Cargando…

Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype

Parathyroid adenomas weighing more than 3.5 g are reported variously as “atypical”, “large” or “giant” parathyroid adenomas. All such adenomas are rare variants accounting for no more than 1.5% of all parathyroid adenomas. Large parathyroid adenomas are often associated with more severe form of the...

Descripción completa

Detalles Bibliográficos
Autores principales: Bhan, Arti, Athimulam, Shobana, Kumari, Poonam, Pal, Rimesh, Bhadada, Sanjay Kumar, Cook, Bernard C., Qiu, Shijing, Rao, Sudhaker D.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9931720/
https://www.ncbi.nlm.nih.gov/pubmed/36817587
http://dx.doi.org/10.3389/fendo.2023.1009516
_version_ 1784889293892747264
author Bhan, Arti
Athimulam, Shobana
Kumari, Poonam
Pal, Rimesh
Bhadada, Sanjay Kumar
Cook, Bernard C.
Qiu, Shijing
Rao, Sudhaker D.
author_facet Bhan, Arti
Athimulam, Shobana
Kumari, Poonam
Pal, Rimesh
Bhadada, Sanjay Kumar
Cook, Bernard C.
Qiu, Shijing
Rao, Sudhaker D.
author_sort Bhan, Arti
collection PubMed
description Parathyroid adenomas weighing more than 3.5 g are reported variously as “atypical”, “large” or “giant” parathyroid adenomas. All such adenomas are rare variants accounting for no more than 1.5% of all parathyroid adenomas. Large parathyroid adenomas are often associated with more severe form of the disease, including osteitis fibrosa cystica (OFC) and share many biochemical, histological, and molecular features of both benign and malignant parathyroid neoplasms, and are considered a distinct clinical entity. However, the pathogenesis of oversized parathyroid adenomas and the often-associated skeletal phenotype remains unclear. We present 5 cases of primary hyperparathyroidism (PHPT) with OFC, an uncommon manifestation of contemporary PHPT, associated with larger parathyroid adenomas, seen in the Bone and Mineral Disorders Clinic of the Henry Ford Health in the last 30 years to illustrate the critical role of vitamin D nutrition in the pathogenesis of both the OFC and adenoma size. The estimated prevalence of OFC was very low 0.2%, 5 of the >3000 surgically confirmed cases of PHPT seen during this time. The mean ± SD values were: age: 36.8 ± 22.1 years (4 of the 5 <36years), serum calcium 11.6 ± 1.1 mg/dl, alkaline phosphatase 799 ± 487 IU/L, PTH 1440 ± 477 pg/ml, 25-hydroxyvitamin D 13.0 ± 8.9 ng/ml, 1,25-dihyroxyvitamin D 26.5 ± 13.7 pg/ml, urine calcium 562 ± 274 mg/day, and parathyroid adenoma weight 4.53 ± 2.2 g. Parathyroidectomy led to the resolution of both the biochemical indices and OFC in each patient without recurrence over >10 years of follow-up. Because OFC is a very rare in the West, but very common areas of endemic vitamin D deficiency, we also examined the relationship between vitamin D nutrition, as assessed by serum 25-hydroxyvitamin D level, and parathyroid adenoma weight as well as prevalence of OFC in two large secularly diverse cohorts of patients with PHPT (Detroit, USA and Chandigarh, India). Based on this relationship and the relative prevalence of OFC in these two large cohorts, we propose that vitamin D nutrition (and perhaps calcium nutrition) best explains both the adenoma size and prevalence of OFC.
format Online
Article
Text
id pubmed-9931720
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-99317202023-02-17 Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype Bhan, Arti Athimulam, Shobana Kumari, Poonam Pal, Rimesh Bhadada, Sanjay Kumar Cook, Bernard C. Qiu, Shijing Rao, Sudhaker D. Front Endocrinol (Lausanne) Endocrinology Parathyroid adenomas weighing more than 3.5 g are reported variously as “atypical”, “large” or “giant” parathyroid adenomas. All such adenomas are rare variants accounting for no more than 1.5% of all parathyroid adenomas. Large parathyroid adenomas are often associated with more severe form of the disease, including osteitis fibrosa cystica (OFC) and share many biochemical, histological, and molecular features of both benign and malignant parathyroid neoplasms, and are considered a distinct clinical entity. However, the pathogenesis of oversized parathyroid adenomas and the often-associated skeletal phenotype remains unclear. We present 5 cases of primary hyperparathyroidism (PHPT) with OFC, an uncommon manifestation of contemporary PHPT, associated with larger parathyroid adenomas, seen in the Bone and Mineral Disorders Clinic of the Henry Ford Health in the last 30 years to illustrate the critical role of vitamin D nutrition in the pathogenesis of both the OFC and adenoma size. The estimated prevalence of OFC was very low 0.2%, 5 of the >3000 surgically confirmed cases of PHPT seen during this time. The mean ± SD values were: age: 36.8 ± 22.1 years (4 of the 5 <36years), serum calcium 11.6 ± 1.1 mg/dl, alkaline phosphatase 799 ± 487 IU/L, PTH 1440 ± 477 pg/ml, 25-hydroxyvitamin D 13.0 ± 8.9 ng/ml, 1,25-dihyroxyvitamin D 26.5 ± 13.7 pg/ml, urine calcium 562 ± 274 mg/day, and parathyroid adenoma weight 4.53 ± 2.2 g. Parathyroidectomy led to the resolution of both the biochemical indices and OFC in each patient without recurrence over >10 years of follow-up. Because OFC is a very rare in the West, but very common areas of endemic vitamin D deficiency, we also examined the relationship between vitamin D nutrition, as assessed by serum 25-hydroxyvitamin D level, and parathyroid adenoma weight as well as prevalence of OFC in two large secularly diverse cohorts of patients with PHPT (Detroit, USA and Chandigarh, India). Based on this relationship and the relative prevalence of OFC in these two large cohorts, we propose that vitamin D nutrition (and perhaps calcium nutrition) best explains both the adenoma size and prevalence of OFC. Frontiers Media S.A. 2023-02-02 /pmc/articles/PMC9931720/ /pubmed/36817587 http://dx.doi.org/10.3389/fendo.2023.1009516 Text en Copyright © 2023 Bhan, Athimulam, Kumari, Pal, Bhadada, Cook, Qiu and Rao https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Endocrinology
Bhan, Arti
Athimulam, Shobana
Kumari, Poonam
Pal, Rimesh
Bhadada, Sanjay Kumar
Cook, Bernard C.
Qiu, Shijing
Rao, Sudhaker D.
Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title_full Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title_fullStr Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title_full_unstemmed Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title_short Large parathyroid adenomas: Potential mechanisms to reconcile adenoma size and disease phenotype
title_sort large parathyroid adenomas: potential mechanisms to reconcile adenoma size and disease phenotype
topic Endocrinology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9931720/
https://www.ncbi.nlm.nih.gov/pubmed/36817587
http://dx.doi.org/10.3389/fendo.2023.1009516
work_keys_str_mv AT bhanarti largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT athimulamshobana largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT kumaripoonam largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT palrimesh largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT bhadadasanjaykumar largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT cookbernardc largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT qiushijing largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype
AT raosudhakerd largeparathyroidadenomaspotentialmechanismstoreconcileadenomasizeanddiseasephenotype