Head and Neck Clinic

Ear, Nose & Throat Journal

1–3
Histoplasma capsulatum Caused a Localized ª The Author(s) 2019
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Tongue Ulcer in a Non-HIV Patient— DOI: 10.1177/0145561319844246
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A Case From Nonendemic Country

Liyanage Shamithra Madhumali Sigera, MBBS, MD1,

Sumith Ruwan Gunawardane, BDS, MBA2,
Matharage Ariyalatha Malkanthi, Diploma MLT1,
Ruwan Duminda Jayasinghe, BDS, MS2, M. A. M. Sitheeque, BDS, MS2,
Wanninayake Mudiyanselage Tilakaratne, MS, FDSRCS (UK), FRCPath (UK), PhD4,
and Primali Iresha Jayasekera, MBBS, MD1

A 56-year-old male, smoker (25 cigarettes per day) for

30 years, was referred to the Oral Medicine Clinic, University
Dental Hospital with nonhealing, gradually enlarging ulcer
on tongue for 4 months.
He complained of a painful ulcer over the left side of his
tongue which was associated with odynophagia. He didn’t have
chronic cough, fever, malaise, loss of appetite or loss of weight,
and systemic symptoms suggestive of inherited or acquired
immunodeficiency including HIV.
His past medical history was marked by diabetes mellitus
and chronic obstructive pulmonary disease, and he was on oral
hypoglycemic drugs.
He denied past orofacial surgeries, trauma, high-risk sexual
behavior, or long-term immunosuppressive drugs. This nonal-
coholic patient had no history of traveling abroad.
He was averagely built, afebrile male. Oral examination
Figure 1. The photo shows the solitary, irregular ulcer on the left
revealed a solitary, 2  4  1 cm size, tender, irregular ulcer posterior tongue with indurations.
on the left posterior tongue with indurations (Figure 1). The
pain intensity was at 10 in the visual analog scale. There was no white cottony growth was observed on the agar (26 C) after
regional lymphadenopathy or hepatosplenomegaly. Rest of the 14 days. The tease mount of the growth revealed hyaline, sep-
general and systemic examinations were normal. tate fungal filaments, and macroconidia (Figure 2). After
Histopathology of first 2 biopsies of the lesion showed 7 days of incubation on blood–brain–heart infusion agar at
chronic granulomatous inflammation. However, they were
negative for Ziehl-Neelsen staining. The patient tested normal
for renal and hepatic biochemical tests, chest radiography, and
1
colonoscopy. He had neutrophil leukocytosis and Erythrocyte Department of Mycology, Medical Research Institute, Colombo, Sri Lanka
2
Sedimentation Rate (ESR) was 22 mm per first hour. He was Faculty of Dental Sciences, University of Peradeniya, Peradeniya, Sri Lanka
3
Department of Oral Medicine and Periodontology, Faculty of Dental Sciences,
negative for HIV, Venereal Disease Research Laboratory test University of Peradeniya, Peradeniya, Sri Lanka
(VDRL), and Mantoux tests. 4
Department of Oral Pathology, Faculty of Dental Sciences, University
He was treated with anti-inflammatory drugs and cellulose- of Peradeniya, Peradeniya, Sri Lanka
based covering agent for 6 weeks and subsequently started with Received: February 21, 2019; revised: March 12, 2019; accepted: March 22,
oral prednisolone. There was an initial response, but he left 2019
against medical advice after 5 days of treatment.
Corresponding Author:
He readmitted 3 months later along with expanded ulcer Liyanage Shamithra Madhumali Sigera, MBBS, MD, Department of Mycology,
with few areas of necrosis and it was biopsied again. Mycolo- Medical Research Institute, Colombo 00080, Sri Lanka.
gical evaluation revealed yeast cells in the direct smear, and Email: shamithra@yahoo.com
2 Ear, Nose & Throat Journal XX(X)
Figure 2. The tease mount of the growth reveals hyaline, septate
fungal filaments, and macroconidia with tubercles and small, round
microconidia (40).
Figure 3. Gomori methenamine silver stain section of the biopsy of
the ulcer shows small oval yeasts cells suggestive of Histoplasma cap-
sulatum (40).
37 C, yeast-like growth was observed, and the isolate was
confirmed as Histoplasma capsulatum.
The histology report revealed mixed inflammatory infiltrate
containing macrophages, neutrophils, and plasma cells. Small
circular organisms positive for Periodic acid–Schiff (PAS) and
Grocott gomori suggestive of histoplasmosis were seen both
within and outside of macrophages (Figure 3).
He was initially started with oral itraconazole, yet converted
to intravenous amphotericin B after 7 days while he was mon-
itoring in the intensive care unit. Under treatment, his physical
condition was aggravated with uncontrolled diabetes mellitus
which was complicated by a secondary bacterial infection.
Evidence of bacterial pneumonia was observed clinically,
microbiologically, and radiologically. Intravenous antibiotics
were started to cover up the pathogen. Unfortunately, the
patient remained frail and died due to sepsis after 1 month from
starting treatments.
Histoplasmosis is a systemic fungal infection caused by
H capsulatum, a dimorphic fungus. 1,2 All together,
3 varieties of H capsulatum have been recognized, in which
H capsulatum var. capsulatum and H capsulatum var duboisii
are human pathogens and H capsulatum var. farciminosum is
an equine pathogen.1
Histoplasmosis is endemic in certain regions of the world,
with a rising incidence in some Asian countries.3,4 However, it
is a rare disease condition in Sri Lanka and only very few cases
have been reported.5-7
The disease has wide range of clinical presentations, ranging
from asymptomatic infection, chronic pulmonary infections, to
disseminated infections.8 Around 30% to 50% of patients with
disseminated histoplasmosis present with oral lesions.8,2,9 They
usually have clinical manifestations of dissemination such as
fever, weight loss, mucocutaneous lesions, lymphadenopathy,
hepatosplenomegaly, and features of adrenal insuffi-
ciency.2,8,10 A localized oral lesion without dissemination is
an uncommon presentation.2,11,12 Since oral lesions could be
the primary manifestation of the dissemination, evaluation of
the patient for the dissemination and possible immunocompro-
mised condition is important.13
These mucosal lesions appear in almost every part of the
oral mucosa, yet the commonest sites are tongue, palate, and
buccal mucosa.12,14 These lesions could be nodular, ulcerative,
verrucous, or plaque-like.11 Oral histoplasmosis mimics other
oral ulcerative lesions such as chronic traumatic ulcers, squa-
mous cell carcinoma, lymphomas, Crohn disease, ulcerative
necrotic gingivitis or stomatitis, tuberculosis, and necrotizing
sialometaplasia.2 Consequently, in the absence of proper
awareness, the clinicians may misdiagnose the condition.10,15
Amphotericin B and certain azoles are effective against
H capsulatum.10 Itraconazole and fluconazole have been
effective in treating head and neck histoplasmosis with
85% to 100% and 86% of cases, respectively.9 The refrac-
tory cases to initial azole treatment should be converted into
amphotericin B.9
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to
the research, authorship, and/or publication of this article.
Funding
The author(s) received no financial support for the research, author-
ship, and/or publication of this article.
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