Journal of Pathology and Translational Medicine

Case Study

Unusual Histology of Eosinophilic Myenteric Ganglionitis: A Case Report: Hyekyung Lee, Dongwook Kang, Heejin Kim, Byungsun Cho, Jeho Jang; J Pathol Transl Med. 2017;51(3):320-324. Published online April 4, 2017; DOI: https://doi.org/10.4132/jptm.2016.09.07

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Eosinophilic myenteric ganglionitis is a disorder characterized by infiltration of the Auerbach myenteric plexus by eosinophils. As a cause of chronic intestinal pseudo-obstruction (CIPO), eosinophilic myenteric ganglionitis has been rarely reported and the majority of the reported cases in the literature were children. We experienced a case of eosinophilic myenteric ganglionitis associated with CIPO in a 53-year-old female patient. Histologic examination of the resected descending colon showed moderate eosinophilic infiltrates with hypogangliosis in the myenteric plexus. Immunohistochemical study revealed increased number of CD4-positive lymphocytes and stronger but scantier glial fibillary acid protein expression in the inflamed myenteric plexus.

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Eosinophilic myenteric ganglionitis: A case in a 14‐year‐old‐male
Anthony Price, Tandis Rastegarlari, Sanober Khowaja, Kade Thompson, Arian P. Lahiji, Michelle M. Felicella, Jing He, Annie Goodwin
JPGN Reports.2024; 5(3): 389. CrossRef
Eosinophilic Myenteric Ganglionitis Presenting as Sigmoid Volvulus: A Brief Report
Alyson Kim, Jeffrey L. Roberson, Lillias H. Maguire, Bo Jian, Nicole M. Saur
The American Surgeon™.2023; 89(11): 5021. CrossRef
Histological characteristics of eosinophilic myenteric ganglionitis: an under-recognised cause of chronic intestinal pseudo-obstruction
Yoichi Akazawa, Takuo Hayashi, Tsuyoshi Saito, Koichiro Niwa, Hirohiko Kamiyama, Noriko Sasahara, Kazuhiro Sakamoto, Akihito Nagahara, Takashi Yao
Virchows Archiv.2019; 474(3): 395. CrossRef
Colonic Pseudo-obstruction With Transition Zone: A Peculiar Eastern Severe Dysmotility
Eun Mi Song, Jong Wook Kim, Sun-Ho Lee, Kiju Chang, Sung Wook Hwang, Sang Hyoung Park, Dong-Hoon Yang, Kee Wook Jung, Byong Duk Ye, Jeong-Sik Byeon, Suk-Kyun Yang, Hyo Jeong Lee, Chang Sik Yu, Chan Wook Kim, Seong Ho Park, Jihun Kim, Seung-Jae Myung
Journal of Neurogastroenterology and Motility.2019; 25(1): 137. CrossRef

Original Article

Eosinophils in Colorectal Neoplasms Associated with Expression of CCL11 and CCL24: Hyuck Cho, Sung-Jig Lim, Kyu Yeoun Won, Go Eun Bae, Gou Young Kim, Ji Won Min, Byeong-joo Noh; J Pathol Transl Med. 2016;50(1):45-51. Published online December 14, 2015; DOI: https://doi.org/10.4132/jptm.2015.10.16

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Abstract PDF

Background
A decrease in the number of tissue eosinophils is known to reflect the malignancy potential of neoplastic lesions and even prognosis. Increased levels of the chemokines CCL11 and CCL24 in serum and tissue are also known to have diagnostic value as serum tumor markers or prognostic factors. The aim of this study was to evaluate the correlation between the degree of tissue eosinophilia and the expression of these chemokines in the glandular and stromal cells of colorectal neoplastic lesions ranging from benign to malignant tumors. Methods: We counted the number of infiltrating eosinophils in neoplastic lesion tissue and we evaluated the expression of CCL11 and CCL24 in glandular cells and stromal cells by immunohistochemical staining. Results: The results showed that the number of eosinophils decreased significantly and the expression of CCL11 and CCL24 in glandular cells decreased with tumor progression, whereas the stromal expression of CCL11 and CCL24 appeared to increase. Conclusions: The discrepancy in CCL11 and CCL24 expression between glandular cells and stromal cells might shed light on how colorectal cancer evades the immune system, which would enable further development of immunotherapies that target these chemokines. Further research on eosinophil biology and the expression pattern of chemokines in tumor cells is needed.

Citations

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Eosinophilia in cancer and its regulation by sex hormones
Sandeep Artham, Ching-Yi Chang, Donald P. McDonnell
Trends in Endocrinology & Metabolism.2023; 34(1): 5. CrossRef
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Parisa Maleki Dana, Fatemeh Sadoughi, Russel J. Reiter, Bahman Yousefi, Zatollah Asemi
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Monoclonal Antibodies in Immunodiagnosis and Immunotherapy.2023; 42(2): 68. CrossRef
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Ayman M. Farouk, Mona K. ElDeeb, Mona H. Kandil, Noha A. ElBanna, Mohamed M. Shamseya, Amel S. Elsedafy, Nevine L. Micheal, Mohamed A. Selimah
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Case Study

CD30-Positive T-Cell Lymphoproliferative Disease of the Oral Mucosa in Children: A Manifestation of Epstein-Barr Virus-Associated T-Lymphoproliferative Disorder: Mineui Hong, Young Hyeh Ko; J Pathol Transl Med. 2015;49(6):525-530. Published online September 30, 2015; DOI: https://doi.org/10.4132/jptm.2015.07.13

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Abstract PDF

Eosinophilic ulcer of the oral mucosa (EUOM) is a very rare, benign, self-limiting ulcerative lesion of the oral cavity of unknown pathogenesis, and belongs to the same spectrum of CD30⁺ T-cell lymphoproliferative disease (LPD) of the oral mucosa. The etiology and pathogenesis of the disease are unknown. We report two cases in children who were initially diagnosed with EUOM and CD30⁺ T-cell LPD, respectively. However, retrospective analysis revealed that a majority of infiltrated atypical T cells were positive for Epstein-Barr virus (EBV). The present cases suggest that the pathogenesis and etiology of EUOM or CD30⁺ T-cell LPD occurring in children are different from those in adults. EUOM or CD30⁺ T-cell LPD in children is a manifestation of EBV-positive T-cell LPD, and should therefore be distinguished from the disease in adults.

Citations

Citations to this article as recorded by

Pediatric oral Epstein-Barr virus associated self-remitting CD30+ lymphoproliferative disorder: A distinct entity
Ziv Schwartz, Robert B. Bowe, Morton Coleman, Cynthia M. Magro
Annals of Diagnostic Pathology.2018; 37: 57. CrossRef

Case Reports

Extranodal NK/T Cell Lymphoma Accompanied by Heavy Eosinophilic Infiltration and Peripheral Blood Eosinophilia, Involving Skeletal Muscles.: Jin Ho Paik, Yoon Kyung Jeon, Heounjeong Go, Chul Woo Kim; Korean J Pathol. 2011;45:S70-S74.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S70

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Abstract PDF

The patient was a 52-year-old female with swelling in both lower legs and peripheral blood eosinophilia. Biopsy specimen revealed the heavy infiltration of eosinophils with sparse small lymphocytes showing mild atypia. The diagnosis was Kimura disease. The symptoms including eosinophilia were relieved by steroid treatment. At 17 months from initial biopsy, the patient developed swelling of the buttock. At 25 months, fever and dyspnea with multiple lung nodules developed. Wedge resection revealed multiple aggregates of CD3(+), CD56(+), Epstein-Barr virus(+) large atypical lymphocytes with necrosis. The patient was finally diagnosed with extranodal NK/T cell lymphoma (NKTL). Epstein-Barr virus in situ hybridization retrospectively performed on the previous biopsies demonstrated Epstein-Barr virus infection in small CD3(+) lymphocytes. The patient expired after 26 months despite chemotherapy. Blood eosinophilia correlated well with disease activity during the clinical course. This case shows not only unusual histologic features, which hampered the correct diagnosis, but also a unique clinical manifestation of NKTL.

Citations

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A case of extranodal NK/T cell lymphoma suspected to be focal myositis of the masseter muscle
Junya YAMASHITA, Daisuke TAKEDA, Tatsuya SHIRAI, Kaito URYU, Nanae YATAGAI, Masaya AKASHI
Japanese Journal of Oral and Maxillofacial Surgery.2025; 71(1): 33. CrossRef
Muscular involvement of extranodal natural killer/T cell lymphoma misdiagnosed as polymyositis: A case report and review of literature
Li-Hui Liu, Qing Huang, Yun-Hai Liu, Jie Yang, Han Fu, Lin Jin
World Journal of Clinical Cases.2020; 8(5): 963. CrossRef
Extranodal natural killer/T-cell lymphoma with paraneoplastic eosinophilic myositis
Jayati Mallick, Jasmine Zain, Dennis D. Weisenburger
Human Pathology: Case Reports.2020; 21: 200391. CrossRef
Extranodal NK/T-cell Lymphoma Mimicking Granulomatous Myositis
Norihiko Kawaguchi, Rumiko Izumi, Masahiro Kobayashi, Maki Tateyama, Naoki Suzuki, Fumiyoshi Fujishima, Juichi Fujimori, Masashi Aoki, Ichiro Nakashima
Internal Medicine.2019; 58(2): 277. CrossRef
Uveitis and Myositis as Immune Complications in Chemorefractory NK/T-Cell Nasal-Type Lymphoma Successfully Treated with Allogeneic Stem-Cell Transplant
Maria José Gómez-Crespo, Aránzazu García-Raso, Jose Luis López-Lorenzo, Teresa Villaescusa, María Rodríguez-Pinilla, José Fortes, Cristina Serrano, Salma Machan, Pilar Llamas, Raúl Córdoba
Case Reports in Hematology.2016; 2016: 1. CrossRef
Prognostic implications of CD30 expression in extranodal natural killer/T-cell lymphoma according to treatment modalities
Wook Youn Kim, Soo Jeong Nam, Sehui Kim, Tae Min Kim, Dae Seog Heo, Chul-Woo Kim, Yoon Kyung Jeon
Leukemia & Lymphoma.2015; 56(6): 1778. CrossRef
Unusual case of metachronous EBV‐associated B‐cell and NK/T‐cell lymphoma mimicking polymyositis‐diagnostic challenges and pitfalls
Esther H.L. Chan, Suat‐Jin Lu, Fredrik Petersson, Kong‐Bing Tan, Wee‐Joo Chng, Siok‐Bian Ng
American Journal of Hematology.2014; 89(1): 110. CrossRef
CD30+ extranodal natural killer/T-cell lymphoma mimicking phlegmonous myositis: A case report
YAN-JIA YANG, YA-XIN LI, YAN-BIN LIU, MEI YANG, KAI LIU
Oncology Letters.2014; 7(5): 1419. CrossRef

Eosinophilic Fasciitis Associated with Overlying Intraepidermal Blister Formation: A Case Report.: Na Rae Kim, Dong Hae Chung, Seung Yeon Ha; Korean J Pathol. 2009;43(5):478-481.; DOI: https://doi.org/10.4132/KoreanJPathol.2009.43.5.478

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Abstract PDF: Eosinophilic fasciitis is a scleroderma-like disease and it may present with paraneoplastic syndrome or as an isolated form of the disease. We report here on a case of eosinophilic fasciitis in a 20-year-old woman who presented with an abrupt onset of subcutaneous limb swelling and peripheral eosinophilia. Pathologically, the specimen was characterized by acute inflammation and thickening of the collagen bundles in the reticular dermis and superficial muscle fascia in addition to the overlying intraepidermal blisters that contained many eosinophils. Eosinophils, some lymphocytes and plasma cells were infiltrated in the superficial muscle fascia and subcutaneous fat. The diagnosis of eosinophilic fasciitis was confirmed by biopsy. It is intriguing that eosinophilic fasciitis showed the microscopic findings of intraepidermal blister with predominant inflammation, and the patient showed a good response to steroid therapy.

Original Articles

Eosinophilic Cellulitis (Wells' Syndrome).: Anhi Lee; Korean J Pathol. 1995;29(3):407-410.

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Abstract PDF: Eosinophilic cellulitis is a rare dermatosis first described by Wells, and characterized by recurrent episodes of sudden outbreaks of erythematous cutaneous swellings often painful or pruritic. Microscopically, the lesion shows diffuse tissue eosinophilia and fibrinoid flame figures, evolution of associated focal necrobiosis, and formation of focal microgranulomas associated with eosinophils. The cause is still unknown but the triggers which have been thought to precipitate the disease include insect bites, parasitic infections such as toxocara, onchocerciasis, nasopharyngeal carcinoma, rheumatoid arthritis and spider bites. However many cases are idiopathic. The author experienced a case of eosinophilic cellulitis of a 52-year-old woman with multiple cutaneous tender plaques of cellulitis for approximately 10 years with history of repeated remission and recurrent episodes. Biopsy was taken from ulcerated edematous nodule of inguinal region under the clinical impression of deep fungal infection, pyoderma gangrenosum and polyarteritis nodosa. Cultures for fungal and common organisms were negative. Histologically, the entire dermis was infiltrated by numerous eosinophils and scattered histiocytes. There were scattered flame figures showing necrobiotic foci in the collagen with accumulation of eosinophils, granulated free cosinophilic granules and histiocytes.

Immunohistochemical Study of the Expression of p53, Pan-ras, c-erbB-2 and PCNA in N-Nitrosomorpholine(NNM)-Induced Hepatocellular Carcinoma of Rats.: Ok Kyung Kim, Ryun Jo Shin; Korean J Pathol. 1995;29(6):727-739.

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Abstract PDF: The focus of this study was o aialyze the morphologic expression of p53, Pan-ras, c-erbB-2, and PCNA in preneoplastic and neoplastic liver lesions induced with NNM of rats. The development of hepatocellular tumors was investigated by histology and electron microscope in 65 Splague-Dawley rats administered with NNM in drinking water at low dose(5 mg/100 ml) and high dose(20 mg/100 ml). Three types of hepatocytic degeneration glycogenotic, eosinophilic and basophilic changes were followed by the appearance of hepatocellullar carcinoma. Hepatocellular carcinoma was increased in number and size according to NNM dosage and to duration of exposure. The histological classifications of hepatocelular carcinoma wer trabecular type, which was which was the most common, large eosinophilic, small cell, adenocarcinomatous and clear cell type. The expression of p53, Pan-ras, c-erbB-2 PCNA was examined by immunohistochemical stains. Eosinophilic degeneration revealed mild positivity at 18-26 weeks for expression of all oncogenic proteins studied and PCNA, whereas precancerous lesions showed variable expression from negative to moderate positivity on PCNA. Hepatocellular carcinoma lesions showed strong positivity for all stains and increased intensity during experimental period. These may indicate that chemical carcinogen produce hepatic eosinophilic degeneration and preCancerOus lesions by genetic mutation, resulting in hepatocellular carcinoma.

Case Reports

Chromophobe Renal Cell Carcinoma.: Yeong Jin Choi, Tae Kon Hwang, Youn Soo Lee, Eun Jung Lee, Seok Jin Kang, Byung Kee Kim, Sang In Shim; Korean J Pathol. 1999;33(4):259-266.

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Abstract PDF: We report 13 chromophobe renal cell carcinomas (10.8%) observed among 120 renal cell carcinomas in adults. The average age was 53 (range: 34-72) years old, and 6 were males and 7 females. The mean tumor size was 10 (range: 5-17) cm, mean nuclear grade 2.4, and mean Robson's stage was 1.9. There were two distinct histologic variants; typical variant (n=9) and eosinophilic variant (n=4). Both of them showed typical light microscopic features and positive reaction with Hale's colloidal iron and carbonic anhydrase II, a marker protein of intercalated cells of renal collecting ducts. A strong positive immunoreactivity for epithelial membrane antigen was noted in the cytoplasm in 12 of 13 tumors. Numerous microvesicles, 180~440 nm in diameter, were identified ultrastructurally. DNA aneuploidy was found in 3 out of 10 cases. Neither local recurrence nor metastasis have been identified during the following period of 4~144 (mean 48) months.

Chronic Hepatitis in the Idiopathic Hypereosinophilic Syndrome: A case report .: Kyeong Hee Kim, Hae Joung Sul, Sung Chul Jun, Dae Young Kang; Korean J Pathol. 1999;33(8):624-626.

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Abstract PDF: Chronic hepatitis associated with the idiopathic hypereosinophilic syndrome has been very rarely reported worldwide. Recently, we experienced a case of chronic hepatitis with piecemeal necrosis as the clinical feature of the idiopathic hypereosinophilic syndrome. The patient was a 49-year-old woman who complained of a mild fever, nausea, vomiting, and pain in the right upper quadrant. The eosinophil count of peripheral blood increased up to 14,020/microliter (64% of WBC). Liver biopsy specimen showed severe porto-periportal inflammation with marked eosinophilic infiltration and ballooning degeneration of hepatocytes. Corticosteroid therapy significantly normalized the eosinophil count of peripheral blood.

Original Articles

Histopathologic Studies of Muscle and Peripheral Nerve Following Ingestion of L-tryptophan in Rats.: Tae Sik Yoon, Tai Seung Kim, In Joon Choi, Jung Soon Shin; Korean J Pathol. 1993;27(4):318-327.

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Abstract PDF: The eosinophilia-myalgia syndrome associated with ingestion of L-tryptophan in man is defined by the CDC(1989) as follows: 1) eosinophil count more than 1,000 cells per microliter; 2) generalized myaligia(at some point during the course of illness) of severity sufficient to affect a patient's ability to pursue his or her usual daily activities; 3) no evidence of any infection(e.g., trichinosis) or neoplasm that would explain either the eosinophila or the myalgia. The pathologic findings of the eosinophilia-myalgia syndrome varies according to the degree of severity and types of inflammatory cells in the skeletal muscle. In order to simulate this syndrome in animals and further determine its histopathogenesis, L-tryptophan was administered to rats in the laboratory at various doses(25 mg/kg, 50 mg/kg, 150 mg/kg) over a set period of time. In this study, positive histopathologic findings were designated according to the inflammatory cell infiltration of the epimysium and epineurium. Most of the pathologic findings in the positive group were that of inflammatory cell infiltration composed mainly of eosinophils in the epimysial, epineurial connective and surrounding adipose tissues. Only a few necrotic muscle fibers were seen, and there was absence of any evidence of inflammatory cell inflitration in endoneurium or axonal degenerations. Of 59 rats which were given L-tryptophan, 27 rats(45.8%) met the criteria and were designated as belongintg to the positive group. Only 2 rats ingested with L-tryptophan(150 mg/kg) for 2 months and 4 months showed an eosinophil count more than 1,000 cells/microliter. The eosinophil count in the positive group showed significantly different levels when compared to the the negative group and control group. On the other hand, there were no significant differences in the electrodiagnostic study and serum CK, SGOT, SGPT level between the positive, negative and control groups. In summary, histopathologic findings similar to the eosinophilia-myalgia syndrome were inducible in rats followign the administration of L-tryptophan.

Eosinophilic Liver Abscess in Patients with Gastric Carcinoma.: Soon Won Hong, Ho Guen Kim, Chan Il Park, Sang In Lee; Korean J Pathol. 1993;27(1):27-33.

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Abstract PDF: Sixteen cases of heavy eosinophil infiltration or eosinophilic abscess of the liver in patients with gastric carcinoma were analyzed to draw attention to this interesting combination and to persue the pathogenetic mechanism. Peripheral blood eosinophilia and hepatic granuloma were found in only 5 and 4 cases, respectively. Neither the patients' stool nor the hepatic tissues disclosed any parasitic worms or eggs, although the skin tests for Clonorchis sinensis and Paragonimus westermani were positive in 2 cases. Among stomach carcinomas, early gastric cancer tended to have more eosinophils than advanced carcinoma, but was less frequently associated with the infiltration of mast cells. In the regional lymph nodes, there was no infiltration of eosinophils even in the presence of tumor metastasis. In the liver, none of the 16 cases had metastatic gastric carcinoma and mast cells were found in only 2 cases. The results suggest that heavy hepatic infiltration of eosinophils in gastric carcinoma patients is not of the parasitic or allergic cause, but of certain eosinophil chemotactic factor which may gain access to accumulate in the liver following released from the gastric carcinoma and transfered through the portal vein.

Case Report

Infantile Solitary Eosinophilic Granuloma of the Lymph Node: A case report.: Sun Hee Sung, Woo Ick Yang, Jae Ok Kim; Korean J Pathol. 1992;26(3):277-282.

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Abstract PDF: Infantile form of histiocytosis X is commonly presented as multiorgan desseminated form such as Letterer-Siwe disease. Lymph node involvement of histiocytosis X is usually accompanied by adjacent bone or skin lesion. Solitary nodal eosinophilic granuloma without evidence of other organ involvement is very rare. A case herein report is a 11 month-old female infant presented with fever and palpable both inguinal lymph nodes. There was neither skin lesion nor hepatosplenomegaly. Laboratory evaluation was within normal range except increased alkaline phosphatase and many neutrophils in urine. Radiologic examination revealed no remarkable bone lesions. And she showed good clinical outcome without evidence of other organ involvements. On microscopic examination of inguinal lymph node it was replaced by infiltration of histiocytes mainly along the sinusoid. Some of histiocytes showed morphologic features of "histiocytosis X cell" having nuclear grooves or multilobulation. Multinulceated giant cells were frequently see. Numerous eosinphils were also infiltrated and showed multifocal microabscess formation. Immunohistochemical staining revealed that majority of histiocytes were postitive for S-100 protein but multinucleated histriocytes, phagocytic histiocytes and those around the abscess were positive for macrophage marker, suck as CD68 and alpha-1-antichymotrypsin. Interestingly some histiocytes showed positivity for both S-100 protein and macrophage marker. These results suggest that histiocytosis X is proliferative disorder of phenotypically heterogenous population of histiocytes in contrast to the theory that it is a proliferative disorder of Langerhans cells.

Original Articles

Eosinophilic Granuloma of the Lung.: Sang Ae Yoon, Won Bo Jo, Yang Seok Chae, Kap No Lee; Korean J Pathol. 1992;26(3):270-276.

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Abstract PDF: Eosinophilic granuloma of the lung, first described by Farrinaci et al. in 1951, is rare. A 35-year-old male smoker presented with recurrent pneumothorax. Open thoracotomy with bleb resection and biopsy was performed. Microscopically there was histological changes consistent with typical eosinophilic granuloma and intertitial fibrosis. The Langerhans cells showed positive reaction for S-100 protein and typical Birbeck granules in their cytoplasm. A brief summary of histopathological aspect of this disease and a review of literature are presented.

A Pathological and Immunohistochemical Study of 9 Cases of Inflammatory Fibroid Polyp.: Nam Hoon Cho, Hyeon Joo Jeong, Ho Guen Kim; Korean J Pathol. 1989;23(1):20-28.

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Abstract PDF: We aimed to study the clinicopathologic features of inflammatory fibroid polyp by histological and immunohistochemical methods. The materials used in this study consisted of 9 cases of inflammatory fibroid polyp: 4 in the stomach, 4 in the small intestine and 1 in the cecum. The results were as follows: Females were affected more frequently than males and the average age was 45 years(range:27-61). In cases of gastric lesion, the size tended to be smaller, the mass was mainly located along the greater curvature side of antrum, and confined to the submucosa. However inflammatory fibroid polyp of the small intestine was over 2.5 cm in size, located along the antemesenteric border, and involved the proper muscle layer. In addition, intussusception was accompanied by polyp in 2 cases of small intestinal lesions. Histologically inflammatory fibroid polyps of the stomach were characterized by prominent lymphocytic infiltration and occasional onion-skinning of stromal cells, whereas plasmocytic infiltration was prominent in those of the small intestine. Main component cells comprising this lesion were confirmed to be fibroblasts by immunohistochemistry which revealed strong reactivity to vimentin in the cytoplasm of slindle cells.

Case Report

Hibernoma: A case report.: Hee Na Kim, Mi Kyung Jee, Seok Jin Gang, Byung Kee Kim, Sun Moo Kim; Korean J Pathol. 1988;22(3):353-355.

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Abstract PDF: Hibernoma is very rarely encountered and is expressed in the various names including "lipoma of brown fat", "fetal lipoma", and "lipoma of embryonic fat". In our knowledge, only about 50 cases have been reported in the literature of the Western world, and no case reports have been published in Korea. In May 1986, we experienced a case of hibernoma occuring in a 16 old years boy. On physical examination, a relatively well defined nodule was noted on the right scapular region. A total removal of the lesion was done under the clinical diagnosis of epidermal inclusion cyst. On gross examination, the specimen consists of three irregularly shaped portions of yellowish brown soft tissue, measuring up to 0.8 cm in greatest dimension. Microscopically, the tumor showed distinct lobular pattern separated by fibrovascular bands. The lobules were composed of multivacuolated or eosinophilic granular cells showing centrally located small unclei. A brief review of the literature was done.

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