A case of a fifteen-year-old patient suffering from rare adenocystic lung carcinoma bronchoplastic surgery

Adenoid cystic carcinoma of the lung is a relatively rare malignant tumor, accounting for 0.04–0.2 % of all primary malignant tumors of the respiratory system. This carcinoma can occur at any age, but it is more common in the 40–60 age group and usually in women. The main treatment method for adenoid cystic carcinoma is surgical. Since tumors of this histological form are often centrally located, options for bronchoplastic operations are considered. In childhood, adenoid cystic carcinoma is extremely rare, and performing bronchoplastic lobectomies in children is associated with several difficulties, such as the smaller diameter of the bronchi compared to adult patients, complicating surgical intervention and subsequent rehabilitation. This clinical case demonstrates the experience of performing a bronchoplastic operation on a 15-year-old patient at the Department of Thoracic Oncology of the National Medical Research Centre for Oncology, Rostov-on-Don, Russian Federation. The patient was hospitalized complaining of prolonged cough, shortness of breath, and chest pain. Adenoid cystic carcinoma of the central type was identified during diagnostics, which included bronchoscopy, computed tomography, and biopsy. The surgical intervention involved performing a bronchoplastic lobectomy, during which the affected lobe of the lung was removed with resection and reconstruction of the bronchus. The operation was performed taking into account the anatomical features of the child's body, which required high precision and surgical skills. The postoperative period proceeded without significant complications, and the patient was under the supervision of a multidisciplinary team of specialists.

This clinical case provides a detailed description of the results of preoperative diagnostic measures, the stages of the operation, and the postoperative follow-up results. Special attention was paid to the difficulties associated with the small diameter of the bronchi in children, which required the use of specialized instruments and techniques. The importance of using modern diagnostic and treatment methods, as well as close interdisciplinary interaction, is emphasized for a successful treatment outcome.

The experience of performing such operations in childhood is extremely important for improving the quality and safety of surgical treatment of adenoid cystic carcinoma and other rare tumors in children. Further observations will be described in stages.

1. Kit OI, Kharagezov DA, Lazutin YuN, Mirzoyan EA, Leyman IA, Tikhonova SN. Sequential bronchoplastic lobectomies in complex treatment for synchronous bilateral multiple primary non-small cell lung cancer: a rare clinical case. South Russian Journal of Cancer. 2022;3(4):67–73. (In Russ.). https://doi.org/10.37748/2686-9039-2022-3-4-7, EDN: NIGIFS

2. Kharagezov DA, Lazutin YuN, Mirzoyan EA, Milakin AG, Stateshny ON, Leiman IA, et al. Клинический случай легочной формы мукормикоза у ребенка с острым лимфобластным лейкозом. South Russian Journal of Cancer. 2021;2(4):38–47. (In Russ.). https://doi.org/10.37748/2686-9039-2021-2-4-5, EDN: PLFQUY

3. Kharagezov DA, Lazutin YuN, Mirzoyan EA, Milakin AG, Stateshny ON, Leyman IA. Neoadjuvant and adjuvant immunotherapy of non-small cell lung cancer (literature review). Environmental issues. 2022;68(2):159–168. (In Russ.). https://doi.org/10.37469/0507-3758-2022-68-2-159-168, EDN: VYYGNS

4. Krifa M, Bdioui A, Lajmi Z, Missaoui N, Hmissa S, Mokni M. Primary adenoid cystic carcinoma of the lung: A case report and literature review. Heliyon. 2021 Feb;7(2):e06206. https://doi.org/10.1016/j.heliyon.2021.e06206

5. Li M, Zhao BR, Liu SQ, An J, Deng PB, Han-Zhang H, et al. Mutational landscape and clonal diversity of pulmonary adenoid cystic carcinoma. Cancer Biol Ther. 2018;19(10):898–903. https://doi.org/10.1080/15384047.2018.1480296

6. Elnayal A, Moran CA, Fox PS, Mawlawi O, Swisher SG, Marom EM. Primary salivary gland-type lung cancer: imaging and clinical predictors of outcome. AJR Am J Roentgenol. 2013 Jul;201(1):W57–63. https://doi.org/10.2214/AJR.12.9579

7. Yokouchi H, Otsuka Y, Otoguro Y, Takemoto N, Ito K, Uchida Y, et al. Primary peripheral adenoid cystic carcinoma of the lung and literature comparison of features. Intern Med. 2007;46(21):1799–1803. https://doi.org/10.2169/internalmedicine.46.0331

8. Grillo HC, Mathisen DJ. Primary tracheal tumors: treatment and results. Ann Thorac Surg. 1990 Jan;49(1):69–77. https://doi.org/10.1016/0003-4975(90)90358-D

9. Tang J, Cao M, Qian L, Fu Y, Tang J, Zhao X. The pure distal left main bronchial sleeve resection with total lung parenchymal preservation: report of two cases and literature review. J Thorac Dis. 2014 Dec;6(12):E294–298. https://doi.org/10.3978/j.issn.2072-1439.2014.12.14

10. Gaissert HA, Mathisen DJ, Grillo HC, Vacanti JP, Wain JC. Tracheobronchial sleeve resection in children and adolescents. J Pediatr Surg. 1994 Feb;29(2):192–197. https://doi.org/10.1016/0022-3468(94)90316-6

11. Kharagezov DA, Mirzoyan EA, Turkin IN, Lazutin YuN, Milakin AG, Stateshny ON, et al. Comparative analysis of the results of bronchoplastic surgical interventions and pneumonectomies. The journal Chief physician of the South of Russia. 2022;(1(82)):10–13. (In Russ.). EDN: HQJKHD

12. Presley CJ, Gross CP, Lilenbaum RC. Optimizing Treatment Risk and Benefit for Elderly Patients With Advanced Non-Small-Cell Lung Cancer: The Right Treatment for the Right Patient. J Clin Oncol. 2016 May 1;34(13):1438–1442. https://doi.org/10.1200/JCO.2015.65.9599

13. Tikhonova SN, Rozenko DA, Ushakova ND, Popova NN, Skopintsev AM, Shulga AV, et al. Optimization of anesthetic tactics in the surgical treatment of multiple primary non-small cell lung cancer. South Russian Journal of Cancer. 2021;2(2):42–49. https://doi.org/10.37748/2686-9039-2021-2-2-5, EDN: MGRWDV

14. Reshetov AV, Elkin AV, Nikolaev GV, Stepanov SS. Broncho- and angioplasty lobectomy as an alternative to pneumonectomy in treatment of non-small-cell lung cancer. Grekov's Bulletin of Surgery. 2018;177(3):19–24. (In Russ.). https://doi.org/10.24884/0042-4625-2018-177-3-19-24, EDN: XRSVVB

15. Levchenko EV, Levchenko NE, Yurin RI, Mikhnin AE, Ergnian SM, Barchuk AS. Long-term results of bronchoplastic surgical interventions compared with pneumonectomies in lung cancer. Questions of Oncology. 2017;63(2):234–239. (In Russ.). https://doi.org/10.37469/0507-3758-2017-63-2-234-239, EDN: YNCXYX

16. Li Z, Chen W, Xia M, Liu H, Liu Y, Inci I, et al. Sleeve lobectomy compared with pneumonectomy for operable centrally located non-small cell lung cancer: a meta-analysis. Transl Lung Cancer Res. 2019 Dec;8(6):775–786. https://doi.org/10.21037/tlcr.2019.10.11

17. Gaissert HA, Mathisen DJ, Grillo HC, Vacanti JP, Wain JC. Tracheobronchial sleeve resection in children and adolescents. J Pediatr Surg. 1994 Feb;29(2):192–197. https://doi.org/10.1016/0022-3468(94)90316-6

18. Yu Y, Song Z, Chen Z, Jian H, Lu S. Chinese pediatric and adolescent primary tracheobronchial tumors: a hospital-based study. Pediatr Surg Int. 2011 Jul;27(7):721–726. https://doi.org/10.1007/s00383-011-2858-8

19. Toker A, Bayrak Y, Dilege S, Kalayci G. Bronchial sleeve resections for carcinoid tumor in the first decade of life. Interact Cardiovasc Thorac Surg. 2004 Jun;3(2):280–282. https://doi.org/10.1016/j.icvts.2003.12.006