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(Chest. 1999;115:595-598.)
© 1999 American College of Chest Physicians

Spontaneous Partial Expectoration of an Endobronchial Carcinoid*

William F. Kelly, MD; Eric A. Crawley, MD; Dan J. Vick, MD and Kenneth M. Hurwitz, MD

* From the Department of Internal Medicine (Dr. Kelly), Pulmonary and Critical Care Medicine Service (Drs. Crawley and Hurwitz), and the Department of Pathology and Area Laboratory Service (Dr. Vick), Walter Reed Army Medical Center, Washington, DC and the Uniformed Services University of Health Sciences, Bethesda, MD.


   Abstract
TOP
 Abstract
Introduction
 Case Report
 Discussion
 References
 
Typical pulmonary carcinoid tumors often present as proximal endobronchial masses discovered during the evaluation of cough and/or hemoptysis. We present a case of a carcinoid tumor that presented with spontaneous partial expectoration. A review of the literature revealed 16 cases of expectoration of fragments from various primary and metastatic tumors. Our case appears to be the first report of the expectoration of a carcinoid tumor.

Key Words: bronchial adenoma • carcinoid • expectoration • pulmonary • sputum


   Introduction
TOP
 Abstract
 Introduction
Case Report
 Discussion
 References
 
Expectoration of large tumor fragments appears to be a rare event. Mackenzie described sputum containing tumor fragments in 1886. Only 16 cases of tumor expectoration have been reported since then. Four of them were primary lung lesions: three squamous cell carcinomas and one mixed tumor.2 ,3 ,4 ,5 The remainder were metastatic lesions in patients with renal and colon carcinomas, osteogenic sarcoma, or malignant melanomas4 ,6 ,7 ,8 ,9 ,10 ,11 ,12 ,13 (Table 1 ). A dermoid cyst presenting with hair expectoration14 and an unspecified tumor cast15 also have been reported. No cases of expectorated carcinoid tumors have been described. We report here a case of spontaneous partial expectoration of a carcinoid tumor in a previously healthy patient.


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  		Table 1. Reported Cases of Lung Tumor Expectoration*

 

   Case Report
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 Abstract
 Introduction
 Case Report
Discussion
 References
 
A 25-year-old white male nonsmoker was in excellent health until he experienced a paroxysm of coughing while watching television. This was immediately followed by an episode of hemoptysis, consisting of a "mouthful" of blood and expectoration of a small piece of "tissue" which he saved. The symptoms resolved, so he deferred seeking medical attention until the next day. He reported having a scant hemoptysis 1 year earlier associated with bronchitis, and he described a mild flushing and cramping abdominal discomfort. He denied having a fever, a recent respiratory infection, shortness of breath, a continued cough, wheezing, chest pain, weight loss, anorexia, night sweats, or exposure to tuberculosis. A physical exam and chest radiograph were unremarkable.

The specimen submitted by the patient was a 1.8 x 0.6 x 0.5-cm piece of tan, soft tissue. An initial examination revealed a neuroendocrine neoplasm (Fig 1 ). Immunohistochemical stains were positive for keratin, chromogranins, synaptophysin, and Leu7, supporting the diagnosis of carcinoid tumor. A chest CT scan revealed a soft-tissue mass in the right middle lobe (Fig 2 ). A bronchoscopy demonstrated a smooth, well-demarcated, round vascular mass nearly obstructing the medial segment of the right middle lobe bronchus (Fig 3 ). A biopsy confirmed the previous findings. A right middle lobectomy revealed a localized carcinoid approximately 2.5 x 1.3 x 0.9 cm in size.



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  		Figure 1. Endobronchial carcinoid tumor fragment expectorated by a 25-year-old patient. Shown are nests and trabeculae of tumor cells with scant cytoplasm and round-to-oval nuclei with clumped chromatin and conspicuous small nucleoli (hematoxylin-eosin, original x400).

 


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  		Figure 2. Chest CT: soft tissue mass in the right middle lobe.

 


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  		Figure 3. Fiberoptic bronchoscopy: smooth well-demarcated round mass nearly obstructing the medial segment of the right middle lobe bronchus.

 

   Discussion
TOP
 Abstract
 Introduction
 Case Report
 Discussion
References
 
Bronchial carcinoids are neuroendocrine tumors that, like small cell carcinomas, derive from Kultchitsky cells of the bronchial mucosa and produce serotonin. They represent < 5% of all primary lung cancers.16 The incidence is highest in the fifth decade of life, and there is no gender predominance or known risk factors. Central carcinoids are the most common type, occurring in 50 to 97% of the reviewed case series.17 ,18 ,19 ,20 They generally arise within a major bronchus and present as slow-growing single polypoid masses. Carcinoid syndrome, consisting of flushing, bronchospasm, diarrhea, and valvular heart disease, with increased serotonin secretion in the blood or 5-hydroxyindoleacetic acid in the urine, is found in < 5% of pulmonary carcinoid patients. These are generally patients with atypical carcinoids or liver metastases.21 Even though our patient's symptoms were suggestive of carcinoid syndrome, urine 5-hydroxyindoleacetic acid levels were normal.

The majority of patients with central carcinoids present with coughing, recurrent lung infections, or hemoptysis.18 ,20 Plain film radiographic studies are generally abnormal, with segmental or lobar collapse and coin lesions among the most common findings.18 ,19 In our case, the chest radiograph was unremarkable. However, the chest CT scan was more sensitive, and the bronchoscopic biopsy was diagnostic. A surgical resection was performed, which is the treatment of choice. Radiation and chemotherapy are generally reserved for symptomatic and metastatic disease. The prognosis in this case is excellent, as 5- and 10-year survival rates of > 90% for localized typical carcinoids have been reported.18 ,19

We were unable to find other reports of carcinoids presenting with tumor expectoration. This is surprising given the endobronchial location of the majority of these tumors, their pedunculated and somewhat friable nature, and their association with coughing and hemoptysis. Similarly, tumor expectoration is a rare manifestation of other primary and metastatic lung tumors (Table 1 ). In some of these cases, as in our case, expectoration was the initial presentation of a malignancy. The expectoration was most often spontaneous; but, in two cases, it was induced by bronchoscopy.4 ,8 Three of the four cases of expectorated primary lung tumors reviewed in Table 1 were squamous cell carcinomas. Although this type of tumor represents only one third of primary lung cancers, it is the most common type of central endobronchial cancer. Tumor necrosis also may yield more readily expectorated tissue. The tumors of extrathoracic primary origin were those that tend to metastasize endobronchially, previously described using the mnemonic HAMS—hypernephroma, adenocarcinoma, melanoma, and sarcoma.22 However, essentially all of the cases predated the AIDS epidemic, in which the mnemonic KLAS—Kaposi's, lymphoma, adenocarcinoma, sarcoma—has been suggested.23

Expectoration of small quantities of cells from primary and metastatic lung tumors may lead to a diagnosis on the basis of sputum cytology. However, endobronchial carcinoids generally are covered with intact bronchial mucosa. These tumor cells rarely appear in sputum and bronchial washings unless the bronchial mucosa is ulcerated or disrupted by a biopsy. Bronchial brushings and needle aspirates also break the overlying epithelium and frequently yield cytologic samples.24 The tumor cells occur singly, in sheets, and in clusters, and are rather monomorphic. They have scant cytoplasm and round-to-oval nuclei containing granular clumped chromatin with small but conspicuous nucleoli.25

The cytologic diagnosis of carcinoid tumor is challenging, and the differential diagnosis includes small cell (oat cell) carcinoma, adenocarcinoma, non-Hodgkin's lymphoma, and reserve cell hyperplasia. Oat cell carcinomas exhibit nuclear molding and a basophilic tumor debris, features not found in carcinoids. They also tend to have finer chromatin, as do reserve cells. Small cell adenocarcinomas have vacuolated cytoplasm and macronucleoli. Lymphomas exhibit protruding and indented nuclei. Immunohistochemical stains of cell blocks from cytology specimens also prove helpful in making the diagnosis of carcinoids. These tumors stain positive for neural markers—neuron-specific enolase, neurofilaments, and Leu7—as do oat cell carcinomas. However, they also stain positive for chromogranins and synaptophysin. Small cell adenocarcinomas stain positive for carcinoembryonic antigen, and lymphomas show positive staining with leukocyte common antigen.25

There may be several reasons why tumor expectoration appears to be a rare event. First, patients may fail to notice, acknowledge, or report such events. This may lead them to discard the expectorated tissue. Second, clinicians may ignore such specimens even when they are submitted, presuming them to be phlegm or blood clot. Finally, although such events may indeed occur more frequently, they simply may be underreported in the medical literature or are not retrieved with the search parameters used. We believe our case to be the first reported instance of "carcinoptysis."

It has been suggested that patients at high risk of developing lung tumors should be advised to save any apparent expectorated tissue.4 Clinicians should consider inquiring about such occurrences and review any submitted specimens. These fragments tend to be larger than standard biopsy specimens and may readily lend themselves to diagnostic studies, despite the lack of immediate tissue fixation. If staging studies fail to demonstrate evidence of malignancy, or if psychiatric or other gain is suspected, DNA analysis remains an option to determine "ownership" of the expectorated material.


   Footnotes
 
The authors are U.S. government institution employees. The opinions or assertions contained herein are the private views of the authors and are not to be construed as official or as reflecting the views of any U.S. government agency.

Correspondence to: Dr. William Kelly, Department of Medicine, Walter Reed Army Medical Center, Washington, DC 20307

Received for publication May 29, 1998. Accepted for publication July 20, 1998.


   References
TOP
 Abstract
 Introduction
 Case Report
 Discussion
 References
 

  1. Mackenzie GH. A practical treatise on the sputum. Edinburgh and London: W & AK Johnston; 1886:50–51
  2. Mital, OP, Agarwala, MC (1968) Expectoration of a bronchogenic carcinoma. Br J Dis Chest 62,52-53[Medline]
  3. Goldstein, AR (1976) Expectoration of bronchogenic tumor tissue. JAMA 236,1271[CrossRef][Medline]
  4. Wigton, R, Rohatgi, PK (1978) Expectoration of bronchogenic carcinoma. J Natl Med Assoc 70,799[Medline]
  5. Kern, WH (1976) Expectoration of bronchogenic tumor tissue [letter]. JAMA 236,2604
  6. Jariwalla, AG, Seaton, A, McCormack, RJM, et al (1981) Intrabronchial metastases from renal carcinoma with recurrent tumor expectoration. Thorax 36,179-182[Abstract]
  7. Daryanani, S, Knausenberger, HP, de Takats, PG, et al (1997) Spontaneous pneumothorax associated with expectoration of a lump of metastatic renal cancer. Clin Oncol (R Coll Radiol) 9,262-263[Medline]
  8. Maytum, CK, Vinson, PP (1936) Pulmonary metastasis from hypernephroma, with ulceration into a bronchus simulating primary bronchial carcinoma. Arch Otolaryngol 23,101-104
  9. Gerle, R, Felson, B (1963) Metastatic endobronchial hypernephroma. Dis Chest 44,225-233[Medline]
  10. Seiler, HH, Clagett, OT, McDonald, JR (1950) Pulmonary resection for metastatic malignancy lesions. J Thorac Surg 19,655-679
  11. Sutton, FD, Vestal, RE, Creagh, CE (1974) Varied manifestations of metastatic pulmonary melanoma. Chest 65,415-419[Abstract/Free Full Text]
  12. Rubin EH. The lung as a mirror of systemic disease. Springfield: Charles C. Thomas Publisher; 1956:283–284
  13. Zias, EA, Owen, RP, Borczuk, A, et al (1998) An unusual presentation of metastatic colon cancer to the lung. Chest 113,244-246[Abstract/Free Full Text]
  14. Ustun, MO, Demircan, A, Paksoy, N, et al (1996) A case of intrapulmonary teratoma presenting with hair expectoration. Thorac Cardiovasc Surg 44,271-273[Medline]
  15. Dieter, RA (1974) Unusual expectoration due to a foreign body [letter]. Arch Surg 108,123
  16. Davila, DG, Dunn, WF, Tazelaar, HD, et al (1993) Bronchial carcinoid tumors. Mayo Clin Proc 68,795-803[ISI][Medline]
  17. Harpole, DH, Feldman, JM, Buchanan, S, et al (1992) Bronchial carcinoid tumors: A retrospective analysis of 126 patients. Ann Thorac Surg 54,50-55[Abstract]
  18. Hurt, R, Bates, M (1984) Carcinoid tumors of the bronchus: A 33 years experience. Thorax 39,617-623[Abstract]
  19. Salminen, U-S, Halttunen, PEA, Mattila, SP, et al (1991) Bronchial carcinoid: A clinical follow-up study of 33 cases. Scand J Thorac Cardiovasc Surg 25,189-194[ISI][Medline]
  20. Schreurs, AJM, Westermann, CJJ, van den Bosch, JMM, et al (1992) A twenty-five-year follow-up of ninety-three resected typical carcinoid tumors of the lung. J Thorac Cardiovasc Surg 104,1470-1475[Abstract]
  21. Rosai J. Ackerman's surgical pathology. 7th ed. St. Louis: CV Mosby; 1989:312–317
  22. Braman, SS, Whitcomb, ME (1975) Endobronchial metastasis. Arch Intern Med 135,543-547[Abstract]
  23. Argyros, GJ, Torrington, KG (1994) Fiberoptic bronchoscopy in the evaluation of carcinoma metastatic to the lung. Chest 105,454-457[Abstract/Free Full Text]
  24. Nguyen, GK (1995) Cytopathology of pulmonary carcinoid tumors in sputum and bronchial brushings. Acta Cytol 39,1152-1160[Medline]
  25. Nguyen G-K, Kline TS. Essentials of exfoliative cytology. New York: Igaku Shoin Medical Publishers; 1992:136–142



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