Journal of Pathology and Translational Medicine

Case Study

Follicular Dendritic Cell Sarcoma of the Inflammatory Pseudotumor-like Variant Presenting as a Colonic Polyp: Shien-Tung Pan, Chih-Yuan Cheng, Nie-Sue Lee, Peir-In Liang, Shih-Sung Chuang; Korean J Pathol. 2014;48(2):140-145. Published online April 28, 2014; DOI: https://doi.org/10.4132/KoreanJPathol.2014.48.2.140

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Follicular dendritic cell (FDC) sarcoma is rare and is classified either as conventional type or inflammatory pseudotumor (IPT)-like variant. Extranodal presentation is uncommon and nearly all gastrointestinal FDC tumors are of the conventional type. IPT-like variant tumors occur almost exclusively in the liver and spleen and are consistently associated with Epstein-Barr virus (EBV). Here we report the case of a 78-year-old woman with an IPT-like FDC sarcoma presenting as a pedunculated colonic polyp. Histologically, scanty atypical ovoid to spindle cells were mixed with a background of florid lymphoplasmacytic infiltrate, which led to an initial misdiagnosis of pseudolymphoma. These atypical cells expressed CD21, CD23, CD35, and D2-40, and were positive for EBV by in situ hybridization, confirming the diagnosis. The patient was free of disease five months after polypectomy without adjuvant therapy. Although extremely rare, the differential diagnosis for colonic polyp should include FDC sarcoma to avoid an erroneous diagnosis. A review of the 24 cases of IPT-like FDC sarcoma reported in the literature reveal that this tumor occurs predominantly in females with a predilection for liver and spleen, and has a strong association with EBV.

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Original Article

Inflammatory Pseudotumor of the Urinary Bladder: An Immunohistochemical and Ultrastructural Study.: Seung Sam Paik, Joo Seob Keum, Moon Hyang Park, Jung Dal Park; Korean J Pathol. 1996;30(5):447-452.

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Abstract PDF: Inflammatory pseudotumor of the urinary bladder is an unusual, benign mesenchymal proliferative lesion of the submucosal stroma easily mistaken for a malignant neoplasm clinically and histologically. We present a case and describe the clinical presentation and radiologic, histologic, histochemical, immunohistochemical, and ultrastructural findings. A 23-year old patient presented with sudden onset of gross painless hematuria for 3 months. There was no previous instrumentation or surgery involving the genitourinary tract. Cystoscopy revealed a large polypoid and ulcerated bladder mass. The lesion consisted of plump spindle shaped, fibroblast-like cells embedded in a myxoid stroma. Mitotic figures were negligible and the lesion showed encroachment on the superficial muscle bundles. The spindle cells were immunoreactive for vimentin and muscle specific actin. Immunohistochemical and ultrastructural findings revealed the fibroblastic-myofibroblastic nature of this lesion. Complete surgical excision by partial cystectomy was successful in eradicating the lesion. The findings are described with a discussion of the pathogenesis and review of the literature.

Case Reports

Inflammatory Pseudotumor of the Kidney.: Hwa Eun Oh, Jeong Seok Moon, Sung Jin Cho, Nam Hee Won; Korean J Pathol. 1997;31(6):592-594.

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Abstract PDF: Inflammatory pseudotumor, originally described in the lung, is a relatively rare tumor-like lesion that occurs in various organs and tissues. It is usually well demarcated from the surrounding tissue, however it can be unfortunately resected as a malignant tumor. A few inflammtory pseudotumor in the kidney have been reported in English literature, but there have been no reports in Korea. We report a case with inflammatory pseudotumor of the kidney. A 48 year old woman had an intermittent flank pain on the right side. An ultrasonographic study suggested a renal cell carcinoma and a nephrectomy was done. Grossly, there were two separate masses with a well demarcated yellowish appearance, measuring 2.3 cm and 1.3 cm in diameter, respectively. Histologically, they were composed of smooth muscle actin positive spindle cells and a large number of foamy histiocytes, lymphocytes, and plasma cells in the fibrotic backgound.

Inflammatory Myofibroblastic Tumor of the Mesentery: A case report.: Sung Jig Lim, Gou Young Kim, Jae Hoon Park, Youn Wha Kim, Yong Koo Park, Ju Hie Lee, Moon Ho Yang; Korean J Pathol. 1999;33(9):729-732.

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Abstract PDF: Inflammatory myofibroblastic tumor or inflammatory pseudotumor is characterized by spindle cell proliferation with inflammatory cell infiltration, predominantly plasma cells and lymphocytes. We have experienced a case of inflammatory myofibroblastic tumor of the mesentery in a 57-year-old male patient with intermittent abdominal pain. On computer tomography, a well demarcated mass was seen in the mesenteric side of the ascending colon. Right hemicolectomy was performed under the impression of the metastatic tumor of lymph nodes. Grossly, a rather well-circumscribed gray white mass was noted in the mesentery of the ascending colon. Microscopically, the lesion consisted of plump spindle cells and accompanying inflammatory cellular infiltrates. The spindle cells were positive for vimentin.

Rosai-Dorfman Disease of the Nose and Salivary Gland: A case report.: Mee Sook Roh, Jin Sook Jeong, Sook Hee Hong; Korean J Pathol. 1999;33(12):1203-1206.

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Abstract PDF: Rosai-Dorfman disease (RDD) is a rare type of benign histiocytosis characterized histologically by intracellular engulfment of lymphocytes. Extranodal RDD may occur as a part of generalized process involving lymph nodes or may involve extranodal sites independent of the lymph node status. We have experienced a case of extranodal Rosai-Dorfman disease of the nose as an initial lesion prior to nodal involvement. The patient was a 20-year-old woman who complained of nasal obstruction for 4 years, remotely, and left submandibular mass for 3 months, recently. Histologically, the lesion taken from nasal cavity, submandibular gland and left upper jugular lymph node all showed an heavy infiltrate consisted of plasma cells, lymphocytes and sheets of macrophages with abundant pale cytoplasm, which replaced organ architecture. The associated focal fibrosis made it difficult to differentiate from inflammatory pseudotumor. Some macrophages demonstrated phagocytosis of lymphocytes, plasma cells and occasionally neutrophils. The macrophages were strongly positive for S-100 protein.

Original Article

Fine Needle Aspiration Cytologic Findings of Inflammatory Pseudotumor of the Lymph Node .: So Young Park, Gyung Yub Gong, Joo Ryung Huh, Eun Sil Yu, In Chul Lee, On Ja Kim; Korean J Cytopathol. 1997;8(1):87-92.

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Abstract PDF: Reports on the cytologic or histopathologic findings of inflammatory pseudotumors are relatively infrequent and most of them have dealt with those involving the lung, liver, genitourinary tract, alimentary tract, spleen, mediastinum, retroperitoneum, etc. Moreover there have not been any cytologic studies of those involving lymph nodes. We present fine needle aspiration cytologic features of inflammatory pseudotumor occurring in a lymph node in a 64 year-old man. The aspirate consisted of proliferating spindle cells and admixed histiocytes, fibroblasts, lymphocytes, and plasma cells. Histiocytes were present either singly or in loosely cohesive small clusters. A few multinucleated giant cells were present as well. However, tuberculosis could be excluded by the absence of typical granuloma, caseation necrosis, or characteristic mixed spindle and inflammatory cell components. Other benign and malignant lymphadenopathies could also be differentiated based on cytologic findings.

Case Reports

Fine Needle Aspiration Cytology of Inflammatory Pseudotumor of the Lung: Report of A Case Misdiagnosed as Adenocarcinoma .: Wan Seop Kim, Eun Kyung Hong, Moon Hyang Park; Korean J Cytopathol. 1999;10(2):145-149.

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Abstract PDF: Cytologic features of inflammatory pseudotumor of the lung have not been described frequently. We report fine needle aspiration cytologic(FNAC) finding of a case of inflammatory pseudotumor misdiagnosed as adenocarcinoma in a 63-year-old man. The FNAC displayed a mixture of histiocytes, myofibroblasts, pneumocytes, and plasma cells. Some histiocytes and myofibroblasts had large nuclei with irregular nuclear membrane and prominent nucleoli, which mislead the diagnosis of adenocarcinoma on FNAC. The heterogeneous cell population is the unique cytologic features of inflammatory pseudotumor, which are helpful to distinguish it from other circumscribed benign and malignant lesions. Familiarity with these features is essential to avoid misdiagnosis and possible overtreatment.

Fine Needle Aspiration Cytology of Inflammatory Myofibroblastic Tumor of Lung: A Case Report.: Gyeongsin Park, Kyungji Lee, Sun Mi Lee, Kyo Young Lee, Sang In Shim, Chang Suk Kang, Youn Soo Lee; Korean J Cytopathol. 2006;17(1):63-68.

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Abstract PDF: Inflammatory myofibroblastic tumor (IMT), normally referred to as inflammatory pseudotumor, is a fairly rare condition. Fine needle aspiration cytology (FNAC) of IMT has only rarely been reported. Here, we describe one such case of pulmonary inflammatory myofibroblastic tumor. A 30-year-old man presented with a 2.8cm-sized mass in his lung. Chest CT revealed a well defined, poorly enhancing mass. FNAC showed some fascicular or swirled clusters of spindle cells, admixed with occasional inflammatory cells and foamy histiocytes. The majority of the tumor cells evidenced bland, elongated nuclei, but infrequent pleomorphic nuclei. Some of the tumor cells evidenced nuclear grooves and intranuclear inclusions. Although the cytological differentiation of IMT from malignant lesions is not immensely problematic, due to the general paucity of cytological and nuclear atypia, a definite cytological diagnosis of IMT cannot be rendered simply by FNAC. Therefore, a diagnosis of IMT may be suggested via exclusive diagnosis.

Epstein-Barr virus-associated Inflammatory Pseudotumor-like Follicular Dendritic Cell Tumor in the Spleen of a Patient with Diffuse Large B Cell Lymphoma: A Case Report and Review of the Literature.: Sun Och Yoon, Hyoungsuk Ko, Baek hui Kim, Ghee Young Kwon, Yoon Kyung Jeon, Chul Woo Kim; Korean J Pathol. 2007;41(3):198-202.

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Abstract PDF: We report a case of an Epstein-Barr virus (EBV)-associated inflammatory pseudotumor-like follicular dendritic cell tumor (IPT-like FDC tumor). The tumor occurred in the spleen of a 64-year-old woman with a history of a diffuse large B-cell lymphoma (DLBCL) of neck nodes that presented four years ago. The splenectomy specimen revealed a 5 cm-sized, tan-colored and well-circumscribed mass. Histologically, spindle or ovoid cells with large vesicular nuclei were admixed with abundant inflammatory cells. Immunohistochemically, spindle cells were positive for FDC marker CD35, but negative for CD20, CD30 and ALK. EBV was detected almost exclusively in spindle cells by EBER in situ hybridization. IPT-like FDC tumors are rare, and are recognized as a distinctive clinicopathologic variant of FDC tumors. Among only 18 similar cases reported in the English language literature, the present case is the first case of a patient with a history of DLBCL.

Follicular Lymphoma with Monoclonal Plasma Cell Differentiation: A Case Report.: Hyun chul Kim, Young Seok Lee, Jung woo Choi, Ae ree Kim, Bom Woo Yeom, Han kyeom Kim, In sun Kim; Korean J Pathol. 2006;40(2):151-155.

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Abstract PDF: We present a case of recurrent follicular lymphoma with an extensive plasma cell component involving infra-auricular lymph nodes in a 64 year-old woman. Immunohistochemical staining showed a strongly positive reaction of the follicles with CD20, bcl-2, bcl-6, CD10 and CD21 on the first biopsy specimen. The intrafollicular and interfollicular plasma cells showed monoclonality for IgG heavy chain and lambda light chain. The histological and immunohistochemical findings in the recurrent tumor were identical with those of the original. Discussion is focused on the importance of the differential diagnosis between reactive lymphoid hyperplasia and other lymphomas having plasmacytic differentiation.

Inflammatory Myofibroblastic Tumor of the Stomach: A Case Report.: Joon Mee Kim, In Suh Park, Lucia Kim, Suk Jin Choi, Jee young Han, Young Chae Chu, Kyoung Rae Kim; Korean J Pathol. 2006;40(2):148-150.

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Abstract PDF: Inflammatory myofibroblastic tumor of the stomach is a rare tumor-like, benign disease with an uncertain pathogenesis. A 15-year-old male presented with epigastric pain. Endoscopic ultrasonography revealed a 2.3 cm sized ovoid intramuscular mass in the lower body of the stomach at the lesser curvature. Histologically, the tumor was composed of smooth muscle actin positive- and vimentin positive spindle cells and there were a large number of lymphocytes, plasma cells and histiocytes in the fibrotic background. The spindle cells were also positive for ALK1, but negative for EBV in situ hybridization.

Xanthomatous Pseudotumor of the Breast: A Brief Case Report.: Lee So Maeng, Se Jeong Oh, Kyoung Mee Kim, Anhi Lee, Chang Suk Kang; Korean J Pathol. 2005;39(5):345-347.

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Abstract PDF: Inflammatory (xanthomatous) pseudotumors of the breast are very rare and this case is the first reported case in Korea. A healthy, pregnant 29-year-old woman presented with a right breast mass. Ultrasonography of the breast revealed a 1.8 x 1.9 x 1.1 cm, sized lobulated, partially spiculated mass in the upper and outer quadrant of the breast. Macroscopically, the mass was well circumscribed, bright yellow, and lobulated. Microscopically, the tumor was composed of foamy histiocytes with multifocal neutrophilic infiltration, accompanying chronic inflammatory cellul infiltration, fibrosis and ductal-lobular atrophy.

Inflammatory Pseudotumor of the Paratesticular Area: A Case Report.: Na Rae Kim, Seung Yeon Ha, Jae Gul Chung, Joungho Han; Korean J Pathol. 2004;38(3):208-211.

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Abstract PDF: Inflammatory pseudotumors of the paratesticular area are rare, and are often reported in the literature by various terms, e.g., proliferative funiculitis, inflammatory myofibroblastic tumor, pseudosarcomatous myofibroblastic proliferation and fibrous pseudotumor. This is one of the most common lesions of that region, and typically presents as a longstanding, painless scrotal mass. Here, we describe a 34 year-old man who has had a palpable scrotal mass for the past 10 years. The excised mass was composed of multiple conglomerated nodules, which had homogeneous rubbery cut surfaces. Histologically, each was a well circumscribed, but unencapsulated mass of hyalinized collagenous tissue interspersed with lymphoplasmacytic cells and lymphoid follicle formation. A small fraction of paucicellular spindle cells was positive for vimentin, smooth muscle actin and CD68. Ultrastructurally, abundant collagen fibrils were mixed with paucicellular spindle cells and inflammatory cells. These spindle cells had abundant rough endoplasmic reticula and myofilaments with focal densities, indicating myofibroblastic differentiation.

Inflammatory Myofibroblastic Tumor (InflammatoryFibrosarcoma) of the Lung: A Case Report.: Minseob Eom, Tae Heon Kim, Jin Kyu Park, Kwang Hwa Park, Soon Hee Jung, Kwang Gil Lee; Korean J Pathol. 2003;37(4):291-295.

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Abstract PDF: Inflammatory myofibroblastic tumor, also known as inflammatory fibrosarcoma, has been frequently diagnosed as inflammatory pseudotumor. Although there are six cases reported as inflammatory pseudotumors or inflammatory myofibroblastic tumors in the lung, no cases of pulmonary inflammatory myofibroblastic tumor with features of inflammatory fibrosarcoma have been reported in Korea. We experienced a case of inflammatory myofibroblastic tumor (inflammatory fibrosarcoma)characterized by high cellularity, severe nuclear pleomorphism, necrosis, or increased mitoticcounts. A 31-year-old male patient with a solitary pulmonary nodule on the routine chest x-rayreceived a right lower lobectomy. The tumor was an ovoid solid mass with multifocal necrosis, showing diffuse irregular proliferation of spindle cells with high cellularity and focal nuclear pleomorphism, admixed with dense lymphoplasmacytic cells. Although spindle cells are focally immunoreactivefor smooth muscle actin, the ultrastructural examination failed to demonstrate smooth muscledifferentiation. In cases of inflammatory myofibroblastic tumor (inflammatory fibrosarcoma), a completeexcision and close follow-up without radical surgery, radiation, or chemotherapy are needed.

Endobronchial Inflammatory Pseudotumor: A case report.: Seoung Wan Chae, Young Hee Choi, Hye KYung Ahn, Young Euy Park; Korean J Pathol. 1994;28(2):213-215.

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Abstract: Inflammatory pseudotumors of the bronchus have been reported infrequently. Histologic diagnosis remains difficult because of their Polymorphic histologic characteristics and confusing terminology, which are also the problems in the diagnosis of intrapulmonary pseudotumors. We report a case of inflammatory pseudotumor in the left main bronchus which occurred in a 37-year-old man. Histologically, the main portion of the tumor was composed of pale eosinophilic spindle shaped cells covered by respiratory epithelium with squamous metaplasia. Mononuclear inflammatory cells, including plasma cells but acute inflammatory cells were also present in the superficial portion.

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