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01.12.2011 | Case report | Ausgabe 1/2011 Open Access

Journal of Medical Case Reports 1/2011

A rare case of "switch on and off" multi-system Langerhans cell histiocytosis in an adult patient

Zeitschrift:
Journal of Medical Case Reports > Ausgabe 1/2011
Autoren:
Georgia Karpathiou, Anastasios Koutsopoulos, Marios E Froudarakis
Wichtige Hinweise

Electronic supplementary material

The online version of this article (doi:10.​1186/​1752-1947-5-302) contains supplementary material, which is available to authorized users.

Competing interests

The authors declare that they have no competing interests.

Authors' contributions

Our patient was admitted under the care of MF during this episode and was followed up in an outpatient setting. All authors contributed equally in writing the manuscript. GK wrote the manuscript, AK provided the histological images and MF corrected and refined the manuscript. All authors read and approved the final manuscript.

Abstract

Introduction

We report the case of a 24-year-old Greek woman with histologically proven osseous and pulmonary Langerhans cell histiocytosis whose lesions had progressively regressed with a "switch on and off" mode. This is the first report in the literature of this mode of presentation of Langerhans cell histiocytosis.

Case presentation

The patient had first presented at the age of 20 years with a solitary lesion of the humerus which spontaneously regressed. At that time, no therapy or smoking cessation was indicated. Four years later she presented with bilateral pneumothorax and pulmonary lesions of Langerhans cell histiocytosis. She had pleurodesis for this disease-related complication, and no further systemic treatment was applied, except with regard to smoking cessation. During the follow-up period, her pulmonary lesions regressed without recurrence six years after the initial lung involvement.

Conclusion

This uncommon case of remission of multi-system Langerhans cell histiocytosis indicates the unpredictable evolution of the disease, raising the question of conservative management in such a patient.

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