CASE REPORTS   

Minerva Pneumologica 2012 December;51(4):163-8

Copyright © 2012 EDIZIONI MINERVA MEDICA

language: English

Intrathoracic hyaline-vascular Castleman’s disease: three different presentations and management

Zairi S. ¹, Marghli A. ¹, Smadhi H. ¹, Ayadi A. ², Zribi H. ¹, Boudaya M. S. ¹, El Mezni F. ², Kilani T. ¹

¹ Department of Thoracic Surgery. Abderrahmen Mami Hospital, Ariana, Medical School of Tunis, University of Tunis El Manar, Tunisia; ² Department of Pathology, Abderrahmen Mami Hospital, Ariana, Medical School of Tunis, University of Tunis El Manar, Tunisia

The unicentric form of Castleman’s disease with its hyaline-vascular type is a rare benign entity. Symptoms are not specific and result in diagnostic delays. A surgical approach is often necessary for diagnosis and complete excision is the best treatment. However, in some cases, surgical resection raises problems. We report three different presentations and management of the unicentric form of Castleman’s disease. The first patient was a 33-year-old woman with a history of chest pain. Thoracic imaging showed a right superior and anterior mediastinal opacity. The patient underwent a complete and difficult surgical resection by a right posterolateral thoracotomy. The second patient, a 29-year-old man, had a history of dry cough and an erosive oral lichen planus. A CT-scan showed compressive middle mediastinal mass. The patient underwent surgical resection with a favorable outcome and complete recovery from the oral lichen planus. The third case was a 24-year-old woman with a history of asthmatic dyspnea. Thoracic imaging revealed a right hilar opacity. Diagnostic and therapeutic thoracotomy was performed. A complete resection of the tumor required a pneumonectomy. Our three cases illustrate the variety of presentations and surgical management of the hyaline-vascular type of Castleman’s disease, which although benign, requires in some cases a major resection. Erosive oral lichen planus associated in one case does not seem coincidental.