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 Table of Contents  
CASE REPORT
Year : 2023  |  Volume : 3  |  Issue : 1  |  Page : 167-169

Rhino-orbito-cerebral mucormycosis in a pregnant woman: A case report


Department of Ophthalmology, RMC, Kakinada, West Bengal, India

Date of Submission22-Jul-2022
Date of Acceptance27-Oct-2022
Date of Web Publication20-Jan-2023

Correspondence Address:
Penumala Viswamithra
11-9-43/1, Dasapalla Hills, Visakhapatnam - 530 003, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_1734_22

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  Abstract 


Mucormycosis is an uncommon, rapidly progressive, angioinvasive fungal infection characterized by large areas of tissue necrosis. We report a case of a 25-year-old primigravida of gestational age 25 weeks with clinically and radiologically proven COVID-19-related rhino-orbito-cerebral mucormycosis (ROCM) stage 3c with gestational diabetes. She was treated with injection liposomal amphotericin B, but her condition rapidly progressed with impending intracranial extension. She was subjected to total exenteration of the left eye with extensive endoscopic sinus surgery and continued on systemic antifungals along with supportive therapy for four weeks after surgery.

Keywords: COVID-19 infection, exenteration, gestational diabetes, rhino-orbito-cerebral mucormycosis, ROCM in pregnancy


How to cite this article:
Viswamithra P, Muralikrishna V, Sultana SS, Sindusha K, Rani BS. Rhino-orbito-cerebral mucormycosis in a pregnant woman: A case report. Indian J Ophthalmol Case Rep 2023;3:167-9

How to cite this URL:
Viswamithra P, Muralikrishna V, Sultana SS, Sindusha K, Rani BS. Rhino-orbito-cerebral mucormycosis in a pregnant woman: A case report. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Feb 1];3:167-9. Available from: https://www.ijoreports.in/text.asp?2023/3/1/167/368166



Mucormycosis is an opportunistic fungal infection caused by saprophytic fungi belonging to the genera Mucor, Rhizopus, and Absidia of order mucorales and class zygomycetes. A major route of infection is via the inhalation of spores.[1] The most common form is rhino-orbito-cerebral mucormycosis (ROCM), with others being pulmonary, cutaneous, disseminated, and gastrointestinal (GI) mucormycosis. ROCM is almost exclusively seen in immunocompromised patients such as those with uncontrolled diabetes, diabetic ketoacidosis (DKA), HIV/AIDS, and organ transplantation. Currently, it is seen in patients with a recent history of concomitant COVID-19 infection. There is no absolute data on whether pregnant women are at increased risk of severe consequences of COVID-19 infection. A majority will experience mild or asymptomatic disease; however, some have increased rates of intensive care unit (ICU) admission and the need for mechanical ventilation.[2] The disease progresses rapidly within a period of a few hours to days, leading to cranial nerve palsies and with features of central nervous system (CNS) involvement.


  Case Report Top


A 25-year-old female primigravida of 25 weeks gestational age came to our institute with complaints of left-sided facial pain, periocular swelling, proptosis, drooping of the upper eyelid with gradual diminution of vision over the past four days. She was diagnosed with COVID-19 infection via real-time reverse transcription polymerase chain reaction (RT-PCR) 17 days prior to visiting our institute. She was treated at a primary health care center with intravenous steroids, O2 inhalation, and other medications, the nature of which was not known exactly. Prior to COVID-19 infection, she had regular antenatal checkups, and her general and obstetric conditions were unremarkable.

On examination, her visual acuity in the right eye was 6/6 and no light perception in the left eye. On examination, there was left periocular edema, proptosis, complete ptosis, mild conjunctival congestion, chemosis, and total ophthalmoplegia. The right eye was normal apart from mild periocular edema.

On deep nasal endoscopy, necrosis of the left inferior and middle turbinates were noted. Broad branching aseptate hyaline hyphae seen on 10% KOH mount and Rhizopus species were isolated on fungal culture.

[Figure 1]a and [Figure 1]b show the clinical and endoscopic findings.
Figure 1: (a) Clinical picture of a 25-year-old primigravida at the time of admission showing left eye periocular edema, proptosis, complete ptosis, and chemosis. (b) Deep nasal endoscopic picture showing necrosis of inferior and middle turbinates

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Contrast magnetic resonance imaging (MRI) brain with orbits and paranasal sinuses (PNS) revealed mucosal thickening involving the paranasal sinuses, thickening of medial, inferior, lateral recti, and superior oblique muscles. Left-sided orbital cellulitis, optic neuritis, perineuritis, mild proptosis along with cavernous sinus involvement and internal carotid artery angioinvasion were seen.

[Figure 2] shows the radiological findings at presentation.
Figure 2: (a) Axial section of MRI paranasal sinuses showing mucosal thickening of bilateral maxillary and ethmoidal sinuses. (b) Axial section of MRI orbits showing thickening of extraocular muscles with involvement of the orbital apex

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The lab reports revealed DKA with elevated blood sugars (FBS: 320 mg/dl, RBS: 520 mg/dl), urine ketone bodies, and metabolic acidosis with elevated C reactive protein of 18.07 mg/L, serum ferritin of 468.99 ng/ml, total count at 20,700 cells/mm3. Complete blood count and renal function tests were normal.

She was immediately admitted. The patient and her husband were explained about the course of the illness, associated morbidity and mortality, teratogenicity, chances of spontaneous abortion, intrauterine death of the fetus, and associated maternal and fetal risk due to general anesthetic agents. She refused any intervention with her pregnancy. After obtaining necessary informed and written consent from the patient and her spouse, medical management was started immediately as per protocol by the Government of Andhra Pradesh which included intravenous liposomal amphotericin B (category B drug).[3],[4]

She was initiated on inj. liposomal amphotericin B 300 mg in 300 ml D5 along with long-acting insulin, injection KCL, and supportive treatment. The patient's condition rapidly deteriorated with impending intracranial extension within 24 hours. She was immediately subjected to functional endoscopic sinus surgery with left modified Denker's procedure, left anterior and posterior ethmoidectomy, left sphenoidotomy with right medial maxillary antrostomy, and left eye total orbital exenteration under general anesthesia.

[Figure 3] is an intraoperative picture showing total orbital exenteration.
Figure 3: Intraoperative picture during orbital exenteration

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The debrided tissue from the sinuses and the left orbit were sent for histopathological examination which revealed features of invasive fungal sinusitis. The patient was continued on intravenous amphotericin B, subcutaneous insulin, and supportive treatment for four weeks postoperatively. Her blood sugars and renal parameters were monitored regularly. During her stay, daily per abdominal examination for monitoring of fetal movements and fetal heart rate was done by the obstetrician. She was discharged four weeks after surgery and continued on oral posaconazole and antihyperglycemic drugs along with supportive treatment. The patient was reviewed weekly for one month, fortnightly for the next five months, and then followed up monthly till date.

[Figure 4] shows the clinical and endoscopic picture at two months of follow-up.
Figure 4: (a) Clinical picture at two months of follow-up showing healthy granulation tissue in the left orbit. (b) Endoscopic picture at two months of follow-up showing healthy nasal mucosa

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Pregnancy was continued till term with weekly ultrasonography to monitor the gestational well-being of the fetus. Six weeks after discharge, at 37 weeks gestational age, she gave birth to a single, healthy, female child with birth weight of 2 kg and with APGAR score 8 to 10 by normal vaginal delivery. Eight weeks after surgery, follow-up diagnostic nasal endoscopy (DNE) was done which revealed normal healthy mucosal tissue.

There was no recurrence till date.


  Discussion Top


Rhino-orbito-cerebral mucormycosis is a potentially fatal fungal infection that commonly affects diabetics and other risk factors include neutropenia, usage of steroids, and immunocompromised state.[5] Pregnancy is not a recognised risk factor; however, COVID-19 infection, intravenous (IV) steroid usage, and gestational diabetes with poor glycemic control increased the risk of mucormycosis in our patient.[6] The fungal hyphae spreads via vessels to the paranasal sinuses and into the CNS via orbit and cribriform plate.[6] Treatment of mucormycosis is challenging. It requires a combination of radical surgical debridement of the involved tissue with aggressive antifungal therapy. Amphotericin B is a first-line drug against mucormycosis.[7]

In this study, our patient was initially started on amphotericin B for four weeks and later changed to tablet posaconazole. Amphotericin B, a systemic polyene, is the safest systemic antifungal drug in pregnancy. They bind to ergosterol, forming transmembrane pores leading to ionic leakage and fungal death. Maternal nephrotoxicity is reported to be similar to that of non-pregnant women. No teratogenicity has been shown in rodents or rabbits at ten times the recommended human dose, even during the first trimester (category B drug).[4] Posaconazole, triazole has expanded antifungal spectrum including mucorales. It is embryotoxic and teratogenic in rodents and rabbits; no human data are available so far (category C drug).[4] However, it is indicated in life-threatening maternal disease without a therapeutic alternative.

Early reporting, early proven diagnosis by direct microscopy from nasal aspirate and by histopathological examination, and patient's willingness to take the risk of systemic antifungals in spite of her antenatal status, response to treatment with amphotericin B, and reversal of risk factors and unknown factors related to immune status of patient with multidisciplinary team approach helped in the treatment of our patient and in the saving of two lives.


  Conclusion Top


To our knowledge, this is the first case of ROCM in an antenatal woman to be reported. Though exact treatment protocols are not available yet, early diagnosis, prompt initiation of amphotericin B therapy with surgical debridement, and supportive care led to successful management of rhino-orbital mucormycosis. With timely, committed, and aggressive management mucormycosis can be concurred.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sen M, Lahane S, Lahane TP, Parekh R, Honavar SG. Mucor in viral land: A Tale of two pathogens. Indian J Ophthalmol 2021;69:244-52.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Wastnedge EAN, Reynolds RM, van Boeckel SR, Stock SJ, Denison FC, Maybin JA, et al. Pregnancy and COVID-19. Physiol Rev 2021;101:303-18.  Back to cited text no. 2
    
3.
Government of Andhra Pradesh – management protocol for mucormycosis. Dated: 18/05/2021.  Back to cited text no. 3
    
4.
Pilmis B, Jullien V, Sobel J, Locuit M, Lortholary O, Charlier C. Antifungal drugs during pregnancy: An updated review. J Antimicrob Chemother 2015;70:14-22.  Back to cited text no. 4
    
5.
Ibrahim AS, Spellberg B, Walsh TJ, Kontoyiannis DP. Pathogenesis of mucormycosis. Clin Infect Dis 2012;54(Suppl 1):S16-22.  Back to cited text no. 5
    
6.
Hayat M, Mushtaq S, Saba S, Saif R. Rhino-orbito-mucormycosis as a presenting manifestation of gestational diabetes mellitus. Indian J Endocr Metab 2011;15:S65-6.  Back to cited text no. 6
    
7.
Juma F, Nagaraj V, Rashid M, Darwish A. Placental mucormycosis in an IVF induced pregnancy in a diabetic patient. Bahrain Med Bull 2019;41:278-80.  Back to cited text no. 7
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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