SPONTAN REGRESYON İZLENEN LANGERHANS HÜCRELİ HİSTİYOSİTOSİZ: OLGU SUNUMU

Year 2015, Volume: 29 Issue: 3, 155 - 159, 01.12.2015

Melike Yüksel Yavuz Ceyda Anar Erdem Yalçınkaya İpek Ünsal Filiz Güldaval Derya Deniz Hüseyin Halilçolar Zekiye Aydoğdu

Abstract

Pulmoner eozinofilik granülom olarak da bilinen Langerhans hücreli histiyositoz, pulmoner Histiyositoz- X sendromuna ait bir interstisyel akciğer hastalığıdır. Genç sigara içen hastalarda daha sık görülmektedir. Tanı klinik ve tipik radyolojik bulgularla konulabilse de esas tanı patolojik incelemeyle konulur. Tedavinin temelini sigarayı bıraktırma oluşturur. Beş aydır eforla artan nefes darlığı yakınması ile polikliniğimize başvuran 39 yaşında bayan hastanın 15 yıl bir paket/gün sigara içme öyküsü mevcuttu. Yüksek rezolüsyonlu toraks bilgisayarlı tomografisinde her iki akciğer parankim alanlarında üst ve orta zonlarda daha belirgin, alt zonlarda daha hafif ve küçük, kaviter ve kistik nodüler lezyonlar mevcuttu. Bronkoalveolar lavaj sıvısının immünohistokimyasal boyamasında S- 100 yüzey antijeni pozitifliği gösteren Langerhans hücreleri görüldü. Sadece akciğer tutulumu olan ve sigaranın bırakılması ile lezyonlarında gerileme görülen olgumuzu sunmayı uygun bulduk.

Keywords

Smoking, Langerhans Hücreli Histiyositosiz, treatment, spontan regression

SPONTANEOUS REMISSION IN LANGERHANS CELL HISTIOCYTOSIS: A CASE REPORT

Abstract

Langerhans Cell Histiocytosis, also known as pulmonary eosinophilic granuloma, is a part of the pulmonary histiyositozis-X syndrome and is a form of interstitial lung disease. It is common among young smoker patients. Although the diagnosis is based on the clinical and typical radiological findings, pathological examination is necessary for final diagnosis. Smoking cessation is main stay of treatment. A 39-year-old female patient referred to our clinic with the complaint of dispne for the last five months. She is a current smoker and had a smoking history of 15 packedyear. In high resolution computed tomography, cystic-cavitary appearances markedly in upper lobes were observed. Bronchoalveolar lavage showed some Langerhans cells with S-100 antigen positivity. We report a case of eosinophilic granuloma in whom spontaneous disease regression was obtained with only smoking cessation.

Keywords

Sigara, Langerhans Hücreli Histiyositosiz, tedavi, spontan regre

References

• Lanzkowsky P. Histiocytosis syndromes. In Manual of Pediatric Hematology and Oncology. 2nd ed. USA: Churchill Livingstone 1995;493- 511.
• Nicholson S.H, Egeler M, Nesbit M.E. The epidemiology of langerhans cell histiocytosis. Hematology/Oncology Clinics of North America 1998; 12(2): 379-84.
• Berna K, Enver Y, Melih B, Gökhan Y, Emel Ç, N ur Y. Spontaneous remission in pulmonary eosinophilic granuloma: a case report Eurasian J Pulmonol 2004; 6(2): 84-8.
• Von Essen S, West W, Sitorius M, Rennard SI. Complete resolution of roentgenographic changes in a patient with pulmonary histiocytosis X. Chest 1990; 98:765–7.
• Cheryl L. Willman, Kenneth L. McClain. An Updte On Clonality, Cytoknes and Viral Etıology in Langerhans Cell Histiocytosis. Hematology/ Oncology Clinics of North America 1998; 12:407-16.
• Badalian-Very G1, Vergilio JA, Fleming M, Rollins BJ. Pathogenesis of Langerhans Cell Histiocytosis. Annu Rev Pathol 2012;24:1-20.
• Fuks L, Kramer MR, Shitrit D, Raviv Y. Pulmonary Langerhans cell histiocytosis and diabetes insipidus in pregnant women: our experience. Lung 2014;192(2):285-7.
• Brauner MW, Grenier P, Tijani K, Battesti JP, Valeyre D. Pulmonary Langerhans cell histiocytosis: Evaluation of lesions on CT scans. Thoracic Radiology 1997;204:497-502.
• Erkan A, Ersin A, Sibel A, Maruf Ş, Kemal B, Levent E. Spontan Pnömotoraksla Seyreden Pulmoner Langerhans Hücreli Histiyositozis: Olgu sunumu. Solunum Hastalıkları Cilt 22, Sayı 2, 2011,63-5.
• Tazi A. Adult pulmonary Langerhans' cell histiocytosis. Eur Respir J 2006;27:1272-85.
• Tazi A, Soler P, Hance A J. Pulmonary Langerhans’ cell histiocytosis. Eur Respir Mon 2000;14:181-93.
• Abi Khalil S, Gourdier AL, Aoun N, Nedelcu C, El Rai S, Moubarak E, Sfeir S, Willoteaux S, Aubé C. Cystic and cavitary lesions of the lung: imaging characteristics and differential diagnosis. J Radiol 2010; 91: 465-73.
• Johnson S. Lymphangioleiomyomatosis: clinical features, management and basic mechanisms. Thorax1999;54:254-26414.
• Kumar N, Sayed S, Vinayak S. Diagnosis of Langerhans cell histiocytosis on fine needle aspiration cytology: a case report and review of the cytology literature. Patholog Res Int 2011; 20:439-518.
• Housini I, Tomashefski JF Jr, Cohen A, Crass J, Cohen A, Crass J, Kleinerman JF. Transbronchial biopsy in patients with pulmonary eosinophilic granuloma. Comparison with findings on open lung biopsy ArchPathol Lab Med 1994; 118: 523-30.
• Şule A, Füsun Öner E, Ali, Beyhan D. Effect of pulse steroid therapy in a patient with Langerhans’ cell histiocytosis. Respirology 2001;6:357-60.
• Ertürk A, Ünsal E, Gülhan M, Canbakan S, Güler M, Çakır E, Gülhan EŞ, Çapan N. A case of pulmonary langerhans’ cell histiocytosis: improved pulmonary lesions after smoking cessation in a female patient.Eurasian J Pulmonol 2007; 9(2):105-9.
• Tazi A, Montcelly L, Bergeron A, Valeyre D, Battesti JP, Hance AJ. Relapsing nodular lesions in the course of adult pulmonary Langerhans cell histiocytosis. Am J Res-pir Crit Care Med 1998; 157:2007-10.
• Bénédicte E, Michèle B, Jean-Paul G, Fabrice T, Christophe B, Thérèse W, Robert L, Jean B, and Jean-François M. Relapsing Pulmonary Langerhans Cell Histiocytosis after Lung Transplantation. American Journal of Respiratory and Critical Care Medicine, 1998; 157 (1): 288-91.
• Ji Won L, Hee Young S, Hyoung Jin K, Hyery Kim June Dong P, Kyung Duk P, Han-Soo K, Sung-Hye P,Kyu-Chang W and Hyo Seop A. Clinical Characteristics and Treatment Outcome of Langerhans Cell Histiocytosis: 22 Years’Experience of 154 Patients at a Single Center Pediatric Hematology and Oncology 2014;31:293-302.