Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 26, 2021; 9(6): 1416-1423
Published online Feb 26, 2021. doi: 10.12998/wjcc.v9.i6.1416
Osseous Rosai-Dorfman disease of tibia in children: A case report
Djandan Tadum Arthur Vithran, Jian-Zhou Wang, Feng Xiang, Jie Wen, Sheng Xiao, Wen-Zhong Tang, Qian Chen
Djandan Tadum Arthur Vithran, Jian-Zhou Wang, Feng Xiang, Jie Wen, Sheng Xiao, Wen-Zhong Tang, Qian Chen, Department of Pediatric Orthopedic, Hunan Provincial People's Hospital, the First Affiliated Hospital of Hunan Normal University, Changsha 410013, Hunan Province, China
Author contributions: Vithran DTA and Wang JZ contributed equally to this work and are co-first authors; Xiang F and Wei J designed the research; Xiao S and Tang WZ collected the data; Chen Q performed the literature review.
Supported by Natural Science Foundation of Hunan Province, China, No. 2019JJ50324.
Informed consent statement: All study participants, or their legal guardian, provided informed written consent prior to study enrollment.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Jie Wen, PhD, Doctor, Department of Pediatric Orthopedic, Hunan Provincial People's Hospital, the First Affiliated Hospital of Hunan Normal University, No. 61 West Jiefang Road, Changsha 410013, Hunan Province, China. cashwj@qq.com
Received: October 9, 2020
Peer-review started: October 9, 2020
First decision: December 3, 2020
Revised: December 15, 2020
Accepted: December 22, 2020
Article in press: December 22, 2020
Published online: February 26, 2021
Processing time: 119 Days and 7.7 Hours
Abstract
BACKGROUND

Rosai-Dorfman disease (RDD), or sinus histiocytosis with massive lymphadenopathy, is a benign histiocytic disorder. Extranodal involvement is common, occurring in > 40% of patients, but bone involvement occurs in < 10% of cases. In addition, primary bone RDD is extremely rare. The majority of patients are adolescents and young adults, and the mean age at onset is 20-years-old.

CASE SUMMARY

We report an 8-year-old Chinese girl who presented to our hospital with an insidious onset of swelling and pain in the middle shaft of her right tibia for 4 mo. We performed total surgical resection of the right tibia lesion and allograft transplantation. A good prognosis was confirmed at the 6 mo follow-up. Pain and swelling symptoms were totally relieved, range of motion of her right knee and ankle returned to normal, and there was no clinical evidence of lesion recurrence at last follow up. Our case is the second reported case of osseous RDD without lymphadenopathy in the shaft of the tibia of a child.

CONCLUSION

Extranodal RDD is a rare disease and can be misdiagnosed easily. Lesion resection and allograft transplantation are an option to treat extranodal RDD in children with good short term result. Pediatric orthopedist should be aware of this rare disease, especially extranodal involvement.

Keywords: Osseous Rosai-Dorfman disease; Children tibia pain and swelling; Lesion resection; Allograft transplantation; Good prognosis; Rare benign disorder; Case report

Core Tip: Extranodal Rosai-Dorfman disease (RDD) is a rare disease and can be misdiagnosed easily. We report an 8-year-old Chinese female case. We performed total surgical resection of the right tibia lesion and allograft transplantation. A good prognosis was confirmed at the 6 mo follow-up. Pain and swelling symptoms were totally relieved, movement range of right knee and ankle return to normal, and there was no clinical evidence of lesion recurrence at last follow up. Our case is the second reported case of osseous RDD without lymphadenopathy in the shaft of tibia of a child.