March 2015

 

RESIDENTS

 

Academic grand rounds: Dean McGee Eye Institute

Conjunctival and cutaneous lesions following zoonotic transmission from an infected cat


by Jared R. Jackson, MD, and Alex Cohen, MD, PhD

 
   
Conjunctival granulomas

Conjunctival granulomas caused by Sporothrix schenckii involving the bulbar and palpebral conjunctiva, as well as the caruncle. A: Right upper lid. B: Left upper lid. C: Right lower lid. D: Left lower lid

Cutaneous lesions

Cutaneous lesions with areas of nodular erythema with ulceration and crusting. A: Left wrist. B: Left hand. C: Left elbow. D: Right forearm Source: Dean McGee Eye Institute

Introduction from R. Michael Siatkowski, MD, residency program director We chose this case to illustrate the broad range of pathology that residents from the Dean McGee Eye Institute encounter, as well as to highlight the work of clinician/scientist Alex Cohen, MD, one of our many award-winning educators at the University of Oklahoma.

Case presentation

A 48-year-old female was referred by an infectious disease specialist for evaluation of conjunctival lesions. The patient reported rescuing a stray cat with multiple ulcerated skin lesions approximately 2 months prior to presentation. Shortly after initial contact with the cat, she noticed erythematous bullae on her upper extremities. These bullae eventually burst, leaving crusted, ulcerated cutaneous lesions on both arms and hands. She denied any history of cat-scratch or trauma. She reported lesions inside both eyelids 1 month later, with bilateral, intermittent blurry vision secondary to excessive tearing. The patient also reported a chronic, low-grade fever during this time. The infectious disease specialist cultured and biopsied the cutaneous lesions; no organisms were identified. Past medical history was positive for arthritis and migraine, for which she was taking meloxicam and hydrocodone/acetaminophen as needed, and gastroesophageal reflux. She had a surgical history of appendectomy, Caesarian section, gastric banding, and tonsillectomy. She denied HIV positivity, history of immunosuppression, and intravenous drug use.

Best corrected visual acuity was 20/20 in each eye. Pupils, confrontation visual fields, and motility were normal. Slit lamp examination revealed raised, erythematous, granulomatous lesions of the palpebral conjunctiva and caruncles bilaterally, left greater than right. The right cornea was clear with no fluorescein staining, and the left cornea exhibited punctate epithelial erosions. The anterior chambers were deep and quiet in each eye. Dilated fundus examination was normal bilaterally. Cutaneous examination revealed erythematous, crusted lesions on the hands and arms.

Discussion

Conjunctival granulomas have multiple etiologies. In a patient with feline contact, Parinaud oculoglandular syndrome caused by Bartonella henselae should be high on the list of possible causes. Our patient did not have the typical glandular involvement or any history of cat scratch as would likely occur with Bartonella infection. Other causes of conjunctival granulomas, such as sarcoidosis, mycotic or parasitic disease, connective tissue disease, tuberculosis, and Kaposi sarcoma should also be considered. Given our patients cutaneous findings and apparent zoonotic transmission, mycotic or parasitic infection was suspected. After discussion with the infectious disease specialist and the veterinarian who treated the patients cat, we were highly suspicious of mycotic infection with Sporothrix spp. The patient was thus started on itraconazole 500 mg daily. The initial ocular treatment approach was to continue aggressive lubrication with artificial tears and monitor response to oral itraconazole therapy. The patient returned to clinic 1 week later with mildly enlarged lesions, especially in the right inferior palpebral conjunctiva, and decreased visual acuity to 20/30 -2 in the right eye and 20/30 in the left, attributed to disrupted tear film and epitheliopathy. After informed consent was obtained, we proceeded with excisional debulking biopsy of the inferior palpebral lesions bilaterally under local anesthesia. Excised tissue was sent for gram stain, culture, and pathologic evaluation. Intralesional and subconjunctival injections of fluconazole 2 mg/mL were administered to all lesions. The patient was also started on neomycin sulfate/polymyxin B sulfate/dexamethasone ophthalmic ointment twice daily, and continued on systemic itraconazole. Histopathology revealed mixed neutrophilic and granulomatous inflammation with giant cells and rare yeast. Gram stain showed no organisms and only a few white blood cells. Cultures grew Sporothrix schenckii.

Sporothrix schenckii is a dimorphic fungus endemic to temperate and tropical areas worldwide. Sporotrichosis typically presents with subacute to chronic cutaneous or lymphocutaneous involvement following inoculation through minor skin trauma contaminated by soil or decomposing vegetable matter.1 Airborne and zoonotic transmission may also occur. Pulmonary, osteoarticular, and disseminated forms have been documented, especially in immunocompromised hosts.1,2 Ocular and periocular tissues may also become involved via direct inoculation, with or without antecedent trauma, or through endogenous spread.3,4

Zoonotic transmission of sporotrichosis from cats to humans may occur without associated penetrating injury.4,5 It is suggested that cats may be the only animal vector that can readily infect humans because feline sporotrichosis is unique in the large number of organisms associated with cutaneous lesions, making non-traumatic transmission possible.5 Inoculation from feline hosts has been documented in various areas, including Brazil where it is epidemic in the Rio de Janeiro region.6 Occasional cases have been reported in India,7 the Netherlands,8 and the United States.4,5,9,10 Our review of the literature reveals this is the first reported case with ocular involvement following zoonotic transmission in the United States. Sporothrix may cause a variety of ophthalmic infections, including keratitis,11 conjunctival3,12,13 and retinal granulomas,14 scleritis,15 necrotizing retinochoroiditis,16 anterior uveitis,17 endophthalmitis,17,18,19,20 cutaneous eyelid infection,21 dacryocystitis,22 and even a mucormycosis-like presentation of invasive sinusitis.23 Ocular sporotrichosis in the United States was initially reported in 1910 by Gifford.12 To our knowledge, there have only been 15 cases of ophthalmic sporotrichosis documented in the United States since that time.

As was the case with our patient, clues in the history and physical exam can lead a physician to investigate Sporothrix spp. as the causative organism. Diagnosis is confirmed with positive culture or histopathology. Cultures can be most effectively obtained from material aspirated from lesions, excised tissue specimens, sputum, or body fluids. Organisms are often present only in small numbers, and may not be detected on potassium hydroxide preparation or gram stain. Growth may be evident in the mold phase within days of culture but may take weeks to definitively identify.1,24 The paucity of organisms in many cases means they are frequently not identified on histopathology, which shows a mixed granulomatous and pyogenic process.1 When Sporothrix spp. are isolated they can be visualized with Gomori methenamine silver stain on high magnification.

Sporotrichosis treatment varies with the location and extent of disease. Cutaneous and lymphocutaneous disease is typically treated with oral itraconazole 200 mg daily until 24 weeks after resolution of the lesions.1,25 Reports of successful treatment of conjunctival sporotrichosis have ranged between 100 and 300 mg of oral itraconazole daily2,6 with or without adjunctive topical fluconazole.3 Our patient may be the first case treated with a combination of oral itraconazole, therapeutic debulking, and intralesional fluconazole.

Outcome

At follow-up 2 weeks later, the patient noted improving symptoms and reported visual acuity back to baseline. Her conjunctival lesions were smaller and her cutaneous lesions were also improving. The neomycin sulfate/polymyxin B sulfate/dexamethasone ointment was tapered, and her conjunctival and cutaneous lesions resolved as she continued oral itraconazole treatment over the next few months.

Take-home points

Sporotrichosis should be suspected in patients with conjunctival granulomas, especially in cases with concomitant cutaneous lesions.

Zoonotic transmission of Sporothrix can occur without mucocutaneous trauma.

Lymphocutaneous and conjunctival sporotrichosis may be effectively treated with oral antifungal therapy, with or without adjunct topical therapy.

Excisional biopsy and intralesional antifungal therapy may also be beneficial in treating conjunctival granulomatous disease.

References

1. Kauffman, CS. Sporothrichosis. In: Goldman L, Schafer AI. Goldmans Cecil Medicine, 24th edition. Maryland Heights: W.B. Saunders, 2011:19851986.

2. Freitas DF, de Siqueira Hoagland B, do Valle AC, et al. Sporotrichosis in HIV-infected patients: report of 21 cases of endemic sporotrichosis in Rio de Janeiro, Brazil. Medical Mycology 2012;50(2):1708.

3. Hampton DE, Adesina A, Chodosh J. Conjunctival sporotrichosis in the absence of antecedent trauma. Cornea 2002;21(8):8313.

4. Reed KD, Moore FM, Geiger GE, et al. Zoonotic transmission of sporotrichosis: case report and review. Clinical Infectious Disease 1993;16(3):3847.

5. Dunstan RW, Langham RF, Reimann KA, et al. Feline sporotrichosis: a report of five cases with transmission to humans. Journal of the American Academy of Dermatology 1986;15(1):3745.

6. Schubach A, de Lima Barros MB, Schubach TM, et al. Primary conjunctival sporotrichosis: two cases from a zoonotic epidemic in Rio de Janeiro, Brazil. Cornea 2005;24(4):4913.

7. Yegneswaran PP, Sripathi H, Bairy I, et al. Zoonotic sporotrichosis of lymphocutaneous type in a man acquired from a domesticated feline source: report of a first case in southern Karnataka, India. International Journal of Dermatology 2009;48(11):1198200.

8. Hugen L, Koumans H. Sporotrichosis in a cat and its owner. Tijdschrift voor Diergeneeskunde 2006;131(24):9189.

9. Rafal ES, Rasmussen JE. An unusual presentation of fixed cutaneous sporotrichosis: a case report and review of the literature. Journal of the American Academy of Dermatology 1991;25:92832.

10. Rees RK, Swartzberg JE. Feline-transmitted sporotrichosis: A case study from California. Dermatology Online Journal 2011;17(6):2.

11. Sun KY, Ch WC. Corneal sporotrichosis. A case report. Chinese Medical Journal 1966;85(1):446. 12. Gifford. Sporotrichosis of the eyeball and eyelids. Ophthalmic Record 1910 p. 573.

13. Kashima T, Honma R, Kishi S, et al. Bulbar conjunctival sporotrichosis presenting as a salmon-pink tumor. Cornea 2010;29(5):5736.

14. Curi AL, Flix S, Azevedo KM, et al. Retinal granuloma caused by Sporothrix schenckii. American Journal of Ophthalmology 2003 Jul;136(1):2057.

15. Brunette I, Stulting RD. Sporothrix schenckii scleritis. American Journal of Ophthalmology 1992;114(3):3701.

16. Font RL, Jakobiec FA. Granulomatous necrotizing retinochoroiditis caused by Sporotrichum schenkii. Report of a case including immunofluorescence and electron microscopical studies. Archives of Ophthalmology 1976;94(9):15139.

17. Cartwright MJ, Promersberger M, Stevens GA. Sporothrix schenckii endophthalmitis presenting as granulomatous uveitis. British Journal of Ophthalmology 1993;77(1):612.

18. Cassady JR, Foerster HC. Sporotrichum schenckii endophthalmitis. Archives of Ophthalmology 1971;85(1):71-4.

19. Levy JH. Intraocular sporotrichosis. Report of a case. Archives of Ophthalmology 1971;85(5):5749. 20. Kurosawa A, Pollock SC, Collins MP, et al. Sporothrix schenckii endophthalmitis in a patient with human immunodeficiency virus infection. Archives of Ophthalmology 1988;106(3):37680.

21. Iyengar SS, Khan JA, Brusco M, et al. Cutaneous Sporothrix schenckii of the human eyelid. Ophthalmic Plastic and Reconstructive Surgery 2010;26(4):3056.

22. Freitas DF, Lima IA, Curi CL, et al. Acute dacryocystitis: another clinical manifestation of sporotrichosis. Memorias do Instituto Oswaldo Cruz 2014;109(2):2624.

23. Agger WA, Caplan RH, Maki DG. Ocular sporotrichosis mimicking mucormycosis in a diabetic. Annals of Ophthalmology 1978;10(6):76771.

24. Falqueto A, Maifrede SB, Ribeiro MA. Unusual clinical presentation of sporotrichosis in three members of one family. International Journal of Dermatology 2012;51(4):4348.

25. Kauffman CA, Bustamante B, Chapman SW, et al. Clinical practice guidelines for the management of sporotrichosis: 2007 update by the Infectious Diseases Society of America. Clinical Infectious Diseases 2007;45(10):125565.

Contact information

Cohen
: Alex-Cohen@dmei.org
Jackson: Jared-Jackson@dmei.org
Siatkowski: RMichael-Siatkowski@dmei.org

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Conjunctival following zoonotic transmission from an infected cat Conjunctival following zoonotic transmission from an infected cat
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