Journal of Pathology and Translational Medicine

Case Study

Thyroid pathology, a clue to PTEN hamartoma tumor syndrome: Yurimi Lee, Young Lyun Oh; J Pathol Transl Med. 2023;57(3):178-183. Published online March 30, 2023; DOI: https://doi.org/10.4132/jptm.2023.03.04

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Phosphatase and tensin homolog (PTEN) hamartoma tumor syndrome (PHTS) is a hereditary disorder caused by germline inactivating mutations in the PTEN tumor suppressor gene. As a type of PHTS, Cowden syndrome is associated with abnormalities of the thyroid, breast, uterus, and gastrointestinal tract. A 52-year-old-woman visited the outpatient clinic of our endocrinology clinic with multiple thyroid nodules and Hashimoto's thyroiditis. Computed tomography imaging revealed a multinodular mass measuring up to 3.5 cm in the left thyroid lobe, causing laryngotracheal airway displacement. The total thyroidectomy specimen revealed multiple follicular adenomas and adenomatous nodules with lymphocytic thyroiditis and lipomatous metaplasia in the background. The patient was suspected of PTHS based on her thyroid pathology, family history, and numerous hamartomatous lesions of the breast, uterus, and skin. Her diagnosis was confirmed through molecular testing. This case demonstrates that pathologists must be well acquainted with thyroid pathology in PHTS.

Citations

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A clinical case of papillary thyroid cancer associated with a PTEN gene defect
R. A. Atanesyan, L. Ja. Klimov, T. M. Vdovina, G. A. Saneeva, E. I. Andreeva, I. A. Stremenkova, R. I. Arakelyan, I. K. Gasparyan
Rossiyskiy Vestnik Perinatologii i Pediatrii (Russian Bulletin of Perinatology and Pediatrics).2025; 69(6): 85. CrossRef
Pediatric cancer predisposition syndromes involving non-central nervous system solid pediatric tumors: a review on their manifestations with a focus on histopathology
B. Schurink, M. Reyes-Múgica, R. R. de Krijger
Virchows Archiv.2025; 486(1): 3. CrossRef
Dedifferentiated Leiomyosarcoma of the Uterine Corpus with Heterologous Component: Clinicopathological Analysis of Five Consecutive Cases from a Single Institution and Comprehensive Literature Review
Suyeon Kim, Hyunsik Bae, Hyun-Soo Kim
Diagnostics.2024; 14(2): 160. CrossRef
Case report: Rare oral manifestations in Cowden syndrome with PTEN mutation
Wei Yuan, Yanbin Liu, Haibin Sun, Ming Su, Lizheng Qin, Xin Huang
Frontiers in Oncology.2024;[Epub] CrossRef
Can thyroid histomorphology identify patients with PTEN hamartoma tumour syndrome?
Melad N Dababneh, Laura Rabinowitz, Gilman Plitt, Charis Eng, Christopher C Griffith
Histopathology.2024; 85(6): 929. CrossRef
A novel mutation in PTEN in anaplastic thyroid carcinoma: A case report
Yanli Zhao
Biomedical Reports.2024;[Epub] CrossRef

Case Report

Solitary Peutz-Jeghers type harmartomatous polyp in duodenum with gastric foveolar epithelium: a case report: Eugene Choi, Junghwan Lee, Youngsoo Park; J Pathol Transl Med. 2023;57(2):128-131. Published online January 10, 2023; DOI: https://doi.org/10.4132/jptm.2022.11.07

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Abstract PDF

Peutz-Jeghers type hamartomatous polyp is known to be associated with Peutz-Jeghers syndrome, which shows characteristic multiple hamartomatous polyp involvement in the gastrointestinal tract, combined with mucocutaneous symptom, familial history of Peutz- Jeghers syndrome or STK11/LTB1 mutation. However, some cases showing histologic appearance of the polyps discovered in Peutz- Jeghers syndrome while lacking other diagnostic criteria of the syndrome have been reported, and these are called solitary Peutz- Jeghers type polyps. Herein, we report a case of solitary Peutz-Jeghers type polyp covered with heterotopic epithelium. The patient was 47-year-old female without any mucocutaneous symptoms nor familial history of Peutz-Jeghers syndrome. Microscopic examination revealed Peutz-Jeghers type hamartomatous polyp in duodenum covered with gastric type foveolar epithelium. Considering the definition of hamartomatous polyp, which is, the abnormal overgrowth of the indigenous epithelial component, the histological feature of current case is noteworthy in a point that it shows proliferation of heterotopic component, rather than the indigenous component.

Citations

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A Solitary Peutz-Jeghers Hamartomatous Polyp in the Gastric Body: A Case Report
Noelia Madera, Noemí Acevedo, Carmen González-Peralta, Rafael Castro, Vismelis Mezquita-Luna
Cureus.2024;[Epub] CrossRef

Case Study

Multicystic Biliary Hamartoma of the Liver: Ji Soo Song, Sang Jae Noh, Baik Hwan Cho, Woo Sung Moon; Korean J Pathol. 2013;47(3):275-278. Published online June 25, 2013; DOI: https://doi.org/10.4132/KoreanJPathol.2013.47.3.275

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Abstract PDF

Multicystic biliary hamartoma (MCBH) is a very rare hamartomatous cystic nodule of the liver, which has recently been described as a new entity of a hepatic nodular lesion. We report a unique case of MCBH with a review of the literatures. A hepatic multicystic mass of segment 3 was detected in a 52-year-old male by abdominal computed tomography, and resection of this lesion was performed. Macroscopic examination revealed a 2.7×2.0 cm nodular mass with a multicystic honeycomb cut surface. Histologically, this lesion consisted of multiple dilated cystic ducts lined by biliary type epithelial cells, periductal glands and connective tissue, which included small amounts of hepatic parenchyma and blood vessels. Recognition of this unusual lesion is essential to avoid confusion with other cystic tumors of the liver, and to learn more about its natural history and response to treatment.

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Multicystic biliary hamartoma with long-term gradual enlargement treated by laparoscopic partial hepatectomy
Satoshi Nishiwada, Tetsuya Tanaka, Yuki Kirihataya, Takeshi Takei, Tomomi Sadamitsu, Masato Takano, Masayoshi Sawai, Atsushi Yoshimura
Clinical Journal of Gastroenterology.2025; 18(3): 527. CrossRef
Technical Considerations in EEG Source Imaging
Benjamin H. Brinkmann
Journal of Clinical Neurophysiology.2024; 41(1): 2. CrossRef
A Case of Multicystic Biliary Hamartoma with a Marked Peribiliary Gland Component Successfully Treated by Purely Laparoscopic Anatomical Liver Resection
Keita Kai, Takao Ide, Tomokazu Tanaka, Kumpei Yukimoto, Hiroyuki Irie, Hirokazu Noshiro, Shinichi Aishima
Journal of Gastrointestinal Cancer.2023; 54(3): 996. CrossRef
Characteristics of multicystic biliary hamartoma: A case report
Jia Lian, Lixia Sun, Yankai Yang, Jun Li, Ye Zhang, Guiqiu Liu, Weijuan Hu
Frontiers in Surgery.2023;[Epub] CrossRef
Recurrent sepsis in a patient with biliary hamartomas
Maria Beatriz Santos, Magda Ponta Garça, Bárbara Vieira, Paulo Ávila, Alexandra Freitas
European Journal of Case Reports in Internal Medicine.2023;[Epub] CrossRef
Hamartoma multiquístico de vías biliares
Victoria Carmona, Iago Justo, Yolanda Rodríguez-Gil, Alberto Marcacuzco, Carmelo Loinaz, Carlos Jiménez
Cirugía Española.2022; 100(12): 800. CrossRef
Multicystic Biliary Hamartoma With Xanthogranulomatous Inflammation on 18F-FDG PET/CT
Nahomi Shono, Yoichi Otomi, Hideki Otsuka, Takayoshi Shinya, Masafumi Harada
Clinical Nuclear Medicine.2022; 47(10): 882. CrossRef
Intrahepatic multicystic biliary hamartoma: A case report
Chen-Yu Wang, Fu-Yang Shi, Wei-Feng Huang, Yan Tang, Ting Li, Guo-Lin He
World Journal of Clinical Cases.2022; 10(26): 9361. CrossRef
A Case of Multicystic Biliary Hamartoma Treated with Left Medial Sectionectomy
Naomi KUROKI, Tomoaki TANAKA, Takanobu SUGASE, Syoji TANIGUCHI, Takashi GOTO, Rintaro KOGA, Takumi KIWAKI, Hiroyuki TANAKA
Nihon Rinsho Geka Gakkai Zasshi (Journal of Japan Surgical Association).2022; 83(2): 395. CrossRef
Multicystic biliary hamartoma
Victoria Carmona, Iago Justo, Yolanda Rodríguez-Gil, Alberto Marcacuzco, Carmelo Loinaz, Carlos Jiménez
Cirugía Española (English Edition).2022; 100(12): 800. CrossRef
Case Report: Incidentally Discovered a Rare Cystic Lesion of Liver: Multicystic Biliary Hamartoma
Wentao Mu, Peng Su, Shanglei Ning
Pathology and Oncology Research.2021;[Epub] CrossRef
Bile Duct Hamartoma Mimicking Liver Metastasis in Suspected Porcelain Gallbladder: a Case Report
Gautham Krishnamurthy, Harjeet Singh, Sravya Deepika Ganti, Ganga Ram Verma
Journal of Gastrointestinal Cancer.2019; 50(4): 1022. CrossRef
A variant of multicystic biliary hamartoma presenting as an intrahepatic cystic neoplasm
Tetsuro Tominaga, Takafumi Abo, Naoe Kinoshita, Tomonori Murakami, Yasunori Sato, Yasuni Nakanuma, Kenich Harada, Junichi Masuda, Takeshi Nagayasu, Atsushi Nanashima
Clinical Journal of Gastroenterology.2015; 8(3): 162. CrossRef
Hamartoma biliar multiquístico intrahepático: presentación de un caso clínico
María Jezabel Fernández-Carrión, Ricardo Robles Campos, Asunción López Conesa, Roberto Brusadín, Pascual Parrilla Paricio
Cirugía Española.2015; 93(9): e103. CrossRef
Intrahepatic Multicystic Biliary Hamartoma: Presentation of a Case Report
María Jezabel Fernández-Carrión, Ricardo Robles Campos, Asunción López Conesa, Roberto Brusadín, Pascual Parrilla Paricio
Cirugía Española (English Edition).2015; 93(9): e103. CrossRef
Multicystic biliary hamartoma: A report of a rare entity and a review of the literature
Rachel E. Beard, Eric U. Yee, Koenraad J. Mortele, Khalid Khwaja
International Journal of Surgery Case Reports.2014; 5(12): 919. CrossRef
Multicystic biliary hamartoma mimicking intrahepatic cholangiocarcinoma: report of a case
Tomoaki Yoh, Ryuji Okamura, Hiroyuki Nakayama, Xue Lin, Yuya Nakamura, Tatsushi Kato
Clinical Journal of Gastroenterology.2014; 7(5): 418. CrossRef

Case Report

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.

Original Article

Newly Formed Hepatic Masses in Children with Biliary Atresia after Kasai Hepatic Portoenterostomy.: Hye Jong Song, Yeon Lim Suh; Korean J Pathol. 2011;45(2):160-169.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.2.160

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Abstract PDF

BACKGROUND
This report describes the clinicopathologic findings of six hepatic masses that developed after Kasai hepatic portoenterostomy (HPE) in six patients with longstanding biliary atresia (BA).
METHODS
Hepatic masses were found in six of 55 pediatric patients who underwent liver transplantation for BA after Kasai HPE from 1997 to 2009. Clinicopathologic analysis was performed and immunohistochemical staining was carried out for CD34, smooth muscle actin (SMA) and cytokeratin 7.
RESULTS
Of the six hepatic masses, two were diagnosed as focal nodular hyperplasia (FNH)-like lesions, two were large regenerative nodules (LRN), one was a mesenchymal hamartoma (MH) and one was a cholangiocarcinoma. The immunohistochemical staining findings for SMA and CD34 were more prominent for the FNH-like nodules than for the cirrhotic background liver. Dysplastic biliary epithelium arising from intestinal metaplasia was found in the cholangiocarcinoma.
CONCLUSIONS
Our findings suggest that FNH-like lesions, LRNs and MH are the results of vascular hemodynamic changes after Kasai HPE and that cholangiocarcinoma is due to recurrent cholangitis after BA. All the lesions in this series must be included in the differential diagnosis of a newly formed hepatic mass in patients after portoenterostomy.

Citations

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Imaging Findings and Management Strategies for Liver Masses in Children with Predisposition Disorders: A Review by the Pediatric LI-RADS Group
Amy B. Kolbe, Michael R. Acord, Geetika Khanna, Cara E. Morin, HaiThuy N. Nguyen, Mitchell A. Rees, Esther Ro, Gary R. Schooler, Judy H. Squires, Ali B. Syed, Elizabeth R. Tang, Alexander J. Towbin, Adina Alazraki
RadioGraphics.2025;[Epub] CrossRef
Features of Nodules in Explants of Children Undergoing Liver Transplantation for Biliary Atresia
Ana M. Calinescu, Anne-Laure Rougemont, Mehrak Anooshiravani, Nathalie M. Rock, Valerie A. McLin, Barbara E. Wildhaber
Journal of Clinical Medicine.2022; 11(6): 1578. CrossRef
Biliary Atresia Patients With Successful Kasai Portoenterostomy Can Present With Features of Obliterative Portal Venopathy
Kalyani R. Patel, Sanjiv Harpavat, Zahida Khan, Sadhna Dhingra, Norma Quintanilla, Mihail Firan, John Goss
Journal of Pediatric Gastroenterology and Nutrition.2020; 71(1): 91. CrossRef

Case Reports

Sclerosing Angiomatoid Nodular Transformation (SANT) in Spleen: A Case Report.: Hyun Jung Lee, Song Yi Choi, Song Mei Huang, Ji Young Sul, Jin Man Kim; Korean J Pathol. 2011;45(1):111-114.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.1.111

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Abstract PDF

Sclerosing angiomatoid nodular transformation (SANT) of spleen is a rare inflammatory tumor-like vascular lesion composed of angiomatoid nodules in a fibrosclerotic background. We report herein on a case of SANT in the spleen with its pathologic features, and review the related literature. A 50-year-old woman presented with mild left upper quadrant discomfort and tenderness and she showed a 6 cm-sized solitary splenic mass on computed tomography. She underwent laparoscopic splenectomy. Grossly, the spleen showed a well circumscribed round-shaped solid mass with multinodular hemorrhagic surfaces. Microscopically, the mass consisted of multiple angiomatoid nodules surrounded by collagen bundles with fibroblasts and a lymphoplasma cell infiltration. Immunohistochemically, the cells of the angiomatoid nodules were positive for CD31, CD30, CD34, alpha-smooth muscle actin, and VWF-VIII, but they were negative for CD8, anaplastic lymphoma kinase protein, and D2-40. The patient has been under close follow-up without recurrence.

Citations

Citations to this article as recorded by

Sclerosing angiomatoid nodular transformation of the spleen presenting rapid growth after adrenalectomy: Report of a case
Yasuo Nagai, Daisuke Satoh, Hiroyoshi Matsukawa, Shigehiro Shiozaki
International Journal of Surgery Case Reports.2017; 30: 108. CrossRef
A Case of Sclerosing Angiomatoid Nodular Transformation of the Spleen: Spoke Wheel Pattern on Computed Tomography
Joo Han Park, Sun Hyuk Hwang, Ki Chan Kim, Si Yeon Kim, Ga Won Song, Jae Ho Han, Joon Seong Park
Korean Journal of Medicine.2014; 86(3): 357. CrossRef
Differentiation of an Unclear Splenic Lesion in a Patient With Cholangiocarcinoma
Constantin Lapa, Ulrich Steger, Christian O. Ritter, Vanessa Wild, Ken Herrmann
Clinical Nuclear Medicine.2014; 39(5): 470. CrossRef
Sclerosing Angiomatoid Nodular Transformation of the Spleen: CT and MRI Features With Pathologic Correlation
Rachel B. Lewis, Grant E. Lattin, Meenakshi Nandedkar, Nadine S. Aguilera
American Journal of Roentgenology.2013; 200(4): W353. CrossRef

First Report of a Gangliocytic Paraganglioma Arising in a Tailgut Cyst.: Yosep Chong, Mee Yon Cho; Korean J Pathol. 2010;44(4):435-440.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.4.435

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Abstract PDF

Here we present the first report of a gangliocytic paraganglioma arising in a tailgut cyst; it occurred in a 56-year-old man. Tailgut cysts are uncommon congenital hamartomatous lesions that arise in the retrorectal presacral space in infants or adults. Benign or malignant tumors associated with tailgut cysts are rarely described; the most common tumors are adenocarcinomas and carcinoid tumors. A gangliocytic paraganglioma is a rare benign tumor that occurs nearly exclusively in the second portion of the duodenum. Rare cases have been reported at other locations, but a tailgut cyst has never been described. In our case, a resected 3.9 x 3.3 x 3 cm mass was composed predominantly of a solid yellow white neuroendocrine tumor within the area of a tailgut cyst. The neuroendocrine component of this tumor was different from previously described carcinoid tumors with respect to the histologic findings of neural differentiation as well as the intermixed typical gangliocytic features highlighted by immunohistochemical stains for S-100 protein and neurofilament. Although an intermixed area of the tailgut cyst and gangliocytic paraganglioma were found in some areas, the pathogenesis of this tumor remains to be elucidated.

Citations

Citations to this article as recorded by

Diagnosis of Tailgut Cyst in Gynecologic Patients: Systematic Review of the Literature
Polina Schwarzman, Salvatore Andrea Mastrolia, Yael Sciaky-Tamir, Joel Baron, Boaz Sheizaf, Giuseppe Trojano, Reli Hershkovitz
Journal of Endometriosis and Pelvic Pain Disorders.2017; 9(3): 168. CrossRef
Fine Needle Aspiration Cytology Diagnosis of Tailgut Cyst: A Rare Entity
Farhan Asif Siddiqui, Rajan Chopra, Yusef Al-Marzooq
Acta Cytologica.2014; 58(2): 217. CrossRef

Subcutaneous Neuromuscular Hamartoma: A case report.: Dong Hoon Kim, Eun Kyung Hong, Jung Dal Lee; Korean J Pathol. 1999;33(1):62-64.

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Abstract PDF: Subcutaneous form of neuromuscular hamartoma is extremely rare and histologically different from the conventional neuromuscular hamartoma of the peripheral nerve or benign Triton tumor by an indistinct nodular growth with ill-defined margin and marked collagen interposition. It is usually not associated with a major nerve. We report a case of subcutaneous neuromuscular hamartoma developed in the forehead of 24-year-old man. The tumor showed proliferation of dense, hyalinized fibrous tissue, in which single or group of mature skeletal muscle fibers and nerve fibers were haphazardly intermixed. Recognition of abnormally arranged muscle and nerve fibers is important not to miss this lesion.

Odontogenic Gingival Epithelial Hamartoma; with Reference to the Expression of Ameloblastin Gene by in situ Hybridization and Immunohistochemistry.: Na Rae Kim, Yeon Lim Suh, Je G Chi, Young Joon Lee, Suk Keun Lee, Jae Il Lee, Chang Yun Lim, Ji Young Park; Korean J Pathol. 2004;38(2):116-120.

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Abstract PDF: Odontogenic gingival epithelial hamartoma (OGEH) is an extremely rare lesion characterized by an abnormal proliferation of odontogenic epithelium. This lesion is thought to arise from the rest of the dental lamina lying dormant in the gingival tissue after odontogenesis. Distinguishing OGEH from the granular cell variant of ameloblastoma and central odontogenic fibroma is important. To date, only eleven cases have been reported, and its pathogenesis remains unclear. We report here on a case of OGEH, where the epithelial strands in the lesion were conspicuously positive for the antisera of cytokeratin 19 and ameloblastin. Tumor cells intensely expressed ameloblastin mRNA by in situ hybridization. To the best of our knowledge, this is the first case of OGEH to which ameloblastin immunohistochemical stain and in situ hybridization were applied. Although our study is limited to a single case, the coexpression of cytokeratin 19 and ameloblastin might indicate the origin and specific cytodifferentiation of OGEH is quite different and unique, when contrasted to other odontogenic tumors.

Nasal Chondromesenchymal Hamartoma: A Case Report.: Jun Kang, Young Ok Hong, Geung Hwan Ahn, Young Min Kim, Hee Jeong Cha, Hye Jeong Choi; Korean J Pathol. 2007;41(4):258-262.

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Abstract PDF: We report a case of nasal chondromesenchymal hamartoma. A 14-year-old boy presented with a 5 cm sized mass in the left maxillary sinus, facial swelling and a loose tooth. A subtotal left maxillectomy with a bone graft was performed. The excised mass was composed of partly encapsulated, solid and cystic fragments of soft tissues. The mass contained chondroid and myxoid areas consisting of mesenchymal tissues including hyaline cartilage, osteoid and spindle cells in various proportions. The hyaline cartilage component was the most prominent. The spindle cell component had a fibrous matrix with variable myxoid or sclerotic changes. Thick hyalinized eosinophilic osteoid-like trabeculae were focally present. Immunohistochemically, all the mesenchymal cells tested positive for vimentin. The chondrocytes tested positive for the S-100 protein, and the spindle cell component showed focal immunoreactivity for smooth muscle actin and desmin. However, the cells were negative to pan-cytokeratin and p63.

Fine Needle Aspiration Cytology of Pulmonary Hamartoma: 3 cases.: Na Hye Myong, Kyung Ja Cho, Ja June Jang; Korean J Pathol. 1989;23(3):355-358.

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Abstract PDF: Fine needle aspiration cytology of three cases of pulmonary hamartoma is presented. Case 1 was in a 67-year-old man with a 7 cm-sized left lung mass. Case 2 and 3 were in 47 and 53 year old females and consisted of 3 cm and 2 cm-sized right lung nodules, respectively. Fine needle aspiration of the masses revealed several fragments of irregularly shaped mature hyaline cartilage or fibromyxoid mesenchyme and sheets of benign epithelial cells in scanty to acellular background. Also scattered were inflammatory cells including lymphocytes, neutrophils and histiocytes and mature fat cells. These features were diagnostic for pulmonary hamartoma and case 1 was histologically confirmed by following surgical excision of the mass. Differential diagnoses about pulmonary hamartoma in the respect of conditions capable of producing cartilage on fine needle aspiration, were discussed.

Hamartoma of the Breast A report of two cases.: Sung Suk Paeng, In Ae Park, Woo Kyoung Moon, Dong Young Noh, Eui Keun Ham; Korean J Pathol. 1999;33(3):199-203.

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Abstract PDF: Hamartoma of the breast is a rare benign lesion presenting as palpable mass. They are, however, sometimes misdiagnosed by pathologist due to their resemblance to other benign or physiologic conditions of the breast. We report two cases of hamartomas of the breast. The clinical diagnosis was fibroadenoma in both cases. One patient was a 49-year-old woman presented with palpable mass for the duration of 5 months. Mammography showed a well demarcated round mass with homogeneous density which is slightly greater than the surrounding glandular tissue. Grossly it was a 3.5 3.0 2.0 cm sized round firm mass covered by thin fibrous tissue. Microscopically it was a well defined mass composed of variable amount of breast lobules, dilated ducts and adipose tissue surrounded by dense hyalinized connective tissue. The adipose tissue accounted for 30% of the entire lesion. Cellular atypism was not found. The other patient was a 18-year-old woman presented with palpable mass of the duration of 3 years, which was increased in size recently. Mammography demonstratesd a well delimited lobulated mass with lucent halo. Ultrasonography showed a well encapsulated low echoic mass admixed with high echoic areas. Grossly, it was a 4.0 4.0 2.0 cm sized well demarcated oval-shaped firm mass with translucent thin fibrous capsule.. Microscopic findings were similar to the former. The amount of adipose tissue was 20%.

Nasal Chondromesenchymal Hamartoma: A case report.: Hyo Jeong Chae, Ji Hye Suk, Sun Kyung Lee; Korean J Pathol. 1999;33(3):225-227.

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Abstract PDF: Nasal chondromesenchymal hamartoma is a distinctive mixed mesenchymal lesion of sinonasal region with a complex histologic appearance and benign clinical course and clinicopathologically similar to those of the mesenchymal hamartoma of the chest wall of infancy. We report a case of nasal chondromesenchymal hamartoma occurred in the right nasal cavity in a 3-month-old female. She was admitted with a history of profuse nasal bleeding and obstruction. CT revealed complex solid and cystic mass, measuring 3.5x2.5x2.5 cm in dimensions which filled the right nasal cavity and extended into ethmoid sinus and cribriform plate. The received piecemeal fragments of tissue were brown tan-colored firm semitranslucent tissue with a cartilaginous appearance. Microscopically, the basic morphologic elements were irregular islands of hyaline cartilage and myxoid to spindle cell stroma with various cellularity.

Original Articles

Fine Needle Aspiration Cytology of Pulmonary Hamartoma.: Tae Jin Lee, Jin Sook Lee, Gyung Yub Gong, Shin Kwang Khang, Jae Y Ro; J Pathol Transl Med. 2000;11(1):19-24.

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Abstract PDF: Pulmonary hamartomas are uncommon benign tumors, usually discovered radiologically as a solitary coin lesion in asymptomatic individual. The approach to the patient with a peripheral lung nodule has changed with the increasing acceptance of fine needle aspiration cytology(FNAC) as a rapid, safe, inexpensive, and highly accurate diagnostic tool. However, a few reports describing the FNAC findings of pulmonary hamartoma have appeared in the cytologic literature and the experience of FNAC is limited. We reviewed all 9 cases of pulmonary hamartoma with histologic confirmation after FNAC seen at Asan Medical Center since 1995 to evaluate cytologic findings and to determine the value of FNAC in identifying that lesion. Originally, seven of nine patients were diagnosed as pulmonary hamartoma, while two patients were diagnosed as inflammatory lesion and adenocarcinoma of each. On review, eight of nine patients were considered as diagnostic of pulmonary hamartoma. The diagnostic findings in FNAC of pulmonary hamartoma were the presence of fibrillary myxoid tissue with spindle cells as well as hyaline cartilage.

Fine Needle Aspiration Cytology of Pulmonary Hamartoma: A Report of Two Cases.: Jeana Kim, Kyoung Mee Kim, Young Sill Kim, An hi Lee, Sang In Shim, Byung Kee Kim; J Pathol Transl Med. 2000;11(1):31-34.

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Abstract PDF: Pulmonary hamartoma is an uncommon benign tumor consisting of a mixture of loose fibromyxoid tissue, cartilage, fat, and cleft-like spaces lined by cuboidal or ciliated epithelium. Cytologically, the presence of a mesenchymal component is essential for the diagnosis of pulmonary hamartoma. We report the fine needle aspiration cytologic findings of two cases of pulmonary hamartoma. Case 1 was a 71-year-old woman with a mass, measuring 1.8X1.5 cm in the upper lobe of the right lung. Case 2 was a 51-year-old woman with a mass, measuring 2.3 x 2.0 cm in the lower lobe of the right lung. Fine needle aspiration cytology of both pulmonary masses revealed several sheets of loose fibromyxoid tissue fragments with focal cartilaginous differentiation and a few clusters of bland cuboidal epithelial cells on the bloody background. The diagnosis was histologically confirmed by needle biopsy.

Case Report

Hamartoma Arising in the Urinary Bladder: A case report.: Young Bae Kim, Tae Sook Hwang, Byung Gon Park, Jin Sook Jeong, Sook Hee Hong; Korean J Pathol. 1993;27(3):283-286.

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Abstract PDF: Hamartoma of the bladder is quite a rare entity which is composed of a disorderly admixture of mature cellular elements normally present in the urinary bladder. There is a great controversy regarding the pathogenesis of this lesion. Whether it is a true hamartomatous lesion or metaplastic lesion developed secondary to the inflammatory process. Similar or identical lesions has often been given by other names such as florid examples of cystitis glandularis. We prefer to cell florid examples of cystitis glandularis rather than hamartoma when it was occurred in an old age higher then 50th decade. Here we report a case of hamartoma of the urinary bladder in 44 years old man. Cystoscopic examination revealed a papillary polypoid mass which was attached to the fundus of bladder by long stalk. The mass measured 1.5 cm in greatest diameter. It was composed of epithelial nests resembling von Brunn's nest, cystitis glandularis or cystitis cystica dispersed in a stroma rich in smooth muscle and fibrous tissue.

Original Article

Infantile Myofibromatosis(Congenital Generalized Fibromatosis): Associated with multiple congenital malformations and basaloid follicular hamartomas in the skin.: Eun Sook Nam, Yoo Hun Kim, Han Kyeom Kim, Insun Kim, Je Geun Chi; Korean J Pathol. 1995;29(6):776-782.

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Abstract PDF: Infantile myofibromatosis with systemic involvement is a very rare disease and is characterized by numerous nodules composed of spindle cells of a myofibroblastic nature. There are often disseminated throughout the subcutis, muscle, skeleton and viscera. We report an autopsy case of infantile myofibromatosis in a stillborn female fetus of 32 weeks of gestation. The nodules, Imm to 2 cm, were found over the whole body and viscera. The involved viscera were the heart, tongue, esophagus, gastrointestinal tract, portal areas of the liver, spleen anc pancreas. There were also associated malformations, viz., frontal meningoencephalocele, flexion defer-mities, syndactyly, cleft palate, agenesis of corpus callosum, pachygyria, diaphragmatic hemia, renal hypoplasia, etc. Multiple basaloid follicular hamartomas of the skin were noted on the face and extremeties. There are no previous reports in the literature of infantile myofibromatosis in conjunction with the above skin lesion and congenital malformations.

Case Reports

Angiomyolipoma of the Nasal Cavity.: Jai Hyang Go; Korean J Pathol. 2005;39(4):284-286.

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Abstract PDF: Nasal angiomyolipomas are exceedingly rare and usually present as small tumors in middle-aged or old men. Their histology is similar to that of the renal counterpart, but they differ in the absence of epithelioid cells, the lack of HMB-45 immunoexpression and no association with tuberous sclerosis. A case of angiomyolipoma occurring in the right nasal cavity was presented in a 44-year-old male patient. The mass was removed by an endoscopic approach. A well-circumscribed, 1.3 cm-sized, ovoid mass was composed of yellowish gray, solid, soft tissue. Histologically, it consisted of bundles of smooth muscles, mature fat cells and thick-walled blood vessels. Nests of seromucous glands were admixed within the mass. Neither epithelioid nor pleomorphic cells were found, and HMB-45-positive cells were not detected. No recurrence has been detected for 2 months after the operation.

Muscular Hamartoma of the Breast: A case report.: Dong Won Min, Sun Hee Sung, In Joon Choi; Korean J Pathol. 1994;28(1):86-89.

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Abstract PDF: Hamartoma of the breast is relatively rare benign tumor, which is a well-circumscribed mass mainly composed of fibrous stroma, a(tipose tissue, ducts and acini. Muscular hamartoma of the breast is mainly composed of smooth muscle, and is extremely rare because proper smooth muscle is normaly absent in the breast except in the nipple. We describe a rare case of muscular hamartoma of the breast in a 38-year-old woman. This tumor was located in the upper outer quadrant and a 3 x 2.5 x 2 cm sized, well-demarcated but not encapsulated mass, The mass consisted mainly of irregularly arranged smooth muscle bundles in the fibrous stroma with lobular units and admixed fat cells. The origin of smooth muscle in hamartoma is not well known.

A Case of Ocular Neurofibromatosis.: Je G Chi, In Ae Park; Korean J Pathol. 1987;21(1):62-65.

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Abstract PDF: Neurofibromatosis is one of the neurocristopathies that involve many system or tissues forming various types of lesion. Almost every tissue or organ can be involved by this disease. However, the eyeball itself is very rarely affected by this process. The findings seen in our case indicate the diversity of lesions in neurofibromatosis, and also suggest hamartomatous nature. Heterotopic ganglion cells and glial cells in uveal tract are not easily understood. We reported here a case of intra-occular neurofibromatosis with its characteristic involvement of the uveal tract, in a 21 year old female. Her ocular symptoms began at her age of 11 as poor vision and were slowly progressive together with multiple facial neurofibromas. The involved left eyeball showed many ganglioneuroglial cell nests in iris, ciliary body and retina. Minute plexiform neurofibromas were also seen in small nerve twigs around the eyeball.

Mesenchymal Hamartoma of the Liver in Beckwith-Wiedemann Syndrome: A Case Report.: Seong Ho Yoo, Hyo Jin Park, Soo Yoen Cho, Chong Jai Kim; Korean J Pathol. 2002;36(6):425-428.

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Abstract PDF: Beckwith-Wiedemann syndrome is a well-known overgrowth syndrome associated with the presence of a wide variety of anomalies and increased risk of cancers. Less frequently, benign neoplasms also develop. We report a female infant with Beckwith-Wiedemann syndrome who developed a mesenchymal hamartoma of the liver. The patient was born with macroglossia, ear lobe crease, and abdominal distension. Laboratory data showed hypoglycemia, and magnetic resonance image revealed both adrenal enlargement, enhancing mass of the pancreas, and multiple hepatic nodules. The histologic findings of the resected distal pancreas and both adrenals were those of Beckwith-Wiedemann syndrome. Microscopic findings of the liver biopsy specimens were compatible with mesenchymal hamartoma. Hamartoma of the urinary bladder, cardiac fibrous hamartoma, and mixed hamartoma of the liver have been documented previously in association with Beckwith-Wiedemann syndrome. However, to our knowledge, this is the first case report of hepatic mesenchymal hamartoma in Beckwith-Wiedemann syndrome. Because of the paucity of hamartomas in childhood, we should be cautious of other features of Beckwith-Wiedemann syndrome and the present case extends the spectrum of tumor formation in this syndrome.

Ectopic Hamartomatous Thymoma: A Case Report along with a Review of the Literature Concerning the Histogenesis and New Nomenclature.: Sang Hee Seok, Dong Hyun Lee, Su Hwan Kang, Young Kyung Bae; Korean J Pathol. 2006;40(4):292-296.

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Abstract PDF: Ectopic hamartomatous thymoma (EHT) is a rare and distinctive benign neoplasm of the lower neck. We here report on a case of EHT arising in the suprasternal area of 47-year-old male patient. The well-circumscribed mass measured 7 x 6 x 4 cm and it predominantly had a solid gray-white cut surface. Microscopically, the tumor consisted of spindle cells, epithelial nests, and mature adipose tissue. The epithelial component was arranged in anastomosing cords, solid nests and variable-sized cysts that were lined by squamous or cuboidal epithelium. The spindle cells revealed the myoepithelial immunohistochemical phenotype. There was no obvious thymic differentiation nor was any normal thymic tissue observed in our case. We think that EHT needs to be reclassified with using different nomenclature to designate its origin and histology. Further, pathologists and clinicians should be aware of the existence of this tumor in the lower neck so as not to mistake it for high-grade sarcoma or spindle cell carcinoma.

Tonsillar Lymphangiomatous Polyp: Report of Two Cases.: Han Suk Ryu, Soo Young Jung, Jae Soo Koh, Seung Sook Lee; Korean J Pathol. 2006;40(5):381-384.

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Abstract PDF: Tonsillar lymphangiomatous polyp is an uncommon hamartomatous lesion that generally arises from the tonsillar surface, and it has rarely been reported in the medical literature. Because of the uncommon clinical and pathological features of these polyps, pathologists and clinicians may experience difficulty in correctly classifying them. We report herein two cases of lymphangiomatous polyp of the tonsil in a 49 year-old man and a 30 year-old man who both presented with a tonsillar mass of the palatine tonsil. Microscopically, there were protruding polypoid lesions that had a core of fibroadipose tissue with dilated multiple lymphatics and aggregated lymphoid tissue. Both patients have been stable since resection.

Mammary Hamartoma: A case report.: Sung Ran Hong, Hy Sook Kim, Byung Jae Cho; Korean J Pathol. 1988;22(4):471-474.

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Abstract PDF: We have recently experienced a case of mammary hamartoma in 29-year-old woman. Hamartoma of the breast is a rare benign tumor-like lesion which is characteristically well demarcated by thin layer of fibrofatty tissue and composed of essentially normal mammary tissue with mammary glandular structure and fibrous stroma containing variable amounts of fat. The lobular architecture is relatively well preserved. It can be readily recognized, but should not be confused with fibroadenoma or mammary dysplasia. It also shows characteristic mammographic picture.

Original Article

Fatty Hamartoma of Epicardium.: Hyun Wook Kang, Jung Ran Kim, Jeong Wook Seo, Je G Chi; Korean J Pathol. 1989;23(1):132-135.

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Abstract PDF: The fatty hamartoma of pericardium is characterized by abnormal accumulations of adipose tissue forming a recognizable mass. We reported a case that could best be put into "fatty hamartoma" group. This 66 year old male presented with atrial fibrillation came in for a anterior mediastinal mass. He received an operation for adenocarcinoma of ascending colon several months ago. The chest CT revealed an inhomogeneous mass in the right anterior mediastinal area, which was not clearly demarcated from the right cardiac border. The heart was deviated to the left side due to the mass. Surgical excision was performed under the impression of cardiac teratoma. The large mass was well encapsulated, and was bright yellow and partly pink, and measured 12x8 cm. Microscopically, the mass consisted predominantly of mature fat cells and abundant fibrous tissue with scattered nests of primitive cardiac muscle cells. Reviewing the literature we found the term "fatty hamartoma" could also refer to rhabdomyolipoma or fibrolipoma. Since it contains entrapped cardiac muscle cells, abundant fibrous tissue, multiple blood vessels and fetal fat cells, it could best the categorized into "hamartoma". The term "fatty" represents it's main component. Therefore we propose the term "fatty hamartoma" that could to be used further.

Case Report

Biliary Cystadeoma of the Liver: Report of a congenital case.: Jai Hyang Go, Young Nyun Park, Woo Hee Jung, Chanil Park; Korean J Pathol. 1995;29(2):241-243.

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Abstract PDF: Biliary cystadenoma of the liver is a rare multilocular cystic neoplasm of biliary origin. it occurs most often in middle aged women and rarely in children. Histogenesis of this tumor is uncertain. It may be developmental in origin arising from aberrant hamartomatous bile ducts or ectopic rests of embryonic biliary cystadenoma of the liver discovered at 8 month of intrauterine fetal life. This case supports its congenital theory.

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