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Table of Contents   
CASE REPORT  
Year : 2019  |  Volume : 30  |  Issue : 5  |  Page : 807-809
Maxillary rehabilitation after complete destruction by fungal osteomyelitis


Department of Oral and Maxillofacial Surgery, Balaji Dental and Craniofacial Hospital, 30, KB Dasan Road, Teynampet, Chennai, India

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Date of Submission11-Sep-2019
Date of Decision04-Oct-2019
Date of Acceptance19-Oct-2019
Date of Web Publication18-Dec-2019
 

   Abstract 


Treatment of uncommon fungal infection such as Rhinosporidiosis is challenging, especially when occurring in a non-immunocompromised patient in non-invasive form. Extensive involvement, extending into maxillary jawbone would need aggressive, chemotherapeutic and surgical approach. There are few reports of successful rehabilitation of maxillary jaw with dental implants after treatment of such fungal infection. After adequate medical treatment, the iliac graft and recombinant bone morphogenetic protein-2 was effectively utilized to reconstruct the lost maxillary bone. Later, dental implants were placed, that osseointegrated well.

Keywords: Fungal osteomyelitis, maxillary rehabilitation, rhinosporidiosis

How to cite this article:
Balaji S M, Balaji P. Maxillary rehabilitation after complete destruction by fungal osteomyelitis. Indian J Dent Res 2019;30:807-9

How to cite this URL:
Balaji S M, Balaji P. Maxillary rehabilitation after complete destruction by fungal osteomyelitis. Indian J Dent Res [serial online] 2019 [cited 2020 Aug 5];30:807-9. Available from: http://www.ijdr.in/text.asp?2019/30/5/807/273435



   Introduction Top


Maxillary fungal osteomyelitis is not uncommon and often requires surgical treatment. It can lead to large scale destruction of the maxillary bone without proper diagnosis, intervention and treatment. Most of the fungal osteomyelitis of jaws occurs in medically compromised patients, possibly owing to poor immune surveillance. Rehabilitation of a severely affected jaw is challenging, as there would have been extensive involvement, regional necrosis, and presence of sequestrum. Staged reconstruction using grafts have been the most common approach. The product of neo-osteogenesis is often compromised and lacks the quantity and quality of normal bone.[1]

Dental implants have evolved as an ideal replacement for lost tooth. The placement of dental implants predisposes to formation of biofilms on it. This biofilms, being new ecological niche may facilitate the formation, growth and maturation of opportunistic pathogens including fungal organisms. A recent systematic review has identified that more than a few dozens of cases of osseointergrated dental implants predisposing the occurrence of osteomyelitis of jaws.[2] Procedures such as sinus lift increase the risk of having a fungal infection.[2],[3],[4] But there are very few case reports of placing dental implants successfully in a treated maxillary osteomyelitis due to fungal ball.[5] The aim of this manuscript is to present the successful management of a case of maxillary fungal infection by Rhinosporidiosis and later place dental implants in the newly developed bone.


   Case Report Top


A 30-year-old male reported to the hospital with pain and swelling in the left half of the upper jaw since 12 months. History revealed that about 14 months back, he was diagnosed to have a cyst in the left maxilla that was surgically removed; however, the infection in the region did not subside even after removal of the cyst. Subsequent examination and exploration by an otorhinolaryngologist revealed a mass of non-native tissue that was successfully removed through functional endoscopic sinus surgery about nine months back. Culture sensitivity and typing of the organism revealed that to be a member of the rhinosporidium species. Patient underwent several unsuccessful intraoral curettages along the left maxillary alveolar area in a bid to remove the foci of infection.

On examination, there was no contributory general medical history. Patient had completely edentulous second quadrant with obvious loss of alveolar bone. The maxillary right permanent premolars, canines and incisors were periodontally compromised with lack of alveolar bone support. The involved area showed that bony destruction was restricted to alveolar bone and not extending into other parts of the maxilla. The infected area of the alveolus was totally removed along with the maxillary right permanent premolars, canines, and incisors. The entire area was reconstructed with rib grafts. Sufficient antibiotic coverage and painkillers were provided. The healing was uneventful and patient resumed normal activities [Figure 1].
Figure 1: (a-c) Pre-operative CT, OPG, and clinical pictures showing edentulous second quadrant with alveolar bone loss and periodontally compromised right permanent premolars, canines, and incisors. (d) Intra –operative view showing harvested rib graft through right infra-mammary incision.

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After about a year, patient had similar pain and swelling. Radiographic examination revealed the persistence of infection. The plates and screws were removed. A complete work up on the infection was done. Help of a Tropical Infectious Diseases specialist was taken. Subsequent to microbiological tests, tablet Voritek 200 mg (VORICONAZOLE) was advised for one month [Figure 2]. Subsequently, the patient was cured of the fungal infection.
Figure 2: (a and b) Pre-operative CT, OPG showing persistent infection along with reconstructed maxillary alveolus with plates and screws. (c) Pre-operative clinical view showing partially edentulous maxilla. (d) previously placed plates and screws removed. (e) Intra-operative view showing reconstructed maxillary alveolus with screws

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After due healing, additional rib bone grafts along with rbBMP2 (recombinant bone morphogenetic protein-2) was placed. The graft was secured with screws. Bone formation was adequate. Later, dental implants were planned for the missing maxillary teeth. Under general anesthesia, the Nobel Biocare dental implants (6) were placed as per standard protocol. Additionally, while closing with sutures, small doses of rhBMP2 were placed to accelerate osteointegration. Within three months, there was adequate osteointegration. Later, superstructures were fabricated and placed [Figure 3] and [Figure 4].
Figure 3: (a and b) Pre-operative CT, OPG showing reconstructed maxillary alveolus with screws. (c) Intra-operative view showing dental implants placed in maxilla. (d) Immediately after suturing

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Figure 4: (a) OPG taken after 3 months showing dental implants with complete Osseo integration. (b) Intra oral view showing cementation of implant supported ceramic prosthesis

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The patient was followed for about 25 months and there has been no abnormality noted till date.


   Discussion Top


The dental implants and related surgical procedures like maxillary sinus augmentation are known to predispose to fungal infection. The placement of dental implants, bone augmentation, and sinus lift surgeries are known to be safe surgical procedures, albeit with low prevalence of complications. All surgical procedures have inherent potential and risk to develop complications such as pain, infection, extreme bone loss, need for additional surgery, prolonged recovery, and nutritional disorders. In our present case, the removal of the cyst and subsequent infection complication related to healing possibly has resulted in this condition.[1],[6]

Clinically, non-invasive forms of fungal infections, associated with maxillary sinus called “fungal balls” are often self-limiting entities. This entity is often associated with local predisposing factors such as local tissue hypoxia or exposure to massive fungal load. Such sinus fungus balls, on a longer term, when situation is favorable, could possibly turn invasive and erode the sinus wall.[6] This could lead to facial pain or obstruct the sinus precipitating other secondary infections. In our present case, possibly the rhinosporidial fungal ball, may be a result of poor local tissue hypoxia resulting due to surgical site complication.[6],[7] The late diagnosis could be a possibility, which could, as in the present case lead to widespread local destruction. Prior researchers have identified that as the organisms in these balls are localized outside the mucosa, they are rendered non-viable for culture. Additionally, fungal culture procedures involve extended durations and carry very low diagnostic sensitivity. Probably in our present case too, this might have contributed to the delayed identification of the species. When the fungal ball turns invasive, widespread destruction along the local cancellous maxillary bone could have occurred.[7]

The complete treatment and removal of the fungal entities probably led to the success of the second surgery. The rapid clearing of tissues and reconstruction, in the presence of fresh bleeding brought in several blood borne progenitor cells to the maxilla, which was further stimulated by rhBMP-2 to differentiate into bone forming osteoblasts. This later gave rise to a good quality and quantity of alveolar bone. Use of rhBMP2 to induce neo-bone formation in critical sized defects and for increasing the alveolar height of residual ridges for dental implants is normal clinical protocol.[8],[9] On this bone, the dental implants were placed, which survived and readily osteointegrated.

In the present case, it is possible that the healing following cystic nucleation was not quite successful, possibly leading to subsequent local fungal ball infection. Subsequent spread of this infection possibly has eroded the remaining maxilla. This residual infection probably has caused the subsequent rejection of the graft. It is also possible that the surgery lead to infection or already the sinus was infected by fungus. Although the exact factors of the infection in this patient were hard to distinguish, in absence of concrete evidence, both conditions need to assume to have contributed to the problem.


   Conclusion Top


To the best of our knowledge, this is the second case of rehabilitation rehabilitation of an edentulous maxillary quadrant with dental implants after reconstruction in a known case of widespread fungal infection. The surgical treatment for such fungal infection, with reconstruction as well as dental implant placement produced good results, and no recurrence was observed.

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.
Anehosur V, Agarwal SM, Joshi VK, Anand J, Krishnamurthy K. Incidence and treatment protocol for maxillofacial fungal osteomyelitis: A 12 year study. J Oral Maxillofacial Surg 2019. doi: 10.1016/j.joms. 2019.06.187.  Back to cited text no. 1
    
2.
Kellesarian SV, Javed F, Romanos GE. Osteomyelitis arising around osseointegrated dental implants: A systematic review. Implant Dent 2018;27:1-10.  Back to cited text no. 2
    
3.
Sohn D, Lee J, Shin H, Choi B, An K. Fungal infection as a complication of sinus bone grafting and implants: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;107:375-80.  Back to cited text no. 3
    
4.
Vereanu AD, Savu MA, Patrascu E, Sarafoleanu C. Transnasal endoscopy – Evaluation and treatment method for patients with sinus lift and dental implants indications. Rom J Rhinol 2015;5:179-84.  Back to cited text no. 4
    
5.
Colletti G, Felisati G, Biglioli F, Tintinelli R, Valassina D. Maxillary reconstruction and placement of dental implants after treatment of a maxillary sinus fungus ball. Int J Oral Maxillofac Implants 2010;25:1041-4.  Back to cited text no. 5
    
6.
Prasad KC, Prasad SC, Mouli N, Agarwal S. Osteomyelitis in the head and neck. Acta Oto-laryngoloica 2007;127:194-205.  Back to cited text no. 6
    
7.
Jiang R, Huang W, Liang K. Characteristics of sinus fungus ball: A unique form of rhinosinusitis. Clin Med Insights Ear Nose Throat 2018;11:1-12.  Back to cited text no. 7
    
8.
Balaji SM. Use of recombinant human bone morphogenetic protein (rhBMP-2) in reconstruction of maxillary alveolar clefts. J Maxillofacial Oral Surg 2009;8:211-7.  Back to cited text no. 8
    
9.
Balaji SM. Augmentation of residual alveolar bone height with tissue engineering for dental implant placement. Indian J Dent Res 2014;25:410-2.  Back to cited text no. 9
[PUBMED]  [Full text]  

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Correspondence Address:
Dr. S M Balaji
Department of Oral and Maxillofacial Surgery, Balaji Dental and Craniofacial Hospital, 30, KB Dasan Road, Teynampet, Chennai - 600 018
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_711_19

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    Abstract
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