Loading…
A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review
Mediastinal teratomas with elements of mature pancreatic tissue are rare. Only a very few cases of pancreatic tissue with nesidioblastosis in teratoma have been reported. Here, we report a case of a 12-year-old male who presented with pleural effusion and was revealed to have a large anterior medias...
Saved in:
Published in: | Endocrine pathology 2016-03, Vol.27 (1), p.21-24 |
---|---|
Main Authors: | , , , |
Format: | Article |
Language: | English |
Subjects: | |
Citations: | Items that this one cites Items that cite this one |
Online Access: | Get full text |
Tags: |
Add Tag
No Tags, Be the first to tag this record!
|
cited_by | cdi_FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663 |
---|---|
cites | cdi_FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663 |
container_end_page | 24 |
container_issue | 1 |
container_start_page | 21 |
container_title | Endocrine pathology |
container_volume | 27 |
creator | Agrawal, Tanupriya Blau, Adam J. Chwals, Walter J. Tischler, Arthur S. |
description | Mediastinal teratomas with elements of mature pancreatic tissue are rare. Only a very few cases of pancreatic tissue with nesidioblastosis in teratoma have been reported. Here, we report a case of a 12-year-old male who presented with pleural effusion and was revealed to have a large anterior mediastinal mass. Biopsy of the mass revealed benign mature teratoma. After biopsy, the teratoma ruptured into the right thoracic cavity. It was then excised and sent to pathology for further evaluation. Preoperatively, there was no evidence of hyperinsulinemia or hypoglycemia. Postoperatively, there was no change in blood glucose levels. Histologically, the mass showed large areas of mature pancreatic tissue flanking a small intestine-like structure. Numerous endocrine cell islets, poorly defined groups of neuroendocrine cells and ductular-insular complexes characteristic of nesidioblastosis were dispersed in the exocrine pancreatic parenchyma. In addition, other parts of the tumor containing keratinizing squamous epithelium with cutaneous adnexal glands, small intestine, and bronchus including cartilage and respiratory epithelium were observed. Some islets contained two or more cell types while others were monophenotypic. Immunohistochemical staining showed pronounced expression of pancreatic polypeptide, moderate expression of somatostatin and insulin and nearly complete absence of glucagon-containing cells. The selective deletion of glucagon might hold clues to an important regulatory mechanism in pancreatic development. |
doi_str_mv | 10.1007/s12022-015-9393-4 |
format | article |
fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_journals_1764950714</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>3952194231</sourcerecordid><originalsourceid>FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663</originalsourceid><addsrcrecordid>eNp1kc1uFDEQhC0EIiHwAFyQJa4ZaI899prbasNPpA2gaHO2POM2ONodL7aHiJfhWfEwAXHh1Jb6qyq5i5DnDF4xAPU6sxbatgHWNZpr3ogH5JR1nW4AtHpY3yBkw7WSJ-RJzrcAjAO0j8lJKzlbCdGekp9rejOGbxPSjc1Io6dX6ILNJYx2T3eYbIkHS-9C-UqvbJkS0s92HBLaEga6CzlPeE4_Yg4uxH5fhTGHfE7t6Oi6x5TsWOhl3mOhF8F7TDiWULVxfEPtknmNx5jKb8U2lDlxTrnG7wHvnpJH3u4zPrufZ-Tm3dvd5kOz_fT-crPeNkP9RWncgF44yZRA1OC5w0Fay9uVQsc9c4p75zrp-l7pleAdH7QXQsKKKQlKSn5GXi6-xxTrMXIxt3FK9QTZVEToDhQTlWILNaSYc0JvjikcbPphGJi5EbM0YmojZm7EzJoX985Tf0D3V_Gnggq0C5DravyC6Z_o_7r-Ajt2mAM</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1764950714</pqid></control><display><type>article</type><title>A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review</title><source>Springer Link</source><creator>Agrawal, Tanupriya ; Blau, Adam J. ; Chwals, Walter J. ; Tischler, Arthur S.</creator><creatorcontrib>Agrawal, Tanupriya ; Blau, Adam J. ; Chwals, Walter J. ; Tischler, Arthur S.</creatorcontrib><description>Mediastinal teratomas with elements of mature pancreatic tissue are rare. Only a very few cases of pancreatic tissue with nesidioblastosis in teratoma have been reported. Here, we report a case of a 12-year-old male who presented with pleural effusion and was revealed to have a large anterior mediastinal mass. Biopsy of the mass revealed benign mature teratoma. After biopsy, the teratoma ruptured into the right thoracic cavity. It was then excised and sent to pathology for further evaluation. Preoperatively, there was no evidence of hyperinsulinemia or hypoglycemia. Postoperatively, there was no change in blood glucose levels. Histologically, the mass showed large areas of mature pancreatic tissue flanking a small intestine-like structure. Numerous endocrine cell islets, poorly defined groups of neuroendocrine cells and ductular-insular complexes characteristic of nesidioblastosis were dispersed in the exocrine pancreatic parenchyma. In addition, other parts of the tumor containing keratinizing squamous epithelium with cutaneous adnexal glands, small intestine, and bronchus including cartilage and respiratory epithelium were observed. Some islets contained two or more cell types while others were monophenotypic. Immunohistochemical staining showed pronounced expression of pancreatic polypeptide, moderate expression of somatostatin and insulin and nearly complete absence of glucagon-containing cells. The selective deletion of glucagon might hold clues to an important regulatory mechanism in pancreatic development.</description><identifier>ISSN: 1046-3976</identifier><identifier>EISSN: 1559-0097</identifier><identifier>DOI: 10.1007/s12022-015-9393-4</identifier><identifier>PMID: 26318442</identifier><language>eng</language><publisher>New York: Springer US</publisher><subject>Biomarkers, Tumor - analysis ; Cell Differentiation ; Child ; Endocrinology ; Glucagon - metabolism ; Humans ; Immunohistochemistry ; Islets of Langerhans - pathology ; Male ; Mediastinal Neoplasms - pathology ; Medicine ; Medicine & Public Health ; Nesidioblastosis - pathology ; Oncology ; Pancreas - pathology ; Pathology ; Teratoma - pathology</subject><ispartof>Endocrine pathology, 2016-03, Vol.27 (1), p.21-24</ispartof><rights>Springer Science+Business Media New York 2015</rights><rights>Springer Science+Business Media New York 2016</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663</citedby><cites>FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>315,786,790,27957,27958</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26318442$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Agrawal, Tanupriya</creatorcontrib><creatorcontrib>Blau, Adam J.</creatorcontrib><creatorcontrib>Chwals, Walter J.</creatorcontrib><creatorcontrib>Tischler, Arthur S.</creatorcontrib><title>A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review</title><title>Endocrine pathology</title><addtitle>Endocr Pathol</addtitle><addtitle>Endocr Pathol</addtitle><description>Mediastinal teratomas with elements of mature pancreatic tissue are rare. Only a very few cases of pancreatic tissue with nesidioblastosis in teratoma have been reported. Here, we report a case of a 12-year-old male who presented with pleural effusion and was revealed to have a large anterior mediastinal mass. Biopsy of the mass revealed benign mature teratoma. After biopsy, the teratoma ruptured into the right thoracic cavity. It was then excised and sent to pathology for further evaluation. Preoperatively, there was no evidence of hyperinsulinemia or hypoglycemia. Postoperatively, there was no change in blood glucose levels. Histologically, the mass showed large areas of mature pancreatic tissue flanking a small intestine-like structure. Numerous endocrine cell islets, poorly defined groups of neuroendocrine cells and ductular-insular complexes characteristic of nesidioblastosis were dispersed in the exocrine pancreatic parenchyma. In addition, other parts of the tumor containing keratinizing squamous epithelium with cutaneous adnexal glands, small intestine, and bronchus including cartilage and respiratory epithelium were observed. Some islets contained two or more cell types while others were monophenotypic. Immunohistochemical staining showed pronounced expression of pancreatic polypeptide, moderate expression of somatostatin and insulin and nearly complete absence of glucagon-containing cells. The selective deletion of glucagon might hold clues to an important regulatory mechanism in pancreatic development.</description><subject>Biomarkers, Tumor - analysis</subject><subject>Cell Differentiation</subject><subject>Child</subject><subject>Endocrinology</subject><subject>Glucagon - metabolism</subject><subject>Humans</subject><subject>Immunohistochemistry</subject><subject>Islets of Langerhans - pathology</subject><subject>Male</subject><subject>Mediastinal Neoplasms - pathology</subject><subject>Medicine</subject><subject>Medicine & Public Health</subject><subject>Nesidioblastosis - pathology</subject><subject>Oncology</subject><subject>Pancreas - pathology</subject><subject>Pathology</subject><subject>Teratoma - pathology</subject><issn>1046-3976</issn><issn>1559-0097</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2016</creationdate><recordtype>article</recordtype><recordid>eNp1kc1uFDEQhC0EIiHwAFyQJa4ZaI899prbasNPpA2gaHO2POM2ONodL7aHiJfhWfEwAXHh1Jb6qyq5i5DnDF4xAPU6sxbatgHWNZpr3ogH5JR1nW4AtHpY3yBkw7WSJ-RJzrcAjAO0j8lJKzlbCdGekp9rejOGbxPSjc1Io6dX6ILNJYx2T3eYbIkHS-9C-UqvbJkS0s92HBLaEga6CzlPeE4_Yg4uxH5fhTGHfE7t6Oi6x5TsWOhl3mOhF8F7TDiWULVxfEPtknmNx5jKb8U2lDlxTrnG7wHvnpJH3u4zPrufZ-Tm3dvd5kOz_fT-crPeNkP9RWncgF44yZRA1OC5w0Fay9uVQsc9c4p75zrp-l7pleAdH7QXQsKKKQlKSn5GXi6-xxTrMXIxt3FK9QTZVEToDhQTlWILNaSYc0JvjikcbPphGJi5EbM0YmojZm7EzJoX985Tf0D3V_Gnggq0C5DravyC6Z_o_7r-Ajt2mAM</recordid><startdate>20160301</startdate><enddate>20160301</enddate><creator>Agrawal, Tanupriya</creator><creator>Blau, Adam J.</creator><creator>Chwals, Walter J.</creator><creator>Tischler, Arthur S.</creator><general>Springer US</general><general>Springer Nature B.V</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7QP</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope></search><sort><creationdate>20160301</creationdate><title>A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review</title><author>Agrawal, Tanupriya ; Blau, Adam J. ; Chwals, Walter J. ; Tischler, Arthur S.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2016</creationdate><topic>Biomarkers, Tumor - analysis</topic><topic>Cell Differentiation</topic><topic>Child</topic><topic>Endocrinology</topic><topic>Glucagon - metabolism</topic><topic>Humans</topic><topic>Immunohistochemistry</topic><topic>Islets of Langerhans - pathology</topic><topic>Male</topic><topic>Mediastinal Neoplasms - pathology</topic><topic>Medicine</topic><topic>Medicine & Public Health</topic><topic>Nesidioblastosis - pathology</topic><topic>Oncology</topic><topic>Pancreas - pathology</topic><topic>Pathology</topic><topic>Teratoma - pathology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Agrawal, Tanupriya</creatorcontrib><creatorcontrib>Blau, Adam J.</creatorcontrib><creatorcontrib>Chwals, Walter J.</creatorcontrib><creatorcontrib>Tischler, Arthur S.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Calcium & Calcified Tissue Abstracts</collection><collection>ProQuest Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>ProQuest Pharma Collection</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><jtitle>Endocrine pathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Agrawal, Tanupriya</au><au>Blau, Adam J.</au><au>Chwals, Walter J.</au><au>Tischler, Arthur S.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review</atitle><jtitle>Endocrine pathology</jtitle><stitle>Endocr Pathol</stitle><addtitle>Endocr Pathol</addtitle><date>2016-03-01</date><risdate>2016</risdate><volume>27</volume><issue>1</issue><spage>21</spage><epage>24</epage><pages>21-24</pages><issn>1046-3976</issn><eissn>1559-0097</eissn><abstract>Mediastinal teratomas with elements of mature pancreatic tissue are rare. Only a very few cases of pancreatic tissue with nesidioblastosis in teratoma have been reported. Here, we report a case of a 12-year-old male who presented with pleural effusion and was revealed to have a large anterior mediastinal mass. Biopsy of the mass revealed benign mature teratoma. After biopsy, the teratoma ruptured into the right thoracic cavity. It was then excised and sent to pathology for further evaluation. Preoperatively, there was no evidence of hyperinsulinemia or hypoglycemia. Postoperatively, there was no change in blood glucose levels. Histologically, the mass showed large areas of mature pancreatic tissue flanking a small intestine-like structure. Numerous endocrine cell islets, poorly defined groups of neuroendocrine cells and ductular-insular complexes characteristic of nesidioblastosis were dispersed in the exocrine pancreatic parenchyma. In addition, other parts of the tumor containing keratinizing squamous epithelium with cutaneous adnexal glands, small intestine, and bronchus including cartilage and respiratory epithelium were observed. Some islets contained two or more cell types while others were monophenotypic. Immunohistochemical staining showed pronounced expression of pancreatic polypeptide, moderate expression of somatostatin and insulin and nearly complete absence of glucagon-containing cells. The selective deletion of glucagon might hold clues to an important regulatory mechanism in pancreatic development.</abstract><cop>New York</cop><pub>Springer US</pub><pmid>26318442</pmid><doi>10.1007/s12022-015-9393-4</doi><tpages>4</tpages></addata></record> |
fulltext | fulltext |
identifier | ISSN: 1046-3976 |
ispartof | Endocrine pathology, 2016-03, Vol.27 (1), p.21-24 |
issn | 1046-3976 1559-0097 |
language | eng |
recordid | cdi_proquest_journals_1764950714 |
source | Springer Link |
subjects | Biomarkers, Tumor - analysis Cell Differentiation Child Endocrinology Glucagon - metabolism Humans Immunohistochemistry Islets of Langerhans - pathology Male Mediastinal Neoplasms - pathology Medicine Medicine & Public Health Nesidioblastosis - pathology Oncology Pancreas - pathology Pathology Teratoma - pathology |
title | A Unique Case of Mediastinal Teratoma with Mature Pancreatic Tissue, Nesidioblastosis, and Aberrant Islet Differentiation: a Case Report and Literature Review |
url | http://sfxeu10.hosted.exlibrisgroup.com/loughborough?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-09-21T22%3A46%3A49IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=A%20Unique%20Case%20of%20Mediastinal%20Teratoma%20with%20Mature%20Pancreatic%20Tissue,%20Nesidioblastosis,%20and%20Aberrant%20Islet%20Differentiation:%20a%20Case%20Report%20and%20Literature%20Review&rft.jtitle=Endocrine%20pathology&rft.au=Agrawal,%20Tanupriya&rft.date=2016-03-01&rft.volume=27&rft.issue=1&rft.spage=21&rft.epage=24&rft.pages=21-24&rft.issn=1046-3976&rft.eissn=1559-0097&rft_id=info:doi/10.1007/s12022-015-9393-4&rft_dat=%3Cproquest_cross%3E3952194231%3C/proquest_cross%3E%3Cgrp_id%3Ecdi_FETCH-LOGICAL-c442t-dcef4d6174ee90f3dec6aa3287ed3f1d73fdd56dbb7984353c9f4460817607663%3C/grp_id%3E%3Coa%3E%3C/oa%3E%3Curl%3E%3C/url%3E&rft_id=info:oai/&rft_pqid=1764950714&rft_id=info:pmid/26318442&rfr_iscdi=true |