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1.
Front Endocrinol (Lausanne) ; 13: 961256, 2022.
Article in English | MEDLINE | ID: mdl-36004344

ABSTRACT

Ectopic thyroid-stimulating hormone (TSH)oma located outside the sella turcica is exceedingly rare and can be associated with significant diagnostic delay. The clinical presentation depends on the anatomical location and size of the ectopic tumor and the degree of thyrotoxicosis. A 71-year-old woman presented with goiter and thyrotoxicosis. Initial investigations revealed elevated free thyroxine (fT4) and tri-iodothyronine (fT3) with inappropriately high-normal TSH. Assay interference was unlikely, pituitary magnetic resonance imaging (MRI) scan was reported as "normal," and germline sequencing was negative for thyroid hormone receptor ß pathogenic variants. One year later, total thyroidectomy for enlarging symptomatic goiter and suspicious nodule revealed multifocal microscopic papillary thyroid carcinoma. Six years later, she presented to an ear, nose, and throat surgeon with nasal congestion, and a sphenoid bone mass was discovered on nasoendoscopy and imaging. Ectopic TSHoma was confirmed on surgical resection, and a review of the initial pituitary MRI scan revealed the mass which had initially been missed. This is the first reported case of an ectopic TSHoma located in the sphenoid bone. Ectopic TSHoma should be considered in patients with inappropriate TSH secretion when more common differentials are excluded including thyroid hormone resistance or pituitary TSHoma.


Subject(s)
Adenoma , Goiter , Pituitary Neoplasms , Thyroid Neoplasms , Thyrotoxicosis , Adenoma/complications , Adenoma/diagnostic imaging , Adenoma/surgery , Aged , Delayed Diagnosis , Female , Goiter/complications , Humans , Pituitary Neoplasms/complications , Pituitary Neoplasms/diagnostic imaging , Pituitary Neoplasms/surgery , Sphenoid Bone , Thyroid Cancer, Papillary/complications , Thyroid Neoplasms/complications , Thyrotoxicosis/complications , Thyrotropin
2.
J Endocr Soc ; 5(12): bvab167, 2021 Dec 01.
Article in English | MEDLINE | ID: mdl-34877444

ABSTRACT

Ovarian hyperthecosis (OHT), severe hyperandrogenism after menopause in the absence of ovarian or adrenal tumors, is usually treated by surgical excision. We report a 58-year-old woman presenting with severe hyperandrogenism (serum testosterone 15.7-31.0 nmol/L, normal female <1.8 nmol/L) with menopausal gonadotropins and virilization but no adrenal or ovarian lesions. Multisteroid profiling by liquid chromatography mass spectrometry (LCMS) of adrenal and ovarian vein samples identified strong gradients in the left ovarian vein (10- to 30-fold vs peripheral blood in 17OHP4, 17 hydroxyprogesterone, 17 hydroxypregnenolone, androstenedione, testosterone, dehydroepiandrosterone) but the right ovarian vein could not be cannulated with the same findings in a second ovarian vein cannulation. OHT diagnosis was confirmed by an injection of a depot pure gonadotropin-releasing hormone (GnRH) antagonist (80 mg Degarelix, Ferring) producing a rapid (<24 hour) and complete suppression of ovarian steroidogenesis as well as serum luteinizing hormone and follicle-stimulating hormone lasting at least 8 weeks, with reduction in virilization but injection site reaction and flushing and vaginal spotting ameliorated by an estradiol patch. Serum testosterone remained suppressed at 313 days after the first dose despite recovery of menopausal gonadotropins by day 278 days. This illustrates use of multisteroid LCMS profiling for confirmation of the OHT diagnosis by ovarian and adrenal vein sampling and monitoring of treatment by peripheral blood sampling. Injection of a depot pure GnRH antagonist produced rapid and long-term complete suppression of ovarian steroidogenesis maintained over 10 months. Hence a depot pure GnRH antagonist can not only rapidly confirm the OHT diagnosis but also induce long-term remission of severe hyperandrogenism without surgery.

3.
Endocr Regul ; 55(3): 163-168, 2021 Sep 13.
Article in English | MEDLINE | ID: mdl-34523297

ABSTRACT

Objective. Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by chronic hypophosphatemia and osteomalacia. We present case of a patient with a protracted clinical course of TIO. TIO profoundly affected every aspect of his life with subsequent profound physical and psychosocial disabilities. Method. The review of a complex clinical presentation, serial laboratory investigations, and imaging modalities of a patient with TIO caused by a mesenchymal tumor. Results. The patient presented with chronic lower back pain, severe bilateral leg weakness, and multiple pathological fractures due to severe osteoporosis. His investigations revealed hypophosphatemia, low 1,25 dihydroxyvitamin D, phosphaturia and normal serum calcium, and parathyroid hormone. Elevated fibroblast growth factor 23 (FGF23) confirmed the diagnosis of TIO and 68Ga-DOTATATE-positron emission tomography/computed tomography (PET/CT) imaging correctly identified a tumor in the left femoral head. His clinical features and biochemical abnormalities promptly recovered after successful surgical resection of the mesenchymal tumor. Conclusion. The present case demonstrated the need to extensively investigate causes of generalized bone pain in patients with hypophosphatemia, as TIO is highly curable. Importantly, 68Ga-DOTATATE PET/CT imaging successfully identified the FGF23 producing tumor, which was undetectable by conventional imaging, favoring its early use in suspected TIO presentation. The present report highlights the importance of timely diagnosis of this complex medical condition, aiming to improve general awareness and enable better clinical outcomes for this rare disorder.


Subject(s)
Neoplasms, Connective Tissue , Osteomalacia , Paraneoplastic Syndromes , Depression , Fibroblast Growth Factor-23 , Humans , Neoplasms, Connective Tissue/complications , Neoplasms, Connective Tissue/diagnostic imaging , Osteomalacia/diagnostic imaging , Osteomalacia/etiology , Pain , Paraneoplastic Syndromes/etiology , Positron Emission Tomography Computed Tomography , Positron-Emission Tomography , Radionuclide Imaging
4.
J Cell Mol Med ; 23(1): 556-567, 2019 01.
Article in English | MEDLINE | ID: mdl-30467960

ABSTRACT

Angiogenesis has a great impact on human health, owing to its participation in development, wound healing and the pathogenesis of several diseases. It has been reported that let-7a is a tumour suppressor, but whether it plays a role in angiogenesis is unclear. Here we showed that let-7a, a microRNA conserved in vertebrates, regulated angiogenesis by concomitantly down-regulating TGFBR3. Overexpression of let-7a or knockdown of TGFBR3 in cell culture inhibited the tube formation and reduced migration rate. Moreover, xenograft experiments showed that overexpression of let-7a or knockdown of TGFBR3 had smaller tumour size. Downstream genes, such as VEGFC and MMP9, were also down-regulated in let-7a overexpression or TGFBR3 knockdown groups. Therefore, our results revealed a novel mechanism that let-7a regulate angiogenesis through post-transcriptional regulation of TGFBR3.


Subject(s)
MicroRNAs/genetics , Neovascularization, Pathologic/genetics , Proteoglycans/genetics , RNA, Messenger/genetics , Receptors, Transforming Growth Factor beta/genetics , Animals , Cell Line , Down-Regulation/genetics , Gene Expression Regulation, Neoplastic/genetics , HEK293 Cells , Human Umbilical Vein Endothelial Cells , Humans , Mice , Mice, Nude , Neovascularization, Pathologic/pathology , RNA Processing, Post-Transcriptional/genetics
5.
Cardiovasc Ther ; 36(6): e12477, 2018 Dec.
Article in English | MEDLINE | ID: mdl-30380183

ABSTRACT

AIMS: To investigate whether there exists a cardio-protective effect of Fasudil, a selective Rho kinase (ROCK) inhibitor, in an experimental murine model of acute viral myocarditis. METHODS: Male BALB/c mice were randomly assigned to three groups: control, myocarditis treated with placebo and myocarditis treated with Fasudil (n = 40 animals per group). Myocarditis was established by intraperitoneal injection with coxsackievirus B3 (CVB3). Twenty-four hours after infection, Fasudil was intraperitoneally administered for 14 consecutive days. Twenty mice were randomly selected from each group to monitor a 14-day survival rate. On day 7 and day 14, eight surviving mice from each group were sacrificed and their hearts and blood were obtained to perform serological and histological examinations. Expression of ROCKs, IL-17, IL-1b, TNFα, RORgt, and Foxp3 were quantified with RT-PCR. Plasma levels of TNF alpha, IL-1 beta, and IL-17 were measured by ELISA. In addition, protein levels of IL-17 and ROCK2 in cardiac tissues were analyzed with Western blot. RESULTS: Fasudil treatment significantly increased survival, attenuated myocardial necrotic lesions, reduced CVB3 replication and expression of ROCK2 and IL-17 in the infected hearts. This treatment also imposed a T-cell subpopulation shift, from Th17 to Treg, in cardiac tissues. CONCLUSIONS: ROCK pathway inhibition was cardio-protective in viral myocarditis with increased survival, decreased viral replication, and inflammatory response. These findings suggest that Fasudil might be a potential therapeutic agent for patients with viral myocarditis.


Subject(s)
1-(5-Isoquinolinesulfonyl)-2-Methylpiperazine/analogs & derivatives , Anti-Inflammatory Agents/pharmacology , Antiviral Agents/pharmacology , Coxsackievirus Infections/prevention & control , Enterovirus B, Human/drug effects , Myocarditis/prevention & control , Myocardium/enzymology , Protein Kinase Inhibitors/pharmacology , rho-Associated Kinases/antagonists & inhibitors , 1-(5-Isoquinolinesulfonyl)-2-Methylpiperazine/pharmacology , Animals , Coxsackievirus Infections/enzymology , Coxsackievirus Infections/pathology , Coxsackievirus Infections/virology , Cytokines/blood , Disease Models, Animal , Enterovirus B, Human/growth & development , Inflammation Mediators/blood , Male , Mice, Inbred BALB C , Myocarditis/enzymology , Myocarditis/pathology , Myocarditis/virology , Myocardium/pathology , Necrosis , T-Lymphocytes, Regulatory/drug effects , T-Lymphocytes, Regulatory/metabolism , T-Lymphocytes, Regulatory/virology , Th1 Cells/drug effects , Th1 Cells/metabolism , Th1 Cells/virology , Viral Load , Virus Replication/drug effects , rho-Associated Kinases/metabolism
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