Ophthalmic Plast Reconstr Surg, Vol. 36, No.
3, 2020 Case Reports
lesion be completely separated from an anatomically normal
caruncle, and that histopathology demonstrates normal ca-
runcular features. All convincing reports of supernumerary
lacrimal caruncle involve the inferomedial palpebral conjunc-
tiva and are typically inferior to the punctum.2 Although the
lesions are typically solitary, 2 double reduplications have been
described..2,7 To the best of the authors’ knowledge, the English
literature does not contain any accounts of a supernumerary lac-
rimal caruncle diagnosed in a child, despite the congenital na-
ture of the lesion.1,2,7–9 The Japanese literature describes 1 lesion
consistent with a supernumerary lacrimal caruncle biopsied in
a 13-year-old male.12
Jakobiec et al. postulated that puberty plays an essen-
tial role in the symptomatology of supernumerary lacrimal
caruncle.2 Pilosebaceous units, which are found in the normal
caruncle, are involved in androgen processing and modulation
and are also under androgen control.13 The enlargement of pilo-
sebaceous units that occurs in response to the release of andro-
gens may be more likely to irritate the ocular surface compared
Downloaded from http://journals.lww.com/op-rs by BhDMf5ePHKbH4TTImqenVAPwFBsBoeDVRxnE4i0B2TSuqqTOmWR3YeEwRrc2fprC on 05/26/2020
with the prepubescent state. The role of puberty, and the cursory
examinations of the palpebral conjunctiva in asymptomatic pe-
diatric patients, may lead to the underdiagnosis of this condition
in younger children. The biannual eye examinations for the child
presented herein to evaluate for ocular manifestations of juve-
nile idiopathic arthritis likely contributed to his early diagnosis.
CONCLUSION
Congenital anomalies of the caruncle are rare. In the
English literature, supernumerary lacrimal caruncle has only
been previously described in adults despite the congenital na-
ture of the lesion. Supernumerary lacrimal caruncle is typically
diagnosed after puberty, in which the androgen-sensitive pilose-
baceous units enlarge and become more irritating to the ocular
surface.
REFERENCES
1. Duke-Elder S. Normal and abnormal development: congenital de-
formities. In: Duke-Elder S, ed. System of Ophthalmology. Vol 3.
St. Louis: CV Mosby, 1964:860–862.
2. Jakobiec FA, Lam H, Bhat P, et al. Non-syndromic supernumerary
caruncles causing ocular irritation after cataract surgery: a critical
review of caruncular dysgeneses. Am J Ophthalmol 2010;149:398–
404.e1–2.
3. Rennie IG, Parsons MA, Benson MT. Neurilemoma of the caruncle:
a clinicopathological report. Br J Ophthalmol 1991;75:749–751.
4. Shields CL, Shields JA. Tumors of the caruncle. Int Ophthalmol
Clin 1993;33:31–36.
5. Duke-Elder S, Cook C. Normal and abnormal development: em-
bryology. In: Duke-Elder S, ed. System of Ophthalmology. Vol. 3.
St. Louis: CV Mosby, 1963:234–235.
6. Whitnall SE. Eyelids. In: Whitnall SE, ed. The Anatomy of the
Human Orbit and Accessory Organs of Vision. 1st ed. London:
Henry Frowde and Hodder & Stoughton, 1921:183–187.
7. Friedman B. The supernumerary caruncle. Arch Ophthalmol
1941;25:664–668.
8. Mansour K, van Bijsterveld OP. Supernumerary caruncle: report of
a case. Ann Ophthalmol 1984;16:677–678.
9. Zamir E, Banin E, Chowers I, et al. Ectopic caruncle. Arch
Ophthalmol 1999;117:1446–1447.
10. Szestyńska-Serwatkowa A. [Case of additional lacrymal caruncle].
Klin Oczna 1975;45:269–271.
11. Reznikov Iu E. [Accessory lacrimal caruncle]. Oftalmol Zh
1987:252 [in Russian].
12. Shimoyama R, Uda S, Nemoto Y. [A case of supernumerary carun-
cle]. Nippon Ganka Gakkai Zasshi 2013;117:438–442.
13. Zouboulis CC, Degitz K. Androgen action on human skin – from
basic research to clinical significance. Exp Dermatol 2004;13(suppl
4):5–10.
© 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e73
Copyright © 2020 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
A Case of Reactivation of Herpes
Simplex Virus Corneal Endotheliitis
Following Periocular Botulinum
Toxin A Injection
Nibedita Das, D.O., D.N.B., F.I.C.O., M.R.C.S.Ed.*,
Joyeeta Das, M.B.B.S., D.N.B.†, and Samar Basak, M.D.,
F.R.C.S.*
Abstract: Botulinum toxin type-A is a standard treatment of
a variety of neurological disorders. Here, the authors report a
rare ocular adverse effect after periocular Botulinum Toxin-A
injection for hemifacial spasm. Three weeks after the injection,
facial spasm was relieved, but the patient had dimness of
vision OS. Examination revealed corneal edema, anterior-
chamber reaction, keratic precipitates, and elevated intraocular
pressure in OS suggesting reactivation of herpes simplex viral
endotheliitis with trabeculitis. The case was successfully treated
with oral acyclovir (400 mg) and topical fluorometholone 0.1%
for 6 weeks. Pre- and posttreatment anterior segment optical
coherence tomography clearly demonstrated the evidence of
keratic precipitates. To the best of the authors' knowledge, this
is the first report of herpetic reactivation in the form of corneal
endotheliitis with trabeculitis after Botulinum toxin-A injection.
The aim of reporting this case is to alert the clinician about the
possibility of such consequence of botulinum toxin injection.
CASE REPORT
Hemifacial spasm is characterized by chronic unilateral in-
voluntary and irregular tonic and clonic contractions of the mus-
cles supplied by the facial nerve. The United States Food and Drug
Administration approved botulinum toxin A (BOTOX, Allergan
Inc., Irvine, CA) for the management of strabismus, blepharo-
spasm, and related facial spasms in 1989. In view of growing evi-
dence supporting its efficacy, it has become a standard first line of
treatment for hemifacial spasm.1,2 Botulinum toxins are exotoxins
produced by Clostridium botulinum with 7 toxin serotypes types
A–G. Botulinum toxin A is preferred because of its longer duration
of action and ease of production. The toxin prevents release of ace-
tylcholine into the synaptic cleft and reduces muscular or glandular
activity. Most common documented reversible side effects of botu-
linum toxin injections for hemifacial spasm include erythema, ec-
chymosis of the region injected, lagophthalmos, dry eyes, drooping
of the mouth, eyelid edema, ptosis, and facial muscle weakness.3
Recently very few cases of reactivations of herpes sim-
plex virus keratitis (HSV) and herpes zoster ophthalmicus have
*Department of Cornea and External Disease, Disha Eye Hospitals,
88(63A) Ghoshpara road, Kolkata, India; and †Department of Orbit and
Oculoplasty, Disha Eye Hospitals, 88(63A) Ghoshpara road, Kolkata, India
Accepted for publication December 12, 2019.
The authors have no financial or conflicts of interest to disclose.
All authors have no financial interest related to manuscript, stock or
ownership of a business entity connected to any product described in the
above article, paid consulting for the company or competing companies, or
patent rights to a drug or piece of equipment.
All authors have no personal or family ownership or potential rights to
more than 1% of the company or competing company and whether they
have any interest in marketing any product, drug, instrument, or piece of
equipment discussed in the manuscript
Address correspondence and reprint requests to Nibedita Das, D.O.,
D.N.B., F.I.C.O., M.R.C.S.Ed., Department of Cornea and external disease,
Disha Eye Hospitals, 88(63A) Ghoshpara road, Kolkata, India 700120.
E-mail: dasnibedita@yahoo.co.in.
DOI: 10.1097/IOP.0000000000001587
 Case Reports Ophthalmic Plast Reconstr Surg, Vol. 36, No. 3, 2020

been reported by some authors after botulinum toxin A injec-
tion for various indications.4–7 Here, we are reporting a case of
this rare complication of botulinum toxin injection which was
manifest as viral corneal endotheliitis without any epithelial
and stromal recurrence. To the best of our knowledge, this is
the first report of herpetic reactivation in the form of corneal
endotheliitis with trabeculitis after Botulinum toxin A injection.
This case report adhered to the ethical principles outlined in
the Declaration of Helsinki as amended in 2013 and proper in-
formed consent was obtained from the patient for publication.
CASE PRESENTATION
A 44-year-old Indian lady presented to our clinic for se-
vere twitching on the left side of her face for the past one and
half years without any fruitful results. She does not have any
past history of trauma, systemic illness, or surgery in the head
and neck region. Her best spectacle corrected visual acuity was
20/20 in OD and 20/40 in OS using Snellen’s chart.
On examination, there was involuntary forceful closure
of her OS eyelid with mild deviation of her face towards the left
side which was also present when the patient was sleeping.
Anterior segment of the OD had normal findings; but the cornea
of the OS showed old epithelial and mid-stromal scar in the in-
ferior paracentral area. An MRI of the brain-orbit and other neu-
rological examinations were normal. A diagnosis of left sided
hemifacial spasm was made. Twenty-five units of Onabotulinum
toxin type A (BOTOX, Allergan Inc., Irvine, CA) were given
by an oculoplastic surgeon according to standard protocol.
Subcutaneous injection into the  pretarsal orbicularis oculi on
left upper and lower eyelid (2.5 units each in medial and lateral
angle of eyelid), and intramuscular injection into the  corruga-
tor, frontalis, and zygomaticus muscles (5 units each spots) were
done.
At the first week follow-up visit her clinical condition was
improved with complete resolution of facial spasms. On the third
week, she presented in the emergency clinic with blurring of
vision in the OS, associated with photophobia and redness. Best
spectacle corrected visual acuity in her OS was reduced to 20/80
by Snellen’s chart. On slit lamp examination there was perilim-
bal conjunctival congestion, an old fusiform stromal scar, mild
corneal edema, and medium keratic precipitates with 2+ flare in
the anterior chamber (Fig. 1A–C). Her intraocular pressure in
the OD was 18 mm Hg and in the OS 30 mm Hg. Clinical fea-
tures were suggestive of viral endotheliitis with trabeculitis. At
this point, on further enquiry, she told us that she had a redness
and dimness of vision in the same eye about 7 years ago. Her
local doctor diagnosed HSV keratitis which responded to acy-
clovir (3%) eye ointment. Aqueous humor tapping for viral pol-
ymerase chain reaction to detect HSV, varicella zoster virus, and
cytomegalovirus was done, and the test reports were negative.
Anterior segment Optical Coherence Tomography using RTvue
model RT100 CAM system (Optovue Inc, Fremont, CA) demon-
strates multiple hyper-reflective dome shaped protrusions from
the endothelium underlying area of stromal edema. On the basis
of history, clinical findings, and temporal events of treatment
(i.e., Botulinum toxin injection), the case was provisionally diag-
nosed as reactivation of herpes simplex endotheliitis possibly
due to periocular botulinum toxin injections. Other differential
diagnosis of the case can be acute anterior uveitis. But on slit
lamp findings, a previous corneal scar along with focal stromal
edema and associated keratic precipitates just behind edema
strongly suggest viral endothelitis. She also had increased intra-
ocular pressure indicating trabeculitis which is quiet common in
viral keratouveitis.
We treated her with oral Acyclovir 400 mg 5 times a day for
7 days and then continued twice daily for 10 weeks; along with

FIG. 1.  Slit lamp and anterior segment-OCT findings before treatment. A, Acute endotheliitis with anterior chamber reaction. B,
Keratic precipitates on endothelium. C, Anterior segment OCT showing keratic precipitates on the endothelium.

FIG. 2.  Slit lamp and anterior segment-OCT findings after treatment. A, Resolved endotheliitis, clear anterior chamber reaction. B, No
keratic precipitates present on endothelium, stromal scar. C, Stromal scar with absent keratic precipitates after treatment.

e74 © 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.

Copyright © 2020 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
 Ophthalmic Plast Reconstr Surg, Vol. 36, No. 3, 2020 Case Reports
timolol maleate (0.5%) 2 times and flurometholone (0.1%) eye
drop in weekly tapered doses starting at 4 times per day follow-
ing a standard treatment protocol.8 After 1 month, she improved
symptomatically with improvement of best spectacle corrected
visual acuity of 6/9 on Snellen’s Chart, corneal edema improved,
keratic precipitates resolved without any anterior chamber reac-
tion (Fig. 2A–C), and her intraocular pressure was normal. She did
not have any recurrence at the last follow up visit after 18 months.
DISCUSSION
Herpes simplex viral keratitis is an important cause of
visual morbidity due to its multiple recurrence and progression.
The virus remains latent in the sensory and autonomic neurons
after infection and its reactivation is thought to be multifactorial.
Reactivation most commonly involves the anterior segment of
the eye, and it manifests as corneal epithelial keratitis, stromal
keratitis, endotheliitis, and/or iritis. According to the Herpetic
Eye Disease Study, demographic data and other predictors, such
as age, gender, and ethnicity, and nonocular herpes, seasonal
variation, history of epithelial keratitis or psychogenic stress,
were not significantly associated with recurrences.9 There is ev-
idence of reactivation in the published literature following the
use of ocular laser or surgical procedures, that is, laser‑assisted
in situ keratomileusis, laser iridotomy, laser trabeculoplasty, cat-
aract surgery, keratoplasty, and collagen crosslinking, and top-
ical prostaglandin analogues like latanoprost.10–13
So far, only a few reports of reactivation of ocular herpes
simplex keratitis and herpes zoster ophthalmicus following the
periocular botulinum toxin injection have been described in the
literature. Lingua6 described a case of reactivated homolateral
herpes simplex keratitis within 3 days after botulinum toxin injec-
tion. Ramappa et al.4 recently described a case of HSV reactiva-
tion following periocular injection of botulinum toxin for spastic
entropion in a 55-year-old male who developed stromal necrotic
keratitis after 1 week. Another case was described by Narang et
al.,5 where a 59-year-old man developed bilateral epithelial ker-
atitis with stromal edema 3 weeks after bilateral botulinum toxin
injection in the lacrimal gland for functional epiphora. In our case,
patient had no epithelial or stromal involvement but she developed
HSV endotheliitis. Like previous cases, our patient was also neg-
ative for HSV 1 in polymerase chain reaction but this negative
result does not refute our diagnosis as HSV endotheliitis as a large
sample study of 140 cases by Kakimaru-Hasegawa et al.14 showed
that proliferation of HSV DNA copies in real time polymerase
chain reaction were least in number among endotheliitis cases than
in other forms of herpetic keratitis. So, the yield of viral DNA by
real time polymerase chain reaction is low in HSV endotheliitis.14
Authors in earlier reports believed that reactivation either
may be coincidence or there may be a causal association which
is difficult to prove and needs further research. In the literature,
some authors hypothesize that stressors in different forms may
include local trauma that exerts a local epigenetic influence on
viral transcription, allowing for viral reactivation.12 Our present
case further strengthens that there may be a possibility of asso-
ciation of periocular botulinum toxin injection and reactivation
of herpes simplex keratitis. This is important because there is
a need for repeated botulinum toxin injection as it has limited
duration of action and hence repeated chance of herpetic reac-
tivation. So, it is important to question the patient about any
prior history of herpetic eye disease or to search for and not
to miss clinical signs of it, before periocular botulinum toxin
injection. The patient should be warned about the possible risk
of herpetic reactivation and appropriate perioperative antiviral
prophylaxis may be recommended in cases with previous her-
petic eye diseases.
© 2019 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. e75
Copyright © 2020 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc. Unauthorized reproduction of this article is prohibited.
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119 patients with hemifacial spasm over 11 years. Clin Neurol
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2. Defazio G, Abbruzzese G, Girlanda P, et al. Botulinum toxin A
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3. Elston JS. Botulinum toxin treatment of hemifacial spasm. J Neurol
Neurosurg Psychiatry 1986;49:827–829.
4. Ramappa M, Jiya PY, Chaurasia S, et al. Reactivation of herpes
simplex viral keratitis following the botulinum toxin injection.
Indian J Ophthalmol 2018;66:306–308.
5. Narang P, Singh S, Mittal V. Bilateral herpes simplex keratitis re-
activation after lacrimal gland botulinum toxin injection. Indian J
Ophthalmol 2018;66:697–699.
6. Lingua RW. Sequelae of botulinum toxin injection. Am J
Ophthalmol. 1985;100:305–307
7. Gadient PM, Smith JH, Ryan SJ. Herpes zoster ophthalmicus fol-
lowing onabotulinumtoxinA administration for chronic migraine:
a case report and literature review. Cephalalgia 2015;35:443–438.
8. Porter SM, Patterson A, Kho P. A comparison of local and systemic
acyclovir in the management of herpetic disciform keratitis. Br J
Ophthalmol 1990;74:283–285.
9. Predictors of recurrent herpes simplex virus keratitis. Herpetic Eye
Disease Study Group. Cornea 2001;20:123–128.
10. White ML, Chodosh J. Herpes Simplex Virus Keratitis: A
Treatment Guideline‑ 2014. This Guideline was approved by the
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Guideline Was Reviewed and Accepted by the Hoskins Centre for
Quality EyeCare, American Academy of Ophthalmology in the
Compendium of Evidence‑Based Eye Care™; June 2014. Accessed
on 16th December 2018
11. Ringvold A, Davanger M, Olsen EG. Changes of the cornea en-
dothelium after ultraviolet radiation. Acta Ophthalmol (Copenh)
1982;60:41–53.
12. Morales J, Shihab ZM, Brown SM, et al. Herpes simplex vi-
rus dermatitis in patients using latanoprost. Am J Ophthalmol
2001;132:114–116.
13. Moshirfar M, Murri MS, Shah TJ, et al. A review of corneal endo-
theliitis and endotheliopathy: differential diagnosis, evaluation, and
treatment. Ophthalmol Ther 2019;8:195–213.
14. Kakimaru-Hasegawa A, Kuo CH, Komatsu N, et al. Clinical appli-
cation of real-time polymerase chain reaction for diagnosis of her-
petic diseases of the anterior segment of the eye. Jpn J Ophthalmol
2008;52:24–31.
Orbital Leakage of Intraocular
Silicone Oil: Case Reports and
Literature Review
Stephanie Lemaitre, M.D., Ph.D.*,
Kaveh Vahdani, M.D., FRCOphth.*, Pierre Escalas, M.D.†,
and Geoffrey E. Rose, D.Sc., FRCOphth.*
Abstract: Silicone oil migration into the periorbital tissues
occurs rarely after vitreo-retinal surgery and two cases
are reported. The first case was a 48-year-old woman who
presented with a blind painful right eye, upper eyelid
*Adnexal Service, Moorfields Eye Hospital, City Road, London EC1V
2PD; and †Polyclinique Atlantique, Avenue Claude Bernard BP 40419,
44800 Saint-Herblain, France
Accepted for publication December 14, 2019.
The authors have no financial or conflicts of interest to disclose.
Address correspondence and reprint requests to Professor Geoffrey E.
Rose, D.Sc., FRCOphth, Adnexal Service, Moorfields Eye Hospital NHS
Foundation Trust, City Road, London EC1V 2PD, United Kingdom. E-mail:
geoff.rose1@nhs.net.
DOI: 10.1097/IOP.0000000000001588