Uncommon presentation of small cell lung carcinoma with ectopic adrenocorticotropic hormone secretion and resistant hypokalemia: A case report

Keywords: Small cell lung cancer, Cushing’s syndrome, Hypokalemia

Abstract

Paraneoplastic syndromes can serve as initial indicators of malignancy, with small cell lung cancer accounting for 13% of new lung cancer diagnoses. The most prevalent paraneoplastic syndrome associated with small cell lung cancer is inappropriate antidiuretic hormone syndrome, followed by ectopic adrenocorticotropic hormone-mediated Cushing’s syndrome. Cushing’s syndrome manifests as hypercortisolemia and presents with diverse symptoms, including central obesity, plethora, menstrual irregularities, hypertension/diabetes mellitus, ecchymoses, osteoporosis, muscle weakness, virilization/hirsutism, skin atrophy, decreased libido, and infertility. This case report details the uncommon presentation of small cell lung carcinoma manifesting with ectopic adrenocorticotropic hormone secretion (EAS), leading to resistant hypokalemia and rhabdomyolysis. This case emphasizes the importance of considering EAS in severe cases of Cushing's syndrome and highlights the diagnostic and therapeutic challenges associated with this condition.

Downloads

Download data is not yet available.

References

Ye L, Li X, Kong X, Wang W, Bi Y, Hu L, et al. Hypomethylation in the promoter region of POMC gene correlates with ectopic overexpression in thymic carcinoids. J Endocrinol. 2005; 185(2):337–343. doi: 10.1677/joe.1.05963. 

Young J, Haissaguerre M, Viera-Pinto O, Chabre O, Baudin E, Tabarin A. Cushing’s síndrome due to ectopic ACTH secretion: an expert operational opinion. Eur J Endocrinol. 2020;182:R29-R58. doi: 10.1530/EJE-19-0877. 

Rodrigues P, Castedo JL, Damasceno M, Carvalho D. Ectopic Cushing's syndrome caused by a pulmonary ACTH-secreting tumor in a patient treated with octreotide. Arq Bras Endocrinol Metabol. 2012;56(7):461-464. doi: 10.1590/s0004-27302012000700009. 

Qiang W, Song S, Chen T, Wang Z, Feng J, Zhang J, et al. A rare case of ectopic ACTH syndrome with rhabdomyolysis. BMC Endocr Disord. 2021;21(1):98. doi: 10.1186/s12902-021-00755-0. 

Zhang Wen, Li Chuanwei, Zeng Chunyu, Hui H, Weimin L. Rhabdomyolysis presenting with severe hypokalemia in hypertensive patients: a case series BMC Research Notes. 2013; 6:155. doi: 10.1186/1756-0500-6-155. 

Isidori AM, Kaltsas GA, Pozza C, Frajese V, Newell-Price J, Reznek RH, et al. The ectopic adrenocorticotropin syndrome: clinical features, diagnosis, management, and long-term follow-up. J Clin Endocrinol Metab. 2006; 91 (2): 371-377. doi: 10.1210/jc.2005-1542. 

Espinosa-de-Los-Monteros AL, Ramírez-Rentería C, Mercado M. Clinical Heterogeneity of Ectopic ACTH Syndrome: A Long-Term Follow-Up Study. Endocr Pract. 2020; 26(12):1435-1441. doi: 10.4158/EP-2020-0368. 

Lopez-Montoya V, Gutierrez-Restrepo J, Grajales JLT, Aristizabal N, Pantoja D, Roman-Gonzalez A, et al. Ectopic Cushing syndrome in Colombia. Arch Endocrinol Metab. 2021;64(6):687-694. doi: 10.20945/2359-3997000000271. 

Sathyakumar S, Paul T, Asha HS, Gnanamuthu BR, Paul MJ, Abraham DT, et al. ECTOPIC CUSHING SYNDROME:A 10-YEAR EXPERIENCE FROM A TERTIARY CARE CENTER IN SOUTHERN INDIA. Endocr Pract. 2017; 23(8):907–914. doi: 10.4158/EP161677.OR. 

Miret M, Horváth-Puhó E, Déruaz-Luyet A, Sørensen HT, Ehrenstein V. Potential paraneoplastic syndromes and selected autoimmune conditions in patients with non-small cell lung cancer and small cell lung cancer: A population-based cohort study. PLoS One. 2017; 12(8):e0181564. doi: 10.1371/journal.pone.0181564. 

Jeong C, Lee J, Ryu S, Lee HY, Shin AY, Kim JS, et al. A Case of Ectopic Adrenocorticotropic Hormone Syndrome in Small Cell Lung Cancer. Tuberc Respir Dis (Seoul). 2015 Oct;78(4):436-9. doi: 10.4046/trd.2015.78.4.436. 

Foray N, Stone T, Johnson A, Ali M, Kulkarni S, Gao J, et al. Severe metabolic alkalosis-a diagnostic dilemma. Respir Med Case Rep. 2018; 25:177-180. doi: 10.1016/j.rmcr.2018.08.019. 

Mahapatra S, Lavina A, Bello A, Mehta's J.P. A Rare Case of Ectopic Adrenocorticotropic Hormone Syndrome Causing Severe Hypokalemia and Metabolic Alkalosis in Small Cell Lung Cancer. Am J Respir Crit Care Med. 2022; 205:A3388. doi: 10.1164/ajrccm-conference.2022.205.1_MeetingAbstracts.A3388. 

Franco F, Carcereny E, Guirado M, Ortega AL, López-Castro R, Rodríguez-Abreu D, et al. Epidemiology, treatment, and survival in small cell lung cancer in Spain: Data from the Thoracic Tumor Registry. PLoS One. 2021;16(6):e0251761. doi:10.1371/journal.pone.0251761. 

Hine J, Schwell A, Kairys N. An Unlikely Cause of Hypokalemia. J Emerg Med. 2017;52(5):e187-e191. doi: 10.1016/j.jemermed.2016.12.011. 

Richa CG, Saad KJ, Halabi GH, Gharios EM, Nasr FL, Merheb MT. Case-series of paraneoplastic Cushing syndrome in small-cell lung cancer. Endocrinol Diabetes Metab Case Rep. 2018; 8;2018:18-0004. doi: 10.1530/EDM-18-0004. 

Zhang X, Meng L, Xiao Y, Chen Z. Case report: Radiofrequency ablation combined with biopsy for Cushing’s syndrome due to ectopic ACTH lesions in the lung. Front. Oncol. 2022;12:1059308. doi: 10.3389/fonc.2022.1059308. 

Suyama K, Naito Y, Yoh K, Niho S, Goto K, Ohmatsu H, et al. Development of Cushing's syndrome during effective chemotherapy for small cell lung cancer. Intern Med. 2011;50(4):335-8. doi: 10.2169/internalmedicine.50.4127. 

Braun J, Grinshpun A, Atlan K, Sachar S, Knigen A, Yosha-Orpaz L, et al. Intractable Hypokalemia: a Red Herring for Rapidly Evolving Ectopic Cushing's Syndrome. Isr Med Assoc J. 2019; 21(1):60-62. 

Lobo Ferreira T, Nunes da Silva T, Canário D, Francisca Delerue M. Hypertension and severe hypokalaemia associated with ectopic ACTH production. BMJ Case Rep. 2018;16;2018:bcr2017223406. doi: 10.1136/bcr-2017-223406. 

Mohib O, Papleux E, Remmelink M, Gottignies P, De Bels D. An ectopic Cushing’s syndrome as a cause of severe refractory hypokalemia in the ICU. Acta Clinica Belgica. 2021; 76:5, 373-378, doi: 10.1080/17843286.2020.1734162. 

Alba EL, Japp EA, Fernandez-Ranvier G, Badani K, Wilck E, Ghesani M, et al. The Mount Sinai Clinical Pathway for the Diagnosis and Management of Hypercortisolism due to Ectopic ACTH Syndrome. J Endocr Soc. 2022; 6(7):bvac073. doi: 10.1210/jendso/bvac073. 

Raj R, Taylor RK, Owen D. A Rapidly Progressive Case of Ectopic Adrenocorticotropic Hormone (ACTH) Syndrome. Am J Case Rep. 2021;22:e934437. doi: 10.12659/AJCR.934437. 

Raff H, Sharma ST, Nieman LK. Physiological basis for the etiology, diagnosis, and treatment of adrenal disorders: Cushing's syndrome, adrenal insufficiency, and congenital adrenal hyperplasia. Compr Physiol. 2014;4(2):739-769. doi:10.1002/cphy.c130035. 

Lacroix A, Feelders RA, Stratakis CA, et al. Cushing's syndrome. Lancet. 2015;386:913-927. doi: 10.1016/S0140-6736(14)61375-1. 


Abstract views: 411
PDF Downloads: 334
Published
2023-12-05
How to Cite
Geçgel, E., Alp, A., Karpuzoğlu, E. K., Genek, D. G., & Huddam, B. (2023). Uncommon presentation of small cell lung carcinoma with ectopic adrenocorticotropic hormone secretion and resistant hypokalemia: A case report. Ukrainian Journal of Nephrology and Dialysis, (1(81), 3-9. https://doi.org/10.31450/ukrjnd.1(81).2024.01

Most read articles by the same author(s)