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Ann Thorac Surg 2002;73:630-632
© 2002 The Society of Thoracic Surgeons


Case report

Bilateral elastofibroma dorsi

Akif Turna, MD*a, Muhammet Ali Yilmaz, MDa, Nur Ürer, MDa, Mehmet Ali Bedirhan, MDa, Atilla Gürses, MDa

a Yedikule Hospital for Chest Disease and Thoracic Surgery, Zeytinburnu, Istanbul, Turkey

Accepted for publication April 13, 2001.

* Address reprint requests to Dr Turna, Cami Sok. Muminderesi Yolu. Emintas Camlik Sit. No: 32/22, Sahrayicedid, Kadikoy, 81080 Istanbul, Turkey
e-mail: aturna{at}turk.net


    Abstract
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 Abstract
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Elastofibroma dorsi was diagnosed in a 48-year-old woman with bilateral subscapular tumor masses diagnosed asynchronously in an interval of 4 months in spite of presence of another lesion at first admittance. She underwent subsequent resections of the lesions. They were diagnosed as elastofibroma. Reevaluation of the initial computerized tomography of thorax indicated an omitted small lesion with a 2-cm diameter and 25.2-day doubling time. Although the real neoplastic nature of elastofibroma is unknown, bilateral presence of the masses with different sizes and relatively short doubling times of the lesions must be kept in mind.


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Elastofibroma, a rare, noncapsulated benign entity is characterized by the proliferation of fibrous tissue with elastin and occurs most often in the infrascapular area of elderly women. This entity was first described by Jarvi and Saxen in 1961 [1]. This lesion usually arises beneath the romboid major and latissimus dorsi muscles subjacent to the inferior angle of the scapula.

Whether elastofibromas are true neoplasms or merely reactive pseudotumors has been a subject of controversy. It has been reported that the operative findings of elastofibroma dorsi are disconcerting because the lesion adheres densely to muscle, periosteum of ribs, and scapula like a malignant lesion [24]. It has been speculated that its pathophysiology represents a reaction to friction of scapula against rib cage [1]. Although studies documenting elastofibroma dorsi had been sporadic and indicated a quite rare thoracic pathology, Brandser and colleagues [5] reported that the prevalance of elastofibroma in an asymptomatic elderly population revealed by computed tomography (CT) was 2%. CT imaging of thorax has a role in identifying the infrascapular ill-defined lesion but is of no pathognomonic value. Although the use of magnetic resonance imaging (MRI) can lead to a presumptive diagnosis in elderly individuals with subscapular lesions, they are often mistaken for neoplasms.

A 48-year-old woman was referred to our hospital because of a back thoracic pain and a palpable mass located under the left scapula. Chest roentgenogram presumed an extrathoracic mass and a thorax CT revealed a poorly circumscribed, heterogeneous left infrascapular soft-tissue mass with diameter of 8 cm (Fig 1). She had no history of hard or heavy labor during her lifetime. MRI of the lesion confirmed the noncapsular, heterogeneous nature of the soft-tissue mass with signal intensity similar to that of skeletal muscle (namely latissimus dorsi muscle) interlaced with strands of fat. Since the MRI and CT examination of the mass does not exclude the possibility of a malignant lesion, CT scans of abdomen and cranium were performed. No radiologic abnormality apart from a cortical atrophy of the brain was found. On gross examination performed in general anesthesia, a firm, rubbery, not encapsulated, malignant-tumor-like lesion was seen. Frozen section examination of the incisional biopsy disclosed a benign tumor which is rich from collagen fibers. The lesion was totally excised with negative margins confirmed by histologic evaluation.



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Fig 1. Computed tomographic (CT) scan of the patient’s chest. Unencapsulated soft tissue density is seen beneath left latissimus dorsi muscle. Meticulous reevaluation of the initial CT scan shows previous omitted right lesion (arrow) which is about one-third of the presented lesion in size.

 
Postoperative course was uneventful, and the patient was followed up monthly. Four months later, the patient was admitted to our hospital because of a right juxtascapular pain and a palpable infrascapular mass. A CT scan showed a right periscapular noncapsulated soft-tissue mass with a diameter of 6 cm (Fig 2). In MRI, a mass 4 cm in diameter was shown to demonstrate signal intensities isointense to that of muscle. A meticulous reevaluation of the previous CT performed 4 months earlier (Fig 1), disclosed a possible initial infrascapular thoracic lesion in the same location which is approximately 2 cm in diameter. Again, a total resection of the mass with marginal muscle tissue was performed. Histologic examination of the lesion was reported to be elastofibroma. The patient was discharged home on the 5th postoperative day.



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Fig 2. Right-sided nonencapsulated soft tissue mass (6 cm in diameter) is found (arrow) 4 months after the left-sided presentation.

 
Grossly, the tumors formed firm, ovoid, ill-defined masses of 10 and 4 cm in diameter, respectively. Histologically, the tumor masses consisted of a mixture of eosinophilic collagen and elastic fibers, in about equal proportions, that extended in an irregular manner into adjacent fibrous and adipose tissue. Elastic fibers had a degenerated beaded appearance. These beads were strongly colored with an elastic stain (Fig 3). Specimens contain areas of interspersed fat. Clinically and pathologically, the lesion studied fits the criteria of a typical elastofibroma [1].



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Fig 3. View of elastofibroma showing numerous elongated and globe-like fibers (Verhoeff elastic-tissue stain; original magnification, x400).

 

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 Abstract
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 Comment
 References
 
It is often difficult to identify the characteristics of soft-tissue tumors of the chest wall. Elastofibroma is well known to pathologists and not rare at autopsy, with 2% reported prevalance [5]. Although these lesions can usually be diagnosed on the basis of their imaging characteristics, increased awareness of these characteristics will decrease misdiagnosis of these lesions as malignancies and avoid unnecessary biopsies and surgeries. However, some researchers believe that imaging features are not sufficient to make the diagnosis of elastofibroma dorsi and believe that biopsy is indicated to exclude more aggressive tumors [6]. Similarly, Mojica and Kuntzman reported that fine-needle aspiration represents the simplest and quickest method of obtaining a definitive diagnosis in elastofibroma dorsi [7], however, they also admitted that, due to the nature of this lesion, a correct diagnosis could inadvertently be missed. In these lesions, CT scan and MRI of the thoracic wall depict only noncapsulated malignant-like masses without discriminating the border between lesion and normal thoracic muscle bundles. However, Yu and associates [8] speculated that T1- and T2-weighted spin-echo MRI showing periscapular soft-tissue neoplasm demonstrating a pattern of linear alternating regions of high and intermediate signal intensity might be sufficient to allow the diagnosis of elastofibroma. Nevertheless, diagnosis of elastofibroma for nonencapsulated periscapular lesion in elderly women must be kept in mind.

Constant trauma between chest wall and scapula with resulting excessive elastin production and collagen degeneration might play a major role in the pathogenesis of this rare proliferative lesion. Whether elastofibroma is a true neoplasm or a reactive fibrous lesion that produces not only collagen, but also abnormal elastic fibers, has been a subject of controversy and remains undetermined [2]. For this reason, despite its low incidence, this lesion should be known to differentiate it from malignant tumors and to avoid wide or radical surgery. Our strategy in those lesions has been to use frozen section histologic evaluation during operation, and in the case of benign histology, to perform complete resection. Several authors reported bilateral presentation of elastofibroma usually with bilateral thoracic pain increased during movement [5, 9], although there is no study reporting rapid progression of the contralateral lesion in such a short period. Briccoli and coworkers reported 1 patient with elastofibroma dorsi who had a second lesion on the other side 2 years after surgical excision of the first lesion [4]. In our patient, radiologic doubling time of the lesion has been calculated to be 25.2 days using the method originally described by Schwartz [10]. However, it has been known to be a very slow-growing process; the need for an explanation for the development of this benign lesion with 1 month doubling time warrants further study. Therefore, the fundamental question of pathogenesis of this lesion is still unanswered, because the presence of a type of collagen not normally found in reparative processes could be explained by either metaplastic or true neoplastic transformation. Inasmuch as it may have the calculated short doubling period, a neoplastic pattern of pathogenesis can be proposed. However, only 1 case of local recurrence has been reported in the literature [6]; malignant transformation of the lesion or metastasis has never been observed.

In conclusion, elastofibroma dorsi is a rare, ill-defined, pseudotumoral lesion of the soft tissues. Despite its low incidence, it is important to differentiate this lesion from other soft-tissue lesions, such as sarcomas and desmoid tumors. Moreover, bilateral occurrence and relatively short doubling time of the lesion must be kept in mind.


    References
 Top
 Abstract
 Introduction
 Comment
 References
 

  1. Jarvi O.H., Saxen A.E. Elastofibroma dorsi. Acta Pathol Microbiol Scand 1961;144(Suppl 51):83.
  2. Marin M.L., Perzin K.H., Markowitz A.M. Elastofibroma dorsi: benign chest wall tumor. J Thorac Cardiovasc Surg 1989;98:234-238.[Abstract]
  3. Brown R.K. Elastofibroma dorsi. New Engl J Med 1966;275:155-156.
  4. Briccoli A., Casasei R., Di Renzo M., Favale L., Bacchini P., Bertoni F. Elastofibroma dorsi. Surg Today 2000;30:147-152.[Medline]
  5. Brandser E.A., Goree J.C., El-Khoury G.Y. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. AJR Am J Roentgenol 1998;171:977-980.[Abstract/Free Full Text]
  6. Nagamine N., Nohara Y., Ito E. Elastofibroma in Okinawa. A clinicopathologic study of 170 cases. Cancer 1982;50:1794-1795.[Medline]
  7. Mojica W.D., Kuntzman T. Elastofibroma dorsi: elaboration of cytologic features and review of its pathogenesis. Diagn Cytopathol 2000;23:393-396.[Medline]
  8. Yu J.S., Weis L.D., Vaughan L.M., Resnick D. MRI of elastofibroma dorsi. J Comput Assist Tomogr 1995;19:601-603.[Medline]
  9. Tighe J.R., Clark A.E., Turvey D.J. Elastofibroma dorsi. J Clin Pathol 1968;21:463-469.[Abstract/Free Full Text]
  10. Schwartz M. A biomathematical approach to clinical tumor growth. Cancer 1961;14:1272-1294.[Medline]



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