Hypercalcemic crisis, unusual presentation of parathyroid adenoma
Main Article Content
Abstract
Hypercalcemic crisis (HC) is an unusual endocrine emergency, de!ned as the presence of serum calcium > 14 mg/dl related to kidney dysfunction, cardiovascular, gastrointestinal and sensory disturbances. It could also be considered in patients with severe symptoms and lower serum calcium levels.
Primary hyperparathyroidism (PHPT) and malignant neoplasms are the most common hypercalcemia etiologies (90% of cases), nevertheless, the former hardly ever occurs as HC. Due to the high mortality associated with
HC, it is crucial to establish early diagnosis and treatment.
We report two patients with HC as the first manifestation of PHPT; the former with atrioventricular (AV) block and the latter with acute pancreatitis. Pathology revealed oxyphilic adenoma in both cases, which is an infrequent histological variant that can have a severe clinical manifestation.
Conclusions: parathyroid adenomas are a rare cause of HC. We consider the histological type observed (oxyphilic adenoma) as a probable conditioning factor. Pancreatitis and especially AV block are rare manifestations of HC. We emphasize the importance of determining
calcium levels in the initial evaluation of all patients with AV block.
Article Details
Derechos de autor: Actualizaciones en Osteología es la revista oficial de la Asociación Argentina de Osteología y Metabolismo Mineral (AAOMM) que posee los derechos de autor de todo el material publicado en dicha revista.
References
Ahmad S, Kuraganti G, Steenkamp D. Hypercalcemic crisis: a clinical review. Am J Med 2015;128:239-45.
Cannon J, Lew JI, Solórzano CC. Parathyroidectomy for hypercalcemic crisis: 40 years’ experience and long-term outcomes. Surgery 2010;148:807-12.
Singh DN, Gupta SK, Kumari N, Krishnani N, Chand G, Mishra A, et al. Primary hyperparathyroidism presenting as hypercalcemic crisis: Twenty-year experience. Indian J Endocrinol Metab 2015;19:100-5.
James PR, Richards PG. Parathyroid crisis; treatment by emergency parathyroidectomy. AMA Arch Surg 1956;72:553-6.
Gücek Haciyanli S, Acar N, Gür EÖ, et al. Severe hypercalcaemia of primary hyperparathyroidism: Could giant adenoma be the real culprit rather than carcinoma? Ann R Coll Surg Engl 2020;102:363-8.
Phitayakorn R, McHenry CR. Hyperparathyroid Crisis: use of bisphosphonates as a bridge to parathyroidectomy. J Am Coll Surg 2008;206:1106-15.
Starker LF, Björklund P, Theoharis C, Long WD 3rd, Carling T, Udelsman R. Clinical and histopathological characteristics of hyperparathyroidism- induced hypercalcemic crisis. World J Surg 2011;35:331-5.
Betea D, Potorac I, Beckers A. Parathyroid carcinoma: Challenges in diagnosis and treatment. Ann Endocrinol (Paris) 2015;76:169-77.
Hundahl SA, Fleming ID, Fremgen AM, Menck HR. Two hundred eighty-six cases of parathyroid carcinoma treated in the U.S. between 1985-1995: a National Cancer Data Base Report. The American College of Surgeons Commission on Cancer and the American Cancer Society. Cancer 1999;86:538-44.
Talat N, Schulte KM. Clinical presentation, staging and long-term evolution of parathyroid cancer. Ann Surg Oncol. 2010; 17:2156-74.
Robert JH, Trombetti A, Garcia A, et al. Primary hyperparathyroidism: can parathyroid carcinoma be anticipated on clinical and biochemical grounds? Report of nine cases and review of the literature. Ann Surg Oncol 2005;12:526-32.
Fleischer J, Becker C, Hamele-Bena D, Breen TL, Silverberg SJ. Oxyphil parathyroid adenoma: a malignant presentation of a benign disease. J Clin Endocrinol Metab 2004; 89:5948-51.
Hoff H, Smith P, Winkler A. Electrocardiographic changes and concentration of calcium in serum following intraveonous injection of calcium chloride. Am J Physiol 1939; 125:162-71
Shah AP, López A, Wachsner RY, Meymandi SK, El-Bialy AK, Ichiuji AM. Sinus node dysfunction secondary to hyperparathyroidism. J Cardiovasc Pharmacol Ther2004;9:145-7.
Vosnakidis A, Polymeropoulos K, Zarogoulidis P, Zarifis I. Atrioventricular nodal dysfunction secondary to hyperparathyroidism. J Thorac Dis. 2013; 5:4-6.
Garg G, Khadgwat R, Khandelwal D, Gupta N. Vitamin D toxicity presenting as hypercalcemia and complete heart block: An interesting case report. Indian J Endocrinol Metab 2012;16(Suppl 2):S423-5.
Thotakura S, Stafford J, Barnett B, Slicker K, Kramer D, Gupta R. Complete Atrioventricular Nodal Block Due to Malignancy-Related Hypercalcemia. Fed Pract 2016;33:23-5.
Arya AK, Bhadada SK, Mukherjee S, et al. Frequency & predictors of pancreatitis in symptomatic primary hyperparathyroidism. Indian J Med Res 2018;148:721-7.
Thareja S, Manrai M, Shukla R, et al. Pancreatitis and hyperparathyroidism: Still a rare association! Med J Armed Forces India 2019;75:444-9.
Misgar RA, Bhat MH, Rather TA, et al. Primary hyperparathyroidism and pancreatitis. J Endocrinol Invest. 2020; 43:1493-8.
Shepherd JJ. Hyperparathyroidism presenting as pancreatitis or complicated by postoperative pancreatitis. Aust N Z J Surg 1996;66:85-7.
Sitges‐Serra A, Alonso M, de Lecea C, Gores PF, Sutherland DER. Pancreatitis and hyperparathyroidism. Br J Surg 1988;75:158-60.
Kopelberg T, Bartsch D, Printz H, Hasse C, Rothmund M. Die Pankreatitis beim primären Hyperparathyreoidismus (pHPT) ist eine Komplikation des fortgeschrittenen pHPT. Dtsch.med.Wschr 1994;119:719-24.
Felderbauer P, Karakas E, Fendrich V, et al. Pancreatitis risk in primary hyperparathyroidism: relation to mutations in the SPINK1 trypsin inhibitor (N34S) and the cystic fibrosis gene. Am J Gastroenterol 2008;103:368-74.
Felderbauer P, Karakas E, Fendrich V, Lebert R, Bartsch DK, Bulut K. Multifactorial genesis of pancreatitis in primary hyperparathyroidism: evidence for protective (PRSS2) and destructive (CTRC) genetic factors. Exp Clin Endocrinol Diabetes 2011;119:26-9.
Eremkina A, Krupinova J, Dobreva E, et al. Denosumab for management of severe hypercalcemia in primary hyperparathyroidism. Endocr Connect 2020;9:1019-27.
Torregrosa JV, Ramos AM. Uso de bifosfonatos en la enfermedad renal crónica. Nefrologia 2010;30:288-96.
Schweitzer VG, Thompson NW, Harness JK, Nishiyama RH. Management of severe hypercalcemia caused by primary hyperparathyroidism. Arch Surg 1978;113:373-81
DeLellis R, Arnold A, Eng C, et al. Parathyroid adenoma. In: Llyod R, Osamura R, Kloppel G, et al (eds). WHO classification of tumours of endocrine organs.4th ed. IARC: Lyon; 2017. pp. 153-8
Wolpert HR, Vickery AL, Wang CA. Functioning oxyphil cell adenomas of the parathyroid gland. A study of 15 cases. Am J Surg Pathol 1989;13:500-4.
Howson P, Kruijff S, Aniss A, et al. Oxyphil Cell Parathyroid Adenomas Causing Primary Hyperparathyroidism: a Clinico-Pathological Correlation. Endocr Pathol 2015;26:250-4.