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Ann Thorac Surg 2003;76:2081-2082
© 2003 The Society of Thoracic Surgeons

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Case report

Calcifying fibrous pseudotumor of the pleura: a rare location

^a Service d’Anatomie et de Cytologie Pathologiques, Tunis, Tunisia
^b Service de Chirurgie Thoracique et Cardio-vasculaire, Hôpital A. Mami, Tunis, Tunisia

Accepted for publication April 22, 2003.

^* Address reprint requests to Dr Ammar, 55 Rue Chedly Gtari, 1013 El Menzeh IX, Tunis, Tunisia
e-mail: ammar.abderrazak{at}planet.tn

	Abstract

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Calcifying fibrous pseudotumor is a rare lesion recently reported in the literature. We report a new case occurring in the pleura, which is an unusual location. The patient, a 38-year-old woman, had a left pleural mass revealed by chest roentgenograms. The tumor was resected by video-assisted thoracotomy. Only 26 cases have been reported in the literature to date, but only 4 cases have been reported in the pleura. The pathogenesis is discussed but remains uncertain. Tumor resection appears to be the best option and the clinical course is benign. However, the number of cases to date is insufficient to establish therapeutic recommendations.

	Introduction

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Calcifying fibrous pseudotumor (CFP) is a recently recognized well-defined fibrous lesion [1, 2]. This unusual entity occurs predominantly in the young and has a good prognosis. We report a case of CFP in the pleura as a rare location.

A 38-year-old woman presented with thoracic pain and was in good health without general symptoms. The patient complained of pain 3 months before presenting at our outdoor consultation. Chest roentgenograms revealed a left pleural mass. Computed tomographic scan confirmed a 6 x 5, 3-cm mass with several calcifications (Fig 1). There was no involvement of the chest wall. Taking into account this clinical pattern, a benign process revealed, such a solitary fibrous tumor of the pleura could be considered. A biopsy was not performed because the mass was surgically resectable, and in this type of procedure the tumoral cell spillage is hardly unavoidable.

View larger version (144K): [in this window] [in a new window]   Fig 1. Computed tomographic scan showing left pleural mass without intercostal space involvement.

On gross examination, the lesion was firm, white, homogeneous, and well limited but nonencapsulated (6 x 5, 3-cm mass). The lesion is mostly composed of dense hyalinized collagenous tissue containing calcifications (Fig 2). The calcifications had a laminated appearance typical of psammoma bodies. The lesion was interspersed with sparse spindle cells and with scattered lymphoid and plasma cells (Fig 3). No atypical features of spindled cells were noted. There was no necrosis and no amyloid substance. Most spindle cells were positive for cytokeratin (Dako, France), vimentin (Dako), and S100 protein (Dako). Only a few cells were labeled with alpha smooth muscle actin (Dako). There was no staining for desmin (Dako) and epithelial membrane antigen (Dako).

View larger version (138K): [in this window] [in a new window]   Fig 2. Microscopic features of calcifying fibrous pseudotumor: dense hyalinized collagenous tissue with rare spindle cells and psammoma bodies. (Hematoxylin & eosin, x100.)

View larger version (137K): [in this window] [in a new window]   Fig 3. High-power view featuring sparse spindle cells mixed with lymphocytes. (Hematoxylin & eosin, x400.)

	Comment

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The lesion is rare with only 26 reported cases in the literature. Patients are always young children or young adults, predominantly women. Discovery is often fortuitous. General symptoms are not infrequent. Calcifying fibrous pseudotumor has been described in soft tissues (trunk, limbs, neck) and in serous locations (peritoneal and pleural) [3, 4] with only four pleural cases.

The pathologic features of CFP are well established. The features include a nonencapsulated, densely hyalinized, fibrotic proliferation with slight infiltration of lymphocytes and plasma cells. Two types of calcifications have been described (ie, psammomatous and dystrophic types). Immuno-labeling is not necessary for diagnosis. Staining for alpha smooth actin is positive in the majority of reported cases and negative in some of the others. Cytokeratins are negative in almost all reported cases. Our case and the one reported by Van Dorpe and associates [5] are the only two cases published to date that show a positive reaction for this marker.

Careful examination of the criteria for this lesion enables separation of similar benign lesions such as solitary fibrous tumors of the pleura, calcified granulomas, calcified pleural plaques, chronic fibrous pleuritis, as well as intrapulmonary lesions such as hyalinizing granuloma, inflammatory pseudotumor, and amyloid. Calcifying fibrous pseudotumor can appear to be multiple pleural lesions [4] simulating mesothelioma. Desmoplastic mesothelioma must also be considered in differential diagnoses.

The cause and pathogenesis remain unclear, but a possible relationship with other pseudotumors (such as nodular fasciitis or inflammatory myofibroblastic tumor) has been proposed by some authors [1, 2]. Van Dorpe and associates [5] reported the case of a 17-year-old girl with multiple peritoneal CFP and an inflammatory myofibroblastic tumor (inflammatory pseudotumor). Some multinodular lesions showed calcifying fibrous pseudotumors next to inflammatory myofibroblastic tumors with transitional stages. This case clearly illustrates a histogenetic relationship between CFP and inflammatory myofibroblastic tumor and suggests that the former is a late sclerosing stage of the latter. However, the possibility that CFP is a distinctive lesion with unique clinical and biological significance cannot be excluded. There are no cytogenetic data reported on CFP, and thus it is not known whether the anaplastic lymphoma kinase gene is involved, as it is in inflammatory myofibroblastic tumors.

Local excision appears to be the best therapy for CFP of the pleura as it occurs in soft tissue. If this lesion is similar to those of soft tissues, it may be expected at a low frequency of local recurrence with a good prognosis.

	References

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1. Rosenthal N.S., Abdul-Karim F.W. Childhood fibrous tumor with psammoma bodies. Clinicopathologic features in two cases. Arch Pathol Lab Med 1988;112:798-800.[Medline]
2. Fetsch J.F., Montgomery E.A., Meis J.M. Calcifying fibrous pseudotumor. Am J Surg Pathol 1993;17(5):502-508.[Medline]
3. Pinkard N.B., Wilson R.W., Lawless N., et al. Calcifying fibrous pseudotumor of pleura. A report of 3 cases of a newly described entity involving the pleura. Am J Clin Pathol 1996;105(2):189-194.[Medline]
4. Heinaut P., Lesage V., Weynand B., Coche E., Noirhomme P. Calcifying fibrous pseudotumor: a patient presenting with multiple pleural lesions. Acta Clin Belg 1999;54(3):162-164.[Medline]
5. Van Dorpe J., Ectors N., Geboes K., D’Hoore A., Scoit R. Is calcifying fibrous pseudotumor a late sclerosing stage of inflammatory myofibroblastic tumor?. Am J Surg Pathol 1999;23(3):329-335.[Medline]

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This Article Abstract Full Text (PDF) Alert me when this article is cited Alert me if a correction is posted Citation Map Services Email this article to a friend Similar articles in this journal Similar articles in PubMed Alert me to new issues of the journal Add to Personal Folders Download to citation manager Author home page(s): Tarek Kilani Permission Requests Citing Articles Citing Articles via HighWire Citing Articles via Google Scholar Google Scholar Articles by Ammar, A. Articles by Kilani, T. Search for Related Content PubMed PubMed Citation Articles by Ammar, A. Articles by Kilani, T. Related Collections Pleura