Ophthal Plast Reconstr Surg, Vol. 30, No.
5, 2014 Case Reports
current case is the first reported case of bilateral, simultaneous
development of pyogenic granuloma after perforated punctal
plug insertion and a description to its management.
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1. Konuk O, Urgancioglu B, Unal M. Long-term success rate of perfo-
rated punctal plugs in the management of acquired punctal stenosis.
Ophthal Plast Reconstr Surg 2008;24:399–402.
2. Offutt WN 4th, Cowen DE. Stenotic puncta: microsurgical puncto-
plasty. Ophthal Plast Reconstr Surg 1993;9:201–5.
3. Edelstein J, Reiss G. The wedge punctoplasty for treatment of punc-
tal stenosis. Ophthalmic Surg 1992;23:818–21.
4. Soiberman U, Kakizaki H, Selva D, et al. Punctal stenosis: defini-
tion, diagnosis, and treatment. Clin Ophthalmol 2012;6:1011–8.
5. Silverberg SG, ed. Principles and Practice of Surgical Pathology
and Cytopathology. 3rd ed. New York, NY: Churchill Livingstone,
1997:1443–4.
6. Akova YA, Demirhan B, Cakmakçi S, et al. Pyogenic granuloma:
a rare complication of silicone punctal plugs. Ophthalmic Surgery
Lasers Imaging 1999;30:584–5.
7. Kim BM, Osmanovic SS, Edward DP. Pyogenic granulomas after
silicone punctal plugs: a clinical and histopathologic study. Am J
Ophthalmol 2005;139:678–84.
8. Musadiq M, Mukherji S, Sandramouli S. Pyogenic granuloma
following silicone punctal plugs: report of two cases. Orbit
2005;24:149–51.
9. Kaido M, Ishida R, Dogru M, et al. Comparison of retention rates
and complications of 2 different types of silicon lacrimal punc-
tal plugs in the treatment of dry eye disease. Am J Ophthalmol
2013;155:648–653, 653.e1.
10. Bourkiza R, Lee V. A review of the complications of lacrimal occlu-
sion with punctal and canalicular plugs. Orbit 2012;31:86–93.
Keloidal Scarring in Hard Palate
Grafts
Meredith S. Baker, M.D.*, Angela R. McAllister, M.D.,
M.P.H.*, Nasreen A. Syed, M.D.*†, and
Richard C. Allen, M.D., Ph.D.*‡
Abstract: Hard palate grafts are a commonly accepted
treatment for lower eyelid retraction and are typically
associated with low complication rates. The authors present
a case of bilateral keloidal scarring within hard palate grafts
requiring surgical excision. A 72-year-old woman who had
undergone bilateral hard palate grafts to her lower eyelids for
lower eyelid retraction was referred for the evaluation of ocular
irritation. She was diagnosed with bilateral hypertrophy of
the grafts with secondary entropion. The grafts were excised;
the pathology was consistent with keloidal scarring. To the
authors’ knowledge, there is no previously reported case of
bilateral keloidal scarring in normal donor tissue, which
required excision of the hard palate grafts. It is important
for oculoplastic surgeons to be aware of this potential
complication when considering, discussing, and consenting
patients for eyelid reconstruction with hard palate grafting.
Departments of *Ophthalmology and Visual Sciences, †Pathology, and
‡Otolaryngology–Head and Neck Surgery, University of Iowa, Iowa City,
Iowa, U.S.A.
Accepted for publication July 2, 2013.
Presented at Fall ASOPRS Meeting, Chicago, IL, on November 15, 2012.
The authors have no financial or conflicts of interest to disclose.
Address correspondence and reprint requests to Richard C. Allen, M.D.,
Ph.D., Department of Ophthalmology and Visual Sciences, University
of Iowa Hospitals and Clinics, 200 Hawkins Drive, Iowa City, IA 52242.
E-mail: Richard-Allen@uiowa.edu
DOI: 10.1097/IOP.0b013e3182a5ba82
© 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e115
T he treatment of hard palate grafts frequently involves the
placement of spacers. Potential spacers include hard palate
grafts, free tarsal grafts, humane and porcine acellular dermal
matrix, fascia, porous polyethylene, sclera, auricular cartilage,
and nasal septum. Hard palate grafts are a commonly accepted
treatment for lower eyelid retraction. The benefits are they are
rigid, mucous membrane-lined, and autologous. In addition,
most studies proclaim low complication rates.1–6 Complications
do exist and can be divided into donor site complications and
recipient site complications.3,7 This study presents a case of
bilateral keloidal scarring localized to hard palate grafts and
requiring surgical excision.
RESULTS
A 72-year-old woman was initially presented for the evaluation
of ocular irritation, discharge, and tearing. She noted that her lower
eyelids had become “more swollen” over the past 6 months. Her ocular
history was significant for thyroid eye disease (TED). Approximately
15 years prior to presentation, she underwent bilateral hard palate
grafts to her lower eyelids for lower eyelid retraction secondary to
TED. Her medical examination was significant for lower eyelid entro-
pion, large nodular lesions on the inferior palpebral conjunctiva of OU
with mild mucoid discharge (Fig. 1), and corneal epitheliopathy. She
was diagnosed with bilateral hypertrophy of the hard palate grafts with
secondary entropion. She subsequently underwent bilateral excision of
the hard palate grafts with bilateral pillar tarsorrhaphies (Fig. 2). The
pathology demonstrated thick bundles of collagen arranged at oblique
angles consistent with keloidal scarring (Fig. 3). Keloidal scarring is
a descriptive term for describing thick collagenous fibers of hypertro-
phic scarring that does not extend beyond the original wound; keloid
is defined by extension beyond the original wound that did not occur
in this case.
DISCUSSION
Hard palate grafts are frequently chosen as the ideal
spacer in lower eyelid surgery.1–6 However, complications do
exist. Donor site morbidities include postoperative pain and
bleeding and, less commonly, oral candidiasis and oronasal
fistula formation.3,7 Complications related to the graft, such
as keratinization causing ocular irritation or graft contrac-
tion or failure requiring reoperation, are also fairly uncom-
mon in the literature.3,7 To the authors’ knowledge, there is
no previously reported case of bilateral keloidal scarring
in previously normal donor tissue, which required excision
of the hard palate grafts. It is important for oculoplastic
surgeons to be aware of this potential complication and to
consider this possible adverse effect when evaluating and
consenting patients for eyelid reconstruction with hard pal-
ate grafting.
REFERENCES
1. Kersten RC, Kulwin DR, Levartovsky S, et al. Management of low-
er-lid retraction with hard-palate mucosa grafting. Arch Ophthalmol
1990;108:1339–43.
2. Cohen M, Shore N. Eyelid reconstruction with hard palate grafts.
Ophthal Plast Reconstr Surg 1992;8:183–195.
3. Sullivan SA, Dailey RA. Graft contraction: a comparison of acel-
lular dermis versus hard palate mucosa in lower eyelid surgery.
Ophthal Plast Reconstr Surg 2003;19:14–24.
4. Swamy BN, Benger R, Taylor S. Cicatricial entropion repair with
hard palate mucous membrane graft: surgical technique and out-
comes. Clin Experiment Ophthalmol 2008;36:348–52.
5. Wearne MJ, Sandy C, Rose GE, et al. Autogenous hard palate
mucosa: the ideal lower eyelid spacer? Br J Ophthalmol 2001;85:
1183–7.
Case Reports Ophthal Plast Reconstr Surg, Vol. 30, No. 5, 2014

FIG. 1.  External photographs demonstrating lower eyelid edema (left) and bilateral large nodular lesions on the inferior palpebral
conjunctiva with eversion of the lower eyelids (right). Note: yellow discoloration from fluorescein staining. The nodular lesions did not
extend beyond the original hard palate grafts.

FIG. 2.  External photograph showing preoperative (left) and postoperative (right) photographs of the patient, with improvement in
lower eyelid edema and entropion after excision of hard palate grafts.

FIG. 3.  A, Histopathology demonstrating thick bundles of collagen arranged at oblique angles in the substantia propria consistent
with keloidal scarring (hematoxylin-eosin, ×20). B, Masson trichrome stain highlighting the collagen (blue) within the substantia nigra
consistent with keloidal scarring (×10).

6. Leibovitch I, Malhotra R, Selva D. Hard palate and free tarsal grafts
as posterior lamella substitutes in upper lid surgery. Ophthalmology
2006;113:489–96.
7. Kim JW, Kikkawa DO, Lemke BN. Donor site complications
of hard palate mucosal grafting. Ophthal Plast Reconstr Surg
1997;13:36–9.
associated with surgical morbidity were identified, and the
embryological origins of these cysts were reviewed.
T he most common orbital choristoma is a dermoid cyst that
accounts for about a half of diagnosed childhood orbital
masses.1 Rarer lesions include conjunctival cysts, ectopic lac-
rimal rests, and congenital cysts associated with the common
sheath of the superior rectus and levator palpebrae superioris.2

Congenital Respiratory Epithelial

Cysts of the Orbit: A Rare Cause of
Major Orbital Impairment
Eugene Tay, F.R.C.Ophth.*†,
Anita Chan Sook Yee, F.R.C.S(Edin).†‡,
Philip J. Luthert, F.R.C.P., F.R.C.Path., F.R.C.Ophth.‡,
and Geoffrey E. Rose, D.Sc., F.R.C.Ophth.*
Abstract: Congenital respiratory epithelial cysts of the orbit
are rare lesions with few reported cases. Extensive disease
may cause an orbital apex syndrome, resulting in significant
visual loss. Two such cases were described, the risk factors
*Adnexal and Orbital Service, Moorfields Eye Hospital, London,
United Kingdom; †Department of Ophthalmology, Singapore National Eye
Centre, Singapore, Singapore; and ‡Department of Pathology, Institute of
Ophthalmology, London, United Kingdom
Accepted for publication July 2, 2013.
GER has received funding from the National Institute of Health Research
(NIHR), the Biomedical Research Centre at Moorfields Eye Hospital
National Health Service (NHS) Foundation Trust and University College
London (UCL) Institute of Ophthalmology. The other authors have no
financial or conflicts of interest to disclose.
Address correspondence and reprint requests to Geoffrey E. Rose, D.Sc.,
F.R.C.Ophth., Adnexal and Orbital Service, Moorfields Eye Hospital, City
Road, London EC1V 2PD, United Kingdom. Geoff.Rose@moorfields.nhs.uk
DOI: 10.1097/IOP.0b013e3182a64f6b

e116 © 2014 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.