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Table of Contents   
CASE REPORT  
Year : 2013  |  Volume : 24  |  Issue : 5  |  Page : 639-641
Alternaria alternata infection associated osteomyelitis of maxilla: A rare disease entity


1 Department of Oral and Maxillofacial Surgery, Institute of Technology and Science, Ghaziabad, Uttar Pradesh, India
2 Department of Microbiology, Institute of Technology and Science, Ghaziabad, Uttar Pradesh, India

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Date of Submission15-Feb-2013
Date of Decision18-Feb-2013
Date of Acceptance28-Mar-2013
Date of Web Publication21-Dec-2013
 

   Abstract 

Alternaria alternata is one of the rarest fungi associated with paranasal sinusitis. Alternaria species are pigmented (also known as dematiaceous or phaeoid) filamentous fungi, which are well-known soil saprophytes and plant pathogens that infrequently cause infection in humans mainly, cutaneous lesions. We present a case of osteomyelitis of maxilla caused by a rare fungus- A. alternata in a diabetic patient with poor glycemic control who was successfully treated with antifungal and surgical debridement over the period of 6 months.

Keywords: Alternaria alternata , diabetes mellitus, maxilla, osteomyelitis, paranasal sinusitis

How to cite this article:
Chhabra V, Rastogi S, Barua M, Kumar S. Alternaria alternata infection associated osteomyelitis of maxilla: A rare disease entity. Indian J Dent Res 2013;24:639-41

How to cite this URL:
Chhabra V, Rastogi S, Barua M, Kumar S. Alternaria alternata infection associated osteomyelitis of maxilla: A rare disease entity. Indian J Dent Res [serial online] 2013 [cited 2018 Sep 19];24:639-41. Available from: http://www.ijdr.in/text.asp?2013/24/5/639/123420
Fungal infections of the paranasal sinuses is being recognized and reported with an increasing frequency. [1] Currens et al. reported the detailed review of the literature of fungal sinusitis. [2] The first well-documented case of invasive fungal sinusitis was reported by Oppe in 1897 and was treated by surgical debridement and irradiation. [3]

Alternaria alternata is one of the rarest fungi associated with paranasal osteomyelitis. [4] The clinical spectrum of the disease includes the following: Hypersensitivity pneumonitis, granulomatous lung disease, bronchial asthma, paranasal sinusitis with and without osteomyelitis, allergic sinusitis and rhinitis, keratitis, peritonitis and cutaneous and subcutaneous deep-tissue infection. Most infections in humans occur in patients with immunologic impairment and far less often in healthy individuals. [5] Diabetes mellitus, considered by some investigators to represent an immunodeficient state is frequently cited in association with fungal sinusitis.

We recount an unusual case of maxillary sinusitis caused by an infrequent fungus- A. alternata in a diabetic patient with poor glycemic control mimicking periodontal infection.


   Case Report Top


A 55-year-old female patient reported to the department of oral and maxillofacial surgery with a chief complaint of pain on the left side of her face and post-nasal discharge since 15-16 months. She gave a history of extraction of left upper posterior teeth at a private dental clinic after which she developed pain and post nasal discharge. Patient was known hypertensive and diabetic under medication.

Pain was dull, continuous with intermittent rise in intensity 2-3 times a day. Extraoral examination revealed bilateral tenderness over malar region with no associated nerve paresthesia or swelling. Intraorally, a 2 × 1 cm fistulous opening in the midline of the anterior palate with no pus discharge was present [Figure 1].
Figure 1: Preoperative intraoral photograph with fistulous opening in the anterior mid palate

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Systemic examination and lab investigations were within normal limits except blood sugar and blood pressure. No immunologic work-up was performed.

Computed tomography (CT) scans revealed partial obliteration of the maxillary sinuses with destruction of the walls of the antrum [Figure 2].
Figure 2: Preoperative axial computed tomography scan with obliteration of the maxillary sinuses as well as buckling of the nasal septum with thickening of the sinus lining in the posterolateral aspect of the sinus

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A provisional diagnosis of chronic sinusitis was reached and the patient was kept on antral triage for 10 days. Patient came back to the department with aggravated signs and symptoms and increased post nasal discharge and nasal stuffiness. On examination, mobility of upper anteriors was elicited along with the loss of periodontal attachment in relation to 11, 21 and 22 with increased size of fistula. A 3D CT presented with massive osteolysis of the hard palate [Figure 3]. A swab culture was obtained from the fistula and was found to be positive with Periodic Acid Schiff, which indicated a fungal etiology. Oral antifungals were given for 1 month (Tablet. Fluconazole 150 mg OD) and significant improvement was seen with reduction of the size of the fistula.

Surgical debridement was carried out under general anesthesia. Amphotericin B administered in post-operative phase for 10 days intravenously. The recovery was uneventful and patient was discharged with oral antifungals (Tablet. Fluconazole 150 mg OD). Patient is on regular follow-up without any further complaints.
Figure 3: 3D reconstruction images with massive osteolysis of the hard palate

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Histopathological examination of the multiple serial sections of antral bone was prepared and stained with hematoxylin and eosin for cellular examination and PAS for fungal detection, which revealed septate hyphae. Mycological examination of the samples subjected to 40% KOH mount was positive for branched septate phaeiod hyphae suggestive of phaeohyphomycosis [Figure 4]. Grayish white wooly colonies appeared within 3 days of inoculation in Sabourauds dextrose agar, which assumed black pigment on further inoculation. Microscopically, Lactophenol cotton blue stain showed branches of septate hyphae with multiple chains of large, brown, muriform conidia with transverse and longitudinal septations. These findings were indicative to A. alternata.
Figure 4: Depicts fungal hyphae in 40% KOH mount

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   Discussion Top


Allergic fungal sinusitis is most common form of fungal sinusitis and it is particularly common in warm, humid climates. Incidence in India has been recorded up to 51% of patients with chronic rhinosinusitis.

Alternaria is a ubiquitous fungus regarded as non-pathogenic contaminants of clinical specimen unless isolated on repeated culture and correlated with clinical findings. Only six species are described as human pathogens in the  Atlas More Details of clinical fungi: A. alternata. Alternaria chlamydospora, Alternaria dianthicola, Alternaria infectoria, Alternaria tenuissima and Alternaria longipes. Three forms of alternariosis can be distinguished: (1) exogenous, superficial form caused dermatopathy; (2) exogenous, unilocular form of traumatic origin; (3) endogenous, multilocular, disseminated form. [6] In our case, the patient was symptomatic after undergoing an extraction of tooth in posterior left maxilla, thereby, of traumatic origin. The infections are not contagious and in immunosupressed individual development of invasive and fulminant disease is much more common than in immunocompetent individual. [6] Current dogma holds that diabetes significantly predisposes patients to fungal infections.

A total of 17 cases of invasive and non-invasive sinusitis caused by Alternaria spp. have been reported since 1977 but, all the cases were associated with immunocompromised state of the patients such as corticosteroid therapy, leukemia and organ transplant. Moreover, all cases were linked with cutaneous manifestations none of the cases associated with oral cause. Our case is first ever case reported in the oral and maxillofacial sciences literature.

The mechanism, however, is not clearly understood and it was found that blood cells involved in immunity of chronic rhinosinusitis showed exaggerated responses to A. alternata, which is commonly airborne. A recent study revealed that Alternaria not only interfere with the nerve impulses in the nasal passages, but also colonizes there, resulting in a full-blown infection. This anomalous response to ubiquitous environmental fungi might explain the persistent airway inflammation in chronic rhinosinusitis patients. [7]

Radiographically, there usually, is a frequent nasal component. The majority of the sinuses show near-complete opacification and are expanded. Non-contrast CT demonstrates hyperattenuating allergic mucin within the lumen of the paranasal sinuses. In our case, mottled erosion of left nasal wall with osteolysis and sequestration extending from frontonasal junction until nasomaxillary junction, anterior to nasolacrimal canal was seen. Mild hyperattenuating soft-tissues indicative of mucosal thickening were seen involving the sinuses bilaterally, which seem to be consistent with a chronic invasive nasomaxillary infection.

The treatment of alternariosis and appropriate antifungal dosages has not been standardized and these issues have not been specifically addressed in the literature. Itraconazole is the treatment of choice as the renal toxicity and concerns about the necessity of extended hospitalization, makes amphotericin B the second choice of drug with surgical debridement/resection to achieve clinical resolution without relapse. [5]


   Conclusion Top


Clinicians should consider and include this species as differential diagnosis in cases of chronic sinusitis that is non-responsive to usual management. Only histopathological examination cannot substantiate and should always be aided by microbiological identification as early diagnosis is essential in order to avoid high morbidity associated with the destructive disease and to instigate treatment before irreversible conditions arise.

 
   References Top

1.Adam RD, Paquin ML, Petersen EA, Saubolle MA, Rinaldi MG, Corcoran JG, et al. Phaeohyphomycosis caused by the fungal genera Bipolaris and Exserohilum. A report of 9 cases and review of the literature. Medicine (Baltimore) 1986;65:203-17.  Back to cited text no. 1
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2.Currens J, Hutcheson PS, Slavin RG, Citardi MJ. Primary paranasal Aspergillus granuloma: Case report and review of the literature. Am J Rhinol 2002;16:165-8.  Back to cited text no. 2
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3.de Carpentier JP, Ramamurthy L, Denning DW, Taylor PH. An algorithmic approach to aspergillus sinusitis. J Laryngol and Otol 1994;108:314-8.  Back to cited text no. 3
    
4.Washburn RG, Kennedy DW, Begley MG, Henderson DK, Bennett JE. Chronic fungal sinusitis in apparently normal hosts. Medicine (Baltimore) 1988;67:231-47.  Back to cited text no. 4
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5.Lyke KE, Miller NS, Towne L, Merz WG. A case of cutaneous ulcerative alternariosis: Rare association with diabetes mellitus and unusual failure of itraconazole treatment. Clin Infect Dis 2001;32:1178-87.  Back to cited text no. 5
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6.Vennewald I, Wollina U. Cutaneous infections due to opportunistic molds: Uncommon presentations. Clin Dermatol 2005;23:565-71.  Back to cited text no. 6
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7.Chakrabarti A, Sharma SC, Chandler J. Epidemiology and pathogenesis of paranasal sinus mycoses. Otolaryngol Head Neck Surg 1992;107:745-50.  Back to cited text no. 7
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Correspondence Address:
Sanjay Rastogi
Department of Oral and Maxillofacial Surgery, Institute of Technology and Science, Ghaziabad, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.123420

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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