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SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia

Disclosure: K. Jones: None. P. Krutilova: None. J.B. McGill: None. Introduction: Humoral hypercalcemia of malignancy is known to be associated with squamous cell carcinomas (SCC), although rarely seen in the setting of skin cancer. We present an interesting case of a young patient with giant condylo...

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Autores principales: Jones, Kai, Krutilova, Petra, McGill, Janet B
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10555027/
http://dx.doi.org/10.1210/jendso/bvad114.552
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author Jones, Kai
Krutilova, Petra
McGill, Janet B
author_facet Jones, Kai
Krutilova, Petra
McGill, Janet B
author_sort Jones, Kai
collection PubMed
description Disclosure: K. Jones: None. P. Krutilova: None. J.B. McGill: None. Introduction: Humoral hypercalcemia of malignancy is known to be associated with squamous cell carcinomas (SCC), although rarely seen in the setting of skin cancer. We present an interesting case of a young patient with giant condyloma acuminatum complicated by SCC presenting with severe hypercalcemia. Case Description: A 37-year-old man presented with pain and purulent drainage due to bacterial superinfection of genital warts. He had recurrent and locally advanced condylomas, first occurring more than 10 years ago. Skin lesions in the genital and perianal area never resolved and he required multiple surgical interventions including sigmoid colostomy placement due to Fournier gangrene. His laboratory evaluation this admission was notable for an elevated calcium level (13.0 mg/dL [8.5-10.3], albumin 3.4 g/dL [3.5-5.0]), phosphorus 3.9 mg/dL [2.3-4.5], and normal kidney function. He did not have history of hypercalcemia and was not taking any calcium supplements. Hypercalcemia prompted work-up including: PTH 4 pg/mL [15-65], 25-OH vitamin D 36 ng/mL [30-80], unremarkable SPEP/UPEP, TSH 0.91 mcIUnit/mL [0.3-4.2]. Initial differential diagnosis included hypercalcemia due to granulomatous process and increased production of calcitriol vs. PTH-related protein (PTHrP) mediated hypercalcemia. 1,25-OH vitamin D was normal (33 pg/mL [18-64]) and PTHrP was elevated (6.7 pmol/L [<4.2]). The patient subsequently underwent biopsy of groin lesions which demonstrated SCC in situ. PET/CT only demonstrated hypermetabolic changes in the perineum and groins bilaterally. Inguinal lymph node biopsy was negative for malignancy. His hypercalcemia persisted despite administration of IV fluids and he was treated with IV zolendronic acid. Calcium level subsequently normalized (9.1 mg/dL). He underwent extensive excision of the tumor involving the right gracilis muscle, left testicle, portions of the perineum, and penile shaft skin. Discussion: While cases of hypercalcemia in malignancy are common, this is an unusual case of giant condyloma acuminata (GCA) heralding development of SCC by elevated calcium levels. GCA also known as Buschke-Loewenstein tumor is a rare disease with estimated incidence of 0.1% in the general population and a male-to-female ratio of 2.7:1, typically associated with human papilloma virus subtypes 6 and 11. It is a locally aggressive tumor with a potential for destructive growth and malignant transformation (estimated rate up to 56%), very high rate of recurrence after excision (66%), and overall mortality of approximately 20%. Early recognition and treatment of GCA is essential in order to prevent devastating complications. Purzycka-Bohdan D, Nowicki RJ, Herms F, et al. The Pathogenesis of Giant Condyloma Acuminatum (Buschke-Lowenstein Tumor): An Overview. Int J Mol Sci. 2022;23(9):4547. Published 2022 Apr 20. doi:10.3390/ijms23094547 Presentation: Saturday, June 17, 2023
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spelling pubmed-105550272023-10-06 SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia Jones, Kai Krutilova, Petra McGill, Janet B J Endocr Soc Bone And Mineral Metabolism Disclosure: K. Jones: None. P. Krutilova: None. J.B. McGill: None. Introduction: Humoral hypercalcemia of malignancy is known to be associated with squamous cell carcinomas (SCC), although rarely seen in the setting of skin cancer. We present an interesting case of a young patient with giant condyloma acuminatum complicated by SCC presenting with severe hypercalcemia. Case Description: A 37-year-old man presented with pain and purulent drainage due to bacterial superinfection of genital warts. He had recurrent and locally advanced condylomas, first occurring more than 10 years ago. Skin lesions in the genital and perianal area never resolved and he required multiple surgical interventions including sigmoid colostomy placement due to Fournier gangrene. His laboratory evaluation this admission was notable for an elevated calcium level (13.0 mg/dL [8.5-10.3], albumin 3.4 g/dL [3.5-5.0]), phosphorus 3.9 mg/dL [2.3-4.5], and normal kidney function. He did not have history of hypercalcemia and was not taking any calcium supplements. Hypercalcemia prompted work-up including: PTH 4 pg/mL [15-65], 25-OH vitamin D 36 ng/mL [30-80], unremarkable SPEP/UPEP, TSH 0.91 mcIUnit/mL [0.3-4.2]. Initial differential diagnosis included hypercalcemia due to granulomatous process and increased production of calcitriol vs. PTH-related protein (PTHrP) mediated hypercalcemia. 1,25-OH vitamin D was normal (33 pg/mL [18-64]) and PTHrP was elevated (6.7 pmol/L [<4.2]). The patient subsequently underwent biopsy of groin lesions which demonstrated SCC in situ. PET/CT only demonstrated hypermetabolic changes in the perineum and groins bilaterally. Inguinal lymph node biopsy was negative for malignancy. His hypercalcemia persisted despite administration of IV fluids and he was treated with IV zolendronic acid. Calcium level subsequently normalized (9.1 mg/dL). He underwent extensive excision of the tumor involving the right gracilis muscle, left testicle, portions of the perineum, and penile shaft skin. Discussion: While cases of hypercalcemia in malignancy are common, this is an unusual case of giant condyloma acuminata (GCA) heralding development of SCC by elevated calcium levels. GCA also known as Buschke-Loewenstein tumor is a rare disease with estimated incidence of 0.1% in the general population and a male-to-female ratio of 2.7:1, typically associated with human papilloma virus subtypes 6 and 11. It is a locally aggressive tumor with a potential for destructive growth and malignant transformation (estimated rate up to 56%), very high rate of recurrence after excision (66%), and overall mortality of approximately 20%. Early recognition and treatment of GCA is essential in order to prevent devastating complications. Purzycka-Bohdan D, Nowicki RJ, Herms F, et al. The Pathogenesis of Giant Condyloma Acuminatum (Buschke-Lowenstein Tumor): An Overview. Int J Mol Sci. 2022;23(9):4547. Published 2022 Apr 20. doi:10.3390/ijms23094547 Presentation: Saturday, June 17, 2023 Oxford University Press 2023-10-05 /pmc/articles/PMC10555027/ http://dx.doi.org/10.1210/jendso/bvad114.552 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
Jones, Kai
Krutilova, Petra
McGill, Janet B
SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title_full SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title_fullStr SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title_full_unstemmed SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title_short SAT256 Buschke Loewenstein Tumor Presenting With Hypercalcemia
title_sort sat256 buschke loewenstein tumor presenting with hypercalcemia
topic Bone And Mineral Metabolism
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10555027/
http://dx.doi.org/10.1210/jendso/bvad114.552
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