Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 6, 2020; 8(3): 614-623
Published online Feb 6, 2020. doi: 10.12998/wjcc.v8.i3.614
Erdheim-Chester disease with asymmetric talus involvement: A case report
Qin Xia, Cheng Tao, Ke-Wei Zhu, Wei-Ye Zhong, Peng-Lin Li, Yi Jiang, Min-Zhi Mao
Qin Xia, Cheng Tao, Ke-Wei Zhu, Wei-Ye Zhong, Peng-Lin Li, Min-Zhi Mao, Department of Orthopedics, The Second Xiangya Hospital, Central South University, Changsha 410011, Hunan Province, China
Yi Jiang, Department of Pathology, The Second Xiangya Hospital, Central South University, Changsha 410011, Hunan Province, China
Author contributions: Mao MZ designed the case report and drafted the manuscript; Xia Q and Li PL analyzed the patient data and reviewed the literature; Tao C and Zhu KW provided treatment advice and performed the surgery; Jiang Y and Zhong WY performed the pathological and immunohistochemical examinations.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
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: Min-Zhi Mao, PhD, Doctor, Department of Orthopedics, The Second Xiangya Hospital, Central South University, No. 139 Middle Renmin Road, Changsha 410011, Hunan Province, China. maominzhi@csu.edu.cn
Received: November 21, 2019
Peer-review started: November 21, 2019
First decision: December 23, 2019
Revised: December 25, 2019
Accepted: January 2, 2020
Article in press: January 2, 2020
Published online: February 6, 2020
Processing time: 76 Days and 20 Hours
Abstract
BACKGROUND

Erdheim-Chester disease (ECD) is a rare multi-system or multi-organ histiocytic proliferative disease with diverse clinical manifestations, and the development of the disease is complex, which makes clinical diagnosis and treatment difficult. The characteristic clinical manifestations include multi-organ involvement, especially in the symmetrical diaphysis and metaphysis of the bilateral extremities. ECD with a unilateral talus lesion is extremely rare. Here, we report an unusual case of ECD invading the asymmetric talus and tibia without involving other organs. The patient had good outcome after surgery.

CASE SUMMARY

We report a case of a 67-year-old man who was referred to our outpatient department because of left ankle chronic pain for 5 years, which exacerbated after a foot sprain 6 mo previously. We discovered multiple sclerotic lesions of the tibia and talus on his previous X-ray films, which were initially missed in a local hospital. Therefore, enhanced computer computed tomography (CT) and magnetic resonance imaging were performed. These examinations showed multiple lesions in the bone marrow cavity of the left tibia, and cortical sclerosis and osteonecrosis of the left talus. Specimens were collected via bone puncture from the two lesions, and a final diagnosis of ECD was confirmed by pathological and immunohistochemical examinations. In addition, other auxiliary examinations including head CT, pulmonary CT, spinal CT, abdominal CT, cardiac ultrasound and thyroid ultrasound showed no obvious abnormalities. The patient underwent surgery for the tibia lesion scraping and talus lesion scraping combined with cement casting. The patient started on a progressive rehabilitation at 4 wk, and felt no pain after surgery. During a 2-year follow-up period, the patient exercised normally without pain, and there were no signs of recurrence.

CONCLUSION

This study shows that surgery treatment may also achieve good results for ECD patients with only bone involvement.

Keywords: Erdheim-Chester disease; Talus; Tibia; Osteosclerosis; Surgery; Case report

Core tip: Erdheim-Chester disease (ECD) is a subtype of non-Langerhans’s cell histiocytosis, for which diagnosis is difficult and no treatment guidelines have been available. In addition, ECD is a systemic disease with multi-organ involvement, and especially affects the symmetrical diaphysis and metaphysis of the bilateral extremities. We report the first case of ECD invading the unilateral tibia and talus without involvement of any other organs. The patient was treated with surgery, and had a good prognosis, which added evidence to the diagnosis and treatment of this disease.