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Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes

BACKGROUND/OBJECTIVE: Ectopic tumoral production of parathyroid hormone (PTH) is rare. The incidence of hyperparathyroidism and osteitis fibrosa cystica (OFC) secondary to ectopic PTH secretion has only been reported in case reports, although infrequent. CASE REPORT: We report a case of a well-diffe...

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Autores principales: Yazdian, Aaron, Minuk, Jeffrey, Hemenway, Gregory, Upadhyaya, Neelam, Vaz, Cherie Lisa
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Association of Clinical Endocrinology 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9508597/
https://www.ncbi.nlm.nih.gov/pubmed/36189140
http://dx.doi.org/10.1016/j.aace.2022.06.002
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author Yazdian, Aaron
Minuk, Jeffrey
Hemenway, Gregory
Upadhyaya, Neelam
Vaz, Cherie Lisa
author_facet Yazdian, Aaron
Minuk, Jeffrey
Hemenway, Gregory
Upadhyaya, Neelam
Vaz, Cherie Lisa
author_sort Yazdian, Aaron
collection PubMed
description BACKGROUND/OBJECTIVE: Ectopic tumoral production of parathyroid hormone (PTH) is rare. The incidence of hyperparathyroidism and osteitis fibrosa cystica (OFC) secondary to ectopic PTH secretion has only been reported in case reports, although infrequent. CASE REPORT: We report a case of a well-differentiated pulmonary neuroendocrine tumor (NET) producing PTH that presented with severe hypercalcemia and OFC. Surgical removal of the pulmonary tumor resulted in resolution of hypercalcemia. Immunocytochemical analysis of the tumor tissue revealed PTH-positive staining. Recovery was complicated by severe hypocalcemia due to hungry bone syndrome. DISCUSSION: To the best of our knowledge, this is the first documented case of a pulmonary NET causing OFC via PTH. We further describe the successful identification and resection of a rare NET and restoration of calcium homeostasis with aggressive calcium and vitamin D repletion. CONCLUSION: Although a rare cause of severe hypercalcemia and OFC, ectopic tumoral production of PTH must be considered in the differential diagnosis. Furthermore, resection of these tumors secreting PTH can lead to a protracted and severe high risk of hungry bone syndrome, which requires aggressive treatment to maintain calcium homeostasis.
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spelling pubmed-95085972022-09-30 Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes Yazdian, Aaron Minuk, Jeffrey Hemenway, Gregory Upadhyaya, Neelam Vaz, Cherie Lisa AACE Clin Case Rep Case Report BACKGROUND/OBJECTIVE: Ectopic tumoral production of parathyroid hormone (PTH) is rare. The incidence of hyperparathyroidism and osteitis fibrosa cystica (OFC) secondary to ectopic PTH secretion has only been reported in case reports, although infrequent. CASE REPORT: We report a case of a well-differentiated pulmonary neuroendocrine tumor (NET) producing PTH that presented with severe hypercalcemia and OFC. Surgical removal of the pulmonary tumor resulted in resolution of hypercalcemia. Immunocytochemical analysis of the tumor tissue revealed PTH-positive staining. Recovery was complicated by severe hypocalcemia due to hungry bone syndrome. DISCUSSION: To the best of our knowledge, this is the first documented case of a pulmonary NET causing OFC via PTH. We further describe the successful identification and resection of a rare NET and restoration of calcium homeostasis with aggressive calcium and vitamin D repletion. CONCLUSION: Although a rare cause of severe hypercalcemia and OFC, ectopic tumoral production of PTH must be considered in the differential diagnosis. Furthermore, resection of these tumors secreting PTH can lead to a protracted and severe high risk of hungry bone syndrome, which requires aggressive treatment to maintain calcium homeostasis. American Association of Clinical Endocrinology 2022-06-18 /pmc/articles/PMC9508597/ /pubmed/36189140 http://dx.doi.org/10.1016/j.aace.2022.06.002 Text en © 2022 AACE. Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Yazdian, Aaron
Minuk, Jeffrey
Hemenway, Gregory
Upadhyaya, Neelam
Vaz, Cherie Lisa
Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title_full Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title_fullStr Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title_full_unstemmed Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title_short Occult Pulmonary Neuroendocrine Tumor Causing Severe Hyperparathyroidism: Diagnostic Challenges and Clinical Outcomes
title_sort occult pulmonary neuroendocrine tumor causing severe hyperparathyroidism: diagnostic challenges and clinical outcomes
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9508597/
https://www.ncbi.nlm.nih.gov/pubmed/36189140
http://dx.doi.org/10.1016/j.aace.2022.06.002
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