Rhino orbital cerebral mucormycosis

Rhino orbital cerebral mucormycosis

Rhino orbital cerebral mucormycosis rapidly became an epidemic following the COVID-19 pandemic 1)

Gutiérrez-Delgado et al searched PubMed database from 1964 to 2014 for all available articles in the English language related to rhino-orbital-cerebral chronic infections caused by fungi of the order Mucorales and found 22 cases 2).

Rhino-orbital-cerebral mucormycosis is usually associated with a poor prognosis and is almost exclusively seen in immunocompromised patients

2015

A unique case of isolated intracranial mucormycosis of a slowly progressive nature in a healthy immunocompetent child. A 4-year-old girl with a clear medical and surgical history presented with complaints of right side facial asymmetry and unsteady gait for a period of 10 months. Clinical and radiographic investigations revealed right-sided lower motor neuron facial palsy caused by an infiltrative lesion on the right cerebellopontine angle. Initial surgical debulking was performed, a biopsy was sent for histopathological examination, and a course of prophylactic antibiotic and antifungal drugs was prescribed. The pathological report confirmed the mucormycosis fungal infection, and intravenous amphotericin B was administered for 3 weeks. One month after admission, the patient left the hospital with complete recovery. Follow-ups after 4, 8 and 12 weeks revealed no sensory or motor neurological deficits. In conclusion, this is a unique case of mucormycosis with regard to the nature and location of the infection, along with the host being a healthy child. Initial surgical exploration is a very critical step in the early diagnosis and treatment of such rare conditions 3).

2014

A 42-year-old man who developed a cerebellar mucor abscess after undergoing hematopoietic stem cell transplant for the treatment of myelodysplastic syndrome. In the post-operative period he was admitted to the neurocritical care unit and received liposomal amphotericin B intravenously and through an external ventricular drain. This patient demonstrates that utilization of an external ventricular drain for intrathecal antifungal therapy in the post-operative period may warrant further study in patients with difficult to treat intracranial fungal abscesses 4).

2013

A case of mucormycosis presenting with extensive necrosis of the maxilla with extension into the retrobulbar and infrabulbar region in an otherwise healthy patient. He underwent extensive debriding surgery followed by amphotericin B first and then oral antifungal therapy, but unfortunately, even after extensive surgery and medical treatment, he did not survive 5).

2010

Yoon et al describe a case of Rhino-orbital-cerebral (ROC) mucormycosis with pericranial abscess occurring in a female patient with uncontrolled diabetes mellitus. The infection initially developed in the right-sided nasal sinus and later progressed through the paranasal sinuses with the invasion of the peri-orbital and frontotemporal region, due to the delayed diagnosis and treatment. Numerous non-septate hyphae of the zygomycetes were identified by a punch biopsy from the nasal cavity and by an open biopsy of the involved dura. The patient was treated successfully with extensive debridement of her necrotic skull and surrounding tissues, drainage of her pericranial abscess and antifungal therapy, including intravenous amphotericin B for 61 days and oral posaconazole for the following 26 days. She returned to a normal life and has had no recurrence since the end of her treatment 15 months ago 6).

2000

A 59-year-old immunocompetent white man sustained a high-pressure water jet injury to the right inner canthus while cleaning an air conditioner filter. He later had “orbital cellulitis” develop that did not respond to antibiotics and progressed to orbital infarction. Imaging studies and biopsy results led to a diagnosis of mucormycosis. Tissue culture grew Apophysomyces elegans, a new genus of the family Mucoraceae first isolated in 1979. Orbital exenteration and radical debridement of involved adjacent structures, combined with intravenous liposomal amphotericin, resulted in patient survival.

After orbital exenteration and debridement of involved adjacent structures along with intravenous liposomal amphotericin, our patient has remained free from relapse with long-term follow-up.

The agent causing this case of rhino-orbital-cerebral mucormycosis (Apophysomyces elegans) contrasts with the three genera most commonly responsible for mucormycosis (Rhizopus, Mucor, and Absidia) in that infections with this agent tend to occur in warm climates, by means of traumatic inoculation, and in immunocompetent patients. Rhino-orbital-cerebral mucormycosis should be considered in all patients with orbital inflammation associated with multiple cranial nerve palsies and retinal or orbital infarction, regardless of their immunologic status. A team approach to management is recommended for early, appropriate surgery and systemic antifungal agents 7).


1)

Soni K, Das A, Sharma V, Goyal A, Choudhury B, Chugh A, Kumar D, Yadav T, Jain V, Agarwal A, Garg M, Bhatnagar K, Elhence P, Bhatia PK, Garg MK, Misra S. Surgical & medical management of ROCM (Rhino-orbito-cerebral mucormycosis) epidemic in COVID-19 era and its outcomes – a tertiary care center experience. J Mycol Med. 2021 Dec 25;32(2):101238. doi: 10.1016/j.mycmed.2021.101238. Epub ahead of print. PMID: 34979299.
2)

Gutiérrez-Delgado EM, Treviño-González JL, Montemayor-Alatorre A, Ceceñas-Falcón LA, Ruiz-Holguín E, Andrade-Vázquez CJ, Lara-Medrano R, Ramos-Jiménez J. Chronic rhino-orbito-cerebral mucormycosis: A case report and review of the literature. Ann Med Surg (Lond). 2016 Feb 6;6:87-91. doi: 10.1016/j.amsu.2016.02.003. eCollection 2016 Mar. PubMed PMID: 26981237; PubMed Central PMCID: PMC4776268.
3)

Al Barbarawi MM, Allouh MZ. Successful Management of a Unique Condition of Isolated Intracranial Mucormycosis in an Immunocompetent Child. Pediatr Neurosurg. 2015;50(3):165-7. doi: 10.1159/000381750. Epub 2015 May 7. PubMed PMID: 25967858.
4)

Grannan BL, Yanamadala V, Venteicher AS, Walcott BP, Barr JC. Use of external ventriculostomy and intrathecal anti-fungal treatment in cerebral mucormycotic abscess. J Clin Neurosci. 2014 Oct;21(10):1819-21. doi: 10.1016/j.jocn.2014.01.008. Epub 2014 May 19. Review. PubMed PMID: 24852901.
5)

Rahman A, Akter K, Hossain S, Rashid HU. Rhino-orbital mucourmycosis in a non-immunocompromised patient. BMJ Case Rep. 2013 Feb 6;2013. pii: bcr2012007863. doi: 10.1136/bcr-2012-007863. PubMed PMID: 23391952; PubMed Central PMCID: PMC3604437.
6)

Yoon YK, Kim MJ, Chung YG, Shin IY. Successful treatment of a case with rhino-orbital-cerebral mucormycosis by the combination of neurosurgical intervention and the sequential use of amphotericin B and posaconazole. J Korean Neurosurg Soc. 2010 Jan;47(1):74-7. doi: 10.3340/jkns.2010.47.1.74. Epub 2010 Jan 31. PubMed PMID: 20157385; PubMed Central PMCID: PMC2817523.
7)

Fairley C, Sullivan TJ, Bartley P, Allworth T, Lewandowski R. Survival after rhino-orbital-cerebral mucormycosis in an immunocompetent patient. Ophthalmology. 2000 Mar;107(3):555-8. PubMed PMID: 10711895.

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