Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report
Abstract Background Hyperparathyroid crisis, or “parathyroid storm” is a rare manifestation of primary hyperparathyroidism, characterized by sudden onset of symptomatic, severe hypercalcemia (> 3.5 mmol/L). Hemorrhage into a parathyroid adenoma has rarely been reported as an inciting or associate...
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SAGE Publishing
2023-01-01
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| Series: | Journal of Otolaryngology - Head and Neck Surgery |
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| Online Access: | https://doi.org/10.1186/s40463-022-00600-x |
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| author | Gia Gill Veena Agrawal Paul Kerr |
| author_facet | Gia Gill Veena Agrawal Paul Kerr |
| author_sort | Gia Gill |
| collection | DOAJ |
| description | Abstract Background Hyperparathyroid crisis, or “parathyroid storm” is a rare manifestation of primary hyperparathyroidism, characterized by sudden onset of symptomatic, severe hypercalcemia (> 3.5 mmol/L). Hemorrhage into a parathyroid adenoma has rarely been reported as an inciting or associated event. We present a case of hemorrhage into a longstanding adenoma presenting with acute onset of profound hypercalcemia and associated complications. Case presentation A 60-year-old male presented to hospital with sudden onset of confusion, muscle weakness, and ataxia. Initial labs showed serum calcium 4.79 mmol/L, parathyroid hormone 2043 ng/L; creatinine 364 μmol/L. Review of the patient’s medical history indicated a 4-year history of recurrent nephrolithiasis, but no prior documented calcium levels. The hypercalcemia did not respond to 5 days of aggressive medical management with fluid resuscitation, denosumab and calcitonin, and later pamidronate and cinacalcet. He continued to deteriorate, requiring intubation and continuous renal replacement therapy. Imaging demonstrated 4.8 cm cystic right paratracheal mass; Technetium (Tc99m) Sestamibi scintigraphy was non-localizing. Urgent parathyroidectomy was completed, revealing a 5 × 3.3 × 1.8 cm hemorrhagic, atypical hypercellular parathyroid. Unfortunately, the patient died from complications from anticoagulation therapy for treatment of deep vein thrombosis 4 weeks after admission. His renal function had not recovered at the time of his death. Conclusion This case gives potential insight into the etiology of hyperparathyroid crisis, and the difficulty in achieving control of hypercalcemia with medical means. Surgical intervention is the definitive management in these cases and should be considered urgently. Graphical Abstract |
| format | Article |
| id | doaj-art-ce3f6b3e396b4844a1a11959281054da |
| institution | Kabale University |
| issn | 1916-0216 |
| language | English |
| publishDate | 2023-01-01 |
| publisher | SAGE Publishing |
| record_format | Article |
| series | Journal of Otolaryngology - Head and Neck Surgery |
| spelling | doaj-art-ce3f6b3e396b4844a1a11959281054da2025-02-02T23:08:47ZengSAGE PublishingJournal of Otolaryngology - Head and Neck Surgery1916-02162023-01-015211810.1186/s40463-022-00600-xPrimary hyperparathyroidism presenting as acute hypercalcemic crisis: a case reportGia Gill0Veena Agrawal1Paul Kerr2Department of Otolaryngology-Head and Neck Surgery, Max Rady College of Medicine, University of ManitobaSection of Endocrinology & Metabolism, Department of Internal Medicine, Max Rady College of Medicine, University of ManitobaDepartment of Otolaryngology-Head and Neck Surgery, Max Rady College of Medicine, University of ManitobaAbstract Background Hyperparathyroid crisis, or “parathyroid storm” is a rare manifestation of primary hyperparathyroidism, characterized by sudden onset of symptomatic, severe hypercalcemia (> 3.5 mmol/L). Hemorrhage into a parathyroid adenoma has rarely been reported as an inciting or associated event. We present a case of hemorrhage into a longstanding adenoma presenting with acute onset of profound hypercalcemia and associated complications. Case presentation A 60-year-old male presented to hospital with sudden onset of confusion, muscle weakness, and ataxia. Initial labs showed serum calcium 4.79 mmol/L, parathyroid hormone 2043 ng/L; creatinine 364 μmol/L. Review of the patient’s medical history indicated a 4-year history of recurrent nephrolithiasis, but no prior documented calcium levels. The hypercalcemia did not respond to 5 days of aggressive medical management with fluid resuscitation, denosumab and calcitonin, and later pamidronate and cinacalcet. He continued to deteriorate, requiring intubation and continuous renal replacement therapy. Imaging demonstrated 4.8 cm cystic right paratracheal mass; Technetium (Tc99m) Sestamibi scintigraphy was non-localizing. Urgent parathyroidectomy was completed, revealing a 5 × 3.3 × 1.8 cm hemorrhagic, atypical hypercellular parathyroid. Unfortunately, the patient died from complications from anticoagulation therapy for treatment of deep vein thrombosis 4 weeks after admission. His renal function had not recovered at the time of his death. Conclusion This case gives potential insight into the etiology of hyperparathyroid crisis, and the difficulty in achieving control of hypercalcemia with medical means. Surgical intervention is the definitive management in these cases and should be considered urgently. Graphical Abstracthttps://doi.org/10.1186/s40463-022-00600-xHyperparathyroidHypercalcemiaParathyroid adenomaAtypical adenoma |
| spellingShingle | Gia Gill Veena Agrawal Paul Kerr Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report Journal of Otolaryngology - Head and Neck Surgery Hyperparathyroid Hypercalcemia Parathyroid adenoma Atypical adenoma |
| title | Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report |
| title_full | Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report |
| title_fullStr | Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report |
| title_full_unstemmed | Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report |
| title_short | Primary hyperparathyroidism presenting as acute hypercalcemic crisis: a case report |
| title_sort | primary hyperparathyroidism presenting as acute hypercalcemic crisis a case report |
| topic | Hyperparathyroid Hypercalcemia Parathyroid adenoma Atypical adenoma |
| url | https://doi.org/10.1186/s40463-022-00600-x |
| work_keys_str_mv | AT giagill primaryhyperparathyroidismpresentingasacutehypercalcemiccrisisacasereport AT veenaagrawal primaryhyperparathyroidismpresentingasacutehypercalcemiccrisisacasereport AT paulkerr primaryhyperparathyroidismpresentingasacutehypercalcemiccrisisacasereport |