- Email: [email protected]
Stensen duct dilation: Case series of minimally invasive treatment
Accepted Manuscript
Stensen’s duct dilation: case series of minimally invasive treatment Le Roux Marc-Kevin MD , Graillon Nicolas MD , Hadj-Sa¨ıd Mehdi , Scemama Ugo MD , Lutz Jean-Christophe , Chossegros Cyrille MD, PHD PII: DOI: Reference:
S2212-4403(19)30106-3 https://doi.org/10.1016/j.oooo.2019.01.078 OOOO 4097
To appear in:
Oral Surg Oral Med Oral Pathol Oral Radiol
Received date: Revised date: Accepted date:
3 November 2018 12 January 2019 16 January 2019
Please cite this article as: Le Roux Marc-Kevin MD , Graillon Nicolas MD , Hadj-Sa¨ıd Mehdi , Scemama Ugo MD , Lutz Jean-Christophe , Chossegros Cyrille MD, PHD , Stensen’s duct dilation: case series of minimally invasive treatment, Oral Surg Oral Med Oral Pathol Oral Radiol (2019), doi: https://doi.org/10.1016/j.oooo.2019.01.078
This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Stensen’s duct dilation: case series of minimally invasive treatment Le Roux Marc-Kevin MD,1,2 Graillon Nicolas MD,1,2 Hadj-Saïd Mehdi,1,2 Scemama Ugo, MD,2,3
CR IP T
Lutz Jean-Christophe,4 Chossegros Cyrille MD, PHD,1,2
1. Oral and Maxillofacial surgery department, University Hospital Center Conception, 147 Boulevard Baille, 13005 Marseille, France
AN US
2. Aix Marseille University, SPMC EA 3279, 27, boulevard Jean-Moulin, 13385 Marseille, France
3. Department of Radiology, Hôpital Nord, Aix-Marseille University, chemin des Bourrely, 13915 cedex 20 Marseille, France
M
4. Stomatology and Maxillo-Facial Surgery Department, Strasbourg University Hospital,
PT
ED
1, place de l’Hôpital, 67091 Strasbourg cedex, France
CE
Author’s affiliation and address:
Le Roux Marc-Kevin Email : [email protected], [email protected],
AC
Phone : 00 33 4 91 43 58 48. Fx : 00 33 4 91 43 54 22 Address: Oral and Maxillofacial surgery department, University Hospital Center Conception, 147 boulevard Baille, 13005 Marseille, France
ACCEPTED MANUSCRIPT
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Abstract:
CR IP T
Stensen’s duct dilation is a rare condition defined by a global or partial idiopathic dilation of the Stensen’s duct. Affected patients usually show either aesthetic features, such as a tubular-shaped swelling in the cheek, or with mildly painful inflammatory episodes. Three women between the ages of 61 and 67 years were diagnosed with Stensen’s duct dilatation after sialoMRI. They were
AN US
treated with botulinum toxin A (BTX-A). Our preliminary results suggest that Botulinum Toxin A was efficient as a suspensive treatment. BTX-A should prevail as a first-line treatment of the early silent symptoms of Stensen’s duct dilation like swelling and aesthetic discomfort. The use
ED
M
of BTX-A avoids more serious procedures and further complications.
CE
PT
Keywords: Salivary glands, Sialadenitis, Botulinum Toxin A, Stensen's Duct
This research did not receive any specific grant from funding agencies in the public, commercial,
AC
or not-for-profit sectors.
ACCEPTED MANUSCRIPT
Introduction:
CR IP T
Non-tumor salivary pathologies can be classified into 3 categories: lithiasis, stenosis, and dilation. In the order of frequency, lithiasis occurs more often, followed by stenosis and dilation.1 One of the etiologies of the salivary gland duct dilation affecting only the Stensen’s duct is
AN US
known as “megaduct”, Stensen’s duct sialectasis, or Stensen’s duct dilation.
The management of ductal deformities depends on their etiology, size and location. Due to its scarcity and unique characteristics, Stensen’s duct dilation must be treated specifically. Patients affected with this pathology indicate only few symptoms or even none at all. They are usually
M
referred either for aesthetic purposes due to a tubular-shaped swelling in the cheek, inflammatory episodes with mild pain, or a purulent flow in the oral cavity. Pruritus or mild discomfort located
ED
in the cheek are often early symptoms of swelling or infections. It is only years later, once annoying symptoms have appeared, patients do refer to a specialist. Wang et al.2 and Seifert et
PT
al.3 found that congenital Stensen’s duct dilation was involved in non-tumor parotid gland
CE
diseases in 3.5% and 1.5% of a series of 200 and 360 patients, respectively . Patients affected with this pathology don’t have a personal history of salivary pathology. Some
AC
cases of familial occurrence have been described in the literature.4,5 The existing articles only mention a conventional diet and lifestyle rules as medical treatment.2,4 Vatin et al.5 and Kandl et al.6 have suggested sialendoscopy as a first step to handle this pathology.7 In 2007, Baumarsch et al.8 described the pull-through technique to avoid the very
ACCEPTED MANUSCRIPT
invasive superficial parotidectomy proposed by Wang.2 Lohia et al., seeking a minimally invasive treatment, recently described marsupialization.9 The purpose of this article is to report the use of botulinum toxin A (BTX-A) in the treatment of
illustrated by the preliminary results of 3 of our cases.
AN US
Patients and methods:
CR IP T
Stensen’s duct dilation as a minimally invasive treatment of salivary diseases. Our experience is
A retrospective study was conducted in the maxillofacial surgery department of Conception’s university hospital in Marseille from September 2014 to September 2018. The inclusion criteria were patients with Stensen’s duct dilation confirmed by MRI and treated
M
with BTX-A. Patients with non-primary cause of ductal dilation were excluded.
ED
The drug used was Botox ® (Allergan, Irvine, California). Vials were reconstituted with 2.5 mL of 0.9% sterile non-preserved saline solution. 50 or 100 IU were delivered blindly in
PT
three specific areas on the sides of the pathological Stensen. The following were used as anatomical landmarks: postero-superiorly the tragus, anteriorly the masseter muscle,
CE
superiorly the zygomatic arch, and postero-inferiorly the sternocleidomastoid muscle. The first injection point was 0.5 cm anterior to the tragus. The second point was 0.5 cm inferior,
AC
and the third point was 0.5 cm inferior to the second and posterior to the ascending branch of the mandible. The collected data for every patient were age, gender, previous and current symptomatology, previous and current treatment. We collected the number and location of the infiltration points, the number of units of BTX-A used and the intervals between
ACCEPTED MANUSCRIPT
infiltrations. The assessment criteria included the types of symptoms such as pain and swelling and their periodicity. Swelling was assessed using patient clinical descriptions or clinically objectified when major.
CR IP T
This study was conducted in accordance with the principles of the Helsinki Declaration (2013) and with the approval of the CIL from University Hospital Conception (RGPD/APHM n°2018-15).
AN US
Results:
Three women aged from 61 to 67-year-old were referred to our department for chronic salivary symptoms. Symptoms were bilateral cheek discomfort relieved by rough parotid
M
massages, right painful swelling (Figure 1) and recurrent parotitis requiring multiples
ED
antibiotics therapies. The time length of the symptoms varied from a few weeks to 30 years.
PT
Symptoms lacked any correlations to meal time and no lithiasis or abnormality was found on the CT-scan. The suspicion of Stensen’s duct dilation was confirmed through a
CE
sialoMRI. It showed a typical sausage-string appearance of Stensen’s duct, confirming the
AC
diagnosis (Figure 2,3,4,5).
The first patient with recurrent parotitis and Lupus syndrome in her personal history went through a sialendoscopy which was proven ineffective. Three months later, she received an
ACCEPTED MANUSCRIPT
infiltration of 50 IU of BTX-A in the left parotid gland but also appeared ineffective. The second dose performed eight months later, carried a higher dose of 100 IU. This time, it allowed a drastic improvement of the patient’s symptoms with the resorption
CR IP T
of swelling and discomfort. Five months later, a third infiltration of 100 UI of BTX-A was performed, following the patient’s demand. After six months, the patient requested another infiltration because the swelling had reappeared. Due to an unexpected health problem, the treatment had to be
AN US
postponed (Table 1).
The second and the third patients were treated respectively by 1 and 3 infiltrations of 100 IU of BTX-A. Infiltration of 100 IU of BTX-A in the right parotid gland was performed for
M
the second patient. After 18 months, the patient hadn’t experienced any inflammatory recurrence, although mild cheek swelling persisted.
ED
The first infiltration of 100 IU of BTX-A alleviated the third patient symptoms. Two additional infiltrations were performed after six and eight months, respectively. Over this
PT
period, the patient systematically refused sialendoscopy under general anesthesia. Six
CE
months later, the patient was hospitalized for four days due to severe left parotitis, which was treated by antibiotics and corticosteroid therapy. Due to clinical worsening, an
AC
interventional sialendoscopy was performed on the left parotid duct with irrigation and soft dilation which fully resolved symptoms.
Discussion:
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We have reported for the first time in medical literature the successful treatment of Stensen’s duct dilation using BTX-A. This simple and reliable procedure can be performed during a consultation and does not imply any severe side effects. Its efficacy has been previously proven in salivary gland diseases, especially for drooling. BTX-A has also been used for sialectasis after
CR IP T
traumatic or postoperative parotid wound.10,11 Most of the patients described in our series needed one infiltration of 100 IU of BTX-A every six months for comfortable symptom relief.
BTX-A infiltration avoided therapeutic escalation. While ultrasonography increases infiltration
AN US
accuracy and therefore yields better reduction of saliva secretion, blind infiltration does not seem to cause additional side effects.12
Although it is a minimally invasive procedure, sialendoscopy requires general anesthesia. Surgical treatments, such as superficial parotidectomy, have a higher risk of complications and
M
are therefore difficult to justify when treating a mild pathology.
ED
BTX-A in the parotid gland reduces the salivary flow while retaining basal secretion. Full effect usually occurs two to eight weeks after BTX-A infiltration.13 The Stensen’s duct size slightly
PT
decreases after such treatment, and most of the inflammatory episodes are avoided.
CE
Regarding Stensen’s duct sialectasis with minor symptoms, no medical treatment was retrieved in the literature to our knowledge, except for antihistaminic medication in case of pruritus, diet and
AC
lifestyle advice.2,4 Because of the sole esthetic inconvenience of this condition and the occasional character of painful episodes, we believe that the least invasive treatment should be considered. BTX-A infiltration avoided therapeutic escalation in mild cases and proved appropriate for patients who could not accept surgery. However, the efficacy is limited in time. In case of intense discomfort and severe inflammatory episodes, BTX-A infiltration could not be used as the first
ACCEPTED MANUSCRIPT
choice of treatment. Other potentially invasive procedures, such as sialendoscopy or surgery, might have to be performed. Sialendoscopy is a satisfactory minimally invasive treatment, thus it can be an alternative in case of BTX-A failure. However, sialendoscopy can be ineffective, especially when there is a blind channel associated with the duct sialectasis. In such case, the best
CR IP T
option seems to be the pull-through of the Stensen’s duct or its marsupialization. Marsupialization is possible only if the dilation is close enough to the oral mucosa. These two techniques should be considered before performing a superficial parotidectomy with Stensen’s duct excision, as it is associated with a high rate of complication.14 BTX-A should prevail as a
AN US
first-line treatment of the early silent symptoms of Stensen’s duct dilation like swelling and aesthetic discomfort. The use of BTX-A helps to avoid more serious procedures and meets
M
patients’ expectations for a minimally invasive treatment.
CE
PT
or not-for-profit sectors.
ED
This research did not receive any specific grant from funding agencies in the public, commercial,
AC
References :
1.
Marchal F, Chossegros C, Faure F, et al. Salivary stones and stenosis. A comprehensive
classification. Revue de Stomatologie et de Chirurgie Maxillo-faciale. 2008;109(4):233-236. 2.
Wang Y, Yu G-Y, Huang M-X, Mao C, Zhang L. Diagnosis and treatment of congenital
ACCEPTED MANUSCRIPT
dilatation of Stensen’s duct: Congenital Dilatation of Stensen’s Duct. The Laryngoscope. 2011;121(8):1682-1686. 3.
Seifert G, Thomsen S, Donath K. Bilateral dysgenetic polycystic parotid glands.
Virchows Arch A Pathol Anat Histol. 1981;390(3):273-288. 4.
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Morphological analysis and differential diagnosis of a rare disease of the salivary glands.
Lee DH, Yoon TM, Lee JK, Lim SC. Congenital dilatation of Stensen’s duct in siblings.
International Journal of Pediatric Otorhinolaryngology. 2015;79(11):1952-1954. Smith M. Familial incidence of sialectasis. Br Med J. 1953;2(4850):1359.
6.
Vatin L, Foletti JM, Collet C, Varoquaux A, Chossegros C. A case of bilateral megaducts:
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5.
Diagnostic and treatment methods. Journal of Stomatology, Oral and Maxillofacial Surgery.
7.
M
2017;118(2):125-128.
Kandl JA, Ong AA, Gillespie MB. Pull-through sialodochoplasty for Stensen’s megaduct:
Baurmash HD. Sialectasis of Stensen’s Duct With an Extraoral Swelling: A Case Report
PT
8.
ED
Sialodochoplasty for Stensen’s Megaduct. The Laryngoscope. 2016;126(9):2003-2005.
9.
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With Surgical Management. Journal of Oral and Maxillofacial Surgery. 2007;65(1):140-143. Lohia S, Joshi AS. Idiopathic sialectasis of the Stensen’s duct treated with
AC
marsupialisation. Case Reports. 2013;2013(nov14 1):bcr2013201548-bcr2013201548. 10.
Lipp A, Trottenberg T, Schink T, Kupsch A, Arnold G. A randomized trial of botulinum
toxin A for treatment of drooling. Neurology. 2003;61(9):1279-1281. 11.
Arnaud S, Batifol D, Goudot P, Yachouh J. Prise en charge non chirurgicale des plaies de
la glande parotide et du canal de Stenon: intérêt de la toxine botulinique. Annales de Chirurgie
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Plastique Esthétique. 2008;53(1):36-40. 12.
Dogu O, Apaydin D, Sevim S, Talas DU, Aral M. Ultrasound-guided versus ‘blind’
intraparotid injections of botulinum toxin-A for the treatment of sialorrhoea in patients with
13.
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Parkinson’s disease. Clinical Neurology and Neurosurgery. 2004;106(2):93-96. Jongerius PH, van den Hoogen FJA, van Limbeek J, Gabreëls FJ, van Hulst K, Rotteveel
JJ. Effect of botulinum toxin in the treatment of drooling: a controlled clinical trial. Pediatrics. 2004;114(3):620-627.
Patel RS, Low T-HH, Gao K, O’Brien CJ. Clinical Outcome After Surgery for 75 Patients
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14.
PT
ED
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With Parotid Sialadenitis. The Laryngoscope. 2007;117(4):644-647.
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CE
Figure 1: Swelling of the right Stensen’s duct or “Mustache sign”
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ED
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Figure 2: SialoMRI of the first patient showing a bilateral Stensen's duct dilation
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ED
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Figure 3: SialoMRI of the second patient showing a right Stensen's duct dilation
M
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Figure 4: 3D SialoMRI of the second patient showing a right Stensen’s duct
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dilation
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PT
ED
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Figure 5: SialoMRI of the third patient showing a left Stensen's duct dilation
Pati ent 1
A Gen ge der 6 Fem 1 ale
Sympt oms Bilater al cheek swellin g rare episod
Lenght of symtoms Since 2014 First visit in the department : 10/2015 First infiltration :
Prior Tratment Sialendoscopie early 2015
Number of BTX-A infiltration required 4 unilateral infiltrations : 1 infiltration of 50 IU 3 infiltrations of 100 IU
Outcome Drastic reductio n of sympto ms Request another
ACCEPTED MANUSCRIPT
Fem ale
Since 1988. Worsening since 04/2016 First visit in the department : 05/2016 First infiltration : 05/2016
Diet and lifestyle rules Phloroglucinol Cure of amoxicilline + acide clavulanique during patent infection
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1 unilateral infiltration : 100 IU
Mild cheek swelling No more inflamm atory episodes at the last visit
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6 5
Self-massage
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3
Since 2008 First visit in the department : 10/2015 First infiltration : 09/2016
M
Fem ale
ED
6 7
infiltratio n at the last visit
09/2016
PT
2
e of pain on the left side Right side cheek swellin g Mild pain at palpati on only Slow worse ning Reccur rent left parotit is Pain and purule nt flow at the ostium Lupus syndro me
3 unilateral infiltrations : 100 IU
Occurren ce of a severe left parotitis treated by sialendos copie No more sympto ms after sialendos copie
Stensen’s duct dilation: case series of minimally invasive treatment Le Roux Marc-Kevin MD , Graillon Nicolas MD , Hadj-Sa¨ıd Mehdi , Scemama Ugo MD , Lutz Jean-Christophe , Chossegros Cyrille MD, PHD PII: DOI: Reference:
S2212-4403(19)30106-3 https://doi.org/10.1016/j.oooo.2019.01.078 OOOO 4097
To appear in:
Oral Surg Oral Med Oral Pathol Oral Radiol
Received date: Revised date: Accepted date:
3 November 2018 12 January 2019 16 January 2019
Please cite this article as: Le Roux Marc-Kevin MD , Graillon Nicolas MD , Hadj-Sa¨ıd Mehdi , Scemama Ugo MD , Lutz Jean-Christophe , Chossegros Cyrille MD, PHD , Stensen’s duct dilation: case series of minimally invasive treatment, Oral Surg Oral Med Oral Pathol Oral Radiol (2019), doi: https://doi.org/10.1016/j.oooo.2019.01.078
This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Stensen’s duct dilation: case series of minimally invasive treatment Le Roux Marc-Kevin MD,1,2 Graillon Nicolas MD,1,2 Hadj-Saïd Mehdi,1,2 Scemama Ugo, MD,2,3
CR IP T
Lutz Jean-Christophe,4 Chossegros Cyrille MD, PHD,1,2
1. Oral and Maxillofacial surgery department, University Hospital Center Conception, 147 Boulevard Baille, 13005 Marseille, France
AN US
2. Aix Marseille University, SPMC EA 3279, 27, boulevard Jean-Moulin, 13385 Marseille, France
3. Department of Radiology, Hôpital Nord, Aix-Marseille University, chemin des Bourrely, 13915 cedex 20 Marseille, France
M
4. Stomatology and Maxillo-Facial Surgery Department, Strasbourg University Hospital,
PT
ED
1, place de l’Hôpital, 67091 Strasbourg cedex, France
CE
Author’s affiliation and address:
Le Roux Marc-Kevin Email : [email protected], [email protected],
AC
Phone : 00 33 4 91 43 58 48. Fx : 00 33 4 91 43 54 22 Address: Oral and Maxillofacial surgery department, University Hospital Center Conception, 147 boulevard Baille, 13005 Marseille, France
ACCEPTED MANUSCRIPT
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Abstract:
CR IP T
Stensen’s duct dilation is a rare condition defined by a global or partial idiopathic dilation of the Stensen’s duct. Affected patients usually show either aesthetic features, such as a tubular-shaped swelling in the cheek, or with mildly painful inflammatory episodes. Three women between the ages of 61 and 67 years were diagnosed with Stensen’s duct dilatation after sialoMRI. They were
AN US
treated with botulinum toxin A (BTX-A). Our preliminary results suggest that Botulinum Toxin A was efficient as a suspensive treatment. BTX-A should prevail as a first-line treatment of the early silent symptoms of Stensen’s duct dilation like swelling and aesthetic discomfort. The use
ED
M
of BTX-A avoids more serious procedures and further complications.
CE
PT
Keywords: Salivary glands, Sialadenitis, Botulinum Toxin A, Stensen's Duct
This research did not receive any specific grant from funding agencies in the public, commercial,
AC
or not-for-profit sectors.
ACCEPTED MANUSCRIPT
Introduction:
CR IP T
Non-tumor salivary pathologies can be classified into 3 categories: lithiasis, stenosis, and dilation. In the order of frequency, lithiasis occurs more often, followed by stenosis and dilation.1 One of the etiologies of the salivary gland duct dilation affecting only the Stensen’s duct is
AN US
known as “megaduct”, Stensen’s duct sialectasis, or Stensen’s duct dilation.
The management of ductal deformities depends on their etiology, size and location. Due to its scarcity and unique characteristics, Stensen’s duct dilation must be treated specifically. Patients affected with this pathology indicate only few symptoms or even none at all. They are usually
M
referred either for aesthetic purposes due to a tubular-shaped swelling in the cheek, inflammatory episodes with mild pain, or a purulent flow in the oral cavity. Pruritus or mild discomfort located
ED
in the cheek are often early symptoms of swelling or infections. It is only years later, once annoying symptoms have appeared, patients do refer to a specialist. Wang et al.2 and Seifert et
PT
al.3 found that congenital Stensen’s duct dilation was involved in non-tumor parotid gland
CE
diseases in 3.5% and 1.5% of a series of 200 and 360 patients, respectively . Patients affected with this pathology don’t have a personal history of salivary pathology. Some
AC
cases of familial occurrence have been described in the literature.4,5 The existing articles only mention a conventional diet and lifestyle rules as medical treatment.2,4 Vatin et al.5 and Kandl et al.6 have suggested sialendoscopy as a first step to handle this pathology.7 In 2007, Baumarsch et al.8 described the pull-through technique to avoid the very
ACCEPTED MANUSCRIPT
invasive superficial parotidectomy proposed by Wang.2 Lohia et al., seeking a minimally invasive treatment, recently described marsupialization.9 The purpose of this article is to report the use of botulinum toxin A (BTX-A) in the treatment of
illustrated by the preliminary results of 3 of our cases.
AN US
Patients and methods:
CR IP T
Stensen’s duct dilation as a minimally invasive treatment of salivary diseases. Our experience is
A retrospective study was conducted in the maxillofacial surgery department of Conception’s university hospital in Marseille from September 2014 to September 2018. The inclusion criteria were patients with Stensen’s duct dilation confirmed by MRI and treated
M
with BTX-A. Patients with non-primary cause of ductal dilation were excluded.
ED
The drug used was Botox ® (Allergan, Irvine, California). Vials were reconstituted with 2.5 mL of 0.9% sterile non-preserved saline solution. 50 or 100 IU were delivered blindly in
PT
three specific areas on the sides of the pathological Stensen. The following were used as anatomical landmarks: postero-superiorly the tragus, anteriorly the masseter muscle,
CE
superiorly the zygomatic arch, and postero-inferiorly the sternocleidomastoid muscle. The first injection point was 0.5 cm anterior to the tragus. The second point was 0.5 cm inferior,
AC
and the third point was 0.5 cm inferior to the second and posterior to the ascending branch of the mandible. The collected data for every patient were age, gender, previous and current symptomatology, previous and current treatment. We collected the number and location of the infiltration points, the number of units of BTX-A used and the intervals between
ACCEPTED MANUSCRIPT
infiltrations. The assessment criteria included the types of symptoms such as pain and swelling and their periodicity. Swelling was assessed using patient clinical descriptions or clinically objectified when major.
CR IP T
This study was conducted in accordance with the principles of the Helsinki Declaration (2013) and with the approval of the CIL from University Hospital Conception (RGPD/APHM n°2018-15).
AN US
Results:
Three women aged from 61 to 67-year-old were referred to our department for chronic salivary symptoms. Symptoms were bilateral cheek discomfort relieved by rough parotid
M
massages, right painful swelling (Figure 1) and recurrent parotitis requiring multiples
ED
antibiotics therapies. The time length of the symptoms varied from a few weeks to 30 years.
PT
Symptoms lacked any correlations to meal time and no lithiasis or abnormality was found on the CT-scan. The suspicion of Stensen’s duct dilation was confirmed through a
CE
sialoMRI. It showed a typical sausage-string appearance of Stensen’s duct, confirming the
AC
diagnosis (Figure 2,3,4,5).
The first patient with recurrent parotitis and Lupus syndrome in her personal history went through a sialendoscopy which was proven ineffective. Three months later, she received an
ACCEPTED MANUSCRIPT
infiltration of 50 IU of BTX-A in the left parotid gland but also appeared ineffective. The second dose performed eight months later, carried a higher dose of 100 IU. This time, it allowed a drastic improvement of the patient’s symptoms with the resorption
CR IP T
of swelling and discomfort. Five months later, a third infiltration of 100 UI of BTX-A was performed, following the patient’s demand. After six months, the patient requested another infiltration because the swelling had reappeared. Due to an unexpected health problem, the treatment had to be
AN US
postponed (Table 1).
The second and the third patients were treated respectively by 1 and 3 infiltrations of 100 IU of BTX-A. Infiltration of 100 IU of BTX-A in the right parotid gland was performed for
M
the second patient. After 18 months, the patient hadn’t experienced any inflammatory recurrence, although mild cheek swelling persisted.
ED
The first infiltration of 100 IU of BTX-A alleviated the third patient symptoms. Two additional infiltrations were performed after six and eight months, respectively. Over this
PT
period, the patient systematically refused sialendoscopy under general anesthesia. Six
CE
months later, the patient was hospitalized for four days due to severe left parotitis, which was treated by antibiotics and corticosteroid therapy. Due to clinical worsening, an
AC
interventional sialendoscopy was performed on the left parotid duct with irrigation and soft dilation which fully resolved symptoms.
Discussion:
ACCEPTED MANUSCRIPT
We have reported for the first time in medical literature the successful treatment of Stensen’s duct dilation using BTX-A. This simple and reliable procedure can be performed during a consultation and does not imply any severe side effects. Its efficacy has been previously proven in salivary gland diseases, especially for drooling. BTX-A has also been used for sialectasis after
CR IP T
traumatic or postoperative parotid wound.10,11 Most of the patients described in our series needed one infiltration of 100 IU of BTX-A every six months for comfortable symptom relief.
BTX-A infiltration avoided therapeutic escalation. While ultrasonography increases infiltration
AN US
accuracy and therefore yields better reduction of saliva secretion, blind infiltration does not seem to cause additional side effects.12
Although it is a minimally invasive procedure, sialendoscopy requires general anesthesia. Surgical treatments, such as superficial parotidectomy, have a higher risk of complications and
M
are therefore difficult to justify when treating a mild pathology.
ED
BTX-A in the parotid gland reduces the salivary flow while retaining basal secretion. Full effect usually occurs two to eight weeks after BTX-A infiltration.13 The Stensen’s duct size slightly
PT
decreases after such treatment, and most of the inflammatory episodes are avoided.
CE
Regarding Stensen’s duct sialectasis with minor symptoms, no medical treatment was retrieved in the literature to our knowledge, except for antihistaminic medication in case of pruritus, diet and
AC
lifestyle advice.2,4 Because of the sole esthetic inconvenience of this condition and the occasional character of painful episodes, we believe that the least invasive treatment should be considered. BTX-A infiltration avoided therapeutic escalation in mild cases and proved appropriate for patients who could not accept surgery. However, the efficacy is limited in time. In case of intense discomfort and severe inflammatory episodes, BTX-A infiltration could not be used as the first
ACCEPTED MANUSCRIPT
choice of treatment. Other potentially invasive procedures, such as sialendoscopy or surgery, might have to be performed. Sialendoscopy is a satisfactory minimally invasive treatment, thus it can be an alternative in case of BTX-A failure. However, sialendoscopy can be ineffective, especially when there is a blind channel associated with the duct sialectasis. In such case, the best
CR IP T
option seems to be the pull-through of the Stensen’s duct or its marsupialization. Marsupialization is possible only if the dilation is close enough to the oral mucosa. These two techniques should be considered before performing a superficial parotidectomy with Stensen’s duct excision, as it is associated with a high rate of complication.14 BTX-A should prevail as a
AN US
first-line treatment of the early silent symptoms of Stensen’s duct dilation like swelling and aesthetic discomfort. The use of BTX-A helps to avoid more serious procedures and meets
M
patients’ expectations for a minimally invasive treatment.
CE
PT
or not-for-profit sectors.
ED
This research did not receive any specific grant from funding agencies in the public, commercial,
AC
References :
1.
Marchal F, Chossegros C, Faure F, et al. Salivary stones and stenosis. A comprehensive
classification. Revue de Stomatologie et de Chirurgie Maxillo-faciale. 2008;109(4):233-236. 2.
Wang Y, Yu G-Y, Huang M-X, Mao C, Zhang L. Diagnosis and treatment of congenital
ACCEPTED MANUSCRIPT
dilatation of Stensen’s duct: Congenital Dilatation of Stensen’s Duct. The Laryngoscope. 2011;121(8):1682-1686. 3.
Seifert G, Thomsen S, Donath K. Bilateral dysgenetic polycystic parotid glands.
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With Surgical Management. Journal of Oral and Maxillofacial Surgery. 2007;65(1):140-143. Lohia S, Joshi AS. Idiopathic sialectasis of the Stensen’s duct treated with
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Figure 1: Swelling of the right Stensen’s duct or “Mustache sign”
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Figure 2: SialoMRI of the first patient showing a bilateral Stensen's duct dilation
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Figure 3: SialoMRI of the second patient showing a right Stensen's duct dilation
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Figure 4: 3D SialoMRI of the second patient showing a right Stensen’s duct
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dilation
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Figure 5: SialoMRI of the third patient showing a left Stensen's duct dilation
Pati ent 1
A Gen ge der 6 Fem 1 ale
Sympt oms Bilater al cheek swellin g rare episod
Lenght of symtoms Since 2014 First visit in the department : 10/2015 First infiltration :
Prior Tratment Sialendoscopie early 2015
Number of BTX-A infiltration required 4 unilateral infiltrations : 1 infiltration of 50 IU 3 infiltrations of 100 IU
Outcome Drastic reductio n of sympto ms Request another
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Fem ale
Since 1988. Worsening since 04/2016 First visit in the department : 05/2016 First infiltration : 05/2016
Diet and lifestyle rules Phloroglucinol Cure of amoxicilline + acide clavulanique during patent infection
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1 unilateral infiltration : 100 IU
Mild cheek swelling No more inflamm atory episodes at the last visit
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Self-massage
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Since 2008 First visit in the department : 10/2015 First infiltration : 09/2016
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infiltratio n at the last visit
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e of pain on the left side Right side cheek swellin g Mild pain at palpati on only Slow worse ning Reccur rent left parotit is Pain and purule nt flow at the ostium Lupus syndro me
3 unilateral infiltrations : 100 IU
Occurren ce of a severe left parotitis treated by sialendos copie No more sympto ms after sialendos copie