Extragenital Adenomatoid Tumor of a Mediastinal Lymph Node
2003; Elsevier BV; Volume: 78; Issue: 3 Linguagem: Inglês
10.4065/78.3.350
ISSN1942-5546
AutoresPhillip A. Isotalo, Antonio G. Nascimento, Victor F. Trastek, Lester E. Wold, John C. Cheville,
Tópico(s)Salivary Gland Tumors Diagnosis and Treatment
ResumoAdenomatoid tumors are benign neoplasms found predominantly in male and female genital tracts. Rare extragenital adenomatoid tumors have been discovered that involve serosal surfaces and nonmesothelial-lined organs such as adrenal glands. Since the discovery of adenomatoid tumors, their histogenetic origin has been debated. Many researchers support a mesothelial cell origin for adenomatoid tumors because these tumors characteristically express a mesothelial phenotype. Tumor derivation from primitive pluripotent mesenchymal cells and coelomic epithelium also has been suggested because of the anatomical distribution of the tumors. Despite their characteristic mesothelial phenotype and histological appearance, adenomatoid tumors have an extensive differential diagnosis that includes vascular neoplasms, malignant mesothelioma, germ cell tumors, and metastatic adenocarcinoma. Recognition of these tumors may be especially difficult when examined at frozen section and when adenomatoid tumors are encountered in rare extragenital sites. We describe an adenomatoid tumor of a mediastinal lymph node that was found incidentally during a redo Collis-Nissen gastroplasty. On frozen section examination, this tumor was misinterpreted as metastatic adenocarcinoma. The hematoxylin-eosin histological, immunohistochemical, and ultrastructural studies confirmed the mesothelial phenotype of this tumor. To our knowledge, this is the first description of a lymph node adenomatoid tumor. Adenomatoid tumors are benign neoplasms found predominantly in male and female genital tracts. Rare extragenital adenomatoid tumors have been discovered that involve serosal surfaces and nonmesothelial-lined organs such as adrenal glands. Since the discovery of adenomatoid tumors, their histogenetic origin has been debated. Many researchers support a mesothelial cell origin for adenomatoid tumors because these tumors characteristically express a mesothelial phenotype. Tumor derivation from primitive pluripotent mesenchymal cells and coelomic epithelium also has been suggested because of the anatomical distribution of the tumors. Despite their characteristic mesothelial phenotype and histological appearance, adenomatoid tumors have an extensive differential diagnosis that includes vascular neoplasms, malignant mesothelioma, germ cell tumors, and metastatic adenocarcinoma. Recognition of these tumors may be especially difficult when examined at frozen section and when adenomatoid tumors are encountered in rare extragenital sites. We describe an adenomatoid tumor of a mediastinal lymph node that was found incidentally during a redo Collis-Nissen gastroplasty. On frozen section examination, this tumor was misinterpreted as metastatic adenocarcinoma. The hematoxylin-eosin histological, immunohistochemical, and ultrastructural studies confirmed the mesothelial phenotype of this tumor. To our knowledge, this is the first description of a lymph node adenomatoid tumor. Adenomatoid tumors are benign neoplasms that have characteristic histological features, including variably dilated acinar structures arranged haphazardly in stromal tissue.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar Most adenomatoid tumors are discovered in male and female genital tracts1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar; however, rare extragenital adenomatoid tumors have been described in various locations, including the adrenal gland,5Angeles-Angeles A Reyes E Munoz-Fernandez L Angritt P Adenomatoid tumor of the right adrenal gland in a patient with AIDS.Endocr Pathol. Spring 1997; 8: 59-64Crossref PubMed Scopus (24) Google Scholar, 6Evans CP Vaccaro JA Storrs BG Christ PJ Suprarenal occurrence of an adenomatoid tumor.J Urol. 1988; 139: 348-349PubMed Google Scholar, 7Rodrigo Gasque CR Marti-Bonmati L Dosda R Gonzalez Martinez AG MR imaging of a case of adenomatoid tumor of the adrenal gland.Eur Radiol. 1999; 9: 552-554Crossref PubMed Scopus (30) Google Scholar, 8Glatz K Wegmann W Papillary adenomatoid tumour of the adrenal gland [letter].Histopathology. 2000; 37: 376-377Crossref PubMed Scopus (25) Google Scholar, 9Raaf HN Grant LD Santoscoy C Levin HS Abdul-Karim FW Adenomatoid tumor of the adrenal gland: a report of four new cases and a review of the literature.Mod Pathol. 1996; 9: 1046-1051PubMed Google Scholar, 10Simpson PR Adenomatoid tumor of the adrenal gland.Arch Pathol Lab Med. 1990; 114: 725-727PubMed Google Scholar, 11Travis WD Lack EE Azumi N Tsokos M Norton J Adenomatoid tumor of the adrenal gland with ultrastructural and immunohistochemical demonstration of a mesothelial origin.Arch Pathol Lab Med. 1990; 114: 722-724PubMed Google Scholar heart,12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar intestinal mesentery,13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar omentum,14Hanrahan JB A combined papillary mesothelioma and adenomatoid tumor of the omentum: report of a case.Cancer. 1963; 16: 1497-1500Crossref PubMed Scopus (53) Google Scholar pleura,15Handra-Luca A Couvelard A Abd Alsamad I et al.Adenomatoid tumor of the pleura: case report [in French].Ann Pathol. 2000; 4: 369-372Google Scholar, 16Ikuta N Tano M Iwata M et al.A case of adenomatoid mesothelioma of the pleura [in Japanese].Nippon Kyobu Shikkan Gakkai Zasshi. 1989; 27: 1540-1544PubMed Google Scholar, 17Kaplan MA Tazelaar HD Hayashi T Schroer KR Travis WD Adenomatoid tumors of the pleura.Am J Surg Pathol. 1996; 20: 1219-1223Crossref PubMed Scopus (73) Google Scholar and retroperitoneum,18Benisch BM A retroperitoneal mesonephric cystadenoma with features of the adenomatoid tumor of the genital tract.J Urol. 1973; 110: 44-46PubMed Google Scholar as well as in a herniorrhaphy specimen.19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar The mesothelial differentiation of adenomatoid tumors is well recognized from the histology, immunophenotype, and ultrastructure of these tumors19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 24McClure RF Keeney GL Sebo TJ Cheville JC Serous borderline tumor of the paratestis: a report of seven cases.Am J Surg Pathol. 2001; 25: 373-378Crossref PubMed Scopus (62) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar; however, despite the obvious differentiation, multiple histogenetic origins have been proposed for these tumors, including derivation from mesothelial cells,19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar, 26Sidhu GS Fresko O Adenomatoid tumor of the epididymis: ultrastructural evidence of its biphasic nature.Ultrastruct Pathol. 1980; 1: 39-47Crossref PubMed Scopus (25) Google Scholar primitive pluripotent mesenchymal cells,4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 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Although hyperplastic mesothelial cells that involve lymph node sinuses have been described previously,28Argani P Rosai J Hyperplastic mesothelial cells in lymph nodes: report of six cases of a benign process that can stimulate metastatic involvement by mesothelioma or carcinoma.Hum Pathol. 1998; 29: 339-346Abstract Full Text PDF PubMed Scopus (53) Google Scholar, 29Brooks JS LiVolsi VA Pietra GG Mesothelial cell inclusions in mediastinal lymph nodes mimicking metastatic carcinoma.Am J Clin Pathol. 1990; 93: 741-748PubMed Google Scholar, 30Clement PB Young RH Oliva E Sumner HW Scully RE Hyperplastic mesothelial cells within abdominal lymph nodes: mimic of metastatic ovarian carcinoma and serous borderline tumor—a report of two cases associated with ovarian neoplasms.Mod Pathol. 1996; 9: 879-886PubMed Google Scholar, 31Cohn DE Folpe AL Gown AM Goff BA Mesothelial pelvic lymph node inclusions mimicking metastatic thyroid carcinoma.Gynecol Oncol. 1998; 68: 210-213Crossref PubMed Scopus (20) Google Scholar, 32Isotalo PA Veinot JP Jabi M Hyperplastic mesothelial cells in mediastinal lymph node sinuses with extranodal lymphatic involvement.Arch Pathol Lab Med. 2000; 124: 609-613PubMed Google Scholar, 33Parkash V Vidwans M Carter D Benign mesothelial cells in mediastinal lymph nodes.Am J Surg Pathol. 1999; 23: 1264-1269Crossref PubMed Scopus (62) Google Scholar, 34Rutty GN Lauder I Mesothelial cell inclusions within mediastinal lymph nodes.Histopathology. 1994; 25: 483-487Crossref PubMed Scopus (29) Google Scholar to our knowledge, this is the first description of a lymph node adenomatoid tumor. A 66-year-old woman was admitted to a Mayo Clinic-affiliated hospital in Rochester, Minn, because of recurrent dysphagia, intermittent chest pain, and regurgitation at night, 2½ years after a Nissen fundoplication for intractable reflux esophagitis resistant to medical therapy. Four months before admission, the patient had an esophago-gastroduodenoscopy that showed a 4-cm diaphragmatic hernia and no evidence of reflux esophagitis. Her surgical history included remote tonsillectomy, total abdominal hysterectomy, bilateral salpingo-oophorectomy, and remote appendectomy. The patient also had a history of rheumatoid arthritis and postoperative pulmonary embolism, the latter having complicated her first Nissen fundoplication. Her medications on admission included cisapride and omeprazole. Physical examination findings were normal for vital signs; cardiorespiratory examination results were unremarkable. The patient underwent redo uncut Collis-Nissen gastroplasty. When the inferior pulmonary ligament was divided, 3 lymph nodes were found and sent for intraoperative frozen section. One lymph node was particularly firm at the time of surgery, and on frozen section examination it was interpreted as metastatic adenocarcinoma. Intraoperative palpation of the left lung and gastroesophageal region revealed no evidence of tumor. When permanent sections were reviewed the day after surgery, the diagnosis was amended from metastatic adenocarcinoma to adenomatoid tumor. On the second postoperative day, the patient developed atrial fibrillation that was managed with digoxin and low-dose intravenous heparin because of her history of postoperative pulmonary embolism. No other postoperative complications developed. Four years later, the patient is alive and asymptomatic with no evidence of malignancy or recurrent esophagitis. All lymph nodes were frozen for intraoperative frozen section examination and stained with toluidine blue. The frozen section remnant was then fixed in 10% neutral-buffered formalin, processed, and then stained with hematoxylin-eosin for histological examination. Immuno-histochemical stains using the standard avidin-biotin-peroxidase complex method were performed on 4-µm sections from formalin-fixed, paraffin-embedded tissues using the following antibodies: calretinin (1:2000, polyclonal; Zymed Laboratories, Inc, South San Francisco, Calif), CD15 (1:10, clone MMA; Becton, Dickinson and Company, San Jose, Calif), CD31 (1:200, clone JC/70a; DakoCytomation California, Inc, Carpinteria, Calif), CD34 (1:50, clone MY10; Becton, Dickinson), polyclonal carcinoembryonic antigen (p-CEA, 1:2000; DakoCytomation), CAM 5.2 (1:50, clone CAM 5.2; Becton, Dickinson), cytokeratins 5/6 (CK5/6, 1:100, clone D5/16B4; Zymed), cytokeratin 7 (CK7, 1:100, clone 0B-TL12/30; DakoCytomation), cytokeratin 20 (CK20, 1:100, clone Ks20.8; DakoCytomation), cytokeratins AE1/AE3 (1:200, clones AE1 and AE3; Zymed), MOC-31 (1:100, clone MOC-31; DakoCytomation), and vimentin (1:500, clone 3B4; DakoCytomation). All immunohistochemical stains were processed with an autostainer (BioTek or Ventana ES, Ventana Medical Systems, Inc, Tucson, Ariz), except for CD15 slides, which were manually stained using the avidin-biotin-peroxidase complex detection system. Ultrastructural studies were performed on lymph node tissue that was deparaffinized in xylene, fixed in 2.5% glutaraldehyde, and treated subsequently with osmium tetroxide. Tissue was embedded in plastic epoxy, and thin sections were cut and stained with uranyl acetate and lead citrate. Electron microscopy was performed with a Zeiss EM9 electron microscope (Carl Zeiss, Inc, Thornwood, NY). Intraoperative frozen section examination revealed 2 benign lymph nodes. A third lymph node, 0.9 cm in diameter, was replaced by a tumor that had a “glandular” pattern. Metastatic adenocarcinoma was diagnosed on frozen section examination. Review of hematoxylin-eosin-stained permanent sections confirmed involvement of a single lymph node by tumor. Pseudoacini and anastomosing channels lined by both epithelioid cells and flattened cells characterized this neoplasm (Figure 1, left). Solid foci of epithelioid cells with intervening fibrous tissue were also present (Figure 1, right). The epithelioid cells had abundant eosinophilic cytoplasm and vesicular nuclei with well-defined, small nucleoli. Nuclear pleomorphism, mitotic activity, and tumor necrosis were absent. No extranodal tumor was identified. Epithelioid cells and flattened cells exhibited strong immunoreactivity for calretinin (Figure 2), CAM 5.2, cytokeratins AE1/AE3, CK7, and vimentin and weak focal staining for CK5/6. The immunoperoxidase stains, CD15, CD31, CD34, CK20, MOC-31, and p-CEA, were all negative. Ultrastructural studies revealed epithelioid cells with prominent, slender microvilli, perinuclear intermediate filaments, and desmosomes (Figure 3). The hematoxylin-eosin histology, immunophenotype, and ultrastructure confirmed the mesothelial differentiation of the neoplasm. An extragenital adenomatoid tumor involving a lymph node was diagnosed.Figure 3Electron microscopy of the nodal adenomatoid tumor shows epithelioid cells with prominent microvilli, perinuclear intermediate filaments, and desmosomes (osmium tetroxide, original magnification x12,600).View Large Image Figure ViewerDownload (PPT) Adenomatoid tumors are well recognized as benign neoplasms of male and female genital tracts.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar Patients with these tumors are commonly asymptomatic, and the tumors are often found incidentally during surgical procedures.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar The epididymis is the most common site of involvement in men; however, adenomatoid tumors also may involve the ejaculatory duct, spermatic cord, tunica albuginea, tunica vaginalis, and even testicular parenchyma.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar, 35Fan K Johnson DF Adenomatoid tumor of ejaculatory duct.Urology. 1985; 25: 653-654Abstract Full Text PDF PubMed Scopus (17) Google Scholar In women, the fallopian tubes and uterus are commonly involved, although ovarian adenomatoid tumors also occur.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar Regardless of their location, adenomatoid tumors have a consistent light microscopic appearance, consisting of plump eosinophilic epithelioid cells and flattened cells, which are organized around tubules and variably dilated acinar structures.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar Individual tumor cells may adopt a signet ring cell morphology, secondary to cytoplasmic vacuolization.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar Intervening stroma consists of fibrous tissue with varying degrees of hyalinized collagen and at times, smooth muscle.1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar Adenomatoid tumors may exhibit adenoid, angiomatoid, cystic, and solid areas3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar and may have an infiltrative pattern that can cause diagnostic difficulties.3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar Although adenomatoid tumors possess a characteristic histological appearance, their differential diagnosis is extensive and includes vascular neoplasms, lymphangioma, malignant mesothelioma, germ cell tumors, carcinoma of the rete testis in males, and metastatic adenocarcinoma, including signet ring cell carcinoma.2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 3Quigley JC Hart WR Adenomatoid tumors of the uterus.Am J Clin Pathol. 1981; 76: 627-635Crossref PubMed Scopus (89) Google Scholar, 9Raaf HN Grant LD Santoscoy C Levin HS Abdul-Karim FW Adenomatoid tumor of the adrenal gland: a report of four new cases and a review of the literature.Mod Pathol. 1996; 9: 1046-1051PubMed Google Scholar, 12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar, 13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar, 17Kaplan MA Tazelaar HD Hayashi T Schroer KR Travis WD Adenomatoid tumors of the pleura.Am J Surg Pathol. 1996; 20: 1219-1223Crossref PubMed Scopus (73) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar Table 1 compares the clinical and histopathologic features of adenomatoid tumors and adenocarcinoma. Adenomatoid tumors may be difficult to diagnose on frozen section examination,2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar especially when rare extragenital adenomatoid tumors are encountered as incidental surgical findings,13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar as occurred in our patient.Table 1Clinical and Histopathologic Features of Adenomatoid Tumors and AdenocarcinomaAdenomatoid tumorsAdenocarcinomaClinical behaviorBenignMalignantHistological features Typically well circumscribedTumors characterized by pseudoacini lined by epithelioid cells and flattened cells with intervening fibrous stromaTumor cells may adopt a signet ring cell morphology; however, the background pattern is that of adenomatoid tumorMultiple histological patterns (adenoid, angiomatoid, cystic, and solid) are typical Typically poorly circumscribedWell-differentiated tumors characterized by infiltrative glands surrounded by a desmoplastic stromal reactionSignet ring cell differentiation may occur, either in the presence or absence of typical adenocarcinomaMultiple histological patterns are uncommonImmunophenotype Diffuse cytokeratin immunoreactivityNo immunoreactivity for certain epithelial/carcinoma markers (eg, CD15, p-CEA, MOC-31) and immuno-reactivity for mesothelial markers (eg, calretinin) Diffuse cytokeratin immunoreactivityStrong immunoreactivity for certain epithelial/carcinoma markers (eg, CD15, p-CEA, MOC-31)UltrastructureTumor cells have a mesothelial phenotype consisting of abundant, long, and slender microvilli, perinuclear intermediate filaments, and desmosomesTumor cells may have microvilli, but they are typically less numerous and shorter, in addition to desmosomes and intermediate filaments Open table in a new tab Although adenomatoid tumors initially were believed to be restricted to the genital tracts,1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar, 2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar rare extragenital adenomatoid tumors occur in a variety of anatomical sites, including the adrenal gland,5Angeles-Angeles A Reyes E Munoz-Fernandez L Angritt P Adenomatoid tumor of the right adrenal gland in a patient with AIDS.Endocr Pathol. Spring 1997; 8: 59-64Crossref PubMed Scopus (24) Google Scholar, 6Evans CP Vaccaro JA Storrs BG Christ PJ Suprarenal occurrence of an adenomatoid tumor.J Urol. 1988; 139: 348-349PubMed Google Scholar, 7Rodrigo Gasque CR Marti-Bonmati L Dosda R Gonzalez Martinez AG MR imaging of a case of adenomatoid tumor of the adrenal gland.Eur Radiol. 1999; 9: 552-554Crossref PubMed Scopus (30) Google Scholar, 8Glatz K Wegmann W Papillary adenomatoid tumour of the adrenal gland [letter].Histopathology. 2000; 37: 376-377Crossref PubMed Scopus (25) Google Scholar, 9Raaf HN Grant LD Santoscoy C Levin HS Abdul-Karim FW Adenomatoid tumor of the adrenal gland: a report of four new cases and a review of the literature.Mod Pathol. 1996; 9: 1046-1051PubMed Google Scholar, 10Simpson PR Adenomatoid tumor of the adrenal gland.Arch Pathol Lab Med. 1990; 114: 725-727PubMed Google Scholar, 11Travis WD Lack EE Azumi N Tsokos M Norton J Adenomatoid tumor of the adrenal gland with ultrastructural and immunohistochemical demonstration of a mesothelial origin.Arch Pathol Lab Med. 1990; 114: 722-724PubMed Google Scholar heart,12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar intestinal mesentery,13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar omentum,14Hanrahan JB A combined papillary mesothelioma and adenomatoid tumor of the omentum: report of a case.Cancer. 1963; 16: 1497-1500Crossref PubMed Scopus (53) Google Scholar pleura,15Handra-Luca A Couvelard A Abd Alsamad I et al.Adenomatoid tumor of the pleura: case report [in French].Ann Pathol. 2000; 4: 369-372Google Scholar, 16Ikuta N Tano M Iwata M et al.A case of adenomatoid mesothelioma of the pleura [in Japanese].Nippon Kyobu Shikkan Gakkai Zasshi. 1989; 27: 1540-1544PubMed Google Scholar, 17Kaplan MA Tazelaar HD Hayashi T Schroer KR Travis WD Adenomatoid tumors of the pleura.Am J Surg Pathol. 1996; 20: 1219-1223Crossref PubMed Scopus (73) Google Scholar peritoneum,19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar and retroperitoneum.18Benisch BM A retroperitoneal mesonephric cystadenoma with features of the adenomatoid tumor of the genital tract.J Urol. 1973; 110: 44-46PubMed Google Scholar The nodal tumor encountered in our patient showed the classic architecture, immunophenotype, and ultrastructural features of an adenomatoid tumor. The neoplasm's strong immunoreactivity for cyto keratins AE1/AE3, CK7, calretinin, and vimentin, focal staining for CK5/6, and negative staining for endothelial markers, MOC-31, CD15, and p-CEA revealed the mesothelial differentiation of this adenomatoid tumor. The ultrastructural findings of slender microvilli, perinuclear intermediate filaments, and desmosomes also supported the tumor's mesothelial phenotype. To our knowledge, this is the first report of a nodal adenomatoid tumor. Since adenomatoid tumors were first recognized by Masson et al36Masson P Riopelle JL Simard LC Le mesotheliome benin de la sphere genitale.Rev Canad Biol. 1942; 1: 720-751Google Scholar and named by Golden and Ash in 1945,1Golden A Ash JE Adenomatoid tumors of the genital tract.Am J Pathol. 1945; 21: 63-79PubMed Google Scholar their histogenesis has been debated.2Lee MJ Dockerty MB Thompson GJ Waugh JM Benign mesotheliomas (adenomatoid tumors) of the genital tract.Surg Gynecol Obstet. 1950; 91: 221-231PubMed Google Scholar, 4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar, 27Soderstrom KO Origin of adenomatoid tumor: a comparison between the structure of adenomatoid tumor and epididymal duct cells.Cancer. 1982; 49: 2349-2357Crossref PubMed Scopus (22) Google Scholar Adenomatoid tumors clearly have a mesothelial phenotype and immunophenotype19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 24McClure RF Keeney GL Sebo TJ Cheville JC Serous borderline tumor of the paratestis: a report of seven cases.Am J Surg Pathol. 2001; 25: 373-378Crossref PubMed Scopus (62) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar; therefore, mesothelial cells have been proposed as the progenitor cells of these tumors.19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 20Delahunt B Eble JN King D Bethwaite PB Nacey JN Thornton A Immunohistochemical evidence for mesothelial origin of paratesticular adenomatoid tumour.Histopathology. 2000; 36: 109-115Crossref PubMed Scopus (68) Google Scholar, 21Delahunt B Eble JN Srigley JR Thornton A Paratesticular adenomatoid tumor: assessment of calretinin immunoexpression and cell proliferation indices.J Urol Pathol. 2000; 12: 105-115Crossref Google Scholar, 22Mackay B Bennington JL Skoglund RW The adenomatoid tumor: fine structural evidence for a mesothelial origin.Cancer. 1971; 27: 109-115Crossref PubMed Scopus (76) Google Scholar, 25Taxy JB Battifora H Oyasu R Adenomatoid tumors: a light microscopic, histochemical, and ultrastructural study.Cancer. 1974; 34: 306-316Crossref PubMed Scopus (102) Google Scholar The origin of adenomatoid tumors from primitive pluripotent mesenchymal cells4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar and coelomic epithelium27Soderstrom KO Origin of adenomatoid tumor: a comparison between the structure of adenomatoid tumor and epididymal duct cells.Cancer. 1982; 49: 2349-2357Crossref PubMed Scopus (22) Google Scholar has also been suggested. The proposed mesothelial origin of adenomatoid tumors does not explain fully why most of these tumors are limited to the genital tracts27Soderstrom KO Origin of adenomatoid tumor: a comparison between the structure of adenomatoid tumor and epididymal duct cells.Cancer. 1982; 49: 2349-2357Crossref PubMed Scopus (22) Google Scholar and why so few of them have been reported developing on serosal surfaces.12Natarajan S Luthringer DJ Fishbein MC Adenomatoid tumor of the heart: report of a case.Am J Surg Pathol. 1997; 21: 1378-1380Crossref PubMed Scopus (42) Google Scholar, 13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar, 14Hanrahan JB A combined papillary mesothelioma and adenomatoid tumor of the omentum: report of a case.Cancer. 1963; 16: 1497-1500Crossref PubMed Scopus (53) Google Scholar, 15Handra-Luca A Couvelard A Abd Alsamad I et al.Adenomatoid tumor of the pleura: case report [in French].Ann Pathol. 2000; 4: 369-372Google Scholar, 16Ikuta N Tano M Iwata M et al.A case of adenomatoid mesothelioma of the pleura [in Japanese].Nippon Kyobu Shikkan Gakkai Zasshi. 1989; 27: 1540-1544PubMed Google Scholar, 17Kaplan MA Tazelaar HD Hayashi T Schroer KR Travis WD Adenomatoid tumors of the pleura.Am J Surg Pathol. 1996; 20: 1219-1223Crossref PubMed Scopus (73) Google Scholar Adenomatoid tumors also develop in non-mesothelial-lined sites such as the adrenal gland5Angeles-Angeles A Reyes E Munoz-Fernandez L Angritt P Adenomatoid tumor of the right adrenal gland in a patient with AIDS.Endocr Pathol. Spring 1997; 8: 59-64Crossref PubMed Scopus (24) Google Scholar, 6Evans CP Vaccaro JA Storrs BG Christ PJ Suprarenal occurrence of an adenomatoid tumor.J Urol. 1988; 139: 348-349PubMed Google Scholar, 7Rodrigo Gasque CR Marti-Bonmati L Dosda R Gonzalez Martinez AG MR imaging of a case of adenomatoid tumor of the adrenal gland.Eur Radiol. 1999; 9: 552-554Crossref PubMed Scopus (30) Google Scholar, 8Glatz K Wegmann W Papillary adenomatoid tumour of the adrenal gland [letter].Histopathology. 2000; 37: 376-377Crossref PubMed Scopus (25) Google Scholar, 9Raaf HN Grant LD Santoscoy C Levin HS Abdul-Karim FW Adenomatoid tumor of the adrenal gland: a report of four new cases and a review of the literature.Mod Pathol. 1996; 9: 1046-1051PubMed Google Scholar, 10Simpson PR Adenomatoid tumor of the adrenal gland.Arch Pathol Lab Med. 1990; 114: 725-727PubMed Google Scholar, 11Travis WD Lack EE Azumi N Tsokos M Norton J Adenomatoid tumor of the adrenal gland with ultrastructural and immunohistochemical demonstration of a mesothelial origin.Arch Pathol Lab Med. 1990; 114: 722-724PubMed Google Scholar and deep to surface mesothelium.23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar, 27Soderstrom KO Origin of adenomatoid tumor: a comparison between the structure of adenomatoid tumor and epididymal duct cells.Cancer. 1982; 49: 2349-2357Crossref PubMed Scopus (22) Google Scholar These adenomatoid tumors may have originated from mesothelial inclusions5Angeles-Angeles A Reyes E Munoz-Fernandez L Angritt P Adenomatoid tumor of the right adrenal gland in a patient with AIDS.Endocr Pathol. Spring 1997; 8: 59-64Crossref PubMed Scopus (24) Google Scholar, 10Simpson PR Adenomatoid tumor of the adrenal gland.Arch Pathol Lab Med. 1990; 114: 725-727PubMed Google Scholar, 11Travis WD Lack EE Azumi N Tsokos M Norton J Adenomatoid tumor of the adrenal gland with ultrastructural and immunohistochemical demonstration of a mesothelial origin.Arch Pathol Lab Med. 1990; 114: 722-724PubMed Google Scholar or even from embolized mesothelial cells.30Clement PB Young RH Oliva E Sumner HW Scully RE Hyperplastic mesothelial cells within abdominal lymph nodes: mimic of metastatic ovarian carcinoma and serous borderline tumor—a report of two cases associated with ovarian neoplasms.Mod Pathol. 1996; 9: 879-886PubMed Google Scholar, 32Isotalo PA Veinot JP Jabi M Hyperplastic mesothelial cells in mediastinal lymph node sinuses with extranodal lymphatic involvement.Arch Pathol Lab Med. 2000; 124: 609-613PubMed Google Scholar However, adenomatoid tumor origin from pluripotent mesenchymal cells,4Jackson JR The histogenesis of the “adenomatoid' tumor of the genital tract.Cancer. 1958; 11: 337-350Crossref PubMed Scopus (67) Google Scholar, 23Mai KT Yazdi HM Perkins DG Isotalo PA Adenomatoid tumors of the genital tract: evidence of mesenchymal cell origin.Pathol Res Pract. 1999; 195: 605-610Crossref PubMed Scopus (24) Google Scholar especially mesenchymal cells associated with the müllerian tract, could explain both the predominance of adenomatoid tumors in the genital tract as well as the rarity of these tumors in the extragenital tract. Defining the exact origin of adenomatoid tumors may be somewhat academic because both mesothelial cells and primitive müllerian mesenchymal cells are derived from the same source: the coelomic epithelium.19Stephenson TJ Mills PM Adenomatoid tumours: an immunohistochemical and ultrastructural appraisal of their histogenesis.J Pathol. 1986; 148: 327-335Crossref PubMed Scopus (63) Google Scholar, 27Soderstrom KO Origin of adenomatoid tumor: a comparison between the structure of adenomatoid tumor and epididymal duct cells.Cancer. 1982; 49: 2349-2357Crossref PubMed Scopus (22) Google Scholar Although the histogenetic origin of adenomatoid tumors may continue to be debated, ongoing recognition of these tumors in extragenital sites is important to prevent diagnostic confusion with other neoplasms, especially metastatic adenocarcinoma.11Travis WD Lack EE Azumi N Tsokos M Norton J Adenomatoid tumor of the adrenal gland with ultrastructural and immunohistochemical demonstration of a mesothelial origin.Arch Pathol Lab Med. 1990; 114: 722-724PubMed Google Scholar, 13Craig JR Hart WR Extragenital adenomatoid tumor: evidence for the mesothelial theory of origin.Cancer. 1979; 43: 1678-1681Crossref PubMed Scopus (69) Google Scholar The discovery of a nodal adenomatoid tumor proves that these tumors are not restricted to the genital tract or to mesothelial-lined surfaces.
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