With survival after retransplant improving over the years, issues regarding multiple retransplant have emerged. Here, we report the 25-year follow-up of a 16-year-old male patient who received 4 successful lung transplant procedures in 1990, 1991, 1995, and 2005

Kessler, Romain

Massard, Gilbert

SecoAntunes, Ana Rita

Zhang, Min

English

peer reviewedWith survival after retransplant improving over the years, issues regarding multiple retransplant have emerged. Here, we report the 25-year follow-up of a 16-year-old male patient who received 4 successful lung transplant procedures in 1990, 1991, 1995, and 2005

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AbstractWith survival after retransplant improving over theyears, issues regarding multiple retransplant haveemerged. Here, we report the 25-year follow-up of a16-year-old male patient who received 4 successfullung transplant procedures in 1990, 1991, 1995, and2005.Key words: Chronic graft dysfunction, Cystic fibrosis,RetransplantIntroductionThe main long-term complication after lung trans -plant is chronic graft dysfunction. Lung retransplantis presently the only curative strategy in themanagement of chronic rejection, provided that thepatient appears fit for this procedure. With thegrowing volume of primary lung transplants, thedemand for lung retransplant has also becomeincreasingly common over the past decade.1-3 Themain indications for lung retransplant include airwaycomplications, acute graft failure, and chronic graftdysfunction. However, lung retransplant for acutegraft failure and early airway complications havedramatically decreased during the past decadebecause of poor reported outcomes. In contrast, lungretransplant for chronic allograft dysfunction hascontinued as survival rates approach those obtainedafter first lung transplant. Several studies have shownthat long-term survival rates after lung retransplantfor chronic rejection have approached the survivalrates of primary lung transplant. As survival afterretransplant becomes more prolonged, complicationsafter multiple lung retransplants emerge.Here, we present a 25-year follow-up of a malepatient with cystic fibrosis who underwent a total of 4successive lung transplant procedures in 1990, 1991,1995, and 2005. To our knowledge, this is one of thefirst cases of reported multiple lung retransp lants.Case ReportThe study patient was diagnosed with cystic fibrosis at6 years old. He was admitted to the intensive care unitwith end-stage disease at age 16 years and was put onthe wait list for lung transplant. After 22 days withartificial ventilation through a tracheotomy, suitabledonor lungs became available and he underwent abilateral lung transplant according to the en bloctechnique with bilateral bronchial anastomoses. Theprocedure, performed in March 1990, showed aninitially favorable clinical outcome. However, severalmonths after transplant, lung function had dete -riorated. Biopsy findings confirmed acute rejection.After failure of steroid bolus therapy, muromonab-CD3 (OKT3, Johnson & Johnson, Highland Park, NJ,USA) was administered without further success. Thepatient was transferred to the intensive care unit andput under artificial ventilation. Because there was anirreversible alteration of graft function, he was againplaced on the emergency transplant list.In January 1991, after 63 days of artificial ven -tilation, a heart-lung transplant was performed. Hisrespiratory function gradually improved until 1993,when a pulmonary biopsy showed grade 1 rejectionassociated with hemosiderosis.In September 1995, he received a left lung trans -plant without cardiopulmonary bypass, whichresulted in a satisfactory outcome posttransplant. He was kept on the standard immunosuppressive Copyright © Başkent University 2019 Printed in Turkey. All Rights Reserved.Multiple Lung Transplant in a Patient Within 25 Years: A Case ReportMin Zhang,1 Ana Rita SecoAntunes,2 Romain Kessler,3 Gilbert Massard4From the 1Department of Cardiothoracic Surgery, the First Affiliated Hospital of ChongqingMedical University, Chongqing, China; the 2Faculty of Medicine, University of Strasbourg,Strasbourg, France; and the 3Department of Respiratory Medicine and the 4Service de ChirurgieThoracique, Hôpitaux Universitaires de Strasbourg, Strasbourg, FranceAcknowledgements: The authors have no sources of funding for this study and have no conflictsof interest to declare.Corresponding author: Gilbert Massard, Service de Chirurgie Thoracique, HôpitauxUniversitaires de Strasbourg, 1, Place de l’hôpital, 67091 Strasbourg, FrancePhone: +33 607 276 108                   E-mail: gilbert.massard@chru-strasbourg.frExperimental and Clinical Transplantation (2019) Case RepORtDOI: 10.6002/ect.2018.0340regi men of the mid-1990s (combined methylpre -dnisolone, cyclosporine, and azathioprine). In March2004, the patient’s situation again started to deteriorate.Chest computed tomography scan findings weresuggestive of right-sided constrictive bronchiolitis andleft-sided bronchiolitis obliterans. Bronchoalveolarlavage revealed infection with methicillin-resistantcoagulase-negative Staphylococcus species. Despitetreatment with vancomycin and pristinamycin,respiratory function deterioration progressedrelentlessly.In October 2005, the patient underwent right lungretransplant without extracorporeal circulation, withextubation 2 days after surgery. One month later, thepatient was hospitalized for infectious bronchitis.Biology revealed coagulase-negative Staphylococcusand Enterococcus infections, which were sensitive to amoxicillin. The patient responded well to theantibiotics and was discharged.In January 2006, the patient presented with pro -ductive cough with sputum. Microbiologic testshowed relapsing Enterococcus and Staphylococcusinfections. An acute transplant rejection due tosuperinfection was suspected, and treatment withantilymphocyte antibody (OKT3) was provided. Thenext 7 years were uneventful. However, in October2013, the patient had a splenectomy for spontaneousrupture of the spleen, leading to the discovery of aposttransplant Epstein-Barr virus-related lymphoma.The latter responded well to 4 cycles of R-CHOP(cyclophosphamide, doxorubicin, vincristine, pred -nisone, plus rituximab). In March 2014, the patientpresented with relapse of Epstein-Barr viruslymphoma. After 4 shots of rituximab, second-linechemotherapy with ifosfamide-etoposide and ofatu -mumab was started. Although the patient had apartial response after the first 2 cycles, performancestatus declined to a World Health Organization 3/4category. The 3rd and 4th cycles of treatment werepoorly tolerated. After several episodes of septicshock, the patient ultimately died in July 2014.DiscussionWith survival of retransplant improving over theyears, issues with regard to multiple retransplantshave emerged. Lung retransplant comprises anincreasing share of lung transplants, and recentlypatients have shown improved outcomes. Themedian survival of patients who undergo lungretransplant has been shown to be 2 years.2 Mediansurvival after lung retransplant for bronchiolitisobliterans syndrome is longer than survival afterretransplant for restrictive chronic lung allograftdysfunction. In a study from Thomas and colleagues,1-year survival was only 50% after lung retransplantfor acute graft failure and airway complications;however, lung retransplant for bronchiolitis obli -terans syndrome showed superior survival at 1 year(78%) and at 5 years (62%). These latter rates aresimilar to survival rates for first-time lung transplantrecipients. The authors concluded that lung retrans -plant may be appropriate in well-selected patientswith chronic lung allograft failure.3Presently, there are no reports about outcomes of multiple lung retransplant procedures. There are many unanswered questions regarding ethics,indication, immunology, and graft tolerance, management of immunosuppression, and long-termoutcomes. Our reported case demonstrates thatprolonged survival is possible: this particular patientunderwent 4 transplants over a 25-year period. Twoof these transplants were followed by survival ofalmost 10 years each.References1. Kawut SM. Lung retransplantation. Clin Chest Med. 2011;32(2):367-377.2. Novick RJ, Stitt LW, Al-Kattan K, et al. Pulmonary retransplantation:predictors of graft function and survival in 230 patients.Pulmonary Retransplant Registry. Ann Thorac Surg. 1998;65(1):227-234.3. Thomas M, Belli EV, Rawal B, Agnew RC, Landolfo KP. Survival afterlung retransplantation in the United States in the current era (2004to 2013): better or worse? Ann Thorac Surg. 2015;100(2):452-457.2 Min Zhang et al/Experimental and Clinical Transplantation (2019) Exp Clin TransplantQ

Multiple Lung Transplant in a Patient Within 25 Years: A Case Report

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Multiple Lung Transplant in a Patient Within 25 Years: A Case Report

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