Journal of Pathology and Translational Medicine

Volume 45(0); July 2011

Case Reports

High-Grade Myxofibrosarcoma Showing Pleomorphic Hyalinizing Angiectatic Tumor-like Appearance: A Case Report.: Mi Seon Kang, Hye Jung Jo, Sung Hee Son; Korean J Pathol. 2011;45:S1-S4.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S1

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Myxofibrosarcomas (MFSs), which consist of multiple nodules with a variable cellular population in a myxoid matrix, are primarily located in subcutaneous tissue. Pleomorphic hyalinizing angiectatic tumors (PHATs) are rare soft-tissue tumors characterized by a proliferation of highly pleomorphic spindle or polygonal cells and abundant ectatic blood vessels in cellular or myxoid stroma. We present here an unusual case of a high-grade MFS with a PHAT-like appearance. A 67-year-old man presented with an asymptomatic subcutaneous mass in the right forearm. The tumor had myxoid, hypo-, and hypercellular areas with highly pleomorphic spindle or polygonal tumor cells that showed frequent mitoses and nuclear pseudoinclusions. Foci of punctuate necrosis and inflammatory infiltration were present throughout the tumor, and abundant ectatic, thick-walled vessels containing blood clots were noted. The tumor cells were immunohistochemically positive for vimentin but negative for CD34, S-100 protein, smooth muscle actin, desmin, and bcl-2.

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Children's kinetic family drawings and their internalizing problem behaviors
Jin Kyung Kim, Joo Hyun Suh
The Arts in Psychotherapy.2013; 40(2): 206. CrossRef

Odontogenic Keratocyst Associated with an Ectopic Tooth in the Maxillary Sinus: A Report of Two Cases and a Review of the Literature.: Hyuk Il Kwon, Won Bong Lim, Ji Sun Kim, Young Jong Ko, In Ae Kim, Suk Ja Yoon, Yoo Duk Choi, Hong Ran Choi, Ok Joon Kim; Korean J Pathol. 2011;45:S5-S10.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S5

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Abstract PDF

Odontogenic keratocysts are benign intraosseous tumors of odontogenic origin that occur most commonly in the jaw. In particular, they have a predilection for the angle and ascending ramus of the mandible. In contrast, odontogenic keratocysts arising in the maxillary sinus are relatively rare. Two such cases are reported herein. In addition, the English literature that concerns odontogenic keratocysts of the maxillary sinus is reviewed.

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Diagnostic Dilemma of Odontogenic keratocyst Mimicking a Dentigerous Cyst: A Case Report
Georgia Benitha, Syed Wali Peeran
International Journal of Head and Neck Pathology.2023; 6(2): 6. CrossRef
Keratocystic odontogenic tumor associated with impacted maxillary third molar extending to the antrum: A challenging diagnosis
Roozbeh Pahlevan, Farnaz Keyhanlou, Sahar Fazel, Fatemeh Shahsavari
Human Pathology: Case Reports.2019; 15: 59. CrossRef
Current Concepts and Occurrence of Epithelial Odontogenic Tumors: II. Calcifying Epithelial Odontogenic Tumor Versus Ghost Cell Odontogenic Tumors Derived from Calcifying Odontogenic Cyst
Suk Keun Lee, Yeon Sook Kim
Korean Journal of Pathology.2014; 48(3): 175. CrossRef
Keratocystic Odontogenic Tumor with an Ectopic Tooth in Maxilla
Basavaraj T. Bhagawati, Manish Gupta, Gaurav Narang, Sharanamma Bhagawati
Case Reports in Dentistry.2013; 2013: 1. CrossRef
A Large Keratocystic Odontogenic Tumor
Saurabh Jolly, Jeevan Lata
World Journal of Dentistry.2013; 4(2): 138. CrossRef

Ectopic Epididymis in Testicular Appendices: Report of Two Cases.: Hyun Soo Kim, Gou Young Kim, Hyung Lae Lee, Youn Wha Kim, Sung Jig Lim; Korean J Pathol. 2011;45:S11-S14.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S11

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Abstract PDF: We report two cases of ectopic epididymal ducts and efferent ductules in the testicular appendices (TAs) of adult men with normally descended testes. In both cases, a sessile TA was incidentally found at the upper pole of the right testis during the scrotal hydrocelectomy. Microscopically, a few closely arranged tubules were detected within the TA. In the first case, the tubules were lined with a pseudostratified columnar epithelium with numerous, long microvilli, and were surrounded by a smooth muscle coat. In contrast, in the second case, the tubules had a wavy luminal surface, because ciliated columnar cells alternated with groups of cuboidal cells. In both cases, strong CD10 immunoreactivity was observed in the luminal border of the lining epithelium. Surgical pathologists should be aware of the presence of both ectopic epididymal ducts and efferent ductules that can occur in TAs, in order to avoid misinterpretation as transected, functional reproductive structures.

The Wolf-Hirschhorn Syndrome in Fetal Autopsy: A Case Report.: Sun Ju Byeon, Jae Kyung Myung, Sung Hye Park; Korean J Pathol. 2011;45:S15-S19.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S15

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Abstract PDF

Wolf-Hirschhorn syndrome (WHS) is a malformation associated with a hemizygous deletion of the distal short arm of chromosome 4. Herein we report a fetal autopsy case of WHS. A male fetus was therapeutically aborted at 17(+0) weeks gestational age, due to complex anomaly and intrauterine growth retardation, which were found in prenatal ultrasonography. His birth weight was 65 g. Mild craniofacial dysmorphism, club feet, bilateral renal hypoplasia, edematous neck, and left diaphragmatic hernia of Bochdalek were found on gross examination. On GTG-banding, the fetus revealed 46,XY,add(4p) karyotype and the mother revealed 46,XX,t(4;18)(p16;q21.1), with normal karyotype of the father. Array comparative genomic hybridization performed on the autopsied lung tissue revealed loss of 4p16.2-->4pter and gain of 18q21.1-->18qter, suggesting 46,XY,der(4)t(4;18)(p16.2;q21.1)mat of fetal karyotype. This suggested deletion of 4p, compatible with WHS inherited from the mal-segregation of a maternal translocation t(4;18)(p16.2;21.1). Therefore, our fetus was both genotypically and phenotypically compatible with WHS.

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A Case of Wolf-Hirschhorn Syndrome with Periventricular Nodular Heterotopia Presenting with Status Epilepticus
Bong Sul Suh, Keun Hyeok Ko, Kon Hee Lee, Tae-Jung Sung
Neonatal Medicine.2015; 22(4): 233. CrossRef
Rhabdoid glioblastoma is distinguishable from classical glioblastoma by cytogenetics and molecular genetics
Sun-Ju Byeon, Hwa Jin Cho, Hae Woon Baek, Chul-Kee Park, Seung-Hong Choi, Se-Hoon Kim, Hee Kyung Kim, Sung-Hye Park
Human Pathology.2014; 45(3): 611. CrossRef

Primary Myoepithelioma of the Testis: A Case Report.: Seong Muk Jeong, Jung Hee Lee, Won Young Park, Na Ri Shin, Woo Gyeong Kim, Gi Yeong Huh, Chang Hun Lee, Hong Koo Ha; Korean J Pathol. 2011;45:S20-S24.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S20

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Abstract PDF

Myoepitheliomas are well-established to occur in the salivary glands, but they have also been described in the breast, upper aerodigestive tract, skin, and soft tissues. We report here on a unique case of primary myoepithelioma that occurred in the right testis of a 28-year-old man. The tumor was entirely confined to the testis and it was clearly separated from the epididymis. Histopathology revealed mixed architectural patterns in which the reticular areas merged into the chondromyxoid stroma. The tumor cells, which were focally immunoreactive to pancytokeratin and S-100 protein, were round to ovoid and spindly arranged in cords, strands, and fascicles. They showed mild nuclear pleomorphism, sparse mitotic figures and a low Ki-67 proliferative index. There was no ductal differentiation in the tumor. To the best of our knowledge, there has been only one case report of a primary testicular myoepithelioma in the English medical literature.

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Primary cutaneous myoepithelial carcinoma: a case report and review of the literature
Markus Winther Frost, Torben Steiniche, Tine Engberg Damsgaard, Lars Bjørn Stolle
APMIS.2014; 122(5): 369. CrossRef
Imprint Cytology of Soft Tissue Myoepithelioma: A Case Study
Seok Ju Park, Ae Ri Kim, Mi Jin Gu, Joon Hyuk Choi, Duk Seop Shin
Korean Journal of Pathology.2013; 47(3): 299. CrossRef

Cystic Nephroma: A Case Report and Comparing Literature Review with Mixed Epithelial and Stromal Tumor of Kidney.: Hyun Jung Kim, Choong Hee Noh, Giyoung Kwon, Eunah Shin, Jung Yeon Kim, Kyeongmee Park; Korean J Pathol. 2011;45:S25-S28.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S25

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Abstract PDF: Cystic nephroma (CN) is a benign cystic neoplasm composed of mixed epithelial and stromal elements. Less than 200 cases have been reported. We had a patient, a 41-year-old woman, who had a huge typical CN. The patient was admitted for a right renal mass that was found incidentally. On laparaoscopic right nephrectomy, there was an encapsulated 7 cm multilocular cystic mass at the upper pole. Microscopically, the cystic wall was lined by a single layer of low cuboidal or hobnail epithelium without a solid area. The thin septa were composed of bland, ovarian type spindle cells. The main differential diagnoses were mixed epithelial and stromal tumor (MEST), low grade multilocular renal cell carcinoma, and tubulocystic carcinoma. The results of immunohistochemical staining were cytokeratin 7/19(+/+) and CD10(-) in lining epithelium, estrogen receptor/progesterone receptor(+/+) in stromal cells. After surgery, she was free of recurrence for 10 months. We report this rare case and compare it with other cystic renal tumors, especially MEST.

Simultaneous Pancreatic Serous Microcystic Adenoma and Intraductal Papillary Mucinous Tumor of the Pancreas: A Case Report.: Hyoung Jong Kwak, Young Kon Kim, Baik Hwan Cho, Woo Sung Moon; Korean J Pathol. 2011;45:S29-S31.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S29

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Abstract PDF: Serous cystadenomas of the pancreas account for approximately a third of pancreatic cystic neoplasms. Their coexistence with a second tumor is extremely rare. We now report a case of a serous microcystic adenoma combined with an intraductal papillary mucinous tumor of the pancreas in a 69-year-old man. Abdominal computed tomography scans demonstrated an incidental cystic mass in the body with cystic dilatation of the duct in the head of the pancreas. Central pancreatectomy with pancreatico-jejunostomy, and cyst excision of the pancreatic head were performed. Histologic examination demonstrated a serous microcystic cystadenoma in the body coexisting with an intraductal papillary mucinous adenoma in the head of the pancreas. This case study highlights the importance of careful intra-operative and pathologic examination for synchronous pancreatic tumors.

Diffuse Pulmonary Meningotheliomatosis: A Case Report.: Jungsuk An, Heejung Park, Joungho Han, Tae Sung Kim, Yong Soo Choi, Moon Seok Choi, Sang Won Um; Korean J Pathol. 2011;45:S32-S35.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S32

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Abstract PDF

Diffuse pulmonary meningotheliomatosis (DPM) is an extremely rare condition. We herein report a unique case of DPM in a 54-year-old woman with a previous history of hepatocellular carcinoma. A chest computed tomography showed diffuse bilateral nodular infiltration, suggesting miliary spread of metastatic hepatocellular carcinoma. The patient underwent a video-assisted thoracoscopic surgery for diagnostic purposes. The cut surface of the lung specimen showed multiple dispersed small nodules, consisting of variably sized nests or whorls of bland epithelioid cells often along the walls of alveolar septa or in a perivascular network within the alveolar interstitium. The tumor cells showed immunoreactivity for epithelial membrane antigen, vimentin, and progesterone receptor. DPM should be included in the differential diagnosis of diffuse multiple small nodules or a reticular pattern in the radiologic studies.

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Minute Pulmonary Meningothelial-Like Nodules Simulating Hematogenous Lung Metastasis: A Case Report
Sang Kook Lee, Gi Jeong Kim, Young Jae Kim, Ah Young Leem, Eu Dong Hwang, Se Kyu Kim, Joon Chang, Young Ae Kang, Song Yee Kim
Tuberculosis and Respiratory Diseases.2013; 75(2): 67. CrossRef

IgG4-Related Sclerosing Sialadenitis: Report of Three Cases.: Ji Seon Bae, Joo Young Kim, Sang Hak Han, Seung Ho Choi, Kyung Ja Cho; Korean J Pathol. 2011;45:S36-S40.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S36

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Abstract PDF

Chronic sclerosing sialadenitis, Mikulicz disease or Kuttner tumor has been recently recognized as a spectrum of IgG4-related sclerosing disease. IgG4-related disease is characterized by a high serum IgG4 level and tissue infiltration of IgG4-positive plasmacytes. We report three cases of chronic sclerosing sialadenitis with variably associated systemic involvement. All patients presented with a submandibular mass or swelling, and all the resected submandibular glands showed diffuse lymphocytic infiltration, lymphoid follicles, and septal fibrosis. Two of the specimens revealed numerous IgG-positive plasma cells, most of which were IgG4-positive on immunohistochemical staining. One of them was associated with dacryoadenitis and hypophysitis. The other patient had ureterorenal lesions. Immunohistochemical study was unavailable in remaining one case, but the histologic features along with elevated IgG level and associated pancreatitis supported the diagnosis. All patients received steroid therapy postoperatively and are doing well. Salivary gland involvement in IgG4-related fibrosclerosis should be recognized in systemic medical pathology.

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A Case of Immunoglobulin G4-Related Sialadenitis and Dacryoadenitis
Kyoung Kyu Lee, Hun Po Cho, Young Min Lee, Jun Ho Park
Korean Journal of Otorhinolaryngology-Head and Neck Surgery.2013; 56(7): 444. CrossRef

Localized Primary Thymic Amyloidosis Presenting as a Mediastinal Mass: A Case Report.: Sang Yun Ha, Jae Jun Lee, Heejung Park, Joungho Han, Hong Kwan Kim, Kyung Soo Lee; Korean J Pathol. 2011;45:S41-S44.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S41

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We herein describe a case of a 55-year-old healthy woman with localized primary thymic amyloidosis presented as a mediastinal mass, found incidentally by chest radiography. Computed tomography revealed a 4.1 cm soft tissue lesion with nodular calcification in the left anterior mediastinum. The resected specimen was a well-defined lobulating mass with calcification. Microscopically, the mass was consisted of amorphous eosinophilc hyalinized substances involving the thymus and intrathymic lymph nodes. These eosinophilic substances showed apple-green bi-refringence under polarized light after staining with Congo red. In immunohistochemical study, they were positive for kappa and lambda light chains and negative for amyloid A. There was no evidence of systemic amyloidosis in clinical investigations. A final diagnosis of localized primary thymic amyloidosis was made.

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EBUS-TBNA diagnosis of localised amyloidosis presenting as mediastinal lymphadenopathy
Avnee Kumar, Barathi Sivasailam, Ellen Marciniak, Janaki Deepak
BMJ Case Reports.2018; 11(1): e226619. CrossRef
Ocular Myasthenia Gravis Associated With Thymic Amyloidosis
Kristin O. Chapman, Debra M. Beneck, Marc J. Dinkin
Journal of Neuro-Ophthalmology.2016; 36(1): 50. CrossRef
Localized Thymic Amyloidosis Presenting with Myasthenia Gravis: Case Report
Seung-Myoung Son, Yong-Moon Lee, Si Wook Kim, Ok-Jun Lee
Journal of Korean Medical Science.2014; 29(1): 145. CrossRef

Esophageal Gland Duct Adenoma.: Yoonjung Kim, Yang Soon Park, Jei So Bang, Ji Yeon Kim, Young Hyeh Ko, Cheol Keun Park, Kyoung Mee Kim; Korean J Pathol. 2011;45:S45-S47.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S45

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Abstract PDF: Benign ductal or glandular neoplasms of the esophagus unrelated to Barrett esophagus are extremely rare. Only 9 cases have been reported in the English language literature. We now report a case of esophageal gland duct adenoma incidentally found in a 73-year-old man. A 0.8 cm-sized, polypoid submucosal lesion in the distal esophagus was removed. Histologically, the lesion was well circumscribed and consisted of several ducts or cysts with focal papillary configurations. Interstitial lymphocytic infiltration with germinal centers was also observed. The lining cells of ducts or cysts were composed of two layers: an inner intensely eosinophilic luminal duct cell layer and an outer myoepithelial cell layer that was accentuated by alpha-smooth muscle actin. There was no significant nuclear atypia or mitosis. Mucin production was occasionally observed in a few goblet cells. To the best of our knowledge, this is the first case of benign ductal or glandular neoplasm of the esophagus among Koreans.

Clinicopathologic Analysis of the Liver Explant with Severe Hepatitis A Virus Infection.: Joo Young Kim, Sung Gyu Lee, Shin Hwang, Ji Hoon Kim, Se Jin Jang, Eunsil Yu; Korean J Pathol. 2011;45:S48-S52.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S48

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Abstract PDF: The incidence of severe hepatitis A virus (HAV) infection has been increasing. However, clinicopathologic features of severe HAV infection that lead to liver transplantation (LT) have not been reported in Korea. We retrieved 16 LT cases with HAV infection during the last 3 years at Asan Medical Center, Seoul, Korea. Fifteen cases progressed to hepatic encephalopathy. Thirteen cases survived with or without complications, and three patients died of sepsis. The explanted liver showed massive or zonal necrosis with moderate to severe cholestasis. The zonal distribution of necrosis was frequently associated with endothelialitis of portal and/or central veins. Degenerative changes of hepatocytes were various in degree and distribution. Viral inclusions were suspected in two cases. Although HAV infection is usually confirmed by serological tests, significant venulitis of central and/or portal veins and viral inclusions, which are rarely observed, can suggest an HAV infection as a cause of massive hepatic necrosis of unknown mechanism.

In Situ Follicular Lymphoma Developed after Hodgkin Lymphoma.: Ho Sung Park, Sang Jae Noh, Jae Yong Kwak, Eun Kee Song, Myung Hee Sohn, Ho Lee, Woo Sung Moon, Kyu Yun Jang; Korean J Pathol. 2011;45:S53-S57.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S53

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Abstract PDF: In situ follicular lymphoma is a newly defined entity among the lymphoid neoplasms and is defined as architecturally normal-appearing lymph nodes and other lymphoid tissues that have one or more follicles that demonstrate bcl-2 overexpressing centrocytes and centroblasts, with or without a monomorphic cytologic appearance suggestive of follicular lymphoma. Here we present a case of in situ follicular lymphoma diagnosed during the follow-up after a complete response to the treatment of lymphocyte-rich classical Hodgkin's lymphoma. In our case, because only a few germinal centers contained bcl-2 overexpressing cells, we missed the diagnosis of in situ follicular lymphoma in the initial histological examination. We could establish the diagnosis only after performing bcl-2 immunostaining in the sequential biopsy. Therefore, we recommend that careful histological examination along with bcl-2 immunostaining is needed in patients with suspicious clinical findings.

Angiomyomatous Hamartoma of Popliteal Lymph Nodes Occurring in Association with Diffuse Pigmented Villonodular Synovitis of Knee.: Hyun Soo Kim, Ki Yong Na, Jae Hoon Lee, Nam Su Cho, Gou Young Kim, Sung Jig Lim; Korean J Pathol. 2011;45:S58-S61.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S58

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Abstract PDF: We report the first case of an angiomyomatous hamartoma (AH) of the popliteal lymph nodes (LNs) occurring in association with diffuse pigmented villonodular synovitis (PVNS) of the knee. AH is a rare benign vascular disease with a predisposition for the LNs of the inguinal region. Twenty-five cases of AH have been reported to date; however, the precise pathogenesis is still undetermined. In the present case, an open synovectomy revealed two of three popliteal LNs in close proximity to the extra-articular component of diffuse PVNS. These LNs demonstrated irregularly distributed thick-walled blood vessels in the hilum. These vessels extended into the medulla and cortex and were associated with haphazardly arranged smooth muscle cells in the sclerotic stroma. These findings are compatible with an AH. Our observations raise the possibility that AH of the popliteal LNs may represent an abnormal proliferative reaction against the inflammatory process caused by PVNS of the knee.

Bilateral Frontal Polymicrogyria: An Autopsy Case Report.: Yi Kyeong Chun, Jong Sun Choi, Je G Chi; Korean J Pathol. 2011;45:S62-S65.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.S1.S62

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Abstract PDF: Bilateral frontal polymicrogyria is a recently recognized syndrome characterized by symmetric polymicrogyria of both frontal lobes that presents with delayed motor and language development, spastic quadriparesis, and variable mental retardation. However, the postmortem findings of this syndrome are not fully elaborated. Here we describe an autopsy case of bilateral frontal polymicrogyria in a male fetus delivered at 22 weeks gestation due to extensive chorioamnionitis. The microscopic findings included a thinned cortical plate with fair neuronal maturation. There were no signs of neuronal damage and the white matter was unremarkable.

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