|Year : 2021 | Volume
| Issue : 3 | Page : 184-188
Corneal perforation as a complication of fungal interface infectious keratitis after deep anterior lamellar keratoplasty
Wael Otaif1, Abdulaziz Ismail Al Somali2, Amar Almulhim3
1 Department of Ophthalmology, King Khalid University, Abha, Saudi Arabia
2 Department of Ophthalmology, College of Medicine, King Faisal University, Al Ahsa, Saudi Arabia
3 Department of Ophthalmology, King Faisal General Hospital, Al Ahsa, Saudi Arabia
|Date of Submission||03-Apr-2021|
|Date of Acceptance||27-Oct-2021|
|Date of Web Publication||31-Dec-2021|
Dr. Wael Otaif
Department of Ophthalmology, King Khalid University, P.O. Box 960, Abha 61421
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Deep anterior lamellar keratoplasty (DALK) is a surgical intervention for corneal diseases that do not affect the endothelium. The creation of an interface between the donor graft and recipient bed is a typical feature of DALK. Interface infectious keratitis (IIK) is an uncommon complication that originates at this point of contact. The onset of IIK following lamellar keratoplasty can cause delayed visual loss and subsequent endophthalmitis, with primarily fungal etiology (e.g., Candida spp.) and occasionally bacterial etiology. Infection of the lamellar interface may be attributed to contamination of the donor material or to precipitating factors such as loose sutures, persistent epithelial defects, and prolonged topical steroid use; fungal IIK is frequently resistant to medical treatment. Here, we describe the previously unreported occurrence of corneal perforation as a complication of fungal IIK after DALK. A 26-year-old otherwise healthy woman underwent uneventful DALK for advanced keratoconus in the left eye. She was discharged with instructions to apply topical prednisolone acetate and topical moxifloxacin. Culture of the donor corneoscleral rim showed growth of Candida glabrata, although the patient exhibited no clinical signs of infection. Approximately 3 months later, the patient exhibited mild blurring of vision in her left eye. Therefore, treatment was modified to topical amphotericin B and oral voriconazole. One week later, the patient developed multiple, sheath-like whitish creamy infiltrates, primarily in the lamellar interface; a positive Seidel test result indicated the presence of corneal perforation. As treatment for IIK, excisional penetrating keratoplasty (PK) was performed, followed by topical amphotericin B and topical prednisolone acetate treatment. During 12 months of follow-up after PK, the corneal graft was clear and there was no clinical evidence of recurrent keratitis. Prompt excisional PK prevented the progression of IIK to endophthalmitis in our patient. Early intervention with excisional PK should be considered when a diagnosis of fungal IIK is suspected in a patient with a positive donor rim culture, and in whom the condition does not respond to medical treatment. This early intervention is essential to prevent delayed treatment, which could result in corneal perforation and endophthalmitis, with ultimately poor visual outcomes.
Keywords: Case report, corneal perforation, deep anterior lamellar keratoplasty, fungal keratitis, interface infectious keratitis, lamellar keratoplasty
|How to cite this article:|
Otaif W, Al Somali AI, Almulhim A. Corneal perforation as a complication of fungal interface infectious keratitis after deep anterior lamellar keratoplasty. Middle East Afr J Ophthalmol 2021;28:184-8
|How to cite this URL:|
Otaif W, Al Somali AI, Almulhim A. Corneal perforation as a complication of fungal interface infectious keratitis after deep anterior lamellar keratoplasty. Middle East Afr J Ophthalmol [serial online] 2021 [cited 2022 Jul 4];28:184-8. Available from: http://www.meajo.org/text.asp?2021/28/3/184/334622
| Introduction|| |
Deep anterior lamellar keratoplasty (DALK) is a surgical intervention for corneal diseases that do not affect the endothelium; these diseases may include keratoconus, partial-thickness corneal scars, and corneal stromal dystrophies. Compared with penetrating keratoplasty (PK), DALK involves a lower risk of endothelial rejection and shorter duration of postoperative steroid medication; it also avoids the need for open-sky surgery and its related complications while enabling earlier suture removal.
The creation of a surface where the donor graft contacts the recipient bed (i.e. the graft–host interface) is a typical feature of DALK. Interface infectious keratitis (IIK) is an uncommon complication that originates at this point of contact. The development of IIK following lamellar keratoplasty may compromise corneal graft clarity, which can lead to visual loss and subsequent progression to endophthalmitis. It has been reported that IIK develops between 2 and 120 days after DALK; the infection is often fungal (most frequently involving Candida spp.) but occasionally demonstrates bacterial etiology. Infection of the lamellar interface may be attributed to contamination of the donor material or to precipitating factors such as loose sutures, persistent epithelial defects, and prolonged topical steroid use. Here, we describe a patient who developed fungal IIK after DALK, which led to corneal perforation. To the best of our knowledge, this is the first report of corneal perforation as a possible complication of fungal IIK after DALK.
| Case Report|| |
A 26-year-old otherwise healthy woman underwent uneventful DALK for advanced keratoconus in the left eye. The preoperative best-corrected visual acuity (BCVA) in the patient's left eye was 20/100; she had no reported history of ocular infection or injury. The donor corneal tissue was obtained from a 61-year-old man who had no history of candidemia or compromised immunity. The cause of donor death was cardiac arrest; the death to preservation interval was 8 h. The donor tissue had been stored in Optisol-GS storage medium (Bausch and Lomb Incorporated, Bridgewater, NJ, USA); additionally, the corneoscleral rim had been sent for routine fungal and bacterial cultures. After DALK, the patient was discharged with instructions to apply topical prednisolone acetate 1% and topical moxifloxacin 0.5% four times per day; the early postoperative period was uneventful.
On postoperative day 7, BCVA in the patient's left eye was 20/40; slit-lamp examination showed a clear graft and quiet anterior chamber. However, on postoperative day 10, the culture of the donor corneoscleral rim showed growth of Candida glabrata, which was sensitive to voriconazole and amphotericin B. Three days later (postoperative day 13), clinical examination of the patient revealed no signs of inflammation and demonstrated a clear graft. Because no clinically active infection was present, no antifungal therapy was initiated at this stage. The patient was followed at 1-week intervals for 1 month, during which she remained asymptomatic with a BCVA of 20/40 in the left eye; no signs of inflammation were noted. However, 1 month later (postoperative day 73), the patient experienced ocular discomfort and irritation. She presented to a local ophthalmologist, who observed a whitish corneal opacity in an otherwise quiet eye. A diagnosis of epithelial ingrowth was made; the patient's treatment remained topical prednisolone acetate 1% and topical moxifloxacin 0.5%.
Two weeks later (postoperative day 87), the patient returned to our hospital with the complaint of mild blurring of vision. Her BCVA was 20/60 in the left eye, while intraocular pressure in the left eye was 16 mmHg; slit-lamp examination revealed multiple, sheath-like whitish creamy infiltrate, primarily located in the lamellar interface. The epithelium was intact, moderate stromal thinning was present, no loose or broken sutures were observed, the anterior chamber was quiet, and the fundus examination results were unremarkable; thus, a diagnosis of IIK was made. Based on the growth of C. glabrata in the donor rim culture, treatment was immediately initiated with topical 0.15% amphotericin B four times per day, in addition to 200 mg oral voriconazole two times per day; the administration of topical prednisolone acetate was tapered and the patient was monitored daily.
One week later (postoperative day 94), the patient exhibited redness, pain, and further vision reduction. Her BCVA was 20/125 in the left eye; slit-lamp examination showed mild conjunctival injection, while infiltration was considerably worse, such that it involved the paracentral and superior interface at the 2 o'clock position [Figure 1]a. A positive Seidel test result indicated corneal perforation at the site of infiltration [Figure 1]b; a 2 + cellular response was noted in the anterior chamber. Fundus examination and B-scan analysis revealed no indications of vitritis. Anterior segment optical coherence tomography confirmed that infiltrates were present at the lamellar interface [Figure 2]. Given the worsening of infiltration with corneal perforation despite medical treatment, as well as the possibility of progression to fungal endophthalmitis, the patient was immediately admitted for excisional PK. In this procedure, an 8.5-mm donor graft was substituted for the infected area of the host cornea. Considerable irrigation of the anterior chamber was performed with intracameral injection of 5 mg/0.1 ml amphotericin B. Both donor graft and lamellar host cornea were sent for bacterial and fungal cultures; the results were positive for C. glabrata. Postoperative treatment included topical amphotericin B 0.15% and topical prednisolone acetate 1%.
|Figure 1: Slit-lamp photograph of the left eye. (a) Multiple, sheath-like whitish creamy infiltrates are present; wound dehiscence is present at the 2 o'clock position (Arrow). (b) Positive Seidel test results are shown with fluorescein staining|
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|Figure 2: Anterior segment optical coherence tomography of the left eye. Infiltrates are present in the lamellar interface|
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At 3 months after PK, the patient's BCVA had reached 20/40 in the left eye. During the remainder of the 18-month follow-up period after PK, it remained at this level; the corneal graft was clear, and there was no clinical evidence of recurrent keratitis. A summary of the patient's clinical course and relevant treatment is shown in [Figure 3].
|Figure 3: Timeline depicting the patient's clinical course and relevant treatment. Abbreviations: BCVA: Best-corrected visual acuity, DALK: Deep anterior lamellar keratoplasty, IIK: Interface infectious keratitis, PK: Penetrating keratoplasty, POD: Postoperative day|
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| Discussion and Conclusions|| |
Fungal IIK is an uncommon – but very serious – complication after DALK. Diagnosis and treatment of fungal IIK are challenging because of factors such as the slow nature of the fungal infection, an early asymptomatic clinical course with similarity to epithelial ingrowth and graft rejection, the use of topical steroids, and a sequestered location within the lamellar interface that can hamper the penetration of topical treatments. Treatment strategies for IIK are not yet well established. Conventional methods for diagnosis and treatment of microbial keratitis are unsuitable because the sequestered location of the infiltrate limits scraping and culture. Consequently, based on clinical presentation and the results of corneoscleral donor rim culture testing, topical and systemic broad-spectrum antifungal treatments are often initiated. However, topical and systemic therapies are reportedly ineffective for preventing the progression of active infection in most affected patients.
A previous study found that fungi were present in donor corneoscleral rim cultures in 2.1% of patients who had undergone keratoplasty; however, only 5.6% of patients with fungi in corneoscleral rim cultures subsequently developed an active clinical infection, suggesting that positive corneoscleral rim culture results may not be accurate predictors of clinical infection. Therefore, it remains unclear whether antifungal treatment should be administered immediately after determination of a positive corneoscleral rim culture result; alternatively, affected patients could be monitored at regular intervals and receive treatment only after observation of clinical symptoms and signs of fungal infection.,,,,
In previous case reports that involved the use of confocal scanning, this noninvasive imaging modality was shown to aid in the diagnosis of fungal IIK because it enabled consistent identification of hyper-reflective granular deposits in the interface in all scanned patients with fungal IIK after DALK; it also allowed the identification of hyphal elements in some scanned patients. Accordingly, confocal scanning may reduce the reliance on culture testing and eliminate the corresponding delay prior to intervention. Further research regarding the sensitivity and specificity of confocal scanning is needed to verify whether it is a suitable method for diagnosis of fungal IIK.,,,
The benefit of adding oral voriconazole to topical antifungal therapy in patients with severe fungal keratitis has been considered. One randomized controlled trial found no additional therapeutic benefit and a significantly higher incidence of adverse events when oral voriconazole was added to topical antifungal treatment. However, that study was limited to filamentous fungi; thus, its results are not necessarily applicable to fungal IIK after lamellar keratoplasty, in which the most frequently reported organisms are Candida spp.
Previously, irrigation of the interface with antifungal agents was reported for four patients with post-DALK fungal IIK, which was expected to preserve the graft and avoid the need for PK. However, irrigation resulted in the rupture of Descemet's membrane in two of these patients, who then required PK;, one patient exhibited posterior perforation with a double anterior chamber that resolved spontaneously without further intervention, whereas the other patient exhibited worsening of infection after irrigation and thus underwent excisional PK.
Notably, graft replacement and interface irrigation with antifungal therapy is another surgical management approach that has been attempted in three patients with post-DALK fungal IIK; it was expected to prevent the need for full-thickness transplant surgery. However, this approach was successful only in one patient for whom the intervention was performed early in the course of infection; excisional PK was needed in the other two patients, both of whom exhibited worsening and recurrence of interface opacity., These findings suggest that early excisional PK constitutes a safe and successful method for treatment of fungal IIK after DALK.
While it has been reported that corneal perforation and endophthalmitis can occur in patients with fungal IIK after Descemet stripping automated endothelial keratoplasty, there have been no prior reports of DALK-related fungal IIK associated with corneal perforation or endophthalmitis. The patient described in this report exhibited an inadequate response to topical and systemic treatment, as well as the potential for corneal perforation. Prompt excisional PK with intracameral injection of an antifungal agent was a successful intervention for this patient, which prevented progression to endophthalmitis. Based on our findings and the results of previous reports, early intervention with excisional PK should be considered when a diagnosis of fungal IIK is suspected in a patient with a positive donor rim culture, and in whom the condition does not respond to medical treatment. This consideration is essential to prevent delayed treatment, which could eventually result in corneal perforation and endophthalmitis, with ultimately poor visual outcomes.
In conclusion, fungal IIK is a vision-threatening complication of lamellar keratoplasty that is frequently resistant to medical treatment; affected patients often require therapeutic excisional PK. A high degree of suspicion is needed for diagnosis because this condition often presents with mild inflammation, following a considerable delay after surgery. In addition, routine donor rim culture at the time of corneal transplantation is important for early diagnosis and treatment. It is crucial to closely monitor any small whitish interface corneal opacity that may occur a few days or weeks after any type of lamellar keratoplasty, and to consider this as an indication of infection, particularly when the donor rim culture results are positive. Because of the risks of perforation and endophthalmitis, early intervention with excisional PK should be considered when there are clear signs of infection despite provision of maximum medical treatment.
Consent for publication
Written informed consent was obtained from the patient for the publication of this report and associated images.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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