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Eur J Cardiothorac Surg 2003;23:134-135
© 2003 Elsevier Science NL


Case report

Recurrence of stage I thymoma in sternum, 13 years after ‘complete’ excision

Rajashekara H.V. Reddy, Rajesh Shah, B. Kumar, James A.C. Thorpe*

Department of Cardiothoracic Surgery, Leeds General Infirmary, Great George Street, Leeds LS1 3EX, UK

Received 11 July 2002; received in revised form 1 October 2002; accepted 7 October 2002.

* Corresponding author. Tel.: +44-113-392-5736; fax: +44-113-392-8436
e-mail: thorpyat{at}aol.com


    Abstract
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Recurrence after complete excision of non-invasive thymoma is infrequent. We report a case of recurrent thymoma in the sternum in a 76-year-old man 13 years after complete surgical resection of stage I thymoma.

Key Words: Thymoma • Sternal tumour • Recurrent thymoma


    1. Introduction
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Thymomas are uncommon neoplasms but form the commonest tumour in the anterior mediastinum. Natural history of thymoma is unpredictable and often have an indolent course. Recurrence, up to 10–30%, may be seen as long as 10 years after complete surgical removal. Surgical re-resection whenever possible seems to be the most favoured approach.


    2. Case report
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
A 76-year-old man presented with 6-month history of chest pain. Clinical examination revealed a diffuse non-tender bony swelling arising from the mid portion of sternum measuring 8x6 cm. Computed tomography (CT) scan of the chest confirmed the presence of sternal tumour involving two to four ribs on right side and also adherence of the right lung to the under surface of the tumour (Fig. 1) . CT guided biopsy was inconclusive.



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Fig. 1. CT scan of the chest showing needle biopsy of large sternal tumour infiltrating adjacent right upper ribs.

 
In 1989 he had ‘complete’ excision of an asymptomatic thymoma by median sternotomy. Mediastinal tumour (14.5x9.5x6 cm) was accidentally detected on chest X-ray taken before inguinal hernia repair. The large encapsulated thymoma was occupying superior mediastinum more into right hemithorax which was excised along with mediastinal pleura on the right side. It was considered stage I thymoma as it was complete encapsulated.

In March 2002 he underwent sternal resection with resection anterior ends of right two to four ribs along with wedge excision of upper lobe of right lung. At surgery the tumour was 9x8x5 cm in diameter involving the subcutaneous tissues, sternum, right upper ribs and mediastinal fibrofatty tissues. The residual defect was reconstructed with a sandwich prosthesis consisting of two layers of marlex mesh and methyl methacrylate cement. The postoperative recovery was uncomplicated apart from a wound seroma, which required needle aspiration.

Histologically it was a mixed thymoma (according to Muller-Hermelink classification) or type AB (in WHO classification). The histological appearances of the recent tumour are similar to those of the mediastinal neoplasm resected in 1989. Although the right lung was found adherent to the tumour on CT scan of the chest it was not involved by the thymoma. The fibrous adhesions were probably due to previous surgery in 1989 where in the mediastinal pleura on the right was excised along with the tumour.


    3. Discussion
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Recurrence of thymoma has been reported after disease free intervals of more than 10 years [14]. Intrathoracic recurrence is more often in early stage (I and II) and distant metastases occur often in stages III and IV thymoma. Recurrence rate for stage I thymoma is around 5% [4,5]. Distant metastases have been documented in liver, brain, vertebrae, ribs and clavicle and also in thoracotomy scar [1,3,4]. Predominantly epithelial tumours and thymic carcinoma or atypical thymoma have the highest recurrence rate [4,6]. Extensive search of English literature did not reveal any report of recurrent stage I thymoma with presentation as sternal tumour.

Sternal recurrence could be possible be due to tumour implant in the sternum at the time original excision or recurrent substernal tumour eroding in to the bone. Local recurrence is attributed to the multicentic origin of thymoma in residual or ectopic thymus [3,5]. In view of high possibility of ectopic thymic tissue in up to 10–15% of population, some recommend extended thymectomy for myasthenia gravis even in the absence of thymoma [5]. Postoperative radiotherapy is likely to reduce the chance of recurrence [2,3,5]. It is generally agreed that patients with stage I thymoma do not need adjuvant therapy.

Survival after complete resection of stage I thymoma is about 95% at 5 years and 86% at 10 years [4]. Prognosis and recurrence is dependent on stage, size, complete resection and histology. Patients with myasthenia are said to have better survival in view of early detection. According to Blumberg et al. 5-year survival of patients with large tumour (size more than 11 cm) is 58% compared to one with smaller tumour with 5-year survival of 84% [4]. Indeed their policy is to consider all patients with thymoma of more than 5 cm or invasive tumors for additional therapy. Patient with predominantly lymphocytic tumour having up to 90% survival at 10 years compared with 60% 10-year survival for patients with predominantly epithelial or mixed variety and atypical thymoma.

Surgical complete excision is the best modality in the management of thymoma, however there remains controversy in treating recurrent thymoma. Re-resection of recurrent thymoma, whenever possible, is associated with greater survival (5 years 85 versus 45% for those without surgery) [24]. Benefits of multimodality treatment will probably remain debatable in the absence of prospective randomised studies in view of the rarity, indolent unpredictable natural history and heterogeneity of thymoma. However atypical thymoma (showing nuclear atypia) or thymic carcinoma and invasive tumour do need aggressive combination therapy.


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

  1. Awad W.I., Symmana P.J., Dussek J.E. Recurrence of stage I thymoma 32 years after total excision. Ann Thorac Surg Dec 1998;66(6):2106-2108.[Abstract/Free Full Text]
  2. Ruffini E., Mancuso M., Oliaro A., Casadio C., Cavallo A., Cianci R., Filosso P.L., Molinatti M., Porrello C., Cappello N., Maggi G. Recurrence of thymoma: analysis of clinicopathological features, treatment and outcome. J Thorac Cardiovasc Surg Jan 1997;113(1):55-63.
  3. Regnard J.-F., Zinzindohoue F., Magdeleinat P., Guibert L., Spaggiari L., Levasseur P. Results of re-resection for recurrent thymomas. Ann Thorac Surg Dec 1997;64(6):1593-1598.[Abstract/Free Full Text]
  4. Blumberg D., Port J.L., Weksler B., Delgado R., Rosai J., Bains M.S., Ginsberg R.J., Martini N., McCormack P.M., Rush V., Burt M.E. Thymoma: a multivariate analysis of factors predicting survival. Ann Thorac Surg Oct 1995;60(4):908-914.
  5. Masaoka A., Yamakawa Y., Niwa H., Fukai I., Kondo S., Kobayashi M., Fujii Y., Monden Y. Extended thymectomy for myasthenia gravis patients: a 20-year review. Ann Thorac Surg 1996;62(3):853-859.[Abstract/Free Full Text]
  6. Kondo K., Sakiyama S., Takahashi K., Uyama T., Monden Y., Shimosato Y. Two cases of repeatedly recurrent atypical thymoma. Chest Jan 1999;115(1):282-285.




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