Recurrence of Langerhans' cell granulomatosis following lung transplantation

Gabbay, Eli; Dark, John H.; Ashcroft, T; Milne, Duncan; Gibson, G J; Healy, M. J. R.; Corris, Paul A.

doi:10.1136/thx.53.4.326

Cited by 54 publications

(31 citation statements)

References 6 publications

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“…A number of patients with very severe respiratory failure or major pulmonary hypertension have been treated with lung transplantation, with results similar to those found in patients with other patterns of diffuse infiltrating lung disease [73,74]. However, recurrence of the disease in the transplant within the first year has been reported, with possible risk factors being resumption of smoking and extrapulmonary involvement [73][74][75][76][77].…”

Section: Course and Prognosismentioning

confidence: 67%

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Adult pulmonary Langerhans' cell histiocytosis

Tazi¹

2006

European Respiratory Journal

281

297

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Adult pulmonary Langerhans' cell histiocytosis is a rare disorder of unknown aetiology that occurs predominantly in young smokers, with an incidence peak at 20-40 yrs of age. In adults, pulmonary involvement with Langerhans' cell histiocytosis usually occurs as a single-system disease and is characterised by focal Langerhans' cell granulomas infiltrating and destroying distal bronchioles.High-resolution computed tomography (HRCT) of the chest is essential to the diagnosis, typically showing a combination of nodules, cavitated nodules, and thick-and thin-walled cysts. A high macrophage count in bronchoalveolar lavage (BAL) fluid is a common but nonspecific finding that merely reflects exposure to tobacco smoke. BAL is useful for eliminating infections and the other infiltrating lung disorders that can be seen in young adults. Langerhans' cells can be identified in BAL fluid, but, in contrast to what was initially hoped, this test shows a very low sensitivity and is rarely useful in the diagnosis of the disease.The definite diagnosis of pulmonary Langerhans' cell histiocytosis requires identification of Langerhans' cell granulomas, which is usually achieved by surgical lung biopsy at a site selected by chest HRCT. In practice, however, lung biopsy is performed on a case-by-case basis.No effective treatment is available to date, and improved understanding of the mechanisms involved in the pathogenesis of pulmonary Langerhans' cell histiocytosis is urgently needed, and should help in the development of specific therapeutic strategies for patients with this orphan disease.

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Section: Course and Prognosismentioning

confidence: 67%

“…Reports of recurrent disease in lungs transplanted into patients with PLCH are further evidence in favour of patient-related factors, the nature of which remain to be determined [73][74][75][76][77].…”

Section: Smoking and Plchmentioning

confidence: 99%

Adult pulmonary Langerhans' cell histiocytosis

Tazi¹

2006

European Respiratory Journal

281

297

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“…LT may be considered in a subgroup of PLCH patients who experience progressive respiratory impairment related to the extension of cystic lung destruction or to the development of severe pulmonary hypertension. To this date, several hundreds of PLCH patients have probably been transplanted worldwide, but with the exception of some postoperative cases of recurrence of the disease that have been reported postLT (2)(3)(4), little information on LT in PLCH is available. Therefore, we designed a retrospective multicenter study to obtain information on LT in PLCH patients with special emphasis on posttransplant survival and frequency of recurrence of the disease.…”

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confidence: 97%

Lung Transplantation for Pulmonary Langerhans' Cell Histiocytosis: A Multicenter Analysis

et al. 2006

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“…Sarcoidosis, lymphangioleiomyomatosis, eosinophilic granulomatosis, panbronchiolitis, giant-cell interstitial pneumonia, bronchoalveolar cell carcinoma, emphysema, and desquamative interstitial pneumonia are among the diseases with case reports of recurring disease after lung transplantation. [5][6][7][8][9][10][11] Recurrence of veno-occlusive disease after transplantation has not been reported until now, and lung transplantation was thought to be definitive treatment for pulmonary veno-occlusive disease. With this 1st report of early recurrence after heart-lung transplantation, we believe that extrapulmonary factors may play a role in the pathogenesis of this disease.…”

Section: Discussionmentioning

confidence: 99%