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J Thorac Cardiovasc Surg 2004;127:584-585
© 2004 The American Association for Thoracic Surgery

Brief communication

Thymic carcinoid combined with myasthenia gravis

Ming-Ho Wu, MDa, Yau-Lin Tseng, MDa,b,*, Fen-Fen Cheng, MDc, Thy-Sheng Lin, MDd

a Department of Surgery, Thoracic Division, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan, Republic of China
b Institute of Clinical Medicine, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan, Republic of China
c Department of Pathology, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan, Republic of China
d Department of Neurology, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan, Taiwan, Taiwan, Republic of China

Received for publication June 4, 2003; accepted for publication September 17, 2003.

* Address for reprints: Yau-Lin Tseng, MD, Department of Surgery, National Cheng Kung University Hospital, No. 138 Sheng-Li Rd, Tainan, Taiwan, Republic of China
tsengyl{at}mail.ncku.edu.tw


Dr Tseng


Thymic carcinoid is a rare cancer and is categorized as one of the families of thymic carcinoma.1-3 Many paraneoplastic syndromes were reported concomitant with thymic carcinoid, including Cushing syndrome, acromegaly, endocrine abnormalities,2,3 and so on. Myasthenia gravis is one of the concomitant paraneoplastic syndromes with thymic neoplasm. Its association with thymic carcinoma, however, has previously only been reported for the cell type of well-differentiated squamous cell carcinoma.4,5 Here we report the case of a patient with thymic carcinoid who underwent en bloc resection of the locally advanced tumor and in whom myasthenia gravis was diagnosed with acute respiratory failure 31 days after surgery.

Clinical summary

A 47-year-old female patient was quite well before an anterior mediastinal mass was found during routine checkup. After thoracic computed tomographic examination (Figure 1), fine-needle aspiration was performed in a local hospital, but without a definitive diagnosis. The patient was then referred to our hospital for further management. During her first admission, physical examination revealed no abnormal neurologic signs. Because of an impression of thymic tumor with local organ invasion, the patient underwent en bloc resection of tumors, including complete thymectomy, tumor excision, partial pericardiectomy, wedge resection of the upper lobe of the left lung, and local mediastinal lymph node dissection. Grossly, two pieces of tumor mass measuring 8 x 6 x 4 cm and 2 x 1.5 x 1 cm were noted. Pathologic reports revealed that it was a thymic carcinoid with cellular atypia (Figure 2). The lung parenchyma was spared, but the subpleural connective tissue and pericardium were invaded. The lymph nodes were free from metastasis. Results of immunohistochemical staining of tumors were positive for synaptophysin, focally positive for neuron- specific enolase, and negative for chromatogranin.



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  		Figure 1. CT scans. A, 2 x 1.5 x 1 cm nodule over right thymus. B, 8 x 6 x 4 cm mass with direct invasion of pericardium and dense adhesion to upper lobe of left lung.

 


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  		Figure 2. Tumor was composed of polygonal cells with finely granular nuclei and moderate eosinophilic cytoplasm. Tumor cells were arranged in solid nests, festoons, and rosettelike pattern. (Hematoxylin and eosin, original magnification x40.)

 
The patient was discharged uneventfully 1 week after the operation. Twenty-four days after discharge, while waiting for radiotherapy, she had dyspnea, orthopnea, dysphagia, and dysphonia. She was sent to the emergency service. Results of edrophonium chloride (Tensilon) and repetitive stimulation tests were positive for a diagnosis of myasthenia gravis. Pulmonary function tests revealed a severe restrictive ventilatory defect. Respiratory failure developed, and the patient underwent endotracheal intubation 2 days after admission, although pyridostigmine was given. Plasma exchange failed because of complicated anaphylactic shock. As an alternative, the patient received intravenous immunoglobulin for 5 days (doses totaling 120 g). She was successfully weaned from the ventilator and extubated 13 days later. Postoperative radiotherapy was then given (total dose 5040 cGy). The patient has been followed up for more than 1 year in the outpatient clinic and is still receiving prednisolone treatment without symptoms or tumor recurrence.

Discussion

Of the 42 patients with thymic carcinoma in our institution from July 1988 to September 2002, 3 had a diagnosis of carcinoid, with 2 of them having no associated paraneoplastic syndrome. Consistent with reports in the literature,4 our previous data indicated that thymic carcinoma with myasthenia gravis only occurred with the cell type of well-differentiated squamous cell carcinoma,5 and the symptoms of myasthenia gravis were always seen before thymic carcinoma was diagnosed.

To our knowledge, thymic carcinoid combined with myasthenia gravis has not been reported before. When a patient with thymic carcinoid demonstrates muscle weakness, no matter whether it develops before or after surgery, myasthenia gravis should be included in the differential diagnosis.

References

  1. Fukai I, Masaoka A, Fujii Y, Yamakawa Y, Yokoyama T, Murase T, et al. Thymic neuroendocrine tumor (thymic carcinoid): a clinicopathologic study in 15 patients. Ann Thorac Surg. 1999;67:208–211[Abstract/Free Full Text]
  2. Moran CA, Suster S. Thymic neuroendocrine carcinoma with combined features ranging from well-differentiated (carcinoid) to small cell carcinoma. Am J Clin Pathol. 2000;113:345–350[Medline]
  3. Jansson JO, Svensson J, Bengtsson BA, Frohman LA, Ahlman H, Wangberg B, et al. Acromegaly and Cushing's syndrome due to ectopic production of GHRH and ACTH by a thymic carcinoid tumor: in vitro responses to GHRH and GHRP-6. Clin Endocrinol. 1998;48:243–250[Medline]
  4. Blumberg D, Burt ME, Bains MS, Downey RJ, Martini N, Rusch V, et al. Thymic carcinoma: current staging does not predict prognosis. J Thorac Cardiovasc Surg. 1998;115:303–309[Abstract/Free Full Text]
  5. Tseng YL, Wang ST, Wu MH, Lin MY, Lai WW, Cheng FF. Thymic carcinoma: involvement of great vessels indicates poor prognosis. Ann Thorac Surg. 2003;76:1041–1045[Abstract/Free Full Text]



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Y.-L. Tseng, J.-M. Chang, I-L. Shu, and M.-H. Wu
Myasthenia gravis developed 30 months after resection of recurrent thymoma.
Eur. J. Cardiothorac. Surg., February 1, 2006; 29(2): 268 - 268.
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