Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report
Abstract Background Solitary fibrous tumor (SFT) is a prototypical mesenchymal neoplasm that induces non-islet cell tumor hypoglycemia (NICTH) due to overproduction of insulin-like growth factor 2 (IGF2). We here report the case of a malignant SFT associated with a hypoglycemia attack. Case presenta...
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| Format: | Article |
| Language: | English |
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SpringerOpen
2018-08-01
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| Series: | Surgical Case Reports |
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| Online Access: | http://link.springer.com/article/10.1186/s40792-018-0508-2 |
| _version_ | 1819049969893507072 |
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| author | Yusuke Arakawa Hidenori Miyake Hidehisa Horiguchi Taku Inokuchi Naoki Hino Takashi Ogasawara Takeshi Kuroda Shinichi Yamasaki |
| author_facet | Yusuke Arakawa Hidenori Miyake Hidehisa Horiguchi Taku Inokuchi Naoki Hino Takashi Ogasawara Takeshi Kuroda Shinichi Yamasaki |
| author_sort | Yusuke Arakawa |
| collection | DOAJ |
| description | Abstract Background Solitary fibrous tumor (SFT) is a prototypical mesenchymal neoplasm that induces non-islet cell tumor hypoglycemia (NICTH) due to overproduction of insulin-like growth factor 2 (IGF2). We here report the case of a malignant SFT associated with a hypoglycemia attack. Case presentation An 81-year-old man with a large subphrenic mass presented with hypoglycemia and loss of consciousness. His serum insulin and IGF1 levels were relatively low, suggesting an excessively high serum IGF2 levels. Preoperative Western blotting of serum confirmed the overproduction of high-molecular-weight IGF2. After total tumor resection, the patient recovered from hypoglycemia without the need for further treatment. Histological examination revealed proliferation of spindle cells and frequent nuclear mitoses with STAT6 and CD34 immunoreactivity, which led to the diagnosis of malignant SFT. IGF2 was strongly upregulated in the tumor upon immunohistochemistry, consistent with the report of NICTH. In addition, the tumor expressed IGF2 receptor (IGF2R) but not IGF1R. Conclusions The present results indicate that the tumor co-expressed IGF2 and IGF2R. IGF2R has not previously been recognized as a tyrosine kinase receptor participating in cell signal transduction. Thus, further case series are required to determine whether IGF2R overexpression reflects the action of an unknown autocrine/paracrine system involving IGF2 for cell proliferation or for the scavenging and degradation of IGF2. |
| first_indexed | 2024-12-21T11:40:36Z |
| format | Article |
| id | doaj.art-d6e60238099e428ba568d7fe83f0cbb7 |
| institution | Directory Open Access Journal |
| issn | 2198-7793 |
| language | English |
| last_indexed | 2024-12-21T11:40:36Z |
| publishDate | 2018-08-01 |
| publisher | SpringerOpen |
| record_format | Article |
| series | Surgical Case Reports |
| spelling | doaj.art-d6e60238099e428ba568d7fe83f0cbb72022-12-21T19:05:19ZengSpringerOpenSurgical Case Reports2198-77932018-08-01411510.1186/s40792-018-0508-2Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case reportYusuke Arakawa0Hidenori Miyake1Hidehisa Horiguchi2Taku Inokuchi3Naoki Hino4Takashi Ogasawara5Takeshi Kuroda6Shinichi Yamasaki7Department of Surgery, Tokushima Municipal HospitalDepartment of Surgery, Tokushima Municipal HospitalDepartment of Pathology, Tokushima Municipal HospitalDepartment of Endocrinology, Tokushima Municipal HospitalDepartment of Surgery, Tokushima Municipal HospitalDepartment of Surgery, Tokushima Municipal HospitalDepartment of Surgery, Tokushima Municipal HospitalDepartment of Surgery, Tokushima Municipal HospitalAbstract Background Solitary fibrous tumor (SFT) is a prototypical mesenchymal neoplasm that induces non-islet cell tumor hypoglycemia (NICTH) due to overproduction of insulin-like growth factor 2 (IGF2). We here report the case of a malignant SFT associated with a hypoglycemia attack. Case presentation An 81-year-old man with a large subphrenic mass presented with hypoglycemia and loss of consciousness. His serum insulin and IGF1 levels were relatively low, suggesting an excessively high serum IGF2 levels. Preoperative Western blotting of serum confirmed the overproduction of high-molecular-weight IGF2. After total tumor resection, the patient recovered from hypoglycemia without the need for further treatment. Histological examination revealed proliferation of spindle cells and frequent nuclear mitoses with STAT6 and CD34 immunoreactivity, which led to the diagnosis of malignant SFT. IGF2 was strongly upregulated in the tumor upon immunohistochemistry, consistent with the report of NICTH. In addition, the tumor expressed IGF2 receptor (IGF2R) but not IGF1R. Conclusions The present results indicate that the tumor co-expressed IGF2 and IGF2R. IGF2R has not previously been recognized as a tyrosine kinase receptor participating in cell signal transduction. Thus, further case series are required to determine whether IGF2R overexpression reflects the action of an unknown autocrine/paracrine system involving IGF2 for cell proliferation or for the scavenging and degradation of IGF2.http://link.springer.com/article/10.1186/s40792-018-0508-2No islet cell tumor hypoglycemiaSolitary fibrous tumorInsulin-like growth factor-2Insulin-like growth factor-2 receptor |
| spellingShingle | Yusuke Arakawa Hidenori Miyake Hidehisa Horiguchi Taku Inokuchi Naoki Hino Takashi Ogasawara Takeshi Kuroda Shinichi Yamasaki Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report Surgical Case Reports No islet cell tumor hypoglycemia Solitary fibrous tumor Insulin-like growth factor-2 Insulin-like growth factor-2 receptor |
| title | Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report |
| title_full | Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report |
| title_fullStr | Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report |
| title_full_unstemmed | Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report |
| title_short | Overexpression of IGF2 and IGF2 receptor in malignant solitary fibrous tumor with hypoglycemia: a case report |
| title_sort | overexpression of igf2 and igf2 receptor in malignant solitary fibrous tumor with hypoglycemia a case report |
| topic | No islet cell tumor hypoglycemia Solitary fibrous tumor Insulin-like growth factor-2 Insulin-like growth factor-2 receptor |
| url | http://link.springer.com/article/10.1186/s40792-018-0508-2 |
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