Acta Scientific Dental Sciences

Case Report Volume 1 Issue 2

Oral Histoplasmosis Masquerading as Squamous Cell Carcinoma

Arundhati1* and Ashok Kumar2

1Consultant Pathologist, Mahavir Cancer Sansthan and Research Institute, Patna, Bihar, India
2Consultant Coardiologist, Medica Heart Institute, Patna, Bihar, India

*Corresponding Author: Arundhati, Consultant Pathologist, Department of Pathology, Mahavir Cancer Sansthan, Patna, Bihar, India.

Received: June 07, 2017; Published: July 21, 2017

Citation: Arundhati and Ashok Kumar. “Oral Histoplasmosis Masquerading as Squamous Cell Carcinoma” Acta Scientific Dental Sciences 1.2 (2017).

Abstract

  Histoplasmosis is a systemic, deep mycotic infection caused by the dimorphic fungus, Histoplasma capsulatum. Systemic histoplasmosis has emerged as an important opportunistic infection in human immunodeficiency virus (HIV) patients and those in endemic areas. Reported cases of histoplasmosis have been low in India with less than 50 cases being reported. We are reporting a case of disseminated histoplasmosis with isolated oral involvement in an HIV seronegative patient. Treatment with oral itraconazole led to remission of the oral lesions.

Keywords: Oral Histoplasmosis; Histoplasma capsulatum; Oral Lesions

Introduction

  Histoplasmosis is a granulomatous systemic mycotic infection caused by thermal dimorphic fungus, Histoplasma capsulatum. It is also known as Darlings disease as Samuel Darling while working in the Canal Zone in Panama in 1905 found it out. He first described its clinical disseminated disease form in a fatal case from Martinique after 30 years of finding it [1].

  Histoplasmosis is endemic in the central eastern United States, especially the Ohio and Mississippi River Valleys, in Central and South America, and Africa, but is less frequently reported in Asia and Europe [2,3].

  Reported cases of histoplasmosis have been low in India with less than 50 cases being reported [4].

  Histoplasma develops as a branching hyphal form in the soil and a yeast form in the host tissue [5]. The organism is commonly found in warm, humid environment that contains bird and bat excreta. H. capsulatum is acquired by inhalation of airborne spores from mycelial fragments of the fungus.

  Histoplasmosis may occur in three forms: (i) Primary acute pulmonary form, (ii) chronic pulmonary and (iii) disseminated form(DH) [6]. Occurrence of disseminated form of histoplasmosis is rare in immunocompetent individual though in immunocompromised and HIV positive patients, 95% of histoplasmosis appears as disseminated infection [3].

  The oral lesions may occur in any part of the oral cavity and the lesions vary from nodules to painful shallow or deep ulcers [3]. Around 30 - 66% of patients with DH have oral lesions, frequently presenting as the initial sign. Generally, lesions of histoplasmosis in the oral cavity are the local manifestation of pulmonary or disseminated disease [7]. but rarely may they be the primary or even the only manifestation of the disease [8].

  In India, histoplasmosis is rare and is endemic only in small regions in West Bengal and Maharashtra [9,10].

  We hereby present a case of oral histoplasmosis from the state of Bihar in an otherwise immunocompetent, HIV negative young male without other subjacent underlying disease, with lesions exclusively in the bilateral retromolar trigone area of oral cavity. Bihar is a non-endemic region for histoplasmosis in India.

Case Report

  The patients complained of symptoms such as occasional fever, asthenia, lassitude, lethargy, weight loss, some difficulty in speech and odynophagia for more than 2 months. On examination, the oral lesions appear as nodular ulcerative lesions of size approximately 2 cm × 2 cm that were painful, localized bilaterally in the retromolar trigone area of buccal mucosa. The ulcers have raised borders covered by a reddish yellow ulcerative necrotic membrane, resembling aphthous ulcers, traumatic ulcers, carcinoma or tuberculosis.

  The ulcer did not bleed and showed no discharge. The lesions were initially budding, papule like and then developed gradually into ulcerating, indurated and painful lesions. The histopathology features were also consistent with those of a chronic specific infection characterized by infiltration of predominantly lymphocytes, formation of the ill formed granulomas composed of histiocytes, containing variable numbers of yeast-like oval structures, measuring 3 - 5µm in diameter morphologically looking like - confirmed by PAS stain. On physical examination, no lymphadenopathy or organomegaly was found.

  On systemic examination, no any relevant finding could be elicited. The patient does not report any risk factor for histoplasmosis. There was no history of oral trauma. He was non-alcoholic, nonsmoker, non-diabetic and no past history for tuberculosis. He did not report a history of travelling and never went out of the local district.

 Figure 1: 
Gross Photograph of the Patient’s Oral Ulcer Before
(A) and after Treatment.

Figure 1: Gross Photograph of the Patient’s Oral Ulcer Before (A) and after Treatment.

 Figure 2: 
Microphotograph Showing Inflammatory Cell
Infiltrate and Macrophages Containing the Yeast form of
H.Capsulatum (H&E X 40)

Figure 2: Microphotograph Showing Inflammatory Cell Infiltrate and Macrophages Containing the Yeast form of H.Capsulatum (H&E X 40)

 Figure 3: 
Yeast-Like Cells were Found when Stained with
Periodic Acid-Schiff Stain (PAS X100).

Figure 3: Yeast-Like Cells were Found when Stained with Periodic Acid-Schiff Stain (PAS X100).

Discussion

  Histoplasmosis infection occurs through the inhalation of conidial forms present in the environment. The fungus is dimorphic intracellular, parasitizing the reticule-endothelial system and involving many organs like spleen, liver, kidney, central nervous system [11,12]. It is primarily a pulmonary disease, and the environmental reservoir is soil. There are two varieties of H. capsulatum that are pathogenic to humans, H. capsulatum var. capsulatum and H. capsulatum var. duboisii, and a third variety that is an equine pathogen, H. capsulatum var. farciminosum, which exists in Africa [13].

  Oral lesions of histoplasmosis can manifest in a variety of forms as ulcerative, nodular, verrucous, vegetative, granulomatous, and plaque-like lesions involving the lips, gingiva, tongue, and palate [14,15,16].

  Frequently, in cases of disseminated histoplasmosis, oral lesions appear as the initial clinical manifestation. Since 1946 only a few cases of apparently initial mucocutaneous histoplasmosis in patients without detectable systemic involvement have been reported [17]. According to one school of thought oral lesions result of hematogenous spread from an unknown infectious focus, and according to another school of thought primary oral lesions can occur by direct inoculation of the fungus in the mucosa [18,19].

  Some investigators suggested that, when no systemic sign or symptom can be detected, oral histoplasmosis should be considered a localized disease, and treated as such (i) Clinical manifestations of histoplasmosis range from asymptomatic lesions to life-threatening disseminated infections, although the disease is self-limiting in immunocompetent patients [20].

  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 [21]. In the present case itraconazole 200 mg once a day has brought about complete clinical remission of the lesion in just 10 days, though medicine is to be continued for one month and patient has been suggested to be in follow up for at least 9 months. The patient’s haematological values along with liver and renal function test were within the normal limits; thus, showing the drug was well tolerated. Facility for serum itraconazole assessment is not available in our set up, so it was not done.

  The gold standard for the diagnosis is the isolation of the H. capsulatum in culture with observation of the conidia, as well as its conversion to yeast forms at 37°C. Diagnosis is usually made by a combination of culture, detection of the organism in tissues, measurement of antibodies, and detection of antigen.

Conclusion

   Many of the patients do not have any identifiable risk factors for Histoplasmosis particularly in non-endemic area. Non-healing ulcer in oral region should not only be looked upon as malignant lesion rather infectious ethology like tuberculosis and histoplasmosis should also be kept in differential diagnosis.

Bibliography

  1. Darling ST. “A protozoan general infection producing pseudotubercles in the lungs and focal necroses in the liver, spleen, and lymph nodes”. JAMA 46 (1906): 1283-1285.
  2. Rappo U., et al. “Expanding the horizons of histoplasmosis: Disseminated histoplasmosis in a renal transplant patient after a trip to Bangladesh”. Transplant Infectious Disease 12.2 (2010): 155-160.
  3. Ferreira OG., et al. “Oral histoplasmosis in Brazil”. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontology 93.6 (2002): 654-659.
  4. Vidyanath S., et al. Journal of Oral and Maxillofacial Pathology 17.1 (2013): 139-142.
  5. Wheat LJ. “Laboratory diagnosis of histoplasmosis: update 2000”. Seminars in Respiratory Infections 16.2 (2001): 131- 140.
  6. Wheat LJ and Kauffman CA. “Histoplasmosis”. Infectious Disease Clinics of North America 17.1 (2003): 1-19.
  7. Loh FC., et al. “Histoplasmosis presenting as hyperplastic gingival lesion”. Journal of Oral Pathology and Medicine 18.9 (1989): 533-536.
  8. Reddy P., et al. “Progressive disseminated histoplasmosis as seen in adults”. American Journal of Medicine 48.5 (1970): 629-636.
  9. Sanyal M. “Skin sensitivity to histoplasmin in Calcutta and its neighbourhood”. Indian Journal of Dermatology, Venereology and Leprology 46.2 (1980): 94-98.
  10. Mukherjee AM., et al. “Histoplasmosis in India with report of two cases”. Journal of the Indian Medical Association 56.5 (1971): 121-125.
  11. Hinshaw M and Longley BJ. “Fungal Diseases”. In: Elder D.A., Lever’ S Histopathology of the Skin 10th ed. Lippincott Williams & Wilkins, a Wolters Kluwer business 2009: 611-613.
  12. Sood N., et al. “Mucocutaneous nasal histoplasmosis in an immunocompetent young adult”. Indian Journal of Dermatology, Venereology and Leprology 73.3 (2007): 182-184.
  13. Kauffman CA. “Histoplasmosis. In: Dismukes WE, Pappas PG, Sobel JD, editors. Clinical mycology. New York, NY: Oxford University Press 2003: 285-298.
  14. Greenberg MS., et al. “Burket’s Oral Medicine”.11th ed. Hamilton BC Decker Inc 2008: 41-73.
  15. Warnakulasuriya KA., et al. “Localised oral histoplasmosis lesions associated with HIV infection”. Journal of Oral Pathology and Medicine 26.6 (1997): 294-296.
  16. Hernández SL., et al. “Oral histoplasmosis associated with HIV infection: a comparative study”. Journal of Oral Pathology and Medicine 33.8 (2004): 445-450.
  17. MIGNOGNA MD., et al. “A case of oral localized histoplasmosis in an immunocompetent patient”. European Journal of Clinical Microbiology and Infectious Diseases 20.10 (2001): 753-755.
  18. Economopoulou P., et al. “Oral histoplasmosis as an indicator of HIV infection”. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and Endodontology 86.2 (1998): 203-206.
  19. Sell SH., et al. “The use of immunofluorescent techniques in diagnosis of clinical histoplasmosis”. Diseases of the chest 54 (1968): 292-295.
  20. Patil K., et al. “Oral histoplasmosis”. Journal of Indian Society of Periodontology 13.3 (2009): 157-159.
  21. Dimukes WE., et al. “Itraconozole therapy for blastomycosis and histoplasmosis. NIAID Mycoses Study Group”. American Journal of Medicine 93.5 (1992): 489-497.

Copyright: © 2017 Arundhati and Ashok Kumar. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.



News and Events

  • Submission Timeline
    Last date for submission of articles is May 05, 2019.
  • Publication Certificate
    Authors will be issued a "Publication Certificate" as a mark of appreciation for publishing their work.
  • Best Papers of the Issue
    The Editors will elect one Best Article after each issue release. The authors of this article will be provided with a certificate of “Best Article of the Issue”.
  • Welcoming Article Submission
    Acta Scientific delightfully welcomes active researchers for submission of articles towards the upcoming issue of respective journals.
  • Contact US