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1.
Chin Med Sci J ; 39(2): 102-110, 2024 Jun 30.
Artículo en Inglés | MEDLINE | ID: mdl-38755752

RESUMEN

Objective To investigate the efficacy of raw corn starch (RCS) in clinical management of insulinoma-induced hypoglycemia. Methods We retrospectively collected clinical data of insulinoma patients who received RCS-supplemented diet preoperatively, and analyzed the therapeutic effects of the RCS intervention on blood glucose control, weight change, and its adverse events. Results The study population consisted of 24 cases of insulinoma patients, 7 males and 17 females, aged 46.08±14.15 years. Before RCS-supplemented diet, all patients had frequent hypoglycemic episodes (2.51±3.88 times/week), concurrent with neuroglycopenia (in 83.3% of patients) and autonomic manifestations (in 75.0% of patients), with the median fasting blood glucose (FBG) of 2.70 (interquartile range [IQR]: 2.50-2.90) mmol/L. The patients' weight increased by 0.38 (IQR: 0.05-0.65) kg per month, with 8 (33.3%) cases developing overweight and 7 (29.2%) cases developing obesity. All patients maintained the RCS-supplemented diet until they underwent tumor resection (23 cases) and transarterial chemoembolization for liver metastases (1 case). For 19 patients receiving RCS throughout the day, the median FBG within one week of nutritional management was 4.30 (IQR: 3.30-5.70) mmol/L, which was a significant increase compared to pre-nutritional level [2.25 (IQR: 1.60-2.90) mmol/L; P < 0.001]. Of them, 10 patients receiving RCS throughout the day for over four weeks had sustained improvement in FBG compared to pre-treatment [3.20 (IQR: 2.60-3.95) mmol/L vs. 2.15 (IQR: 1.83-2.33) mmol/L; P < 0.001). Five patients who received RCS only at night also had a significant increase in FBG within one week of nutritional management [3.50 (IQR: 2.50-3.65) mmol/L vs. 2.20 (IQR:1.80-2.60) mmol/L; P < 0.001], but only one patient who continued to receive RCS for over four weeks did not have a significant improvement in FBG. No improvement in weight gain was observed upon RCS supplementation. Mild diarrhea (2 cases) and flatulence (1 case) occurred, and were relieved by reduction of RCS dose. Conclusion The RCS-supplemented diet is effective in controlling insulinoma-induced hypoglycemia.


Asunto(s)
Hipoglucemia , Insulinoma , Almidón , Humanos , Femenino , Persona de Mediana Edad , Masculino , Insulinoma/complicaciones , Insulinoma/terapia , Adulto , Almidón/uso terapéutico , Estudios Retrospectivos , Glucemia/metabolismo , Neoplasias Pancreáticas/complicaciones , Anciano
2.
Front Immunol ; 15: 1384406, 2024.
Artículo en Inglés | MEDLINE | ID: mdl-38596681

RESUMEN

Introduction: The autoimmune response in type 1 diabetes (T1D), in which the beta cells expressing aberrant or modified proteins are killed, resembles an effective antitumor response. Defective ribosomal protein products in tumors are targets of the anti-tumor immune response that is unleashed by immune checkpoint inhibitor (ICI) treatment in cancer patients. We recently described a defective ribosomal product of the insulin gene (INS-DRiP) that is expressed in stressed beta cells and targeted by diabetogenic T cells. T1D patient-derived INS-DRiP specific T cells can kill beta cells and are present in the insulitic lesion. T cells reactive to INS-DRiP epitopes are part of the normal T cell repertoire and are believed to be kept in check by immune regulation without causing autoimmunity. Method: T cell autoreactivity was tested using a combinatorial HLA multimer technology measuring a range of epitopes of islet autoantigens and neoantigen INS-DRiP. INS-DRiP expression in human pancreas and insulinoma sections was tested by immunohistochemistry. Results: Here we report the induction of islet autoimmunity to INS-DRiP and diabetes after ICI treatment and successful tumor remission. Following ICI treatment, T cells of the cancer patient were primed against INS-DRiP among other diabetogenic antigens, while there was no sign of autoimmunity to this neoantigen before ICI treatment. Next, we demonstrated the expression of INS-DRiP as neoantigen in both pancreatic islets and insulinoma by staining with a monoclonal antibody to INS-DRiP. Discussion: These results bridge cancer and T1D as two sides of the same coin and point to neoantigen expression in normal islets and insulinoma that may serve as target of both islet autoimmunity and tumor-related autoimmunity.


Asunto(s)
Diabetes Mellitus Tipo 1 , Insulinoma , Neoplasias Pancreáticas , Humanos , Diabetes Mellitus Tipo 1/genética , Diabetes Mellitus Tipo 1/terapia , Autoinmunidad/genética , Insulinoma/genética , Insulinoma/terapia , Insulinoma/complicaciones , Autoantígenos , Insulina , Epítopos , Inmunoterapia/métodos
3.
J Clin Endocrinol Metab ; 109(4): 1109-1118, 2024 Mar 15.
Artículo en Inglés | MEDLINE | ID: mdl-37925662

RESUMEN

Insulinomas are hormone-producing pancreatic neuroendocrine neoplasms with an estimated incidence of 1 to 4 cases per million per year. Extrapancreatic insulinomas are extremely rare. Most insulinomas present with the Whipple triad: (1) symptoms, signs, or both consistent with hypoglycemia; (2) a low plasma glucose measured at the time of the symptoms and signs; and (3) relief of symptoms and signs when the glucose is raised to normal. Nonmetastatic insulinomas are nowadays referred to as "indolent" and metastatic insulinomas as "aggressive." The 5-year survival of patients with an indolent insulinoma has been reported to be 94% to 100%; for patients with an aggressive insulinoma, this amounts to 24% to 67%. Five percent to 10% of insulinomas are associated with the multiple endocrine neoplasia type 1 syndrome. Localization of the insulinoma and exclusion or confirmation of metastatic disease by computed tomography is followed by endoscopic ultrasound or magnetic resonance imaging for indolent, localized insulinomas. Glucagon-like peptide 1 receptor positron emission tomography/computed tomography or positron emission tomography/magnetic resonance imaging is a highly sensitive localization technique for seemingly occult, indolent, localized insulinomas. Supportive measures and somatostatin receptor ligands can be used for to control hypoglycemia. For single solitary insulinomas, curative surgical excision remains the treatment of choice. In aggressive malignant cases, debulking procedures, somatostatin receptor ligands, peptide receptor radionuclide therapy, everolimus, sunitinib, and cytotoxic chemotherapy can be valuable options.


Asunto(s)
Hipoglucemia , Insulinoma , Tumores Neuroendocrinos , Neoplasias Pancreáticas , Humanos , Insulinoma/diagnóstico , Insulinoma/terapia , Insulinoma/complicaciones , Receptores de Somatostatina/uso terapéutico , Neoplasias Pancreáticas/terapia , Neoplasias Pancreáticas/tratamiento farmacológico , Hipoglucemia/diagnóstico , Hipoglucemia/etiología , Hipoglucemia/terapia , Tumores Neuroendocrinos/complicaciones
4.
In Vivo ; 37(6): 2402-2408, 2023.
Artículo en Inglés | MEDLINE | ID: mdl-37905620

RESUMEN

BACKGROUND/AIM: Pancreatic neuroendocrine tumors (PNETs) are pancreatic neoplasms with neuroendocrine features, divided into functioning and non-functioning. The non-functioning PNETs are the largest group, and their morbidity is the result of their potential to invade surrounding tissues and metastasize. The functioning PNETs produce hormonal symptoms due to over-secretion of specific hormones. They constitute 1% to 2% of all pancreatic tumors. The use of novel imaging methods has rendered their detection more frequent. Insulinoma, the most common functioning PNET, comprises 35-40% of all functioning PNETs. Its clinical presentation is due to hyperinsulinemia and the subsequent hypoglycemia. Glucagonoma accounts for 5% of all PNETs and is the fourth most frequent functioning PNET, following insulinoma, gastrinoma, and vipoma. Its symptoms are due to the massive secretion of glucagon and ensuing hyperglycemia. The co-existence of two PNETs is a very rare entity. This report aimed to describe cases of concomitant insulinomas and glucagonomas. MATERIALS AND METHODS: A review of the literature was performed using the PubMed database and Cochrane library aiming to identify reported cases of concomitant pancreatic insulinoma and glucagonoma. Specifically, the research was conducted using the keywords, separately and in various combination, including insulinoma, glucagonoma, cystic, pancreatic neuroendocrine tumors and hypoglycemia. Only publications in English were included in the present study. RESULTS: A total of 8 cases of concomitant pancreatic insulinoma and glucagonoma were identified, corresponding to the period 1992-2021. CONCLUSION: Concomitant insulinoma and glucagonoma are rare and challenging. A multidisciplinary approach is necessary for diagnosis, prognosis, and therapy.


Asunto(s)
Glucagonoma , Hipoglucemia , Insulinoma , Tumores Neuroectodérmicos Primitivos , Tumores Neuroendocrinos , Neoplasias Pancreáticas , Humanos , Insulinoma/diagnóstico , Insulinoma/terapia , Glucagonoma/diagnóstico , Glucagonoma/terapia , Tumores Neuroendocrinos/terapia , Neoplasias Pancreáticas/patología , Hipoglucemia/diagnóstico , Hipoglucemia/etiología
5.
J Neuroendocrinol ; 35(8): e13318, 2023 08.
Artículo en Inglés | MEDLINE | ID: mdl-37578384

RESUMEN

This ENETS guidance paper aims to provide practical advice to clinicians for the diagnosis, treatment and follow-up of functioning syndromes in pancreatic neuroendocrine tumours (NET). A NET-associated functioning syndrome is defined by the presence of a clinical syndrome combined with biochemical evidence of inappropriately elevated hormonal levels. Different hormonal syndromes can be encountered in pancreatic NET patients, including insulinoma, gastrinoma as well as the rare glucagonoma, VIPoma, ACTHoma, PTHrPoma, carcinoid syndrome, calcitoninoma, GHRHoma and somatostatinoma. The recommendations provided in this paper focus on the biochemical, genetic and imaging work-up as well as therapeutic management of the individual hormonal syndromes in well-differentiated, grade 1-3, functioning NET with the primary tumour originating in the pancreas, and for specific subtypes also in the duodenum.


Asunto(s)
Gastrinoma , Glucagonoma , Insulinoma , Tumores Neuroendocrinos , Neoplasias Pancreáticas , Humanos , Tumores Neuroendocrinos/diagnóstico , Tumores Neuroendocrinos/terapia , Neoplasias Pancreáticas/diagnóstico , Neoplasias Pancreáticas/terapia , Insulinoma/diagnóstico , Insulinoma/terapia , Gastrinoma/diagnóstico , Gastrinoma/terapia , Glucagonoma/diagnóstico , Glucagonoma/terapia
7.
J Clin Endocrinol Metab ; 106(12): 3477-3486, 2021 11 19.
Artículo en Inglés | MEDLINE | ID: mdl-34343300

RESUMEN

CONTEXT: Insulinoma is the most common pancreatic functional neuroendocrine neoplasm, yet little information on recent clinical practice in patients with insulinoma, especially malignant insulinoma, is available. OBJECTIVE: This work aims to clarify the characteristics and practice patterns in patients with insulinoma using a national inpatient database. METHODS: Using the Japanese Diagnosis Procedure Combination database, we retrospectively identified patients with insulinoma admitted between 2010 and 2018. We compared background characteristics and therapeutic interventions between patients with benign and malignant insulinoma. We also estimated the incidence of insulinoma using the number of patients with newly diagnosed insulinoma in 2012. RESULTS: We identified 844 patients with benign insulinoma and 102 patients with malignant insulinoma. Patients with malignant insulinoma were younger (median, 55.5 vs 66.0 years, P < .001) and less likely to be female (55.9% vs 65.3%, P = .061) than patients with benign insulinoma. Analysis of therapeutic interventions revealed that patients with malignant insulinoma more frequently received medications (71.6% vs 49.6%, P < .001) but less frequently underwent pancreatic surgery (57.8% vs 72.0%, P = .003). Older patients were a smaller proportion of those undergoing surgery and a larger proportion of those managed with medications without surgery (P < .001). The incidence of insulinoma was estimated to be 3.27 (95% CI, 2.93-3.61) individuals per million Japanese adult population per year. CONCLUSION: The present study using a nationwide database had a larger sample size than previous studies and revealed definitive differences in patient characteristics and therapeutic patterns between benign and malignant insulinoma.


Asunto(s)
Bases de Datos Factuales/estadística & datos numéricos , Pacientes Internos/estadística & datos numéricos , Insulinoma/epidemiología , Neoplasias Pancreáticas/epidemiología , Adulto , Anciano , Femenino , Estudios de Seguimiento , Humanos , Incidencia , Insulinoma/patología , Insulinoma/terapia , Japón/epidemiología , Masculino , Persona de Mediana Edad , Neoplasias Pancreáticas/patología , Neoplasias Pancreáticas/terapia , Pronóstico , Estudios Retrospectivos
8.
Ann Endocrinol (Paris) ; 82(2): 112-120, 2021 Apr.
Artículo en Inglés | MEDLINE | ID: mdl-33839123

RESUMEN

In 1953, for the first time, Paul Wermer described a family presenting endocrine gland neoplasms over several generations. The transmission was autosomal dominant and the penetrance was high. Forty years later in 1997, the multiple endocrine neoplasia type 1 (MEN1) gene was sequenced, thus enabling diagnosis and early optimal treatment. Patients carrying the MEN1 gene present endocrine but also non-endocrine tumors. Parathyroid, pancreatic and pituitary impairment are the three main types of endocrine involvement. The present article details therapeutic management of hyperparathyroidism, neuroendocrine pancreatic tumors and pituitary adenomas in patients carrying the MEN1 gene. Significant therapeutic progress has in fact been made in the last few years. As concerns the parathyroid glands, screening of family members and regular monitoring of affected subjects now raise the question of early management of parathyroid lesions and optimal timing of parathyroid surgery. As concerns the duodenum-pancreas, proton-pump inhibitors are able to control gastrin-secreting syndrome, reducing mortality in MEN1 patients. Mortality in MEN1 patients is no longer mainly secondary to uncontrolled hormonal secretion but to metastatic (mainly pancreatic) disease progression. Tumor risk requires regular monitoring of morphological assessment, leading to iterative pancreatic surgery in a large number of patients. Finally, pituitary adenomas in MEN1 patients are traditionally described as aggressive, invasive and resistant to medical treatment. However, regular pituitary screening showed them to be in fact infra-centimetric and non-secreting in the majority of patients. Consequently, it is necessary to regularly monitor MEN1 patients, with regular clinical, biological and morphological work-up. Several studies showed that this regular monitoring impairs quality of life. Building a relationship of trust between patients and care provider is therefore essential. It enables the patient to be referred for psychological or psychiatric care in difficult times, providing long-term support and preventing any breakdown in continuity of care.


Asunto(s)
Neoplasia Endocrina Múltiple Tipo 1/genética , Neoplasia Endocrina Múltiple Tipo 1/terapia , Neoplasias de la Mama/genética , Neoplasias de la Mama/terapia , Femenino , Gastrinoma/genética , Gastrinoma/terapia , Predisposición Genética a la Enfermedad , Humanos , Hiperparatiroidismo/genética , Hiperparatiroidismo/terapia , Insulinoma/genética , Insulinoma/terapia , Masculino , Tumores Neuroendocrinos/genética , Tumores Neuroendocrinos/terapia , Neoplasias Pancreáticas/genética , Neoplasias Pancreáticas/terapia , Paratiroidectomía , Neoplasias Hipofisarias/genética , Neoplasias Hipofisarias/terapia , Neoplasias del Timo/genética , Neoplasias del Timo/terapia
9.
Rev. esp. enferm. dig ; 113(1): 48-51, ene. 2021. ilus, tab
Artículo en Español | IBECS | ID: ibc-199889

RESUMEN

INTRODUCCIÓN: el insulinoma pancreático esporádico es un tumor con frecuencia benigno, generalmente solitario y menor de 20 mm. Cuando es funcionante, los pacientes experimentan síntomas relacionados con la producción excesiva de insulina. El diagnóstico es clínico, con confirmación bioquímica y radiológica. El tratamiento suele ser quirúrgico, pero en la actualidad se está desarrollando la ablación guiada por ultrasonido endoscópico. MATERIAL Y MÉTODOS: realizamos una serie de casos prospectiva de pacientes con insulinoma pancreático con síntomas de hipoglicemia severa, a quienes se les ofreció el tratamiento ablativo con etanol al 96 % guiado por ultrasonido endoscópico como tratamiento alternativo a la cirugía. RESULTADOS: el éxito técnico y clínico se consiguió en el 100 % de los casos. No se presentaron complicaciones asociados al procedimiento. CONCLUSIÓN: el tratamiento del insulinoma sintomático mediante ablación por etanolización guiada por ultrasonido endoscópico podría ser una alternativa segura y eficaz a la cirugía en pacientes no candidatos o que rechacen la intervención quirúrgica


No disponible


Asunto(s)
Humanos , Femenino , Adulto , Anciano de 80 o más Años , Insulinoma/diagnóstico por imagen , Insulinoma/terapia , Hipoglucemia/complicaciones , Etanol/administración & dosificación , Radioterapia Guiada por Imagen/instrumentación , Neoplasias Pancreáticas/diagnóstico por imagen , Estudios Prospectivos , Endosonografía/métodos , Ultrasonografía Intervencional/métodos , Biopsia por Aspiración con Aguja Fina Guiada por Ultrasonido Endoscópico/métodos , Neoplasias Pancreáticas/terapia
10.
Dig Dis Sci ; 66(5): 1533-1539, 2021 05.
Artículo en Inglés | MEDLINE | ID: mdl-32529519

RESUMEN

BACKGROUND: Insulinoma, owing to the low incidence and small volume of the tumor, is often undiagnosed. The 72-h fast test is centered on diagnosing insulinoma; however, it cannot be performed on outpatients. Our aim was to evaluate the results of a 3-h oral glucose tolerance test (3-h OGTT) for insulinoma diagnosis. METHODS: Thirty-seven patients with insulinoma were enrolled for comparison with 42 control subjects. All patients underwent 3-h OGTT with measurements of insulin and C-peptide. The secretion ratios of insulin and C-peptide at 1, 2, and 3 h were calculated by comparison with their values at 0 h. We used logistic regression analysis to establish the predictive models and compared the diagnostic efficiency by receiver operating characteristic analysis. RESULTS: The fasting insulin and C-peptide levels of insulinoma patients were both higher; however, the concentrations at 1 h and 2 h were both lower (P < 0.05). The levels at 3 h were not significantly different (P > 0.05). Our final logistic regression model was constructed as follows: logit (P) = 8.305 - 0.441 × insulin 2 h/0 h ratio - 1.679 × C-peptide 1 h/0 h ratio. A cutoff value of > 0.351 showed the highest diagnostic accuracy, with an area under the curve of 0.97, a sensitivity of 86.5%, and a specificity of 95.2%. CONCLUSIONS: The 2-h/0-h insulin ratio, as well as the 1-h/0-h C-peptide ratio, has high diagnostic efficiency for insulinoma. The 2-h OGTT can be an alternative test for diagnosing insulinoma in outpatient settings.


Asunto(s)
Péptido C/sangre , Prueba de Tolerancia a la Glucosa , Insulina/sangre , Insulinoma/diagnóstico , Neoplasias Pancreáticas/diagnóstico , Adulto , Anciano , Atención Ambulatoria , Femenino , Humanos , Insulinoma/sangre , Insulinoma/terapia , Masculino , Persona de Mediana Edad , Neoplasias Pancreáticas/sangre , Neoplasias Pancreáticas/terapia , Valor Predictivo de las Pruebas , Reproducibilidad de los Resultados , Estudios Retrospectivos , Vías Secretoras , Factores de Tiempo
12.
Rev. cuba. cir ; 59(4): e994, oct.-dic. 2020.
Artículo en Español | LILACS, CUMED | ID: biblio-1149851

RESUMEN

RESUMEN Introducción: La Nesidioblastosis es una rara afección pancreática que provoca hipoglucemia por hipersinsulinismo endógeno en la infancia. Es poco habitual en el adulto; solo se han publicado casos aislados desde su descripción. Objetivo: Caracterizar la presentación de una hipoglucemia hiperinsulínica en un paciente adulto con Nesidioblastosis. Caso clínico: Paciente adulto de 35 años, blanco, sexo masculino, sospecha de insulinoma, con episodios de hipoglucemia en ayunas o tras un ejercicio que revertía con la ingesta de alimentos o soluciones glucosadas. Se le realizó pancreatectomía de un 85 por ciento y en el estudio histológico se detectó una Nesidioblastosis. Conclusiones: Es infrecuente en el adulto, realizar su diagnóstico es difícil, se llega a la cirugía con el conocimiento de un estado hiperinsulínico endógeno, sin la certeza de su origen(AU)


ABSTRACT Introduction: Nesidioblastosis is a rare pancreatic condition that causes hypoglycemia due to endogenous hypersinsulinism in childhood. It is unusual in adults; only isolated cases have been published including its description. Objective: To characterize a case of hyperinsulinic hypoglycemia, in an adult patient with nesidioblastosis. Clinical case: A 35-year-old, white, male, adult patient with suspected insulinoma, with episodes of hypoglycemia in the fasting state or after exercise that was reversed with ingestion of food or glucose solutions. 85 percent pancreatectomy was performed and nesidioblastosis was detected in the histological study. Conclusions: It is rare in adults, making its diagnosis is difficult, and surgery is reached with the knowledge of an endogenous hyperinsulinic state, without the certainty of its origin(AU)


Asunto(s)
Humanos , Masculino , Adulto , Pancreatectomía/métodos , Nesidioblastosis/diagnóstico , Hipoglucemia/diagnóstico por imagen , Insulinoma/terapia
13.
J BUON ; 25(3): 1302-1314, 2020.
Artículo en Inglés | MEDLINE | ID: mdl-32862570

RESUMEN

Insulinoma is the most common pancreatic neuroendocrine tumor (NET). Insulinomas are most commonly benign, well-differentiated NETs, whereas malignant neoplasms account for approximately 5-10% of all cases. Management includes conservative treatment with drugs targeting insulin-induced hypoglycemia, non-operative invasive procedures, as well as curative open or laparoscopic tumor resection. The current review aimed to summarize the current literature evidence on insulinoma and investigate the advantages and complications of available treatments.


Asunto(s)
Insulinoma/diagnóstico , Insulinoma/terapia , Humanos , Laparoscopía/métodos , Neoplasias Pancreáticas/diagnóstico , Neoplasias Pancreáticas/terapia
15.
Am J Case Rep ; 21: e923356, 2020 Jun 14.
Artículo en Inglés | MEDLINE | ID: mdl-32535614

RESUMEN

BACKGROUND Hypoglycemia is rare in individuals without drug-treated diabetes mellitus. In a seemingly well individual, the differential diagnosis of hypoglycemia narrows to 2 major categories: 1) accidental, surreptitious, or intentional hypoglycemia, or 2) endogenous hyperinsulinism (EHH). Insulinomas are the most common cause of EHH. Localization of insulinomas can be challenging, as most tumors are less than 2 cm in size and may be present in any part of the pancreas. In fact, almost 30% of neuroendocrine tumors (NET) cannot be located preoperatively by traditional imaging techniques such as computerized tomography (CT) or magnetic resonance imaging (MRI). CASE REPORT This report describes a case of metastatic insulinoma in a patient with a complex medical history. CT with contrast of the abdomen identified 1 lesion located in the pancreas body. Endoscopic ultrasound (EUS) identified an additional 3 to 4 hypoechoic lesions in the pancreatic neck and body. 68-Gallium Dotatate scanning identified 3 distinct lesions within the pancreas and a right posterior rib sclerotic lesion. CONCLUSIONS Reliance upon traditional imaging techniques (CT/MRI) for tumor localization would not have identified the multifocal pancreatic lesions and the metastatic bone lesion. Accurate identification of multifocal, metastatic insulinomas requires multiple imaging modalities, including first-line non-invasive imaging (CT or MRI) followed by second-line imaging (EUS or nuclear imaging).


Asunto(s)
Diazóxido/uso terapéutico , Hipoglucemia/tratamiento farmacológico , Insulinoma/diagnóstico por imagen , Insulinoma/terapia , Neoplasias Pancreáticas/diagnóstico por imagen , Neoplasias Pancreáticas/terapia , Técnicas de Ablación , Neoplasias Óseas/secundario , Diagnóstico Diferencial , Endosonografía , Femenino , Humanos , Hallazgos Incidentales , Insulinoma/patología , Persona de Mediana Edad , Neoplasias Pancreáticas/patología , Costillas/patología , Tomografía Computarizada por Rayos X
16.
Eur J Endocrinol ; 182(4): 439-446, 2020 Apr.
Artículo en Inglés | MEDLINE | ID: mdl-32061159

RESUMEN

INTRODUCTION: Management of malignant insulinomas is challenging due to the need to control both hypoglycaemic syndrome and tumor growth. Literature data is limited to small series. AIM OF THE STUDY: To analyze clinico-pathological characteristics, treatments and prognosis of patients with malignant insulinoma. MATERIALS AND METHODS: Multicenter retrospective study on 31 patients (male: 61.3%) diagnosed between 1988 and 2017. RESULTS: The mean age at diagnosis was 48 years. The mean NET diameter was 41 ± 31 mm, and 70.8% of NETs were G2. Metastases were widespread in 38.7%, hepatic in 41.9% and only lymph nodal in 19.4%. In 16.1% of the cases, the hypoglycaemic syndrome occurred after 46 ± 35 months from the diagnosis of originally non-functioning NET, whereas in 83.9% of the cases it led to the diagnosis of NET, of which 42.3% with a mean diagnostic delay of 32.7 ± 39.8 months. Surgical treatment was performed in 67.7% of the cases. The 5-year survival rate was 62%. Overall survival was significantly higher in patients with Ki-67 ≤10% (P = 0.03), insulin level <60 µU/mL (P = 0.015) and in patients who underwent surgery (P = 0.006). Peptide Receptor Radionuclide Therapy (PRRT) was performed in 45.1%, with syndrome control in 93% of patients. CONCLUSIONS: Our study includes the largest series of patients with malignant insulinoma reported to date. The hypoglycaemic syndrome may occur after years in initially non-functioning NETs or be misunderstood with delayed diagnosis of NETs. Surgical treatment and Ki67 ≤10% are prognostic factors associated with better survival. PPRT proved to be effective in the control of hypoglycaemia in majority of cases.


Asunto(s)
Insulinoma/mortalidad , Tumores Neuroendocrinos/mortalidad , Neoplasias Pancreáticas/mortalidad , Femenino , Humanos , Hipoglucemia/etiología , Hipoglucemia/mortalidad , Hipoglucemia/patología , Insulinoma/patología , Insulinoma/terapia , Masculino , Persona de Mediana Edad , Tumores Neuroendocrinos/patología , Tumores Neuroendocrinos/terapia , Neoplasias Pancreáticas/patología , Neoplasias Pancreáticas/terapia , Pronóstico , Estudios Retrospectivos , Tasa de Supervivencia
17.
J Endocrinol Invest ; 43(2): 139-148, 2020 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-31368049

RESUMEN

PURPOSE: Insulinomas are a rare type of neuroendocrine tumors, originating in the pancreas, difficult to diagnose and to treat. Due to its rarity, insulinomas are a not well-known pathological entity; thus, the diagnostic process is frequently a medical challenge with many possible differential diagnoses. The diagnostic process varies between non-invasive procedures, such as the fasting test or imaging techniques, and invasive ones. Insulinomas are rarely malignant, but the glycemic imbalance correlated with this tumor can frequently alter the quality of life of the patients and the consequent hypoglycemia can be extremely dangerous. Moreover, insulinomas can be associated with different genetic syndromes, such as Multiple Endocrine Neoplasia 1, accompanied by other specific symptoms. There are many different treatment strategies, depending on the need to control symptoms or control diseases progression, the only curative one being surgery. METHODS AND RESULTS: We reviewed the evidences present in the literature on insulinomas and reported its main clinical characteristics and management strategies. CONCLUSION: The aim of this review of the literature is to present the current knowledge on insulinomas, exploring the main clinical characteristics, the diagnostic tools, and the therapeutic strategies.


Asunto(s)
Insulinoma/metabolismo , Páncreas/fisiología , Neoplasias Pancreáticas/metabolismo , Animales , Humanos , Insulinoma/patología , Insulinoma/terapia , Páncreas/patología , Neoplasias Pancreáticas/patología , Neoplasias Pancreáticas/terapia , Receptores de Somatostatina/metabolismo
18.
J Labelled Comp Radiopharm ; 62(10): 656-672, 2019 08.
Artículo en Inglés | MEDLINE | ID: mdl-31070270

RESUMEN

Insulinomas, neuroendocrine tumors arising from pancreatic beta cells, often show overexpression of the glucagon-like peptide-1 receptor. Therefore, imaging with glucagon-like peptide analog exendin-4 can be used for diagnosis and preoperative localization. This review presents an overview of the development and clinical implementation of exendin-based tracers for nuclear imaging, and the potential use of exendin-4 based tracers for optical imaging and therapeutic applications such as peptide receptor radionuclide therapy or targeted photodynamic therapy.


Asunto(s)
Diagnóstico por Imagen/métodos , Exenatida/química , Exenatida/uso terapéutico , Insulinoma/diagnóstico por imagen , Insulinoma/terapia , Animales , Humanos , Insulinoma/tratamiento farmacológico , Fotoquimioterapia
20.
BMC Endocr Disord ; 18(1): 98, 2018 Dec 27.
Artículo en Inglés | MEDLINE | ID: mdl-30591061

RESUMEN

BACKGROUND: Insulinoma is a rare tumour representing 1-2% of all pancreatic neoplasms and it is malignant in only 10% of cases. Locoregional invasion or metastases define malignancy, whereas the dimension (> 2 cm), CK19 status, the tumor staging and grading (Ki67 > 2%), and the age of onset (> 50 years) can be considered elements of suspect. CASE PRESENTATION: We describe the case of a 68-year-old man presenting symptoms compatible with hypoglycemia. The symptoms regressed with food intake. These episodes initially occurred during physical activity, later also during fasting. The fasting test was performed and the laboratory results showed endogenous hyperinsulinemia compatible with insulinoma. The patient appeared responsive to somatostatin analogs and so he was treated with short acting octreotide, obtaining a good control of glycemia. Imaging investigations showed the presence of a lesion of the uncinate pancreatic process of about 4 cm with a high sst2 receptor density. The patient underwent exploratory laparotomy and duodenocephalopancreasectomy after one month. The definitive histological examination revealed an insulinoma (T3N1MO, AGCC VII G1) with a low replicative index (Ki67: 2%). CONCLUSIONS: This report describes a case of malignant insulinoma responsive to octreotide analogs administered pre-operatively in order to try to prevent hypoglycemia. The response to octreotide analogs is not predictable and should be initially assessed under strict clinical surveillance.


Asunto(s)
Insulinoma/terapia , Octreótido/administración & dosificación , Neoplasias Pancreáticas/terapia , Tejido Adiposo/patología , Anciano , Duodeno/fisiología , Duodeno/cirugía , Humanos , Hipoglucemia/prevención & control , Insulinoma/diagnóstico , Insulinoma/patología , Metástasis Linfática/patología , Masculino , Invasividad Neoplásica/patología , Pancreatectomía , Neoplasias Pancreáticas/diagnóstico , Neoplasias Pancreáticas/patología , Cuidados Preoperatorios , Somatostatina/análogos & derivados
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