Isolated Primary Corneal Acremonium Eumycetoma

CASE REPORT

Isolated Primary Corneal Acremonium Eumycetoma: Case Report and Literature Review Downloaded from https://journals.lww.com/pages by 3Jrx4YHfwj7MGHRJzzbB/Vh+RluNcJj9DQ8r7raYNtcYsHeTucI+Qo+3Z8glmHegxxEiUgxMyHj8ipwW55JFxvOK+GREvy8lb/6MyVxxs3vkQ+dNkkPbO1HM+jCAwp5QWP0xhB6s8S4IxCeTseXeSzkBMO/QfunFX7EYf/N+B+k= on 09/24/2018

Ritika Mukhija, MD,* Noopur Gupta, MS, PhD,* Anita Ganger, MS,* Seema Kashyap, MD,† Nishat Hussain, MD,‡ Murugesan Vanathi, MD,* and Radhika Tandon, MD*

Purpose: To report an unusual case of isolated Acremonium eumycetoma presenting as a protuberant mass over the cornea.

Methods: Case report and literature review. Results: A 55-year-old male patient referred to our center with a case of perforated corneal ulcer with uveal tissue prolapse was examined in the casualty department and found to have central melt, approximately 8 mm, along with suspected uveal tissue prolapse. A provisional diagnosis of sloughed corneal ulcer with uveal prolapse was made along with differential diagnoses of fungal ball and infected foreign body granuloma. Tectonic penetrating keratoplasty under general anesthesia was planned. Intraoperatively, the suspected uveal (brown colored) tissue was found to be an epicorneal mass growing over an intact and infiltrated cornea. Histopathological and microbiological analysis of the epicorneal mass and host cornea revealed it to be a fungal ball (mass full of septate hyphae) with growth of Acremonium species on culture. The patient was administered topical and oral antifungal agents postoperatively, in addition to topical antibiotics and cycloplegics.

Conclusions: Isolated corneal Acremonium eumycetoma masquerading as a perforated corneal ulcer with prolapsed uveal tissue is a rare entity. Surgical intervention and appropriate antimicrobial therapy are key to successful outcome. Key Words: fungal keratitis, Acremonium, eumycetoma (Cornea 2018;00:1–3)

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ungal keratitis is a significant cause of ocular morbidity, especially in tropical and subtropical areas, with sequelae leading to irreversible corneal blindness.1 Acremonium, previously known as Cephalosporium, appear as hyaline septate hyphae on microscopic examination and are considered as an Received for publication May 30, 2018; revision received July 26, 2018; accepted July 27, 2018. From the *Cornea, Cataract, and Refractive Surgery Services, Dr. Rajendra Prasad Centre for Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi, India; †Ocular Pathology, All India Institute of Medical Sciences, New Delhi, India; and ‡Ocular Microbiology, All India Institute of Medical Sciences, New Delhi, India. The authors have no funding or conflicts of interest to disclose. Correspondence: Noopur Gupta, MS, PhD, Cornea, Cataract and Refractive Surgery Services, Dr. Rajendra Prasad Centre For Ophthalmic Sciences, All India Institute of Medical Sciences, New Delhi 110029, India (e-mail: [email protected]). Copyright © 2018 Wolters Kluwer Health, Inc. All rights reserved.

Cornea Volume 00, Number 00, Month 2018

unusual etiological agent for keratitis.2,3 Eumycetoma, or a fungal ball, is characterized by a colonization by true fungi within the entire mass of tissue, not just the surface.4 It has been reported that Acremonium is the causative pathogen.5,6 Herein, we report a patient presenting with isolated epicorneal and corneal Acremonium eumycetoma which was initially misdiagnosed as perforated corneal ulcer with uveal tissue prolapse. To the best of our knowledge, no case report of primary, isolated corneal Acremonium/Cephalosporium eumycetoma is documented in the literature.

CASE REPORT A 55-year-old male patient presented to a local ophthalmologist with history of sudden onset, painful, progressive diminution of vision in the right eye for the past 2 months and was diagnosed with a suspected fungal corneal ulcer with secondary glaucoma. On follow-up, he presented with a brown-colored mass protruding from the eye and was then diagnosed as a case of perforated corneal ulcer with uveal tissue prolapse as per the patient’s records. He was referred to a tertiary care center for further management. On presentation to our ocular emergency department, visual acuity was perception of light with accurate projection of rays in the right eye, and 6/12 (or 20/40) in the left eye. Intraocular pressure was digitally normal in the right eye. Diffuse conjunctival congestion with an unusual brownish corrugated mass protruding from the right eye, measuring approximately 8 mm in diameter and 5 to 6 mm in height with surrounding corneal haze, was observed on clinical examination. A provisional diagnosis of sloughed corneal ulcer with uveal or choroidal tissue prolapse was made, with a differential diagnosis of either a corneal fungal ball or a foreign body granuloma. The patient was taken for tectonic penetrating keratoplasty (PK) under general anesthesia as an emergency procedure using nonoptical grade tissue after obtaining written informed consent. Intraoperatively, the suspected uveal tissue was found to be an epicorneal mass growing over an intact cornea. After piecemeal removal of the mass, diffuse and apparently full-thickness involvement of the underlying cornea with brown-colored tissue was observed (Figs. 1A–H). Hence, a therapeutic PK was performed supplemented with intracameral voriconazole (50 mg/0.1 mL) injection. The epicorneal mass and host cornea were divided and both were sent for histopathological and microbiological analysis, which showed corneal infiltration with septate fungal hyphae, and the pigmented mass was reported to be a fungal ball consisting entirely of septate fungal hyphae (Figs. 2A–C). The same was corroborated with growth of Acremonium species on fungal culture in Sabouraud dextrose agar medium. Furthermore, a lactophenol cotton blue mount from slide culture revealed thin septate hyphae with erect unbranched phialides, oblong, one- or 2-celled conidia clustered at the tips of phialides that were easily disrupted from clusters, suggestive of Acremonium species (Fig. 2D). www.corneajrnl.com |

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FIGURE 1. A, Intraoperative image showing the protuberant pigmented mass at the beginning of the operation (simulating corneal melt with uveal prolapse). B and C, Intraoperative images showing piecemeal removal of the pigmented mass. D, Intraoperative image showing an intact cornea beneath the mass. E, Intraoperative image showing corneal infiltration after complete removal of the mass along with complete scraping and epithelial debridement. F, Intraoperative image showing measurement of the corneal pathology. G, Intraoperative image showing exudates in the anterior chamber. H, Intraoperative image at the end of the keratoplasty. Postoperatively, the patient was treated with antifungals and cycloplegics. Visual acuity on the first postoperative day was perception of light with accurate projection of rays with normal intraocular pressure. No pathological infiltration of the anterior segment was found on ultrasound biomicroscopy. On follow-up, the graft lost its transparency and the patient has been registered for optical keratoplasty of the right eye for visual rehabilitation. No signs of recurrence of infection were noted up to the 6-month followup visit.

DISCUSSION Acremonium eumycetoma, which usually develops following trauma, is a rare fungal infection of the skin, subcutaneous tissue, and bones.7,8 Eumycetoma is characterized by colonization of filamentous fungi as a fungal ball and the peculiar presence of sulfur granules, which is sometimes apparent on naked eye examination. Although a few reports of Acremonium keratitis have been found in the literature,

FIGURE 2. A, Histopathological cut section of the cornea fixed in formalin and stained by silver methenamine depicting infiltration with fungal hyphae (stained as black). B, Histopathological cut section of the cornea stained by hematoxylin and eosin depicting infiltration with fungal hyphae. C, Histopathological cut section of the pigmented mass stained by silver methenamine depicting numerous fungal hyphae (stained as black). D, Lactophenol cotton blue mount from slide culture depicting thin septate hyphae with erect unbranched phialides suggestive of Acremonium species.

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Acremonium eumycetoma has never been reported with typical features of a fungal ball and sulfur granules.9 We herein report, for the first time, culture-proven Acremonium eumycetoma in the eye, presenting as a de novo epicorneal pigmented mass. Our patient presented with isolated epicorneal and corneal eumycetoma without any inciting factor such as trauma or surgery, although minor trauma and poor hygiene cannot be completely ruled out as the patient was from an agricultural background, with frequent exposure to dust, soil, and vegetative matter. Similar presentation of a black-colored mass over the cornea, initially misdiagnosed as intraocular malignant melanoma and later found to be prolapsed orbital aspergilloma, has been previously reported.10 In our patient, the deeper corneal layers were also involved, so full thickness tectonic penetrating keratoplasty was performed. In cases with superficial corneal involvement with fungal ball formation, lamellar keratoplasty with antifungal therapy may prove effective. Our patient belonged to lower socioeconomic status and rural background; hence, high cost of medical therapy, frequent follow-up, higher chances of recurrence, and poor compliance were determining factors in favor of full-thickness surgical intervention. This rare manifestation of an uncommon disease highlights the importance of keeping a high level of clinical suspicion in such cases and undertaking necessary microbiological and histopathological evaluation to reach an accurate diagnosis

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Isolated Primary Corneal Acremonium Eumycetoma

and subsequently formulate an appropriate management plan. Primary epicorneal pigmented eumycetoma is as such an unusual presentation and has never before been reported with Acremonium as the causative pathogen. REFERENCES 1. Gupta N, Vashist P, Tandon R, et al. Prevalence of corneal diseases in the rural indian population: the corneal opacity rural epidemiological (CORE) study. Br J Ophthalmol. 2015;99:147–152. 2. Jurkunas U, Behlau I, Colby K. Fungal keratitis: changing pathogens and risk factors. Cornea. 2009;28:638–643. 3. Fincher RM, Fisher JF, Lovell RD, et al. Infection due to the fungus Acremonium (cephalosporium). Medicine (Baltimore). 1991;70:398– 409. 4. Gooptu S, Ali I, Singh G, et al. Mycetoma foot. J Fam Community Med. 2013;20:136–138. 5. Fahal A, Mahgoub ES, Hassan AME, et al. Mycetoma in the Sudan: an update from the mycetoma research centre, university of khartoum, Sudan. Wanke B, ed. PLoS Negl Trop Dis. 2015;9:e0003679. 6. Cordoba A, Fraenza L. Mycetoma from acremonium sp. Ann Dermatol Venereol. 2005;132:194. 7. Geyer AS, Fox LP, Husain S, et al. Acremonium mycetoma in a heart transplant recipient. J Am Acad Dermatol. 2006;55:1095–1100. 8. Guevara D, Wongkittiroch K, Goodman M, et al. Acremonium mycetoma: a case report and discussion. Cutis. 2011;88:293–295, 299. 9. Alfonso JF, Baamonde B, Santos J, et al. Acremonium fungal infection in 4 patients after laser in situ keratomileusis. J Cataract Refract Surg. 2004;30:262–272. 10. Naik MN, Vemuganti GK, Honavar SG. Primary orbital aspergilloma of the exenterated orbit in an immunocompromized patient. Indian J Med Microbiol. 2006;24:233–234.

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