Palabras clave
adenoma atípico
tumores pardos
lesiones líticas óseas
hipercalcemia
cirugía de paratiroides
seguimiento clínico
Cómo citar
Resumen
Introducción: el adenoma atípico de paratiroides (APA) es una neoplasia infrecuente con comportamiento intermedio entre el adenoma benigno y el carcinoma, caracterizado por hipercalcemia severa y, en casos avanzados, lesiones óseas líticas como los tumores pardos. Estas manifestaciones pueden simular procesos metastásicos, dificultando el diagnóstico oportuno.
Objetivo: describir un caso clínico de APA gigante con múltiples tumores pardos que simulaban metástasis óseas, destacando los hallazgos clínicos, diagnósticos e histopatológicos, así como la evolución posquirúrgica y su seguimiento a largo plazo.
Presentación del caso: mujer de 33 años con dolor óseo crónico, debilidad muscular progresiva y pérdida ponderal significativa. Al examen físico destacaban tumoración mandibular y masa cervical. Los estudios bioquímicos revelaron hipercalcemia (14,16 mg/dl), PTH elevada (725 pg/ml) y fosfatasa alcalina aumentada (1183 U/l). Las imágenes mostraron múltiples lesiones líticas óseas compatibles con tumores pardos. La gammagrafía SESTAMIBI localizó un adenoma en el lóbulo tiroideo izquierdo. La paciente fue sometida a paratiroidectomía, confirmándose el diagnóstico de APA. El seguimiento por dos años mostró normalización bioquímica y mejoría clínica y radiológica sostenida.
Discusión y conclusión: el APA es un diagnóstico de exclusión que requiere evaluación histopatológica e inmunohistoquímica para diferenciarlo de entidades malignas. Su presentación clínica puede ser engañosa y confundirse con patología metastásica. La cirugía sigue siendo el pilar del tratamiento, con un rol fundamental del seguimiento a largo plazo para detectar recurrencias o progresión. Este caso subraya la importancia de considerar etiologías endocrinas como el APA ante lesiones óseas agresivas. El diagnóstico preciso y el abordaje oportuno permiten revertir complicaciones graves y mejorar el pronóstico funcional del paciente.
Citas
Soto-Pedre E, Newey PJ, Leese GP. Stable incidence and increasing prevalence of primary hyperparathyroidism in a population-based study in Scotland. J Clin Endocrinol Metab. 2023;108(10):e1117-24. https://doi.org/10.1210/clinem/dgad201
Garifullin AI, Absaliamova RM, Dubinina A, Yufanova AI, Smirnova VM, Ishbulatova JT, et al. Principales tumores de la glándula paratiroides. ??????????? ????????. 2022;16(1):4-12. https://doi.org/10.14341/serg12756
Siqueira H, Lima C, Furtado A, Ferreira A, Canto M, Cunha J, et al. ODP121 Recurrent atypical parathyroid adenoma: a case report. J Endocr Soc. 2022;6(supl. 1):A176. https://doi.org/10.1210/jendso/bvac150.362
Boro H, Alam S, Kubihal V, Khatiwada S, Kubihal S, Agarwal S, et al. Atypical parathyroid adenoma: severe manifestations in an adolescent girl. Pediatr Endocrinol Diabetes Metab. 2022;28(1):91-100. https://doi.org/10.5114/pedm.2021.109127
Limon MA, Murtaza Ali M, Banisaeed N, Shannon S, Sharma A, Jakoby M. PSAT198 atypical parathyroid adenoma is an unusual cause of primary hyperparathyroidism. J Endocr Soc. 2022;6(supl. 1):A208-9. https://doi.org/10.1210/jendso/bvac150.429
Aydemir E, Ünsal Y, Ate? C, Cander S, Gül Ö, Ersoy C, et al. Differences between atypical parathyroid tumors and parathyroid adenomas in patients with primary hyperparathyroidism. Res Square. 2022. https://doi.org/10.21203/rs.3.rs-1658291/v1
Erickson LA, Mete O, Juhlin CC, Perren A, Gill AJ. Overview of the 2022 WHO Classification of Parathyroid Tumors. Endocr Pathol. 2022;33(1):64-89. https://doi.org/10.1007/s12022-022-09709-1
Bilezikian JP, Brandi ML, Eastell R, Silverberg SJ, Udelsman R, Marcocci C, 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. https://doi.org/10.1210/jc.2014-1413
Gill AJ. Understanding the genetic basis of parathyroid carcinoma. Endocr Pathol. 2014;25(1):30-4. https://doi.org/10.1007/s12022-013-9294-3
Shane E. Clinical review: parathyroid carcinoma. J Clin Endocrinol Metab. 2001;86(2):485-93. https://doi.org/10.1210/jcem.86.2.7207
Guise TA, Wysolmerski JJ. Cancer-Associated Hypercalcemia. N Engl J Med. 2022;386(15):1443-51. https://doi.org/10.1056/nejmcp2113128
El-Hajj Fuleihan G, Clines GA, Hu MI, Marcocci C, Murad MH, Piggott T, et al. Management of hypercalcemia of malignancy in adults: An Endocrine Society clinical practice guideline. J Clin Endocrinol Metab. 2023;108(3):507-28. https://doi.org/10.1210/clinem/dgac621
Walker MD, Shane E. Hypercalcemia: a review. JAMA. 2022;328(16):1624-36. https://doi.org/10.1001/jama.2022.18331
Lee CT, Chen HC, Lai LW, Yong KC, Lien YH. Effects of furosemide on renal calcium handling. Am J Physiol Renal Physiol. 2007;293(4):F1231-7. https://doi.org/10.1152/ajprenal.00038.2007
LeGrand SB, Leskuski D, Zama I. Narrative review: furosemide for hypercalcemia: an unproven yet common practice. Ann Intern Med. 2008;149(4):259-63. https://doi.org/10.7326/0003-4819-149-4-200808190-00007
Jain N, Reilly RF. Hungry bone syndrome. Curr Opin Nephrol Hypertens. 2017;26(4):250-5. https://doi.org/10.1097/mnh.0000000000000327
Witteveen JE, van Thiel S, Romijn JA, Hamdy NA. Hungry bone syndrome: still a challenge in the post-operative management of primary hyperparathyroidism: a systematic review of the literature. Eur J Endocrinol. 2013;168(3):R45-53. https://doi.org/10.1530/eje-12-0528
Goldfarb M, Gondek SS, Lim SM, Farra JC, Nose V, Lew JI. Postoperative hungry bone syndrome in patients with secondary hyperparathyroidism of renal origin. World J Surg. 2012;36(6):1314-9. https://doi.org/10.1007/s00268-012-1560-x
Lee IT, Sheu WH, Tu ST, Kuo SW, Pei D. Bisphosphonate pretreatment attenuates hungry bone syndrome after parathyroid operation in subjects with primary hyperparathyroidism. J Bone Miner Metab. 2006;24(3):255-8. https://doi.org/10.1007/s00774-005-0680-x
Pal R, Gautam A, Bhadada SK. Role of bisphosphonates in preventing postoperative hungry bone syndrome in primary hyperparathyroidism: a meta-analysis and need for randomized controlled trials. Drug Res. 2021;71(2):108-9. https://doi.org/10.1055/a-1325-0351
Stan R, Dumitrache S, Zubaci A, Iliescu M, Tarna M, Grosu I, et al. Atypical parathyroid adenomas, a challenging diagnosis and management - case report. Endocr Abstracts. 2023:90. https://doi.org/10.1530/endoabs.90.EP204
Galani A, Morandi R, Dimko M, Molfino S, Baronchelli C, Lai S, et al. Atypical parathyroid adenoma: clinical and anatomical pathologic features. World J Surg Oncol. 2021;19(1):19. https://doi.org/10.1186/s12957-021-02123-7
Cetani F, Marcocci C, Torregrossa L, Pardi E. Atypical parathyroid adenomas: challenging lesions in the differential diagnosis of endocrine tumors. Endocr Relat Cancer. 2019;26(7): R441-64. https://doi.org/10.1530/erc-19-0135
Truran PP, Johnson SJ, Bliss RD, Lennard TW, Aspinall SR. Parafibromin, galectin-3, PGP9.5, Ki67, and cyclin D1: using an immunohistochemical panel to aid in the diagnosis of parathyroid cancer. World J Surg. 2014;38(11):2845-2854. https://doi.org/10.1007/s00268-014-2700-2
Singhal AA. Atypical ultrasound appearances of enlarged parathyroid. En: Singhal AA, autor. Atlas of Parathyroid Sonography. Singapur: Springer; 2023. https://doi.org/10.1007/978-981-19-7919-4_3
Bhan A, Athimulam S, Kumari P, Pal R, Bhadada SK, Cook B, et al. Large parathyroid adenomas: potential mechanisms to reconcile adenoma size and disease phenotype. Front Endocrinol. 2023;14:1009516. https://doi.org/10.3389/fendo.2023.1009516
Deb P, Devarasetty M, Vulpala A, Nalla S. ODP091 Giant parathyroid adenoma presenting with pseudoclubbing - a case report. J Endocr Society. 2022;6(supl. 1):A163. https://doi.org/10.1210/jendso/bvac150.334
Mahmodlou R, Sedokani A, Pezeshk A, Najafinejad B. Giant parathyroid adenoma: a case report. J Med Case Rep. 2022;16(1):150. https://doi.org/10.1186/s13256-022-03401-y
Hoshi M, Takami M, Kajikawa M, Teramura K, Okamoto T, Yanagida I, et al. A case of multiple skeletal lesions of brown tumors, mimicking carcinoma metastases. Arch Orthop Trauma Surg. 2008;128(2):149-54. https://doi.org/10.1007/s00402-007-0312-0
Alharmoodi A, Alsheryani M, Alhajeri L, Alshaikh M, Aby Ali, Alfelasi M. Brown tumor secondary to hyperparathyroidism: a case report. Am J Surg Case Rep. 2023;5(2):2-3. http://dx.doi.org/10.31487/j.AJSCR.2023.02.03
Gite V, Majumder A, Dhakane M. Brown tumor of the rib associated with primary hyperparathyroidism misdiagnosed as metastatic deposit - A case report. Arch Citol Histopatol Res. 2023;8(1):63-5. https://doi.org/10.18231/j.achr.2023.015
Hiremath R, Moudgalya A. Case report: brown tumor, a masquerader of malignancy. Indian J Musculoskelet Radiol. 2023;5(1):56-60. http://doi.org/10.25259/IJMSR_23_2022
Henderson S, Hamour S. Brown tumour. BMJ. 2023:381. https://doi.org/10.1136/bmj-2022-071564.r1
Keyser JS, Postma GN. Brown tumor of the mandible. Am J Otolaryngol. 1996;17(6):407-10. https://doi.org/10.1016/s0196-0709(96)90075-7
Turek D, Haefliger S, Ameline B, Alborelli I, Calgua B, Hartmann W, et al. Brown tumors belong to the spectrum of KRAS-driven neoplasms. Am J Surg Pathol. 2022;46(11):1577-82. https://doi.org/10.1097/pas.0000000000001963
Shah S, Fujikawa P, Brand K, Munshi V, Patel KB. Giant parathyroid adenoma: a case report. Cureus. 2023;15(1):e34140. https://doi.org/10.7759/cureus.34140
Al-Hassan MS, Mekhaimar M, El Ansari W, Darweesh A, Abdelaal A. Giant parathyroid adenoma: a case report and review of the literature. J Med Case Rep. 2019;13(1):332. https://doi.org/10.1186/s13256-019-2257-7
Ko?ciuszko MM, Adamska A, Pop?awska-Kita A, Siewko K, Rydzewska-Roso?owska A, My?liwiec J, et al. Hypercalcaemic crisis due to parathyroid adenoma of atypical location. Endokrynol Pol. 2022;73(1):169-70. https://doi.org/10.5603/ep.a2022.0007
Besrour C, Rojbi I, Mchirgui N, Ben Nacef I, Khiari K. Atypical parathyroid adenoma: a case report. Clin Case Rep. 2022;10(12):e06785. https://doi.org/10.1002/ccr3.6785
Kim EK, Krupinova J, Belousov PV, Trutneva KA, Mokrysheva N. Treatment of parathyroid cancer: current status and near-term prospects. Ann Russ Acad Med Sci. 2022;77(5):362-70. https://doi.org/10.15690/vramn2132
Ta B, Bennett MJ. Refractory Hypercalcemia Secondary to Metastatic Parathyroid Carcinoma Treated With Immunotherapy. JCEM Case Rep. 2024;2(7):luae127. Published 2024 Jul 15. https://doi.org/10.1210/jcemcr/luae127
Jiao Y, Wang Q, Yin Y, et al. Clinical and genetic analysis of atypical parathyroid adenoma. Frontiers in Endocrinology. 2023;14:1027598. https://doi.org/10.3389/fendo.2023.1027598
Maser RE, Lenhard MJ, Pohlig RT, Balagopal P, Abdel-Misih R. Effect of parathyroidectomy on osteopontin and undercarboxylated osteocalcin in patients with primary hyperparathyroidism. Endocr Res. 2018;43(1):21-8. https://doi.org/10.1080/07435800.2017.1369432
Ko WC, Liu CL, Lee JJ, Liu TP, Wu CJ, Cheng SP. Osteocalcin is an independent predictor for hungry bone syndrome after parathyroidectomy. World J Surg. 2020;44(3):795-802. https://doi.org/10.1007/s00268-019-05251-0
Syka?a M, Szumowski P, Mojsak M, Abdelrazek S, ?ukowski ?, Lipinska D, et al. Assessment of clinical utility of assaying FGF-23, klotho protein, osteocalcin, NTX, and sclerostin in patients with primary hyperparathyroidism. J Clin Med. 2021;10(14):3089. https://doi.org/10.3390/jcm10143089
Ellakany SA, Hashaad DI, Merghany AF. Serum osteocalcin level in end-stage renal disease patients on maintenance hemodialysis after parathyroidectomy in relation to parathyroid hormone level. Egypt J Intern Med. 2019;31:795-803. https://doi.org/10.4103/ejim.ejim_84_19
Lu J, Wei YM, Zang P, Wang W, Feng Z, Yuan Y, et al. Circulating osteocalcin is associated with time in range and other metrics assessed by continuous glucose.monitoring in type 2 diabetes. Diabetol Metab Syndr. 2022;14(1):109. https://doi.org/10.1186/s13098-022-00863-4
Rianon N, Alex G, Callender G, Jimenez C, Hu M, Grubbs E, et al. Preoperative serum osteocalcin may predict elevated postoperative parathyroid hormone levels in patients with primary hyperparathyroidism. World J Surg. 2012;36(6):1320-6. https://doi.org/10.1007/s00268-012-1432-4
Esta obra está bajo una licencia internacional Creative Commons Atribución-NoComercial-SinDerivadas 4.0.
Derechos de autor 2025 Revista Colombiana de Endocrinología, Diabetes & Metabolismo