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Ann Thorac Surg 2007;83:1523-1524
© 2007 The Society of Thoracic Surgeons


Case Reports

Rare Pleural Recurrence of Typical Pulmonary Carcinoid Tumor 30 Years After Lobectomy

Askari P. Townshend, BMBS, BMedScia,*, Bhanu Lakshminarayanan, BMBSa, Antonio E. Martin Ucar, FRCSa, Zia R. Chaudry, MRCPathb, John P. Duffy, C-Tha

a Department of Thoracic Surgery, Nottingham City Hospital, Nottingham, United Kingdom
b Department of Histopathology, Nottingham City Hospital, Nottingham, United Kingdom

Accepted for publication November 7, 2006.

* Address correspondence to Dr Townshend, Department of Thoracic Surgery, Nottingham City Hospital, 46 Wimbledon Rd, Nottingham, NG5 1GU UK (Email: askaritownshend{at}hotmail.com).


    Abstract
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A 68-year-old man, who underwent a left lower lobectomy for a typical carcinoid tumor 30 years previously, presented with a 2-year history of left-sided chest pain. A computed tomographic scan, an octreotide scan, and pleural biopsy all showed recurrence of carcinoid tumor in the pleura. He underwent left chest wall resection, and histopathologic examination confirmed the diagnosis of typical carcinoid tumor. A review of the literature indicates that this is only the second reported case of pleural recurrence after previous complete resection of typical pulmonary carcinoid tumor. This case is even more unusual in that the recurrence presented so long after surgery.


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Pulmonary carcinoid tumors are uncommon. Typical carcinoid tumors are less aggressive, and metastasize less frequently than atypical tumors. They carry an excellent prognosis after complete surgical excision. However, recurrence is described in the literature and may occur many years after surgery.

A 68-year-old retired tobacco worker underwent a left lower lobectomy in 1971 for a typical carcinoid tumor of the left lower lobe. The tumor was completely excised and the lymph nodes were negative. No further therapy was given before discharge from follow-up.

The patient presented again in 2005 with a 2-year history of mild left-sided chest pain with no associated symptoms. Past medical history included Parkinson’s disease, myocardial infarction, hypertension, and diet-controlled diabetes mellitus. He had stopped smoking at the time of his original surgery.

A chest roentgenogram showed opacity in the left lower zone (Fig 1), and a computed tomographic scan revealed three lesions (two in the pleura and one in the pericardium) (Fig 2). An octreotide scan confirmed localized lesions suitable for resection. A computed tomographic-guided biopsy of the pleural mass was performed and histologic appearances were consistent with recurrence of typical carcinoid tumor.


Figure 1
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Fig 1. Chest roentgenogram shows left-sided chest wall lesion (arrow).

 

Figure 2
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Fig 2. Computer tomographic scan shows two lesions in the parietal and one in the pericardial pleura (arrows).

 
Surgery was performed through a left posterolateral thoracotomy in the fifth intercostal space. Adhesions from previous surgery were divided and a lobulated pleural mass was excised en-bloc with a portion of the sixth and seventh ribs. A further lesion located on the pericardium was also excised. The defect was covered with Prolene mesh (Ethicon, Somerville, NJ) and soft tissue. The patient made an uncomplicated recovery and was discharged home on postoperative day 6.

Histopathologic examination of the surgical specimen revealed a typical carcinoid tumor. Nests of carcinoid tumor (composed of uniform cells with central vesicular nuclei and granular eosinophilic cytoplasm) were seen in a fibrovascular stroma. Tumor cells were positive for neuroendocrine markers CD56, chromogranins, and synaptophysin, and were patchily positive for Berep 4 epithelial membrane antigen, protein gene product 9.5, and carcinoembryonic antigen were negative.


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Twenty-five percent of carcinoid tumors are pulmonary in origin and comprise as much as 5% of all primary lung tumors [1]. They are grouped as either typical or atypical by histopathologic examination. As much as 90% of tumors are typical [1, 2]. These rarely metastasize; therefore they have a much better prognosis than atypical tumors that metastasize in 40% to 50% of cases.

Computed tomography is a useful technique for localizing bronchial carcinoids, but it can not differentiate between typical and atypical tumors because they have similar imaging features. The 111In-labelled octreotride can be used to detect early recurrence and metastases.

Chemotherapy and radiation therapy have been tried for metastatic disease with little success. If possible, the treatment of choice is complete surgical resection with lymph node dissection [2]. The largest series to date was done by Cardillo and colleagues [2] who reviewed 163 patients during a 12-year span. Excellent survival rates were reported with an overall 5-year survival of over 90% (98.6% for typical and 70.1% for atypical tumors). These findings are consistent with earlier series [3, 4].

Prognosis was related to the stage of the tumor rather than the histologic subtype, but not gender, age, or smoking. This was in agreement with a series of 96 patients by Fiala and colleagues [4], although Filosso and colleagues [5] found that survival was "significantly related to the histological type" as well as pathologic stage in 126 patients.

Recurrence of disease is rare in typical tumors (5.5% [5], 2% [3], 2.6% [4], and <1% [2]), but as much as 10 times more common in atypical tumors. Recurrence in the pleura is extremely unusual, and a comprehensive search of the literature revealed only two case reports of this [6], with one from an atypical tumor (10 years after surgery) and another from a typical tumor (4 years after surgery). As recurrence is possible, although rare, wedge resection may not be effective. Cardillo and colleagues [2] concluded, "Bronchial carcinoid tumors require formal anatomic resection plus radical mediastinal lymphadenectomy either in typical or atypical subtype."

Our case is even more unusual in that the time from resection of the primary lesion to recurrence was 30 years. This suggests that these patients should be followed-up for recurrence for longer than 5 years, which is presently the standard practice.


    References
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  1. Fink G, Krelbaum T, Yellin A, et al. Pulmonary carcinoid: presentation, diagnosis, and outcome in 142 cases in Israel and review of 640 cases from literature Chest 2001;119:1647-1651.[Medline]
  2. Cardillo G, Sera F, Di Martino M, et al. Bronchial carcinoid tumours: nodal status and long-term survival after resection Ann Thorac Surg 2004;77:1781-1785.[Abstract/Free Full Text]
  3. Ferguson MK, Landreneau RJ, Hazelrigg SR, et al. Long-term outcome after resection for bronchial carcinoid tumors Eur J Cardiothorac Surg 2000;18:156-161.[Abstract/Free Full Text]
  4. Fiala P, Petraskova K, Cernohorsky S, Kinkor Z, Krepela E, Zatloukal P. Bronchial carcinoid tumors: long-term outcome after surgery Neoplasma 2003;50:60-65.[Medline]
  5. Filosso PL, Rena O, Donati G, et al. Bronchial carcinoid tumors: surgical management and long-term outcome J Thorac Cardiovasc Surg 2002;123:303-309.[Abstract/Free Full Text]
  6. Bonnette P, Epardeau B, Frachon I, et al. Report of 2 cases of pleural recurrences of surgically treated bronchogenic carcinoidsDiagnostic and therapeutic problems. Rev Mal Respir 1999;16:85-88.[Medline]




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