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Abstract
A 30-year-old euthyroid male patient presented with a gradually progressive, painless mass in the neck that moved with deglutition, associated with generalized weakness, needing assistance for activities of daily life, and a pathological fracture. Biochemical reports revealed hyperparathyroidism and hypercalcemia. Radiological investigations revealed a complex mass in the postero-medial aspect of the left thyroid lobe, generalized bone resorption with healed pathological fracture, and nephrolithiasis. FNAC was unable to rule out parathyroid neoplasm. The patient underwent a left hemithyroidectomy and a parathyroidectomy. Aggressive IV calcium therapy managed the post-operative hungry bone syndrome. Post-operative histopathology suggested giant parathyroid adenoma of the left superior parathyroid gland.
Parathormone, secreted from the parathyroid glands, is responsible for the maintenance of calcium homeostasis in the body.[1] Increased secretion of parathyroid hormone results in primary hyperparathyroidism (or PHPT), causing symptoms characterized by the mnemonic “stones, bones, groans, thrones, and psychiatric overtones’’.[2,3] Osteoporosis and bone fractures are the most common symptoms of primary hyperparathyroidism. Calcium deposits in the renal parenchyma and recurrent nephrolithiasis lead to decreased kidney function and phosphate retention.[4,5]
PHPT has been reported in 1% of the adult population and in 2% after the age of 55 years in Western series.[6] In an Indian series, the male: female ratio was 1.7: 1. Almost three quarters (71.5%) of cases were less than 40 years of age, lower than the fifth and sixth decades reported from developed nations.[7] The common causes of primary hyperparathyroidism are sporadic, solitary adenomas (80-85%), parathyroid hyperplasia (10%), and parathyroid carcinoma (2-5%).[8,9] Parathyroid adenomas (PTAs) are usually small, measuring < 2 cm and weighing < 1 gm. GPTAs (Giant PTAs) are adenomas that weigh more than 3.5 gm and are usually rare.
Both PTA and GPTA present with the syndrome of primary hyperparathyroidism.[10]
Most hyperparathyroidism patients are asymptomatic or have nonspecific symptoms such as fatigue, pain, and weakness that are generally missed in the initial phases. Most patients with hypercalcemia are discovered incidentally on routine laboratory screening. The present case report shows a delay in the diagnosis of a symptomatic case of hyperparathyroidism, caused by parathyroid adenoma, due to the difficulty in the diagnosis of such cases in rural areas.
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References
- Prihantono P, Palinggi E, Haryasena H, et al. Surgical treatment for parathyroid adenoma: a case report. Open Access Macedonian Journal of Medical Sciences 2019;7(15):2497. DOI: https://doi.org/10.3889/oamjms.2019.418
- Neagoe RM, Sala DT, Borda A, et al. Clinicopathologic and therapeutic aspects of giant parathyroid adenomas - three case reports and short review of the literature. Romanian J Morphol Embryol 2014;55(2 Suppl):669-74.
- Shields CL, Hasanreisoglu M, Saktanasate J, et al. Sclerochoroidal calcification: Clinical features, outcomes, and relationship with hypercalcemia and parathyroid adenoma in 179 eyes. Retina 2015;35:547–54. DOI: https://doi.org/10.1097/IAE.0000000000000450
- Bilezikian JP, Cusano NE, Khan AA, et al. Primary hyperparathyroidism. Nature reviews Disease Primers 2016;2:16033.
- Baum M. The bone-kidney axis. Current Opinion in Pediatrics 2014;26(2):177. DOI: https://doi.org/10.1097/MOP.0000000000000071
- AACE/AAES position statement. Endocrine Practice 2005;11(1):50–4. DOI: https://doi.org/10.4158/EP.11.2.125
- Pradeep PV, Jayashree B, Mishra A, et al. Systematic review of primary hyperparathyroidism in India: the past, present, and the future trends. International journal of Endocrinology 2011;2011(1):921814. DOI: https://doi.org/10.1155/2011/921814
- Lofrese JJ, Lappin SL. Physiology, parathyroid. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing, 2019. Available from: https://www.ncbi.nlm.nih.gov/books/ NBK-482510/
- Bilezikian JP, Cusano NE, Khan AA, et al. Primary hyperparathyroidism. Nature reviews Disease Primers 2016;2:16033. DOI: https://doi.org/10.1038/nrdp.2016.33
- Madkhali T, Alhefdhi A, Chen H, et al. Primary hyperparathyroidism. Ulus Cerrahi Derg 2016;32(1):58-66. DOI: https://doi.org/10.5152/UCD.2015.3032
- Bilezikian JP, Brandi ML, Eastell R, et al. Guidelines for the management of asymptomatic primary hyperparathyroidism: summary statement from the Fourth International Workshop. J Clin Endocrinol Metab 2014;99(10):3561-9. DOI: https://doi.org/10.1210/jc.2014-1413
- Sisodiya R, Kumar S, Palankar N, et al. Case report on giant parathyroid adenoma with review of literature. Indian Journal of Surgery 2013;75:21-2.
- Spanheimer PM, Stoltze AJ, Howe JR, et al. Do giant parathyroid adenomas represent a distinct clinical entity? Surgery 2013;154(4):714–8.
- Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. In Annales de Chirurgie 2002;127(3):198-202.
- Vilallonga R, Zafon C, Migone R, et al. Giant intrathyroidal parathyroid adenoma. J Emerg Trauma Shock 2012;5(2):196-8. DOI: https://doi.org/10.4103/0974-2700.96497
- Al-Hassan MS, Mekhaimar M, El Ansari W, et al. Giant parathyroid adenoma: a case report and review of the literature. Journal of Medical Case Reports 2019;13(1):1-9.
- Rutledge S, Harrison M, O'Connell M, et al. Acute presentation of a giant intrathyroidal parathyroid adenoma: a case report. J Med Case Rep 2016;10(1):286. DOI: https://doi.org/10.1186/s13256-016-1078-1
- Sisodiya R, Kumar S, Palankar N, et al. Case report on giant parathyroid adenoma with review of literature. Indian J Surg 2011;75(Suppl 1):21–2. DOI: https://doi.org/10.1007/s12262-011-0306-6
- Castro MA, López AA, Fragueiro LM, et al. Giant parathyroid adenoma: differential aspects compared to parathyroid carcinoma. Endocrinol Diabetes Metab Case Rep 2017;2017:1. DOI: https://doi.org/10.1530/EDM-17-0041
- Aggarwal V, Mishra A, Bhargav PR, et al. Giant parathyroid adenoma. ANZ J Surg 2009;79(1–2):91. DOI: https://doi.org/10.1111/j.1445-2197.2008.04815.x
- Spanheimer PM, Stoltze AJ, Howe JR, et al. Do giant parathyroid adenomas represent a distinct clinical entity? Surgery 2013;154(4):714-8. DOI: https://doi.org/10.1016/j.surg.2013.05.013
- Ajmi S, Sfar R, Trimeche S, et al. Scintigraphic findings in hungry bone syndrome following parathyroidectomy. Revista Espanola de Medicina Nuclear 2010;29(2):81-3. DOI: https://doi.org/10.1016/j.remn.2009.10.003
- Agarwal G, Prasad KK, Kar DK, et al. Indian primary hyperparathyroidism patients with parathyroid carcinoma do not differ in clinicoinvestigative characteristics from those with benign parathyroid pathology. World J Surg 2006;30:732-42.
- Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. Ann Chir 2002;127:198-202.
- Agarwal G, Prasad KK, Kar DK, et al. Indian primary hyperparathyroidism patients with parathyroid carcinoma do not differ in clinicoinvestigative characteristics from those with benign parathyroid pathology. World J Surg 2006;30:732-42. DOI: https://doi.org/10.1007/s00268-005-0366-5
- Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. Ann. Chir 2002;127:198–202. DOI: https://doi.org/10.1016/S0003-3944(02)00717-4
- Al-Hassan MS, Mekhaimar M, El Ansari W, et al. Giant parathyroid adenoma: a case report and review of the literature. Journal of Medical Case Reports 2019;13(1):1-9. DOI: https://doi.org/10.1186/s13256-019-2257-7
References
Prihantono P, Palinggi E, Haryasena H, et al. Surgical treatment for parathyroid adenoma: a case report. Open Access Macedonian Journal of Medical Sciences 2019;7(15):2497. DOI: https://doi.org/10.3889/oamjms.2019.418
Neagoe RM, Sala DT, Borda A, et al. Clinicopathologic and therapeutic aspects of giant parathyroid adenomas - three case reports and short review of the literature. Romanian J Morphol Embryol 2014;55(2 Suppl):669-74.
Shields CL, Hasanreisoglu M, Saktanasate J, et al. Sclerochoroidal calcification: Clinical features, outcomes, and relationship with hypercalcemia and parathyroid adenoma in 179 eyes. Retina 2015;35:547–54. DOI: https://doi.org/10.1097/IAE.0000000000000450
Bilezikian JP, Cusano NE, Khan AA, et al. Primary hyperparathyroidism. Nature reviews Disease Primers 2016;2:16033.
Baum M. The bone-kidney axis. Current Opinion in Pediatrics 2014;26(2):177. DOI: https://doi.org/10.1097/MOP.0000000000000071
AACE/AAES position statement. Endocrine Practice 2005;11(1):50–4. DOI: https://doi.org/10.4158/EP.11.2.125
Pradeep PV, Jayashree B, Mishra A, et al. Systematic review of primary hyperparathyroidism in India: the past, present, and the future trends. International journal of Endocrinology 2011;2011(1):921814. DOI: https://doi.org/10.1155/2011/921814
Lofrese JJ, Lappin SL. Physiology, parathyroid. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing, 2019. Available from: https://www.ncbi.nlm.nih.gov/books/ NBK-482510/
Bilezikian JP, Cusano NE, Khan AA, et al. Primary hyperparathyroidism. Nature reviews Disease Primers 2016;2:16033. DOI: https://doi.org/10.1038/nrdp.2016.33
Madkhali T, Alhefdhi A, Chen H, et al. Primary hyperparathyroidism. Ulus Cerrahi Derg 2016;32(1):58-66. DOI: https://doi.org/10.5152/UCD.2015.3032
Bilezikian JP, Brandi ML, Eastell R, et al. Guidelines for the management of asymptomatic primary hyperparathyroidism: summary statement from the Fourth International Workshop. J Clin Endocrinol Metab 2014;99(10):3561-9. DOI: https://doi.org/10.1210/jc.2014-1413
Sisodiya R, Kumar S, Palankar N, et al. Case report on giant parathyroid adenoma with review of literature. Indian Journal of Surgery 2013;75:21-2.
Spanheimer PM, Stoltze AJ, Howe JR, et al. Do giant parathyroid adenomas represent a distinct clinical entity? Surgery 2013;154(4):714–8.
Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. In Annales de Chirurgie 2002;127(3):198-202.
Vilallonga R, Zafon C, Migone R, et al. Giant intrathyroidal parathyroid adenoma. J Emerg Trauma Shock 2012;5(2):196-8. DOI: https://doi.org/10.4103/0974-2700.96497
Al-Hassan MS, Mekhaimar M, El Ansari W, et al. Giant parathyroid adenoma: a case report and review of the literature. Journal of Medical Case Reports 2019;13(1):1-9.
Rutledge S, Harrison M, O'Connell M, et al. Acute presentation of a giant intrathyroidal parathyroid adenoma: a case report. J Med Case Rep 2016;10(1):286. DOI: https://doi.org/10.1186/s13256-016-1078-1
Sisodiya R, Kumar S, Palankar N, et al. Case report on giant parathyroid adenoma with review of literature. Indian J Surg 2011;75(Suppl 1):21–2. DOI: https://doi.org/10.1007/s12262-011-0306-6
Castro MA, López AA, Fragueiro LM, et al. Giant parathyroid adenoma: differential aspects compared to parathyroid carcinoma. Endocrinol Diabetes Metab Case Rep 2017;2017:1. DOI: https://doi.org/10.1530/EDM-17-0041
Aggarwal V, Mishra A, Bhargav PR, et al. Giant parathyroid adenoma. ANZ J Surg 2009;79(1–2):91. DOI: https://doi.org/10.1111/j.1445-2197.2008.04815.x
Spanheimer PM, Stoltze AJ, Howe JR, et al. Do giant parathyroid adenomas represent a distinct clinical entity? Surgery 2013;154(4):714-8. DOI: https://doi.org/10.1016/j.surg.2013.05.013
Ajmi S, Sfar R, Trimeche S, et al. Scintigraphic findings in hungry bone syndrome following parathyroidectomy. Revista Espanola de Medicina Nuclear 2010;29(2):81-3. DOI: https://doi.org/10.1016/j.remn.2009.10.003
Agarwal G, Prasad KK, Kar DK, et al. Indian primary hyperparathyroidism patients with parathyroid carcinoma do not differ in clinicoinvestigative characteristics from those with benign parathyroid pathology. World J Surg 2006;30:732-42.
Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. Ann Chir 2002;127:198-202.
Agarwal G, Prasad KK, Kar DK, et al. Indian primary hyperparathyroidism patients with parathyroid carcinoma do not differ in clinicoinvestigative characteristics from those with benign parathyroid pathology. World J Surg 2006;30:732-42. DOI: https://doi.org/10.1007/s00268-005-0366-5
Lalanne-Mistrih ML, Ognois-Ausse P, Goudet P, et al. Giant parathyroid tumors: characterization of 26 glands weighing more than 3.5 grams. Ann. Chir 2002;127:198–202. DOI: https://doi.org/10.1016/S0003-3944(02)00717-4
Al-Hassan MS, Mekhaimar M, El Ansari W, et al. Giant parathyroid adenoma: a case report and review of the literature. Journal of Medical Case Reports 2019;13(1):1-9. DOI: https://doi.org/10.1186/s13256-019-2257-7