Journal of Pathology and Translational Medicine

Case Study

Morphologic variant of follicular lymphoma reminiscent of hyaline-vascular Castleman disease: Jiwon Koh, Yoon Kyung Jeon; J Pathol Transl Med. 2020;54(3):253-257. Published online February 5, 2020; DOI: https://doi.org/10.4132/jptm.2019.12.17

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Follicular lymphoma (FL) with hyaline-vascular Castleman disease (FL-HVCD)-like features is a rare morphologic variant, with fewer than 20 cases in the literature. Herein, we report a case of FL-HVCD in a 37-year-old female who presented with isolated neck lymph node enlargement. The excised lymph node showed features reminiscent of HVCD, including regressed germinal centers (GCs) surrounded by onion skin-like mantle zones, lollipop lesions composed of hyalinized blood vessels penetrating into regressed GCs, and hyalinized interfollicular stroma. In addition, focal areas of abnormally conglomerated GCs composed of homogeneous, small centrocytes with strong BCL2, CD10, and BCL6 expression were observed, indicating partial involvement of the FL. Several other lymphoid follicles showed features of in situ follicular neoplasia. Based on the observations, a diagnosis of FL-HVCD was made. Although FLHVCD is very rare, the possibility of this variant should be considered in cases resembling CD. Identification of abnormal, neoplastic follicles and ancillary immunostaining are helpful for proper diagnosis.

Citations

Citations to this article as recorded by

Unicentric Castleman Disease: Illustration of Its Morphologic Spectrum and Review of the Differential Diagnosis
Siba El Hussein, Andrew G. Evans, Hong Fang, Wei Wang, L. Jeffrey Medeiros
Archives of Pathology & Laboratory Medicine.2024; 148(1): 99. CrossRef
Finding a Needle in the Haystack
Hung-Yu Lin, Yi-Jen Peng, Yi-Ying Wu, Ping-Ying Chang
Journal of Medical Sciences.2023; 43(6): 292. CrossRef
Analysis of immunophenotypic features in hyaline vascular type Castleman disease
Yu Chang, Yu Ma, Chen Chang, Wensheng Li
Diagnostic Pathology.2023;[Epub] CrossRef
In‐situ follicular neoplasia: a clinicopathological spectrum
Gurdip S Tamber, Myriam Chévarie‐Davis, Margaret Warner, Chantal Séguin, Carole Caron, René P Michel
Histopathology.2021; 79(6): 1072. CrossRef

Case Reports

"Chordoid" Meningioma with Systemic Manifestations of Castleman Syndrome: A case report.: Hwa Sun Lee, Hweon Ok Kim, Do Youn Park, Mee Yeong Sol, Kang Suek Suh, Sun Kyung Lee; Korean J Pathol. 1996;30(3):255-260.

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Abstract PDF: Chordoid meningioma is a recently established meningeal tumor and is characterized by a chordoma like histologic appearance, peritumoral lymphoplasma cell infiltrates causing systemic manifestations similar to Castleman syndrome and having a good prognosis. We experienced a case of chordoid meningioma in a 25 year-old woman. The patient preoperatively manifested iron-resistant hypochromic microcytic anemia, polyclonal gammopathy with beta-gamma bridging and detected a huge mass in the right temporo-parietal convexity of the brain. Microscopically, the mass was composed of nests and cords of cuboid, partly vacuolated cells in a mucoid matrix, simulating chordoma. The tumor was surrounded by masses of lymphoplasma cells around vessels, many of the plasma cells contained Russell bodies. Ultrastructural findings showed intranuclear cytoplasmic invaginations, microvilli protruding from cytoplasmic surfaces and well formed desmosomes. Some portions of tumor cell surface were covered by stretches of basal lamina.

Carcinoma Showing Thymus-Like Differentiation (CASTLE) of the Thyroid Gland: A case report.: Mi Jin Gu, Young Ran Shim, Joon Hyuk Choi, Won Hee Choi; Korean J Pathol. 1999;33(6):460-462.

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Abstract PDF: Carcinoma Showing Thymus-Like Differentiation (CASTLE) is a rare tumor, which occurs in the thyroid gland and surrounding soft tissue, or soft tissue of the neck. It is thought to originate from ectopic thymus or branchial pouch remnants. We report a case of CASTLE of the thyroid gland in a 42-year-old woman. Grossly, a nodular, partly well demarcated, grayish yellow, 3.0 2.0 cm sized, solid mass was found in the right thyroid gland. Microscopically, the tumor was divided into lobules of variable size and shape, nests and cords with thin and thick fibrous septa which were infiltrated by lymphocytes and plasma cells. The tumor cells were large, polygonal and had vesicular nuclei with prominent nucleoli and eosinophilic cytoplasm. Some cells, especially in the central portion of the nests had abundant eosinophilic cytoplasm and showed squamoid feature.

An Unusual Stroma-Rich Variant of Castleman's Disease of the Hyaline-Vascular Type: A Case Report.: Ji Han Jung, Gyeongsin Park, Hyun Joo Choi, Jinyoung Yoo, Seok Jin Kang, Kyo Young Lee; Korean J Pathol. 2007;41(4):266-270.

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Abstract PDF: The stroma-rich variant of Castleman's disease of the hyaline-vascular type (CDHV) is a rare entity that shows overgrowth of a variety of stromal cells in the widened interfollicular (IF) area. We report here on a case of a stroma-rich variant of CDHV in an 18-year-old man who presented with an asymptomatic solitary neck mass he'd had for 1 year. Histologically, an enlarged lymph node fulfilled the criteria of CDHV, along with vague nodularity of a widened IF area. The nodular lesion consisted of numerous vessels and a proliferation of spindle cells. Immunohistochemically, the spindle cells were positive for vimentin and smooth muscle actin, they were negative for desmin, CD21, CD34, CD68, ALK-1, and S-100 protein. This stromal lesion is typically hyperplastic and clinically benign, and it must be distinguished from neoplastic stromal proliferation associated with Castleman's disease because of its potential for recurrence and metastasis.

Angiofollicular Lymph Node Hyperplasia(Castleman's disease): 3 cases report.: Jeong Hee Park, Gil Ro Han, Hee Jin Chang, Jin Hee Sohn, Jung Il Suh; Korean J Pathol. 1992;26(3):298-305.

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Abstract PDF: Angiofollicular lymph node hyperplasia(AFLNH) was first described in 1956 by Castleman et al. It was initially reported as a solitary mediastinal mass but multicentric and extranodal disease is now well known. Histologically two distinct variants, e.g. the hyaline vascular type and the plasma cell type, of AFLNH are recognized. And the plasma cell type is typically associated with clinical syndrome consisting of fever, anemia, elevated erythrocyte sedimentation rate and polyclonal hypergammaglobulinemia. Recently, we experineced three cases of AFLNH. Histologically, two cases were hyaline vascular type, that were presented as a right supraclavicular mass of 49-year-old female, and as an anterior mediastinal mass of 53-year-old female. The remaining one case was plasma cell type that was presented as a left axillary mass of 63-year-old male. The former two cases showed typical features of hyaline vascular type but in case 1, exuberant proliferation of hyalinized vessels of capillary size was characteristic feature. The latter case of plasma cell type characteristically showed clinical syndrome consisting of fever, hypoalbuminemia, polyclonal hypergammaglobulinemia. All cases were presented as a single mass and they were well after surgical excision.

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