obliterans (BO) after bone marrow transplantation (BMT). The patient is a 29-year-old woman who underwent BMT because of aplastic anemia in 1995. In 1996, BO developed in the patient because of chronic graft-versus-host disease. In May 2000, a LDLLT was performed. Pulmonary function tests showed improvement of both vital capacity and forced expiratory volume in 1 second. At present, 91 months after BMT and 38 months after lung transplantation, the patient is in good health. LDLLT may offer a therapeutic option for the treatment of BO after BMT.
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Introduction
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Living-donor lobar lung transplantation (LDLLT) represents a therapeutic option not only for children but also for some adults with severe end-stage pulmonary diseases [1, 2]. The diseases for which LDLLT is indicated have been expanding to include many types of end-stage pulmonary diseases. We report a case in which LDLLT was used successfully to treat severe bronchiolitis obliterans (BO) after bone marrow transplantation (BMT).
The patient is a 29-year-old woman (height: 149 cm, weight: 33.5 kg) who noticed some purpura on her extremities and was diagnosed with aplastic anemia in November 1995. She underwent BMT from an unrelated, HLA-identical and ABO-mismatched (donor: A+, recipient: B+) donor in December 1995. The preparative regimen consisted of cyclophosphamide (50 mg/kg x 4), total body irradiation (4 Gy), and total lymphoid irradiation (6 Gy). The immunosuppressive regimen consisted of cyclosporine, prednisone, and short-term methotrexate. The early posttransplant course was nearly uneventful, with just a grade 1 skin lesion as evidence of an acute episode of graft-versus-host disease (GVHD).
In September 1996, a severe cough developed in the patient and dyspnea upon exertion was suggestive of BO that was due to chronic GVHD. Pulmonary function tests showed severe airflow obstruction (Fig 1). As her cough and dyspnea got more severe, cyclosporine was replaced with tacrolimus in 1998, and home oxygen therapy was started with 1 L/min O2 in 1999. She also suffered several episodes of left spontaneous pneumothorax and underwent bullectomy twice. A chest roentgenogram and a computed tomography scan revealed emphysematous change in her lungs, with pneumothorax of the left lung. In 2000, the patient's pulmonary functions worsened (Fig 1), and she was in respiratory distress with a severe cough and sputum. The patient was totally bedridden and totally dependent on oxygen support. In April 2000, she could not sleep most of the day because of the severe cough.
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Fig 1. Pulmonary function test before and after living-donor lobar lung transplant. (VC = vital capacity (diamonds); FEV1 = forced expiratory volume in 1 second (squares).)
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At the request of the family, LDLLT was performed in May 2000 with a right lower lobe from the patient's brother (27 years old, height: 160 cm, weight: 58.0 kg) with two antigen mismatches of HLA-A and HLA-B loci, and a left lower lobe from her mother (51 years old, height: 150 cm, weight: 41.0 kg) with two mismatches of HLA-A and HLA-B loci. This donor-recipient combination was minor ABO-incompatible (recipient: A+, donors: O+). Immediately after the reperfusion was done, the patient suffered severe pulmonary edema in her right graft and required a differential ventilation with positive end-expiratory pressure of 15 cm H2O in the right and 5 cm H2O in the left lobe, inhalation of nitric oxide (16.0 to 19.7 ppm), and an intravenous infusion of high-dose methylprednisolone. The day after the transplantation, the pulmonary edema and gas exchange improved, and the tracheal tube was removed on postoperative day 4.
The patient was initially treated with triple immunosuppressants consisting of tacrolimus, mycophenolate mofetil, and prednisone, but 8 days after the transplantation, we had to use azathioprine instead of mycophenolate mofetil because she had severe diarrhea. Episodes of acute rejection were diagnosed clinically on postoperative days 6 and 11, and were successfully treated with the administration of pulsed intravenous methylprednisolone. Because of the minor ABO-incompatible transplantation, anti-A-IgG antibody was detected in the patient's serum on day 11 of the transplant, increased to 1:32 on day 26, started to decrease on day 37, and became undetectable on day 72. Her hemoglobin level decreased from 11.6 g/dL to 6.6 g/dL on day 20 with signs of mild hemolysis. The reticulocyte count remained low until day 51, when her hemoglobin level began to increase with reticulocytosis. She required a 2800 mL red blood cell transfusion during this period. A pathologic examination of the explanted lungs confirmed the diagnosis of BO with interstitial pneumonia.
Forty days after the transplantation, she was discharged from the hospital. Pulmonary function tests showed improvement of both vital capacity and forced expiratory volume in one second (Fig 1). At present, 91 months after BMT and 38 months after lung transplantation (LT), the patient is in good health without any signs of acute rejection, infection, or BO. Both of the donors are also in good health and have returned to their full-time jobs.
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Comment
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Chronic GVHD is a systemic, multiorgan syndrome that is a major cause of morbidity and mortality in long-term survivors of allogeneic BMT. The most important risk factor for chronic GVHD is thought to be a prior history of acute GVHD, and so the prevention of acute GVHD is important to decrease the risk of chronic GVHD [3]. Respiratory dysfunction, which includes BO, organizing pneumonia, diffuse alveolar damage, and lymphocytic interstitial pneumonia, is the major cause of death after BMT.
A review of eight cases of LT for BO after BMT shows that seven of these cases involved cadaver donors (three single LT [4–6] and four double LT [6–8]). One case involved a living-donor single lobar LT from a mother to her daughter, who had previously received a BMT from the same donor [9, 10]. The underlying hematologic diseases (including our case) included three cases of aplastic anemia, two of acute lymphoblastic leukemia, and one each of acute myeloid leukemia, chronic myelogenous leukemia, Wiskott-Aldrich syndrome, and an unknown immune defect. In three of these cases, the patient died after LT. The first case, a 27-year-old who underwent a cadaveric single LT, was pathologically diagnosed as having BO, which lead to death 271 days later [5]. In a report of two other cases from The Memorial Sloan-Kettering Cancer Center [6], 1 patient died from progressive respiratory failure secondary to chronic rejection 6 years after LT, and another patient died from infection 3 years after LT. Five other patients were doing well with hematologic remission, without any signs of recurrent BO, from 9 to 72 months after LT. In our case, more than 7 years after BMT and 3 years after LT, the patient is in good health, with good pulmonary function, without any signs of acute rejection, infection, or BO.
We conclude that LDLLT offers a therapeutic option for patients who develop BO after BMT.
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References
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- Starnes VA, Barr ML, Schenkel FA, et al. Experience with living-donor lobar transplantation for indications other than cystic fibrosis. J Thorac Cardiovasc Surg. 1997;114(6):917–921; discussion 921–2[Abstract/Free Full Text]
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Abstract
Introduction
