Anterior segment optical coherence tomography and ultrasound biomicroscopy in the diagnosis of subconjunctival mycosis mimicking nodular scleritis

  1. Pratima Vishwakarma 1,
  2. Somasheila I Murthy 1,
  3. Vineet Joshi 1 and
  4. Dilip Kumar Mishra 2
  1. 1 Shantilal Shangvi Cornea Institute, LV Prasad Eye Institute, Hyderabad, India
  2. 2 Ophthalmic Pathology Services, LV Prasad Eye Institute, Hyderabad, India
  1. Correspondence to Dr Somasheila I Murthy; smurthy@lvpei.org

Publication history

Accepted:09 Jan 2023
First published:17 Jan 2023
Online issue publication:17 Jan 2023

Case reports

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Abstract

A female patient in her 20s presented with a bulbar conjunctival mass lesion that was diagnosed as nodular scleritis. It was treated with topical and oral steroids in another hospital. Imaging was done using anterior segment optical coherence tomography and ultrasound biomicroscopy, which helped to rule out scleritis and subconjunctival cysticercosis. Histopathology of the excision biopsy specimen revealed fungal filaments. Topical antifungals were started, and the condition resolved without recurrence after therapy. Although rare, infections should be considered in the differential diagnosis of conjunctival mass lesions. Imaging can help to rule out other entities and guide towards appropriate management.

Background

Subconjunctival mycosis (fungal ball) is a rare presentation and can be easily misdiagnosed as nodular episcleritis or scleritis. Sporothrix schenckii, a dimorphic fungal pathogen, has been identified as the most common aetiological agent.1–4 Previously published case reports and series have shown that these lesions respond well to antifungal agents, such as topical fluconazole, oral itraconazole or potassium iodide.1–4 In this case report, we report the utility of ocular imaging techniques in diagnosing subconjunctival mycosis and eliminating other differential diagnoses.

Case presentation

A woman in her 20s presented with reports of a painless, progressive mass lesion over the conjunctival surface of her left eye. The problem was observed over a month prior to the visit. She was diagnosed with nodular scleritis in another hospital. She was treated with topical and oral prednisolone in tapering doses for 1 month. There was no history of penetrating trauma or foreign body entry. On examination, her vision was 20/20 in both eyes. A solid, non-mobile, congested nodular lesion was observed over the inferotemporal bulbar conjunctiva (figure 1A). The lesion showed internal transilluminance on an optical section of the slit-lamp biomicroscope (figure 1B). Our main differential was conjunctival cysticercosis, and we decided to investigate further.

Figure 1

: Slit-lamp photograph of the left eye showing inferotemporal nodular bulbar conjunctival lesion with congestion (A) and the transillumination effect (B). AS-OCT (C, D). The arrows in (D) indicate the anterior and posterior edges of the lesion. UBM (E, F) image showing subconjunctival cystic lesion without scleral invasion. The arrows delineate the edges of the lesion. AS-OCT, anterior segment optical coherence tomography; UBM, ultrasound biomicroscopy.

Investigations

She underwent a spectral domain anterior segment optical coherence tomography (AS-OCT) (Optovue, Fremont, California) scan which revealed a smooth nodular elevated lesion with thickened zones of hyperreflectivity (figure 1C,D) caused by the compaction of the subconjunctival tissue, suggesting a cyst wall. The posterior extent of the lesion could not be delineated because of the resolution limits of the machine. Ultrasound biomicroscopy (UBM) (Aviso S, Quantel Medical, Paris, France V.5.0.0) confirmed that the lesion was confined to the subconjunctival space. It also showed the uniform hyperechoic nature of the mass without extending to the anterior sclera. No hyperreflective lesion corresponding to a scolex was observed (figure 1E,F).

Differential diagnosis

The patient was initially diagnosed with nodular scleritis. Clinically, the lesion appeared raised with congestion and tenderness similar to nodular scleritis. However, the lack of severe pain at onset and no improvement despite treatment with steroids for adequate duration ruled it out. Nodular episcleritis is a similar, painless differential. However, it also responds to steroids. We considered a diagnosis of conjunctival cysticercosis because of the cystic appearance and the presence of transillumination under a slit-lamp biomicroscope. However, ultrasonography confirmed the absence of the pathognomonic hyperechoic scolex within the lesion.5 Another differential diagnosis was a benign conjunctival stromal tumour. It appears similar to a subconjunctival solid mass; however, it lacks the transillumination effect.6 Finally, melanoma and amelanotic melanoma might also mimic this presentation; however, the lack of feeder vessels and the presence of transillumination rule them out. Moreover, AS-OCT would have shown its characteristic features, such as hyper-reflective subepithelial lesion with normal thickness and the appearance of a basal epithelium or an epithelial cleavage, indicating atypical melanocytes.7–9

Treatment

The patient underwent an excision biopsy of the cyst under local anaesthesia. A peritomy was performed adjacent to the lesion to excise the cyst in toto. Pus was observed on the cyst surface, oozing out of the cystic cavity. The cyst walls were necrotic and had to be excised piecemeal. The bed was gently debrided, and an amniotic membrane graft (AMG) was placed over the bare scleral area and secured with fibrin glue. The patient was started on topical levofloxacin 1.5% six times a day and oral ciprofloxacin 500 mg two times per day for a week postoperatively.

The pus was sent for microscopy (smears seen with grams and potassium hydro-oxide calcofluor white stains), and the cyst wall fragments were sent for histopathology. The pus was inoculated in blood agar, chocolate agar, potato dextrose agar and thioglycolate broth. The microbiology of the samples was negative on the smear and culture. Histopathology of the excised tissue showed plaque of fungal filaments with necrosed fibrocollagenous tissue (figure 2A). The Gomori methenamine silver stain confirmed thin septate fungal filaments with few cut ends of fungal hyphae (figure 2B,C). This established the diagnosis of subconjunctival mycosis. We started the patient on topical natamycin 5% 2 hourly (stepped down after 21 days) and oral ketoconazole 200 mg two times per day. Both medications were stopped after 36 days.

Figure 2

: (A) H&E stained sample with necrotic tissue showing multiple fungal filaments (40x) (black arrows); (B) GMS stained sample showing an entangled mass of fungal filaments (black arrows) with 10x and (C) 40x (red arrows). Slit-lamp photograph after excision biopsy: (D) 2 days post-surgery, showing conjunctival congestion with AMG in place; (E) 10 days post-surgery, AMG is integrating and congestion has decreased; (F) 2 months post-surgery, AMG well-integrated with mild conjunctival fibrosis. AMG, amniotic membrane graft; GMS, Gomori methenamine silver.

Outcome and follow-up

One week postoperatively, mild congestion was noted over the bulbar conjunctiva in the area of the cyst excision and AMG placement. The antifungals were continued for 5 weeks. Four months postoperatively, the condition and the congestion over the area of excision resolved with mild conjunctival fibrosis and the integration of the AMG (figure 2D–F). No recurrences were noted.

Discussion

Subconjunctival mycosis (fungal ball) is a rare diagnosis in a healthy setting. Zoonotic transmission, especially via contact with cats, is the most common cause. Other causes include trauma with vegetative matter.1 3 In two large case series from South America (21 and 26 cases, respectively), the endemic nature of ocular sporotrichosis was reported.3 4 Arinelli et al 3 showed ocular involvement of the adnexa in 20 cases of sporotrichosis. They also showed one case involving the palpebral conjunctiva. Ramírez Soto4 focused mainly on bulbar conjunctivitis. They found sporotrichosis cultures in all 26 cases using conjunctival swabs. These cases showed bulbar conjunctival nodular involvement, and mycosis was not the first diagnosis. The patients were initially misdiagnosed and treated for scleritis (similar to our case), inflamed pterygium, or conjunctival tumours.3 6 7

In a case report by Sayyad et al, an initial diagnosis of melanoma was made for a pigmented bulbar conjunctival lesion. The patient was a contact lens wearer who underwent scleral buckle surgery 2 years ago. A fungal infection from Scytalidium sp was confirmed after removing the mass.7

Similar cases have been reported with pigmented bulbar conjunctival lesions mimicking melanoma. Histopathological examination of the excision biopsy sample revealed dematiaceous fungi.10–12 Similar to our case, steroids were initiated in these cases too.3 Despite the administration of steroids, there was no propagation of the infection. This can be attributed to the sequestration of the fungal colony within the cyst wall, possibly limiting the penetration of the steroids.

There are several differentials for this entity, including scleritis and melanoma. Therefore, it is paramount to detect whether this is a relatively benign lesion, such as a fungal ball. In this context, we found both AS-OCT and UBM useful in the diagnosis of this condition. The images on both OCT and UBM show a hyperreflective or hyperechoic cystic lesion, respectively. UBM can confirm the subconjunctival location of the lesion and the absence of scleral invasion. The absence of scolex in the UBM can help rule out conjunctival cysticercosis.

Excision biopsy is the gold standard for establishing the final diagnosis and can be curative in most cases. In our case, the fungal species could not be identified because the culture did not grow. This could be because only the pus was cultured, not the solid material from the abscess, such as the bed and sides of the lesion. Histopathology can show characteristic granulomas.3 In cases with tarsal conjunctival involvement, symblepharon formation has also been observed.3 In our case, AMG was placed over the excision area to promote rapid healing and prevent further ocular sequelae, such as severe conjunctival fibrosis or symblepharon formation.

We conclude that this clinical entity needs a high index of suspicion in non-endemic areas. Imaging is useful to eliminate other differentials. The use of steroids should be deferred till the diagnosis is established by histopathological assessment of the excised lesion. To the best of our knowledge, this is the first report to highlight the role of imaging for this entity.

Learning points

  • Subconjunctival mycosis can be a rare presentation and can be misdiagnosed as other entities, such as scleritis.

  • The role of anterior segment optical coherence tomography and ultrasound biomicroscopy imaging is to delineate the level of the lesion and rule out differentials, such as nodular scleritis, conjunctival cysticercosis, and conjunctival stromal tumour. Therefore, they can be useful in these cases.

  • A definite diagnosis can be established by excision biopsy.

  • Treatment with oral and topical antifungals helps to resolve the infection.

Ethics statements

Patient consent for publication

Footnotes

  • Contributors All persons designated as authors qualify for authorship, and all those who qualify are listed. Each author has participated sufficiently in the work to take public responsibility for appropriate portions of the content. PV collected the data, conceptualised and drafted the manuscript. SIM edited the manuscript. VJ, and DKM finalised the article. PV, VJ and DKM were involved in the management of the patients and contributed to the interpretation of the results.

  • Funding This study was funded by Hyderabad Eye Research Foundation (LEC-BHR-R-11-22-958).

  • Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

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