Pulmonary Langerhans Cell Histiocytosis Mimicking Metastasis of Cancer: Two Cases Report and Literature Review_Chinese Journal of Respiratory and Critical Care Medicine

Authors：

ZhaoTingting ¹ , XiaWei ² , ZhaoQi ¹ , MaMiao ¹ , LiYan ¹ , MiaoLiyun ¹ , SunQi ³ ,  ZhangYingwei ¹

1. Department of Respiratory Medicine, The Affiliated Drum Tower Hospital of Nanjing University, Nanjing, Jiangsu, 210008, China;
2. ;
3. ;

Corresponding author：

ZhangYingwei, Email: yingwei16@aliyun.com

Keywords：

Pulmonary Langerhans cell histiocytosis; Metastasis of cancer; Misdiagnosis; HRCT; PET/CT

DOI：

10.7507/1671-6205.2016059

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Objective To investigate the clinical, radiographic characteristics and differential diagnosis of pulmonary Langerhans cell histiocytosis (PLCH) mimicking metastasis of cancer in radiography. Methods Clinical data of 2 patients with PLCH manifesting as metastatic cancer on HRCT and PET/CT were retrospectively analyzed. Patients reported as PLCH on WanFang Database, China Knowledge Resource Integrated Database and Pubmed were reviewed to screen misdiagnosis literature and further analyzed the clinical and radiographic characteristics. Results Two cases both presented with cough and sputum. ¹⁸F-FDG PET/CT showed increased ¹⁸F-FDG up-take in both nodules in the lungs. One patient presented with multiple nodules, diffuse multiple cystic changes in lungs and osteoclasia in the right 4^th rib on HRCT who was diagnosed by a video-assisted thoracoscopic biopsy of rib biopsy. The other patient presented with diffuse multiple nodules on HRCT who was diagnosed by a video-assisted thoracoscopic biopsy of lung biopsy. The pathological characteristics of both biopsy specimen demonstrated infiltration by Langerhans cells (LC) and eosinophils. The LC were positive for CD1a. Literature review found seven PLCH cases who were misdignosed as depression, eosinophilic pneumonia, interstitial lung disease involvement of autoimmune disorders and malignant tumor. Conclusion When clinician faced with a patient suspected as metastatic cancer by HRCT and PET/CT, it is reasonable to consider PLCH as a differential diagnosis and obtain the pathological information as soon as possible so that better prognosis can be achieved through early intervention.

Citation： Zhao Tingting, Xia Wei, Zhao Qi, Ma Miao, Li Yan, Miao Liyun, Sun Qi, Zhang Yingwei. Pulmonary Langerhans Cell Histiocytosis Mimicking Metastasis of Cancer: Two Cases Report and Literature Review. Chinese Journal of Respiratory and Critical Care Medicine, 2016, 15(3): 250-255. doi: 10.7507/1671-6205.2016059 Copy

1.	Misaki H, Yamauchi T, Arai H, et al. Secondary malignant fibrous histiocytoma following refractory Langerhans cell histiocytosis. J Clin Exp Hematop, 2009, 49:33-37.
2.	Niksarlioglu YO, Gurel B, Tezel GG, et al. Langerhans' cell histiocytosis mimicking metastatic carcinoma of the lung. Respirology, 2009, 14:456-458.
3.	Feuillet S, Louis L, Bergeron A, et al. Pulmonary Langerhans cell histiocytosis associated with Hodgkin's lymphoma. Eur Respir Rev, 2010, 19:86-88.
4.	黄华萍, 李羲.朗罕细胞组织细胞增多症误诊1例.临床肺科杂志, 2009, 14:127-128.
5.	刘蔺, 陈磊, 王永生, 等.朗格汉斯组织细胞增多症误诊为抑郁症1例报告.临床肺科杂志, 2012, 17:2131.
6.	Song H, Lee D, Park SY, et al. Single pulmonary Langerhans cell histiocytosis mimicking metastasis in lung cancer: imaging with (18)F-FDG PET/CT. Nucl Med Mol Imaging, 2015, 49:243-245.
7.	González García A, Callejas Rubio JL, Ríos Fernández R, et al. Pulmonary Langerhans histiocytosis: an uncommon cause of interstitial pneumonia in a patient with Sj gren syndrome. Clin Rheumatol, 2016, 35:825-828.
8.	Brownlee NA, Mahar A, Sporn TA. Bilateral pulmonary nodules in a 37-year-old woman with malignant melanoma. Pulmonary Langerhans cell histiocytosis (eosinophilic granuloma). Arch Pathol Lab Med, 2005, 129:e136-e137.
9.	Szturz P, Adam Z, Rehák Z, et al. Lymphoma-like course in aggressive adult multisystem Langerhans cell histiocytosis and the benefit of PET/CT imaging in evaluation of diffuse metabolic activity of lung parenchyma. Vnitr Lek, 2010, 56:1177-1193.
10.	Suri HS, Yi ES, Nowakowski GS, et al. Pulmonary Langerhans cell histiocytosis. Orphanet J Rare Dis, 2012, 7:16.
11.	Bhardwaj H, Bhardwaj B. Differentiating pulmonary lymphangioleiomyomatosis from pulmonary Langerhans cell histiocytosis and Birt-Hogg-Dube syndrome. Lung India, 2013, 30:372-373.
12.	Krajicek BJ, Ryu JH, Hartman TE, et al. Abnormal fluorodeoxyglucose PET in pulmonary Langerhans cell histiocytosis. Chest, 2009, 135:1542-1549.
13.	Lee HJ, Ahn BC, Lee SW, et al. The usefulness of F-18 fluorodeoxyglucose positron emission tomography/computed tomography in patients with Langerhans cell histiocytosis. Ann Nucl Med, 2012, 26:730-737.
14.	Stacher E, Beham-Schmid C, Terpe HJ, et al. Pulmonary histiocytic sarcoma mimicking pulmonary Langerhans cell histiocytosis in a young adult presenting with spontaneous pneumothorax: a potential diagnostic pitfall. Virchows Arch, 2009, 455:187-190.
15.	Jülg BD, Weidner S, Mayr D, et al. Pulmonary manifestation of a Langerhans cell sarcoma: case report and review of the literature. Virchows Arch, 2006, 448:369-374.
16.	Ohtsuki Y, Uomoto M, Hachisuka Y, et al. A rare case of coexistence of pulmonary adenocarcinoma with Langerhans' cell histiocytosis. Med Mol Morphol, 2008, 41:175-178.
17.	Kaya A, Sava Y, Sen E, et al. Histiocytosis X and bronchopulmonary adenocarcinoma: a rare coexistence. Can Respir J, 2002, 9:431-432.
18.	Schreiber J, Waldburg N. FDG-PET/CT in pulmonary and cardiac Langerhans' cell histiocytosis and metastatic non-small cell lung cancer. Nucl Med Biol, 2012, 39:1173-1174.
19.	Sadoun D, Vaylet F, Valeyre D, et al. Bronchogenic carcinoma in patients with pulmonary histiocytosis X. Chest, 1992, 101:1610-1613.
20.	Lee JS, Ko GH, Kim HC, et al. Langerhans cell sarcoma arising from Langerhans cell histiocytosis: a case report. J Korean Med Sci, 2006, 21:577-580.
21.	Etienne B, Bertocchi M, Gamondes JP, et al. Relapsing pulmonary Langerhans cell histiocytosis after lung transplantation. Am J Respir Crit Care Med, 1998, 157:288-291.

1. Misaki H, Yamauchi T, Arai H, et al. Secondary malignant fibrous histiocytoma following refractory Langerhans cell histiocytosis. J Clin Exp Hematop, 2009, 49:33-37.
2. Niksarlioglu YO, Gurel B, Tezel GG, et al. Langerhans' cell histiocytosis mimicking metastatic carcinoma of the lung. Respirology, 2009, 14:456-458.
3. Feuillet S, Louis L, Bergeron A, et al. Pulmonary Langerhans cell histiocytosis associated with Hodgkin's lymphoma. Eur Respir Rev, 2010, 19:86-88.
4. 黄华萍, 李羲.朗罕细胞组织细胞增多症误诊1例.临床肺科杂志, 2009, 14:127-128.
5. 刘蔺, 陈磊, 王永生, 等.朗格汉斯组织细胞增多症误诊为抑郁症1例报告.临床肺科杂志, 2012, 17:2131.
6. Song H, Lee D, Park SY, et al. Single pulmonary Langerhans cell histiocytosis mimicking metastasis in lung cancer: imaging with (18)F-FDG PET/CT. Nucl Med Mol Imaging, 2015, 49:243-245.
7. González García A, Callejas Rubio JL, Ríos Fernández R, et al. Pulmonary Langerhans histiocytosis: an uncommon cause of interstitial pneumonia in a patient with Sj gren syndrome. Clin Rheumatol, 2016, 35:825-828.
8. Brownlee NA, Mahar A, Sporn TA. Bilateral pulmonary nodules in a 37-year-old woman with malignant melanoma. Pulmonary Langerhans cell histiocytosis (eosinophilic granuloma). Arch Pathol Lab Med, 2005, 129:e136-e137.
9. Szturz P, Adam Z, Rehák Z, et al. Lymphoma-like course in aggressive adult multisystem Langerhans cell histiocytosis and the benefit of PET/CT imaging in evaluation of diffuse metabolic activity of lung parenchyma. Vnitr Lek, 2010, 56:1177-1193.
10. Suri HS, Yi ES, Nowakowski GS, et al. Pulmonary Langerhans cell histiocytosis. Orphanet J Rare Dis, 2012, 7:16.
11. Bhardwaj H, Bhardwaj B. Differentiating pulmonary lymphangioleiomyomatosis from pulmonary Langerhans cell histiocytosis and Birt-Hogg-Dube syndrome. Lung India, 2013, 30:372-373.
12. Krajicek BJ, Ryu JH, Hartman TE, et al. Abnormal fluorodeoxyglucose PET in pulmonary Langerhans cell histiocytosis. Chest, 2009, 135:1542-1549.
13. Lee HJ, Ahn BC, Lee SW, et al. The usefulness of F-18 fluorodeoxyglucose positron emission tomography/computed tomography in patients with Langerhans cell histiocytosis. Ann Nucl Med, 2012, 26:730-737.
14. Stacher E, Beham-Schmid C, Terpe HJ, et al. Pulmonary histiocytic sarcoma mimicking pulmonary Langerhans cell histiocytosis in a young adult presenting with spontaneous pneumothorax: a potential diagnostic pitfall. Virchows Arch, 2009, 455:187-190.
15. Jülg BD, Weidner S, Mayr D, et al. Pulmonary manifestation of a Langerhans cell sarcoma: case report and review of the literature. Virchows Arch, 2006, 448:369-374.
16. Ohtsuki Y, Uomoto M, Hachisuka Y, et al. A rare case of coexistence of pulmonary adenocarcinoma with Langerhans' cell histiocytosis. Med Mol Morphol, 2008, 41:175-178.
17. Kaya A, Sava Y, Sen E, et al. Histiocytosis X and bronchopulmonary adenocarcinoma: a rare coexistence. Can Respir J, 2002, 9:431-432.
18. Schreiber J, Waldburg N. FDG-PET/CT in pulmonary and cardiac Langerhans' cell histiocytosis and metastatic non-small cell lung cancer. Nucl Med Biol, 2012, 39:1173-1174.
19. Sadoun D, Vaylet F, Valeyre D, et al. Bronchogenic carcinoma in patients with pulmonary histiocytosis X. Chest, 1992, 101:1610-1613.
20. Lee JS, Ko GH, Kim HC, et al. Langerhans cell sarcoma arising from Langerhans cell histiocytosis: a case report. J Korean Med Sci, 2006, 21:577-580.
21. Etienne B, Bertocchi M, Gamondes JP, et al. Relapsing pulmonary Langerhans cell histiocytosis after lung transplantation. Am J Respir Crit Care Med, 1998, 157:288-291.

Chinese Journal of Respiratory and Critical Care Medicine

Pulmonary Langerhans Cell Histiocytosis Mimicking Metastasis of Cancer: Two Cases Report and Literature Review

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