Ann Thorac Surg 2007;83:1196-1197
© 2007 The Society of Thoracic Surgeons
Case Reports
Facial Flushing Due to Recurrent Bronchial Carcinoid
Somshekar Ganti, FRCSa,*,
Richard Milton, FRCSa,
Leslie Davidson, FRCPb,
Andrew Thorpe, FRCSa
a Department of Thoracic Surgery, St. James University Hospital, Leeds
b Department of Histopathology, Leeds General Infirmary, Leeds, United Kingdom
Accepted for publication July 18, 2006.
* Address correspondence to Dr Ganti, Department of Thoracic Surgery, St. James University Hospital, Beckett Street, Leeds LS9 7T, United Kingdom (Email: gantisom{at}hotmail.com).
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Abstract
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Carcinoid syndrome is quite a rare presentation in bronchial carcinoid. A review of the literature suggests a figure of 2% to 7% in various series. This is usually associated with recurrent carcinoid tumor in the presence of hepatic metastasis. We discuss a patient who presented with flushing attacks 13 years after a left pneumonectomy. Further investigation found that the patient had recurrence at the pneumonectomy stump and in subcarinal lymph nodes.
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Introduction
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Bronchial carcinoids represent about 1% to 2% of all pulmonary neoplasms. The respiratory tract is the second most common location for carcinoid tumors after the gastrointestinal tract [1]. Although most bronchial carcinoids behave in an indolent manner, they all have a potential to metastasize and are considered malignant neoplasms [1]. Bronchial carcinoids display a spectrum of clinical behavior and histologic differentiation, ranging from low-grade typical carcinoids through intermediate-grade atypical carcinoids to the high-grade large cell neuroendocrine carcinomas and small cell carcinomas [2]. Bronchial carcinoids can be central or peripheral. Patients with central tumors are usually symptomatic and present with cough, hemoptysis, and recurrent pulmonary infections [3]. Surgery remains the main treatment for typical carcinoids, with excellent long-term survival [4].
A 52-year-old woman presented with hemoptysis. A chest roentgenogram and subsequent computed tomography (CT) scan revealed a suspicious lesion at the division of the left main bronchus. A histologic examination of a specimen obtained through rigid bronchoscopy confirmed typical carcinoid tumour, and she underwent a left thoracotomy and left pneumonectomy in 1993. Microscopic examination of the resected tumour revealed nests of relatively monomorphic epithelioid cells having medium-to-low nuclear cytoplasmic ratios, abundant granular eosinophilic cytoplasm and monomorphic ovoid nuclei with clumped chromatin, and in most cases, inconspicuous single nucleoli. There was no evidence of necrosis, and there were occasional mitotic figures, less than 1 in every 6 mm2 of the sections, confirming it to be a typical carcinoid tumour.
The patient was symptom free for 13 years, at which time she started experiencing severe flushing attacks. Initially, these attacks were put down to the onset of menopause. However, there was no symptomatic relief despite high-dose hormone replacement therapy.
Because of her history, a 24-hour urinary 5-hydroxy indole acetic acid (5HIAA), and plasma serotonin and 5HIAA levels were measured, all of which were within normal limits. Results of an abdominal CT were normal, but a chest CT revealed suspicious lesions around the left main bronchus stump and the aortopulmonary window (Fig 1). These lesions were strongly positive on a subsequent octreotide scan, confirming it to be a recurrence of her carcinoid tumor. She underwent a redo thoracotomy via the pneumonectomy space and removal of recurrent disease (Fig 2). Her postoperative recovery was uneventful, and to date, she remains symptom-free.
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Fig 1. Computed tomography scan of the thorax shows recurrent tumor near the aortopulmonary window. (Hematoxylin and Eosin stain. Magnification is 40x.)
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Fig 2. Histologic specimen of the resected tumor shows monomorphic epithelioid cells with granular eosinophilic cytoplasm in nests and tubules.
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Comment
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According to Warner and colleagues [5], bronchial carcinoids are not able to provoke the syndrome because (1) pulmonary tissue contains large amounts of monoamine oxidase that inhibits the serotonin, (2) the functional capacity of pulmonary carcinoids is much less compared with gastroenteric carcinoids, and they are detected early and excised before they reach a size where they are able to produce a large amount of serotonin, and (3) metastasis to the liver produces the syndrome because the liver is not able to metabolize the large amounts of serotonin produced. In a literature review by Ricci and colleagues [6], 64 cases of bronchial carcinoids presented with the syndrome, of which only 5 patients did not have liver metastasis; however, a satisfactory search for hepatic metastasis was performed in only 1 of these patients.
Although there are a few reports of cases of the carcinoid syndrome in bronchial carcinoid, they were usually present before the excision of the primary tumor. The presence of carcinoid syndrome after successful surgical excision of primary tumor without any evidence of abdominal disease is usually suggestive of nodal recurrence [7], as in our case. This is an interesting case of recurrent bronchial carcinoid presenting with the carcinoid syndrome 13 years after curative surgery in the absence of abdominal disease [8].
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References
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- Buck JL, Sobin LH. Carcinoids of the gastrointestinal tract Radiographics 1990;10:1081-1095.[Abstract]
- Beasley MB, Thunnissen FB, Hasleteon P, et al. Carcinoid tumourIn: Travis WD, Brambilla E, Muller-Hermelink HK, Harris CC, editors. The World Health Organization Classification of Tumors. Pathology and Genetics of Tumors of the Lung, Pleura, Thymus and Heart. Lyon: IARC Press; 2004. pp. 59-62.
- Rosado de Christenson ML, Abbott GF, Kirejczyk WM, Galvin JR, Travis WD. Thoracic carcinoids: radiologic-pathologic correlation Radiographics 1999;19:707-736.[Abstract/Free Full Text]
- Ducrocq X, Thomas P, Massard G, et al. Operative risk and prognostic factors of typical bronchial carcinoid tumors Ann Thorac Surg 1998;65:1410-1414.[Abstract/Free Full Text]
- Warner RR, Kirschner PA, Warner GM. Serotonin production by bronchial adenomas without the carcinoid syndrome JAMA 1961;178:1175-1179.[Abstract/Free Full Text]
- Ricci C, Patrassi N, Massa R, Mineo C, Benedetti-Valentini FJ. Carcinoid syndrome in bronchial adenoma Am J Surg 1973;126:671-677.[Medline]
- Bernstein C, McGoey J, Lertzman M. Recurrent bronchial carcinoid tumor Chest 1989;95:693-694.
- Chen LC, Travis WD, Krug LM. Pulmonary neuroendocrine tumors: what (little) do we know? J Natl Compr Canc Netw 2006;4:623-630.[Medline]
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Abstract
Introduction
