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Eur J Cardiothorac Surg 2002;22:839-841
© 2002 Elsevier Science NL

Case report

Bilateral elastofibroma dorsi: proper positioning for an accurate diagnosis

Murat Karaa*, Erkan Dikmena, Simay Altan Karab, Pinar Atasoyc

a Department of Thoracic Surgery, University of Kirikkale, School of Medicine, 71100 Kirikkale, Turkey
b Department of Radiology, University of Kirikkale, School of Medicine, 71100 Kirikkale, Turkey
c Department of Pathology, University of Kirikkale, School of Medicine, 71100, Kirikkale, Turkey

Received 17 June 2002; received in revised form 15 July 2002; accepted 30 July 2002.

* Corresponding author. Esenlik sokak 7/10, TR-06540, Asaiayranci, Ankara, Turkey. Tel.: +90-318-225-4511; fax: +90-318-225-2819
e-mail: muratkara66{at}hotmail.com


   Abstract
Top
 Abstract
1. Introduction
 2. Case report
 3. Discussion
 References
 
Elastofibroma dorsi is a rare, slow-growing, ill-defined soft tissue tumor of the chest wall, most commonly located beneath the rhomboid major and latissimus dorsi muscles. It is usually unilateral, and bilateral involvement occurs in only 10% of patients. We report herein a case of a 56-year-old woman with bilateral elastofibroma dorsi. The patient was found to have bilateral involvement of the tumor following a physical examination with proper positioning. An elastofibroma should be considered in the differential diagnosis of tumors deeply located at the periscapular area. Meticulous physical examination may reveal a nonpalpable coexisting tumor with deep subscapular location and help the accurate diagnosis of bilateral elastofibroma dorsi, hence preventing a secondary operation.

Key Words: Elastofibroma • Thoracic surgery


   1. Introduction
Top
 Abstract
 1. Introduction
2. Case report
 3. Discussion
 References
 
Elastofibroma dorsi is a nonencapsulated benign tumor of the chest wall, characterized by the proliferation of fibrous tissue with elastin. It often presents as a nontender mobile mass subjacent to the inferior angle of the scapula in elderly women, and usually arises unilaterally beneath the rhomboid major and latissimus dorsi muscles [1,2].

We herein report a case of a 56-year-old woman with elastofibroma dorsi presenting with severe pain. The patient was found to have bilateral involvement following a meticulous physical examination. We tried to emphasize the significance of physical examination with proper positioning in patients with a possible contralateral nonpalpable tumor of elastofibroma dorsi to prevent a further operation.


   2. Case report
Top
 Abstract
 1. Introduction
 2. Case report
3. Discussion
 References
 
A 56-year-old housewife was admitted to our hospital with a severe back thoracic pain and palpable mass located to the right infrascapular area. She had a 6-month history of pain and clicking sensation at the periscapular area with arm motion. Physical examination revealed a palpable mass, measuring about 6x10 cm, located at the infrascapular area with a clicking sensation when the arm was mobilized. Laboratory data were within the normal limits. Computed tomography (CT) showed a right subscapular heterogeneous soft tissue mass. Considering the possible diagnosis of an elastofibroma dorsi at such a localization, we performed the physical examination again for the contralateral side. The patient was positioned as to stand with the arms slightly elevated forward and adducted. We eventually noticed another nontender and smaller mass measuring 4x6 cm located at the left subscapular area, which had been unnoticed at the previous examination. Magnetic resonance imaging (MRI) was performed at a field strength of 1.5 Tesla and confirmed the bilateral involvement of the infrascapular soft tissue tumor (Fig. 1) . The signal intensity of both lesions were similar to that of skeletal muscle interlaced with strands of fat.



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  		Fig. 1. Axial FSE T2-weighted images showing bilateral hypointense heterogeneous masses beneath the scapulas (arrows).

 
The surgical approach included bilateral posterolateral incisions made over the lesion with the arms positioned to expose the mass under general anesthesia. A firm, rubbery, nonencapsulated, malignant tumor-like lesion measuring 3x5x9 cm was observed following the division of the latissimus dorsi muscle on the right side. The tumor was attached to the periosteum of the underlying 6th–7th ribs, inferior angle of the scapula, and was densely adherent to striated muscle. The tumor was totally excised by sharp and blunt dissection with negative margins confirmed by histologic evaluation. The same procedure was performed on the contralateral side (Fig. 2A) . On histological examination, the tumors showed a mixture of intertwining swollen, eosinophilic collagen and elastic fibers admixed with aggregates of mature fat cells. The elastic fibers demonstrated a degenerated beaded appearance having an irregular serrated margin on Weigert's elastin stain (Fig. 2B). Both masses were diagnosed as elastofibroma. The postoperative course was uneventful. The patient remains well and disease-free 14 months after surgery.



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  		Fig. 2. (A) Macroscopic view of the operative specimen. (B) Microscopic view showing altered elastic fibers within a collagenous matrix (Weigert's elastin stain, x100).

 

   3. Discussion
Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
References
 
Elastofibroma dorsi is a rare benign tumor of the connective tissue first described by Järvi and Saxén [3]. Whether elastofibromas are true neoplasms or merely reactive pseudotumors producing abnormal elastic fibers has been a subject of controversy [2,4]. Elastofibroma was first considered to result from friction of the inferior scapula angle against the thoracic wall, however, the detection of new cases in different sites favored other hypotheses such as underlying genetic disposition, inherent enzymatic defect, or systemic involvement [5,6]. Increased prevalence has been reported in manual workers which may imply that repetitive minor trauma plays a major role in the etiology [5,6]. However, as in many reported cases, the patient presented was not a manual worker, and she had no history of hard or heavy labor during her lifetime. Conclusively, a multifactorial etiology may be more explanatory for elastofibroma because none of the theories can entirely explain the whole spectrum of the presented cases.

Women are more affected, with a ratio as high as 13:1 [1]. The reason of female predominance also remains obscure. The great majority of elastofibromas occur in the infrascapular or subscapular region. Another frequent site is the infraolecranon area. Other sites have been reported, e.g. the greater trochanter, deltoid muscle, ischial tuberosity, breast and foot [4–6]. Elastofibroma dorsi is usually unilateral, and bilateral involvement occurs in only 10% of patients [5]. More than 50% of patients with elastofibroma dorsi are asymptomatic. Patients often have mild pain. Severe pain as in the presented case has rarely been reported [1,5]. Some patients may experience a clicking or catching sensation with the arm motion as in our case. A rubbery, asymptomatic mass located at the periscapular region is usually palpable and barely noticeable when the arm lies against the chest. Unless the tumor is visible or palpable, an elastofibroma cannot be detected when the patient stands in a normal position. The patient should be positioned as to stand with the arms slightly elevated forward and adducted to make the tumor protrude, particularly in patients with nonpalpable and small tumors [7]. Likewise, the left-sided tumor in our case was very likely to go unnoticed unless we had suspected a bilateral occurrence and re-examined the patient after proper positioning.

A radiological survey outlined the prevalence of elastofibroma dorsi as high as 2% in asymptomatic patients [1]. In addition, an autopsy series [8] in individuals older than 55 years old revealed a greater prevalence of 24% in women and 11% in men. Another study of 100 autopsies reported a rate of 13% for elastofibroma and 81% for pre-elastofibroma-like morphological changes [9]. These data suggest that the real prevalence of elastofibroma should be greater than could be expected. Besides, previous reports have noted coincidental asymptomatic elastofibroma dorsi opposite to the symptomatic side [1,10]. Thus, careful physical and radiological evaluation is of great importance in a patient with a possible diagnosis of elastofibroma dorsi. Preoperative diagnosis of bilateral involvement will provide the excision of the tumors at one session. Otherwise, a secondary subsequent operation as experienced by some authors will be inevitable [2,5].

Chest X-ray, CT, and MRI are helpful in the diagnosis of elastofibroma dorsi. Chest X-ray may reveal soft tissue mass, or scapular elevation as a secondary finding. CT often shows poor differentiation of tumor edges from surrounding muscle planes and continuity with adjacent intercostal muscles. Despite the fact that elastofibroma dorsi is revealed by CT, the diagnosis of the contralateral tumor is often missed at initial presentation as in our case, because CT lacks sufficient contrast resolution to differentiate streaks of abnormal tissue from elastic tissue and will show elastofibroma dorsi only when it reaches a sufficient size to be detected [1,2]. On MR imaging, a typical feature of elastofibroma dorsi is that the interposed areas of decreased heterogeneous signal intensity also appear as low signal intensity on T2-weighted sequences. In contrast to CT, MRI was more sensitive in revealing the bilateral involvement of the tumor in our case. It is suggested that when a mass of interposed fat and soft tissue is located deep in relation to the scapula, no further investigation is needed to make the diagnosis of elastofibroma dorsi [1]. However, imaging features are not always sufficient to make the diagnosis of elastofibroma dorsi, thus biopsy, preferably excision of the tumor, is indicated to exclude it from fibromatosis, lipoma and fibrolipoma or from more aggressive tumors such as sarcomas or desmoid tumors [4–6].

Treatment of elastofibroma dorsi involves complete excision of the tumor. Surgical excision is recommended when elastofibroma dorsi causes functional disability, compression symptoms, pain, disfiguring/swelling on the chest wall, or when the tumor dimensions exceed 5 cm in diameter [4,5]. The poorly defined margins and the fixation of the tumor to muscles, periosteum of ribs, and scapula make dissection difficult, thus the procedure requires general anesthesia. Although its incidence is very low, elastofibroma should be known in the differential diagnosis of soft tissue tumors of the chest wall such as sarcomas and desmoid tumors to avoid an unnecessary radical and wide excision. Marginal excision carries a very low risk of recurrence and only one case with local recurrence has been reported in the literature [6]. Furthermore, neither malignant transformation nor metastases resulting from elastofibroma have been reported. We have not observed local recurrence during the 14 months of follow-up in the presented case.

In conclusion, lesions located deep beneath the inferior tip of the scapula in the chest wall should arouse suspicion of an elastofibroma. Meticulous physical examination with proper positioning may reveal a coexisting, contralateral tumor located at the subscapular area, and hence prevent a further operation.


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

  1. Brandser E.A., Goree J.C., El-Khoury G.Y. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. Am J Roentgenol 1998;171:977-980.[Abstract/Free Full Text]
  2. Turna A., Yilmaz M.A., Ürer N., Bedirhan M.A., Gürses A. Bilateral elastofibroma dorsi. Ann Thorac Surg 2002;73:630-632.[Abstract/Free Full Text]
  3. Järvi O.H., Saxén A.E. Elastofibroma dorsi. Acta Pathol Microbiol Scand 1961;51(Suppl 144):83-84.
  4. Marin M.L., Perzin K.H., Markowitz A.M. Elastofibroma dorsi: benign chest wall tumor. J Thorac Cardiovasc Surg 1989;98:234-238.[Abstract]
  5. Briccoli A., Casadei R., Di Renzo M., Favale L., Bacchini P., Bertoni F. Elastofibroma dorsi. Surg Today 2000;30:147-152.[Medline]
  6. Nagamine N., Nohara Y., Ito E. Elastofibroma in Okinawa. A clinicopathologic study of 170 cases. Cancer 1982;50:1794-1805.[Medline]
  7. Vastamaki M. Elastofibroma scapulae. Clin Orthop 2001;392:404-408.
  8. Järvi O.H., Lansimies P.H. Subclinical elastofibromas in the scapular region in an autopsy series. Acta Pathol Microbiol Scand 1975;83:87-108.
  9. Giebel G.D., Bierhoff E., Vogel J. Elastofibroma and pre-elastofibroma – a biopsy and autopsy study. Eur J Surg Oncol 1996;22:93-96.[Medline]
  10. Kransdorf M.J., Meis J.M., Montgomery E. Elastofibroma: MR and CT appearance with radiologic - pathologic correlation. Am J Roentgenol 1992;159:575-579.[Abstract/Free Full Text]



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This Article
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Erkan Dikmen
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Right arrow Articles by Atasoy, P.
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