Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 26, 2019; 7(2): 228-235
Published online Jan 26, 2019. doi: 10.12998/wjcc.v7.i2.228
Successful treatment of invasive fungal rhinosinusitis caused by Cunninghamella: A case report and review of the literature
Yong-Cai Liu, Min-Li Zhou, Ke-Jia Cheng, Shui-Hong Zhou, Xue Wen, Cheng-Dong Chang
Yong-Cai Liu, Min-Li Zhou, Ke-Jia Cheng, Shui-Hong Zhou, Department of Otolaryngology, the First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
Xue Wen, Cheng-Dong Chang, Department of Pathology, the First Affiliated Hospital, College of Medicine, Zhejiang University, Hangzhou 310003, Zhejiang Province, China
Author contributions: Cheng KJ and Zhou SH designed the report; Zhou ML, Wen X, and Chang CD collected the patient’s clinical data; Liu YC and Zhou ML analyzed the data and wrote the paper.
Supported by Zhejiang Province Public Welfare Fund, No. 2014C33203; and National Natural Science Foundation of China, No. 81770976.
Informed consent statement: Consent was obtained from relatives of the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts 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 which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: Shui-Hong Zhou, PhD, Professor, Department of Otolaryngology, the First Affiliated Hospital, College of Medicine, Zhejiang University, 79 Qingchun Road, Shangcheng District, Hangzhou 310003, Zhejiang Province, China. 1190051@zju.edu.cn
Telephone: +86-571-87236894 Fax: +86-571-87236895
Received: October 2, 2018
Peer-review started: October 2, 2018
First decision: November 1, 2018
Revised: November 12, 2018
Accepted: December 14, 2018
Article in press: December 15, 2018
Published online: January 26, 2019
Processing time: 117 Days and 22 Hours
Abstract
BACKGROUND

Invasive fungal rhinosinusitis (IFR) caused by Cunninghamella is very rare but has an extremely high fatality rate. There have been only seven cases of IFR caused by Cunninghamella reported in English and, of these, only three patients survived. In this article, we present another case of IFR caused by Cunninghamella, in which the patient was initially treated successfully but then deteriorated due to a relapse of leukemia 2 mo later.

CASE SUMMARY

A 50-year-old woman presented with a 2-mo history of right ocular proptosis, blurred vision, rhinorrhea and nasal obstruction. Nasal endoscopic examination showed that the middle turbinate had become necrotic and fragile. Endoscopic sinus surgery and enucleation of the right orbital contents were performed successively. Additionally, the patient was treated with amphotericin B both systematically and topically. Secretion cultivation of the right eye canthus showed infection with Cunninghamella, while postoperative pathology also revealed fungal infection. The patient’s condition gradually stabilized after surgery. However, the patient underwent chemotherapy again due to a relapse of leukemia 2 mo later. Unfortunately, her leukocyte count decreased dramatically, leading to a fatal lung infection and hemoptysis.

CONCLUSION

Aggressive surgical debridements, followed by antifungal drug treatment both systematically and topically, are the most important fundamental treatments for IFR.

Keywords: Cunninghamella; Invasive fungal rhinosinusitis; Acute myeloid leukemia; Treatment; Prognosis; Case report

Core tip: Invasive fungal rhinosinusitis (IFR) caused by Cunninghamella is very rare but has an extremely high fatality rate. There have been only seven cases of IFR caused by Cunninghamella reported in English and, of these, only three patients survived. The middle turbinate has been found to be the most common site of invasion, followed by the maxillary and ethmoid sinuses. Antifungal drug treatment and/or surgical treatments are the most important fundamental treatments. In this article, we present another case of IFR caused by Cunninghamella, in which the patient was initially treated successfully but then deteriorated due to a relapse of leukemia 2 mo later.