Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report
Abstract Background Splenic cord capillary hemangioma is a rare benign vascular lesion classified as a splenic hamartoma. On the other hand, non-islet cell tumor hypoglycemia (NICTH) is one of the rare causes of spontaneous hypoglycemia and is considered to be one of the paraneoplastic syndromes. To...
Main Authors: | , , , , , , |
---|---|
Format: | Article |
Language: | English |
Published: |
BMC
2019-12-01
|
Series: | Diagnostic Pathology |
Subjects: | |
Online Access: | https://doi.org/10.1186/s13000-019-0915-0 |
id |
doaj-9c48db4e4919424cb51451e5289cbda4 |
---|---|
record_format |
Article |
spelling |
doaj-9c48db4e4919424cb51451e5289cbda42020-12-20T12:17:09ZengBMCDiagnostic Pathology1746-15962019-12-011411510.1186/s13000-019-0915-0Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case reportTatsuaki Daimon0Takeo Kosaka1Minoru Horinaga2Junichi Saito3Yoshito Ueyama4Shoji Matsuzaki5Mototsugu Oya6Department of Urology, Keio University School of MedicineDepartment of Urology, Keio University School of MedicineDepartment of Urology, Inagi Municipal HospitalDepartment of Surgery, Inagi Municipal HospitalDepartment of Pathology, Inagi Municipal HospitalDepartment of Urology, Inagi Municipal HospitalDepartment of Urology, Keio University School of MedicineAbstract Background Splenic cord capillary hemangioma is a rare benign vascular lesion classified as a splenic hamartoma. On the other hand, non-islet cell tumor hypoglycemia (NICTH) is one of the rare causes of spontaneous hypoglycemia and is considered to be one of the paraneoplastic syndromes. To the best of our knowledge, this is the first reported case of a splenic cord capillary hemangioma with NICTH. Case presentation A 25-year-old male was referred to our hospital with hypoglycemia. Except for his low blood sugar, there were no abnormal findings from laboratory tests, which included an endocrinological examination. Enhanced computed tomography confirmed the presence of a solid mass measuring about 6 cm in the retroperitoneum, and a tumorectomy was performed. During this operation, it became clear that the tumor turned out to be a splenic parenchyma, and as a result, a total splenectomy was performed. Microscopically, we diagnosed this as a cord capillary hemangioma, and through immunohistochemistry, we found that some tumor cells were positive for insulin-like growth factor -II. Fortunately, the hypoglycemia-related symptoms disappeared after surgical resection was performed. The patient is still alive and well without evidence of local tumor recurrence 15 years after the operation. Conclusions Splenic cord capillary hemangioma, one of the types of splenic hamartomas, is a very rare benign vascular lesion and might be associated with hypoglycemia thought to be NICTH.https://doi.org/10.1186/s13000-019-0915-0Insulin-like growth factor-IINon-islet cell tumor hypoglycemiaSplenic cord capillary hemangiomaSplenic hamartomaSplenic tumor |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Tatsuaki Daimon Takeo Kosaka Minoru Horinaga Junichi Saito Yoshito Ueyama Shoji Matsuzaki Mototsugu Oya |
spellingShingle |
Tatsuaki Daimon Takeo Kosaka Minoru Horinaga Junichi Saito Yoshito Ueyama Shoji Matsuzaki Mototsugu Oya Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report Diagnostic Pathology Insulin-like growth factor-II Non-islet cell tumor hypoglycemia Splenic cord capillary hemangioma Splenic hamartoma Splenic tumor |
author_facet |
Tatsuaki Daimon Takeo Kosaka Minoru Horinaga Junichi Saito Yoshito Ueyama Shoji Matsuzaki Mototsugu Oya |
author_sort |
Tatsuaki Daimon |
title |
Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
title_short |
Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
title_full |
Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
title_fullStr |
Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
title_full_unstemmed |
Splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
title_sort |
splenic cord capillary hemangioma with non-islet cell tumor hypoglycemia: a case report |
publisher |
BMC |
series |
Diagnostic Pathology |
issn |
1746-1596 |
publishDate |
2019-12-01 |
description |
Abstract Background Splenic cord capillary hemangioma is a rare benign vascular lesion classified as a splenic hamartoma. On the other hand, non-islet cell tumor hypoglycemia (NICTH) is one of the rare causes of spontaneous hypoglycemia and is considered to be one of the paraneoplastic syndromes. To the best of our knowledge, this is the first reported case of a splenic cord capillary hemangioma with NICTH. Case presentation A 25-year-old male was referred to our hospital with hypoglycemia. Except for his low blood sugar, there were no abnormal findings from laboratory tests, which included an endocrinological examination. Enhanced computed tomography confirmed the presence of a solid mass measuring about 6 cm in the retroperitoneum, and a tumorectomy was performed. During this operation, it became clear that the tumor turned out to be a splenic parenchyma, and as a result, a total splenectomy was performed. Microscopically, we diagnosed this as a cord capillary hemangioma, and through immunohistochemistry, we found that some tumor cells were positive for insulin-like growth factor -II. Fortunately, the hypoglycemia-related symptoms disappeared after surgical resection was performed. The patient is still alive and well without evidence of local tumor recurrence 15 years after the operation. Conclusions Splenic cord capillary hemangioma, one of the types of splenic hamartomas, is a very rare benign vascular lesion and might be associated with hypoglycemia thought to be NICTH. |
topic |
Insulin-like growth factor-II Non-islet cell tumor hypoglycemia Splenic cord capillary hemangioma Splenic hamartoma Splenic tumor |
url |
https://doi.org/10.1186/s13000-019-0915-0 |
work_keys_str_mv |
AT tatsuakidaimon spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT takeokosaka spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT minoruhorinaga spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT junichisaito spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT yoshitoueyama spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT shojimatsuzaki spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport AT mototsuguoya spleniccordcapillaryhemangiomawithnonisletcelltumorhypoglycemiaacasereport |
_version_ |
1724376847221784576 |