A rare histopathological fi nding after lung resection in a child

Czech version

Authors: V. Chrenkova ¹; P. Omaník ¹; L. Plank ²; J. Babala ¹
Authors‘ workplace: Klinika detskej chirurgie, Národný ústav detských chorôb, Bratislava, Slovenská republika ¹; Ústav patologickej anatómie, JLF UK, Martin, Slovenská republika ²
Published in: Klin Onkol 2021; 34(2): 142-146
Category: Case Report
doi: https://doi.org/10.48095/ccko2021142

Overview

Background: The authors present a case of a patient with an extremely rare lung tumor in a child. Case: A 9-year-old girl with a 3-day history of dyspnea and stabbing pain in the xiphoid region, irradiating to the area under the left costal margin, both in rest and in physical activities. She was primarily examined in a regional hospital with bounded homogenous focus of the superior right lung lobe on the X-ray. After initial treatment with antibiotics and persistent finding on X-ray, a CT scan of the chest was performed. It revealed an irregular oval lesion of a non-homogenous structure with a contrast dye in the S3 region merged to pericardium and parietal pleura, which the presence of several micronodules. Based on the negative tumor markers, positive PET-CT scan and a negative etiology, biopsy or eventually a lesion exstirpation were indicated. Right-sided thoracotomy, mass enucleation and exstirpation of nodular lesions were performed 2.5 months after the onset of difficulties. Postoperative recovery was uneventful, no sign of recurrence occurred during a follow-up period. The final histological finding was verified as an inflammatory myofibroblastic tumor – an extremely rare pulmonary pathology in the pediatric population. Conclusion: Inflammatory myofibroblastic tumor can be mimicking IgG4 sclerosing disease and inflammatory pseudotumor. It is essential to distinguish between these affections because of different (i.e. surgical vs. conservative) treatment approach.

Keywords:

inflammatory myofibroblastic tumor – inflammatory pseudotumor – IgG4 – lung tumor

Sources

1. Liu L, Kong X, Lu X, Cao D. Pediatric endobronchial inflammatory myofibroblastic tumor: a case report and review of literature. Clin Pract 2016; 6 (4): 853. doi: 10.4081/cp.2016.853.

2. Soyer T, Talim B, Karnak I et al. Surgical treatment of childhood inflammatory myofibroblastic tumors. Eur J Pediatr Surg 2017; 27 (4): 319–323. doi: 10.1055/s-0036-1593380.

3. Camela F, Gallucci M, di Palmo E et al. Pulmonary inflammatory myofibroblastic tumor in children: a case report and brief review of literature. Front Pediatr 2018; 6: 35. doi: 10.3389/fped.2018.00035.

4. Dalton BGA, Thomas PS, Sharp NE et al. Inflammatory myofibroblastic tumors in children. J Pediatr Surg 2015; 51 (4): 541–544. doi: 10.1016/j.jpedsurg.2015.11.015.

5. Gleason BC, Hornick JL. Inflammatory myofibroblastic tumour: where are we now? J Clin Pathol 2008; 61 (4): 428–437. doi: 10.1136/jcp.2007.049387.

6. Štiková Z, Ptáková N, Horáková M et al. Inflamatorní myofibroblastický tumor dělohy – kazuistika. Cesk Patol 2019; 55 (4): 239–243.

7. Siminovich M, Galluzzo L, López J et al. Inflammatory myofibroblastic tumor of the lung in children: anaplastic lymphoma kinase (ALK) expression and clinico-pathological correlation. Pediatr Dev Pathol 2012; 15 (3): 179–186. doi: 10.2350/11-10-1105-OA.1.

8. SurabhiVR, Chua S, Patel RP. Inflammatory myofibroblastic tumors: current update. Radiol Clin North Am 2016; 54 (3): 553–563. doi: 10.1016/j.rcl.2015.12.005.

9. Pio L, Varela P, Eliott MJ et al. Pediatric airway tumors: a report from the international network of pediatric airway teams (INPAT). Laryngoscope 2019; 130 (4): E243–E251. doi: 10.1002/lary.28062.

10. Giuseppucci C, Reusmann A, Giubergia V et al. Primary lung tumors in children: 24 years of experience at a referral center. Pediatr Surg Int 2016; 32 (5): 451–457. doi: 10.1007/s00383-016-3884-3.

11. Fabre D, Fadel E, Singhal S et al. Complete resection of pulmonary inflammatory pseudotumors has excellent long-term prognosis. J Thorac Cardiovasc Surg 2009; 137 (2): 435–440. doi: 10.1016/j.jtcvs.2008.07.009.

12. Oguz B, Ozcan HN, Omay B et al. Imaging of childhood inflammatory myofibroblastic tumor. Pediatr Radiol 2015; 45 (11): 1672–1681. doi: 10.1007/s00247-015-3377-x.

13. Pešek M. Nová WHO klasifikace nádorů plic. Onkologie 2016; 10 (1): 20–24.

14. Bhagat P, Bal A, Das A et al. Pulmonary inflammatory myofibroblastic tumor and IgG4-related inflammatory pseudotumor: a diagnostic dilemma. Virchows Arch 2013; 463 (6): 743–747. doi: 10.1007/s00428-013-1493-2.

15. Karnak BI, Șenocak ME, Ciftci AO et al. Inflammatory myofibroblastic tumor in children: diagnosis and treatment. J Pediatr Surg 2001; 36 (6): 908–912. doi: 10.1053/jpsu.2001.23970.

16. Saab ST, Hornick JL, Fletcher CD et al. IgG4 plasma cells in inflammatory myofibroblastic tumor: inflammatory marker or pathogenic link? Mod Pathol 2011; 27 (7): 606–612. doi: 10.1038/modpathol.2010.226.

17. Chun YS, Wang L, Nascimento AG et al. Pediatric inflammatory myofibroblastic tumor: anaplastic lymphoma kinase (ALK) expression and prognosis. Pediatr Blood Cancer 2005; 45 (6): 796-801. doi: 10.1002/pbc.20294.

18. Coffin CM, Patel A, Perkins S et al. ALK1 and p80 expression and chromosomal rearrangements involving 2p23 in inflammatory myofibroblastic tumor. Mod Pathol 2001; 14 (6): 569–576. doi: 10.1038/modpathol.3880352.

19. Watanabe H, Yamasaki N, Miyazaki T et al. Successful treatment based on molecular biological assessment of invasive anaplastic lymphoma kinase-positive inflammatory myofibroblastic tumor of the lung. Surg Case Rep 2019; 5 (1): 118. doi: 10.1186/s40792-019-0674-x.

20. Coffin CM, Hornick JL, Fletcher CD. Inflammatory myofibroblastic tumor: comparison of clinicopathologic, histologic, ad immunohistochemical features including ALK expression in atypical and aggressive case. Am J Surg Pathol 2007; 31 (4): 509–520. doi: 10.1097/01.pas.0000213393.57322.c7.

21. Adamkov J, Kašparová P, Česák T. Zánětlivý pseudotumor imitující intrakraniální, konvexitární meningeom – kazuistika. Cesk Slov Neurol N 2015; 78/111 (2): 233–236.

22. Butrynski JE, D’Adamo DR, Hornick JL et al. Crizotinib in ALK-rearranged inflammatory myofibroblastic tumor. N Engl J Med 2010; 363 (18): 1727–1733. doi: 10.1056/NEJMoa1007056.