Rhinocerebral mucormycosis in immunocompetent patients: a case report and review of literature

被引:2
作者
Ajdari, Atra [1 ]
Zolfagharypoor, Azin [2 ]
Firouzifar, Mohammadreza [3 ]
Akbarpour, Maliheh [4 ]
机构
[1] Iran Univ Med Sci, Student Res Comm, Sch Med, Tehran, Iran
[2] Shahid Beheshti Univ Med Sci, Sch Med, Tehran, Iran
[3] Univ Tehran Med Sci, Otorhinolaryngol Res Ctr, Otolaryngol Head & Neck Surg, Tehran, Iran
[4] Guilan Univ Med Sci, Otorhinolaryngol Res Ctr, Sch Med, Dept Otolaryngol & Head & Neck Surg, Rasht, Iran
关键词
Mucormycosis; Immunocompetent; Survival; APOPHYSOMYCES-ELEGANS; INDOLENT MUCORMYCOSIS; CEREBRAL MUCORMYCOSIS; ZYGOMYCOSIS; SINUS;
D O I
10.1007/s15010-023-02147-z
中图分类号
R51 [传染病];
学科分类号
100401 ;
摘要
IntroductionMucormycosis of the head and neck region is a rare but aggressive fungal infection that usually involves immunocompromised patients. More infrequently, this infection can also occur in people with no otherwise known underlying immunological deficit. This rarity usually causes a delay in diagnosis and may severely decrease the chance of survival in these patients. In this study, we present an extreme case of mucormycosis in an immunocompetent patient. By conducting a thorough review of the literature, we aim to increase our knowledge on this matter. Our goal is to improve diagnosis and start treatment at an earlier stage.Case presentation and methodsOur patient was a 31-year-old man who presented with bilateral face numbness, neck pain, headache, and a necrotic palatal lesion 45 days after a dental root canal procedure. There was extensive involvement of facial and skull base bony and soft tissues. Through two debridement sessions and intravenous antifungal treatment, the patient was discharged with near-complete disease resolution. We identified 48 cases in the literature that matched our study criteria. We searched the current literature for proven cases of mucormycosis in the head and neck region who didn't have any underlying disease. We extracted their data and added the data of our patient. Then, we re-analyzed them using descriptive analysis, chi-square, and binary logistic regression to better understand the different factors for survival and disease burden in these patients.Results49 patients were analyzed in this study. The mean age was 46.93 +/- 15.75 (min 16 and max78 years old). The most prevalent subsite to be involved was the sino-nasal mucosa, followed by the surrounding soft tissues and the orbit. While both orbit and intracranial tissue involvement differed significantly between surviving and deceased patients, only intracranial tissue involvement could be used to predict survival. The overall survival rate was 91.8%.ConclusionAlthough very rare, mucormycosis can occur in immunocompetent patients. Physicians should consider mucormycosis when faced with refractory conditions and unusual symptoms such as exposed bones, facial numbness, headaches, and intractable pain. Complementary imaging (CT scan with or without MRI) and histopathological examination are critical for timely diagnosis or exclusion of this potentially fatal yet treatable disease.
引用
收藏
页码:673 / 684
页数:12
相关论文
共 46 条
  • [1] Case of mucormycosis of mandible after self-extraction of teeth incidentally detected to have chronic granulomatous disease: Case report and literature review
    Agarwal, Shubham
    Anand, Abhishek
    Ranjan, Piyush
    Meena, Ved Prakash
    Ray, Animesh
    Dutta, Rimlee
    Jadon, Ranveer Singh
    Vikram, Naval Kishore
    [J]. MEDICAL MYCOLOGY CASE REPORTS, 2020, 28 : 55 - 59
  • [2] Orbital mucormycosis in immunocompetent children; review of risk factors, diagnosis, and treatment approach
    Amanati, Ali
    Barzegar, Hamide
    Pouladfar, Gholamreza
    Dashti, Anahita Sanaei
    Abtahi, Mohamad Bagher
    Khademi, Bijan
    Ashraf, Mohammad Javad
    Badiee, Parisa
    Hamzavi, Seyedeh Sedigheh
    Kashkooe, Ali
    [J]. BMC INFECTIOUS DISEASES, 2020, 20 (01)
  • [3] Ambereen Aafiya, 2021, Clin Infect Pract, V12, P100099, DOI 10.1016/j.clinpr.2021.100099
  • [4] Arafat Y., 2021, WORLD J DENT, V12, P423, DOI [10.5005/jp-journals-10015-1864, DOI 10.5005/JP-JOURNALS-10015-1864]
  • [5] Rhinocerebral Mucormycosis: A Ten-Year Single Centre Case Series
    Balai, Edward
    Mummadi, Sangha
    Jolly, Karan
    Darr, Adnan
    Aldeerawi, Husham
    [J]. CUREUS JOURNAL OF MEDICAL SCIENCE, 2020, 12 (11)
  • [6] Bharathi Ramakrishnan, 2012, J Oral Maxillofac Pathol, V16, P308, DOI 10.4103/0973-029X.99100
  • [7] Isolated palate ulcer due to mucormycosis
    Bist, S. S.
    Varshney, Saurabh
    Bisht, Manisha
    Gupta, Nitin
    Bhatia, Rajat
    [J]. INDIAN JOURNAL OF OTOLARYNGOLOGY AND HEAD & NECK SURGERY, 2008, 60 (01) : 79 - 82
  • [8] An Emergent Entity: Indolent Mucormycosis of the Paranasal Sinuses. A Multicenter Study
    Celis-Aguilar, Erika
    Burgos-Paez, Alan
    Villanueva-Ramos, Nadia
    Solorzano-Barron, Jose
    De La Mora-Fernandez, Alma
    Manjarrez-Velazquez, Juan
    Verdiales-Lugo, Sergio
    Escobar-Aispuro, Lucero
    Becerril, Perla
    Valdez-Flores, Ana
    Javier Merino-Ramirez, Francisco
    Beatriz Caballero-Rodriguez, Carmen
    [J]. INTERNATIONAL ARCHIVES OF OTORHINOLARYNGOLOGY, 2019, 23 (01) : 92 - 100
  • [9] Cumulative Mortality and Factors Associated With Outcomes of Mucormycosis After COVID-19 at a Multispecialty Tertiary Care Center in India
    Choksi, Twinkle
    Agrawal, Anamika
    Date, Purva
    Rathod, Darshana
    Gharat, Anuja
    Ingole, Avinash
    Chaudhari, Bhushan
    Pawar, Nitin
    [J]. JAMA OPHTHALMOLOGY, 2022, 140 (01) : 66 - 72
  • [10] Atypical presentations of fungal osteomyelitis during post COVID-19 outbreak - Case series
    Chugh, Ankita
    Pandey, Akhilesh Kumar
    Goyal, Amit
    Soni, Kapil
    Jain, Vidhi
    Thirunavukkarasu, Balamurugan
    Vedant, Deepak
    Kumar, Deepak
    Kumar, Pravin
    [J]. JOURNAL OF ORAL AND MAXILLOFACIAL SURGERY MEDICINE AND PATHOLOGY, 2022, 34 (05) : 622 - 627