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Eur J Cardiothorac Surg 2003;23:251-253
© 2003 Elsevier Science NL

Case report

Synchronous bilateral typical pulmonary carcinoid tumors

Division of General Thoracic Surgery, University Hospital, 3010 Berne, Switzerland

Received 3 June 2002; received in revised form 22 October 2002; accepted 28 October 2002.

* Corresponding author. Tel.: +41-31-632-2330; fax: +41-31-632-2327
e-mail: ralph.schmid{at}insel.ch

	Abstract

Top Abstract 1. Introduction 2. Case report 3. Discussion References

 
A 73-year-old woman was admitted with bilateral pulmonary nodules, one on each side, which were suspected to be metastases of an unknown primary tumor. Enucleation of the lesion in the upper right lobe was performed. The histological examination showed a typical carcinoid tumor. A subsequent octreotid scan revealed high suspicion of a carcinoid in the left lower lobe as well. Formal right upper lobectomy and left lower lobectomy were performed in staged procedures. The radiological 1 year control with CT-scan demonstrated no further pathological lesions. To the best of our knowledge this is the first description of curable synchronous pulmonary carcinoid tumors.

Key Words: Pulmonary carcinoid • Typical • Synchronous • Bilateral

	1. Introduction

Top Abstract 1. Introduction 2. Case report 3. Discussion References

 
Carcinoid tumors of the lung, such as of the intestinal tract, are derived from neuroendocrine Kulchitsky cells located within the bronchial or intestinal mucosa. The term Karzinoide was first used by Oberndorfer in 1907 to describe tumors which were histologically similar to a carcinoma but appeared to have a much better prognosis. Carcinoid tumors are uncommon lung malignancies accounting for approximately 2% of all lung neoplasms [1]. These tumors may produce a number of peptides such as serotonin, but in contrary to the intestinal manifestation association of lung carcinoids with a carcinoid syndrome is rare (2.6%). It is well known that typical carcinoid tumors may be cured after surgical resection and have an excellent 10 year patient survival rate of 87% [5]. We report an exceptional case of synchronous bilateral typical pulmonary carcinoid.

	2. Case report

Top Abstract 1. Introduction 2. Case report 3. Discussion References

 
A 73-year-old female was admitted with a diagnosis of lung metastases of unknown origin. She was initially seen by her general practitioner with worsening of a dry cough. The patient was in excellent general condition and regularly engaged in long distance hiking. She had no tobacco addiction. Her past medical history was unremarkable except for asymptomatic cholecystolithiasis and epilepsy treated by Phenytoin. On physical examination there were no pathological findings.

Chest X-ray showed a pulmonary nodule in the right upper lobe. The CT-scan of the chest revealed a second nodule in the left lower lobe near the hilum. Both nodules were 10 mm in diameter (Fig. 1a,b) . Laboratory investigations were normal. Spirometry showed normal age-related lung function tests. Bronchoscopy revealed no pathological findings including negative brush cytology and transbronchial biopsy. Brain CT-scan and skeletal isotope-scan showed no metastases. As the lesion was too deep to be thoracoscopically resected, an open tumor resection was performed in the right upper lobe. The histological examination showed a typical carcinoid. Formal right upper lobectomy with mediastinal lymphadenectomy was consecutively performed a few days later, as enucleation of the tumor on the right side is not oncologically sufficient for the treatment of a carcinoid tumor. No lymph node metastases were detected. The postoperative course was uneventful and the patient was discharged on the 7th day postoperatively. To identify the nature of the nodule in the left lower lobe an octreotid scan was performed which showed positive enhancement close to the left hilus (Fig. 2) .

View larger version (95K): [in this window] [in a new window]   Fig. 1. (a) CT-scan with the tumor in the upper right lobe (arrow). (b) The second tumor in the left lower lobe (arrow).

View larger version (108K): [in this window] [in a new window]   Fig. 2. Octreotid-scan with positive enhancement in the left lower lobe (arrow).

	3. Discussion

Top Abstract 1. Introduction 2. Case report 3. Discussion References

 
Although multiple lung nodules seen on radiological investigations are mainly caused by metastases, systematic evaluation of the histological diagnosis is needed, especially in the absence of an unknown primary localization.

The synchronous occurrence of more than one primary lung tumor of the non-small cell lung cancer type is an unusual event and may carry a bad prognosis, but if both lesions are in an early stage the patient may be cured [2,3]. Therefore, even when both tumors are highly suspect for malignancy, as for example in the FDG-PET, histological diagnosis of both sides should be performed.

Carcinoid tumors do not have specific radiological signs and therefore could be mistaken as benign lesions or multiple metastases. Carcinoids are classically located as single nodules close to the hilus, and rarely they present peripherally and exceptionally multifocal [4]. In most cases the typical carcinoid tumors grow slowly and rarely metastasize (5–10%). Patients with typical carcinoid tumors have an excellent prognosis, with a survival rate at 10 years of up to 87% [5]. They represent one extreme of the whole spectrum of neuroendocrine tumors of the lung, consisting of typical carcinoid, atypical carcinoid, large cell neuroendocrine carcinoma (LCNEC), and small cell lung cancer (SCLC) [6]. Although these entities probably share the same original cell, the prognosis is very different. Tumorlets are aggregations of cells which are similar to typical carcinoid cells but less than 0.5 cm in diameter and they are considered as benign tumors. In our case tumorlets were only found on histological examination, and were not seen either on CT-scan or on octreotid scan perhaps because both were only 2–2.5 mm in diameter. A lesion in the contralateral lung is not considered a as tumorlet.

Synchronous carcinoid tumors seem to be extremely rare and are probably underestimated as the systematic examination of the resected lobes with a confirmed carcinoid tumor shows neuroendocrine cell hyperplasia in three-quarters of patients [7]. This seems to be the first case to be reported with synchronous carcinoid tumors in both lungs which were cured surgically. In the literature one patient with multiple synchronous carcinoid tumors mixed with multiple tumorlets has been reported [8]. This might indicate that genetic factors are involved, but their exact role remains unclear. No consensus exists on how to follow up patients after lobectomy for a typical carcinoid. We propose a CT-scan every year for 5 years and thereafter a chest X-ray yearly to exclude the development of metachronous carcinoid tumors which are reported up to 12 years after the first diagnosis [9].

Typical carcinoid tumors are potentially curable even when they occur synchronously in both lungs. The management of such cases requires careful investigation, planning, and an aggressive treatment strategy.

	Acknowledgments

 
The authors thank Dr M. Gugger, Institute of Pathology and Dr S. Weidner, Division of Nuclear Medicine for their support.

	References

Top Abstract 1. Introduction 2. Case report 3. Discussion References

 

1. Vadesz P., Palffy G., Egervary M., Schaff Z. Diagnosis and treatment of bronchial carcinoid tumours: clinical and pathological review of 120 operated patients. Eur J Cardiothorac Surg 1993;7:8-11.[Abstract]
2. Carey F.A., Donnelly S.C., Walker W.S., Cameron E.W., Lamb D. Synchronous primary lung cancers: prevalence in surgical material and clinical implications. Thorax 1993;48(4):344-346.[Abstract]
3. Martini M.D., Melamed M.R. Multiple primary lung cancers. J Thorac Cardiovasc Surg 1975;70:606-611.[Abstract]
4. Okike N., Bernatz P.E., Woolner L.B. Carcinoid tumours of the lung. Ann Thorac Surg 1976;22:270-277.[Abstract]
5. McCaughan B.C., Martini N., Bains M.S. Bronchial carcinoids: review of 124 cases. Thorac Cardiovasc Surg 1985;89:8-17.
6. Travis W.D., Sobin L.H. Histologic typing of lung and pleural tumours: international histologic classification of tumours. New York: Springer-Verlag, 1999.
7. Miller R.R., Muller N.L. Neuroendocrine cell hyperplasia and obliterative bronchiolitis in patients with peripheral carcinoid tumors. Am J Surg Pathol 1995;19(6):653-658.[Medline]
8. Miller M.A., Mark G.J., Kanarek D. Multiple peripheral pulmonary carcinoids and tumorlets of carcinoid type, with restrictive and obstructive lung disease. Am J Med 1978;65(2):373-378.[CrossRef][Medline]
9. Manoutcheri M.A., Bosse U., Fuzesi L., Klinke F. Multicentric metachronous central carcinoid of the lung: a case report. Thorac Cardiovasc Surg 1999;47(5):337-339.[Medline]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Robert Stein Ralph A. Schmid Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Beshay, M. Articles by Schmid, R. A. Search for Related Content PubMed PubMed Citation Articles by Beshay, M. Articles by Schmid, R. A. Related Collections Lung - cancer