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Evolution of Pleural Solitary Fibrous Tumors Causing Severe Hypoglycemia after Exceptionally Long Asymptomatic Periods : Report of Two Surgical Cases

Suzuki, Takaya ; Notsuda, Hirotsugu ; Oishi, Hisashi ; Niikawa, Hiromichi ; Watanabe, Tatsuaki ; Watanabe, Yui ; Onodera, Ken ; Takeda, Tetsuto ; Sugawara, Ringo and Noda, Masafumi , et al. (2024) In The Tohoku journal of experimental medicine 263(1). p.11-16
Abstract

Non-islet cell tumor hypoglycemia (NICTH) is one of the paraneoplastic syndromes manifesting severe hypoglycemia caused by aberrant production of high-molecular-weight insulin-like growth factor 2 (big-IGF2). Two surgical cases of extremely large thoracic solitary fibrous tumors (SFT) with unusual history of NICTH are presented. One case manifested severe hypoglycemia after four years of the first complete surgical resection of the tumor with potential malignant transformation, and the other case showed severe hypoglycemia after ten years of the first detection of the tumor. Meticulous laboratory testing, including serum endocrinological tests and western immunoblotting before and after surgery was performed, and both cases were... (More)

Non-islet cell tumor hypoglycemia (NICTH) is one of the paraneoplastic syndromes manifesting severe hypoglycemia caused by aberrant production of high-molecular-weight insulin-like growth factor 2 (big-IGF2). Two surgical cases of extremely large thoracic solitary fibrous tumors (SFT) with unusual history of NICTH are presented. One case manifested severe hypoglycemia after four years of the first complete surgical resection of the tumor with potential malignant transformation, and the other case showed severe hypoglycemia after ten years of the first detection of the tumor. Meticulous laboratory testing, including serum endocrinological tests and western immunoblotting before and after surgery was performed, and both cases were diagnosed as NICTH. Both patients underwent open thoracic surgery. The patients showed normal glucose and hormone levels immediately after the resection of responsible tumors with elevated blood insulin concentration. SFTs are generally considered benign; however, life-threatening hypoglycemia can happen regardless of treatment. Careful follow-up of the tumor growth is warranted.

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publishing date
type
Contribution to journal
publication status
published
subject
keywords
Humans, Hypoglycemia/etiology, Insulin-Like Growth Factor II/metabolism, Solitary Fibrous Tumor, Pleural/surgery, Tomography, X-Ray Computed
in
The Tohoku journal of experimental medicine
volume
263
issue
1
pages
11 - 16
publisher
Tohoku University Medical Press
external identifiers
  • scopus:85194112033
  • pmid:38325831
ISSN
0040-8727
DOI
10.1620/tjem.2024.J012
language
English
LU publication?
no
id
024b749c-9d9e-4268-ab36-b5fa609e1107
date added to LUP
2025-01-12 01:56:01
date last changed
2025-06-15 16:17:01
@article{024b749c-9d9e-4268-ab36-b5fa609e1107,
  abstract     = {{<p>Non-islet cell tumor hypoglycemia (NICTH) is one of the paraneoplastic syndromes manifesting severe hypoglycemia caused by aberrant production of high-molecular-weight insulin-like growth factor 2 (big-IGF2). Two surgical cases of extremely large thoracic solitary fibrous tumors (SFT) with unusual history of NICTH are presented. One case manifested severe hypoglycemia after four years of the first complete surgical resection of the tumor with potential malignant transformation, and the other case showed severe hypoglycemia after ten years of the first detection of the tumor. Meticulous laboratory testing, including serum endocrinological tests and western immunoblotting before and after surgery was performed, and both cases were diagnosed as NICTH. Both patients underwent open thoracic surgery. The patients showed normal glucose and hormone levels immediately after the resection of responsible tumors with elevated blood insulin concentration. SFTs are generally considered benign; however, life-threatening hypoglycemia can happen regardless of treatment. Careful follow-up of the tumor growth is warranted.</p>}},
  author       = {{Suzuki, Takaya and Notsuda, Hirotsugu and Oishi, Hisashi and Niikawa, Hiromichi and Watanabe, Tatsuaki and Watanabe, Yui and Onodera, Ken and Takeda, Tetsuto and Sugawara, Ringo and Noda, Masafumi and Sakurai, Kanako and Nagao, Mototsugu and Fukuda, Izumi and Okada, Yoshinori}},
  issn         = {{0040-8727}},
  keywords     = {{Humans; Hypoglycemia/etiology; Insulin-Like Growth Factor II/metabolism; Solitary Fibrous Tumor, Pleural/surgery; Tomography, X-Ray Computed}},
  language     = {{eng}},
  month        = {{05}},
  number       = {{1}},
  pages        = {{11--16}},
  publisher    = {{Tohoku University Medical Press}},
  series       = {{The Tohoku journal of experimental medicine}},
  title        = {{Evolution of Pleural Solitary Fibrous Tumors Causing Severe Hypoglycemia after Exceptionally Long Asymptomatic Periods : Report of Two Surgical Cases}},
  url          = {{http://dx.doi.org/10.1620/tjem.2024.J012}},
  doi          = {{10.1620/tjem.2024.J012}},
  volume       = {{263}},
  year         = {{2024}},
}