Neuroendocrine tumor of thymus with ectopic production of adrenocorticotropic hormone in a 13-year-old child. Case report, 5-year follow-up
- 作者: Slashchuk K.1, Reinberg M.1, Bazarova E.1, Kareva M.1, Przhiyalkovskaya E.1, Degtiarev M.1, Pikin O.2
-
隶属关系:
- Endocrinology Research Center
- Hertsen Moscow Oncology Research Institute – branch of the National Medical Research Radiological Centre
- 期: 卷 26, 编号 1 (2024)
- 页面: 106-111
- 栏目: Articles
- URL: https://journals.rcsi.science/1815-1434/article/view/258581
- DOI: https://doi.org/10.26442/18151434.2024.1.202616
- ID: 258581
如何引用文章
全文:
详细
Neuroendocrine tumors (NET) of the thymus are rare, usually aggressive, and prone to metastatic lesion. Ectopic adrenocorticotropic hormone (ACTH) secretion in thymic NET (TNEN) is associated with poor prognosis. Most of TNET express somatostatin receptors, it allows the use of somatostatin receptor scintigraphy (SRS) and SPECT/CT or PET/CT with 68Ga-labeled peptides for diagnosis and staging of the disease. Surgery (macroscopic-complete resection) is the mainstay treatment for TNET. Now, there are no unequivocal data in adjuvant therapy and its effectiveness. We present a case report of the pediatric patient with well differentiated atypical ACTH-producing thymic carcinoid. This localization was verified by whole body somatostatin receptor scintigraphy with 111In-DTPA-octreotide (Octreoscan). The patient proceeded with the surgical treatment followed by remission of hypercorticism without adjuvant chemotherapy. Tumor recurrence with redevelopment of ACTH-ectopic syndrome was detected after 67 months of observation.
作者简介
Konstantin Slashchuk
Endocrinology Research Center
Email: slashuk911@gmail.com
ORCID iD: 0000-0002-3220-2438
SPIN 代码: 3079-8033
Cand. Sci. (Med.)
俄罗斯联邦, MoscowMaria Reinberg
Endocrinology Research Center
Email: e.bazarova.bq@gmail.com
ORCID iD: 0009-0002-1632-2197
еndocrinologis
俄罗斯联邦, MoscowEkaterina Bazarova
Endocrinology Research Center
编辑信件的主要联系方式.
Email: e.bazarova.bq@gmail.com
ORCID iD: 0009-0009-7608-4726
endocrinologist
俄罗斯联邦, MoscowMaria Kareva
Endocrinology Research Center
Email: i_marusya@mail.ru
ORCID iD: 0000-0003-1320-6561
SPIN 代码: 5089-0310
D. Sci. (Med.)
俄罗斯联邦, MoscowElena Przhiyalkovskaya
Endocrinology Research Center
Email: e.bazarova.bq@gmail.com
ORCID iD: 0000-0001-9119-2447
SPIN 代码: 9309-3256
Cand. Sci. (Med.), Assoc. Prof.
俄罗斯联邦, MoscowMikhail Degtiarev
Endocrinology Research Center
Email: germed@mail.ru
ORCID iD: 0000-0001-5652-2607
SPIN 代码: 7725-7831
Department Head
俄罗斯联邦, MoscowOleg Pikin
Hertsen Moscow Oncology Research Institute – branch of the National Medical Research Radiological Centre
Email: e.bazarova.bq@gmail.com
ORCID iD: 0000-0001-6871-6804
SPIN 代码: 2381-5969
D. Sci. (Med.)
俄罗斯联邦, Moscow参考
- Taal BG, Visser O. Epidemiology of neuroendocrine tumours. Neuroendocrinology. 2004;80(Suppl. 1):3-7. doi: 10.1159/000080731
- Yao JC, Hassan M, Phan A, et al. One hundred years after “carcinoid”: Epidemiology of and prognostic factors for neuroendocrine tumors in 35,825 cases in the United States. J Clin Oncol. 2008;26(18):3063-72. doi: 10.1200/JCO.2007.15.4377
- Gaur P, Leary C, Yao JC. Thymic neuroendocrine tumors: A SEER database analysis of 160 patients. Ann Surg. 2010;251(6):1117-21. doi: 10.1097/SLA.0b013e3181dd4ec4
- Thakker RV, Newey PJ, Walls GV, et al. Clinical practice guidelines for multiple endocrine neoplasia type 1 (MEN1). J Clin Endocrinol Metab. 2012;97(9):2990-3011. doi: 10.1210/jc.2012-1230
- Jia R, Sulentic P, Xu JM, et al. Thymic neuroendocrine neoplasms: Biological behaviour and therapy. Neuroendocrinology. 2017;105(2):105-14. doi: 10.1159/000472255
- Sathyakumar S, Paul TV, Asha HS, et al. Ectopic Cushing syndrome: A 10-year experience from a tertiary care center in Southern India. Endocr Pract. 2017;23(8):907-14. doi: 10.4158/EP161677
- Neary NM, Lopez-Chavez A, Abel BS, et al. Neuroendocrine ACTH-producing tumor of the thymus – Experience with 12 patients over 25 years. J Clin Endocrinol Metab. 2012;97(7):2223-30. doi: 10.1210/jc.2011-3355
- Alexandraki KI, Grossman AB. The ectopic ACTH syndrome. Rev Endocr Metab Disord. 2010;11(2):117-26. doi: 10.1007/s11154-010-9139-z
- Баранова О.Д., Румянцев П.О., Слащук К.Ю., Петров Л.О. Радионуклидная визуализация и терапия у пациентов с нейроэндокринными опухолями. Эндокринная хирургия. 2018;11(4):178-90 [Baranova OD, Roumiantsev PO, Slashchuk KY, Petrov LO. Radionuclide imaging and therapy in patients with neuroendocrine tumors. Endocrine Surgery. 2017;11(4):178-90 (in Russian)]. doi: 10.14341/serg9572
- Moran CA, Suster S. Neuroendocrine carcinomas (carcinoid tumor) of the thymus: A clinicopathologic analysis of 80 cases. Am J Clin Pathol. 2000;114(1):100-10. doi: 10.1309/3PDN-PMT5-EQTM-H0CD
- Shah MH, Goldner WS, Benson AB, et al. Neuroendocrine and adrenal tumors, version 2.2021. JNCCN J Natl Compr Cancer Netw. 2021;19(7):839-67. doi: 10.6004/jnccn.2021.0032
- Ose N, Maeda H, Inoue M, et al. Results of treatment for thymic neuroendocrine tumours: multicentre clinicopathological study. Interact Cardiovasc Thorac Surg. 2018;26(1):18-24. doi: 10.1093/icvts/ivx265
- Wick MR, Scheithauer BW, Weiland LH, et al. Primary thymic carcinomas. Am J Surg Pathol. 1982;6(7):613-30. doi: 10.1097/00000478-198210000-00003
- Lau J, Ioan Cvasciuc T, Simpson D, et al. Continuing challenges of primary neuroendocrine tumours of the thymus: A concisereview. Eur J Surg Oncol. 2022;48(12):2360-8. doi: 10.1016/j.ejso.2022.07.017
- Lang M, Hackert T, Anamaterou C. Long-term effect of everolimus in recurrent thymic neuroendocrine neoplasia. Clin Endocrinol (Oxf). 2021;95(5):744-51. doi: 10.1111/cen.14572
- Yliaska I, Tokola H, Ebeling T, et al. Thymic neuroendocrine tumors in patients with multiple endocrine neoplasia type 1. Endocrine. 2022;77(3):527-37. doi: 10.1007/s12020-022-03099-4
- Kulke MH, Lenz HJ, Meropol NJ, et al. Activity of sunitinib in patients with advanced neuroendocrine tumors. J Clin Oncol. 2008;26(20):3403-10. doi: 10.1200/JCO.2007.15.9020
- Pulido GE, Castellano DE, Garcia-Carbonero R, et al. PAZONET: Results of a phase II trial of pazopanib as a sequencing treatment in progressive metastatic neuroendocrine tumors (NETs) patients (pts), on behalf of the Spanish task force for NETs (GETNE) – NCT01280201. J Clin Oncol. 2012;30(15 Suppl.):4119. doi: 10.1200/jco.2012.30.15_suppl.4119
- Wang S, Gu Z, Zhu L, et al. Genetic insights into thymic carcinomas and thymic neuroendocrine neoplasms denote prognosis signatures and pathways. Chin Med J (Engl). 2023;136(22):2712-21. doi: 10.1097/CM9.0000000000002852
- Barroso-Sousa R, Lerario AM, Evangelista J, et al. Complete resolution of hypercortisolism with sorafenib in a patient with advanced medullary thyroid carcinoma and ectopic ACTH (adrenocorticotropic hormone) syndrome. Thyroid. 2014;24(6)1062-6. doi: 10.1089/thy.2013.0571
- Miyata R, Hamaji M, Omasa M, et al. The treatment and survival of patients with postoperative recurrent thymic carcinoma and neuroendocrine carcinoma: a multicenter retrospective study. Surg Today. 2021;51(4):502-10. doi: 10.1007/s00595-020-02102-7
- Girard N. Neuroendocrine tumors of the thymus: The oncologist point of view. J Thorac Dis. 2017;9(Suppl. 15):S1491-500. doi: 10.21037/jtd.2017.08.18