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CASE REPORT
Three Hundred Sixty-Degree Fuchs Superficial Marginal
Keratitis Managed With Annular Lamellar Keratoplasty
Albert Y. Cheung, MD,*† Enrica Sarnicola, MD,*‡ Khaliq H. Kurji, MD,*†
Brad M. Genereux, OD,*† and Edward J. Holland, MD*†
Purpose: To report a case of extensive Fuchs superficial marginal
keratitis managed with annular lamellar keratoplasty.
Methods: Interventional case report.
Results: A 72-year-old man presented with 20/80 best-corrected visual
acuity in his left eye and demonstrated 360-degree peripheral deep
immune stromal keratitis and pseudopterygia with peripheral stromal
thinning. During superficial keratectomy with amniotic membrane
transplantation, the thin cornea was perforated while excising pseudopterygia in the superonasal quadrant. Surgery was aborted. Anterior
segment optical coherence tomography demonstrated a severely thinned
cornea (240 mm nasally, 360 mm temporally) with overlying pseudopterygia peripherally. After allowing 3 months for the cornea to heal, the
decision was made to perform lamellar annular (or “donut”) keratoplasty. The patient had an unremarkable postoperative course, with
20/50 best-corrected visual acuity 10 months after keratoplasty.
Conclusions: We report an extensive case of Fuchs superficial
marginal keratitis treated with 360-degree annular lamellar keratoplasty. This technique provides tectonic support to decrease the
likelihood of future perforation while also improving vision by
modifying the ectatic cornea. Anterior segment optical coherence
tomography may be a helpful tool preoperatively to avoid severely
thinned areas (eg, during pseduopterygium removal) and to ensure
complete removal of the ectatic cornea.
Key Words: Fuchs superficial marginal keratitis, annular lamellar
keratoplasty, anterior segment optical coherence tomography, (AS-OCT)
(Cornea 2017;0:1–3)
F
uchs superficial marginal keratitis is an uncommon chronic
disorder characterized by episodic ocular inflammation
with marginal corneal infiltrates and progressive peripheral
stromal thinning. More common in young to middle-aged
Received for publication August 11, 2017; revision received September 5,
2017; accepted September 15, 2017.
From the *Cincinnati Eye Institute, Cincinnati, OH; †Department of Ophthalmology, University of Cincinnati, Cincinnati, OH; and ‡Department of
Medicine Surgery and Neuroscience, University of Siena, Siena, Italy.
E. J. Holland has consulted for Alcon Laboratories, Allergan, Bausch &
Lomb, Kala Pharmaceuticals, Mati Pharmaceuticals, Omeros, PRN, RPS,
Senju Pharmaceuticals, Shire, TearLab, and TearScience. The authors
have no funding or conflicts of interest to disclose.
Reprints: Edward J. Holland, MD, 580 South Loop Rd, Suite 200 Edgewood,
KY 41017 (e-mail: [email protected]).
Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.
Cornea Volume 0, Number 0, Month 2017
adults, centripetal marginal keratitis is often demarcated
from the central cornea by a sinuous gray line.1 The
process does not start from all parts of the margin at the
same time, does not advance uniformly, and does not reach
the central cornea. Pseudopterygium may develop in areas
of recurrent keratitis. Both traumatic and spontaneous
perforation has been described with this entity.1 We report
a unique, severe case with 360-degree involvement that
developed traumatic perforation at the time of superficial
keratectomy, ultimately treated with 360-degree annular
lamellar keratoplasty.
CASE REPORT
A 72-year-old man was referred to a cornea subspecialty
practice for worsening, blurry vision in his left eye over the last 10 to
15 years. His ocular history included a remote history of recurrent
episodes of redness and foreign body sensation in the left eye. He also
had undergone cataract extraction in the right eye 5 months before.
There was no history of eye trauma or contact lens wear. The medical
history was significant for hypertension, hypercholesterolemia, and
a previous cerebral vascular accident.
On initial examination, his best-corrected visual acuity was
20/20 in the right eye with 20.50 sphere and 20/80 in the left eye
with 23.25 + 1.25 · 010. Slit-lamp examination revealed mild
Meibomian gland dysfunction bilaterally. There was 360-degree
peripheral deep immune stromal keratitis and pseudopterygium
(Fig. 1A) with stromal thinning in the left eye. There was a 3+
nuclear sclerotic cataract and areas of peripheral late staining in the
left eye. Anterior segment examination was unremarkable in the right
eye with a normal cornea and well-positioned intraocular lens.
Corneal sensation was intact bilaterally. Posterior examination was
normal bilaterally.
With extensive pseudopterygia and potential limbal stem cell
deficiency, the decision was made to perform superficial keratectomy
with amniotic membrane transplantation. During superficial keratectomy,
the thin cornea was perforated while excising pseudopterygia in the
superonasal quadrant. Two 10-0 nylon sutures were placed to reform the
eye, and the rest of the pseudopterygia were repositioned with Tisseel
fibrin glue.
A diagnosis of extensive Fuchs superficial marginal keratitis was
made in the setting of peripheral thinning and pseudopterygia in areas of
previous keratitis. Anterior segment optical coherence tomography (ASOCT) demonstrated a severely thinned 360-degree peripheral cornea
(240 mm nasally, 360 mm temporally) with overlying pseudopterygia
(Fig. 2). The central cornea appeared uninvolved with increased thickness
(700 mm). Previous topography noted steep keratometry readings (53.32
and 47.07 D, astigmatism 6.25 D) with irregular astigmatism. After
allowing 3 months for the cornea to heal, the decision was made to
perform lamellar annular (or “donut”) keratoplasty (Fig. 3). This was
performed by partial thickness hand trephination of the host
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Copyright 2017 Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
1
Cornea Volume 0, Number 0, Month 2017
Cheung et al
FIGURE 1. Preoperative slit-lamp
photograph of the left eye demonstrating 360-degree peripheral keratitis
and pseudopterygia, yet the central
cornea is spared (A). Postoperative
slit-lamp photograph 10 months after
360-degree annular (or “donut”)
lamellar keratoplasty demonstrating
a clear annular graft with mild superior
peripheral vascularization and no
thinning (B).
FIGURE 2. AS-OCT demonstrating a severely thinned
360-degree peripheral/midperipheral cornea (measured with
calipers) with overlying pseudopterygia.
Progressive stromal thinning is a risk factor for traumatic and spontaneous perforation.3,4 It may be difficult to
determine the degree of thinning secondary to accompanying
pseudopterygium. Care must be taken during surgical
intervention because perforation may accidentally occur, as
in our case.4 Recurrence of the disease has been noted to occur
even after lamellar keratoplasty, although this may be
peripheral to the borders of the graft.5
AS-OCT may be an invaluable preoperative tool as it
will allow differentiation of the pseudopterygia tissue from the
underlying thinned corneal tissue. Although topography may
measure the cumulative thickness encompassing both
pseudopterygia and corneal tissue, AS-OCT will allow
differentiation based on cross-sectional appearance. Ellis
estimated corneal thickness in 4 eyes with Fuchs superficial
marginal keratitis to be 20% to 25% of normal corneal
thickness.6 AS-OCT noted the thinnest areas in our case
nasally to be 34% of central corneal thickness although
AS-OCT cuts were not taken through the superonasal
quadrant, which was likely the thinnest area (and the site of
the perforation). Interestingly, the AS-OCT images demonstrated thinning to be greatest in the mid-periphery. This
ectatic nature was described in multiple cases by Gifford.1
Recommended surgical treatment may entail combining
superficial keratectomy with a conjunctival autograft or
amniotic membrane transplantation to try to retard recurrent
pseudopterygia formation through the same mechanism that
prevents recurrence after excision of traditional pterygia.7,8
Kotecha and Raber7 noted that this treatment seemed to
suppress the flare-ups of marginal keratitis. For areas of
previous or impending perforation, an annular corneoscleral
lamellar patch graft can provide tectonic support.4 Given the
role the conjunctiva may play in inflammation, a conjunctival
autograft in conjunction with a lamellar patch graft has also
been recommended.5
A component of decreased vision in our case was likely
secondary to corneal steepening and astigmatism from
progressive thinning. Superficial keratectomy with amniotic
membrane transplantation was attempted to control inflammation
and prevent progression of thinning.8 Once the degree of thinning
was noted after perforation, we performed 360-degree annular
(or “donut”) lamellar keratoplasty to provide tectonic support.
This was a novel way to treat such extensive disease while
also sparing the central, uninvolved cornea. Compared with
2
Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.
cornea with 11- and 5.5-mm trephines. Lamellar dissection was
performed for the intervening cornea between the 2 trephinations
(preserving the central 5.5 mm of the host cornea). An 11-mm
trephine was then used to create the donor corneal button, and the
5.5-mm trephine was used to punch a central button out of this
11-mm button to create an annular graft. After the donor
Descemet and endothelium were removed, the “donut” button
was sutured into place with 10-0 nylon sutures. Postoperatively,
the patient was placed on topical difluprednate 0.05% 4 times
daily (qid), lifitegrast 5% twice daily (bid), levofloxacin 0.5% qid,
and cyclopentolate 1% 3 times daily (tid) (to deepen the anterior
chamber). The patient has had an unremarkable postoperative
course, with 20/50 best-corrected visual acuity (20.50 + 1.00 ·
090) 10 months after keratoplasty (Fig. 1B).
DISCUSSION
Fuchs superficial marginal keratitis was described in the
late 19th century with characteristic findings of episodic bouts
of inflammation leading to progressive marginal infiltrates and
peripheral thinning in an irregular, nonuniform manner with or
without pseudopterygia.2 Although our patient was older than
the more common demographic (young to middle-aged
adults), it is possible that his disease had quieted or burnt
out as he had a history of recurrent unilateral episodes of
redness and foreign body sensation. Although extensive
marginal infiltrates have been described,1 the 360-degree
nature of the pseudopterygia and thinning in our patient make
this case unique.
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Copyright 2017 Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
Cornea Volume 0, Number 0, Month 2017
Management of Fuchs Superficial Marginal Keratitis
FIGURE 3. Intraoperative photographs demonstrating 360-degree
annular (or “donut”) lamellar
keratoplasty. Preoperatively, the
cornea demonstrated peripheral
pseudopterygia but a clear center
(A). The host cornea was hand-trephinated partial thickness with 5.5and 11-mm trephines (B and C).
Lamellar dissection was performed
for the intervening cornea between
the 2 trephinations (preserving the
central 5.5 mm of the host cornea)
with Vannas scissors and a crescent
blade (D). An 11-mm trephine was
then used to create the donor corneal button, and the 5.5-mm trephine was used to punch a central
button out of this 11-mm button,
creating an annular graft. The donor
Descemet and endothelium were
removed, and the graft was then
thinned manually with Vannas scissors (E). The “donut” button was
finally sutured into place with 10-0
nylon sutures (F).
penetrating keratoplasty, lamellar keratoplasty has the benefits of
decreasing the chance of rejection, faster visual recovery,
structural benefits in the setting of trauma, and a decreased
chance of intraoperative open-sky complications.
We report an extensive case of Fuchs superficial
marginal keratitis treated with 360-degree annular lamellar
keratoplasty. This technique provides tectonic support to
decrease the likelihood of future perforation while also
improving vision by modifying the ectatic cornea. Ophthalmologists should consider a diagnosis of Fuchs superficial
marginal keratitis even when 360-degree peripheral involvement is present. AS-OCT may be a helpful tool preoperatively to help avoid severely thinned areas (eg, during
pseudopterygia removal) and to ensure complete removal
of the ectatic cornea. AS-OCT imaging of other cases will
help to further characterize this condition.
Copyright © 2017 Wolters Kluwer Health, Inc. All rights reserved.
REFERENCES
1. Gifford SR. Marginal dystrophy of cornea: furrow keratitis. Am J
Ophthalmol. 1925;8:16–23.
2. Fuchs E. Lehrbuch der Augenheilkunde. Leipzig and Wien: F. Deuticke;
1889.
3. Bierly JR, Dunn JP, Dawson CR, et al. Fuchs’ superficial marginal
keratitis. Am J Ophthalmol. 1992;113:541–545.
4. Goldberg MA, Lubniewski AJ, Williams JM, et al. Cystic hydrops and
spontaneous perforation in Fuchs’ superficial marginal keratitis. Am J
Ophthalmol. 1996;121:91–93.
5. Brilakis HS, Nordlund ML, Holland EJ. Recurrence of Fuchs marginal
keratitis within a lamellar graft. Cornea. 2004;23:639–640.
6. Ellis OH. Superficial marginal keratitis. Am J Ophthalmol. 1939;22:
161–168.
7. Kotecha A, Raber IM. Superficial keratectomy and conjunctival autograft
for Fuchs’ superficial marginal keratitis. Cornea. 2001;20:214–216.
8. Tseng SCG, Prabhaswat O, Lee SH. Amniotic membrane transplantation
for conjunctival surface reconstruction. Am J Ophthalmol. 1997;124:
765–774.
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Copyright 2017 Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.
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