Solitary fibrous tumour of the liver with IGF-II-related hypoglycaemia. A case report

Famà, Fausto; Bouc, Yves Le; Barrande, Gaëlle; Villeneuve, A.; Berry, Miles G.; Pidoto, Rocco Roberto; Marc, Olivier Saint

doi:10.1007/s00423-008-0329-z

Cited by 37 publications

(44 citation statements)

References 27 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…Interestingly, association to hypoglycemia, possibly mediated by insulin growth factors, has been described in 4% to 10% of cases (7)(8)(9)(10), more often in SFT arising from peritoneum.…”

Section: Introductionmentioning

confidence: 99%

Sunitinib Malate and Figitumumab in Solitary Fibrous Tumor: Patterns and Molecular Bases of Tumor Response

Stacchiotti

Negri

Palassini

et al. 2010

Molecular Cancer Therapeutics

View full text Add to dashboard Cite

Antiangiogenic treatment activity has been reported in solitary fibrous tumor (SFT), a rare and little chemosensitive sarcoma. We explored the activity of sunitinib malate (SM) in SFT and studied receptor tyrosine kinase (RTK) activation profile. Eleven patients with progressive metastatic SFT resistant to chemotherapy were treated with continuous-dosing 37.5 mg/d SM on a named-use basis. One of them also received the insulin-like growth factor I receptor (IGFIR) inhibitor figitumumab after developing secondary resistance to SM. Besides, biochemical, molecular, and fluorescence in situ hybridization analyses were done in eight naïve SFTs whose cryopreserved material was available to clarify RTK upstream and downstream signaling. In two cases treated with SM and belonging to the naïve series, both pretreatment and posttreatment samples were available. Ten patients were evaluable for response to SM. The best response according to the Choi criteria was six partial response (all with Response Evaluation Criteria in Solid Tumors stable disease), one stable disease, and three progressive disease. Responses lasted >6 months in five patients. The eight naïve samples showed high expression/phosphorylation of PDGFRB, epidermal growth factor receptor, and IGFIR/IR, in the presence of their cognate ligands. Downstream pathways revealed expression/activation of Akt, extracellular signal-regulated kinase 1-2 and, closely related to SFT subtypes, of S6 and 4E-BP1. In two patients, whose pretreatment and posttreatment clinical and molecular status were available, biochemical data confirmed the activity of SM, although they also suggested a possible time-dependent shift of dominant RTK from PDGFRB to IGFIR/insulin receptor. A Response Evaluation Criteria in Solid Tumors partial response to figitumumab corroborated these findings. SM has antitumor activity in SFT, possibly through a PDGFRB-mediated mechanism, but treatments with IGFIR/ insulin receptor and possibly epidermal growth factor receptor inhibitors are worth testing. Mol Cancer Ther; 9(5); 1286-97. ©2010 AACR.

show abstract

“…Interestingly, association to hypoglycemia, possibly mediated by insulin growth factors, has been described in 4% to 10% of cases (7)(8)(9)(10), more often in SFT arising from peritoneum.…”

Section: Introductionmentioning

confidence: 99%

Sunitinib Malate and Figitumumab in Solitary Fibrous Tumor: Patterns and Molecular Bases of Tumor Response

Stacchiotti

Negri

Palassini

et al. 2010

Molecular Cancer Therapeutics

View full text Add to dashboard Cite

show abstract

“…Los TFSH son tumores primitivos con diferenciación multidireccional que se originan en el mesénquima y que ocasionalmente comprometen el hígado 6,7 . La mayoría de los TFSH son benignos pero algunos pueden tener un comportamiento maligno 6,15 .…”

Section: Discussionunclassified

“…Los TFSH se presentan con mayor frecuencia en las mujeres (72%) 8 . La edad promedio de presentación es de 55 a 59 años pero se han reportado entre los 16 y 83 años de edad [4][5][6][7][8][9][10][11][12][13][14][15][16] . nuestro paciente, masculino, se encontraba dentro del promedio de edad habitual de presentación de estos tumores.…”

Section: Discussionunclassified

“…El tumor fibroso solitario del hígado (TFSH) constituye un hallazgo tan raro que se ha publicado únicamente como reportes de caso y hasta el presente año se encontraron sólo 45 casos publicados en la literatura inglesa [4][5][6][7][8][9][10][11][12][13][14][15] . Las manifestaciones clínicas son inespecíficas, habitualmente referidas al efecto de masa y compresión que el TFSH produce sobre los órganos vecinos [4][5][6][7][8][9][10][11] . El diagnóstico es habitualmente incidental y en algunos casos dirigido durante el estudio radiológico de los síntomas del paciente [11][12][13][14] .…”

Section: Introductionunclassified

“…El diagnóstico es habitualmente incidental y en algunos casos dirigido durante el estudio radiológico de los síntomas del paciente [11][12][13][14] . El tratamiento consiste en la resección tumoral pero el pronóstico no se ha establecido claramente aún debido al limitado número de casos [4][5][6][7][8][9][10][11][12][13][14][15] . El diagnóstico definitivo se establece con la inmunohistoquímica debido a que histológicamente presenta características similares a los tumores estromales y mesenquimatosos.…”

Section: Introductionunclassified

See 2 more Smart Citations

Tumor Fibroso Solitario Del Lóbulo Caudado Del Hígado

Valenzuela

et al. 2015

Rev Chil Cir

View full text Add to dashboard Cite

Solitary fibrous tumor of the liver caudate lobeintroduction: Solitary fibrous tumor of the liver (SFTL) constitutes such a rare finding that it has been published only as case reports and to the present date there are only 45 cases published in English medical literature. This article describes the case of a patient treated at our institution with SFTL. case report: A male 59-years-old patient was diagnosed with a SFTL incidentally found in a computed abdominal tomography taken by another causes, tumor was originating from the caudate lobe. The patient was asymptomatic. At surgery, the tumor was observed originating directly from the caudate lobe through a pedicle measuring approximately 3 cm. The tumor was resected sectioning its implantation pedicle and ligating all arterial, venous and biliary vessels. The SFTL was well encapsulated, measured 15 x 9 x 6 cm and weighted 794 g. The histological diagnosis was a SFTL and was confirmed by immunohistochemistry with monoclonal antibodies that reacted positively to CD34 and vimentin and negatively to CD117, S100, smooth-muscle α-actin and desmin. conclusions: SFTL is an uncommon tumor. The present case is the first reported in Chilean medical literature and presented all the habitually described radiologic, surgical and pathological characteristics.

show abstract

Activation of the Akt‐mTOR pathway and receptor tyrosine kinase in patients with solitary fibrous tumors

Yamada

Kohashi

Fushimi

et al. 2013

Cancer

View full text Add to dashboard Cite

BACKGROUND Solitary fibrous tumors (SFTs) are soft tissue tumors of intermediate malignancy that rarely metastasize. Although unresectable SFTs are reported to have a poor prognosis, to the authors' knowledge there is currently no effective therapy. Molecular target therapy is a promising approach for patients with unresectable tumors, but knowledge of the molecular biology of SFTs is currently insufficient to support such therapy. The current study investigated the activation of receptor tyrosine kinases (RTKs) and the Akt‐mammalian target of rapamycin (Akt‐mTOR) pathway in SFTs as therapeutic targets. METHODS The phosphorylation statuses of Akt‐mTOR pathway proteins (p‐Akt, p‐mTOR, phosphorylated 4E‐binding protein [p‐4EBP1], and phosphorylated S6 ribosomal protein [p‐S6RP]) and RTKs (phosphorylated platelet‐derived growth factor receptor‐α [p‐PDGFRα], p‐PDGFRβ, p‐c‐met, and phosphorylated insulin‐like growth factor‐1 receptor‐β [p‐IGF‐1Rβ]) were assessed by immunohistochemistry in 66 samples of SFTs, and the data were compared with clinicopathological and histopathological findings. The expression of phosphorylated proteins was assessed by Western blot analysis in 6 frozen samples. RESULTS The immunohistochemical results were as follows: p‐Akt, 56.0% (nuclear and cytoplasmic staining); p‐mTOR, 69.6% (nuclear and cytoplasmic staining); p‐4EBP1, 80.3% (nuclear and cytoplasmic staining); p‐S6RP, 69.6% (cytoplasmic staining); p‐PDGFRα, 39.0% (cytoplasmic staining); p‐PDGFRβ, 52.0% (cytoplasmic staining); p‐c‐met, 37.8% (nuclear staining) and 19.6% (cytoplasmic staining); and p‐IGF‐1Rβ, 16.6% (nuclear staining). Phosphorylation of the Akt‐mTOR pathway proteins was correlated with one another except for p‐Akt with S6RP. p‐PDGFRβ and p‐IGF‐1Rβ were correlated with p‐Akt. Moreover, significant relationships were noted between disease‐free survival or overall survival and the presence of hypoglycemia, necrosis, cystic and myxoid degeneration, and atypical findings. CONCLUSIONS The Akt/mTOR pathway was activated in approximately 50% of the cases of SFTs and was associated with RTKs, which were phosphorylated at different rates. Thus, the Akt‐mTOR pathway may be involved in the tumorigenesis of SFTs. Cancer 2014;120:864–876. © 2013 American Cancer Society.

show abstract

Solitary fibrous tumour of the liver with IGF-II-related hypoglycaemia. A case report

Abstract: We report for the first time a demonstrable relationship between benign liver SFT and NICTH.

Cited by 37 publications

References 27 publications

Sunitinib Malate and Figitumumab in Solitary Fibrous Tumor: Patterns and Molecular Bases of Tumor Response

Sunitinib Malate and Figitumumab in Solitary Fibrous Tumor: Patterns and Molecular Bases of Tumor Response

Tumor Fibroso Solitario Del Lóbulo Caudado Del Hígado

Activation of the Akt‐mTOR pathway and receptor tyrosine kinase in patients with solitary fibrous tumors

Contact Info

Product

Resources

About