Necrotizing fungal preseptal cellulitis secondary to Candida orthopsilosis in a diabetes mellitus patient – A rare case report and review of literature

Nirupama Kasturi; Tanmay Gokhale; Sandip Sarkar; Farnaz Yazeer; Nelzo Thomas; Rakesh Singh

doi:10.4103/ijo.IJO_2380_21

CASE REPORT

Year : 2022 | Volume : 2 | Issue : 2 | Page : 546-548

Necrotizing fungal preseptal cellulitis secondary to Candida orthopsilosis in a diabetes mellitus patient – A rare case report and review of literature

Nirupama Kasturi¹, Tanmay Gokhale¹, Sandip Sarkar¹, Farnaz Yazeer¹, Nelzo Thomas², Rakesh Singh³
¹ Department of Ophthalmology, Jawaharlal Postgraduate Medical Education and Research, Puducherry, India
² Department of Plastic Surgery and Burn, Jawaharlal Postgraduate Medical Education and Research, Puducherry, India
³ Department of Microbiology, Jawaharlal Postgraduate Medical Education and Research, Puducherry, India

Date of Submission	12-Sep-2021
Date of Acceptance	08-Dec-2021
Date of Web Publication	13-Apr-2022

Correspondence Address:
Sandip Sarkar
Department of Ophthalmology, Jawaharlal Postgraduate Medical Education and Research, Puducherry - 605 006
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_2380_21

Abstract

Non-albicans candida infection is an emerging threat in immunocompromised patients like those with diabetes mellitus, bone marrow transplant recipients, or HIV-positive patients. We report a rare case of necrotizing preseptal cellulitis extending to the cheek caused by Candida orthopsilosis in a young diabetic patient. Fungal etiology must be considered as a possible cause of preseptal and facial cellulitis in the setting of uncontrolled diabetes mellitus, which is unresponsive to broad-spectrum antibiotics even in the absence of a local nidus of infection in the paranasal sinuses or oral cavity.

Keywords: Candida orthopsilosis, ectropion, preseptal cellulitis

How to cite this article:
Kasturi N, Gokhale T, Sarkar S, Yazeer F, Thomas N, Singh R. Necrotizing fungal preseptal cellulitis secondary to Candida orthopsilosis in a diabetes mellitus patient – A rare case report and review of literature. Indian J Ophthalmol Case Rep 2022;2:546-8

How to cite this URL:
Kasturi N, Gokhale T, Sarkar S, Yazeer F, Thomas N, Singh R. Necrotizing fungal preseptal cellulitis secondary to Candida orthopsilosis in a diabetes mellitus patient – A rare case report and review of literature. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 18];2:546-8. Available from: https://www.ijoreports.in/text.asp?2022/2/2/546/342952

The article conforms to the Tenets of Declaration of Helsinki, and written informed consent for publication (including the images) has been obtained from the parents of the patient. Preseptal cellulitis commonly results from a bacterial infection that occurs from an extension of sinusitis, trauma, insect bite, or infected adjacent structures.^[1] Immunocompromised individuals like diabetics are prone to rhino sino-orbital fungal infections such as mucormycosis and aspergillosis. These fungal infections have shown a steady upward trend mirroring the increased number of immunocompromised patients with increased life expectancies.^[2] Preseptal cellulitis of fungal origin remains a rare entity, and due to exogenous inoculation of non-albicans candida species, it has not been reported previously.^[3–7]

Case Report

A 32-year-old male presented with rapidly progressing swelling over the left side of the face and eyelids with an inability to open the left eye for 5 days. There was no antecedent history of trauma, insect bite, or foreign body entry. There was no history of any fever, nasal discharge, or dental complaints. The patient was a known case of Type 2 diabetes mellitus that affected him in his childhood and was on irregular oral hypoglycemic agents (OHA). There was no history of COVID-19 infection among his family members and no history of contact with COVID-19 patients. His general and systemic examination was within the normal limit. On ophthalmic examination, the left upper and lower eyelids showed tense preseptal diffuse swelling associated with induration and blackish eschar extending onto the left cheek [Figure 1]a. The visual acuity assessment, intraocular pressure, and further anterior and posterior segment examination were not possible owing to tense preseptal edema and inability to retract the eyelids even with a Desmarres retractor. Routine blood investigations were unremarkable, but the random blood sugar (RBS = 542 mg/dL), fasting blood sugar (FBS = 386 mg/dL), and Hb1Ac (9.6) values were highly elevated with the presence of ketone bodies in urine. Diagnostic nasal endoscopy and oropharyngeal examination yielded no evidence of a nidus of infection. Contrast-enhanced computed tomography (CECT) of the orbits and paranasal sinuses revealed soft tissue thickening and attenuation involving the left temporalis muscle extending into the left preseptal space with an abscess 1.5 × 1.0 × 0.5 cm along the inferolateral aspect [Figure 2]. There was no evidence of any orbital extension. Based on the initial clinical presentation and radiological evidence, the diagnosis of preseptal cellulitis was established. The patient was started on treatment with intravenous broad-spectrum antibiotics empirically with intravenous crystalline penicillin q6H and ceftriaxone q12H to cover both gram-positive and gram-negative organisms and with metronidazole q8H, supplemented by aggressive glycemic control with insulin. Debridement of the eschar was deferred in view of the uncontrolled blood sugar and presence of features of diabetic ketoacidosis (DKA). Observing no improvement in the clinical outcome, debridement of the eschar and drainage of the preseptal abscess were performed after 2 days of admission. The debrided necrotic tissue was sent for microbiological assessment. Fungal culture of the necrotic tissue bits on Sabouraud dextrose agar (SDA) yielded yeast, which was identified as Candida orthopsilosis species, and it showed sensitivity toward posaconazole, amphotericin B, and fluconazole. The patient was immediately switched to intravenous posaconazole 300 mg twice daily (BID) on day 1 followed by 300 mg IV daily dosage. Two days later, following the initiation of antifungal drugs, there was a dramatic clinical improvement. The tense preseptal edema reduced, and we were able to retract the eyes to examine further ocular structures. The anterior and posterior segments of the left eye were found to be within normal limits. The pupillary reactions of the left eye were brisk. The patient was discharged 10 days later on oral fluconazole 150 mg BID for 2 weeks. On subsequent follow-up, the patient had developed cicatricial ectropion of the upper eyelid, for which a paramedian tarsorrhaphy was done to prevent exposure keratitis. The patient underwent ectropion [Figure 1]b release with full-thickness skin grafting and upper lid reconstruction after 6 weeks [Figure 3]a and [Figure 3]b. We have been following the patient for the last 6 months from the reconstructive surgery, and the patient is able to open and close eyelids and has not developed any further complications.

Figure 1: Clinical photograph showing (a) necrotizing preseptal cellulitis with eschar of the left eye extending to the cheek at presentation. (b) Patient developed ectropion of the upper eyelid at 6 weeks following initial debridement

Click here to view

Figure 2: CECT scan image showing left preseptal cellulitis with abscess. CECT = contrast-enhanced computed tomography

Click here to view

Figure 3: (a and b) Postoperative clinical photographs showing full-thickness skin graft in the left upper eyelid

Click here to view

Discussion

Preseptal cellulitis caused by a bacterial infection typically has a benign clinical course when appropriate antibiotic treatment is given. In some cases, it may lead to complications such as lid abscess and orbital cellulitis. Necrosis of the eyelid is uncommon due to the excellent vascularity of the lids. However, in fungal cellulitis, intravascular dissemination of fungal species can produce a deep-seated infectious vasculitis, thrombosis, and infarction leading to gangrene.^[8] Candidal facial cellulitis is a non-hematogenous infection caused by candida species, most commonly Candida albicans, and only a few cases have been documented worldwide. Codere and co-authors reported the first case of facial cellulitis caused by C. albicans.^[4] The patient presented with bilateral dacryocystitis following midfacial trauma, which was treated with bilateral dacryocystorhinostomy along with amphotericin B therapy. Other cases of facial cellulitis caused by C. albicans in uncontrolled diabetic patients were associated with a nidus of infection in the oral cavity or maxillary sinus.^[6],[7] Invasive disease caused by non-albicans candida can occur without prior colonization and is frequently transmitted nosocomially via contaminated external sources. In our patient, we did not find any evidence of a local or systemic nidus of infection.

C. orthopsilosis is a non-albicans candida species, which rarely causes human infection. Only a few cases of C. orthopsilosis-related infections like septic arthritis have been reported in the literature, and there are no reports on the infection causing preseptal or facial cellulitis.^[9] These organisms are susceptible to most of the available antifungal drugs, including natamycin, amphotericin B, and posaconazole. None of the previously reported cases had an eschar formation requiring debridement or had any evidence of vision-threatening complications or sequelae, like corneal exposure due to cicatricial ectropion, which was an essential aspect in the management of this case.

Conclusion

Fungal etiology must be considered as a possible cause of preseptal and facial cellulitis that progresses to tissue necrosis with eschar formation in the setting of uncontrolled diabetes mellitus, which is unresponsive to broad-spectrum antibiotics even in the absence of a local nidus of infection in the paranasal sinuses or oral cavity. Early debridement of eschar and lid reconstruction for ectropion may be required to prevent vision-threatening complications due to exposure keratitis.

Patient consent

Written informed consent was obtained from the patient for publication of his data and images in the journal.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Stevens DL, Bisno AL, Chambers HF, Dellinger EP, Goldstein EJ, Gorbach SL, et al. Practice guidelines for the diagnosis and management of skin and soft-tissue infections. Clin Infect Dis 2005;41:1373-406.
2.	Ranjith K, Sontam B, Sharma S, Joseph J, Chathoth KN, Sama KC, et al. Candida species from eye infections: Drug susceptibility, virulence factors, and molecular characterization. Invest Ophthalmol Vis Sci 2017;58:4201-9.
3.	Kwak OS, Kang MI, Kim JB, Kim MW, Kim YK. A rare case of facial Candida albicans cellulitis in an uncontrolled diabetic patient. Mycoses 2009;52:379-81.
4.	Codere F, Anderson R. Bilateral candida albicans dacryocystitis with facial cellulitis. Can J Ophthalmol 1982;17:176-7.
5.	Obi E, Roy A, Bates V, Sandy C. Bilateral chronic fungal dacryocystitis caused by candida dubliniensis in a neutropenic patient. J Clin Pathol 2006;59:1194-5.
6.	Chen HM, Shih CC, Yen KL, Wang SM, Kuo YS, Kuo MY, et al. Facial candida albicans cellulitis occurring in a patient with oral submucous fibrosis and unknown diabetes mellitus after local corticosteroid injection treatment. J Oral Pathol Med 2004;33:243-5.
7.	Peker E, Zor F, Toprak ME, Bariş E. Facial Candidal abscess in a patient with unknown type 2 diabetes mellitus. J Maxillofac Oral Surg 2015;14:995-8.
8.	Jun P, Russell M, El-Sayed I, Dillon W, Glastonbury C. Invasive facial fungal infections: Orofacial soft-tissue infiltration in immunocompromised patients. Radiol Case Rep 2013;8:813.
9.	Heslop OD, De Ceulaer K, Rainford L, Nicholson AM. A case of candida orthopsilosis associated septic arthritis in a patient with systemic lupus erythematosus (SLE). Med Mycol Case Rep 2015;7:1-3.