Ann Thorac Surg 2006;82:2265-2266
© 2006 The Society of Thoracic Surgeons
Case Reports
Typical Bronchopulmonary Carcinoid Tumors: A Ramifying Bronchial Presentation With Metastastic Behavior
João-Carlos Das-Neves-Pereira, MD, PhDa,
Leandro Luongo de Matos, MDb,
Claire Danel, MD, PhDa,
Damila Trufelli, MDb,
Marc Riquet, MD, PhDa,*
a Hôpital Européen Georges Pompidou, Thoracic Surgery Department, Paris V University, Paris, France
b Hospital das Clinicas Thoracic Surgery Department, São Paulo University Medical School, São Paulo, Brazil
Accepted for publication May 3, 2006.
* Address correspondence to Dr Riquet, Paris University, Hopital Européen Georges Pompidou, Thoracic Surgery Department, 20-40 rue Leblanc, Cedex 15, 75908 Paris. (Email: marc.riquet{at}hop.egp.ap-hop-paris.fr).
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Abstract
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Bronchopulmonary typical carcinoid tumors (BTCT) are neuroendocrine neoplasms with histologic low grade characteristics considered benign. However, despite reassuring histologic classification, some of them demonstrate an aggressive nature and metastastic behavior. During a not yet concluded study aiming at establishing criteria to predict this metastatic behavior, three uncommon cases were observed. Metastasis occurred despite typical carcinoid microscopic features in 3 female patients of African origin presenting at macroscopic examination as ramifying bronchopulmonary typical carcinoid tumors following the bronchial tree. We suggest that clinical ramifying presentation may be related to metastatic behavior, even for bronchopulmonary typical carcinoid tumors not displaying histologic criteria for atypical carcinoid tumors.
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Introduction
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Since the description in 1906 of a carcinoid tumor by Obberdorfen, the 1999 World Health Organization classification [1], and Travis criteria for neuroendocrine carcinoma [2], bronchopulmonary typical carcinoid tumors (BTCT) are considered as low aggressive and well differentiated carcinomas without metastatic behavior.
Unfortunately, patients operated on for BTCT may have metastases develop, some of them evolving to death [3, 4].
Several authors have tried to define clinical, histologic, immunohistochemical, and molecular biology prognostic criteria, which could help diagnosing the subgroup of BTCT patients who will have metastases develop. Using a logistic regression predictive model [3], we have already studied features related to metastatic potential: male gender, older age, high proliferative rate, Bax/Bcl-2 expression [4], high tumoral microvessel density, low extracellular matrix fiber density, and heparanase expression [5].
During a not yet concluded multicenter Franco–Portuguese–Brazilian study researching further clinical, immunohistochemical, and extracellular matrix characteristics related to metastatic potential, 286 BTCT pathologic samples were revised in two major university hospitals by French and Brazilian pathologists with a large experience in bronchial neuroendocrine carcinomas. Three patients whose tumors had the current histologic criteria for BTCT presented a particular clinical pattern and evolution.
The clinical, surgical, and postoperative data of the patients are shown in Table 1. All three were nonsmoking African females living in the Caribbean Sea area. They had BTCT tumors T2N0M0 (stage IB), but with an uncommon ramified macroscopic presentation following the bronchial tree (Fig 1). Microscopically, all three of these lesions spread along the bronchial submucosa in a ramified pattern from the lobar bronchus up to the subpleural region without any mitosis figure nor necrosis in microscopic analysis (Fig 2). Immunomarkers were not contributive for aggressiveness prediction; however all 3 patients presented metastases during follow-up.
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Fig 2. Microscopic view showing the submucosal spread of typical carcinoid tumor spreading along the bronchial tree (arrows) (magnification, x100).
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Comment
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Nowadays it is well known that even BTCT tumors with a pure histologic pattern may have a metastatic potential develop and sometimes a lethal issue [3]. Morphometric and immunohistochemical biomarkers have been looked for to identify this more aggressive subgroup of typical carcinoids [3–5], but in these three cases the criteria were not predictive of potential metastatic profile. The uncommon branched submucous spread from lobar bronchus up to the subpleural region was the only feature associated with hematogenic metastatic behavior. None of the other 283 patients had such a ramified submucous malignant spread.
Although we describe only three such cases, we suggest being attentive to new cases like these. Effectively, close follow-up using imaging examinations as computed tomography or OctreoScan (Mallinckrodt Medical, Inc, St. Louis, MO) [6], or both, could be offered for all future patients identified with a biologically aggressive tumor, and patients with this specific clinical presentation could also benefit from such a targeted follow-up, perhaps allowing more early and suitable recurrence management.
Our study does not permit determining whether there is any relationship between this particular presentation and the African female origin and geographical area. Perhaps it would be interesting to review data in such populations screening for ramifying BTCT tumors.
Ramified BTCT following the bronchial tree seemed to be an uncommon clinical and pathologic presentation. We suggest that it is a subtype with a metastatic potential, despite typical histologic classification. Further studies must be conducted to clarify the pathology of this entity.
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References
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- Travis WD, Colby TV, Corrin B, et al. Histological typing of lung and pleural tumors, WHO International histological classification of tumor. Berlin: Springer; 1999.
- Travis WD. Pathology of lung cancer Clin Chest Med 2002;23:65-81.[Medline]
- das Neves Pereira JC, Milanez de Campos JR, Capellozi VL, et al. Nuclear and environment morphometric profile in tumor size and nodal metastasis of resected typical carcinoids Pathol Res Pract 2004;200:459-467.[Medline]
- Trufelli D, Matos LL, Das Neves Pereira JC, et al. Bcl 2 family proteins and lymph node metastasis in bronchopulmonary carcinoid tumors Eur J Cancer 2005;3:S341.
- Matos LL, Das Neves Pereira JC, Trufelli D, et al. Heparanase expression in lung carcinoid tumors by immunohistochemistry Eur J Cancer 2005;3:S342.
- Bini A, Grazia M, Stella F, et al. The role of somatostatin receptor scintigraphy (OctreoScan) during follow-up of patients after bronchial carcinoid resection: a prospective study J Cardiovasc Surg (Torino) 2005;46:318-319.[Medline]
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Abstract
Introduction
