Use of septal button in hard palate perforation owing to mucormycosis: a case reportYusuf Çağdaş Kumbul , Vural Akın , Hasan Yasan , Mehmet Emre Sivrice

Department of Otorhinolaryngology Head and Neck Surgery, Suleyman Demirel University Research and Training Hospital, Isparta, Turkey

ABSTRACTMucormycosis (MM) is a fatal opportunistic fungal infection that usually occurs in immunosuppressed patients and in patients with uncontrolled diabetes mellitus (DM). Defects owing to MM (eg, hard palate) increase morbidity. Therefore, these defects must be treated with the most appropriate method. A 65-year-old female patient presented to our clinic with complaints of malodorous discharge from the palate and nose and right-sided headache for 1 week. She had DM and was not compatible with antidiabetic treatment. Samples were taken from the infected palate for histopathological examination and fungal culture. Rhizopus was detected on the fungal culture. After extensive debridement, the hard palate defect was closed with nasal septal button (NSB). Obturator prostheses, created by taking an impression of the palatal defects, are used to close palatal perforations. NSB is an alternative to close these perforations, because NSB is easy to apply and can reduce morbidity. Keywords: Debridement, mucormycosis, nasal septal button, palate, rhizopus

Introduction

Mucormycosis (MM) is a fatal opportunistic fungal infection that usually occurs in immunosuppressed patients and in pa-tients with uncontrolled diabetes mellitus (DM) (1). The mem-bers of the order Mucorales, such as Rhizopus, Absidia, and Mucor, are responsible for it (1-3). The most common form is rhinocerebral MM (1, 2, 4). which primarily affects the pal-ate and paranasal sinuses. If the infection is not controlled, it spreads to the orbit and intracranial areas. Generally, emergen-cy surgical debridement combined with amphotericin B ad-ministration is the treatment of choice (3, 4).

A nasal septal button (NSB) is used as an alternative to surgi-cal repair for treating septum perforation. NSBs are made of silicone, plastic, or acrylic. In addition to the one-piece types, there are 2 separate leaf-shaped types that can be mounted at the lesion level. They are preferred by patients who cannot tolerate surgery or who do not want surgical treatment owing to the advantages of NSBs, such as easy application, no inter-ventional procedure, and being able to have it fixed with local anesthesia as an outpatient (5).

In this article, we aim to discuss the clinical features of a pa-tient with MM and our experience of using NSB for treating the hard palate perforation in light of the literature.

Case Presentation

A 65-year-old female patient presented to our clinic with com-plaints of malodorous discharge from the palate and nose and right-sided headache for 1 week. The patient had no medical history of surgery and trauma; however, she had DM and was not compatible with antidiabetic treatment. According to the patient, in the previous month, her fasting blood glucose levels were between 400 and 500 mg/dL. On physical examination, a 5 × 3-mm perforation was observed on the right side of the mid-line of the hard palate (Figure 1). Purulent discharge was present at the perforation margins. Nasal examination revealed that the right lower turbinate was black, and malodorous discharge came from the right nasal cavity. Ear, nasopharynx, oropharynx, and larynx examination results were normal. The complete blood count test results were normal, and the C-reactive protein level was 56.7 mg/L. The fasting blood glucose level was 201 mg/dL. Blood biochemistry parameters were within the normal range. Liver and kidney function tests and electrolytes were also nor-mal. Computed tomography revealed opacity in the right para-nasal sinuses (except for the frontal sinus). Spread in the pter-ygopalatine fossa and the right orbit was suspected (Figure 2). Samples were taken from the infected palate for histopatholog-ical examination and fungal culture. Rhizopus was detected in the fungal culture. Histopathologic examination revealed fungal

Corresponding Author: Vural Akın, vuralakin92@gmail.com Received: December 8, 2020 Accepted: April 2, 2021Available online at www.b-ent.be

DOI: 10.5152/B-ENT.2021.20216

CASE REPORTGENERAL OTOLARYNGOLOGY & HEAD AND NECK SURGERY

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Cite this article as: Kumbul YÇ, Akın V, Yasan H, Sivrice ME. Use of septal button in hard palate perforation owing to mucormycosis: a case report. B-ENT 2021; 17(1): 49-52.

CC BY 4.0: Copyright@Author(s), “Content of this journal is licensed under a Creative Commons Attribution 4.0 International License.”

hyphae congruent with mucor and necrotic tissue. We decided to perform emergency surgical debridement. Wide surgical de-bridement was performed in the right maxillary sinus, right lower turbinate, medial wall of the right maxillary sinus, and sphenoid and ethmoid sinuses using the Caldwell-Luc technique com-bined with endoscopic sinus surgery (Figures 3 and 4). NSB was used on the palate to prevent oronasal reflux during the post-operative period (upper layer facing nasal cavity and lower layer facing oral cavity) (Figures 5-8). After receiving amphotericin B and ampicillin-sulbactam treatment for a month postopera-tively, the patient was discharged with the NSB. We suggested

fixing the hard palate with a local flap 4 weeks later. The patient was being followed-up strictly without MM relapse at the time of writing this article. Informed consent was obtained from the patient.

Discussion

The most important aspect in the prognosis of MM is early di-agnosis and treatment. Survival has been reported to increase with decrease in time between appearance of the symptoms and beginning of treatment (4). Surgical and medical treat-ments are usually combined (2, 4). Surgical treatment involves debridement of necrotic tissues (2, 4). External approach-es such as lateral rhinotomy and endoscopic approaches are used for debridement.6 With the use of amphotericin B, MM mortality has decreased significantly. Surgical debridement is also important for treating MM (2, 4). The vascular involvement weakens blood circulation to the infected areas; thus, intrave-nous (IV) amphotericin B administration is difficult because it cannot penetrate the infected tissue. Therefore, topical am-photericin B may be added to the treatment (4). In addition, the disease may not be controlled by a single debridement. Re-current debridements may be required (2).

Main Points:

• Mucormycosis is a life-threatening and opportunistic fungal infection, and treatment for mucormycosis should be started as soon as possible.

• If mucormycosis treatment is delayed, bone defects may oc-cur in the palate and paranasal sinuses.

• Bone defects must be treated to reduce morbidity.• Nasal septal button is an alternative treatment method that

can be used to close bone defects in the hard palate.

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Kumbul et al. A distinct indication of septal button B-ENT 2020; 17(1): 49-52

Figure 1. Perforation of the hard palate before the debridement Figure 3. View of the hard palate after the debridement

Figure 2. Computed tomography of the preoperative paranasal sinus in coronal section

Figure 4. Computed tomography image of the paranasal sinus taken after the debridement

In our case, after the diagnosis of MM, emergency surgical de-bridement was performed. After that, amphotericin B treat-ment was started. The blood glucose levels were checked strictly and maintained in the normal range, thereby, eliminat-ing the predisposing factor uncontrolled DM. After the surgical debridement, the defect in the hard palate was left to second-ary healing, and the infection was expected to clear completely for further surgical repair and the surgical area was expected to become healthy. During this process, the palatal defect was closed using an NSB.

The palate is one of the most affected sites in rhinocerebral MM (3, 4). Perforation on the palate might occur because of necrosis caused by vascular involvement, which was observed in our patient. In these cases, the defect caused by the in-fection becomes larger because of surgical debridement. The repair of these defects is important to reduce morbidity. Ob-

turator prostheses are created by taking an impression of the palatal defects (7). However, owing to the definite diagnosis of MM, emergency surgery was performed in our patient. There-fore, palatal impressions could not be taken. In our case, we waited until the infection was under control before attempt-ing surgical repair of the hard palate perforation. During this process, NSB was applied to prevent oronasal reflux and to support nutrition and speech function. An obturator was not needed due to good patient compliance and functional satis-faction of the NSB.

In the literature, there are cases in which NSBs have been used in different anatomical regions as in our patient. Mirza et al. (8) performed total tracheoesophageal puncture for speech function after total laryngectomy and closed the nonfunc-

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Figure 5. Nasal septal button (EON Meditech, Surat, Gujarat, India)

Figure 7. Implementation of the nasal septal button to the hard palate

Figure 8. View of the nasal septal button from right nasal cavity

Figure 6. Implementation of the nasal septal button to the hard palate

tional fistula as a result of wound infection with an NSB, pre-venting aspiration of oesophageal contents. Ünsal et al. (9) reported 4 such cases. Khan et al. (10) reported 2 cases that developed pharyngocutaneous fistula after total laryngecto-my; they closed the fistula with an NSB. In addition, drug abuse may cause perforations in the oral and nasal cavity (11, 12). Tri-marchi et al. (12) reported that they closed a perforation with NSB in patients who developed palate perforation secondary to cocaine use. Consistent with the findings of these cases in the literature, in our case, NSB was well tolerated, and the ex-pected functional results were achieved successfully.

The main advantage of NSB is its easy application. In addition, it prevents the escape of nutrients and saliva into the nasal cavity and therefore plays an important role in the prevention of secondary infections. NSB may be an alternative to tem-porary repair of appropriate-sized hard palate perforations or permanent closure in high-risk surgical patients (elderly pa-tients, patients with severe heart failure, or patients with se-vere pulmonary diseases). However, the blood circulation of the tissue between the leaves of an NSB can be impaired. We believe that this will be observed mostly in the initial days after NSB application. To prevent this, blood circulation of the tissue should be strictly controlled. Therefore, we recommend strict observation of this area during the initial days. If there is a risk of dislocation of NSB, it may be possible to fix it with a suture to prevent aspiration.

This is the first case in which NSB was used for palate per-foration due to MM. We believe that it will contribute to the treatment in future cases. NSB, which has traditionally been used in the treatment of septum perforations, can be used for closure of wall defects between 2 passages, as in our case. The main advantages of NSB are its easy installation and reduced morbidity.

Informed Consent: Written informed consent was obtained from the patients who agreed to take part in the study.

Peer-review: Externally peer-reviewed.

Author Contributions: Concept –Y.Ç.K., V.A.; Design –Y.Ç.K., V.A.; Su-pervision – Y.Ç.K., H.Y., M.E.S.; Resources – Y.Ç.K.; Materials – V.A..; Data

Collection and/or Processing – V.A.; Analysis and/or Interpretation – H.Y., M.E.S.; Literature Search – Y.Ç.K., V.A.; Writing Manuscript – Y.Ç.K., V.A.; Critical Review – H.Y., M.E.S.

Conflict of Interest: The authors have no conflict of interest to de-clare.

Financial Disclosure: The authors declared that this study has re-ceived no financial support.

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