Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma

Autores
Kelly, Maury; Martín, María Julia; Figueroa, Verónica; Yogui, Analía; Gentili, Claudia Rosana; Brunetto, Oscar
Año de publicación
2019
Idioma
inglés
Tipo de recurso
documento de conferencia
Estado
versión publicada
Descripción
Epithelioid hemangioendothelioma (HEE) is a tumor of vascular origin, infrequent in the pediatric age and even more infrequent at intestinal level. To our knowledge, there are no previous reports of pediatric patients with malignant humoral hypercalcemia associated with this tumor. Humoral mechanism is seen more often in lung, uterine cérvix, skin and esophagus tumors. The presence of hypercalcemia appears to be an ominous prognostic sign.Objective: To report the first case of a patient with hypercalcemia related to PTH-rP associated with Colon HEE in a pediatric patient.Case description: A 3 months years old boy was admited because of clinical worsening and palpable abdominal mass. Initial laboratory investigation revealed hypercalcemia with the following workup: PTH: 1.65 pg/ml, calcium: 25.1 mg/dl, phosphorus 2.9 mg/dl 25ohvitamine D: 25.2 ng/ml, Urine catecholamines were normal. Ultrasound visualized a highly vascularized tumor with calcifications in retroperitoneum of 8×6×6 cm located between liver and right kidney. Biphosphonates and Calcitonin were iniciated without improvement. Biopsy reported epithelioid hemangioendothelioma and angiography revealed tumor irrigated by the middle colic artery. Selective embolization was performed with spongostan and 24 hours later, tumor exeresis was achieved. PTHrP mRNA was identified in the tumor. After surgery the patient attained normocalcemic state, PTH levels normalized and remained normocalcemic to date, 18 months later.Conclusion: We report the first case of PTH-rp related hypercalcemia, with mesocolon epithelioid hemangioendothelioma a pediatric patient. PTH-rp mRNA was detected at tumor level, and the patient resolved hypercalcemia with tumor resection, remaining normocalcemic and with normal PTH levels since then. Selective embolization was important in order to facilitate tumor resection successfully, and improving morbidity and mortality of this surgery.Disclosure: The authors declared no competing interests.
Fil: Kelly, Maury. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Martín, María Julia. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; Argentina
Fil: Figueroa, Verónica. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Yogui, Analía. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Gentili, Claudia Rosana. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; Argentina
Fil: Brunetto, Oscar. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
9th International Conference on Children´s Bone Health
Salzburg
Austria
9th International Conference on Children´s Bone Health
Materia
HYPERCALCEMIA
PARATHYROID HORMONE
PEDIATRIC PATIENT
COLON HEMANGIOENDOTHELIOMA
Nivel de accesibilidad
acceso abierto
Condiciones de uso
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
Repositorio
CONICET Digital (CONICET)
Institución
Consejo Nacional de Investigaciones Científicas y Técnicas
OAI Identificador
oai:ri.conicet.gov.ar:11336/216933
Acceder
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oai_identifier_str oai:ri.conicet.gov.ar:11336/216933
network_acronym_str CONICETDig
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network_name_str CONICET Digital (CONICET)
spelling Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon HemangioendotheliomaKelly, MauryMartín, María JuliaFigueroa, VerónicaYogui, AnalíaGentili, Claudia RosanaBrunetto, OscarHYPERCALCEMIAPARATHYROID HORMONEPEDIATRIC PATIENTCOLON HEMANGIOENDOTHELIOMAhttps://purl.org/becyt/ford/3.2https://purl.org/becyt/ford/3Epithelioid hemangioendothelioma (HEE) is a tumor of vascular origin, infrequent in the pediatric age and even more infrequent at intestinal level. To our knowledge, there are no previous reports of pediatric patients with malignant humoral hypercalcemia associated with this tumor. Humoral mechanism is seen more often in lung, uterine cérvix, skin and esophagus tumors. The presence of hypercalcemia appears to be an ominous prognostic sign.Objective: To report the first case of a patient with hypercalcemia related to PTH-rP associated with Colon HEE in a pediatric patient.Case description: A 3 months years old boy was admited because of clinical worsening and palpable abdominal mass. Initial laboratory investigation revealed hypercalcemia with the following workup: PTH: 1.65 pg/ml, calcium: 25.1 mg/dl, phosphorus 2.9 mg/dl 25ohvitamine D: 25.2 ng/ml, Urine catecholamines were normal. Ultrasound visualized a highly vascularized tumor with calcifications in retroperitoneum of 8×6×6 cm located between liver and right kidney. Biphosphonates and Calcitonin were iniciated without improvement. Biopsy reported epithelioid hemangioendothelioma and angiography revealed tumor irrigated by the middle colic artery. Selective embolization was performed with spongostan and 24 hours later, tumor exeresis was achieved. PTHrP mRNA was identified in the tumor. After surgery the patient attained normocalcemic state, PTH levels normalized and remained normocalcemic to date, 18 months later.Conclusion: We report the first case of PTH-rp related hypercalcemia, with mesocolon epithelioid hemangioendothelioma a pediatric patient. PTH-rp mRNA was detected at tumor level, and the patient resolved hypercalcemia with tumor resection, remaining normocalcemic and with normal PTH levels since then. Selective embolization was important in order to facilitate tumor resection successfully, and improving morbidity and mortality of this surgery.Disclosure: The authors declared no competing interests.Fil: Kelly, Maury. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); ArgentinaFil: Martín, María Julia. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; ArgentinaFil: Figueroa, Verónica. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); ArgentinaFil: Yogui, Analía. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); ArgentinaFil: Gentili, Claudia Rosana. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; ArgentinaFil: Brunetto, Oscar. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina9th International Conference on Children´s Bone HealthSalzburgAustria9th International Conference on Children´s Bone HealthBiocientifica2019info:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/conferenceObjectCongresoJournalhttp://purl.org/coar/resource_type/c_5794info:ar-repo/semantics/documentoDeConferenciaapplication/pdfapplication/pdfapplication/pdfhttp://hdl.handle.net/11336/216933Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma; 9th International Conference on Children´s Bone Health; Salzburg; Austria; 2019; 1-12052-1219CONICET DigitalCONICETenginfo:eu-repo/semantics/altIdentifier/url/www.bone-abstracts.orginfo:eu-repo/semantics/altIdentifier/url/https://www.bone-abstracts.org/ba/0007/ba0007p218info:eu-repo/semantics/altIdentifier/url/https://doi.org/10.1530/boneabs.7.P218Internacionalinfo:eu-repo/semantics/openAccesshttps://creativecommons.org/licenses/by-nc-sa/2.5/ar/reponame:CONICET Digital (CONICET)instname:Consejo Nacional de Investigaciones Científicas y Técnicas2025-10-15T14:22:02Zoai:ri.conicet.gov.ar:11336/216933instacron:CONICETInstitucionalhttp://ri.conicet.gov.ar/Organismo científico-tecnológicoNo correspondehttp://ri.conicet.gov.ar/oai/requestdasensio@conicet.gov.ar; lcarlino@conicet.gov.arArgentinaNo correspondeNo correspondeNo correspondeopendoar:34982025-10-15 14:22:03.289CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicasfalse
dc.title.none.fl_str_mv Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
title Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
spellingShingle Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
Kelly, Maury
HYPERCALCEMIA
PARATHYROID HORMONE
PEDIATRIC PATIENT
COLON HEMANGIOENDOTHELIOMA
title_short Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
title_full Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
title_fullStr Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
title_full_unstemmed Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
title_sort Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma
dc.creator.none.fl_str_mv Kelly, Maury
Martín, María Julia
Figueroa, Verónica
Yogui, Analía
Gentili, Claudia Rosana
Brunetto, Oscar
author Kelly, Maury
author_facet Kelly, Maury
Martín, María Julia
Figueroa, Verónica
Yogui, Analía
Gentili, Claudia Rosana
Brunetto, Oscar
author_role author
author2 Martín, María Julia
Figueroa, Verónica
Yogui, Analía
Gentili, Claudia Rosana
Brunetto, Oscar
author2_role author
author
author
author
author
dc.subject.none.fl_str_mv HYPERCALCEMIA
PARATHYROID HORMONE
PEDIATRIC PATIENT
COLON HEMANGIOENDOTHELIOMA
topic HYPERCALCEMIA
PARATHYROID HORMONE
PEDIATRIC PATIENT
COLON HEMANGIOENDOTHELIOMA
purl_subject.fl_str_mv https://purl.org/becyt/ford/3.2
https://purl.org/becyt/ford/3
dc.description.none.fl_txt_mv Epithelioid hemangioendothelioma (HEE) is a tumor of vascular origin, infrequent in the pediatric age and even more infrequent at intestinal level. To our knowledge, there are no previous reports of pediatric patients with malignant humoral hypercalcemia associated with this tumor. Humoral mechanism is seen more often in lung, uterine cérvix, skin and esophagus tumors. The presence of hypercalcemia appears to be an ominous prognostic sign.Objective: To report the first case of a patient with hypercalcemia related to PTH-rP associated with Colon HEE in a pediatric patient.Case description: A 3 months years old boy was admited because of clinical worsening and palpable abdominal mass. Initial laboratory investigation revealed hypercalcemia with the following workup: PTH: 1.65 pg/ml, calcium: 25.1 mg/dl, phosphorus 2.9 mg/dl 25ohvitamine D: 25.2 ng/ml, Urine catecholamines were normal. Ultrasound visualized a highly vascularized tumor with calcifications in retroperitoneum of 8×6×6 cm located between liver and right kidney. Biphosphonates and Calcitonin were iniciated without improvement. Biopsy reported epithelioid hemangioendothelioma and angiography revealed tumor irrigated by the middle colic artery. Selective embolization was performed with spongostan and 24 hours later, tumor exeresis was achieved. PTHrP mRNA was identified in the tumor. After surgery the patient attained normocalcemic state, PTH levels normalized and remained normocalcemic to date, 18 months later.Conclusion: We report the first case of PTH-rp related hypercalcemia, with mesocolon epithelioid hemangioendothelioma a pediatric patient. PTH-rp mRNA was detected at tumor level, and the patient resolved hypercalcemia with tumor resection, remaining normocalcemic and with normal PTH levels since then. Selective embolization was important in order to facilitate tumor resection successfully, and improving morbidity and mortality of this surgery.Disclosure: The authors declared no competing interests.
Fil: Kelly, Maury. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Martín, María Julia. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; Argentina
Fil: Figueroa, Verónica. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Yogui, Analía. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
Fil: Gentili, Claudia Rosana. Consejo Nacional de Investigaciones Científicas y Técnicas. Centro Científico Tecnológico Conicet - Bahía Blanca. Instituto de Ciencias Biológicas y Biomédicas del Sur. Universidad Nacional del Sur. Departamento de Biología, Bioquímica y Farmacia. Instituto de Ciencias Biológicas y Biomédicas del Sur; Argentina
Fil: Brunetto, Oscar. Gobierno de la Ciudad de Buenos Aires. Hospital General de Niños Pedro Elizalde (ex Casa Cuna); Argentina
9th International Conference on Children´s Bone Health
Salzburg
Austria
9th International Conference on Children´s Bone Health
description Epithelioid hemangioendothelioma (HEE) is a tumor of vascular origin, infrequent in the pediatric age and even more infrequent at intestinal level. To our knowledge, there are no previous reports of pediatric patients with malignant humoral hypercalcemia associated with this tumor. Humoral mechanism is seen more often in lung, uterine cérvix, skin and esophagus tumors. The presence of hypercalcemia appears to be an ominous prognostic sign.Objective: To report the first case of a patient with hypercalcemia related to PTH-rP associated with Colon HEE in a pediatric patient.Case description: A 3 months years old boy was admited because of clinical worsening and palpable abdominal mass. Initial laboratory investigation revealed hypercalcemia with the following workup: PTH: 1.65 pg/ml, calcium: 25.1 mg/dl, phosphorus 2.9 mg/dl 25ohvitamine D: 25.2 ng/ml, Urine catecholamines were normal. Ultrasound visualized a highly vascularized tumor with calcifications in retroperitoneum of 8×6×6 cm located between liver and right kidney. Biphosphonates and Calcitonin were iniciated without improvement. Biopsy reported epithelioid hemangioendothelioma and angiography revealed tumor irrigated by the middle colic artery. Selective embolization was performed with spongostan and 24 hours later, tumor exeresis was achieved. PTHrP mRNA was identified in the tumor. After surgery the patient attained normocalcemic state, PTH levels normalized and remained normocalcemic to date, 18 months later.Conclusion: We report the first case of PTH-rp related hypercalcemia, with mesocolon epithelioid hemangioendothelioma a pediatric patient. PTH-rp mRNA was detected at tumor level, and the patient resolved hypercalcemia with tumor resection, remaining normocalcemic and with normal PTH levels since then. Selective embolization was important in order to facilitate tumor resection successfully, and improving morbidity and mortality of this surgery.Disclosure: The authors declared no competing interests.
publishDate 2019
dc.date.none.fl_str_mv 2019
dc.type.none.fl_str_mv info:eu-repo/semantics/publishedVersion
info:eu-repo/semantics/conferenceObject
Congreso
Journal
http://purl.org/coar/resource_type/c_5794
info:ar-repo/semantics/documentoDeConferencia
status_str publishedVersion
format conferenceObject
dc.identifier.none.fl_str_mv http://hdl.handle.net/11336/216933
Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma; 9th International Conference on Children´s Bone Health; Salzburg; Austria; 2019; 1-1
2052-1219
CONICET Digital
CONICET
url http://hdl.handle.net/11336/216933
identifier_str_mv Hypercalcemia and parathyroid hormone-related peptide expression in a 3 months old boy with Colon Hemangioendothelioma; 9th International Conference on Children´s Bone Health; Salzburg; Austria; 2019; 1-1
2052-1219
CONICET Digital
CONICET
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language eng
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info:eu-repo/semantics/altIdentifier/url/https://www.bone-abstracts.org/ba/0007/ba0007p218
info:eu-repo/semantics/altIdentifier/url/https://doi.org/10.1530/boneabs.7.P218
dc.rights.none.fl_str_mv info:eu-repo/semantics/openAccess
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
eu_rights_str_mv openAccess
rights_invalid_str_mv https://creativecommons.org/licenses/by-nc-sa/2.5/ar/
dc.format.none.fl_str_mv application/pdf
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dc.coverage.none.fl_str_mv Internacional
dc.publisher.none.fl_str_mv Biocientifica
publisher.none.fl_str_mv Biocientifica
dc.source.none.fl_str_mv reponame:CONICET Digital (CONICET)
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reponame_str CONICET Digital (CONICET)
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repository.name.fl_str_mv CONICET Digital (CONICET) - Consejo Nacional de Investigaciones Científicas y Técnicas
repository.mail.fl_str_mv dasensio@conicet.gov.ar; lcarlino@conicet.gov.ar
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