Case Report
Paranasal Aspergillus Fungal Infection in Immune Compromised and Uncontrolled Diabetic Patients: A Report of 5 Cases along with Review of Literature
Harish Saluja1*, B M Rudagi1, Shivani Sachdeva2, Semmit Shah1, Anuj Dadhich1, Parul Tandon1
and Vinayak More1
1Department of Oral & Maxillofacial Surgery, Rural Dental College, India
2Department of Periodontics, Rural Dental College, India
*Corresponding author: Harish Saluja, Department of Oral & Maxillofacial Surgery, Rural Dental College, Loni Tal, Rahata, Ahmednagar, Maharashtra – 413736, India
Published: 06 Dec, 2017
Cite this article as: Saluja H, Rudagi BM, Sachdeva
S, Shah S, Dadhich A, Tandon P,
et al. Paranasal Aspergillus Fungal
Infection in Immune Compromised
and Uncontrolled Diabetic Patients: A
Report of 5 Cases along with Review of
Literature. Clin Surg. 2017; 2: 1805.
Abstract
Aspergillus is the most common fungal pathogen in sinus disease, with the maxillary sinus being
predominantly involved. Fungal infections are common in several conditions that lower the
immunity of the patient like uncontrolled diabetes, long term antibiotic & steroid therapy, radio,
chemotherapy, immunosuppressive treatment & immunodeficient diseases. Among these diabetes
is the condition which is increasing day by day in India and is one of the predisposing factor for
head & neck fungal infection especially aspergillus fungal infection. Diabetes has emerged as a major
healthcare problem in India. According to Diabetes Atlas published by the International Diabetes
Federation (IDF), there were an estimated 40 million persons with diabetes in India in 2007 and
this number is predicted to rise to almost 70 million people by 2025. The countries with the largest
number of diabetic people will be India, China and USA by 2030. Here we present 5 cases of invasive
aspergillus fungal infection of paranasal sinus region in uncontrolled diabetic patients, along with
review of literature emphasizing much more incidence of aspergillus infection in facial region in
patients suffering from uncontrolled diabetes. This review will help us in better understanding of
maxillary sinus fungal infections.
Keywords: Diabetes; Fungal infection; Aspergillosis; Paranasal; Maxillary sinus
Introduction
Head & Neck aspergillosis, particularly of the maxillary antrum, has been reported as occurring in both healthy and immunologically compromised individuals. It is one of the most rapidly progressing and lethal form of infection. Aspergillosis occur in both invasive and noninvasive forms, the former is more likely to occur in patients with debilitating illnesses and is a major cause of morbidity & mortality in immunocompromised patients [1,2]. In the immunocompromised patient, infection mortality has risen to 50% in many institutions [3]. Fungal infections are common in several conditions that lower the immunity of the patient like uncontrolled diabetes, long term antibiotic & steroid therapy, radio, chemotherapy, immunosuppressive treatment & immunodeficient diseases [4]. Among these diabetes is the condition which is increasing day by day in India and is one of the predisposing factor for head & neck fungal infection especially aspergillus fungal infection. Diabetes has emerged as a major healthcare problem in India. According to Diabetes Atlas published by the International Diabetes Federation (IDF), there were an estimated 40 million persons with diabetes in India in 2007 and this number is predicted to rise to almost 70 million people by 2025. The countries with the largest number of diabetic people will be India, China and USA by 2030 [5,6]. Here we present 5 cases of aspergillus fungal infection of paranasal sinus region in diabetic patients, along with review of literature emphasizing much more incidence of aspergillus infection in facial region in diabetic patients.
Case Presentations
Case 1: A of 70 year-old male known case of diabetes, reported with chief complaint of pain in
left maxillary posterior region since last 6 months, nasal congestion, headache & numbness on upper
lip region since last 2 months. Intraoral examination showed edentulous necrotic bone involving
hard palate & upper alveolus completely on left side & up to premolar region of right side measuring
approximately 8 cm in length. Even some perforations were also present in palate (Figure 1),.
Radiologic examination revealed obliteration of left maxillary sinus
showing involvement of left pre-maxilla region with extension into
left maxillary sinus & left Zygomatic region. Lateral wall of left side
maxillary sinus was completely eroded. The radiologic appearance of
the paranasal sinus is a focal radio dense shadow in the sinus. The
clinical & radiological diagnosis was invasive fungal infection &same
was confirmed as Aspergillosis in histology. Patient sugar levels were
controlled & the patient was operated for surgical debridement &
curettage of necrotic bone. Whole of the necrotic palate was detached
from surrounding attachments & removed. Curettage was done
up to left Zygomatic arch region, lining of the sinus was removed
completely, even some part of Zygomatic bone was curetted out.
The operated area was irrigated with combination of Amphotericin
powder & normal saline. Patient was kept on injection Amphotericin
B 25 mg in 500 cc of saline twice along with antibiotics for next 5
days & the blood urea & creatinine levels were monitored regularly.
From 6th post operative day patient was shifted to tablet Fluconazole
150 mg OD. At the same time operated site was irrigated twice daily
with normal saline mixed with Amphotericin powder. After 6 months
oral rehabilitation was done with obturator cum complete denture
prosthesis to improve quality of patient’s life.
Case 2: This case was of 40 year male patient reporting with
complaint of swelling on right side of face & upper posterior
mobile teeth since last 4 months. On intraoral examination exposed
alveolar bone was there in right maxillary posterior region with
pus discharge from the same site (Figure 2). Patient was a known
case of uncontrolled diabetes since last 3 years & was not taking
any medication. Histopathological diagnosis for the lesion was
aspergillosis & lesion was also involving maxillary sinus. After sugar
control complete lesion was curetted from maxillary sinus along
with necrotic alveolar part, under general anesthesia. Postoperatively
patient was managed by systemic antifungal drugs.
Cases 3-5:
They were also diabetic patients & suffered from
aspergillus fungal infection of paranasal sinus region (Figures 3-7).
These three patients were suspected of odontogenic origin. All 3
patients were managed surgically along with postoperative systemic
antifungal therapy and treatment for the odontogenic infection was
also given.
Figure 1
Figure 2
Figure 3
Figure 4
Figure 5
Discussion
Mycosis of paranasal sinus has become a more common disease
[7]. Of all the fungal infections aspergillosis is third commonest
fungal disease in human beings. Aspergillus is the most common
fungal pathogen in sinus disease, with the maxillary sinus being
predominantly involved. The recent rise in mycotic nasal & paranasal
infections is due to both improved diagnostic research and an
increase of the conditions that favour fungal infections, which
increase in immunocompromised patients because of changing life styles etc [8,9]. The priest Botanist Micheli first described Aspergillosis
(1729) [10]. In 1847 Sluyter first identified Aspergillus in a woman
dying of pulmonary infection [11]. First report of involvement of
maxilla was by Morrel Mackenzie (1893) while Zorinko (1893) [12]
described maxillary sinus infection. The causative organism is mainly
spore forming filamentous fungus which occurs as a saprophyte in
soil, mud on seeds, fruit, grain seeds & plants. These colonies grow
in a wide range and can grow in temperature up to 50°C [13]. There
are several species of aspergillus which are associated with human
diseases but Aspergillus fumigates & Aspergillus flavus being most
dominant. A. flavus is most destructive one because of potent toxins
[13]. Other reason for its growth was hypoxia & anaerobic conditions
because of sinus obstruction. The fungus itself does not contain
any chlorophyll & therefore does not require any light for growth
but it needs host supplying it with nutrient in the form of glucose,
nitrogen, sulphur, phosphorous, potassium, calcium, magnesium and
iron [14]. Aspergillus in sinus appears as a Ball shaped mass which
contains Ca & P salts & therefore may sometimes mimics foreign
body on radiographs [14]. The etiopathogenesis of paranasal fungal
infection is a debatable topic, but there are three main accepted
theories including odontogenic, aerogenic, and mixed origins. The
odontogenic school of thought maintains that the pathogenesis is
based on an initial colonization of the maxillary sinus by means of
iatrogenic oral-antral communication. This theory holds that the
Zinc Oxide which can be found in endodontic sealers paralyzes
the epithelial cilia or causes edema and hyperemia of soft tissue,
affecting Schneiderian membrane epithelial function [15]. Aspergillus
of paranasal sinus occurs in 2 forms, noninvasive and invasive [4].
The first or noninvasive, type reassembles chronic bacterial sinusitis
with symptoms of unilateral nasal obstruction, pressure feelings
and drainage of foul, gelatinous substance. Radio graphically
usually documents only a single cloudy sinus. The invasive type is
more aggressive with signs of malignancy. This type might extend
to the orbit, cheek or adjacent sinus & may cause displacement or
bony erosion [4]|. The invasive type is mostly associated with some
immunocompromised condition. Hora differentiated Aspergillus
as infiltrating and non-infiltrating with incidence of bone invasion
as the primary difference [16]. At our institute during a period of 3
years from 2008-2011 we encounter 5 aspergillus patients involving
paranasal area & all the patients were suffering from long standing
diabetes. Literature also shows patients suffering from diabetes are
the main victims.
Involvement of paranasal sinuses may result in mucoid or purulent
nasal discharge, facial swelling, pain, tenderness, and fever, proptosis
of eye, sensory nerve disturbances, and epistaxis. In some cases even
sphenoid sinus also got involved and sometimes fungus invade the
orbit and optic nerve from the sphenoid sinus. A fatality rate of
16% has been reported in medical literature for uncontrolled fungal
infections [3]. Schubert et al. reported fatal hemorrhage in paranasal
aspergillus patient [17]. This fungus invades the arteries, form
thrombi with in blood vessel and cause necrosis of hard & soft tissues.
Thrombosis of the internal maxillary artery or descending palatine
artery from a fungal infection could result in necrosis of a portion of
the maxilla. After entering the vessels; fungus can sometimes involve
sinuses & orbit [18]. Uncontrolled diabetes mellitus can alter the
normal immunologic response of patient to infections. In these types
of patients because of decreased granulocytic count phagocytic ability
becomes low with altered polymorph nuclear response [18]. Diabetes
mellitus is a chronic disorder that affects a large segment of human
population & is a major health problem. Immunologic research
demonstrated several defects in host immune defense mechanism
in diabetic subjects. Phagocytic capabilities of PMN are adversely
affected by hyperglycemia in rat module. Several PMN defects occur
in diabetic subjects, including impaired migration, phagocytosis,
intracellular killing & chemotaxis, which may be due to decreased
PMN membrane fluidity. Generalised immunologic defects such as
those raise the suspicion that diabetic patients may be at an overall
increased risk of infection [19]. Invasive mycotic infections present
a problem in therapy but have a fairly good prognosis if treated with
radical local surgery along with control of underlying disease. In our
all 5 cases surgical debridement was done and along with this patient
was on systemic antifungal therapy and even in one case antifungal
was used for local delivery to operating area.
Figure 6
Figure 7
Conclusion
Diabetic patients are more prone to paranasal fungal sinus
infections, in which one of the main etiology can be of odontogenic
origin, so dental problems should be considered seriously in diabetic
patients as these can turn up into these types of sequels. Diabetic
or any immunocompromised patient having necrosed and exposed
maxillary bone with tooth extraction history in the same region
should alert a clinician possibility of some fungal infection.
Three principles should be followed to treat diabetic patients
having paranasal fungal infections.
Firstly, control the underlying diabetes by suitable measures.
Secondly, remove affected bone along with sinus membrane. Lastly
Systemic use of antifungal drugs, even some times antifungal drugs
can be used for local irrigation to the affected area.
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