J Korean Med Sci.  2010 May;25(5):658-662. 10.3346/jkms.2010.25.5.658.

Thoracic Air-leak Syndromes In Hematopoietic Stem Cell Transplant Recipients with Graft-versus-Host Disease: A Possible Sign for Poor Response to Treatment and Poor Prognosis

Affiliations
  • 1Department of Thoracic and Cardiovascular Surgery, Seoul St. Mary's Hospital, The Catholic University of Korea, College of Medicine, Seoul, Korea.
  • 2Department of Thoracic and Cardiovascular Surgery, St. Mary's Hospital, The Catholic University of Korea, College of Medicine, Seoul, Korea. sungbo@catholic.ac.kr
  • 3Department of Thoracic and Cardiovascular Surgery, St. Vincent's Hospital, The Catholic University of Korea, College of Medicine, Seoul, Korea.

Abstract

Bronchiolitis obliterans (BO) or bronchiolitis obliterans organizing pneumonia (BOOP) is one of manifestations of graft-versus-host disease (GVHD), a complication of hematopoietic stem cell transplantation (HSCT). Recently there are reports about thoracic air-leakage syndrome (TALS), but real incidence, clinical course, and implications of TALS remain unclear. Retrospective review of 18 TALS patients among 2,177 patients who received allogeneic HSCT between January 2000 to July 2007 was done. Clinical manifestations, treatments, and outcomes of TALS were reviewed. The incidence of TALS was 0.83% (18/2,177). The onset of TALS was mean 425.9+/-417.8 days (60-1,825 days) after HSCT, and the duration was mean 16.3+/-21 days (2-90 days). The most common types of TALS were spontaneous pneumothroax (n=14), followed by subcutaneous emphysema (n=6), pneumomediastinum (n=5), interstitial emphysema (n=2), and pneumopericardium (n=1). TALS persisted in six patients, who died during the same hospitalization. The 12 patients recovered from TALS, but only 2 survived, while others died due to aggravation of GVHD. TALS may complicate BO/BOOP and be an initial manifestation of BO/BOOP. TALS is hard to be resolved, and even after the recovery, patients die because of aggravation of GVHD. We suggest specifically in HSCT patients, when once developed, TALS seems hard to be cured, and as a result, be related to high fatality.

Keyword

Pleural Diseases; Bronchiolitis Obliterans; Hematopoietic Stem Cell Transplantation; Graft vs Host Disease

MeSH Terms

Adolescent
Adult
Comorbidity
Female
Graft vs Host Disease/*mortality/*surgery
Hematopoietic Stem Cell Transplantation/*mortality
Hemothorax/*mortality
Humans
Incidence
Korea
Male
Middle Aged
Pneumothorax/*mortality
Prognosis
Risk Assessment
Risk Factors
Survival Analysis
Survival Rate
Syndrome
Treatment Outcome
Young Adult

Reference

1. Kotloff RM, Ahya VN, Crawford SW. Pulmonary complications of solid organ and hematopoietic stem cell transplantation. Am J Respir Crit Care Med. 2004. 170:22–48.
Article
2. Dudek AJ, Mahaseth H, Defor TE, Weisdorf DJ. Bronchiolitis obliterans in chronic graft-versus-host disease: Analysis of risk factors and treatment. Biol Blood Marrow Transplant. 2003. 9:657–666.
3. Franquet T, Giemenez A, Torrubia S, Sabate JM, Rodriguez-Arias JM. Spontaneous pneumothorax and pneumomediastinum in IPF. Eur Radiol. 1999. 108–113.
Article
4. Galanis E, Litzow MR, Tefferi A, Scott JP. Spontaneous pneumomediastinum in patient with bronchiolitis obliterans after bone marrow transplantation. Bone Marrow Transplant. 1997. 20:695–696.
5. Taouli B, Cadi M, Leblond V, Grenier PA. Invasive aspergillosis of the mediastinum and left hilum: CT features. AJR Am J Roentgenol. 2004. 183:1224–1226.
Article
6. Toubai T, Tanaka J, Kobayashi N, Honda T, Miura Y, Ogawa T, Imai K, Ogasawara M, Kiyama Y, Higa T, Imamura M, Kasai M. Mediastinal emphysema and bilateral pneumothoraces with chronic GVHD in patients after allogeneic stem cell transplantation. Bone Marrow Transplant. 2004. 33:1159–1163.
Article
7. Shulman HM, Sale GE, Lerner KG, Barker EA, Weiden PL, Sullivan K, Gallucci B, Thomas ED, Storb R. Chronic cutaneous graftversus-host disease in man. Am J Pathol. 1978. 91:545–570.
8. Schubert MM, Sullivan KM, Morton TH, Izutsu KT, Peterson DE, Flournoy N, Truelove EL, Sale GE, Buckner CD, Storb R. Oral manifestations of chronic graft-v-host disease. Arch Intern Med. 1984. 144:1591–1595.
Article
9. McDonald GB, Shulman HM, Sullivan KM, Spencer GD. Intestinal and hepatic complications of human bone marrow transplantation. Part II. Gastroenterology. 1986. 90:770–784.
Article
10. Vogel M, Brodoefel H, Bethge W, Faul C, Hartmann J, Schimmel H, Wehrmann M, Claussen CD, Horger M. Spontaneous thoracic air-leakage syndrome in patients following allogeneic hematopoietic stem cell transplantation: Causes, CT-follow up and patient outcome. Eur J Radiol. 2006. 60:392–397.
Article
11. Ratanatharathorn V, Ayash L, Lazarus HM, Fu J, Uberti JP. Chronic graft versus host disease: clinical manifestation and therapy. Bone Marrow Transplant. 2001. 28:121–129.
12. Franquet T, Rodríguez S, Hernandez JM, Martino R, Gimenez A, Hidalgo A, Domingo P. Air-leak syndromes in hematopoietic stem cell transplant recipients with chronic GVHD. High resolution CT findings. J Thorac Imaging. 2007. 22:335–340.
13. Roca J, Grañeña A, Rodriguez-Roisin R, Alvarez P, Agusti-Vidal A, Rozman C. Fatal airway disease in an adult with chronic graft-versus-host disease. Thorax. 1982. 37:77–78.
Article
14. Yamanoha A, Nagasaki A, Nakachi S, Kinjo S, Takasu N. Air-leak syndromes associated with bronchiolitis obliterans after allogeneic peripheral blood stem cell transplantation. Int J Hematol. 2007. 85:95–96.
15. Tanvetyanon T, Toor AA, Stiff PJ. Post-transplant air-leak syndrome. Br J Haematol. 2004. 126:758.
Article
16. Chadwick C, Marven SM, Vora AJ. Autologous blood pleurodesis for pneumothorax complicating graft-versus-host disease related bronchiolitis obliterans. Bone Marrow Transplant. 2004. 33:451–453.
17. Trisolini R, Bandini G, Stanzani M, Chilosi M, Cancellieri A, Boaron M, Poletti V. Morphologic changes leading to bronchiolitis obliterans in a patient with delayed non-infectious lung disease after allogeneic bone marrow transplantation. Bone Marrow Transplant. 2001. 28:1167–1170.
Article
18. Wintermark M, Schnyder P. The macklin effect - a frequent etiology for pneumomediastinum in severe blunt trauma. Chest. 2001. 120:543–547.
19. Chakravorty I, Oldfield W, Gómez C. Rapidly progressive Bronchiolitis Obliterans Organising Pneumonia presenting with pneumothorax, persistent air leak, acute respiratory distress syndrome and multi-organ dysfunction: a case report. J Med Case Reports. 2008. 2:I45.
Article
20. Philit F, Wiesendanger T, Archimbaud E, Mornex JF, Brune J, Cordier JF. Post-transplant obstructive lung disease ("bronchiolitis oblieterans"): a clinical comparative study of bone marrow and lung transpla patients. Eur Respir J. 1995. 8:551–558.
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