IP Annals of Prosthodontics and Restorative Dentistry

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Get Permission Pruthi: Prosthodontic rehabilitation of patients with Rhinocerebral mucormycosis: An update of evidence


Introduction

Mucormycosis is a fatal disease with serious manifestations as it leads to extensive tissue necrosis and spreads rapidly through vascular route.1 Incidental findings while examining a patient for common dental pain or facial swelling may benefit in the early diagnosis of the disease. Depending upon the type and stage of disease, a course of treatment may lead to aggressive resection of various structures such as the maxilla, alveolus, and extraction of otherwise healthy teeth. Rhino orbito cerebral mucormycosis (RCOM), which is reported to be the most common type in developing countries, may also lead to additional loss of orbital and associated facial structures. 2 Consequences include a physically debilitated patient with enormous psychological trauma. Prosthodontists play a pivotal role in improving the functional and overall quality of life of such patients by fabricating maxillary obturators and facial prostheses.

Santos et al, in a recent article on Mucormycosis, presented a review of literature based on PubMed search, wherein the authors included data from 19 papers including their own case.3 The authors highlighted that there were only 3 documented cases with history of Mucormycosis and rehabilitation with palatal obturator between 2007-2019. With no intention to negate the efforts of the authors and based on my experience with rehabilitation of patients with maxillofacial defects, a literature search was performed to update the evidence on patients who suffered with Mucormycosis who underwent prosthodontic rehabilitation with obturators.

Table 1

Presentation of clinical data (Search engine: PubMed)

Author/ Year Country

Age/ Gender

Genus/ Species

Risk Factors

Location

Imaging tests

Treatment

Rehabilitation

Follow-up

Articles between 2007-2019

1.

Pruthi G et al., 2010 4, India

55/ Male

Unspecified

Diabetes, Rhino cerebral mucormycosis

Aramany Class IV

Unspecified

Right maxillary resection

Definitive maxillary obturator and silicone eye prosthesis

Unspecified

2.

I. Abu El-Naaj et al./ 2013 5, Israel, case 1

15/ Female

Zygomycete compatible with mucormycosis

Acute Lymphoid Leukemia, Rhinocerebral Mucormycosis

Aramany Class IV

Computed tomography, MRI, Fiberoptic Endoscopic Sinus Surgery (FESS) for tissue culture and debridement

Liposomal Amphotericin B Posaconazole Left total hemimaxillectomy, left ethmoidectomy and partial left sphenoidectomy

Definitive maxillary obturator

18-month follow-up

I. Abu El-Naaj et al./ 2013 5, Israel, case 2

56/ Female

Unspecified

B-cell lymphoma Rhinocerebral Mucormycosis

Unspecified

Computed tomography

Subtotal maxillectomy. Isovuconazole, Amphotericin B, Voriconazole Posaconazole

Could not be done

Death

I. Abu El-Naaj et al./ 2013 5, Israel, case 3

25/ Female

Unspecified

Acute Myeloid Leukemia Rhinocerebral Mucormycosis

Unspecified

Computed tomography

Total maxillectomy. Amphotericin-B Posaconazole,

Could not be done

Death

I. Abu El-Naaj et al./ 2013 5, Israel, case 4

22/ Male

Unspecified

Chronic Myeloid Leukemia Rhinocerebral Mucormycosis

Unspecified

Computed tomography

Subtotal maxillectomy. Amphotericin-B, Voriconazole

Could not be done

Death

I. Abu El-Naaj et al./ 2013 5, Israel, case 5

75/ Male

Unspecified

Acute Myeloid leukemia Rhinocerebral Mucormycosis

Unspecified

Computed tomography

Subtotal maxillectomy. Amphotericin-B, Voriconazole

Could not be done

Death

I. Abu El-Naaj et al./ 2013 5, Israel, case 6

64 / Female

Unspecified

Aplastic anemia Rhinocerebral Mucormycosis

Unspecified

Computed tomography

Total maxillectomy. Amphotericin-B, Voriconazole

Could not be done

Death

3.

Hatami et al./ 2013 6, Iran

65/ Male

Unspecified

Diabetes, Diabetic keto acidosis, Rhinocerebral mucormycosis

Open communication between the oral, nasal, and orbital cavities***

Not specified

Resected portions included anterior part of hard palate, nasal septum and conchae, left maxillary sinus, and orbital contents

Magnet retainer intraoral definitve obturator and facial prosthesis

2 years

4.

Gowda et al./ 2013 7, India

52/Male

Unspecified

Type II Diabetes Mellitus

Aramany Class 1

Panoromic radiograph

Left hemimaxillectomy

Interim hollow bulb obturator, Definitve implant and magnet retained obturator

6 months

5.

Vidyasankari et al./ 2014 8, India

62/ Male

Unspecified

Rhino cerebral mucormycosis

Maxilla***

Not specified

Left eye exenteration and left maxillectomy

Orbital prosthesis and a definitive intra-oral obturator

Not specified

6.

Faheemuddin M et al./ 2014 9, Pakistan

49/ Female

Unspecified

Diabetes, Hypertension,

Maxillae***

Not specified

Bilateral total maxillectomy

Definitive Maxillary obturator

2 months

7.

Shah R et al./ 2014 10, India, Case 1

48/ Female

Unspecified

Mucormycosis

Maxilla, completely edentulous***

Orthopanto- mography, Computed tomography

Maxillectomy

Definitive maxillary obturator

5 years

8.

Shah R et al./ 2014 10, India, (Case 2)

42/ Female

Unspecified

Mucormycosis

Maxilla***

Not specified

Bilateral oronasal openings

Definitive maxillary obturator

>1 year

9.

Kalaskar et al./ 2016 11, India

18 months/ male

Unspecified

Rhinocerebral mucormycosis

Aramany class IV

Occlusal radiograph

Amphotericin B

Palatal obturator

3 months

10.

Inbarajan et al./ 2018 12, India

60/ Female

Unspecified

Uncontrolled Type 2 Diabetes Mellitus, Mucormycosis

Maxillary defect with completely edentulous arch***

Not mentioned

Surgical debridement, oronasal fistula

Definitive maxillary obturator

3 months

11.

Manjunath et al./ 2018 13, India

Elderly female

Unspecified

Uncontrolled Type 2 Diabetes Mellitus, Mucormycosis

Maxilla***

Computed tomography

Surgical debridement Amphotericin B

Interim maxillary obturator

3 weeks

12.

Salinas TJ et al./ 2019 14, Rochester, Minnesota

32/ Female

Unspecified

Lymphoblastic Lymphoma, Chemotherapy, Invasive mucormycosis

Aramany Class VI

Unspecified

Serial debridement of the maxilla, anterior maxillectomy, right intranasal, and alar resection followed by microvascular free flap

Interim acrylic obturator followed by metal-ceramic fixed prosthesis supported by 8 osseointegrated dental implants.

Unspecified

13.

Mani UM et al./ 2019 15, India

64/Male

Zygomyces

Uncontrolled diabetes

Aramany class IV

Unspecified

Total maxillectomy on left side and right subtotal maxillectomy

Split thickness graft was done in lateral wall of the defect, maxillary definitive 2-piece hollow obturator

14.

Punjabi et al./ 2019 16, India

50/ Male

Unspecified

Uncontrolled diabetes, Mucormycosis

Aramany class VI

Not specified

Resection of hard palate and maxilla

Definitive obturator with silicone relined titanium bulb

Not specified

Case reports after 2019

15.

Pandilwar PK et al./ 2020 17, India

60/ Male

Unspecified

Uncontrolled diabetes Rhinomaxillary Mucormycosis

Completely edentulous maxilla***

Orthopantomogram and Cone-beam computed tomography

Total maxillectomy

Interim palatal obturator

Not specified

16.

Pandilwar PK et al./ 2020 17, India

67/ Male

Unspecified

Uncontrolled diabetes, Mucormycosis

Maxilla***

Cone-beam computed tomography

Surgical debridement of maxilla Amphotericin B

Palatal obturator

2 months

17.

Mohamed et al./ 2020 18, India

48/ Male

Unspecified

Invasive mucormycosis

Left maxilla***

Unspecified

Hemi maxillectomy

Delayed surgical obturator

Not specified

18.

Mohamed et al./ 2020 18, India

55/ Male

Unspecified

Mucormycosis

Maxilla***

Unspecified

Bilateral complete maxillectomy

Delayed surgical obturator

Not specified

19.

Eswaran et al./ 2021 19, India**

31/ Male

Mucorales

Covid 19, Mucormycosis

Aramany class IV

MRI, Computed tomography

Liposomal Amphotericin B, Polymixin B injection, Bilateral oral Posaconazole maxillectomy and right frontal craniectomy with Debridement and repair with Titanium mesh.

Interim obturator

One month

20.

Ravi MB et al./ 2022 20, India (case 1) *

34/ Female

Rhizopus

Uncontrolled Diabetes / COVID 19/ Mucormycosis

Aramany class I

Unspecified

Surgical debridement and resection of right maxilla, Piperacillin, Tazobactam Posaconazole Insulin

Hollow sectional magnet retained prosthesis

Unspecified

21.

Ravi MB et al./ 2022 20, India (case 2) *

60/ Male

Broad aseptate hyphae of Mucorales.

Diabetes / COVID 19/ Right Rhinosinomaxillary Mucormycosis with Left Mucormycosis

Aramany Class IV

Gadolinium enhanced MRI, a plain Computed tomography of para nasal sinuses

Right Total Maxillectomy and left Hemi maxillectomy Posaconazole Amphotericin

Hollow bulb obturator

24, 48, and 72 hours of denture insertion.

22.

Kondaka S et al./2022 21, India*

40/ Male

Not mentioned

Diabetes/ Post Covid Mucorm- ycosis

Bilateral maxille- ctomy***

Post surgical debridement CBCT

Left total Maxillectomy, right subtotal maxillectomy and left orbital decompression along with the resection of the left zygomatic arch and rim.

Obturator supports by patient specific implants

Till 90 days.

23.

Rathee M et al./2022 22, India (Case 1)*

50/Male

Not mentioned

Diabetes/ Post Covid Rhinocerebral Mucormycosis

Aramany Class II

CT scan

Surgical debridement

Immediate Surgical obturator

Not specified

24.

Rathee M et al./2022 22, India (Case 2)*

24/Male

Not mentioned

Early onset Diabetes, Post COVID Mucormycosis

Aramany Class III

Not specified

Surgical debridement

Immediate Surgical obturator

Not specified

25.

Rathee M et al./2022 22, India (Case 3)*

40/Male

Not mentioned

Diabetes, Post COVID Mucormycosis

Aramany Class III

Not specified

Surgical debridement

Immediate Surgical obturator

Not specified

26.

Rathee M et al./2022 22, India (Case 4)

46/Female

Not mentioned

Diabetes, PCM

Aramany Class III

Functional endoscopic sinus surgery

Surgical debridement

Interim obturator

3 months or more

27.

Rathee M et al./2022 22, India (Case 5)

56/Male

Not mentioned

Post Covid Rhinocerebral mucormycosis

Completely edentulous***

Not mentioned

Partial maxillectomy

Interim obturator/ Magnet retained definitive 2 part prosthesis

Not mentioned

28.

Rathee M et al./2022 22, India (Case 6)

48/Male

Not mentioned

Post Covid Rhinocerebral mucormycosis

Completely edentulous***

Not mentioned

Total maxillectomy

Interim obturator/ Definitive magnet retained obturator

Not mentioned

29.

Rathee M et al./2022 22, India (Case 7)

65/Male

Not mentioned

Diabetes, Post Covid Rhinocerebral mucormycosis

Class IV

Not mentioned

Partial maxillectomy

Interim obturator

Not mentioned

30.

Rathee M et al./2022 22, India (Case 8)

46/ Male

Not mentioned

Rhinocerebral mucormycosis

Not specified

Not mentioned

Partial maxillectomy

Interim obturator

Not mentioned

31.

Rathee M et al./2022 22, India (Case 9)

38/Female

Not mentioned

Diabetic, PCM

Aramany Class I? / Closed flap***

Not mentioned

Hemi maxillectomy

Definitive cast partial denture

32.

Artopoulou I et al./ 2022 23, Greece

53/Male

Rhizopus

Diabetes, PCM

Closed with palatal flap***

CT scan

Bilateral subtotal maxillectomy with aggressive debride ment

Definitive obturator with bilateral acrylic projections

6 months

[i] *Post Covid Mucormycosis

[ii] **On Google scholar, ***Difficult to decipher or classify according to Aramany’s classification (1978)

Table 2

Presentation of clinical data (Search engine: Google scholar, Google)

Author/ Year Country

Age/ Gender

Genus/ Species

Risk Factors

Location

Imaging tests

Treatment

Rehabilitation

Follow-up

1.

Dhiman R et al./ 2007, India,

17/ Male

Unspecified

Uncontrolled diabetes and Mucormycosis

Aramany class IV

Not mentioned

Subtotal right maxillectomy and enucleation of right eye

Magnet-retained, silicone eye prosthesis and a polymethyl-methacrylate hollow bulb obturator.

Unspecified

Rehabilitation of a rhinocerebral mucormycosis patient. The Journal of Indian Prosthodontic Society. 2007;7(2):88-91.

2.

Rathee et al./ 2013, India

68/ Male

Unspecified

Uncontrolled diabetes, Rhinocerebral Mucormycosis

Anterior maxilla***

None

Surgical debridement of maxilla

Interim palatal obturator

Not mentioned

Management of Palatal Perforation in an Immunocompromised Diabetic Patient with Mucormycosis Using Surgical and Interim Obturator. Int J Clin Cases Investig 2013;5:63:67.

3.

Naveen et al./ 2015, India

48/ Male

Unspecified

Uncontrolled diabetes, Rhinocerebral Mucormycosis

Maxilla, Aramany Class IV

Computed tomography, panoromic radiograph

Partial maxillectomy, Amphotericin B

Hollow bulb definitive obturator

Not specified

Mucormycosis of the Palate and its Post-Surgical Management: A Case Report. J Int Oral Heal 2015;7:134.

4.

Ilusika et al./ 2018, India

47/ Male

Mucorales

Diabetes, Mucormycosis

Aramany Class VI

Computed tomography

Fluconazole tablets 200 mg for 12 days, Surgical debridement

Definitive obturator

5 months

Enhancing granulation in a postmucormycotic maxillectomy defect with honey: A review of literature and illustrative case. Niger J Basic Clin Sci 2018;15:156-60.

5.

Abrol et al./ 2019, India

44/ Male

Unspecified

Mucormycosis with osteomyelitis

Aramany Class IV

Not mentioned

Surgical debridement

Definitive maxillary obturator

2 months

Prosthodontic Management of Sub-Total Maxillectomy: A Case Report. Chronicles Dent Res 2019;8:61-5.

6.

Bandari et al./2021, India*

65/ Male

Unspecified

Post covid mucormycosis

Aramany class IV

Not mentioned

Left maxillectomy

Hollow bulb obturator

Unspecified

Prosthetic Rehabilitation of A Post-Covid Mucormycosis Maxillectomy Defect Using A Fused Two-Piece Hollow Obturator: A Fabrication Technique. Eur J Mol & Clin Med 2021;7:8564-9.

7.

Rafique et al./ 2020, Pakistan

65/ Female

Unspecified

Type 2 Diabetes, Mucormycosis

Maxilla and extraoral defect**

Not mentioned

Partial maxillectomy, completely edentulous

Magnet retained defintive obturator

One week

Restoration of a post-surgical defect by magnetic maxillofacial prosthesis: A case report. J Univ Med

8.

Prakash M et al./ 2020, India

26/ Male

Unspecified

Kidney transplantation, Mucormycosis

Maxilla**

Unspecified

Partial maxillectomy

Cast metal maxillary definitive obturator

Unspecified

Prosthodontic Rehabilitation of Maxillary Defect in a Patient with Mucormycosis. J Evol Med Dent Sci 2020;9:3163-7.

9.

Mishra et al./ 2021, India

64/ Male

Unspecified

Rhinocerebral mucormycosis

Right maxilla, Aramany class II

Unspecified

Right maxillectomy

Cast metal maxillary defintive obturator

Unspecified

Prosthetic Rehabilitation of Maxillectomy Defects, with Single-Piece Open-Hollow Bulb Definitive Obturator. J Evol Med Dent Sci 2021;10:1169-74.

10.

Garde J et al./2021, India*

55/ Female

Broad, aseptate, ribbon shaped hyaline fungal hyphae seen.

Post covid mucormycosis

Aramany class IV

CT-PNS: MRI-PNS

Bilateral Subtotal Maxillectomy

Definitive maxillary obturator

1 month and at 8 months

Restoring a Smile Post Covid-19 Associated Mucormycosis: A Case Report. J Dental Sci 2021, 6(4): 000314.

11.

Shilpa et al./ 2021, India

52/ Male

Unspecified

Diabetes mellitus

Aramany Class VI

Unspecified

Hemi maxillectomy

Cast metal hollow obturator

3 months

A case report on prosthetic rehabilitation of a patient with hemimaxillectomy: A modified technique. J Int Oral Heal. 2021;13:306-9.

12.

Chinta A et al./ 2022, India

24/Male

Unspecified

Diabetes mellitus Rhinosinomaxillary mucormycosis and right Mucormycosis

Aramany Class IV

Anterior diagnostic rhinoscopy

Left total maxillectomy and right hemimaxillectomy

Definitive hollow acrylic obturator

3 months

Prosthodontic rehabilitation of a mucormycosis patient: a case report

13.

Shalimon A et al./2023*, India

47/Male

Unspecified

Rhino orbital mucormycosis (Post COVID?)*8

Not clear**

Presented after resection

Right hemi maxillectomy

Cast metal obturator

Not specified

Prosthetic rehabilitation of post-COVID mucormycosis. J Interdiscip Dentistry 2023;13:43-7

14.

Nagpal A et al./ 2022, India

24/Male

Unspecified

Mucormycosis

Brown’s class IIb**

Presented after resection

Unilateral maxillary defect not crossing midline

Cast metal obturator

Not specified

Prosthetic Rehabilitation of Mucormycosis Patient By Cast Partial Denture: A Case Report. Bull. Env. Pharmacol. Life Sci., Spl Issue2 2022 : 235-237

[i] *Post covid mucormycosis, ***Difficult to decipher or classify according to Aramany’s classification (1978)

Figure 1

Age and sex distribution of presented patients in Tables.

https://s3-us-west-2.amazonaws.com/typeset-prod-media-server/1b3ad72d-32c4-4df8-8fc1-d5b17629b1f0image1.jpeg

Figure 2

Geographic distribution of presented cases.

https://s3-us-west-2.amazonaws.com/typeset-prod-media-server/1b3ad72d-32c4-4df8-8fc1-d5b17629b1f0image2.jpeg

Data Acquisition and Observations

Literature search was performed on PubMed and Google scholar/ Google for patients who had a history of Mucormycosis and underwent prosthodontic rehabilitation from 2007 to 18th May 2023. 14 more cases could be found on PubMed (Table 1) and 5 on google scholar (Supplementary table with references) with same etiology and treatment during the same time frame (2007-2019).4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16 In addition, Abu El-Naaj et al (2013) have reported 5 patients with history of mucormycosis who succumbed to the disease before they could be rehabilitated with maxillary obturator. 5

I. E. Ali, et al reported prosthodontic rehabilitation of post mucormycosis defects from 30 case reports published between 2010 to 2021. 24 18 more patients on PubMed 17, 18, 19, 20, 21, 22, 23 and 9 on google scholar [Table 2] were reported with history of mucormycosis followed by fabrication of palatal obturator from 2020 to May 2023. Mean age and gender wise distribution of patients who were rehabilitated after surgical management of mucormycosis has been depicted in Figure 1. A clear predilection of male patients is evident over females with age range between 18 months to 75 years.

As aptly emphasized by other authors that most of the cases were reported from India, a discussion about post covid mucormycosis is worth mentioning. Our data shows that approximately 80% of patients were reported from India. Previous literature has also reported global prevalence of mucormycosis to be 70 times more in India, though other countries also faced the heat of the situation. 2 Covid-19 virus infected patients with diabetes were predisposed at a higher rate to the invasion of fungus owing to a compromised host defence mechanism. One of the scientific rationales behind this could be existence of proviral and profungal cellular host factors in olfactory epithelium of diabetic patients, which makes them vulnerable to upper respiratory infections, owing to their higher glucose levels and lowered immunity. 25 In general, the spores of fungus find a favourable niche in pterygomandibular fossae from where they invade different structures.26 Although the data is extensive on different manifestations of post covid mucormycosis, Moorthy et al have specifically documented history of 11 patients who underwent maxillectomy, out of which 4 patients did not survive this deadly fungal infection.27 Recent case reports have documented 10 patients with history of post covid mucormycosis (PCM) who were efficiently rehabilitated with obturators (Table 1, Table 2). 19, 20, 21, 22, 23, 28, 29

Diabetes mellitus was the most significant associated risk factor in 27 patients followed by Acute Myeloid Leukemia based on data reported here and earlier by Santos et al. Rhizopus oryzae has been reported to be the most common causative organism as high glucose levels, insulin resistance and ketoacidosis encourage its growth.30 Amphotericin B remained the first-choice drug followed by Posaconazole. Follow up period was inconsistent in all the case reports with only one patient followed till 5 years by Shah et al. 10 Probable reasons for short term follow up could be because of aggressive nature of disease, poor general health of patients, uncertain family support and difficult accessibility to healthcare services, especially in developing countries.

Few authors performed extensive debridement in patient diagnosed with mucormycosis but did not mention any prosthetic rehabilitation. 31, 32 So, here i would like to emphasize that diagnosis and management of patients with mucormycosis consist of a multidisciplinary team, where a huge onus lies on the maxillofacial prosthodontist. They play a crucial role by helping in social reintegration of patients by their functional and aesthetic rehabilitation. Various authors have provided either a hollow obturator, sectional prosthesis, or innovative designs to retain prothesis in orbito maxillary defects but there are many case reports in which the performance of obturator was not mentioned. Another critical aspect is reporting of patient satisfaction, which was measured subjectively with Oral health Impact Profile – 14 questionnaires in only two case reports and were found to be improved on Likert scale after prosthodontic rehabilitation. 9, 12 Artopoulou I et al used Obturator Functioning Scale and Distress Scale to evaluate the patient satisfaction and psychological status respectively. 23

Early diagnosis of condition, treatment of underlying pathology, thorough debridement, antifungal drugs, local care of post debridement wound, psychological counselling and prosthodontic rehabilitation are integral components of a comprehensive treatment plan for such patients. 25 In a breakthrough research, Sharma et al have proposed the future of intranasal sprays with anti-inflammatory, anti-diabetic and antiviral action which would help in prophylactic control of mucormycosis in covid infected and/ or diabetic patients. Exact formulation of drugs is still unpublished, but it can be a promising therapy for preventing the spread of infection. 25

I would like to highlight the huge discrepancy which was noticed among the references quoted in Table 1 and reference list given at the end of article by Santos et al. 3 So, the readers should follow correct references to prevent further error while quoting the literature. Last but not the least, this article has reported those case reports which have been already documented in literature, though the number of patients who might have suffered with PCM must be much larger. Data from large tertiary care hospitals from India need to be added for complete reporting of the evidence. Follow up of the patients should be provided by the authors as an integral part of comprehensive treatment plan. In view of incomplete reporting and lack of adequate follow up in most of the case presentations to report patient survival, improvement in quality of life or performance analysis of the prosthesis delivered in terms of functional, esthetic, or psychological benefit, I am unable to document any robust evidence on effect of prosthodontic rehabilitation of such patients.

Another interesting finding is that although maximum number of defects were classified as Class IV (n=12) according to Aramany’s classification, 33 many authors did not classify the post resection defects. One of the reasons could be inability to fit the post mucor especially post covid mucor defects into any class of defects due to extensive and emergency management with overburdening of the system. Authors should ensure to consider this important component of presenting any case report and formulation of a more comprehensive single classification system to classify maxillofacial defects may be considered in future.

Conflict of Interest

There are no conflicts of interest in this article.

Source of Funding

None.

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Article type

Short Communication


Article page

117-128


Authors Details

Gunjan Pruthi*


Article History

Received : 26-05-2023

Accepted : 02-06-2023


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