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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 29  |  Issue : 4  |  Page : 317-320

Secondary Manifestation of Allergic Bronchopulmonary Aspergillosis on Tongue: A Rare Case


1 Department of Oral Medicine and Radiology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India
2 Department of Oral and Maxillofacial Pathology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab, India

Date of Submission26-Oct-2017
Date of Acceptance16-Jan-2018
Date of Web Publication15-Feb-2018

Correspondence Address:
Dr. Bhawandeep Kaur
Department of Oral Medicine and Radiology, Genesis Institute of Dental Sciences and Research, Ferozepur, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_103_17

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   Abstract 


Bronchopulmonary aspergillosis (BPA) most commonly complicates the course of bronchial asthma and cystic fibrosis. The clinical presentation of allergic BPA (ABPA) is very similar to pulmonary tuberculosis, and signs and symptoms are usually poorly-controlled asthma, hemoptysis, and expectoration of mucus plugs, malaise, and fever. Intraoral presentation of aspergillosis is relatively a very rare condition, and the most commonly affected sites are the gingivae, followed by maxillary sinus, hard plate, soft plate, and tongue. Here, we are presenting a rare case report of nonhealing ulcer on right lateral border of tongue secondary to ABPA.

Keywords: Allergic bronchopulmonary aspergillosis, fungal, intraoral, tongue


How to cite this article:
Kaur B, Padda S, Bhandari R, Brar R. Secondary Manifestation of Allergic Bronchopulmonary Aspergillosis on Tongue: A Rare Case. J Indian Acad Oral Med Radiol 2017;29:317-20

How to cite this URL:
Kaur B, Padda S, Bhandari R, Brar R. Secondary Manifestation of Allergic Bronchopulmonary Aspergillosis on Tongue: A Rare Case. J Indian Acad Oral Med Radiol [serial online] 2017 [cited 2019 Aug 21];29:317-20. Available from: http://www.jiaomr.in/text.asp?2017/29/4/317/225463




   Introduction Top


Aspergillosis is predominantly an opportunistic fungal infection and commonly isolated in the lungs of immunocompromised patients.[1] Based on the manifestations, aspergillosis can be categorized under three principal entities: (1) invasive aspergillosis involving several organ systems (particularly pulmonary disease), (2) pulmonary aspergilloma, and (3) allergic bronchopulmonary aspergillosis (ABPA).[2] According to Denning et al. (2013), globally 4.8 million people are suffering from ABPA, but the prevalence of ABPA in asthmatic patients is only 2.5%.[2]

Intraoral manifestation of aspergillosis is relatively rare, as only approximately 25 cases are reported in literature.[3] The most commonly affected intraoral sites are gingivae, followed by maxillary sinus, hard and soft plate, and tongue. Out of all the three variants of the aspergillosis, primary invasive aspergillosis is the most common variant having intraoral manifestations.[3] Here, we are presenting a rare case report of nonhealing ulcer on right lateral border of tongue occurring secondary to ABPA. To the best of authors' knowledge, this is the first case report of oral manifestation of ABPA.


   Case Report Top


A 56-year-old male patient reported to the Department of Oral Medicine and Radiology with the chief complaint of pain and burning sensation on right posterior-lateral border of tongue associated with limited movement of tongue for the past 1 year. Since its onset, the patient has been undergoing treatment from local physician, but there has been no clinical improvement. His past medical history revealed history of dyspnea and cough with sputum discharge for the past 2 years and was under medication for the same. He was getting symptomatic relief, but no significant clinical improvement was achieved. Intraorally, on inspection, a solitary ulcer of about 1.5 cm in diameter on the right posterior-lateral border of tongue having slightly raised margins was seen. On palpation, ulcer was found to be tender, floor was slightly erythematous, base and margins were not indurated, and there was no discharge from the ulcer [Figure 1].
Figure 1: Preoperative intraoral photograph showing ulcer on right lateral border of tongue

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Complete blood investigation and chest X-ray were recommended for further evaluation. Complete blood investigation revealed high level of erythrocyte sedimentation rate, which was raised up to the level of 20 mm. Chest radiograph shows diffuse consolidations bilaterally with dilated bronchi [Figure 2]. Both these findings raised the suspicion of tuberculosis. However, smear from the nonhealing ulcer and sputum of the patient was negative for acid fast bacilli. Mantoux test was also negative for tuberculosis. Kveim test was also done to rule out the sarcoidosis. Further blood investigations revealed that the total serum immunoglobulin E (IgE) level and allergen-specific IgE level of Aspergillosis fumigatus were raised up to the level of 46.10 kUA/l. This level has been considered under the category of high level of allergen-specific antibody. Eosinophil count was also raised (1288 cu/mm). The allergen specific IgE level estimation is considered to be the useful test for the diagnosis of ABPA.
Figure 2: Pretreatment chest radiograph showing diffuse consolidations on right and left lungs with dilated bilateral bronchi

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Incisional biopsy was performed using Er:YAG laser with soft tissue tip (Syneron, 2940 nm, 3 W, 35 Hz, 0.8 mm) and sent to the Department of Oral and Maxillofacial Pathology for histological evaluation. The hematoxylin and eosin (H&E) stained section showed stratified squamous epithelium, which was disrupted at places overlying a fibrovascular connective tissue stroma. The connective tissue exhibited an area of circumscribed collection of epitheloid histiocytes and lymphocytes organized in the form of granuloma. Giant cells and caseous necrosis were absent. On Ziehl–Neelsen staining, Mycobacterium bacilli were not demonstrated, and on Periodic acid–Schiff staining, hyphae were not demonstrated. The above histopathological findings were suggestive of chronic granulomatous inflammatory pathology [Figure 3] and [Figure 4]. With no other evident organic cause, the diagnosis of nonhealing ulcer secondary to ABPA was given and the patient was subjected to treatment based on this diagnosis.
Figure 3: Histopathological picture depicting an area of circumscribed collection of epithelioid histiocytes and lymphocytes organized in the form of granuloma

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Figure 4: Complete healing of the intraoral lesion

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Treatment

According to the Infectious Diseases Society of America (IDSA) guidelines for aspergillosis,[4] the regimen followed under the guidance of Department of General Medicine was the systemic therapy of antifungal agent, i.e., oral itraconazole 100 mg twice daily, initially given for a period of 1 month. Oral prednisolone 30 mg/day for 15 days on alternate days for 6 weeks was prescribed, reducing 5 mg thereafter for every 2 weeks. No separate medication for the nonhealing ulcer was given. Patient was periodically followed-up and showed marked clinical improvement in local as well as systemic signs and symptoms. Clinical improvement was evident with a reduction in dyspnea, wheezing, cough, and nasal obstruction, and marked healing of intraoral ulcer [Figure 5]. This was accompanied by a reduction in total IgE and blood eosinophil count. Recovery was also noticeable on the X-ray chest, 6 months after diagnosis [Figure 5]. There was no clinical or laboratory evidence of adverse therapy-related effects. Based on the initial investigations and therapeutic treatment response, the provisional diagnosis of nonhealing ulcer secondary to ABPA was confirmed.
Figure 5: Post treatment chest radiograph after 6 months showing reduced consolidations on right and left lung

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   Discussion Top


First case of aspergillosis was reported in United Kingdom by Hinson et al. in 1952.[5] Even after five decades of research, this disease is still under-diagnosed, with as many as one-third of cases being initially misdiagnosed as pulmonary tuberculosis in the developing countries.[6] ABPA most commonly complicates the course of bronchial asthma and cystic fibrosis (CF).[7] The clinical presentation of ABPA is very similar to pulmonary tuberculosis, and signs and symptoms are usually poorly-controlled asthma, hemoptysis, expectoration of mucus plugs, malaise, and fever.[8] The diagnosis can be made based on the combination of clinical, hematological, immunological, and radiological findings. Furthermore, type I skin test against Aspergillus antigen, elevated levels of specific IgG and IgE antibodies against A. fumigatus, raised levels of blood IgE, central bronchiectasis, and eosinophilia may help physician to confirm the diagnosis.[9]

Diagnostic criteria for allergic bronchopulmonary aspergillosis

Predisposing conditions:

  • Bronchial asthma, CF.


Obligatory criteria:

  • Elevated serum total IgE levels (>1000 IU/ml)
  • Elevated serum IgG and/or IgE against A. fumigatus.


Other criteria (at least three of five):

  • Immediate type I reaction to A. fumigatus antigen
  • Presence of serum A. fumigatus precipitins
  • Transient and/or permanent chest radiographic opacities
  • Eosinophil count >1000 cells/ul in peripheral blood
  • Central bronchiectasis on high-resolution computed tomography chest.[8],[9]


Literature reveals 25 reported cases of aspergillosis having intraoral manifestations. The mean age of reported patients is 37 years (range, 9–78 years).[10] Case reported here affected a 56-year-old male. Oral aspergillosis affects predominately male patients (69%) to female gender (31%). The most commonly affected sites are the gingivae (19 cases), followed by the hard palate (5 cases), maxillary sinus (3 cases), soft palate (1 case), and tongue (1 case).[10] Correa et al.[3] reported a case of invasive oral aspergillosis on the tongue with acute myeloid leukemia. They have diagnosed the case based on the microscopic invasion of the epithelium, connective tissue, and muscle of the tongue by fungal hyphae branching at 45o angle.[3] In the present case, lesion was also on the tongue but not of invasive type of aspergillosis as no fungal hyphae invading underneath connective tissue branching at 45o were demonstrated. This ulceration was probably a localized allergic manifestation of aspergillosis in addition to systemic ABPA. To the best of authors' knowledge, this was the first ever reported case of aspergillosis on tongue secondary to ABPA.

Aggressive forms of oral aspergillosis may involve hard and soft tissues. The main symptoms are gingival pain and facial swelling, followed by excessive salivation. In the present case, because of location and ulceration of the lesion, the patient displayed glossodynia and dysphagia. As per the diagnostic criteria for ABPA, in our patient the predisposing factor was bronchial asthma, as the patient was taking medicine for the same from local physician. Total serum IgE level and IgE against A. fumigatus were marked raised, which further confirms the obligatory criteria for the diagnosis of ABPA. In the section of other criteria for diagnosis of ABPA, in our case report, patient showed marked elevation in the level of eosinophil count, presence of serum precipitating antibodies to A. fumigatus, and radiopacities on the right and left side of lung.

Initially, the corticosteroids considered as the first-line therapy for ABPA reduce the immune response mounted to the presence of Aspergillus in the airways. Nevertheless, long-term systemic corticosteroid therapy is not recommended as there is no evidence that it prevents the progression of lung destruction and it may be ineffective in end-stage fibrosis. Based on the IDSA guidelines for aspergillosis,[4] we have prescribed combination of itraconazole and corticosteroid therapy and significant improvement was noticed. The decrease in serum total IgE level, the improvement in healing of ulcer, and reduction of consolidation on chest X-ray demonstrate that the patient responded well to the treatment with oral itraconazole and oral prednisolone. This therapy has been seen to reduce exacerbations, improve laboratory and lung function parameters, and prevent disease progression. In the future, we will continue to monitor our patient for any systemic clinical deterioration, as literature show that relapses in ABPA are common, despite initial response to therapy.


   Conclusion Top


The readers should thoroughly understand the differential diagnosis of solitary ulcer on the tongue and should also correlate the systemic conditions with local manifestations.

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.
Stevens DA, Kan VL, Judson MA, Morrison VA, Dummer S, Denning DW, et al. Practice guidelines for diseases caused by Aspergillus. Infectious Diseases Society of America. Clin Infect Dis 2000;30:696-709.  Back to cited text no. 1
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2.
Denning DW, Pleuvry A, Cole DC. Global burden of allergic bronchopulmonary aspergillosis with asthma and its complication chronic pulmonary aspergillosis in adults. Med Mycol 2013;51:361-70.  Back to cited text no. 2
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3.
Correa ME, Soares AB, de Souza CA, Cintra ML, Jorge J, Almeida OP, et al. Primary aspergillosis affecting the tongue of a leukemic patient. Oral Dis 2003;9:49-53.  Back to cited text no. 3
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4.
Walsh TJ, Anaissie EJ, Denning DW, Herbrecht R, Kontoyiannis DP, Marr KA, et al. Treatment of aspergillosis: Clinical practice guidelines of the Infectious Diseases Society of America. Clin Infect Dis 2008;46:327-60.  Back to cited text no. 4
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5.
Hinson KF, Moon AJ, Plummer NS. Bronchopulmonary aspergillosis. Thorax 1952;7:317-33.  Back to cited text no. 5
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6.
Aggarwal R, Gupta D, Aggarwal AN, Saxena AK, Saikia B, Chakrabarti A, et al. Clinical significance of decline in serum IgE levels in allergic bronchopulmonary aspergillosis. Respir Med 2010;104:204-10.  Back to cited text no. 6
    
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Aggarwal R. Allergic bronchopulmonary aspergillosis. Chest 2009;135:805-26.  Back to cited text no. 7
    
8.
Aggarwal R, Hazarika B, Gupta D, Aggarwal AN, Chakrabarti A, Jindal SK. Aspergillus hypersensitivity in patients with chronic obstructive pulmonary disease: COPD as a risk factor for ABPA? Med Mycol 2010;48:988-94.  Back to cited text no. 8
    
9.
Aggarwal R, Khan A, Garg M, Aggarwal AN, Gupta D. Pictorial essay: Allergic bronchopulmonary aspergillosis. Indian J Radiol Imaging 2011;21:242-52.  Back to cited text no. 9
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10.
Bor O, Dinleyici EC, Kiraz N, Dundar E, Akgun NA. Successful treatment of tongue aspergillosis caused by Aspergillus flavus with liposomal amphotericin B in a child with acute lymphoblastic leukemia. Med Mycol 2006;44:767-70.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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