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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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  • 159 Download
  • 2 Web of Science
  • 2 Crossref
AbstractAbstract PDF
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

Citations to this article as recorded by  
  • 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
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
  • 1,232 View
  • 153 Download
AbstractAbstract 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.
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
  • 7,226 View
  • 75 Download
  • 15 Crossref
AbstractAbstract 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.

Citations

Citations to this article as recorded by  
  • 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
  • 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
  • 3,031 View
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AbstractAbstract 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
  • 3,467 View
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  • 2 Crossref
AbstractAbstract 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

Citations to this article as recorded by  
  • 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
  • 3,821 View
  • 50 Download
  • 4 Crossref
AbstractAbstract 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
  • 3,262 View
  • 23 Download
  • 2 Crossref
AbstractAbstract 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
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.
  • 1,687 View
  • 15 Download
AbstractAbstract 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
Subcutaneous Neuromuscular Hamartoma: A case report.
Dong Hoon Kim, Eun Kyung Hong, Jung Dal Lee
Korean J Pathol. 1999;33(1):62-64.
  • 1,756 View
  • 34 Download
AbstractAbstract 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.
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.
  • 1,519 View
  • 14 Download
AbstractAbstract 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.
  • 1,376 View
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AbstractAbstract 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.
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.
  • 1,658 View
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AbstractAbstract 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.
Original Articles
Fine Needle Aspiration Cytology of Pulmonary Hamartoma.
Tae Jin Lee, Jin Sook Lee, Gyung Yub Gong, Shin Kwang Khang, Jae Y Ro
Korean J Cytopathol. 2000;11(1):19-24.
  • 1,584 View
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AbstractAbstract 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
Korean J Cytopathol. 2000;11(1):31-34.
  • 1,543 View
  • 15 Download
AbstractAbstract 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
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.
  • 1,677 View
  • 25 Download
AbstractAbstract 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.

J Pathol Transl Med : Journal of Pathology and Translational Medicine