PMID- 33140265 OWN - NLM STAT- MEDLINE DCOM- 20220117 LR - 20220117 IS - 1936-0568 (Electronic) IS - 1936-055X (Print) IS - 1936-055X (Linking) VI - 15 IP - 3 DP - 2021 Sep TI - Epstein-Barr Virus-Associated Langerhans Cell Histiocytosis of the Thyroid Gland. PG - 1054-1058 LID - 10.1007/s12105-020-01247-8 [doi] AB - Thyroid gland involvement of Langerhans cell histiocytosis (LCH) is extremely rare in both systemic and isolated disease. The role of viral infection in LCH development is not yet fully understood. Although several viruses are proposed as etiologic factors, such as Epstein-Barr virus (EBV) and human herpesvirus 6 (HHV-6), they seem to play a bystander role in LCH. A 29-year old female patient with a prior history of multisystemic LCH (pituitary gland and skull bone), presented with a thyroid nodule. The patient underwent a total thyroidectomy and the histological examination revealed nodular lesions composed of sheets and clusters of histiocytes in the inflammatory background. The histiocytes stained positive for S-100 and CD1a and were negative for HHV-8, cytomegalovirus, and VE1 (anti-BRAF(V600E)) on immunohistochemistry. The EBER in situ hybridization for EBV showed frequent positive-stained cells. The conventional PCR analysis for EBV was positive and qPCR analysis confirmed a significant DNA copy number difference (p = 0.02) between the tumor and adjacent non-neoplastic thyroid tissue. PCR analysis for HHV-6, HPV, HSV was negative in both tumor and benign samples. In conclusion, the presented case showed a rare thyroid involvement by LCH associated with EBV infection, which has not been reported before. Further studies are required to investigate a possible etiologic link between EBV infection and LCH. CI - (c) 2020. Springer Science+Business Media, LLC, part of Springer Nature. FAU - Munkhdelger, Jijgee AU - Munkhdelger J AUID- ORCID: 0000-0003-3483-2342 AD - Department of Pathology, Kameda Medical Center, 929 Higashi-cho, Kamogawa, Chiba, 296-8602, Japan. FAU - Vatanasapt, Patravoot AU - Vatanasapt P AUID- ORCID: 0000-0001-6037-8994 AD - Department of Otorhinolaryngology, Faculty of Medicine, Khon Kaen University, Khon Kaen, 40002, Thailand. AD - HPV and EBV Carcinogenesis Research Group, Khon Kaen University, 40002, Khon Kaen, Thailand. FAU - Pientong, Chamsai AU - Pientong C AUID- ORCID: 0000-0003-2797-043X AD - HPV and EBV Carcinogenesis Research Group, Khon Kaen University, 40002, Khon Kaen, Thailand. AD - Department of Microbiology, Faculty of Medicine, Khon Kaen University, Khon Kaen, 40002, Thailand. FAU - Keelawat, Somboon AU - Keelawat S AUID- ORCID: 0000-0002-4180-7914 AD - Department of Pathology, Faculty of Medicine, Chulalongkorn University, Bangkok, 10330, Thailand. FAU - Bychkov, Andrey AU - Bychkov A AUID- ORCID: 0000-0002-4203-5696 AD - Department of Pathology, Kameda Medical Center, 929 Higashi-cho, Kamogawa, Chiba, 296-8602, Japan. bychkov.andrey@kameda.jp. AD - Department of Pathology, Faculty of Medicine, Chulalongkorn University, Bangkok, 10330, Thailand. bychkov.andrey@kameda.jp. AD - Department of Pathology, Nagasaki University Graduate School of Biomedical Sciences, Nagasaki, 852-8523, Japan. bychkov.andrey@kameda.jp. LA - eng PT - Case Reports PT - Journal Article DEP - 20201102 PL - United States TA - Head Neck Pathol JT - Head and neck pathology JID - 101304010 SB - IM MH - Adult MH - Epstein-Barr Virus Infections/*complications MH - Female MH - Histiocytosis, Langerhans-Cell/*pathology/*virology MH - Humans MH - Thyroid Diseases/*pathology/*virology PMC - PMC8385082 OTO - NOTNLM OT - Epstein-Barr virus OT - Langerhans cell histiocytosis OT - Thyroid COIS- The authors declare that there is no conflict of interest. EDAT- 2020/11/04 06:00 MHDA- 2022/01/18 06:00 PMCR- 2021/11/02 CRDT- 2020/11/03 05:49 PHST- 2020/07/30 00:00 [received] PHST- 2020/10/26 00:00 [accepted] PHST- 2020/11/04 06:00 [pubmed] PHST- 2022/01/18 06:00 [medline] PHST- 2020/11/03 05:49 [entrez] PHST- 2021/11/02 00:00 [pmc-release] AID - 10.1007/s12105-020-01247-8 [pii] AID - 1247 [pii] AID - 10.1007/s12105-020-01247-8 [doi] PST - ppublish SO - Head Neck Pathol. 2021 Sep;15(3):1054-1058. doi: 10.1007/s12105-020-01247-8. Epub 2020 Nov 2.