Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2022; 10(19): 6688-6694
Published online Jul 6, 2022. doi: 10.12998/wjcc.v10.i19.6688
Successful treatment of Morbihan disease with total glucosides of paeony: A case report
Li-Feng Zhou, Rong Lu
Li-Feng Zhou, Department of Dermatology, The 942nd Hospital of the PLA Joint Logistic Support Force, Yinchuan 750004, Ningxia Hui Autonomous Region, China
Rong Lu, Department of Pathology, The 942nd Hospital of the PLA Joint Logistic Support Force, Yinchuan 750004, Ningxia Hui Autonomous Region, China
Author contributions: Zhou LF and Lu R contributed equally to this work; Zhou LF consulted and recorded the patient’s medical history, reviewed the literature, collected the clinical data and images, and wrote the manuscript; Lu R participated in the formulation of the manuscript, literature search, collection of pathological images, image editing, and manuscript modification; all authors approved the final version for submission.
Informed consent statement: Informed consent was obtained from the patient for the publication of this case report.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Rong Lu, MMed, Director, Department of Pathology, The 942nd Hospital of the PLA Joint Logistic Support Force, No. 893 Shengli South Street, Xingqing District, Yinchuan 750004, Ningxia Hui Autonomous Region, China. lrzlf@sohu.com
Received: January 13, 2022
Peer-review started: January 13, 2022
First decision: April 8, 2022
Revised: April 15, 2022
Accepted: May 17, 2022
Article in press: May 17, 2022
Published online: July 6, 2022
Processing time: 162 Days and 4.5 Hours
Abstract
BACKGROUND

Morbihan disease is a rare cutaneous disorder characterized by non-pitting edema and erythema of the upper two-thirds of the face. In severe cases, orbital and facial contour changes may affect the visual field, and there is no guideline for the standard treatment of this disease. Existing treatment methods have been reported to be associated with long medication cycle, easy recurrence after drug withdrawal, and multiple adverse reactions.

CASE SUMMARY

A 55-year-old Chinese woman presented to our hospital with non-pitting edema and erythema of the upper two thirds of her face for 5 mo. Physical examination showed obvious edema and erythema on the upper face. The boundary was unclear, the lesions were hard and non-pitting, and infiltration was obvious by touch. Pathological examination revealed mild hyperkeratosis of the epidermis, nodular inflammatory lesions in the dermis, epithelioid granuloma, and inflammatory cell infiltration with lymphocytes and histiocytes around skin appendages and blood vessels. Alcian blue staining, acid fast staining, silver staining and periodic acid-Schiff staining were negative. The patient was diagnosed with Morbihan disease. She was treated with prednisone acetate and tripterygium wilfordii polyglycoside tablets for 4 mo, and the edema was slightly reduced, but transaminase levels were significantly increased. Compound glycyrrhizin capsules were administered for liver protection for 1 mo; however, facial edema did not significantly improve and transaminase levels continued to increase. Total glucosides of paeony capsules were then administered for 4 mo, and transaminase level returned to normal and the patient’s facial edema disappeared completely.

CONCLUSION

Total glucosides of paeony has a remarkable effect in Morbihan disease, without adverse reactions.

Keywords: Morbihan disease; Morbihan syndrome; Total glucosides of paeony; Prednisone acetate; Tripterygium wilfordii; Case report

Core Tip: Morbihan disease is a rare skin disease characterized by non-pitting edema and erythema of the upper two-thirds of the face. The orbital and facial contour changes can affect the field of vision and cause disfiguring damage if not treated in time. The specific pathogenesis is unclear, and there is no standard treatment guideline for this disease. Thus, it is necessary to identify a safe and effective treatment. We treated a case of Morbihan disease with total glucosides of paeony and achieved very good results with no adverse reactions. The treatment of Morbihan disease with total glucosides of paeony has not previously been reported.