Authors
Bagirov M.A.1, 3, Chashchina M.V.1, Tarasov R.V.1, 2, 3, Sadovnikova S.S.1, Asoyan G.A.1, Khozikov A.S.1
1 Central Scientific Research Institute of Tuberculosis, Moscow
2 Moscow Medical University «REAVIZ», Moscow
3 Russian Medical Academy of Continuous Professional Education, Moscow
Abstract
Relevance: Cystic pulmonary hypoplasia (CPH), or congenital malformation of the respiratory tract of the lungs (RTL) accounts for more than half of all detected lung malformations in children and is often complicated by the addition of chronic infectious diseases of the respiratory system, including mycobacterial origin (tuberculosis, mycobacteriosis of the lungs). Diagnosis of both congenital lung defects and associated respiratory infection is often difficult.
Aim: To demonstrate an integrated approach to treatment and long-term results of a rare case of a combination of congenital malformation of the respiratory tract of the lungs (cystic hypoplasia of the lower lobe of the right lung) manifested in adolescence with mycobacteriosis of the lungs caused by M.kansasii.
Materials: the medical history of a teenage patient with a combination of mycobacterial infection and congenital malformation of the respiratory tract was studied.
Results: The patient underwent a video-assisted thoracoscopic (VATS) lower lobectomy of the right lung with removal of a conglomerate of bronchopulmonary lymph nodes. There were no complications during the operation or in the postoperative period. The drains were removed on the 6th day. Specific antimycobacterial therapy and courses of broad-spectrum antibiotics were performed before and after surgery. 7 months after the operation, a follow-up clinical and X-ray examination showed a favorable course of the long-term postoperative period and positive X-ray dynamics.
Conclusion: This clinical observation demonstrates the high effectiveness of treatment of a child with a complicated course of combined pathology «CPH+RTL» due to the use of an integrated approach – a course of specific etiotropic therapy and timely surgical intervention.
Keywords: cystic lung hypoplasia (clh), congenital malformation of the pulmonary airways (cmpa), non-tuberculous pulmonary disease (ntm-ld), mycobacterium kansasii, video-assisted thoracoscopic lung resection (vats), case report.
References
1. The WHO Practical Guide to Tuberculosis. Module 5. Management of tuberculosis in children and adolescents. Geneva: World Health Organization, 2022 License: CC BY-NC-SA 3.0 IGO. (In Russ.)
2. Hoefsloot W, van Ingen J, Andrejak C, et al. The geographic diversity of nontuberculous mycobacteria isolated from pulmonary samples: an NTM-NET collaborative study. Eur Respir J. 2013; 42(6): 1604-1613. doi: 10.1183/09031936.00149212.
3. Gubkina MF, Sterlikova SS, Petrakova IYu, et al. Cases of Mycobacteriosis with Intrathoracic Lymph Node Involvement in Infants. Tuberculosis and Lung Diseases. 2024; 102(5): 76-83. (In Russ.) doi: 10.58838/ 2075-1230-2024-102-5-76-83.
4. Volodich OS, Nuraliev SM, Kozak AR et al. Pulmonary disseminated mycobacteriosis combined with congenital lung malformation in a 15-year-old adolescent. Pediatria. 2018; 97(2): 213-217. (In Russ.)
5. Galimov OV, Khanov VO, Мukhamadyanov GS, et al. Successful Surgery of Severely Comorbid Cystic Pulmonary Hypoplasia. Creative Surgery and Oncology. 2020; 10(4): 291-295. (In Russ.) doi: 10.24060/2076-3093-2020-10-4-291-295.
6. Ovsyannikov DY, Frolov PA, Semenov PA. Congenital malformation of pulmonary respiratory tract. Pediatria. 2018; 97(1): 152-161. (In Russ.)
7. Stocker JT. Congenital Pulmonary Airway Malformation: a new name for and an expanded classification of congenital cystic adenomatoid malformation of the lung. Histopathology. 2002; 41(2): 424-430.
8. Chikkannaiah P, Kangle R, Hawal M. Congenital cystic adenomatoid malformation of lung: Report of two cases with review of literature. Lung India. 2013; 30(3): 215-18. doi: 10.4103/0970-2113.116272.
9. Isakov YuF, Volodin NN, Geraskin AV. Neonatal surgery. M.: Dynasty; 2011. Р.313-320. (In Russ.)
10. Tsai AY, Liechty KW, Hedrick HL, et al. Outcomes after postnatal resection of prenatally diagnosed asymptomatic cystic lung lesions. J. Pediatr. Surg. 2008; 43: 513-517.
11. Farrugia MK, Raza SA, Gould S, Lakhoo K. Congenital lung lesions: classification and concordance of radiological appearance and surgical pathology. Pediatr. Surg. Int. 2008; 24: 987-991.
12. Galiagina NA, Khaletskaia OV, Outcomes of congenital cystic lung malformations in children abd their management according to clinical progression. Modern Technologies in Medicine. 2014; 6(2): 77-84. (In Russ.)
13. Stashuk GA, Vishnyakova MV, Shcherbina VI, et al. Congenital adenomatoid lung type 1 malformation in a newborn. Almanac of Clinical Medicine. 2015; 43: 127-130. (In Russ.) doi: 10.18786/2072-0505-2015-43-127-130.
14. Bondarenko VN, Shtantze VA, Goponyako SV, et al. Clinical and radiological characteristics of mycobacteriosis of the lungs. Problems of health and ecology. Problems of health and ecology. 2017; 2(52): 38-43. (In Russ.)
