Images in…

Rhino-­orbito-­cerebral mucormycosis causing cranial

1
Radiodiagnosis, Pacific Institute DESCRIPTION
of Medical Sciences, Udaipur, Perineural extension of fungal infection is a rare
Rajasthan, India complication that had been observed in post-­
2
Radiodiagnosis, Pacific Institute
COVID-19 immunocompetent mucormycosis
of Medical Sciences Umarda
patients during this pandemic. Rhino-­ orbito-­
Campus, Udaipur, India
cerebral mucormycosis is an opportunistic fungal
Correspondence to infection caused by members of family Mucora-
Dr Rajaram Sharma; ceae. Mucormycosis proved to be the lethal disease Figure 2 (A) T1-­weighted fat-­saturated postcontrast,
​hemantgalaria13@​gmail.​com in untreated patients with diabetes or patients on axial image shows contiguous involvement of the
immunosuppressive therapy with the same pattern preseptal soft tissue, eye globe and optic nerve up to
Accepted 19 August 2021 of spread and severity.1 2 Rhino-­orbito-­cerebral the optic chiasma on the right side (white arrow). (B)
mucormycosis has been declared endemic by the The path of spread can also be evaluated on a diffusion-­
Indian government recently. Due to the aggres- weighted image (white arrow).
sive pattern of spreading in this disease, it rapidly
involves the surrounding structures. Cerebral
extension of the organism occurs either by direct
affected nerve represents neuritis. However, it can
extension, haematogenous route or perineural
progress into a full-­blown abscess. The trigeminal
spread. Over 2 months, we have observed nine
nerve is a large cranial nerve with a unique course
patients with perineural spread involving cranial
that enables us to easily demonstrate this evolution
nerves during this endemic. The peculiar imaging
line. MRI stands to be the best diagnostic and non-­
findings in a perineural spread include thickened
invasive imaging technology that produces three-­
nerves with abnormal enhancement and perineural
dimensional detailed anatomical images that help
fat stranding. Initially, the enhancement in the
determine disease spreading with treatment.1 This
article highlights the role of MRI in the diagnosis of
perineural complications observed in rhino-­orbito-­
cerebral mucormycosis.
Here, we present two representative cases of a
55-­year-­old woman and a 60-­ year-­old man who
presented to our hospital with facial pain, facial
numbness and high-­grade fever. On initial exam-
ination, periorbital soft tissue swelling and facial
redness were found with a history of hospitalisa-
tion for COVID-19 pneumonitis in the recent past.
The patients were administered steroids during the
previous hospital stay. On relevant laboratory inves-
tigations, both the patients had deranged blood
glucose levels.
The patients underwent an MRI of the paranasal
sinuses and brain, revealing orbital cellulitis, pansi-
Figure 1 (A) T1-­weighted fat-­saturated (T1W nusitis, abnormal signals in the premaxillary skin,
FS) postcontrast, axial image shows heterogenous retro-­antral fat and masticator space extending to
enhancement in the right ethmoid sinus (white arrow), the right cavernous sinus (figure 1A).
orbital apex (black arrow), extending to right cavernous The inferior turbinates and paranasal sinuses
sinus (thick white arrow) and then till fifth nerve nucleus also had non-­ enhancing areas on postcontrast
(thick black arrow). (B) Short tau inversion recovery) MRI, representing necrosis. In the first patient, the
image in the coronal plane shows abnormally increased mandibular division of the right trigeminal nerve
© BMJ Publishing Group
Limited 2021. No commercial signals along the right mandibular nerve (white arrows). was thickened and showed heterogeneous signals
re-­use. See rights and (C) Diffusion-­weighted image in the axial plane depicts on all MRI sequences (figure 1B). The diffusion-­
permissions. Published by BMJ. restricted diffusion in the mandibular nerve (white weighted image (DWI) image showed diffusion
arrow). (D) T1W FS postcontrast image in coronal plane restriction in the abscess in the nerve (figure 1C).
To cite: Sharma K, Tiwari T,
Goyal S, et al. BMJ Case demonstrates peripheral enhancement (white arrows) On postcontrast, T1 fat-­ suppressed images, the
Rep 2021;14:e245756. with central liquefied non-­enhancing collection (black nerve revealed peripheral enhancement with central
doi:10.1136/bcr-2021- arrow) along mandibular nerve passing through foramen liquefied non-­enhancing collection, represented an
245756 ovale. abscess (figure 1D).
Sharma K, et al. BMJ Case Rep 2021;14:e245756. doi:10.1136/bcr-2021-245756 1
Images in…
In another similar patient, there was the involvement of the
Patient’s perspective preseptal soft tissue, eye globe and optic nerve up to the optic
chiasma on the right side (figure 2A) and the path of spread was
Patient 1—I feel unfortunate to have this disease. The doctors seen extending along the optic nerve on a DWI (figure 2B).
are trying their best to save my life. I need to be courageous and There are very few documented cases available in the litera-
hopeful and I am trying for that. I feel ok to share my disease ture describing the perineural spread of fungal infection, espe-
findings with the doctor community (some sort of publication in cially after this COVID-19 era; however, similar cases have
their language). been reported previously in patients with uncontrolled diabetes
Patient 2—I feel frustrated as I got COVID-19 infection during by Galletti et al.1 A surgical biopsy from the inferior turbinate
the pandemic and then this fungal infection in this endemic. was done, which revealed non-­septate fungal hyphae. Both the
My eye cannot be saved and this feels horrible, but doctors are patients managed with antifungal drugs; however, they did not
hopeful that after removing my eye, my condition will improve. improve much. Both the patients were receiving treatment in our
I feel doctors should be made aware of such rare diseases. They hospital till the writing of this article.
need to publish more such cases.
Contributors KS and TT contributed in planning, conduct, reporting, conception
and design, acquisition of data or analysis and interpretation of data. SG and RS
contributed in acquisition of data and interpretation of the data.
Learning points
Funding The authors have not declared a specific grant for this research from any
funding agency in the public, commercial or not-­for-­profit sectors.
►► An emergency CT scan should be performed to access
invasive rhinosinusitis in patients with immunosuppressed or Competing interests None declared.
uncontrolled diabetes presenting with symptoms limited to Patient consent for publication Obtained.
paranasal sinuses. If there is a suspicion of central nervous Provenance and peer review Not commissioned; externally peer reviewed.
system (CNS) or orbital involvement, MRI stands to be the This article is made freely available for use in accordance with BMJ’s website
ideal imaging modality. terms and conditions for the duration of the covid-19 pandemic or until otherwise
►► If any of these modalities raises suspicion for rhino-­orbito-­ determined by BMJ. You may use, download and print the article for any lawful,
cerebral mucormycosis, immediate biopsy and antifungal non-­commercial purpose (including text and data mining) provided that all copyright
notices and trade marks are retained.
treatment must be started. Even if the first biopsy comes
negative for fungal elements and imaging shows typical ORCID iD
findings, the condition should still be treated as invasive Rajaram Sharma http://​orcid.​org/​0000-​0003-​1126-​5875
fungal sinusitis and a second biopsy should be performed.
►► Nerve abscess is an extremely uncommon entity, can only
REFERENCES
be diagnosed if the radiologist is familiar with the pathology 1 Galletti B, Freni F, Meduri A, et al. Rhino-­Orbito-­Cerebral mucormycosis in diabetic
and has a high suspicion. MRI is the ideal diagnostic tool disease mucormycosis in diabetic disease. J Craniofac Surg 2020;31:e321–4.
for this pathology that also helps in accessing the disease 2 Anselmo-­Lima WT, Lopes RP, Valera FCP, et al. Invasive fungal rhinosinusitis in
progression. immunocompromised patients. Rhinology 2004;42:141–4.

Copyright 2021 BMJ Publishing Group. All rights reserved. For permission to reuse any of this content visit
https://www.bmj.com/company/products-services/rights-and-licensing/permissions/
BMJ Case Report Fellows may re-use this article for personal use and teaching without any further permission.
Become a Fellow of BMJ Case Reports today and you can:
►► Submit as many cases as you like
►► Enjoy fast sympathetic peer review and rapid publication of accepted articles
►► Access all the published articles
►► Re-use any of the published material for personal use and teaching without further permission
Customer Service
If you have any further queries about your subscription, please contact our customer services team on +44 (0) 207111 1105 or via email at [email protected].
Visit casereports.bmj.com for more articles like this and to become a Fellow

2 Sharma K, et al. BMJ Case Rep 2021;14:e245756. doi:10.1136/bcr-2021-245756