Localized Synchronous Pulmonary Langerhans Cell Sarcoma and Langerhans Cell Histiocytosis

A synchronous presentation of pulmonary lesions Langerhans cell sarcoma (LCS) and histiocytosis (LCH) is extremely rare. We report the case a 57-year-old man with 3 nodules, 1 in left lung 2 right lung. In 2-stage operation, he first underwent segmentectomy, nodule was diagnosed as LCS. Thereafter, nodules disappeared. The residual resected by segmentectomy LCH. This complete resection localized, LCS whom LCH manifested simultaneously lungs, respectively, were completely resected. 37-pack-year active smoking history referred to our hospital an abnormal opacity on his chest radiograph. Chest computed tomography (CT) revealed 23-mm upper lobe, 20-mm lower 6-mm cavitating middle lobe (Figure 1a). 18F-Fluorodeoxyglucose positron emission tomography/CT (FDG PET/CT) significant uptake (maximum standardized value [SUVmax], 4.8), (SUVmax, 2.9), hilar lymph nodes 2.6; Figures 1b, 1c). Bronchoscopic findings suggestive adenocarcinoma. operation planned for (cT1 cN1 M0, c-stage ⅡB, TNM classification, 8th edition) followed (cT1b N0 ⅠA2 cT1a ⅠA1). His preoperative function test showed obstructive ventilatory defect (forced expiratory volume second, 3200 mL; forced second/forced vital capacity ratio, 67.25%; capacity, 132.5% predicted; diffusing carbon monoxide, 76.2%). intraoperative confirmed that tumor infiltrating across fissure. high FDG PET/CT enlarged had infiltrated into artery from A5 A8. As we could not separate these artery, performed lingulectomy S8 segmentectomy. size 38 × 23 12 mm. postoperative pathologic diagnosis node metastasis; histopathologic examination proliferation cells acidophilic granular cytoplasm rumpled tissue nuclei kidney bean–shaped 2a). dense cellularity. On immunohistochemical analysis, positive S100, CD1a (Figures 2b, 2c), CD163, MIB-1 labeling index 30% 40% 3b ). Mitotic activity 15 22 mitotic figures per 10 high-power fields; however, region mass 2d).Figure 3(A) Hematoxylin eosin staining nodule. It shows minimal cellular atypia. (B, C) Comparison nodules. (B) (C) 12%.View Large Image Figure ViewerDownload Hi-res image Download (PPT) Two months after initial follow-up CT changed size, whereas Bone scintigraphy bone marrow aspiration did reveal patient thoracoscopic S6 lobe. 21 17 13 3a). low (<1 figure fields). CD1a, 12% 3c). are antigen-presenting dendritic cells. Tumors derived rare divided subgroups, LCS, according World Health Organization classification.1Yang C.J. Lee J.Y. Wu C.C. Yin H.L. Lien C.T. Liu Y.C. An unusual mediastinal invasion multiple intrapulmonary 52-year-old man.Chest. 2012; 141: 253-258Abstract Full Text PDF PubMed Scopus (10) Google Scholar benign clonal cells, neoplastic histiocytosis, which clearly exhibit malignant characteristics. particular multisystem diseases. Lesions found bones, pituitary gland, thyroid nodes, skin.1Yang generally reveals cysts micronodules <10 mm predominantly lobes.2Suri H.S. Yi E.S. Nowakowski G.S. Vassallo R. Pulmonary histiocytosis.Orphanet J Rare Dis. 7: 16Crossref (118) localized >20 mm, case, larger than those usually bilateral.1Yang Scholar,3Lee J.S. Ko G.H. Kim H.C. Jang I.S. Jeon K.N. J.H. arising histiocytosis: report.J Korean Med Sci. 2006; 21: 577-580Crossref (55) reported both LCS.2Suri Scholar,4Chen Y.W. Chang Hou P.N. S.L. Lai M.F. characteristics imaging sarcoma.Clin Nucl Med. 37: 495-497Crossref (6) difficult distinguish between them findings. Therefore, biopsies involving surgical procedure or bronchoscopy required accurate be made. primary lesion often organs other such skin nodes. need consider biopsy specimens cases. Treatment strategies have yet been established. related smoking, early cessation important treatment.2Suri disappeared cessation. relationship clear. Patients diseases received chemotherapy.5Lauritzen A.F. Delsol G. Hansen N.E. et al.Histiocytic sarcomas monoblastic leukemias. clinical, histologic, immunophenotypical study.Am Clin Pathol. 1994; 102: 45-54Crossref (69) no chemotherapy because Five cases at reported.1Yang Scholar,5Lauritzen Scholar,6Julg B.D. Weidner S. Mayr D. manifestation sarcoma: review literature.Virchows Arch. 448: 369-374Crossref (37) only cases, including ours, isolated seen.3Lee patients treatment. coworkers3Lee lobectomy 4-cm tended grow; multiple, small, bilateral seen. addition, same Considering systemic diseases, close manifestations needed regardless authors wish thank Dr Keiichiro Honma diagnosis. funding sources disclose.