Auris Nasus Larynx 42 (2015) 119–122
Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
The relationship between the fistula tract and the facial nerve in type II first branchial cleft anomalies Burak Ertas a,*, Rıza Onder Gunaydin b, Omer Faruk Unal a a b
Department of Otorhinolaryngology, Acibadem University, Medical School, Maslak, Istanbul, Turkey Department of Otorhinolaryngology, Hacettepe University, Medical School, Sihhiye, Ankara, Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 March 2014 Accepted 22 August 2014 Available online 16 September 2014
Objective: To share our experience involving seven patients with type II first branchial cleft anomalies (hereafter, type II anomalies), to determine whether the location of the external fistula openings of the anomalies are associated with the location of the facial nerve tract, and elucidate the relationship between the location of the fistula opening and the facial nerve. Methods: The medical records of seven patients who underwent surgery from 2005 to 2013 for type II anomalies were retrospectively examined. The relationship between the fistula opening and the facial nerve was evaluated in each patient with respect to whether the fistula opening was superior or inferior to the mandibular angle. All patients underwent partial parotidectomy, facial nerve exposure, and total excision of the mass together with connection of a small cuff of the external auditory canal skin to the fistula tract. Results: The fistula tracts were located medially to the facial nerve in two patients, and both fistulae had openings inferior to the mandibular angle. The fistula tracts were located laterally to the facial nerve in the remaining five patients: one patient had no external opening, one had an opening inferior to the mandibular angle, and the remaining three had openings superior to the mandibular angle. Conclusion: Because type II anomalies are rare, their diagnosis is difficult. Surgery of such lesions is challenging and associated with a high risk due to their proximity to the facial nerve. We believe that the location of the fistula opening may help to identify the relationship between the anomalous lesion and facial nerve. Studies involving larger series of cases are needed to confirm our hypothesis; however, because of the rarity of this specific anomaly, it will not be easy to compile a large number of cases. We believe that our study will encourage further investigation on this subject. ß 2014 Published by Elsevier Ireland Ltd.
Keywords: Branchial anomaly Tract of branchial fistula Facial nerve
1. Introduction Type II first branchial cleft anomalies (hereafter, type II anomalies) constitute a group of rarely encountered congenital anomalies of the neck and head region. Such anomalies account for less than 8% of all branchial anomalies [1,2]. They are typically connected to the external auditory canal or manifest as cysts, sinuses, or fistulae with inflammatory openings on the neck [3]. During embryological development, five branchial arches can be identified, all of which traverse mesodermal arches that are separated by grooves or clefts. The first branchial cleft is unique in that it is the only cleft that does not become obliterated
* Corresponding author at: Department of Otorhinolaryngology, Acibadem University, Medical School, Buyukdere Cad. No: 40 Acibadem Maslak Hastanesi KBB Klinigi, Maslak, Istanbul, Turkey. Tel.: +90 2123044937; mobile: +90 5333265828. E-mail address: [email protected] (B. Ertas). http://dx.doi.org/10.1016/j.anl.2014.08.017 0385-8146/ß 2014 Published by Elsevier Ireland Ltd.
completely by the eighth week of gestation and persists as a definitive structure in the embryo, as it penetrates the underlying mesoderm toward the first pharyngeal pouch. The dorsal portion produces the external auditory meatus; the middle portion forms the cavum conchae; and the ventral portion disappears. The membrane at the bottom of the cleft becomes the outer layer of the tympanic membrane. The ventral portion of the first pharyngeal pouch evolves to form the eustachian tube. First branchial anomalies are an uncommon group of lesions that arise from incomplete closure of the ventral portion of the first branchial cleft. These anomalies are closely related to the parotid gland and have variable relations to the facial nerve. The relationship of a first cleft anomaly to the parotid gland has been demonstrated to be variable, presumably because of the later development of the gland. The fate of the first branchial apparatus is complete by the sixth or seventh week of development, which means that a first branchial anomaly would be formed by this period. The parotid gland, on the other hand, first appears during
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
120 Table 1 Summary of patients in the present clinical series. Patient number
Age in years
Sex
Previous surgery
Finding in external canal
Location of fistula opening (above or below mandibular angle)
Relationship with facial nerve
Surgery
Follow-up
Complications
1
4
F
Drainage
Opening at 6 o’clock
Below
Medial
2 years
Mild external canal narrowing
2
1
M
None
Above
Lateral
4 years
–
3
3
M
Drainage
Bulging posteriorly Bulging inferiorly
Parotidectomy incision: complete excision with partial external canal resection Same as above
Above
Lateral
Same as above
2 years
4 5
4 2
M F
None None
Below Below
Medial Lateral
Same as above Same as above
4 years 5 years
6
3
M
None
Above
Lateral
Same as above
2 months
7
2
M
Drainage Drainage of three abscesses Inadequate excision of fistula opening None
Mild external canal narrowing + scarring at the previous drainage site – Mild marginal paresis –
None
No opening; only a bulge behind auricle
Lateral to main truncus
Same as above
4 years
–
the sixth week of development. The facial nerve and its muscles migrate upward between the sixth and eighth weeks of development. It can be inferred from the embryological patterns that a first branchial cleft anomaly may be found superficial to, deep to, or within the branches of the facial nerve [4]. While type I lesions are of pure ectodermal origin and form cystic masses, type II anomalies may contain a mixture of tissues such as skin, cartilage, and adnexal structures originating from both ectodermal and mesodermal tissues [2,5,6]. They are frequently complicated with infections, causing significant morbidity. Surgical excision is the preferred mode of treatment, but accurate identification of the relationship between the cleft and facial nerve is of prime importance to avoid injury to the facial nerve [7]. A correct diagnosis is also important because misdiagnoses frequently lead to insufficient draining and partial excisions, thus inducing recurrence. In this retrospective study, we investigated the clinical features of seven patients with type II anomalies and examined the relationship between the location of the fistula opening and the facial nerve.
All patients had type II anomalies according to Work’s classification [5]. Six patients had fistulae and one had a cyst according to Olsen’s classification [1]. All patients were treated with various antibiotics before referral to our clinic. Five patients underwent no surgical intervention before referral. Two patients (28.5%) underwent previous surgery: cyst incision and draining in one patient and incomplete surgical resection in the other. Typical clinical symptoms were bulging or inflammation on the neck due to a cyst, sinus or fistula connection to the external auditory canal, and leakage after opening to the skin. All patients underwent diagnostic neck ultrasonography. Histopathological findings revealed that all patients had branchial cleft anomalies. The mass was excised in a single-phase surgery in all patients. Following creation of a modified Blair incision, the facial nerve tract was exposed during partial parotidectomy, and a small cuff of external auditory canal skin related to the fistula tract plus peripheral inflammatory tissues were excised together with the epithelial tract (Figs. 1–3, belong to the first patient described in Table 1). The neuromonitoring of facial nerves using a Xomed
2. Patients and methods The complete medical records of seven patients who were surgically treated for type II anomalies from July 2005 to 2013 in our otorhinolaryngology clinic were retrospectively evaluated. Magnetic resonance imaging, operational notes, histopathological findings, and all records related to diagnosis and therapy were reviewed. All data revealing any relationship between the facial nerve and the branchial cleft were duly analyzed. Patients’ records are summarized in Table 1. Two and five patients were female and male, respectively, with a mean age of 2.7 years at the time of intervention. Five lesions were located on the right and two were on the left. Fistula openings were located over the mandibular angle in three patients and below the mandibular angle in three patients; one patient had no external fistula opening. There were no other pathologic findings in the patients’ ear, throat, or nose in head and neck examinations. Their general physical examinations also revealed no pathologic findings.
Fig. 1. Intraoperative photograph. (a) Fistula tract opening on the skin.
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
121
Fig. 2. Intraoperative photograph. (a) Branchial fistula tract. (b) Facial nerve. (c) External auditory canal.
Fig. 3. Intraoperative photograph. (a) External auditory canal defect. (b) Facial nerve.
neuromonitoring probe was performed during surgery in all of seven patients. The relationship between the cleft’s tract and the facial nerve was evaluated according to the location of the fistula opening, specifically whether it was above or below the mandibular angle. Two of the fistula tracts were located medially to the facial nerve, and both had fistula openings below the mandibular angle. In the remaining five patients, the tracts were lateral to the facial nerve; one had the fistula opening inferior to the mandibular angle, and three had fistula openings superior to the mandibular angle. The fifth patient had no external fistula opening, but only a postauricular bulge. The average follow-up period was 3.14 years (range, 1.00–4.00 years). There were no recurrences during that period, and no extra surgical intervention was required. Postoperatively, two patients developed mild narrowing of the external auricular canal, and one developed mild temporary paresis of the marginal branch of the facial nerve. One patient developed scar tissue on the cheek secondary to previous drainage.
1923 to 2000 that included a total of 158 cases. They reported that in younger patients and in patients presenting with fistulae, the lesions were located deeper than the facial nerve, and in patients in whom the lesions were connected to the external auditory canal or had sinus tracts, the lesions were located superficial to the facial nerve [7]. In two of our patients (28.5%), the tract was located deeper than the facial nerve, while in the remaining five (71.5%), it was located laterally. Of the 18 patients described by Magdy and Ashram as to have type 1 branchial cleft anomalies, only 9 had a type 1b anomaly. Of these nine patients, in three the tract was located deeper, in four it was superficial, and in two it was located between the branches of the facial nerve [9]. In a series of 41 cases exploring the relationship between branchial anomalies and the facial nerve, the sex and age of the patients and the side and location of the lesions were examined. However, no significant differences in these parameters were observed [10]. A review of the English-language medical literature yielded no other study examining the relationship between the fistula tracts/openings and the facial nerve. We explored our idea that the location of the fistula opening, whether superior or inferior to the mandibular angle, may be an indicator of the relationship between the branchial cleft and facial nerve. Although our findings do not support a generalized concept, larger series may reveal the presence of such a relationship. All of our patients were younger than 5 years. Some authors recommend that the most appropriate timing for surgery in patients with first branchial cleft anomalies is over 4 years of age. It is presumed that surgery will be easier after complete maturation of the facial nerve, with a decreased risk of probable injuries to the nerve [10,11]. However, we believe that delaying surgery may not be optimal because of the risk of recurrent infections, which may result in the formation of fibrotic and inflammatory tissues in the affected region, thus making it even more difficult to identify the facial nerve and increasing the risk of complications. Therefore, we advocate immediate surgery once the diagnosis has been established. The patients with type 1b branchial cleft anomalies described by Magdy and Ashram were older (4–17 years of age) than our patients [9]. The recommended surgical technique for first branchial cleft anomalies begins with the incision used for parotid tumor excisions and proceeds with careful exposure of the facial nerve
3. Discussion First branchial cleft anomalies originate from incomplete closure of the ectodermal part of the branchial cleft. Whether the defect presents as a fistula, sinus, or cyst depends on the degree of closure [8]. Almost all first branchial cleft anomalies are located at the triangular region surrounded by the tip of the chin, external auditory canal, and body of the hyoid bone. The relationship between these anomalies and the facial nerve is quite variable, with three main variants: with the branchial cleft fistula located deeper than the facial nerve, superficial to the facial nerve, or between the nerve’s branches. It is difficult to identify this relationship preoperatively. Recurrence after previous surgical procedures or infection in this region may lead to inflammatory adhesion in the surrounding tissues and further complicate the already demanding operation in a complex anatomical setting. Such difficulties may further increase the risk of injury to the facial nerve, negatively impacting the patient’s quality of life. We carefully reviewed the medical records of seven patients surgically treated during an 8-year period, looking for any data that would help us to predict the relationship between the branchial fistula tract and the facial nerve and thus decrease the risk of facial nerve injury. D’Souza et al. reviewed 55 studies published from
122
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
and subsequent mass resection, taking care to protect the nerve [12,13]. Although some authors recommend injecting methylene blue to display the fistula tract, we do not use such an approach because the dye may leak into neighboring tissues and obscure the surgical area. Due to the relationship between the facial nerve and cleft lesion, it is recommended that a partial or superficial parotidectomy be performed if the lesion is found to be deeply located after exposure of the facial nerve [13]. The incision and subsequent explorative surgery must clearly identify the relationship between the lesion and the facial nerve. 4. Conclusion Diagnosis of a first branchial cleft anomaly is rather difficult because such anomalies are quite rare. Misdiagnosing these lesions as tumors or various other inflammatory incidents may delay the correct diagnosis and lead to insufficient and/or erroneous treatments. Surgical treatment of such cases is difficult and carries a high risk due to the lesion’s proximity to the facial nerve. Neither physical examination nor imaging techniques can clearly define the relationship between the lesion and the facial nerve. We presume that the location of the fistula opening may be helpful in defining this relationship. Studies with larger series are needed to confirm this presumption, but due to the rarity of first branchial cleft anomalies, it will not be easy to gather a large number of cases for such a study. We believe that although our study did not involve many patients, it is a stepping-stone to further studies.
Conflict of interest All authors are required to declare any financial support or relationship that may pose a conflict of interest. References [1] Olsen KD, Maragos NE, Weiland LH. First branchial cleft anomalies. Laryngoscope 1980;90:423–36. [2] Al-Mahdi AH, Al-Khurri LE, Atto GZ, Dhaher A. Type II first branchial cleft anomaly. J Craniofac Surg 2013;24:1832–5. http://dx.doi.org/10.1097/SCS. 0b013e3182997e12. [3] Triglia JM, Nicollas R, Ducroz V, Koltai PJ, Garabedian EN. First branchial cleft anomalies: a study of 39 cases and a review of the literature. Arch Otolaryngol Head Neck Surg 1998;124:291–5. [4] Solares CA, Chan J, Koltai PJ. Anatomical variations of the facial nerve in first branchial cleft anomalies. Arch Otolaryngol Head Neck Surg 2003;129:351–5. [5] Work WP. Newer concepts of first branchial cleft defects. Laryngoscope 1972;82:1581–93. [6] Waldhausen JH. Branchial cleft and arch anomalies in children. Semin Pediatr Surg 2006;15:64–9. [7] D’Souza AR, Uppal HS, De R, Zeitoun H. Updating concepts of first branchial cleft defects: a literature review. Int J Pediatr Otorhinolaryngol 2002;62:103–9. [8] Carlson BM. Human embryology and developmental biology. St Louis: Mosby; 2004. 346. [9] Magdy EA, Ashram YA. First branchial cleft anomalies: presentation, variability and safe surgical management. Eur Arch Otorhinolaryngol 2013;270:1917–25. http://dx.doi.org/10.1007/s00405-012-2287-x [Epub ahead of print]. [10] Guo YX, Guo CB. Relation between a first branchial cleft anomaly and the facial nerve. Br J Oral Maxillofac Surg 2012;50:259–63. [11] May M, editor. The facial nerve. New York: Thieme; 1986. p. 574. [12] Jakubikova J, Stanik R, Stanikova A. Malformations of the first branchial cleft: duplication of the external auditory canal. Int J Pediatr Otorhinolaryngol 2005;69: 255–61. [13] Agaton-Bonilla FC, Gay-Escoda C. Diagnosis and treatment of branchial cleft cysts and fistulae: a retrospective study of 183 patients. Int J Oral Maxillofac Surg 1996;25:449–52.
Contents lists available at ScienceDirect
Auris Nasus Larynx journal homepage: www.elsevier.com/locate/anl
The relationship between the fistula tract and the facial nerve in type II first branchial cleft anomalies Burak Ertas a,*, Rıza Onder Gunaydin b, Omer Faruk Unal a a b
Department of Otorhinolaryngology, Acibadem University, Medical School, Maslak, Istanbul, Turkey Department of Otorhinolaryngology, Hacettepe University, Medical School, Sihhiye, Ankara, Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 30 March 2014 Accepted 22 August 2014 Available online 16 September 2014
Objective: To share our experience involving seven patients with type II first branchial cleft anomalies (hereafter, type II anomalies), to determine whether the location of the external fistula openings of the anomalies are associated with the location of the facial nerve tract, and elucidate the relationship between the location of the fistula opening and the facial nerve. Methods: The medical records of seven patients who underwent surgery from 2005 to 2013 for type II anomalies were retrospectively examined. The relationship between the fistula opening and the facial nerve was evaluated in each patient with respect to whether the fistula opening was superior or inferior to the mandibular angle. All patients underwent partial parotidectomy, facial nerve exposure, and total excision of the mass together with connection of a small cuff of the external auditory canal skin to the fistula tract. Results: The fistula tracts were located medially to the facial nerve in two patients, and both fistulae had openings inferior to the mandibular angle. The fistula tracts were located laterally to the facial nerve in the remaining five patients: one patient had no external opening, one had an opening inferior to the mandibular angle, and the remaining three had openings superior to the mandibular angle. Conclusion: Because type II anomalies are rare, their diagnosis is difficult. Surgery of such lesions is challenging and associated with a high risk due to their proximity to the facial nerve. We believe that the location of the fistula opening may help to identify the relationship between the anomalous lesion and facial nerve. Studies involving larger series of cases are needed to confirm our hypothesis; however, because of the rarity of this specific anomaly, it will not be easy to compile a large number of cases. We believe that our study will encourage further investigation on this subject. ß 2014 Published by Elsevier Ireland Ltd.
Keywords: Branchial anomaly Tract of branchial fistula Facial nerve
1. Introduction Type II first branchial cleft anomalies (hereafter, type II anomalies) constitute a group of rarely encountered congenital anomalies of the neck and head region. Such anomalies account for less than 8% of all branchial anomalies [1,2]. They are typically connected to the external auditory canal or manifest as cysts, sinuses, or fistulae with inflammatory openings on the neck [3]. During embryological development, five branchial arches can be identified, all of which traverse mesodermal arches that are separated by grooves or clefts. The first branchial cleft is unique in that it is the only cleft that does not become obliterated
* Corresponding author at: Department of Otorhinolaryngology, Acibadem University, Medical School, Buyukdere Cad. No: 40 Acibadem Maslak Hastanesi KBB Klinigi, Maslak, Istanbul, Turkey. Tel.: +90 2123044937; mobile: +90 5333265828. E-mail address: [email protected] (B. Ertas). http://dx.doi.org/10.1016/j.anl.2014.08.017 0385-8146/ß 2014 Published by Elsevier Ireland Ltd.
completely by the eighth week of gestation and persists as a definitive structure in the embryo, as it penetrates the underlying mesoderm toward the first pharyngeal pouch. The dorsal portion produces the external auditory meatus; the middle portion forms the cavum conchae; and the ventral portion disappears. The membrane at the bottom of the cleft becomes the outer layer of the tympanic membrane. The ventral portion of the first pharyngeal pouch evolves to form the eustachian tube. First branchial anomalies are an uncommon group of lesions that arise from incomplete closure of the ventral portion of the first branchial cleft. These anomalies are closely related to the parotid gland and have variable relations to the facial nerve. The relationship of a first cleft anomaly to the parotid gland has been demonstrated to be variable, presumably because of the later development of the gland. The fate of the first branchial apparatus is complete by the sixth or seventh week of development, which means that a first branchial anomaly would be formed by this period. The parotid gland, on the other hand, first appears during
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
120 Table 1 Summary of patients in the present clinical series. Patient number
Age in years
Sex
Previous surgery
Finding in external canal
Location of fistula opening (above or below mandibular angle)
Relationship with facial nerve
Surgery
Follow-up
Complications
1
4
F
Drainage
Opening at 6 o’clock
Below
Medial
2 years
Mild external canal narrowing
2
1
M
None
Above
Lateral
4 years
–
3
3
M
Drainage
Bulging posteriorly Bulging inferiorly
Parotidectomy incision: complete excision with partial external canal resection Same as above
Above
Lateral
Same as above
2 years
4 5
4 2
M F
None None
Below Below
Medial Lateral
Same as above Same as above
4 years 5 years
6
3
M
None
Above
Lateral
Same as above
2 months
7
2
M
Drainage Drainage of three abscesses Inadequate excision of fistula opening None
Mild external canal narrowing + scarring at the previous drainage site – Mild marginal paresis –
None
No opening; only a bulge behind auricle
Lateral to main truncus
Same as above
4 years
–
the sixth week of development. The facial nerve and its muscles migrate upward between the sixth and eighth weeks of development. It can be inferred from the embryological patterns that a first branchial cleft anomaly may be found superficial to, deep to, or within the branches of the facial nerve [4]. While type I lesions are of pure ectodermal origin and form cystic masses, type II anomalies may contain a mixture of tissues such as skin, cartilage, and adnexal structures originating from both ectodermal and mesodermal tissues [2,5,6]. They are frequently complicated with infections, causing significant morbidity. Surgical excision is the preferred mode of treatment, but accurate identification of the relationship between the cleft and facial nerve is of prime importance to avoid injury to the facial nerve [7]. A correct diagnosis is also important because misdiagnoses frequently lead to insufficient draining and partial excisions, thus inducing recurrence. In this retrospective study, we investigated the clinical features of seven patients with type II anomalies and examined the relationship between the location of the fistula opening and the facial nerve.
All patients had type II anomalies according to Work’s classification [5]. Six patients had fistulae and one had a cyst according to Olsen’s classification [1]. All patients were treated with various antibiotics before referral to our clinic. Five patients underwent no surgical intervention before referral. Two patients (28.5%) underwent previous surgery: cyst incision and draining in one patient and incomplete surgical resection in the other. Typical clinical symptoms were bulging or inflammation on the neck due to a cyst, sinus or fistula connection to the external auditory canal, and leakage after opening to the skin. All patients underwent diagnostic neck ultrasonography. Histopathological findings revealed that all patients had branchial cleft anomalies. The mass was excised in a single-phase surgery in all patients. Following creation of a modified Blair incision, the facial nerve tract was exposed during partial parotidectomy, and a small cuff of external auditory canal skin related to the fistula tract plus peripheral inflammatory tissues were excised together with the epithelial tract (Figs. 1–3, belong to the first patient described in Table 1). The neuromonitoring of facial nerves using a Xomed
2. Patients and methods The complete medical records of seven patients who were surgically treated for type II anomalies from July 2005 to 2013 in our otorhinolaryngology clinic were retrospectively evaluated. Magnetic resonance imaging, operational notes, histopathological findings, and all records related to diagnosis and therapy were reviewed. All data revealing any relationship between the facial nerve and the branchial cleft were duly analyzed. Patients’ records are summarized in Table 1. Two and five patients were female and male, respectively, with a mean age of 2.7 years at the time of intervention. Five lesions were located on the right and two were on the left. Fistula openings were located over the mandibular angle in three patients and below the mandibular angle in three patients; one patient had no external fistula opening. There were no other pathologic findings in the patients’ ear, throat, or nose in head and neck examinations. Their general physical examinations also revealed no pathologic findings.
Fig. 1. Intraoperative photograph. (a) Fistula tract opening on the skin.
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
121
Fig. 2. Intraoperative photograph. (a) Branchial fistula tract. (b) Facial nerve. (c) External auditory canal.
Fig. 3. Intraoperative photograph. (a) External auditory canal defect. (b) Facial nerve.
neuromonitoring probe was performed during surgery in all of seven patients. The relationship between the cleft’s tract and the facial nerve was evaluated according to the location of the fistula opening, specifically whether it was above or below the mandibular angle. Two of the fistula tracts were located medially to the facial nerve, and both had fistula openings below the mandibular angle. In the remaining five patients, the tracts were lateral to the facial nerve; one had the fistula opening inferior to the mandibular angle, and three had fistula openings superior to the mandibular angle. The fifth patient had no external fistula opening, but only a postauricular bulge. The average follow-up period was 3.14 years (range, 1.00–4.00 years). There were no recurrences during that period, and no extra surgical intervention was required. Postoperatively, two patients developed mild narrowing of the external auricular canal, and one developed mild temporary paresis of the marginal branch of the facial nerve. One patient developed scar tissue on the cheek secondary to previous drainage.
1923 to 2000 that included a total of 158 cases. They reported that in younger patients and in patients presenting with fistulae, the lesions were located deeper than the facial nerve, and in patients in whom the lesions were connected to the external auditory canal or had sinus tracts, the lesions were located superficial to the facial nerve [7]. In two of our patients (28.5%), the tract was located deeper than the facial nerve, while in the remaining five (71.5%), it was located laterally. Of the 18 patients described by Magdy and Ashram as to have type 1 branchial cleft anomalies, only 9 had a type 1b anomaly. Of these nine patients, in three the tract was located deeper, in four it was superficial, and in two it was located between the branches of the facial nerve [9]. In a series of 41 cases exploring the relationship between branchial anomalies and the facial nerve, the sex and age of the patients and the side and location of the lesions were examined. However, no significant differences in these parameters were observed [10]. A review of the English-language medical literature yielded no other study examining the relationship between the fistula tracts/openings and the facial nerve. We explored our idea that the location of the fistula opening, whether superior or inferior to the mandibular angle, may be an indicator of the relationship between the branchial cleft and facial nerve. Although our findings do not support a generalized concept, larger series may reveal the presence of such a relationship. All of our patients were younger than 5 years. Some authors recommend that the most appropriate timing for surgery in patients with first branchial cleft anomalies is over 4 years of age. It is presumed that surgery will be easier after complete maturation of the facial nerve, with a decreased risk of probable injuries to the nerve [10,11]. However, we believe that delaying surgery may not be optimal because of the risk of recurrent infections, which may result in the formation of fibrotic and inflammatory tissues in the affected region, thus making it even more difficult to identify the facial nerve and increasing the risk of complications. Therefore, we advocate immediate surgery once the diagnosis has been established. The patients with type 1b branchial cleft anomalies described by Magdy and Ashram were older (4–17 years of age) than our patients [9]. The recommended surgical technique for first branchial cleft anomalies begins with the incision used for parotid tumor excisions and proceeds with careful exposure of the facial nerve
3. Discussion First branchial cleft anomalies originate from incomplete closure of the ectodermal part of the branchial cleft. Whether the defect presents as a fistula, sinus, or cyst depends on the degree of closure [8]. Almost all first branchial cleft anomalies are located at the triangular region surrounded by the tip of the chin, external auditory canal, and body of the hyoid bone. The relationship between these anomalies and the facial nerve is quite variable, with three main variants: with the branchial cleft fistula located deeper than the facial nerve, superficial to the facial nerve, or between the nerve’s branches. It is difficult to identify this relationship preoperatively. Recurrence after previous surgical procedures or infection in this region may lead to inflammatory adhesion in the surrounding tissues and further complicate the already demanding operation in a complex anatomical setting. Such difficulties may further increase the risk of injury to the facial nerve, negatively impacting the patient’s quality of life. We carefully reviewed the medical records of seven patients surgically treated during an 8-year period, looking for any data that would help us to predict the relationship between the branchial fistula tract and the facial nerve and thus decrease the risk of facial nerve injury. D’Souza et al. reviewed 55 studies published from
122
B. Ertas et al. / Auris Nasus Larynx 42 (2015) 119–122
and subsequent mass resection, taking care to protect the nerve [12,13]. Although some authors recommend injecting methylene blue to display the fistula tract, we do not use such an approach because the dye may leak into neighboring tissues and obscure the surgical area. Due to the relationship between the facial nerve and cleft lesion, it is recommended that a partial or superficial parotidectomy be performed if the lesion is found to be deeply located after exposure of the facial nerve [13]. The incision and subsequent explorative surgery must clearly identify the relationship between the lesion and the facial nerve. 4. Conclusion Diagnosis of a first branchial cleft anomaly is rather difficult because such anomalies are quite rare. Misdiagnosing these lesions as tumors or various other inflammatory incidents may delay the correct diagnosis and lead to insufficient and/or erroneous treatments. Surgical treatment of such cases is difficult and carries a high risk due to the lesion’s proximity to the facial nerve. Neither physical examination nor imaging techniques can clearly define the relationship between the lesion and the facial nerve. We presume that the location of the fistula opening may be helpful in defining this relationship. Studies with larger series are needed to confirm this presumption, but due to the rarity of first branchial cleft anomalies, it will not be easy to gather a large number of cases for such a study. We believe that although our study did not involve many patients, it is a stepping-stone to further studies.
Conflict of interest All authors are required to declare any financial support or relationship that may pose a conflict of interest. References [1] Olsen KD, Maragos NE, Weiland LH. First branchial cleft anomalies. Laryngoscope 1980;90:423–36. [2] Al-Mahdi AH, Al-Khurri LE, Atto GZ, Dhaher A. Type II first branchial cleft anomaly. J Craniofac Surg 2013;24:1832–5. http://dx.doi.org/10.1097/SCS. 0b013e3182997e12. [3] Triglia JM, Nicollas R, Ducroz V, Koltai PJ, Garabedian EN. First branchial cleft anomalies: a study of 39 cases and a review of the literature. Arch Otolaryngol Head Neck Surg 1998;124:291–5. [4] Solares CA, Chan J, Koltai PJ. Anatomical variations of the facial nerve in first branchial cleft anomalies. Arch Otolaryngol Head Neck Surg 2003;129:351–5. [5] Work WP. Newer concepts of first branchial cleft defects. Laryngoscope 1972;82:1581–93. [6] Waldhausen JH. Branchial cleft and arch anomalies in children. Semin Pediatr Surg 2006;15:64–9. [7] D’Souza AR, Uppal HS, De R, Zeitoun H. Updating concepts of first branchial cleft defects: a literature review. Int J Pediatr Otorhinolaryngol 2002;62:103–9. [8] Carlson BM. Human embryology and developmental biology. St Louis: Mosby; 2004. 346. [9] Magdy EA, Ashram YA. First branchial cleft anomalies: presentation, variability and safe surgical management. Eur Arch Otorhinolaryngol 2013;270:1917–25. http://dx.doi.org/10.1007/s00405-012-2287-x [Epub ahead of print]. [10] Guo YX, Guo CB. Relation between a first branchial cleft anomaly and the facial nerve. Br J Oral Maxillofac Surg 2012;50:259–63. [11] May M, editor. The facial nerve. New York: Thieme; 1986. p. 574. [12] Jakubikova J, Stanik R, Stanikova A. Malformations of the first branchial cleft: duplication of the external auditory canal. Int J Pediatr Otorhinolaryngol 2005;69: 255–61. [13] Agaton-Bonilla FC, Gay-Escoda C. Diagnosis and treatment of branchial cleft cysts and fistulae: a retrospective study of 183 patients. Int J Oral Maxillofac Surg 1996;25:449–52.
