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Crocodile tears syndrome: botulinum toxin treatment

under EMG guidance

Sibel Kizkin we considered that it might be more appropriate to car-


Selim Doganay ry out the injection procedure under electromyographic
Handan Isin Ozisik (EMG) guidance, in order to inject botulinum toxin selec-
Cemal Ozcan tively into the lacrimal gland and to protect the above-
mentioned muscles.
In this report, we present the findings of the injection of
Faculty of Medicine, Inonu University and Turgut Ozal
botulinum toxin, under EMG guidance, via a conjunctival
Medical Center, Malatya, Turkey
approach, in a patient presenting crocodile tears syn-
drome following idiopathic peripheral facial paralysis
Reprint requests to: Dr Sibel Kizkin
(PFP).
Inonu Universitesi Tıp Fakultesi
Noroloji Anabilim Dalı
TR-44160 Malatya, Turkey
Case report
E-mail: skizkin@inonu.edu.tr
A 19-year-old male patient presented with a 6-month
history of left PFPwithout concomitant medication. Two
Accepted for publication: February 17, 2005 months after the onset of the PFP, his left eye had be-
gun to present tearing during meals. With the exception
of a slightly decreased left nasolabial fold, there were
Summary no pathological findings on neurological examination.
PFP was categorized as Grade II according to the
Crocodile tears syndrome is one of the rare complica- House Brackmann Facial Nerve Grading System (at
tions of facial paralysis. There have been several re- rest: normal tone and symmetry; motion: complete clo-
ports of cases in which botulinum toxin was found to sure of the eyelid with minimum effort, minimal oral
be useful in the treatment of crocodile tears syndrome. asymmetry). Blink reflexes were investigated by EMG
The adverse effects, due to the paralytic action of botu- and a positive response was found after stimulation of
linum toxin, have been reported to involve the palpe- the ipsilateral supraorbital nerves, from both the m. or-
bral muscle, lateral rectus and superior rectus. There- bicularis oculi and m. orbicularis oris, which suggested
fore, we considered that it might be more appropriate to aberrant reinnervation (Fig. 1). Ophthalmological exam-
carry out the injection procedure under electromyo- ination showed normal visual acuity in both eyes. Fun-
graphic guidance in order to inject botulinum toxin se- dus examination was bilaterally normal. Cornea epithe-
lectively into the lacrimal gland and protect the above- lium was healthy and disclosed no epithelial surface
mentioned muscles. pathology.
In conclusion, we recommend EMG guidance in the The patient was examined clinically, and tearing was as-
treatment of crocodile tears syndrome with botulinum sessed by Schirmer’s test before and 1, 4, 12 weeks af-
toxin. ter the injection. According to this test procedure, one
end of a strip of thin filter paper (5 mm wide and 30 mm
KEY WORDS: botulinum toxin, crocodile tears syndrome, elec -
tromyography.

Introduction

Crocodile tears syndrome, also called gustolacrimal re-


flex, paroxysmal lacrimation and Bogorad’s syndrome,
is one of the rare complications of Bell’s palsy or trau-
matic facial paralysis. It was first described by a Russ-
ian neuropathologist in 1913, and is characterized by in-
appropriate and sometimes excessive lacrimation pro-
voked by eating (1,2).
Recently, there have been several reports of cases in
which botulinum toxin was found to be useful in the
treatment of crocodile tears syndrome (1-6). The ad-
verse effects, due to the paralytic action of botulinum
toxin, have been reported to involve the palpebral mus-
cle, lateral rectus and superior rectus (1,3-6). Therefore, Figure 1 - Blink reflexes.

Functional Neurology 2005; 20(1): 35-37 35


S. Kizkin et al.

long) was inserted into the lower conjunctival sac, while tectable by EMG (Fig. 3). The needle only entered the
the other end was allowed to hang over the margin of lacrimal gland. The treatment was not painful.
the lower lid. The tears wet the strip of filter paper, pro- Within 4 days of the botulinum toxin injection, the patient
ducing a moisture measurement. Basal tear secrection noticed a reduction of tear secretion during eating, and
and tear secrection after gustatory stimulation (the pa- a complete disappearance after 7 days. Side effects
tient ate a sweet) were measured. such as eye burning, ptosis, double vision and visual
Basal tear secretion (without gustatory stimulation) was problems were not observed after botulinum toxin injec -
found to be 15 mm in the right eye and 17 mm in the left tion.
eye. Repetition of the Schirmer’s test during salivary One week after treatment (Tables I and II), the
stimulation gave values of 16 mm and over 30 mm, re- Schirmer’s test gave values of 14 mm (left eye) and 15
spectively (Tables I and II). mm (right eye) during gustatory stimulation (eating a
sweet). The patient had no discomfort 12 weeks after
the procedure.
Table I - Schirmer’s test results without gustatory stimula-
tion.

Right eye Left eye

Before treatment 15 mm 17 mm
1 week after treatment 16 mm 16 mm
4 weeks after treatment 15 mm 17 mm
12 weeks after treatment 16 mm 16 mm

Table II - Schirmer ’s test results with gustatory stimulation.

Right eye Left eye

Before treatment 16 mm over 30 mm


Figure 3 - Electrophysiological silence of the lacrimal gland.
1 week after treatment 15 mm 14 mm
4 weeks after treatment 15 mm 16 mm
12 weeks after treatment 16 mm 15 mm Discussion

Crocodile tears are seen in 3.3 to 6.5% of patients fol-


lowing Bell’s palsy (7,8). The syndrome may also occur
Before botulinum toxin injection, the conjunctiva mucosa following acoustic neurinoma surgery, in maxillofacial
was anesthetized with proparacaine HCL(Alcaine 0.5%; trauma, lepra, Paget’s disease, vascular diseases and
Alcon, Belgium). The EMG needle was inserted in the cervicooculoacoustic syndrome (3,9,10).
palpebral portion of the lacrimal gland following ectropi- A misdirection of regenerating parasympathetic fibres is
onization of the upper eyelid. Six units of botulinum tox- widely accepted as the pathogenetic mechanism under-
in (Botox; Allergan, Irvine, Calif.) were injected into the lying crocodile tears syndrome. Gustatory fibres from fa-
palpebral portion of the lacrimal gland (Fig. 2), after it cial or glossopharyngeal nerves may be misled through
had been established that muscle activity was not de- n. petrosus superficialis major to the lacrimal gland,
causing the eye on that same side to present tearing
when the patient eats or drinks (1,3,6). It may rarely be
accompanied by loss of taste and by facial spasm.
Many methods have been used to treat crocodile tears
including drugs such as antihistaminics, anticholinergics
(8), intraorbital alcohol or cocaine injection (1), Vidian
neurectomy (11,12), and subtotal resection of the
lacrimal gland (13). However, none of these treatment
options has been unanimously accepted due to persist-
ent side effects in some of them (e.g., eye burning in
Vidian neurectomy), or inadequate results.
Recent reports suggest that botulinum toxin injection in-
to the lacrimal gland of patients with crocodile tears syn-
drome may effectively abolish the reflex symptom. Bot-
ulinum toxin-A affects neurotransmission by inhibiting
the release of acetylcholine at the neuromuscular junc-
Figure 2 - Botulinum toxin was injected into the palpebral por- tion and at cholinergic autonomic nerve terminals (1,5,
tion of the lacrimal gland. 6,14,15). Botulinum toxin has been applied both tran-

36 Functional Neurology 2005; 20(1): 35-37


EMG-guided botulinum toxin treatment of crocodile tears

scutaneously and transconjunctivally to the lacrimal 13. Yavuzer R, Basterzi Y, Akata F. Botulinum toxin A for the
gland in patients with crocodile tears syndrome (2,5,6, treatment of crocodile tears. Plast Reconstr Surg 2002;
14). Riemann et al. (6) injected 2-5 U botulinum toxin in- 110:369-370
to the palpebral portion of the lacrimal gland, obtaining 14. Keegan DJ, Geerling G, Lee JP, Blake G, Collin JR, Plant
a noticeable reduction in the patient’s complaint in a GT. Botulinum toxin treatment for hyperlacrimation sec-
week; moreover, no side effects were observed. Mon- ondary to aberrant regenerated seventh nerve palsy or
taya et al. (1), on the other hand, first applied 10 U tran- salivary gland transplantation. Br J Ophthalmol 2002;86:
scutaneously. When this proved ineffective, an addition- 43-46
al 10 U botulinum toxin was administered transconjunc- 15. Hofmann RJ. Treatment of Frey’s syndrome (gustatory
tivally. But it was reported that this application caused sweating) and ‘crocodile tears’ (gustatory epiphora) with
side effects such as eye burning and ptosis resulting purified botulinum toxin. Ophthal Plast Reconstr Surg
2000;16:289-291
from involvement of the m. levator palpebra. However,
16. Riemann R, Pfennigsdorf S, Riemann E, Naumann M.
the desired result was achieved with a lower dose using
Successful treatment of crocodile tears by injection of bot-
the transconjunctival rather than the transcutaneous
ulinum toxin into the lacrimal gland. Ophthalmology 1999;
technique.
106:2322-2324
Botulinum toxin injection may have a paralytic effect on
17. Valenca MM, Valenca LP, Lima MC. Idiopathic facial paral-
m. levator palpebra, m. rectus superior, and m. rectus ysis (Bell’s palsy): a study of 180 patients. Arq Neurop-
lateralis due to the proximity of these muscles to the siquiatr 2001;59:733-739
lacrimal gland. To avoid these side effects, we per- 18. Miszke A, Kotowiecki E. Crocodile tears syndrome. Neurol
formed transconjunctival botulinum toxin injection under Neurochir Pol 1975;9:81-87
EMG guidance. Symptoms completely improved one 19. Irving RM, Viani L, Hardy DG, Baguley DM, Moffat DA.
week after application and no complications occurred in Nervus intermedius function after vestibular schwannoma
the related eye and adnexes. Follow-up ophthalmologi- removal: clinical features and pathophysiological mecha-
cal examination with Schirmer’s test 12 weeks after in- nisms. Laryngoscope 1995;105:809-813
jection showed persistence of the botulinum toxin effect. 10. Magliulo G, Cordeschi S, Sepe C, de Vincentiis M. Taste
In conclusion, the use of EMG guidance could increase and lacrimation after acoustic neuroma surgery. Rev
the efficiency and safety of botulinum injection in cases Laryngol Otol Rhinol (Bord) 1998;119:167-170
with crocodile tears syndrome. We recommend EMG 11. Kirtane MV, Ogale SB, Merchant SN. Vidian neurectomy
guidance in the treatment of crocodile tears syndrome for crocodile tears. Indian J Ophthalmol 1984;32:221-223
with botulinum toxin. 12. Chandra R. Treatment of a case of crocodile-tears by Vid-
ian neurectomy. J Laryngol Otol 1967;81:669-671
13. McCoy FJ, Goodman RC. The crocodile tear syndrome.
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Functional Neurology 2005; 20(1): 35-37 37

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