About MEAJO | Editorial board | Search | Ahead of print | Current Issue | Archives | Instructions to authors | Online submission | Subscribe | Advertise | Contact | Login 
Middle East African Journal of Ophthalmology Middle East African Journal of Ophthalmology
Users Online: 208   Home Print this page Email this page Small font sizeDefault font sizeIncrease font size


 
  Table of Contents 
CASE REPORT
Year : 2012  |  Volume : 19  |  Issue : 3  |  Page : 340-342  

Optic nerve thickening and infarction as the first evidence of orbital involvement with mucormycosis


1 Department of Ophthalmology, College of Medicine, King Saud University, Riyadh, Saudi Arabia
2 Department of Radioliogy, College of Medicine, King Saud University, Riyadh, Saudi Arabia

Date of Web Publication3-Jul-2012

Correspondence Address:
Adel H Alsuhaibani
King Abdulaziz University Hospital, King Saud University, PO Box 245, Riyadh 11411
Saudi Arabia
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0974-9233.97957

Rights and Permissions
   Abstract 

We report a 53-year-old female with uncontrolled diabetes mellitus who presented with decreased vision in the right eye for a few hours duration. Orbital computed tomography and magnetic resonance imaging were performed at presentation and showed a thickening and infarction of the right optic nerve with no other orbital abnormalities. A few days later, the patient developed necrosis in the region of the right medial canthus and nasal mucosa. Tissue biopsy confirmed the diagnosis of mucormycosis.

Keywords: Infarction, Mucormycosis, Optic Nerve, Orbit


How to cite this article:
Alsuhaibani AH, Al-Thubaiti G, Al Badr FB. Optic nerve thickening and infarction as the first evidence of orbital involvement with mucormycosis. Middle East Afr J Ophthalmol 2012;19:340-2

How to cite this URL:
Alsuhaibani AH, Al-Thubaiti G, Al Badr FB. Optic nerve thickening and infarction as the first evidence of orbital involvement with mucormycosis. Middle East Afr J Ophthalmol [serial online] 2012 [cited 2019 Sep 22];19:340-2. Available from: http://www.meajo.org/text.asp?2012/19/3/340/97957


   Introduction Top


Mucormycosis is an opportunistic fungal infection caused by Mucorales. [1] It is a potentially lethal infection that generally affects patients who are immunocompromised due to systemic disease. [1] In this case report, we present a middle aged female with rhino- orbito-cerebral mucormycosis who initially presented with thickening and infarction of the right optic nerve and no other orbital abnormalities on orbital imaging few days prior to the development of necrosis in the right medial canthal region and nasal mucosa.


   Case Report Top


A 53-year-old female with poorly controlled diabetes mellitus presented to the emergency room complaining of decreased vision in the right eye that began a few hours prior. Other complaints were redness and swelling of the right cheek for 2 days. Previous ocular history included uncomplicated cataract surgery in the left eye 2 years prior to presentation. Her vital signs were stable. Visual acuity was 20/100 OD and 20/30 OS with a relative afferent pupilary defect on the right side. A mild restriction of ocular motility in all fields of gaze in the right eye was observed. There was no proptosis in either eye. There was marked swelling with mild redness in the lower part of right cheek, with tenderness over the right cheek. There were no signs of abscess. Mild swelling of the right lower lid was present. Fundus examination showed moderate non-proliferative diabetic retinopathy with normal macula and optic disc in both eyes. The rest of the ophthalmic examination was unremarkable. Endoscopic nasal examination showed congested nasal mucosa with no pus or necrosis. Oral cavity examination was normal. The patient was found to have high blood sugar (517 mg/dl) and ketones in the urine. Orbital computed tomography (CT) showed thickening of the right optic nerve with no other orbital abnormalities (no contrast was used as the patient was allergic to the contrast material) [Figure 1]. Para-nasal sinus CT revealed anterior ethmoidal sinus disease, and nicely aerated posterior ethmoidal, sphenoidal and maxillary sinuses bilaterally. The patient was admitted for controlling her diabetes mellitus, management of right cheek cellulitis, and further investigation for the visual loss and restricted ocular motility in the right eye. The patient was started on intravenous amphotericin B, and broad spectrum antibiotics based on clinical suspicion after obtaining blood sample and nasal swab for culture and sensitivity. On the day of admission, magnetic resonance imaging (MRI) was performed. Diffusion-weighted images (DWI) showed high signal intensity along the right optic nerve with hypointense right optic nerve on the apparent diffusion coefficient (ADC) map [Figure 2]. The rest of the orbit, orbital apex, and cavernous sinus were all normal bilaterally [Figure 3]. MRI of the brain was unremarkable. The vision in the right eye decreased dramatically to no light perception with complete external and internal ophthalmoplegia within two days of the initial presentation. The vision and ocular motility continued to remain normal in the left eye. Five days following admission, the patient developed necrosis (eschar) in the right medial canthal region and nasal mucosa. The tissue biopsy confirmed the diagnosis of mucormycosis (Rhizopus organisms). Despite aggressive medical management and wide surgical debridement, the patient died on the 10 th day of admission.
Figure 1: (a and b) Axial and coronal orbital computed tomography showing diffuse mild thickening of the right optic nerve with anterior ethmoidal sinus disease

Click here to view
Figure 2: (a) MR imaging showing high signal intensity along right optic nerve on diffusion-weighted images (DWI) (arrow), (b) Apparent diffusion coefficient (ADC) map showing hypointense signal along right optic nerve (arrow). There is no obvious signal-intensity change in the left optic nerve in the DWI (c) and ADC (d)

Click here to view
Figure 3: Magnetic resonance imaging T2 weighted image of the orbit shows normal optic nerve in both sides

Click here to view



   Discussion Top


Rhino-orbito-cerebral mucormycosis is an acute and fulminant infection. Patients present with nonspecific complaints such as headache, low-grade fever, facial swelling, sinusitis, and ocular symptoms. Due to its lethal nature, rhino-orbito-cerebral mucormycosis must be recognized early and treated aggressively without waiting for onset of tissue necrosis. [2] Orbital involvement results from spread through the nasolacrimal duct, thin medial orbital wall and presents with proptosis, medial rectus thickening and minimal obliteration of fat shadow. [3],[4] Optic nerve infarction that developed two weeks following presentation was previously reported in association with marked orbital inflammatory process in a patient with mucormycosis. [5] Here we report a patient who presented with an isolated optic nerve thickening and infarction as the first and only sign of orbital involvement. The infraction of the optic nerve was apparent by orbital MRI findings of high signal intensity along the right optic nerve on DWI, hypointense right optic nerve on ADC map, normal fluid-attenuated inversion recovery (FLAIR) and postcontrast MRI. [5] These findings may result from an invasion of the blood vessel walls by the organisms leading to occlusion or thrombosis of the optic nerve sheath, blood vessels, or ophthalmic artery. [6] Direct optic nerve infection by mucormycosis may occur. Isolated optic nerve involvement suggests spread of the infection from the primary focus through one of the branches of ophthalmic artery. Thus, optic nerve infarction and thickening may represent the onset of this potentially lethal condition that warrants early and aggressive treatment.

 
   References Top

1.Yohai RA, Bullock JD, Aziz AA, Markert RJ. Survival factors in rhino-orbito-cerebral mucormucosis. Surv Ophthalmol 1994;39:3-22.  Back to cited text no. 1
[PUBMED]  [FULLTEXT]  
2.Anselmo-Lima WT, Lopes RP, Valera FC, Demarco RC. Invasive fungal rhinosinusitis in immunocompromised patients. Rhinology 2004;42:141-4.  Back to cited text no. 2
[PUBMED]    
3.Rangel-Guerra RA, Martínez HR, Sáenz C, Bosques-Padilla F, Estrada-Bellmann I. Rhinocerebral and systemic mucormycosis: Clinical experience with 36 cases. J Neurol Sci 1996;143:19-30.  Back to cited text no. 3
    
4.Nithyanandam S, Jacob MS, Battu RR, Thomas RK, Correa MA, D'Souza O. Rhino-orbito-cerebral mucormycosis. A retrospective analysis of clinical features and treatment outcomes. Indian J Ophthalmol 2003;51:231-6.  Back to cited text no. 4
[PUBMED]  Medknow Journal  
5.Mathur S, Karimi A, Mafee MF. Acute optic nerve infarction demonstrated by diffusion-weighted imaging in a case of rhinocerebral mucormycosis. AJNR Am J Neuroradiol 2007;28:489-90.  Back to cited text no. 5
[PUBMED]    
6.Downie JA, Francis IC, Arnold JJ, Bott LM, Kos S. Sudden blindness and total ophthalmoplegia in mucormycosis: A clinicopathologic correlation. J Clin Neuroophthalmol 1993;13:27-34.  Back to cited text no. 6
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3]


This article has been cited by
1 Mimics and chameleons of optic neuritis
Dinushi Weerasinghe,Christian Lueck
Practical Neurology. 2016; 16(2): 96
[Pubmed] | [DOI]



 

Top
  
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
   Introduction
   Case Report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed4328    
    Printed96    
    Emailed0    
    PDF Downloaded126    
    Comments [Add]    
    Cited by others 1    

Recommend this journal