Cargando…

SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa

Disclosure: L.A. Robles: None. T. Ayub: None. I. Haque: None. C.E. Guillen Lopez, MD: None. S. Guntupalli: None. Introduction: Hypercalcemia of malignancy (HCM) associated with squamous cell carcinoma (SCC) of cutaneous origin is rare, with an estimated prevalence of 4.6%. We present a case of paran...

Descripción completa

Detalles Bibliográficos
Autores principales: Robles, Lydia A, Ayub, Tooba, Haque, Ikram, Guillen Lopez, Claudia E, Guntupalli, Saroja
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10553914/
http://dx.doi.org/10.1210/jendso/bvad114.555
_version_ 1785116287462014976
author Robles, Lydia A
Ayub, Tooba
Haque, Ikram
Guillen Lopez, Claudia E
Guntupalli, Saroja
author_facet Robles, Lydia A
Ayub, Tooba
Haque, Ikram
Guillen Lopez, Claudia E
Guntupalli, Saroja
author_sort Robles, Lydia A
collection PubMed
description Disclosure: L.A. Robles: None. T. Ayub: None. I. Haque: None. C.E. Guillen Lopez, MD: None. S. Guntupalli: None. Introduction: Hypercalcemia of malignancy (HCM) associated with squamous cell carcinoma (SCC) of cutaneous origin is rare, with an estimated prevalence of 4.6%. We present a case of paraneoplastic hypercalcemia from PTHrP production by SCC arising in the setting of long-standing hidradenitis suppurativa (HS). Clinical Case: A 48-year-old male with a 20-year history of severe HS presents with concerns of disease deterioration and HS flare of buttocks, as well as, constipation, weakness, anorexia, and weight loss. Patient had recently discontinued treatment with Adalimumab for three weeks due to Covid-19 infection. Physical examination was significant for extensive hidradenitis to bilateral buttocks with granulation tissue at the base, surrounding induration, and scarring. Laboratory significant for corrected calcium 14.6 mg/dL (8.6-10.2 mg/dL), albumin 3.2, PTH 5 pg/mL(15-65 pg/mL), phosphorus 1.7mg/dL, ionized calcium 1.93 mm/L (1.15-1.30 mm/L), Vitamin D 25 mg/mL, 1,25 Vitamin D 14 pg/mL. Imaging with CT pelvis with innumerable peripherally enhancing centrally hypoattenuating lesions involving bilateral gluteus. Initially, calcium responded temporarily to IV hydration, calcitonin, and Pamidronate. Further work-up revealed PTHrP 24 pg/mL (11-20 pg/mL) and surgical pathology of bilateral buttocks with fragments of moderately to poorly differentiated SCC with perineural invasion of small caliber nerves and metastasis to lymph nodes. Patient remained significantly hypercalcemic during hospital course and received four doses of Pamidronate and eight doses of calcitonin. Diagnosis of bisphosphonate refractory hypercalcemia was established. Denosumab was given after patient became lethargic, and improvement of hypercalcemia followed. Oncology and Radiation therapy stated further treatment was not feasible given the lack of curative options and poor performance status, patient and family opted to pursue comfort care. Conclusion: Hypercalcemia presents in up to 30% of patients with malignancies and tends to predict a poor prognosis. Many mechanisms lead to HCM, with PTHrP production being the most common. Despite the prevalence of HCM, the risk of malignant transformation of HS to SCC is rare, with limited cases reported to date. Additionally, limited literature demonstrates mortality rates greater than 40% and involvement of buttocks, especially in men, exhibits the most malignant potential. Therefore, if a diagnosis of SCC in HS is made, an aggressive approach to management is advised to prevent significant morbidity and mortality. Bisphosphonates are the first-line therapy of HCM, but recent guidelines recommend treating severe HCM with a combination of calcitonin with either bisphosphonate or denosumab as the initial management. Furthermore, in those with bisphosphonate refractory HCM, treatment with denosumab would be the next step, as presented in our case. Presentation: Saturday, June 17, 2023
format Online
Article
Text
id pubmed-10553914
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher Oxford University Press
record_format MEDLINE/PubMed
spelling pubmed-105539142023-10-06 SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa Robles, Lydia A Ayub, Tooba Haque, Ikram Guillen Lopez, Claudia E Guntupalli, Saroja J Endocr Soc Bone And Mineral Metabolism Disclosure: L.A. Robles: None. T. Ayub: None. I. Haque: None. C.E. Guillen Lopez, MD: None. S. Guntupalli: None. Introduction: Hypercalcemia of malignancy (HCM) associated with squamous cell carcinoma (SCC) of cutaneous origin is rare, with an estimated prevalence of 4.6%. We present a case of paraneoplastic hypercalcemia from PTHrP production by SCC arising in the setting of long-standing hidradenitis suppurativa (HS). Clinical Case: A 48-year-old male with a 20-year history of severe HS presents with concerns of disease deterioration and HS flare of buttocks, as well as, constipation, weakness, anorexia, and weight loss. Patient had recently discontinued treatment with Adalimumab for three weeks due to Covid-19 infection. Physical examination was significant for extensive hidradenitis to bilateral buttocks with granulation tissue at the base, surrounding induration, and scarring. Laboratory significant for corrected calcium 14.6 mg/dL (8.6-10.2 mg/dL), albumin 3.2, PTH 5 pg/mL(15-65 pg/mL), phosphorus 1.7mg/dL, ionized calcium 1.93 mm/L (1.15-1.30 mm/L), Vitamin D 25 mg/mL, 1,25 Vitamin D 14 pg/mL. Imaging with CT pelvis with innumerable peripherally enhancing centrally hypoattenuating lesions involving bilateral gluteus. Initially, calcium responded temporarily to IV hydration, calcitonin, and Pamidronate. Further work-up revealed PTHrP 24 pg/mL (11-20 pg/mL) and surgical pathology of bilateral buttocks with fragments of moderately to poorly differentiated SCC with perineural invasion of small caliber nerves and metastasis to lymph nodes. Patient remained significantly hypercalcemic during hospital course and received four doses of Pamidronate and eight doses of calcitonin. Diagnosis of bisphosphonate refractory hypercalcemia was established. Denosumab was given after patient became lethargic, and improvement of hypercalcemia followed. Oncology and Radiation therapy stated further treatment was not feasible given the lack of curative options and poor performance status, patient and family opted to pursue comfort care. Conclusion: Hypercalcemia presents in up to 30% of patients with malignancies and tends to predict a poor prognosis. Many mechanisms lead to HCM, with PTHrP production being the most common. Despite the prevalence of HCM, the risk of malignant transformation of HS to SCC is rare, with limited cases reported to date. Additionally, limited literature demonstrates mortality rates greater than 40% and involvement of buttocks, especially in men, exhibits the most malignant potential. Therefore, if a diagnosis of SCC in HS is made, an aggressive approach to management is advised to prevent significant morbidity and mortality. Bisphosphonates are the first-line therapy of HCM, but recent guidelines recommend treating severe HCM with a combination of calcitonin with either bisphosphonate or denosumab as the initial management. Furthermore, in those with bisphosphonate refractory HCM, treatment with denosumab would be the next step, as presented in our case. Presentation: Saturday, June 17, 2023 Oxford University Press 2023-10-05 /pmc/articles/PMC10553914/ http://dx.doi.org/10.1210/jendso/bvad114.555 Text en © The Author(s) 2023. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (https://creativecommons.org/licenses/by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Bone And Mineral Metabolism
Robles, Lydia A
Ayub, Tooba
Haque, Ikram
Guillen Lopez, Claudia E
Guntupalli, Saroja
SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title_full SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title_fullStr SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title_full_unstemmed SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title_short SAT259 A Rare Case Of Bisphosphonate Refractory Hypercalcemia; Cancer Masquerading As Hidradenitis Suppurativa
title_sort sat259 a rare case of bisphosphonate refractory hypercalcemia; cancer masquerading as hidradenitis suppurativa
topic Bone And Mineral Metabolism
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10553914/
http://dx.doi.org/10.1210/jendso/bvad114.555
work_keys_str_mv AT robleslydiaa sat259ararecaseofbisphosphonaterefractoryhypercalcemiacancermasqueradingashidradenitissuppurativa
AT ayubtooba sat259ararecaseofbisphosphonaterefractoryhypercalcemiacancermasqueradingashidradenitissuppurativa
AT haqueikram sat259ararecaseofbisphosphonaterefractoryhypercalcemiacancermasqueradingashidradenitissuppurativa
AT guillenlopezclaudiae sat259ararecaseofbisphosphonaterefractoryhypercalcemiacancermasqueradingashidradenitissuppurativa
AT guntupallisaroja sat259ararecaseofbisphosphonaterefractoryhypercalcemiacancermasqueradingashidradenitissuppurativa