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  Table of Contents    
CASE REPORT  
Year : 2016  |  Volume : 59  |  Issue : 2  |  Page : 227-228
Primary cutaneous histoplasmosis mimicking basal cell carcinoma of the eyelid: A case report and review of literature


1 Department of Ocular Pathology, Oculoplasty and Ocular Oncology, Aravind Eye Hospital and Postgraduate Institute, Madurai, Tamil Nadu, India
2 Department of Orbit, Oculoplasty and Ocular Oncology, Aravind Eye Hospital and Postgraduate Institute, Madurai, Tamil Nadu, India

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Date of Web Publication9-May-2016
 

   Abstract 

We present the case of a 46 years old gentleman with longstanding ulcerative lesion of the right upper eyelid. Biopsy of the lesion revealed intracellular periodic acid–Schiff positive round to oval organisms with a surrounding clear halo suggestive of histoplasmosis. Histoplasma capsulatum specific polymerase chain reaction was positive. The patient was treated with oral antifungals and showed a prompt response to treatment. We report this rare case of primary cutaneous histoplasmosis of the eyelid in an otherwise healthy adult patient.

Keywords: Eyelid biopsy, eyelid malignancy, primary cutaneous histoplasmosis

How to cite this article:
Radhakrishnan S, Adulkar NG, Kim U. Primary cutaneous histoplasmosis mimicking basal cell carcinoma of the eyelid: A case report and review of literature. Indian J Pathol Microbiol 2016;59:227-8

How to cite this URL:
Radhakrishnan S, Adulkar NG, Kim U. Primary cutaneous histoplasmosis mimicking basal cell carcinoma of the eyelid: A case report and review of literature. Indian J Pathol Microbiol [serial online] 2016 [cited 2018 Oct 22];59:227-8. Available from: http://www.ijpmonline.org/text.asp?2016/59/2/227/182017



   Introduction Top


Histoplasmosis is deep mycosis caused by the dimorphic fungus Histoplasma capsulatum. The respiratory tract is the usual portal of entry of the fungus. Disruption of soil is one of the most common means of releasing conidia, which are inhaled and eventually settle in lung parenchyma. A primary cutaneous histoplasmosis is an uncommon form of this disease, and that presenting as an eyelid lesion is even rare. We present a case of eyelid histoplasmosis presenting as a malignant lid tumor.


   Case Report Top


A 46-year-old man presented to our clinic with an ulcerative lesion in the right upper eyelid associated with pain and serosanguinous discharge of 1 month duration. On examination, his best-corrected visual acuity in the right eye was 6/6, N6. There was a pigmented ulcerative lesion with on the right upper eyelid, involving the medial two-third of the upper lid [Figure 1]. There was no regional lymphadenopathy. A swab from the base of the ulcer was taken and sent for microbiological examination. A working diagnosis of basal cell carcinoma was made. The patient underwent a biopsy of the lesion. Histopathological examination of the biopsy specimen showed acanthotic skin with ulceration with dense dermal chronic inflammation. There was inflammatory cell infiltration with necrosis, histiocytic proliferation, and giant cell formation. Many round to oval budding dimorphic fungi with yeast like spores were seen inside the giant cells and macrophages as well as tissue [Figure 2]a. Periodic acid–Schiff (PAS) staining revealed intracellular PAS positive round to oval organisms with a surrounding clear halo [Figure 2]b, suggestive of histoplasmosis. However, initial fungal culture did not yield any organisms. As disseminated histoplasmosis was suspected, a serology of HIV was performed, and the result was negative. An H. capsulatum specific polymerase chain reaction was performed on the biopsy sample and was positive in our patient. The patient showed prompt response to itraconazole in a dose of 200 mg twice daily for 1 month.
Figure 1: External color photograph of the patient showing pigmented ulcerated lesion involving the upper lid margin in the right eye

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Figure 2: Histopathological photomicrograph. (a) Inflammatory cell infiltration with necrosis, histiocytic proliferation, and giant cell formation in the dermal layers. Many round to oval budding dimorphic fungi with yeast like spores were seen inside the giant cells and macrophages (arrow) (H and E). (b) Intracellular periodic acid–Schiff positive round to oval organisms with a surrounding clear halo (asterix) (Periodic Acid–Schiff staining)

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   Discussion Top


Primary cutaneous histoplasmosis is very rare and occurs by local trauma or inoculation.[1]

Cutaneous histoplasmosis typically appears as oral ulcerations, but also reportedly as exfoliative-like erythroderma, erythema nodosum, molluscum-like papules, granulomatous plaques, mild eczematous dermatitis, papulonecrotic lesions, erythema multiforme, folliculitis/pustules, and a conjunctival granuloma.[2] Disseminated histoplasmosis can present with skin manifestations such as maculopapular rash. Though endemic in Africa and some parts of America, there have been few reports from India.[3],[4]

Review of literature revealed four reports until date of cutaneous histiocytosis presenting clinically as a malignancy.[5],[6],[7],[8] Three of these were manifestations of disseminated disease and in one patient reported by Merin et al.,[5] systemic disease could not be evaluated as the patient expired from complications of throat malignancy. Two of these were involving the eyelids.

Sen et al.,[6] reported a young patient with an ulcerative lesion of his left lower eyelid, medial canthus, and the medial quarter of the upper eyelid. The patient also had evidence of systemic involvement in the form of lung parenchymal disease and lymphadenopathy. The second case reported by Merin et al.,[5] was an ulcerated exophytic nodule along the palpebral conjunctival edge of the left lower eyelid margin, causing a mechanical ectropion in a 79-year-old immunosuppressed patient with throat malignancy. In this patient, however, it could not be ascertained as to whether it was primary cutaneous disease or manifestation of systemic disease.

O'Dorisio et al.,[9] reported several elevated, discrete lesions on the left eyelid of a 77-year-old man, who presented 17 years after untreated disseminated disease. The multiple, elevated lesions did not mimic a cutaneous malignancy. Our case is a primary cutaneous presentation of histoplasmosis in an immunocompetent patient.


   Conclusion Top


The unique presentation mimicking a basal cell carcinoma of the eyelid demonstrates the importance of considering the diagnosis and the need for histopathologic evaluation. Once the diagnosis of histoplasmosis is confirmed, systemic investigations to rule out disseminated disease must be performed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Kauffman CA. Histoplasmosis: A clinical and laboratory update. Clin Microbiol Rev 2007;20:115-32.  Back to cited text no. 1
    
2.
Lucas AO. Cutaneous manifestations of African histoplasmosis. Br J Dermatol 1970;82:435-47.  Back to cited text no. 2
    
3.
Nair SP, Vijayadharan M, Vincent M. Primaty cutaneous histoplasmosis. Indian J Dermatol Venereol Leprol 2000;66:151-3.  Back to cited text no. 3
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4.
Sanyal M, Thammayya A. Skin sensitivity to histoplasmin in Calcutta and its neighbourhood. Indian J Dermatol Venereol Leprol 1980;46:94-8.  Back to cited text no. 4
  Medknow Journal  
5.
Merin MR, Fung MA, Eisen DB, Lin LK. Histoplasmosis presenting as a cutaneous malignancy of the eyelid. Ophthal Plast Reconstr Surg 2011;27:e41-2.  Back to cited text no. 5
    
6.
Sen S, Bajaj MS, Vijayaraghavan M. Histoplasmosis of the eyelids – A case report. Indian J Pathol Microbiol 1999;42:495-7.  Back to cited text no. 6
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7.
Anandi V, Walter A, Jacob M, John L, Koshi G. Histoplasmosis simulating cutaneous carcinoma. Indian J Pathol Microbiol 1991;34:149-51.  Back to cited text no. 7
    
8.
Golda N, Feldman M. Histoplasmosis clinically imitating cutaneous malignancy. J Cutan Pathol 2008;35 Suppl 1:26-8.  Back to cited text no. 8
    
9.
O'Dorisio TM, Jasper DA, Sullivan J. A cutaneous manifestation of untreated disseminated histoplasmosis. Chest 1975;67:616-8.  Back to cited text no. 9
    

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Correspondence Address:
Namrata G Adulkar
Department of Orbit, Oculoplasty and Ocular Oncology, Aravind Eye Hospital and Postgraduate Institute, 1 Anna Nagar, Madurai - 652 020, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.182017

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