Purpose
To describe the clinical and epidemiologic characteristics of sporotrichosis in ocular adnexa and give an insight into factors associated with this condition.
Design
Retrospective case series and literature review.
Methods
We retrospectively reviewed all cases of sporotrichosis in ocular adnexa between 2004 and 2014 in the Santa Teresa Clinic of Abancay, Peru and reviewed all case reports of sporotrichosis in ocular adnexa in the literature.
Results
We reviewed records of 21 patients with sporotrichosis in ocular adnexa; 12 (57.1%) of them were male and their median age surrounded 9 years. In our series, 19 patients had lesions in the eyelids and 2 in the eyebrows. The lymphocutaneous form occurred in 62% of them. Ten patients (47.6%) were cured with potassium iodide. Among 65 patients with sporotrichosis in ocular adnexa (our 21 patients and 44 from the literature), the average age was 9 years, and 78% were ≤15 years of age; 54% were male. The lesions were more frequent on the eyelids (n = 53 [82%]), followed by the lacrimal gland (n = 5), conjunctiva (n = 4), and eyebrows (n = 3). The lymphocutaneous clinical form (54%) was the most frequent. Fifty patients were cured: 31 of them with potassium iodide, 16 with itraconazole, and 3 with a combination including potassium iodide, itraconazole, and fluconazole. Twenty-nine patients (44.6%) resided in a hyperendemic region, and 5 patients reported contact with cats that had sporotrichosis.
Conclusions
Sporotrichosis in ocular adnexa is associated with children ≤15 years of age, and in 82% of these cases the disease is limited to the eyelids.
Sporotrichosis is a rare mycosis in the ocular adnexa caused by various species of the genus Sporothrix . “Ocular adnexa” refers to tissues and structures surrounding the eye, including the orbital soft tissue, lacrimal glands, conjunctiva, eyelids, and eyebrows. Sporotrichosis in ocular adnexa typically manifests in the eyelids and eyebrows with accompanying regional lymphadenopathy (lymphocutaneous form) following traumatic inoculation, or as fixed-cutaneous ulceration or conjunctivitis following exposure. Acute dacryocystitis by Sporothrix spp has been recently reported, although it is still uncommon. In recent decades, there has been increased recognition of this condition as more cases have been reported in the palpebral and conjunctival region. All these manifestations can lead to severe and chronic complications, since optimal management remains unclear.
To date, reports on sporotrichosis in ocular adnexa are limited, and no comparative study has been published. Therefore, in this report, we describe clinical and epidemiologic findings in 21 cases of sporotrichosis with ocular adnexa lesions and review all case reports of the literature to describe the clinical manifestations, diagnosis, treatment, epidemiology, and outcomes of patients with these manifestations and give insight into factors associated with this condition.
Methods
Patient Selection
In this retrospective study, we evaluated the clinical and epidemiologic features of patients with sporotrichosis in ocular adnexa. The study was approved by the institutional review board of the Institute of Tropical Diseases and Biomedicine of the National University of San Antonio Abad of Cusco, Peru. We conducted a detailed retrospective review and analysis of clinical records of patients who were diagnosed with sporotrichosis in ocular adnexa from January 2004 to December 2012 at the Santa Teresa Clinic of Abancay and a private laboratory in Abancay, Peru.
Definitions
Sporotrichosis in ocular adnexa is defined as lesions of the eyelids, the lacrimal gland (dacryocystitis), conjunctiva, and eyebrows. Skin lesions (eyelid and eyebrow) may be papular, nodular, ulcerative, or infiltrative (plaque-like) or may show a combination of these features. Dacryocystitis is defined as inflammation of the lacrimal sac, with reddening, edema, and the presence of a painful area of induration overlying the nasolacrimal sac, specifically just below the anatomic boundary of the medial canthal ligament. Conjunctival sporotrichosis is defined as a granulomatous lesion accompanied by local hyperemia, secretion, and edema, with or without regional lymph node enlargement. On the basis of these definitions, cases were included in the review if they fulfilled the following criteria: (1) a lesion or clinical syndrome consistent with sporotrichosis in ocular adnexa, and (2) cases diagnosed on the basis of histologic analysis or positive culture, with the recovery of the isolate from tissue or clinical sample. Cases in which isolation of fungi from tissue or a lesion sample occurred in the setting of widely disseminated disease in the eyeball were excluded; that is, cases of endogenous endophthalmitis as part of disseminated disease were excluded as caused by Sporothrix , and because the intraocular sporotrichosis is associated with immunocompromised patients.
Patient Data
We reviewed all available paper-based and electronic clinical records pertaining to these cases using a standard data collection form. We collected information on exposure history, demographic features, clinical presentation, treatment, and outcome.
Literature Review
We searched PubMed and regional databases (Latin American and Caribbean Health Sciences Literature [LILACS] and the Scientific Electronic Library Online [SciELO]) to identify case reports and case series of sporotrichosis in ocular adnexa. We applied no language or date restrictions. Combinations of the keywords “sporotrichosis,” “mycosis,” “palpebral,” “eyelid,” “eyebrow,” “lacrimal gland,” “conjunctiva,” “dacryocystitis,” “ Sporothrix ,” and “ Sporothrix schenckii ” were used for the literature search. The search was supplemented by reviewing the reference lists of all selected studies. We did our last search on January 30, 2015. Patients were excluded if they had intraocular sporotrichosis.
The following information was extracted from each report: (1) characteristics of patients (sex, age, country of origin, underlying conditions); (2) characteristics of infection (infectious agent, site of infection, diagnosis methods); (3) type of intervention (treatment regimens and outcomes); and (4) publication year. However, because of the lack of complete information in some publications, it was imposible to use all the avalible data in some reports.
Statistical Analysis
Descriptive results are presented as medians for continuous variables and as frequencies and percentages for categorical variables. Statistical analysis was performed using SPSS software version 19.0 (SPSS, Inc, Chicago, Illinois, USA). The χ 2 test, if needed, was used in comparing patient outcomes. Differences with P < .05 were considered significant.
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