Abstract

BackgroundCastleman's disease is a rare form of localized lymph node hyperplasia of uncertain etiology. Although the mediastinum is the most common site of involvement, rare cases occurring in lymph node bearing tissue of other localization have been reported, including only a few intramuscular cases. Unicentric and multicentric Castleman's disease are being distinguished, the latter harboring an unfavorable prognosis.Case PresentationHere, we present a case of unicentric Castleman's disease in a 37-year-old woman without associated neoplastic, autoimmune or infectious diseases. The lesion was located in the femoral region of the right lower extremity and surgically resected after radiographic workup and excisional biopsy examinations. The tumor comprised lymphoid tissue with numerous germinal centers with central fibrosis, onion-skinning and rich interfollicular vascularization. CD23-positive follicular dendritic cells were detected in the germinal centers and numerous CD138-positive plasma cells in interfollicular areas. The diagnosis of mixed cellularity type Castleman's disease was established and the patient recovered well.ConclusionsIn conclusion, the differential diagnosis of Castleman's disease should be considered when evaluating a sharply demarcated, hypervascularized lymphatic tumor located in the extremities. However, the developmental etiology of Castleman's disease remains to be further examined.

Highlights

  • Castleman’s disease is a rare form of localized lymph node hyperplasia of uncertain etiology

  • Castleman’s disease is regarded as a polyclonal lymphoid proliferation of unknown etiology, designated as angiofollicular lymph node hyperplasia [1]. It was first described by Benjamin Castleman in 1954 as “localized mediastinal lymph node hyperplasia resembling thymoma” [2]

  • We present the clinico-pathological characteristics with a review of the literature

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Summary

Conclusions

It is important to bear the differential diagnosis of Castleman’s disease in mind when evaluating a sharply demarcated, hypervascularized lymphatic hyperplasia located in the extremities. The etiology of Castleman’s disease remains to be further examined in future studies. 3. Chen WC, Jones D, Ho CL, Cheng CN, Tseng JY, Tsai HP, Chang KC: Cytogenetic anomalies in hyaline vascular Castleman disease: report of two cases with reappraisal of histogenesis. 6. Hakozaki M, Tajino T, Yamada H, Kikuchi S, Hashimoto Y, Konno S: Intramuscular Castleman’s disease of the deltoid: a case report and review of the literature. Pre-publication history The pre-publication history for this paper can be accessed here: http://www.biomedcentral.com/1471-2407/11/352/prepub doi:10.1186/1471-2407-11-352 Cite this article as: Schaefer et al.: Unicentric castleman’s disease located in the lower extremity: a case report. IMS, HG, SS and MK acquired the radiographic, gross and histological pictures, and all authors read and approved of the final manuscript. Competing interests The authors declare that they have no competing interests

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