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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 7  |  Issue : 1  |  Page : 70-72

Rhino-oculo-cerebral mucormycosis with acute kidney injury


Department of Nephrology, Indira Gandhi Institute of Medical Sciences, Patna, Bihar, India

Date of Submission24-Dec-2020
Date of Decision15-Mar-2021
Date of Acceptance18-Mar-2021
Date of Web Publication28-Jun-2021

Correspondence Address:
Prit Pal Singh
Room No. 14, Department of Nephrology, Old Administrative Block, Indira Gandhi Institute of Medical Sciences, Sheikhpura, Patna - 800 014, Bihar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jigims.jigims_9_20

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  Abstract 


Rhino-oculo-cerebral mucormycosis (ROCM) is a rare but life-threatening opportunistic fungal infection that usually occurs in immunocompromised patients. Initial symptoms can be nonspecific causing a delay in diagnosis. We report a case of type 2 diabetic female with acute kidney injury who presented with fever, rhino-orbital swelling, and painful diminution of vision of the left eye secondary to infective ROCM. She was treated with liposomal amphotericin B and surgical debridement. She recovered from ROCM and her renal dysfunction improved, but she lost vision in her left eye. Our case highlights the need for early recognition of signs and symptoms so that the treatment can be initiated early before further invasion results in complete destruction of the eyeball and fatal intracranial lesions.

Keywords: Acute kidney injury, diabetes mellitus, immunocompromise, liposomal amphotericin B, rhino-oculo-cerebral mucormycosis


How to cite this article:
Singh PP, Mazumdar P, Krishna A, Kumar O. Rhino-oculo-cerebral mucormycosis with acute kidney injury. J Indira Gandhi Inst Med Sci 2021;7:70-2

How to cite this URL:
Singh PP, Mazumdar P, Krishna A, Kumar O. Rhino-oculo-cerebral mucormycosis with acute kidney injury. J Indira Gandhi Inst Med Sci [serial online] 2021 [cited 2021 Sep 24];7:70-2. Available from: http://www.jigims.co.in/text.asp?2021/7/1/70/318930




  Introduction Top


Rhino-oculo-cerebral mucormycosis (ROCM) is a life-threatening infection caused by fungi of the order of Phycomycetes and class Mucorales. Rhizopus species are the most common cause of invasive mucormycosis in humans, with rhino-cerebral infection being the most common clinical presentation.[1] Mucormycosis accounts for 8.3%–13% of all fungal infections.[2] Patients with diabetes, prolonged corticosteroid use, hematological malignancies, chronic kidney disease, and other immunocompromised states are at higher risk.

Initial symptoms can be nonspecific making a delay in diagnosis until the disease has progressed significantly and invaded the paranasal sinuses, orbit, hard palate, and brain.[1] Hence, diagnosis at the earliest is critical for initiating appropriate management and better outcome. We report a case of ROCM in a 62-year-old diabetic female with acute kidney injury who recovered after treatment although with sequelae.


  Case Report Top


A 62-year-old female presented with high-grade fever, pain, and diminution of vision in the left eye for 2–3 days. It was accompanied by pain and redness in the left eye along with gradually progressive periorbital and left maxillary swelling for the last 5 days [Figure 1].
Figure 1: Sloughing of cornea with periorbital and left maxillary swelling

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On examination, her heart rate was 102/min, blood pressure 154/82 mmHg, and respiratory rate 14/min. Redness of the left maxillary area, ptosis of the left eye, hard palate erosion with eschar, and small ulcer with grayish eschar on the lateral wall of the left nasal cavity were observed [Figure 2].
Figure 2: Grayish eschar on the lateral wall of the left nasal cavity and hard palate

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On ophthalmic examination, left pupil was found to be fixed and dilated with palsy of cranial nerves III, IV, and VI. Visual acuity in the unaffected right eye was 6/9 and in the left eye was 6/60; however, over the next 48 h, the lesion progressed further leading to sloughing of the cornea.

Laboratory findings revealed hemoglobin 9.4 g/dl, leukocyte count 16,000/cumm, blood urea nitrogen 84 mg/dL, serum creatinine 4.7 mg/dL, along with raised erythrocyte sedimentation rate and C-reactive protein. Random blood glucose level was 460 mg/dL, and further workup revealed a diagnosis of diabetes mellitus. Arterial blood gas showed metabolic acidosis (pH 7.20, serum bicarbonate 16 mEq/l). Urine examination for ketone body was negative.

Conjunctival smear and scrapings from the nasal cavity and hard palate were taken which revealed aseptate hyphae with irregular branching, typical of Mucormycosis on 10% KOH mount. Culture of scrapings showed Rhizopus growth after 7 days in Sabouraud's medium (Sabouraud's dextrose agar).

Magnetic resonance imaging (MRI) of the brain revealed mucosal thickening in the left ethmoid and maxillary sinuses with the involvement of the medial wall of the left orbit, along with enhancement of the intraconal fat and rectus muscles of the left eye. Thus, a diagnosis of ROCM was made.

Treatment with liposomal amphotericin B (150 mg/day) was started on day 2 of hospitalization with monitoring of renal function and serum electrolytes. Antibiotics were also used to combat secondary infections, and blood sugar was controlled with insulin along with the correction of dehydration and acidosis. On day 2, after assessment by the ear-neck-throat and ophthalmology department, the patient underwent debridement of all infected and necrotic tissues on an urgent basis.

After 5th day of starting amphotericin B, the patient started responding to the treatment. Liposomal amphotericin B was continued for 21 days, and the patient improved symptomatically with decrease in periorbital and maxillary swelling and serum creatinine levels decreasing to 2.2 mg/dl. On the last follow-up in the nephrology outpatient department, her serum creatinine was 1.4 mg/dL, spot urinary protein creatinine ratio was 0.3, and she had controlled glycemic status.


  Discussion Top


ROCM is a rare but life-threatening condition that usually occurs in immunocompromised patients.[1] The most common mode of entry is by inhalation of aerosolized spores, and they are commonly found in the mucosa of the oral and nasal cavities in immunocompetent patients with very little pathogenic sequelae. However, in immunocompromised patients, infection can occur due to the decrease in the phagocytic containment of the organism. Fungal invasion produces a fibrin reaction and formation of “mucor thrombi” which causes thrombosis of vessels, leading to ischemia, infarction, and subsequent black necrotic eschar formation that is characteristic.[3] Invasion occurs through the bony wall of the sinuses into the orbit, retro-orbital area, and skull base.

Patients who are diabetic and have underlying chronic kidney disease like our patient are more susceptible as an acidic environment enhances angioinvasion of the fungus. At low pH, reduced ability of the serum to bind iron results in a high-iron, glucose-rich, acid milieu.[4] Moreover, fungal hyphae produce a substance called rhizoferrin, which binds iron avidly and this iron–rhizoferrin complex helps in fungal growth.[4] Hyperglycemia and concurrent acidosis cause impairment of neutrophil chemotaxis, decrease in myeloperoxidase activity, and decrease adherence of neutrophils to the endothelial wall, thus impairing the oxidative and nonoxidative fungicidal mechanisms.[5]

In patients with intracranial involvement, fatality rates can exceed 80%, whereas prognosis and survival rates for patients without intracranial involvement are better. Despite advances in diagnosis and treatment, a high mortality rate of 30%–70% still exists.[6]

Management includes early diagnosis, correction of the underlying predisposing factors (if possible), appropriate surgical debridement of infected tissue, and appropriate antifungal therapy. Radical surgical debridement of infective and necrotic tissue should be performed on a priority basis as was done in our patients. Pharmacological agents are only fungistatic.[7] The preferred agents are liposomal amphotericin B whereas posaconazole can be used as an alternative.[7] Treatment initiated early especially within the first 5 days improves prognosis[6] and prevents complete destruction of the eyeball, thus preventing the need for complicated procedures such as exenteration, as was seen with our patient, thereby reducing morbidity and mortality.


  Conclusion Top


ROCM is a rare but life-threatening condition. Management involves a multidisciplinary approach consisting of early diagnosis, appropriate antifungal agents, and urgent surgical debridement of involved tissue. Our case highlights the need for a high index of suspicion in rapidly progressive cases of sinusitis and orbital cellulitis, especially in those with predisposing factors such as diabetes and chronic kidney disease, so that an early diagnosis can be made and treatment initiated early, to reduce morbidity and mortality.

Declaration of patient consent

We certify that patient consent has been taken for participation in the study and for publication of clinical details and images. Patients understand that the names, initials would not be published, and all standard protocols will be followed to conceal their identity.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Petrikkos G, Skiada A, Lortholary O, Roilides E, Walsh TJ, Kontoyiannis DP. Epidemiology and clinical manifestations of mucormycosis. Clin Infect Dis 2012;54:S23-34.  Back to cited text no. 1
    
2.
Pana ZD, Seidel D, Skiada A, Groll AH, Petrikkos G, Cornely OA, et al. Invasive mucormycosis in children: An epidemiologic study in European and non-European countries based on two registries. BMC Infect Dis 2016;16:667.  Back to cited text no. 2
    
3.
Narayanan S, Panarkandy G, Subramaniam G, Radhakrishnan C, Thulaseedharan NK, Manikath N, et al. The “black evil” affecting patients with diabetes: A case of rhino orbito cerebral mucormycosis causing Garcin syndrome. Infect Drug Resist 2017;10:103-8. doi: 10.2147/IDR.S130926.  Back to cited text no. 3
    
4.
McNab AA, McKelvie P. Iron overload is a risk factor for zygomycosis. Arch Ophthalmol 1997;115:919-21.  Back to cited text no. 4
    
5.
Wali U, Balkhair A, Al-Mujaini A. Cerebro-rhino orbital mucormycosis: An update. J Infect Public Health 2012;5:116-26.  Back to cited text no. 5
    
6.
Yohai RA, Bullock JD, Aziz AA, Markert RJ. Survival factors in rhino-orbital cerebral mucormycosis. Surv Ophthalmol 1994;39:3-22.  Back to cited text no. 6
    
7.
Goldstein JC, Spellberg B, Walsh TJ, Kontoyiannis TP, Edwards J Jr., Ibrahim A. Recent advances in the management of mucormycosis: From bench to bedside. Clin Infect Dis 2009;48:1743-51.  Back to cited text no. 7
    


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