Surgical aspects of treating a child with histiocytosis of the lungs

Cover Page
  • Authors: Stalmakhovich V.N.1, Sapukhin E.V.2, Li I.B.2, Kaygorodova I.N.2, Dmitrienko A.P.2, Ursulenko E.V.2, Dyukov A.A.2
  • Affiliations:
    1. Irkutsk State Medical Academy of Postgraduate Education (ISMAPgE – Branch Campus of the FSBEI FPE RMACPE MOH Russia)
    2. Irkutsk State Regional Children’s Clinical Hospital
  • Issue: Vol 9, No 2 (2019)
  • Pages: 84-90
  • Section: CLINICAL OBSERVATIONS
  • URL: https://rps-journal.ru/jour/article/view/551
  • DOI: https://doi.org/10.30946/2219-4061-2019-9-2-84-90

Abstract


Objective: to present a case of successful treatment of rare severe lung lesions in a child.

Methods: the article analyzes the result of the treatment of lung histiocytosis in a child aged 4 years. The clinical manifestation of the disease began with an increase in the parotid lymph nodes, otitis, pneumonia, and rapidly progressive respiratory failure. On the roentgenogram, bulle transformation of both lungs was noted. The severity of the disease was complicated by bilateral pneumothorax, the formation of bronchopleural fistulas. Histiocytosis of the lungs from Langerhans cells (GCR) was determined morphologically when examining tissue taken at thoracoscopy. Endoscopically, the lung was without signs of inflammation, with multiple subpleural air cavities. The child underwent chemotherapy (vinblastine, prednisone), repeated drainage of the pleural cavity with recurrent intense pneumothorax. For health reasons, a longitudinal sternotomy was performed, total decortication of the costal pleura on both sides (mechanical pleurodesis), which contributed to subsequent fixation of the lung to the chest wall and prevention of recurrent total stressed pneumothorax. Twice developed limited pneumothorax was stopped by drainage and chemical pleurodesis.

Results: Against the background of ongoing complex therapy (drainage of the pleural cavity, mechanical and chemical pleurodesis, chemo- and antibacterial therapy), the condition improved (clinically and radiographically). The child was discharged on day 152 of treatment. On the control MSCT of the lungs, eight months after discharge, there are minimal signs of bullous transformation, and the symptoms of pneumosclerosis of both lungs are moderate. There are no manifestations of pulmonary hypertension.

Conclusion: verification of the diagnosis of pulmonary GLC is very difficult. The complex treatment requires the participation of a wide range of doctors, including a pediatric surgeon.


About the authors

Viktor N. Stalmakhovich

Irkutsk State Medical Academy of Postgraduate Education (ISMAPgE – Branch Campus of the FSBEI FPE RMACPE MOH Russia)

Author for correspondence.
Email: stal.irk@mail.ru
ORCID iD: 0000-0002-4885-123X

Russian Federation

Dr. Sci (Med), Professor, head of the Department of Pediatric Surgery, IGMAPO

Microdistrict Jubilee, 100, Irkutsk, 664033; phone: +7(902) 176-17-82

eLirary SPIN: 9042-5092

Eduard V. Sapukhin

Irkutsk State Regional Children’s Clinical Hospital

Email: sapukhin@yandex.ru
ORCID iD: 0000-0001-5470-7384

Russian Federation

Deputy Chief Surgery Doctor, Endoscopist

b. Gagarin 4, Irkutsk, 664022; phone: +7(902)767-84-75

Igor B. Li

Irkutsk State Regional Children’s Clinical Hospital

Email: tokio197@mail.ru
ORCID iD: 0000-0003-1979-4616

Russian Federation

Head of the operating unit, the doctor the children’s surgeon of surgical department

b. Gagarin 4, Irkutsk, 664022; тел. +7(924)602-08-03

Irina N. Kaygorodova

Irkutsk State Regional Children’s Clinical Hospital

Email: kaygorodova_ir@mail.ru
ORCID iD: 0000-0002-2332-9285

Russian Federation

Cand. Sci (Med), head of the surgical Department

b. Gagarin 4, Irkutsk, 664022; phone: +7(914)871-84-03

Anastasia P. Dmitrienko

Irkutsk State Regional Children’s Clinical Hospital

Email: AnDmitr2013@yandex.ru
ORCID iD: 0000-0002-0003-8792

Russian Federation

Cand. Sci (Med), head Department of endoscopy

b. Gagarin 4, Irkutsk, 664022; phone: +7(902)561-66-72

eLirary SPIN: 3415-9266

Elena V. Ursulenko

Irkutsk State Regional Children’s Clinical Hospital

Email: irk.oncogem@mail.ru
ORCID iD: 0000-0002-5987-2343

Russian Federation

Hematologist, Department of Oncology

b. Gagarin 4, Irkutsk, 664022; phone: +7 (3952) 24-15-82

Andrey A. Dyukov

Irkutsk State Regional Children’s Clinical Hospital

Email: duk@mail.ru
ORCID iD: 0000-0001-6007-1298

Russian Federation

Cand. Sci (Med), head of the Department of purulent surgery

Phone: +7(950)125-53-56

References

  1. Степанян И. Э. Поражения легких при гистиоцитозах. Практическая пульмонология. 2011; (1): 59-63
  2. Ханин А. Л., Викторова И. Б., Николаева Л. П., Головнин В. И. Три случая лангергансоклеточного гистиоцитоза легких. Медицина в Кузбассе. 2012; 11(1): 4-10
  3. Allen C. E., Merad M., McClain K. L. Langerhans-Cell Histiocytosis. The New England journal of medicine. 2018; 379 (9): 856-68. doi: 10.1056/NEJMra1607548
  4. Фролова И. Г., Величко С. А., Гольдберг А. В., Тузиков С. А., Миллер С. В., Родионов Е. О., Евтеев Л. А., Бондарь Л. Н., Перелмутер В. М. Пульмональный лангергансоклеточный гистиоцитоз легких: клиническое наблюдение в стадии раннего поражения. Бюллетень сибирской медицины. 2017; 16(2):195-201. doi: 10.20538/1682-0363-2017-2-195-201
  5. Детская гематология. Клинические рекомендации. Под ред. А. Г. Румянцева, А. А. Масчана, Е. В. Жуковской. - М.: ГЭОТАР-Медия, 2015. - 62 с.
  6. Дорош О. И., Цымбалюк-Волошин И.П., Полищук Р. С., Дубей Л. Я., Воробель О. И. Козлова О. И., Трояновская О. О., Степанюк О. И., Скоропад Л. Л., Кицера Н. И., Середич Л. П., Мих А. М., Грищук Н. Б., Кузьменко А. И. Гистиоцитоз из клеток Лангерганса: особенности клинико-лабораторных проявлений и течения болезни. Здоровье ребёнка. 2014; 5 (56): 40-50
  7. Юсупова Л. А., Юнусова Е. И., Гараева З. Ш., Мавлютова Г. И. Гистиоцитоз X. Практическая медицина. 2014; 84(8): 7-11.
  8. Aricò M. Langerhans cell histiocytosis in children: from the bench to bedside for an updated therapy. British journal of haematology. 2016; 173(5): 663-70. doi: 10.1111/bjh.13955. https://onlinelibrary.wiley.com/doi/full/10.1111/bjh.13955
  9. Krooks J., Minkov M., Weatherall A. G. Langerhans cell histiocytosis in children: diagnosis, differential diagnosis, treatment, sequelae, and standardized follow-up. Journal of the American Academy of Dermatology. 2018; 78(6): 1047-56. doi: 10.1016/j.jaad.2017.05.060.
  10. Jezierska M., Stefanowicz J., Romanowicz G., Kosiak W., Lange M. Langerhans cell histiocytosis in children - a disease with many faces. Recent advances in pathogenesis, diagnostic examinations and treatment. Postȩpy dermatologii i alergologii. 2018; 35(1): 6-17. doi: 10.5114/pdia.2017.67095. Epub 2018 Feb 20. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5872238/
  11. Asilsoy S., Yazici N., Demir S., Erbay A., Koçer Е., Sarıalioğlu F. A different cause for respiratory disorder in children: cases with pulmonary Langerhans cell histiocytosis. The clinical respiratory journal. 2017; 11(2):193-9. doi: 10.1111/crj.12324.
  12. Brown N. A., Elenitoba-Johnson K.S.J. Clinical implications of oncogenic mutations in pulmonary Langerhans cell histiocytosis. Current opinion in pulmonary medicine. 2018; 24(3): 281-6. doi: 10.1097/MCP.0000000000000470.
  13. Çıtak E. Ç., Ak E., Sağcan F., Balcı Y., Bozdoğan-Arpacı R, Kuyucu N. Primary pulmonary Langerhans cell histiocytosis associated with smoking in an adolescent boy. The Turkish journal of pediatrics. 2017; 59(5): 586-9. doi: 10.24953/turkjped.2017.05.013. http://www.turkishjournalpediatrics.org/uploads/pdf_TJP_1769.pdf
  14. Halder R., Anantharaman M. B., Seth T., Mahapatra M., Saxena R. Pulmonary Langerhans cell histiocytosis: a not so benign lesion. Journal of pediatric hematology/oncology. 2019; 41(2):122-4. doi: 10.1097/MPH.0000000000001371.
  15. Kanik-Yuksek S., Ozkaya-Parlakay A., Gulhan B., Ozyoruk D., Karakus E., Cinel G., Emir S., Tezer H. A rare diagnosis in children: isolated pulmonary Langerhans cell histiocytosis. The clinical respiratory journal. 2018; 12(1): 355-6. doi: 10.1111/crj.12480.
  16. Wang D., Cui L., Li Z. G., Zhang L, Lian H. Y., Ma H. H., Zhao Y. Z., Zhao X.X, Wang T. Y., Zhang R. Clinical research of pulmonary langerhans cell histiocytosis in children. Chinese medical journal. 2018; 131(15): 1793-8. doi: 10.4103/0366-6999.237400. https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6071458/

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