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 Table of Contents  
CASE REPORT
Year : 2014  |  Volume : 26  |  Issue : 4  |  Page : 446-449

Diagnosing a solitary oral ulcer: An enigma


Department of Oral Medicine and Radiology, GITAM Dental College and Hospital, Visakhapatnam, Andhra Pradesh, India

Date of Submission04-Mar-2015
Date of Acceptance16-Apr-2015
Date of Web Publication22-Apr-2015

Correspondence Address:
Jayshree Agrawal
Department of Oral Medicine and Radiology, GITAM Dental College and Hospital, Rushikonda, Visakhapatnam - 530 045, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.155658

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   Abstract 

Diagnosing solitary oral ulcers are always a challenging task which requires a through history taking, clinical examination and charting out clear differential diagnosis. Many solitary oral ulcers appear quite similar and may pose a challenge to the diagnostician; however, early diagnosis of such lesions enables to initiate the treatment at an early stage. In this paper, a case of oral histoplasmosis involving the palate along with the differential diagnosis is reported wherein the history of close association of the patient with birds clinched the diagnosis, thus emphasizing the importance of minute aspects of history taking.

Keywords: Grocott′s methanamine silver stain, oral fluconazole, oral histoplasmosis, solitary oral ulcer


How to cite this article:
Yellarthi PK, Bala V, Agrawal J, Damera A. Diagnosing a solitary oral ulcer: An enigma. J Indian Acad Oral Med Radiol 2014;26:446-9

How to cite this URL:
Yellarthi PK, Bala V, Agrawal J, Damera A. Diagnosing a solitary oral ulcer: An enigma. J Indian Acad Oral Med Radiol [serial online] 2014 [cited 2021 Dec 7];26:446-9. Available from: https://www.jiaomr.in/text.asp?2014/26/4/446/155658


   Introduction Top


Diagnosing single ulcers in the oral cavity has always been challenging for an oral diagnostician, where in the entire treatment depends on the diagnosis of such lesions. Careful history recording in all aspects is very essential. In the present case, after excisional biopsy, the diagnosis of histoplasmosis was revealed which was confirmed following the disclosure of the patient's close association with pet birds, thus, emphasizing the fact that minor aspects in history taking should not be taken for granted and are indeed of paramount importance in arriving at the final diagnosis. A case of a solitary ulcer of oral histoplasmosis as the only presenting sign is reported here in a 23-year-old female immunocompetent patient.


   Case Report Top


A 23-year-old female patient reported with a painful oral ulcer on the soft palate since 2 months. The patient also gave a history of occurrence of ulcers in other areas of her mouth which were noticed few months earlier to the present ulcer, lasting a few days. The present reported ulcer was lasting for over 2 months causing dysphasia and with no sign of healing. A negative history of trauma was reported by the patient along with no noteworthy medical and personal history. Physical examination revealed right and left submandibular lymphadenopathy. Her vital signs were within normal limits with no signs of anemia.

Clinical examination revealed a solitary, irregularly shaped, shallow ulcer measuring about 2 × 1.5 cm, at the junction of the hard and soft palate on the left side. The edge of the ulcer was sloping and the floor was covered with slough with a surrounding erythematous halo [Figure 1]. Palpation revealed the ulcer to be tender with no discharge. Topographical maxillary occlusal radiograph revealed no bony changes. A cytology smear demonstrated diffused acute inflammatory cells with predominant neutrophils and a few superficial squamous cells suggesting a chronic infectious ulcer. Considering the history and the chronicity of the present ulcer, with the size exceeding more than 1 cm in diameter and a positive cytological smear for inflammatory cells, an initial diagnosis of recurrent aphthous major was made.
Figure 1: Ulcer at the junction of hard and soft palate on the left side

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A negative trauma history excluded traumatic ulcer from the differential diagnosis. Solitary irregularly shaped painful ulcer on the palate with palpable lymph nodes was also suggestive of infectious ulcers like tubercular or syphilitic ulcer. However, in the present case, although her medical history was non-contributory, they were considered in the differential diagnoses, due to the patient's low socio-economic status along with the persistent nature of the ulcer. Oral deep-seated fungal infection, though rare in the oral cavity, was considered in the differential diagnoses due to the similarity in the clinical presentation. Necrotizing sialometaplasia (NS) often presenting as a deep-seated ulcer and squamous cell carcinoma, the most common malignancy condition occurring in the oral cavity, were also considered in the differential diagnoses; as early lesions of such conditions may easily be missed, sometimes creating a diagnostic dilemma. With a provisional diagnosis of recurrent aphthae major, the patient was prescribed a topical anesthetic application and tetracycline mouthwash, thrice daily for 2 weeks. On recall, no reduction in symptoms or the size of the ulcer was noticed.

An incisional biopsy was planned before which routine hematological tests were carried out and all the values were within normal limits. Serological tests like venereal diseases research laboratory (VDRL) test and Mantoux test were negative, and non-reactive to HIV (tridot), HbsAg and Anti-HCV antibodies. The histopathological examination correlated the cytological smear findings, presenting mixed inflammatory cell infiltrate consisting of mainly lymphocytes and histiocytes, confirming the diagnosis of a chronic ulcer. The patient was then prescribed a systemic immunomodulator (Levamisole 150 mg for 4 days in a week, for 4 weeks). On recall visit, no reduction in size and symptoms of the ulcer was reported; hence, surgical excision of the entire ulcer was performed. The wound was packed with collagen granules and closure was done with collagen membrane [Figure 2]. Hematoxylin-eosin staining and subsequent microscopic examination of the obtained specimen [Figure 3], revealed a breach in the overlying epithelium with infiltration of chronic inflammatory cells in the superficial connective tissue. In the deep connective tissue, some sporangia along with Langerhans giant cells with caseating necrosis were seen, providing a histopathological diagnosis of histoplasmosis which was confirmed using Grocott's methanamine silver stain [Figure 4]. After knowing the histological diagnosis, re-questioning the patient revealed her close association with birds as her pets. Further systemic evaluation was carried out, which was negative for any disseminated form of the disease. Oral fluconazole 150 mg once daily for 1 month was initiated, after which the lesion started showing excellent response within the first month [Figure 5]. The patient was advised to continue the same medication for another 3 months for complete remission and maintenance with monthly follow-up. On subsequent reviews, complete healing of the lesion was noticed [Figure 6]. Patient is currently on maintenance phase with regular follow-up.
Figure 2: Post-surgical wound closure with collagen membrane

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Figure 3: Deep connective tissue showing sporangia along with Langerhan's giant cells and caseating necrosis (Hematoxylin and eosin, 40×)

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Figure 4: Histoplasmosis sporangia confirmed using Grocott's methanamine silver stain (40×)

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Figure 5: Partial remission of ulcer after 1 month recall with fl uconazole therapy

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Figure 6: Complete remission of ulcer after 3 months recall with fluconazole therapy. Surgical scar is noticeable

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   Discussion Top


After its first description, by Samuel Darling in 1905, systemic histoplasmosisis is also known as darlings disease. [1] The disease is caused by the inhalation of fungus Histoplasma capsulatum, which usually grows in warm and humid environments containing dust or contaminated with bird and bat excreta. [2] Histoplasmosis is encountered among laborers and agricultural workers around the globe; however, in India, more cases have been reported in eastern India and Vellore district of Tamil Nadu state. [3] In the present case, the patient was a resident of south India, having a close association with birds.

The immune status of the host determines the course of the disease, with oral lesions in 25-45% of the patients. [4] Rarely oral histoplasmosis has been reported in an immune-competent host without detectable systemic involvement as seen in the present case report. Although cases of oral histoplasmosis is a secondary manifestation of the pulmonary or disseminated forms, this claim has been questioned by many authors, as the possible occurrence of oral histoplasmosis could be due to the direct inoculation of the fungus into the oral mucosa. [5] Weight loss, asthenia, chronic cough and fever are the common presenting signs and symptoms, but none of these were reported in the present case.

Padhye et al. suggested the common site of occurrence of histoplasmosis in India to be in the oral cavity including the buccal mucosa, tongue, palate and gingiva. [3] In the present case, the lesion was seen on the soft palate. Review of literature suggested that histoplasmosis is commonly seen in men, with a male to female ratio of 9:1 and the average range of occurrence between 26 and 65 years similar to the findings in the present case. [6] Nodules, vegetative lesions or painful shallow ulcerations with dysphasia are the common manifestations of oral histoplasmosis. [5] Clinical manifestations of histoplasmosis range from asymptomatic lesions to life-threatening disseminated infections, although the disease is self-limiting in immunocompetent patients. [6]

Amphotericin B has been the drug of choice and has been extensively used in the management since the late 1950's. [7] As Amphotericin B is difficult to administer in I.M. or I.V. route and very often poorly tolerated by the patient, requirement of other antifungal agents as the treatment options has considerably expanded. Azole antifungal drugs like ketoconazole 200 mg tablet and itraconazole 100 mg capsule have off late been the drugs of choice, in most healthy and non-HIV-infected patients. [8] Patients have reported intolerance to these drugs and also are at a significant risk for drug interactions. Although, fluconazole being moderately effective in induction and maintenance therapy for fungal infection in both HIV and non-HIV patients, it has emerged as an effective alternative in treating histoplasmosis due to fewer drug interactions. [9],[10]

In the present case fluconazole has brought about complete remission of the lesion. Following fluconazole therapy for 3 months, the patient's hematological values along with liver and renal function test were within the normal limits; thus, showing the drug was well tolerated.


   Conclusion Top


Solitary oral ulcers may pose a challenge in diagnosis and a careful history recording in all aspects is very essential.

 
   References Top

1.
Sivapathasundaram B, Gururaj N. Mycotic infections of the oral cavity. In: Rajendran R, Sivapathasundaram B, editors. Shafer's Textbook of Oral Pathology. 5 th ed. New Delhi: Elsevier; 2006. p. 1199-227.  Back to cited text no. 1
    
2.
Miller RL, Gould AR, Skolnick JL, Epstein WM. Localized oral histoplasmosis: A regional manifestation of mild chronic disseminated histoplasmosis. Oral Surg Oral Med Oral Pathol 1982;53:367-74.  Back to cited text no. 2
    
3.
Padhye AA, Pathak AA, Katkar VJ, Hazare VK, Kaufman L. Oral histoplasmosis in India: A case report and an overview of cases reported during 1968-92. J Med Vet Mycol 1994;32:93-103.  Back to cited text no. 3
    
4.
Muñante-Cárdenas JL, de Assis AF, Olate S, Lyrio MC, de Moraes M. Treating oral histoplasmosis in an immunocompetent patient. J Am Dent Assoc 2009;140:1373-6.  Back to cited text no. 4
    
5.
Sadat SM, Rita SN, Kahhar MA. Oral histoplasmosis: Report of two cases. J Bangladesh Coll Phys Surg 2012;30:229-33.  Back to cited text no. 5
    
6.
Patil K, Mahima VG, Prathibha Rani RM. Oral histoplasmosis. J Indian Soc Periodontol 2009;13:157-9.  Back to cited text no. 6
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7.
Gallis HA, Drew RH, Pickard WW. Amphotericin B: 30 years of clinical experience. Rev Infect Dis 1990;12:308-29.  Back to cited text no. 7
    
8.
Dimukes WE, Bradsher RW Jr, Cloud GC, Kauffman CA, Chapman SW, George RB, et al. Itraconozole therapy for blastomycosis and histoplasmosis. NIAID Mycoses Study Group. Am J Med 1992;93:489-97.  Back to cited text no. 8
    
9.
Kobayashi GS, Travis SJ, Medoff G. Comparison of flucanozole and amphotericin B in treating histoplasmosis in immunosuppressed mice. Antimicrob Agents Chemother 1987;31:2005-6.  Back to cited text no. 9
    
10.
Como JA, Dismukes WE. Oral azole drugs as systemic antifungal therapy. N Engl J Med 1994;153:263-72.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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