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
|
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 |
Table 2
|
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 |
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.