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Artículo

In vitro osteoclastogenesis from Gaucher patients' cells correlates with bone mineral density but not with Chitotriosidase

Bondar, Constanza MaríaIcon ; Mucci, Juan MarcosIcon ; Crivaro, Andrea NataliaIcon ; Ormazabal, Maximiliano EmanuelIcon ; Ceci, Romina; Oliveri, María BeatrizIcon ; González, D.; Rozenfeld, Paula AdrianaIcon
Fecha de publicación: 10/2017
Editorial: Elsevier Science Inc
Revista: Bone
ISSN: 8756-3282
Idioma: Inglés
Tipo de recurso: Artículo publicado
Clasificación temática:
Otras Medicina Básica

Resumen

Gaucher disease (GD) is caused by mutations on the gene encoding for the lysosomal enzyme glucocerebrosidase. Type I GD (GD1) patients present anemia, hepatosplenomegaly and bone alterations. In spite of treatment, bone alterations in GD patients persist, including poor bone mineral density (BMD). Mechanisms leading to bone damage are not completely understood, but previous reports suggest that osteoclasts are involved. Chitotriosidase (CHIT) is the most reliable biomarker used in the follow up of patients, although its correlation with bone status is unknown. The aim of this work was to study the pro-osteoclastogenic potential in patients and to evaluate its correlation with CHIT activity levels and clinical parameters. PBMCs from treated patients and healthy controls were cultured in the presence of M-CSF, and mature osteoclasts were counted. BMD, blood CHIT activity and serum levels of CTX, BAP, and cytokines were evaluated in patients. We found that blood CHIT activity and osteoclast differentiation were significantly increased in patients, but no correlation between them was observed. Interestingly, osteoclast numbers but not CHIT, presented a negative correlation with BMD expressed as Z-score. CTX, BAP and serum cytokines involved in bone remodeling were found altered in GD1 patients. These results show for the first time a correlation between osteoclast differentiation and BMD in GD1 patients, supporting the involvement of osteoclasts in the bone pathology of GD1. Our results also suggest that an altered immune response may play an important role in bone damage.
Palabras clave: Bone Mineral Density , Bone Pathology , Chitotriosidase , Gaucher Disease , Osteoclasts
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info:eu-repo/semantics/openAccess Excepto donde se diga explícitamente, este item se publica bajo la siguiente descripción: Atribución-NoComercial-SinDerivadas 2.5 Argentina (CC BY-NC-ND 2.5 AR)
Identificadores
URI: http://hdl.handle.net/11336/47678
URL: https://www.sciencedirect.com/science/article/pii/S8756328217302466
DOI: https://dx.doi.org/10.1016/j.bone.2017.07.020
Colecciones
Articulos(INIGEM)
Articulos de INSTITUTO DE INMUNOLOGIA, GENETICA Y METABOLISMO
Articulos(IIFP)
Articulos de INST. DE ESTUDIOS INMUNOLOGICOS Y FISIOPATOLOGICOS
Citación
Bondar, Constanza María; Mucci, Juan Marcos; Crivaro, Andrea Natalia; Ormazabal, Maximiliano Emanuel; Ceci, Romina; et al.; In vitro osteoclastogenesis from Gaucher patients' cells correlates with bone mineral density but not with Chitotriosidase; Elsevier Science Inc; Bone; 103; 10-2017; 262-269
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