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J Thorac Cardiovasc Surg 2006;131:1412-1413
© 2006 The American Association for Thoracic Surgery

Brief Communication

Giant thymolipoma in association with a high production of carbohydrate antigen 19-9 and an increased acetylcholine antibody titer

Valerio Perna, MD * , Ricardo Morera, MD, Ricard Ramos, MD, Ivan Macia, MD, Juan Moya, MD

Thoracic Surgery Department, Hospital Universitario de Bellvitge, Barcelona, Spain.

Received for publication December 27, 2005; accepted for publication January 24, 2006.

* Address for reprints: Valerio Perna, Ronda-del General Mitre 65 4-1, Barcelona 08017, Spain. (Email: 37987vp{at}comb.es).

During preoperative examination for chronic sinusitis, a 26-year-old man displayed an abnormal mass shadow on chest radiography. The patient was referred to us for further evaluation.

Chest radiography revealed a large mass in the mid-lower mediastinum that silhouetted bilateral heart borders and conformed to the shape of the heart, simulating cardiomegaly (Figure 1).


Figure 1
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  		Figure 1. Chest radiography showing a large mediastinal mass extending into both the right and left hemithoraces.

 
Physical examination revealed no abnormalities. Laboratory data were within the normal range, except for an incidental finding of increased serum carbohydrate antigen 19-9 (CA 19-9) level at 150.67 U/mL (normal, <40 U/mL) and an increased acetylcholine antibody titer. Computed tomography (CT) of the chest revealed a large inhomogeneous anterior mediastinal mass composed of fatty areas that surrounded the heart entirely and extended into both hemithoraces (Figure 2). CT-guided fine-needle aspiration biopsy revealed thymolipoma.


Figure 2
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  		Figure 2. Spiral computed tomographic scan identified a massive heterogeneous mass with fat attenuation, with soft tissue extending widely into the bilateral inferior hemithoraces.

 
The tumor was approached through a double thoracotomy. The mass was spread expansively anterior to the pericardium, wrapping the left phrenic nerve and extending inferiorly into both hemithoraces. The tumor was well encapsulated, measuring 17 x 13 x 5 cm and weighing 700 g at the right lobe and measuring 21 x 17 x 15 cm and weighing 1000 g at the left lobe. The postoperative course was uneventful, and serum CA 19-9 levels normalized within 6 weeks. After 6 months, the patient maintained normal serum CA 19-9 levels, and changes of the acetylcholine antibody titer were not observed.

Discussion

Thymolipoma is a rare, benign, and slow-growing tumor accounting for 2% to 9% of all thymic neoplasms. The tumor occurs most frequently in young adults, with no sex predilection. Most patients with thymolipoma are asymptomatic and display large anterior mediastinal masses on chest radiography when the tumor grows on the left side of the chest. A histopathologic evaluation, such as fine-needle biopsy, is needed to differentiate thymolipoma from lipoma, thymic hyperplasia, and liposarcoma; CT and magnetic resonance imaging can narrow the differential diagnoses.

Although several theories have been proposed, the pathogenesis of thymolipoma remains controversial. One theory, citing the haphazard mixture of thymic parenchyma and fat, holds that it is a thymic hamartoma. 1 Go A malformative theory 2 Go is supported by a reported case in which parathyroid tissue was admixed with a tumor resembling thymolipoma. This case suggests a possible origin from aberrant development of the third pharyngeal pouch. Other theories suggest that thymolipoma represents fatty regression of a thymoma or of a previously hyperplastic thymus. Perhaps the most intriguing theory is of thymolipoma representing a benign tumor of specialized thymic stroma 3 Go that maintains its relationship with the thymic epithelium as it grows. In this scenario thymolipoma would be analogous to other tumors of specialized stroma, such as fibroadenoma of the breast and adenofibroma of the uterus. Cases of malignant tumor of specialized thymic stroma, thymoliposarcoma, have been reported 4 Go to further support this analogy.

According to our review of the literature, there has been described only a single case of thymolipoma that seemed to produce CA 19-9, 5 Go a protein originally identified on the surface of colon cancer cells by using murine monoclonal antibodies. Interestingly, the thymolipoma in our case is associated with an increased acetylcholine antibody titer without clinical evidence of myasthenia gravis. Studies of larger case series are necessary to elucidate the relationships between a high production of CA 19-9 and an increased acetylcholine antibody titer in patients with thymolipoma.

References

  1. Moran CA, Rosado-de-Christenson M, Suster S. Thymolipoma. clinicopathologic review of 33 cases. Mod Pathol 1995;8:741-744.[Medline]
  2. van Hoeven KH, Brennan MF. Lipothymoadenoma of the parathyroid. Arch Pathol Lab Med 1993;117:312-314.[Medline]
  3. Hall GFM. A case of thymolipoma with observations on a possible relationship to intrathoracic lipomata. Br J Surg 1948;36:321-324.
  4. Havlicek F, Rosai J. A sarcoma of thymic stroma with features of liposarcoma. Am J Clin Pathol 1984;82:217-224.[Medline]
  5. Shinichi M, Kan O, Masafumi H, Masakatsu H, Takayuki S. Thymolipoma with high production of carbohydrate antigen 19-9. J Thorac Cardiovasc Surg 2004;127:1845-1847.[Free Full Text]




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