Case Resolution
Sporotrichosis primarily involves the skin and surrounding lymphatics following traumatic inoculation of S. schenckii. The lesion at inoculation site can be multiple, may remain as such or nodules appear along the lymphatics. The histopathology is usually non-specific and variable. Demonstration of cigar-shaped, oval to round or single budding forms of the yeast is diagnostic but is rarely possible due to their scanty presence. Clinicopathologic correlation is thus important for diagnosis in most cases while culture of S. schenckii will be confirmatory. The initial diagnosis in our patients was mainly clinical correlating with histology/culture.
Hematogenous spread or multiple traumatic implantations of the fungus is perhaps responsible for cutaneous dissemination and may even be seen in individuals apparently without any predisposing factors for immunosuppression. Although inoculation of the infected material usually occurs after thorn pricks, cuts or blunt injuries, contamination of a pre-existing wound with infected material is perhaps more important for clinical development of the disease. This is also evident in our case 1 who had multifocal or disseminated cutaneous sporotrichosis. He primarily started with noduloulcerative BCC (rodent ulcer) which got contaminated possibly following frequent topical application of herbal pastes perhaps infected with causative fungus. He also developed clinicopathologically typical lymphocutaneous sporotrichosis involving right upper limb apparently from contamination of some innocuous occupational injury over hand most likely from facial lesion/herbal pastes.
Extracutaneous disseminated sporotrichosis can present as sinusitis, osteoarticular, pulmonary, ocular or central nervous system disease. Although osteoarticular sporotrichosis is an uncommon manifestation of systemic disease, it remains the most common extracutaneous presentation and has been reported in immunocompetent as well as in immunocompromised patients. Pulmonary disease is rare and occurs following inhalation of conidia. Clinically cough, low grade fever, weight loss, mediastinal lymphadenopathy, cavitation resembling tuberculosis, and fibrosis are usual. Our case 2, an otherwise immunocompetent patient, perhaps started with pulmonary disease that was mistaken for pulmonary tuberculosis. She developed disseminated systemic sporotrichosis in the form of multiple skin lesions, osteomyelitis of a phalanx and most likely effusion of a knee joint. Skin lesions when developed provided prima facie evidence of systemic disease, later confirmed mycologically. Although treatment with SSKI benefited her, only amphotericin B or itraconazole should be preferred for extracuateous sporotrichosis.
Skin lesions are mostly asymptomatic, rarely mildly pruritic or painful. Apart from classic cutaneous sporotrichosis uncommon lesional morphology mimicking facial cellulitis, erysipelas, rosacea, sarcoidosis, BCC, keratoacanthoma, soft tissue sarcoma and pyoderma gangrenosum has been described. To the best of our knowledge intensely pruritic, nodular prurigo-like lesions as seen in our case 3 are not reported hitherto in the literature. Trauma from repeated scratching apparently contributed in formation and spread of new lesions due to autoinoculation along excoriations resulting in linear and bizarre configuration. Although granulomatous histology was suggestive of mycobacterial (typical or atypical) infection, there was no response to ATT while favorable response to SSKI was suggestive as well as curative in her.
Despite being from this high endemic region all our patients remained undiagnosed before dermatological consultation was sought. To overcome the problem of delayed or no diagnosis, importance of a strong clinical suspicion thus needs be emphasized. Timely initiation of an effective treatment is imperative to prevent chronicity and morbidity. A favorable response to SSKI is almost diagnostic especially in the absence of mycological support.Response is usually evident within 2 weeks and healing occurs in 4-8 weeks. However, the duration of therapy remains uncertain and treatment must be continued for another 4-8 weeks after the clinical cure for mycological cure. All our patients showed clinical cure with SSKI in 12 weeks. However, when cost is not a consideration, itraconazole (100-200 mg/d) given for 3-6 months is the drug of choice in all forms of sporotrichosis as well as in patients intolerant to SSKI.
