J Korean Med Sci.  2011 Feb;26(2):304-307. 10.3346/jkms.2011.26.2.304.

Extranodal Interdigitating Dendritic Cell Sarcoma Presenting in the Pleura: A Case Report

• Han HS
• Lee OJ
• Lim SN
• An JY
• Lee KM
• Choe KH
• Lee KH
• Kim ST

• KMID: 1782124
• DOI: http://doi.org/10.3346/jkms.2011.26.2.304

Abstract

Interdigitating dendritic cell sarcoma (IDCS) is an extremely rare neoplasm arising from the antigen-presenting cells of the immune system. This disease usually involves the lymph nodes, and rarely, extranodal sites may be affected. The authors report a case of extranodal IDCS presenting in the pleura. A 32-yr-old man presented with progressive chest pain. Imaging studies showed diffuse pleural thickening with pleural effusion. Morphological and immunohistochemical analysis of an incisional biopsy of the pleura were consistent with a diagnosis of IDCS; tumor cells were positive for S100 and CD45, but negative for CD1a, CD21, CD35, B cell and T cell markers. The patient was administered chemotherapy, but died of progressive disease. Although its incidence is extremely rare, this case suggests that extranodal IDCS should be considered in the differential diagnosis of undifferentiated neoplasms and that immunohistochemical staining be performed using appropriate markers.

Keyword

Dendritic Cell Sarcoma; Interdigitating; Pleura

MeSH Terms

Adult
Dendritic Cell Sarcoma, Interdigitating/diagnosis/*pathology
Fatal Outcome
Humans
Male
Pleura/*pathology
Pleural Neoplasms/diagnosis/*pathology
Tumor Markers, Biological

Figure

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  Fig. 1 Computed tomography of the chest showed irregular pleural thickening and pleural effusion in the left lung.

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  Fig. 2 Pathologic features of an incisional biopsy of the pleura. (A) The growth pattern of the tumor is infiltrative with a desmoplastic stroma (H&E, × 100). (B) The tumor cells shows oval to spindled and indented nuclei and abundant cytoplasm with indistinct border (H&E, × 400).

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  Fig. 3 Immunohistochemistry showed that the tumor cells were positive for (A) S100 and (B) CD 45 (× 400).

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  Fig. 4 Corresponding positron emission tomography (PET)-CT image showed 18-fluoro-deoxyglucose (FDG) uptake (standardized uptake value, SUV, 10.5 and 9.8), by thickened pleura of the left hemithorax and the whole axial skeleton, which was compatible with malignant tissue.

Reference

1. Fonseca R, Yamakawa M, Nakamura S, van Heerde P, Miettinen M, Shek TW, Myhre Jensen O, Rousselet MC, Tefferi A. Follicular dendritic cell sarcoma and interdigitating reticulum cell sarcoma: a review. Am J Hematol. 1998. 59:161–167.

2. Pileri SA, Grogan TM, Harris NL, Banks P, Campo E, Chan JK, Favera RD, Delsol G, De Wolf-Peeters C, Falini B, Gascoyne RD, Gaulard P, Gatter KC, Isaacson PG, Jaffe ES, Kluin P, Knowles DM, Mason DY, Mori S, Müller-Hermelink HK, Piris MA, Ralfkiaer E, Stein H, Su IJ, Warnke RA, Weiss LM. Tumours of histiocytes and accessory dendritic cells: an immunohistochemical approach to classification from the International Lymphoma Study Group based on 61 cases. Histopathology. 2002. 41:1–29.

3. Kairouz S, Hashash J, Kabbara W, McHayleh W, Tabbara IA. Dendritic cell neoplasms: an overview. Am J Hematol. 2007. 82:924–928.

4. De Pas T, Spitaleri G, Pruneri G, Curigliano G, Noberasco C, Luini A, Andreoni B, Testori A, de Braud F. Dendritic cell sarcoma: an analytic overview of the literature and presentation of original five cases. Crit Rev Oncol Hematol. 2008. 65:1–7.

5. Efune G, Sumer BD, Sarode VR, Wang HY, Myers LL. Interdigitating dendritic cell sarcoma of the parotid gland: case report and literature review. Am J Otolaryngol. 2009. 30:264–268.

6. Gaertner EM, Tsokos M, Derringer GA, Neuhauser TS, Arciero C, Andriko JA. Interdigitating dendritic cell sarcoma. A report of four cases and review of the literature. Am J Clin Pathol. 2001. 115:589–597.

7. Kawachi K, Nakatani Y, Inayama Y, Kawano N, Toda N, Misugi K. Interdigitating dendritic cell sarcoma of the spleen: report of a case with a review of the literature. Am J Surg Pathol. 2002. 26:530–537.

8. Pillay K, Solomon R, Daubenton JD, Sinclair-Smith CC. Interdigitating dendritic cell sarcoma: a report of four paediatric cases and review of the literature. Histopathology. 2004. 44:283–291.

9. Luk IS, Shek TW, Tang VW, Ng WF. Interdigitating dendritic cell tumor of the testis: a novel testicular spindle cell neoplasm. Am J Surg Pathol. 1999. 23:1141–1148.

10. Rupar G, Beham-Schmid C, Gallé G, Zigeuner R, Langner C. Interdigitating dendritic cell sarcoma of urinary bladder mimicking large intravesical calculus. Urology. 2005. 66:1109.

11. Banner B, Beauchamp ML, Liepman M, Woda BA. Interdigitating reticulum-cell sarcoma of the intestine: a case report and review of the literature. Diagn Cytopathol. 1997. 17:216–222.

12. Androulaki A, Giaslakiotis K, Lazaris AC. Interdigitating dendritic cell sarcoma/tumour of the tonsil. Br J Haematol. 2005. 131:415.

13. Yamakawa M, Matsuda M, Imai Y, Arai S, Harada K, Sato T. Lymph node interdigitating cell sarcoma. A case report. Am J Clin Pathol. 1992. 97:139–146.