Journal of Cytology
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IMAGES IN CYTOPATHOLOGY  
Year : 2019  |  Volume : 36  |  Issue : 2  |  Page : 131-132
Cytologic features of pulmonary langerhans cell histiocytosis in bronchial washing: A report of two cases


1 Department of Pathology, Gil Medical Center, Gachon University College of Medicine, Incheon, Republic of Korea
2 Department of Pathology, Dong-A University College of Medicine, Busan, Republic of Korea

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Date of Web Publication8-Mar-2019
 

How to cite this article:
Kang M, Ha SY, Roh MS. Cytologic features of pulmonary langerhans cell histiocytosis in bronchial washing: A report of two cases. J Cytol 2019;36:131-2

How to cite this URL:
Kang M, Ha SY, Roh MS. Cytologic features of pulmonary langerhans cell histiocytosis in bronchial washing: A report of two cases. J Cytol [serial online] 2019 [cited 2019 Sep 19];36:131-2. Available from: http://www.jcytol.org/text.asp?2019/36/2/131/251458




Pulmonary Langerhans cell histiocytosis (PLCH) is a rare disease characterized by the proliferation of CD1a-positive Langerhans cells. In most cases, lung biopsy is performed for diagnosis.[1] However, bronchioalveolar lavage (BAL), a less invasive diagnostic tool, is a possible diagnostic method for PLCH. Here, we report the cytologic features of two cases of PLCH in bronchial washing specimen.

The first case was a 55-year-old male patient who was admitted to our hospital for cough, myalgia, and febrile sensation. He was a 30 pack-years current smoker. He had a diagnosis of pulmonary tuberculosis 15 years ago. Chest X-ray and computed tomography (CT) revealed multiple air cysts in both lungs with predilection for mid and upper zones [Figure 1]a. He underwent bronchoscopy with bronchial washing cytologic examination. The smear of bronchial washing showed high cellularity. Most cell types were histiocytes, with indistinct cell borders having abundant cytoplasm, irregular nuclear contour, and grooves. These cells were accompanied by lymphocytes, alveolar macrophages, and multinucleated giant cells. No eosinophils were found. On immunocytochemical stains, 8% of cells were positive for CD1a (MTB1, 1:100; Novocastra, Newcastle, UK) and S-100 protein (polyclonal, 1:600; Dako, Glostrup, Denmark) [Figure 1]b and [Figure 1]c. The second case was a 48-year-old female patient who visited our hospital for 5-month exertional dyspnea. She was a 10 pack-years current smoker, with a history of central diabetes insipidus and hypertension. Chest CT revealed bilateral numerous thin-walled cystic lesions, mainly on the upper lobes. According to bronchoscopic examination, there was no endobronchial lesion. Bronchial washing showed many histiocytes, multinucleated giant cells, and rarely atypical cells with nuclear grooves. By immunocytochemistry, a few cells were positive for S-100 protein. Thoracoscopic wedge resection was performed for lung biopsy. Many CD1a-positive Langerhans cells were noted in the resected specimen [Figure 1]d. Approval was obtained from the Institutional Review Board (No. GCIRB2018-110) of our hospital.
Figure 1: Pulmonary Langerhans cell histiocytosis. (a) Multiple cysts on chest computed tomography. (b) Hypercellular smear (Papanicolau stain, ×400). (c) Irregular nuclear groove with abundant cytoplasm of Langerhans cells (Papanicolau stain, ×1000) (insert. CD1a). (d) Infiltration of Langerhans cells with lymphocytes (insert, CD1a)

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PLCH is related to cigarette smoke. In typical clinical settings and radiologic findings, the diagnosis of PLCH is established without biopsy. However, many atypical cases require further evaluation for differential diagnosis.[1] BAL is regarded as a diagnostic tool in some literatures.[2] The presence of more than 5% of CD1a-positive cells in BAL fluid defines PLCH.[2] In our first case, the diagnosis of PLCH was possible from following considerations: history of smoking, 8% of CD1a-positive cells in BAL fluid analysis, and multicystic lesions on CT.

The histologic findings of PLCH are characterized by granulomatous nodules with prominent inflammation composed of Langerhans cells and eosinophils. Langerhans cells have irregular convoluted nuclei with occasional nuclear grooves and are positive for CD1a and S-100 protein.[1] There were many Langerhans cells and lymphocytes in both cases. However, eosinophils or neutrophils were not noted. The eosinophil rates in BAL were varied from 1% to 16% in previous reports.[3] In our cases, no eosinophils were found in BAL analysis. These findings might be correlated with multiple cysts on radiologic examination. Cysts and fibrous scars are formed with the lack of Langerhans cells in the advanced stage.

The histiocyte-rich lesions in bronchial washing are diverse. These include common and rare diseases, such as pneumonia, interstitial lung disease, malignancy, Rosai–Dorfman disease, and Erdheim–Chester disease. However, in most cases, it may not be difficult to make the diagnosis using clinicoradiologic features. From our experience, histiocytes in pneumonia or asbestosis were similar with Langerhans cells in bronchial washing [Figure 2]a,[Figure 2]b,[Figure 2]c. Therefore, immunocytochemical stain may be necessary in cases with suspicion of PLCH.
Figure 2: Comparision of histiocytes in bronchial washing. (a) Pulmonary Langerhans cell histiocytosis. (b) Pneumonia. (c) Asbestosis

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In this report, we described the bronchial washing cytologic features of PLCH. Although fine needle aspiration cytologic features have been reported in some literatures,[4],[5] bronchial washing specimens are not well noted. When many histiocytes having convoluted nuclei with abundant cytoplasm were noted in bronchial washing, it is important to consider the possibility of PLCH. Even without eosinophils, additional immunocytochemical stain of Langerhans cells may be necessary.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Roden AC, Yi ES. Pulmonary langerhans cell histiocytosis: An update from the pathologists' perspective. Arch Pathol Lab Med 2016;140:230-40.  Back to cited text no. 1
    
2.
Auerswald U, Barth J, Magnussen H. Value of CD-1-positive cells in bronchoalveolar lavage fluid for the diagnosis of pulmonary histiocytosis X. Lung 1991;169:305-9.  Back to cited text no. 2
    
3.
Kim T, Kwon HJ, Eom M, Kim SW, Sin MH, Jung SH, et al. Bronchial washing cytology of pulmonary langerhans cell histiocytosis: A case report. J Pathol Transl Med 2017;51:444-7.  Back to cited text no. 3
    
4.
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;2011:439518.  Back to cited text no. 4
    
5.
Hang JF, Siddiqui MT, Ali SZ. Fine needle aspiration of langerhans cell histiocytosis: A Cytopathologic study of 37 cases. Acta Cytol 2017;61:96-102.  Back to cited text no. 5
    

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Correspondence Address:
Dr. Seung Yeon Ha
21565, Gachon University Gil Medical Center, 21 Namdongdaero 774 Beon-gil, Namdong-gu, Incheon
Republic of Korea
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JOC.JOC_80_18

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