First case report of isolated aspergillus dacryoadenitis.

We report a case of isolated Aspergillus dacryoadenitis. A 23-year-old male presented with dull ache, diffuse swelling in superolateral quadrant of the right orbit and proptosis for 4 months. Ocular examination showed conjunctival congestion, discharge in the fornix and palpable lacrimal gland (LG) mass. Routine hematological investigations followed by computed tomography scan of orbits were done. He did not respond to a course of systemic and topical antibiotics. Lateral orbitotomy with extended lid crease incision was performed with excision biopsy of LG. Abundant blackish material was found in the LG intraoperatively. The specimen was sent for histopathological examination (HPE). HPE report showed Aspergillus. Thorough ENT and systemic evaluation ruled out any other site with the fungus. To the best of our knowledge, this is the first case report of Aspergillus infection in LG.

Orbital aspergillosis is a relative uncommon orbital infection. Orbital aspergillosis usually arises from nasal and paranasal fungal sinusitis. Usually seen in immunocompromised individuals.

Case Report

A 23-year-old male presented with dull ache, redness, diffuse swelling in superolateral quadrant of the right orbit and proptosis for 4 months [Fig. 1a]. He had no history of trauma, sinusitis, steroid intake, diabetes, and no other significant medical history. On examination, his best-corrected visual acuity of the right eye (RE) was 20/15, N6 and left eye (LE) was 20/20, N6. RE examination showed diffuse right upper lid edema. A firm, tender mass was palpated in the superotemporal quadrant of the orbit. Hertel's exophthalmometry showed 2 mm proptosis of the RE with 1 mm inferior displacement. Extraocular movements were full. Slit lamp examination of RE revealed conjunctival congestion with discharge and rest of the anterior segment examination was within normal limit. Fundus examination was normal. LE examination was unremarkable. General and systemic examinations were normal. Routine hematological investigations were normal. Computed tomography (CT) scan showed a 17 mm × 19 mm × 20 mm well-defined, homogenous, enhancing lacrimal gland (LG) mass with no indentation of the globe [Fig. 1b–d]. No calcification or bony erosion was noted. Acute dacryoadenitis was suspected and was given a course of systemic and topical antibiotics (tablet amoxicillin and clavulanate potassium, 500 mg/125 mg and topical moxifloxacin eye drops). The patient did not respond. Lateral orbitotomy with extended lid crease incision was performed with excision biopsy of LG. Abundant blackish material was found in the LG intraoperatively [Fig. 2]. The specimen was sent for histopathological examination (HPE) in formalin and in saline for KOH preparation. HPE report showed lobules of benign LG acini surrounded by dense lymphoplasmacytic inflammation [Figs. 3 and 4]. Occasional necrotizing granulomas and pigmented island of narrow, septate, branching fungal hyphae along with few nonbudding spores suggestive of Aspergillus infection. The KOH preparation also showed filamentous, branching, and septate hyphae. Culture was done in sabouraud dextrose agar which isolated Aspergillus. Thorough ENT examination was done but did not reveal any evidence of sinus pathology. A diagnosis of isolated LG aspergillosis was therefore made. We started patient on systemic antifungals, i.e., tablet Itraconazole 200 mg TDS for first 3 days followed by maintenance dose twice day for 3 months. The patient was kept under follow-up of 1, 2, 6 weeks.

Figure 1

(a) Clinical photograph of the patient with right eye proptosis inward and downward. (b-d) Well-defined, homogenous, enhancing lacrimal gland mass with no indentation of the globe and without calcification

Figure 2

Intraoperative photographs showing exposure and excision of the lacrimal gland and black material within the lacrimal gland mass

Figure 3

Lacrimal gland acini with dense inflammation and fungal colonies (H and E)

Figure 4

Infiltration of nerve by fungal hyphae and filamentous branching septate fungi (Gram-stain)

Discussion

Our patient had clinical features of dacryoadenitis. The etiology of dacryoadenitis can be infectious and inflammatory. Infectious causes are viruses, bacteria, fungi, and parasites.[1] Though infectious causes include fungi, isolated fungal dacryoadenitis has never been reported.

Aspergillus is a ubiquitous fungus found particularly in soil and decaying vegetation. There are four main types of Aspergillus, i.e. Aspergillus fumigatus, Aspergillus flavus, Aspergillus lentulus, and Aspergillus nidulans. Of which Aspergillus fumigates and A. flavus are the most common fungal contaminants of the sinuses, and thus have the potential to infect the orbit. Cases of invasive sino-orbital aspergillosis in healthy individuals have often been reported from Sudan, India, and other tropical areas.[2] Since these areas share a hot and humid climate favorable for fungal growth and possibly a larger number of fungal spores in the environment, even healthy individuals are exposed to fungal spores resulting in increased risk of sino-orbital aspergillosis. Nevertheless, it is most commonly seen in immunocompromised hosts. Risk factors for aspergillosis are summarized in Table 1.

Table 1

Risk factors for aspergillosis[34]

The spectrum of Aspergillus infection is complex and may present in four fundamental patterns:[5] Allergic, noninvasive, invasive, and fulminant. In nonimmunocompromised individuals, two modes (allergic, noninvasive) of presentation of Aspergillus are typically observed, both of which are usually less aggressive. Allergic Aspergillus sinusitis is a cause of chronic sinusitis seen in atopic but otherwise normal individuals[6] and is thought to be a combination of type I and type III immunological reactions to Aspergillus antigens via granulomatous inflammation. Noninvasive disease results in the formation of an aspergilloma (a fungus ball) and behaves like chronic sinusitis.[7] In contrast, immunocompromised patients typically demonstrate either invasive aspergillosis, which is associated with granulomatous inflammation and fibrosis, or fulminant aspergillosis, which is characterized by diffuse vascular invasion, thrombosis, and ultimately tissue necrosis.[7] Showers of Aspergillus emboli from an infected valve or other source may infarct small vessels, while larger vessels may become thrombosed in situ.[5]

We need to put other causes of LG inflammatory diseases and neoplasms as differential diagnosis. Both CT and MRI have advantages in diagnosing orbital aspergillosis. The presence of dense intraluminal calcifications on CT scan is highly indicative of aspergillosis, but their absence does not rule it out since the calcifications may be present in only 50% of infected patients. When present, calcifications with a density >2000 HU are particularly suggestive of aspergillosis.[8] Fine-needle aspiration is one of the procedures to be indicated, especially in patients too ill to undergo more definitive biopsy or when an orbitotomy is not acceptable to the patient. Orbital aspergillosis in LG has never been reported in the literature till to date. Hence, one should suspect fungal infection in a case presenting as dacryoadenitis and not responding or improving with empiric antibiotic therapy.

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Conflicts of interest

There are no conflicts of interest.