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Second branchial cleft anomalies
Operative Techniques in Otolaryngology (2017) ], ]]]–]]]
Second branchial cleft anomalies Nathan C. Grohmann, MD, Heather C. Herrington, MD From the Division of Otolaryngology, University of Vermont Medical Center, Burlington, Vermont KEYWORDS Branchial apparatus; branchial cleft cyst
Second branchial anomalies represent the most common type of branchial anomalies. Their anatomical course is well understood based on knowledge of the embryologic branchial apparatus. These anomalies tend to present in children, though slow-growing cysts may first be noted well into adulthood. The most common presenting sign is a slowly growing lateral neck mass. Surgical excision remains the mainstay of therapy. r 2017 Elsevier Inc. All rights reserved.
Introduction
Histology
Second branchial anomalies include cysts, sinuses, and fistulae. They represent the most common branchial anomalies. Understanding the embryology and resulting anatomy of these anomalous structures can allow one to predict their course and implement safe, effective surgical interventions with low recurrence rates. Second branchial cleft anomalies commonly occur along the anterior border of the sternocleidomastoid muscle. Sinus and fistulous tracts of second, third, and fourth branchial anomalies all exit near one another, owing to their common embryologic origin in the cervical sinus of His. Second branchial anomaly tracts course between the external and internal (derived from the third arch) carotid arteries, superior to the glossopharyngeal and hypoglossal nerves, and finally end in the tonsillar fossa, usually along the posterior pillar. Some authors have further subclassified second branchial anomalies based on minor anatomical variations of the tract,1 but these classifications have not been broadly applied.
Squamous epithelium, pseudostratified columnar (respiratory) epithelium, and combinations thereof have been reported to line second branchial lesions. Lymphoid, sebaceous, and salivary tissues have also been associated with second branchial anomalies. Cysts are more commonly lined by squamous epithelium, whereas sinuses and fistulae tend to be lined by ciliated, columnar epithelium.1 Cholesterol crystals may be seen in cystic fluid.
Address reprint requests and correspondence: Nathan C. Grohmann, MD, Division of Otolaryngology, University of Vermont Medical Center, 111 Colchester Ave, WP4 ENT Clinic, Burlington, VT 05401. E-mail address: [email protected] http://dx.doi.org/10.1016/j.otot.2017.05.013 1043-1810/r 2017 Elsevier Inc. All rights reserved.
Presentation Generally, second branchial cleft cysts present as nontender, stable-to-slowly-enlarging lateral neck masses that may acutely increase in size during an upper respiratory tract infection, leading occasionally to respiratory compromise, torticollis, and dysphagia. Fistulae and sinuses are usually diagnosed earlier than cysts, owing to the more obvious presence of a skin opening with chronic drainage along the anterior border of the sternocleidomastoid. In a review of 52 patients, the 3 most common presenting symptoms of branchial anomalies were drainage from a cervical sinus opening, neck mass, and repeated infection.2 Second branchial anomalies are the most common and have been reported to account for 95% of all branchial anomalies.1 However, a more recent review of 74 branchial anomalies found that second branchial anomalies accounted for 69%.3 Reviews of the proportion of cysts, sinuses, and
2 fistulas have also varied widely. One review of mostly adults noted cysts accounting for approximately 80% of anomalies,4 whereas another review of children found sinuses, fistulae, and cysts accounting for 45%, 27%, and 27%, respectively.3 Another review of pediatric patients also found sinuses were more common.2 Perhaps this discrepancy relates to the fact that cysts often are found much later in life than sinuses and fistulae, as it can take many years for cysts to enlarge and become noticeable. In a retrospective review of 74 branchial anomalies, the average age of presentation for a cyst was 4.1 years, 3.6 years for a sinus, and 2.6 years for a fistula.3 Approximately one-third of second branchial anomalies had been previously infected before diagnosis. Of that third, 71% of patients had more than 1 infection. Of the anomalies with a skin opening, 85% presented with drainage and 35% presented with a history of infection. For those with both a lateral neck skin opening and drainage, 59% were ultimately diagnosed as a sinus and 37% as a fistula. Sinuses and fistulas were more likely to present with infection (67%) compared with cysts (21%). Previous reviews, including a relatively large Spanish series, have noted that second branchial anomalies are seen more commonly in women, with a female to male ratio of 2:1 or even higher.4 However, this female predominance has not been duplicated in later studies.3,5 There also appears to be no predilection for right or left laterality.
Evaluation The diagnosis of a second branchial anomaly can be straightforward. A careful history and physical examination, as well as a high index of suspicion and clinical awareness, are all important factors in establishing a diagnosis. Any unexplained neck mass, recurrent neck infection, or abscess should be suggestive of a possible branchial anomaly. Although not exhaustive, the differential diagnosis of second branchial anomalies includes reactive lymphadenopathy, lymphadenitis, abscess, lipoma, lymphatic or vascular malformation, lymphoma, ectopic thyroid and thyroglossal duct cysts, tuberculosis adenitis, and cervical thymic cysts.3 In adults, it is always important to exclude a cystic cervical lymph node metastasis by fine needle aspiration to avoid surgical misadventure. Clinical examination alone has been reported as 50%-60% accurate when used to diagnose a second branchial anomaly.6,7 Consequently, several authors have examined the role of preoperative imaging in establishing an accurate diagnosis. Computed tomography scans with and without fistulography, magnetic resonance imaging, and ultrasound have all been employed in the workup of second branchial anomalies. Computed tomography is the beststudied imaging modality, and preoperative diagnosis of branchial anomalies has a reported accuracy rate as high as 93%.3 Ultrasound can be a useful tool in preoperative evaluation due to its low cost, absence of ionizing radiation, and ability to be performed in an office setting. A large
Operative Techniques in Otolaryngology, Vol ], No ], ] 2017 review from China found that the diagnostic accuracy of ultrasound was 66.2%.8 Use of magnetic resonance imaging in the evaluation of second branchial anomalies has been described in case reports as accurate, though not necessarily practical due to its expense and possible need for sedation in young children.9 Routine laboratory testing is not required in the workup of most patients with second branchial anomalies. Most often, second branchial cleft anomalies are not associated with additional anomalies. However, one syndrome is worth noting. Branchiootorenal syndrome, an autosomal dominant disorder with variable expressivity, is characterized by hearing loss (conductive, sensorineural, or mixed), ear defects (outer, middle, and inner have all been described), branchial anomalies, and renal abnormalities.
Treatment options Surgical excision has been shown to be safe and effective for addressing second branchial anomalies and remains the mainstay of treatment. However, several studies have trialed sclerotherapy as an alternative to surgery in branchial cleft cysts. Kim et al10 injected OK-432, a lyophilized mixture of low-virulence Group A Streptococcus pyogenes and penicillin G potassium, directly into branchial cleft cysts in a prospective study of 23 patients. Fourteen of 18 patients with unilocular cysts experienced complete regression. Of the 5 patients with multilocular cysts, 2 showed a partial response and 3 showed no response to OK-432 sclerotherapy. Side effects of OK-432 therapy are noted to be minor and include mild fever, bloody drainage, and temporary injection site pain. Based on the best available evidence, sclerotherapy is not the standard of care and should only be offered to patients with unilocular branchial cleft cysts.
Indications for surgery Once discovered, surgical excision of second branchial anomalies is almost uniformly recommended given the propensity of recurrent infection and cosmetic deformity associated with these lesions. Generally, surgical excision is delayed until a child is at least 3 months old. Surgical excision should not be performed during an acute infection. Rather, one should allow adequate time for the infection and associated inflammation to subside with appropriate antimicrobial therapy before proceeding with surgical excision, typically 2-4 weeks after resolution of the acute infection.
Details of surgical technique Before incision, the patient should be placed supine on the operating table with the neck extended by placement of a small shoulder roll. If it is known that a fistulous tract is present, then preoperative dosing with an antibiotic to cover oral cavity microbes such as ampicillin/sulbactam or
Grohmann and Herrington
Second Branchial Anomalies
Figure 1 Catheter within tract showing anatomical relationship of tract to vital structures.
clindamycin is preferable. If a cyst is present and no communication with the oropharynx is suspected, then a preoperative antibiotic such as cefazolin to cover skin flora is appropriate. If an external skin opening is present, the period of time between induction and skin preparation provides an ideal opportunity to further delineate the extent of the anomaly if incompletely or not previously demonstrated by preoperative imaging studies. General anesthesia will allow for muscle relaxation that may make probing of the lesion easier. It may be possible to cannulate the tract with a 2-0 monofilament suture, arterial line catheter, ophthalmic probe, 5-Fr pediatric suction catheter, or a similarly small, nonpenetrating catheter. We prefer to use a 5-Fr pediatric suction catheter, as sterile saline can be injected through the catheter, allowing it to more easily “float” through the sinus or fistulous tract. After induction of anesthesia, an external skin opening may also be injected with methylene blue via angiocatheter to further delineate the fistulous or sinus tract. We prefer to place an adhesive dressing such as a Tegaderm over the skin opening to avoid skin staining with methylene blue. Presence of methylene blue in the oropharynx can also definitively confirm the presence of a fistula. A direct laryngoscopy can also be considered to evaluate for a peritonsillar sinus or fistula tract opening.
3 Second branchial anomalies are most often approached surgically via a lateral transverse neck incision. If a skin opening is present, the incision should include an ellipse of skin encompassing the opening (Figure 1). Otherwise, the incision is placed over the cyst, typically at least 2-3 cm below the body of the mandible to avoid injury to the marginal mandibular branch of the facial nerve. Dissection proceeds through the platysma muscle and subplatysmal flaps are elevated superiorly toward the body of the mandible and inferiorly toward the inferior aspect of the cyst. Cysts may be found both superficial and deep to the deep cervical fascia. Consequently, careful dissection is necessary to ensure complete removal. The anterior border of the sternocleidomastoid muscle is identified and delineated so that it may be retracted laterally. Next, the carotid sheath is identified medial to the body of the sternocleidomastoid muscle. The tract is bluntly dissected from the surrounding structures as it courses between the internal and external carotid arteries and superior to the glossopharyngeal and hypoglossal nerves. As the dissection continues superiorly, it may be necessary to use a second stepladder incision (Figure 2). Otherwise, one could extend the incision posterosuperiorly toward the mastoid using a “hockey stick” incision to gain additional access. More recently, endoscope-assisted second branchial cleft cyst excision via small lateral cervical11 and retroauricular hairline12 approaches have been proposed and may eventually become more popular given the possibility of improved cosmesis. Careful dissection must be undertaken to avoid injury to cranial nerves 10, 11, and 12, though nerve monitoring is usually not necessary. Once the proximal opening into the
Figure 2
Stepladder incision for surgical excision.
Operative Techniques in Otolaryngology, Vol ], No ], ] 2017
4
of the patient to the hospital, comorbidities, and surgeon preference.
Complications
Figure 3
Oropharyngeal view of tract.
tonsillar fossa is identified superiorly, the tract can be ligated and divided (Figure 3). One must avoid premature transection of the tract, as it can be extremely difficult to find again. To avoid the aforementioned situation in the case of a fistula or sinus tract, we have found it helpful to place a 5 Fr suction catheter through the skin opening into the tract and secure it in place with a 2-0 silk suture ligature. The catheter tends to provide a semirigid structure that aids in dissection. In the case of a fistula, the catheter can be seen entering the oropharynx, a small cuff of tissue around the catheter can be incised intraorally, and the catheter and entire fistulous tract can be delivered through the mouth. The oropharyngeal wound should then be closed with interrupted, absorbable sutures. Excision of most second branchial anomalies is relatively avascular, and use of a drain postoperatively is based on the extent of dissection and surgeon preference. We generally close the incision in multiple layers; the platysma and dermis are closed with absorbable monofilament suture and the skin with topical glue.
Surgical excision is an effective means of dealing with second branchial anomalies with recurrence rates noted later. Major complications are rare. Complications of second branchial anomaly surgery that do not require surgical intervention include minor wound infections, seromas, and stitch extrusion. Hypertrophic scars and keloids have been reported, as with any neck incision. Hypoglossal and superior laryngeal nerve injury, most often temporary, have also been reported.4 Although seemingly straightforward, misdiagnosis can occur in 15% of cases or more.13 Initial correct diagnosis is crucial because prior evidence shows that the recurrence rates after surgical excision of branchial anomalies are 14% and 22% with previous infection and surgery, respectively, whereas the recurrence rate for a primary lesion is 3%.14 Because sinuses and fistulae are more likely to become infected preoperatively than cysts (67% vs 21%, respectively),3 they are also more likely to recur. Additionally, absence of an epithelial lining identified at the time of original excision is associated with a much higher risk of recurrence.3
Conclusions Second branchial anomalies, including cysts, sinuses, and fistulae are relatively common congenital neck lesions, representing approximately 69%-95% of all branchial anomalies. Sinuses and fistulae are generally discovered earlier and are more likely to become infected and recur than cysts. Cysts most commonly present as painless, slowly enlarging lateral neck masses, whereas fistulae and sinuses often present with draining skin openings anterior to the sternocleidomastoid. Complete surgical excision is generally curative and carries a low risk of complications, though recurrence is more likely to occur in previously infected lesions.
Disclosure Postoperative care Postoperative care for most patients after second branchial anomaly excision is generally not complex. In patients with anomalies that extend to the pharynx requiring excision of tissue from the tonsillar fossae, oral feeding can resume safely within the immediate postoperative period, though a soft diet may be better tolerated during the first few days after surgery. Postoperative antibiotics are generally not recommended. Overnight monitoring is not required, assuming no injury to surrounding neurovascular structures; however, this decision is based on patient age, whether or not a neck drain was placed, caregiver reliability, proximity
The authors reported no proprietary or commercial interest in any product mentioned or concept discussed in this article.
References 1. Waldhausen JH: Branchial cleft and arch anomalies in children. Semin Pediatr Surg 15(2):64-69, 2006 2. Choi SS, Zalzal GH: Branchial anomalies: A review of 52 cases. Laryngoscope 105(9, pt 1):909-913, 1995 3. Schroeder JW Jr., Mohyuddin N, Maddalozzo J: Branchial anomalies in the pediatric population. Otolaryngol Head Neck Surg 137(2): 289-295, 2007
Grohmann and Herrington
Second Branchial Anomalies
4. 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 25(6):449-452, 1996 5. Prasad Sampath Chandra, Azeez Arun, Thada Nikhil Dinaker, et al: Branchial anomalies: Diagnosis and management. Int J Otolaryngol 2014, [9 pp, article ID 237015], 2014 6. Neel HB, Pemberton J: Lateral cervical cysts and fistulas. Surgery 18:267-286, 1945 7. Shedden WM: Branchial cysts and fistulas. N Engl J Med 205:800-811, 1931 8. Hu YJ, Li YD, Qu XZ, et al: Clinical analysis of branchial cleft cyst (fistula): Report of 284 cases. Shanghai Kou Qiang Yi Xue 17 (5):461-464, 2008 9. Black CJ, O'Hara JT, Berry J, et al: Magnetic resonance imaging of branchial cleft abnormalities: Illustrated cases and literature review. J Laryngol Otol 124(2):213-215, 2010
5 10. Kim MG, Lee NH, Ban JH, et al: Sclerotherapy of branchial cleft cysts using OK-432. Otolaryngol Head Neck Surg 141(3):329-334, 2009 11. Chen J, Chen W, Zhang J, et al: Endoscope-assisted second branchial cleft cyst resection via an incision along skin line on lateral neck. Eur Arch Otorhinolaryngol 271(10):2789-2793, 2014 12. Chen L, Huang X, Lou X, et al: A comparison between endoscopicassisted second branchial cleft cyst resection via retroauricular hairline approach and conventional second branchial cleft cyst resection. Lin Chung Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 27(22):1258-1262, 2013 13. Kenealy JF, Torsiglieri AJ Jr., Tom LW: Branchial cleft anomalies: A five-year retrospective review. Trans Pa Acad Ophthalmol Otolaryngol 42:1022-1025, 1990 14. Reiter D: Third branchial cleft sinus: An unusual cause of neck abscess. Int J Pediatr Otorhinolaryngol 4(2):181-186, 1982
Second branchial cleft anomalies Nathan C. Grohmann, MD, Heather C. Herrington, MD From the Division of Otolaryngology, University of Vermont Medical Center, Burlington, Vermont KEYWORDS Branchial apparatus; branchial cleft cyst
Second branchial anomalies represent the most common type of branchial anomalies. Their anatomical course is well understood based on knowledge of the embryologic branchial apparatus. These anomalies tend to present in children, though slow-growing cysts may first be noted well into adulthood. The most common presenting sign is a slowly growing lateral neck mass. Surgical excision remains the mainstay of therapy. r 2017 Elsevier Inc. All rights reserved.
Introduction
Histology
Second branchial anomalies include cysts, sinuses, and fistulae. They represent the most common branchial anomalies. Understanding the embryology and resulting anatomy of these anomalous structures can allow one to predict their course and implement safe, effective surgical interventions with low recurrence rates. Second branchial cleft anomalies commonly occur along the anterior border of the sternocleidomastoid muscle. Sinus and fistulous tracts of second, third, and fourth branchial anomalies all exit near one another, owing to their common embryologic origin in the cervical sinus of His. Second branchial anomaly tracts course between the external and internal (derived from the third arch) carotid arteries, superior to the glossopharyngeal and hypoglossal nerves, and finally end in the tonsillar fossa, usually along the posterior pillar. Some authors have further subclassified second branchial anomalies based on minor anatomical variations of the tract,1 but these classifications have not been broadly applied.
Squamous epithelium, pseudostratified columnar (respiratory) epithelium, and combinations thereof have been reported to line second branchial lesions. Lymphoid, sebaceous, and salivary tissues have also been associated with second branchial anomalies. Cysts are more commonly lined by squamous epithelium, whereas sinuses and fistulae tend to be lined by ciliated, columnar epithelium.1 Cholesterol crystals may be seen in cystic fluid.
Address reprint requests and correspondence: Nathan C. Grohmann, MD, Division of Otolaryngology, University of Vermont Medical Center, 111 Colchester Ave, WP4 ENT Clinic, Burlington, VT 05401. E-mail address: [email protected] http://dx.doi.org/10.1016/j.otot.2017.05.013 1043-1810/r 2017 Elsevier Inc. All rights reserved.
Presentation Generally, second branchial cleft cysts present as nontender, stable-to-slowly-enlarging lateral neck masses that may acutely increase in size during an upper respiratory tract infection, leading occasionally to respiratory compromise, torticollis, and dysphagia. Fistulae and sinuses are usually diagnosed earlier than cysts, owing to the more obvious presence of a skin opening with chronic drainage along the anterior border of the sternocleidomastoid. In a review of 52 patients, the 3 most common presenting symptoms of branchial anomalies were drainage from a cervical sinus opening, neck mass, and repeated infection.2 Second branchial anomalies are the most common and have been reported to account for 95% of all branchial anomalies.1 However, a more recent review of 74 branchial anomalies found that second branchial anomalies accounted for 69%.3 Reviews of the proportion of cysts, sinuses, and
2 fistulas have also varied widely. One review of mostly adults noted cysts accounting for approximately 80% of anomalies,4 whereas another review of children found sinuses, fistulae, and cysts accounting for 45%, 27%, and 27%, respectively.3 Another review of pediatric patients also found sinuses were more common.2 Perhaps this discrepancy relates to the fact that cysts often are found much later in life than sinuses and fistulae, as it can take many years for cysts to enlarge and become noticeable. In a retrospective review of 74 branchial anomalies, the average age of presentation for a cyst was 4.1 years, 3.6 years for a sinus, and 2.6 years for a fistula.3 Approximately one-third of second branchial anomalies had been previously infected before diagnosis. Of that third, 71% of patients had more than 1 infection. Of the anomalies with a skin opening, 85% presented with drainage and 35% presented with a history of infection. For those with both a lateral neck skin opening and drainage, 59% were ultimately diagnosed as a sinus and 37% as a fistula. Sinuses and fistulas were more likely to present with infection (67%) compared with cysts (21%). Previous reviews, including a relatively large Spanish series, have noted that second branchial anomalies are seen more commonly in women, with a female to male ratio of 2:1 or even higher.4 However, this female predominance has not been duplicated in later studies.3,5 There also appears to be no predilection for right or left laterality.
Evaluation The diagnosis of a second branchial anomaly can be straightforward. A careful history and physical examination, as well as a high index of suspicion and clinical awareness, are all important factors in establishing a diagnosis. Any unexplained neck mass, recurrent neck infection, or abscess should be suggestive of a possible branchial anomaly. Although not exhaustive, the differential diagnosis of second branchial anomalies includes reactive lymphadenopathy, lymphadenitis, abscess, lipoma, lymphatic or vascular malformation, lymphoma, ectopic thyroid and thyroglossal duct cysts, tuberculosis adenitis, and cervical thymic cysts.3 In adults, it is always important to exclude a cystic cervical lymph node metastasis by fine needle aspiration to avoid surgical misadventure. Clinical examination alone has been reported as 50%-60% accurate when used to diagnose a second branchial anomaly.6,7 Consequently, several authors have examined the role of preoperative imaging in establishing an accurate diagnosis. Computed tomography scans with and without fistulography, magnetic resonance imaging, and ultrasound have all been employed in the workup of second branchial anomalies. Computed tomography is the beststudied imaging modality, and preoperative diagnosis of branchial anomalies has a reported accuracy rate as high as 93%.3 Ultrasound can be a useful tool in preoperative evaluation due to its low cost, absence of ionizing radiation, and ability to be performed in an office setting. A large
Operative Techniques in Otolaryngology, Vol ], No ], ] 2017 review from China found that the diagnostic accuracy of ultrasound was 66.2%.8 Use of magnetic resonance imaging in the evaluation of second branchial anomalies has been described in case reports as accurate, though not necessarily practical due to its expense and possible need for sedation in young children.9 Routine laboratory testing is not required in the workup of most patients with second branchial anomalies. Most often, second branchial cleft anomalies are not associated with additional anomalies. However, one syndrome is worth noting. Branchiootorenal syndrome, an autosomal dominant disorder with variable expressivity, is characterized by hearing loss (conductive, sensorineural, or mixed), ear defects (outer, middle, and inner have all been described), branchial anomalies, and renal abnormalities.
Treatment options Surgical excision has been shown to be safe and effective for addressing second branchial anomalies and remains the mainstay of treatment. However, several studies have trialed sclerotherapy as an alternative to surgery in branchial cleft cysts. Kim et al10 injected OK-432, a lyophilized mixture of low-virulence Group A Streptococcus pyogenes and penicillin G potassium, directly into branchial cleft cysts in a prospective study of 23 patients. Fourteen of 18 patients with unilocular cysts experienced complete regression. Of the 5 patients with multilocular cysts, 2 showed a partial response and 3 showed no response to OK-432 sclerotherapy. Side effects of OK-432 therapy are noted to be minor and include mild fever, bloody drainage, and temporary injection site pain. Based on the best available evidence, sclerotherapy is not the standard of care and should only be offered to patients with unilocular branchial cleft cysts.
Indications for surgery Once discovered, surgical excision of second branchial anomalies is almost uniformly recommended given the propensity of recurrent infection and cosmetic deformity associated with these lesions. Generally, surgical excision is delayed until a child is at least 3 months old. Surgical excision should not be performed during an acute infection. Rather, one should allow adequate time for the infection and associated inflammation to subside with appropriate antimicrobial therapy before proceeding with surgical excision, typically 2-4 weeks after resolution of the acute infection.
Details of surgical technique Before incision, the patient should be placed supine on the operating table with the neck extended by placement of a small shoulder roll. If it is known that a fistulous tract is present, then preoperative dosing with an antibiotic to cover oral cavity microbes such as ampicillin/sulbactam or
Grohmann and Herrington
Second Branchial Anomalies
Figure 1 Catheter within tract showing anatomical relationship of tract to vital structures.
clindamycin is preferable. If a cyst is present and no communication with the oropharynx is suspected, then a preoperative antibiotic such as cefazolin to cover skin flora is appropriate. If an external skin opening is present, the period of time between induction and skin preparation provides an ideal opportunity to further delineate the extent of the anomaly if incompletely or not previously demonstrated by preoperative imaging studies. General anesthesia will allow for muscle relaxation that may make probing of the lesion easier. It may be possible to cannulate the tract with a 2-0 monofilament suture, arterial line catheter, ophthalmic probe, 5-Fr pediatric suction catheter, or a similarly small, nonpenetrating catheter. We prefer to use a 5-Fr pediatric suction catheter, as sterile saline can be injected through the catheter, allowing it to more easily “float” through the sinus or fistulous tract. After induction of anesthesia, an external skin opening may also be injected with methylene blue via angiocatheter to further delineate the fistulous or sinus tract. We prefer to place an adhesive dressing such as a Tegaderm over the skin opening to avoid skin staining with methylene blue. Presence of methylene blue in the oropharynx can also definitively confirm the presence of a fistula. A direct laryngoscopy can also be considered to evaluate for a peritonsillar sinus or fistula tract opening.
3 Second branchial anomalies are most often approached surgically via a lateral transverse neck incision. If a skin opening is present, the incision should include an ellipse of skin encompassing the opening (Figure 1). Otherwise, the incision is placed over the cyst, typically at least 2-3 cm below the body of the mandible to avoid injury to the marginal mandibular branch of the facial nerve. Dissection proceeds through the platysma muscle and subplatysmal flaps are elevated superiorly toward the body of the mandible and inferiorly toward the inferior aspect of the cyst. Cysts may be found both superficial and deep to the deep cervical fascia. Consequently, careful dissection is necessary to ensure complete removal. The anterior border of the sternocleidomastoid muscle is identified and delineated so that it may be retracted laterally. Next, the carotid sheath is identified medial to the body of the sternocleidomastoid muscle. The tract is bluntly dissected from the surrounding structures as it courses between the internal and external carotid arteries and superior to the glossopharyngeal and hypoglossal nerves. As the dissection continues superiorly, it may be necessary to use a second stepladder incision (Figure 2). Otherwise, one could extend the incision posterosuperiorly toward the mastoid using a “hockey stick” incision to gain additional access. More recently, endoscope-assisted second branchial cleft cyst excision via small lateral cervical11 and retroauricular hairline12 approaches have been proposed and may eventually become more popular given the possibility of improved cosmesis. Careful dissection must be undertaken to avoid injury to cranial nerves 10, 11, and 12, though nerve monitoring is usually not necessary. Once the proximal opening into the
Figure 2
Stepladder incision for surgical excision.
Operative Techniques in Otolaryngology, Vol ], No ], ] 2017
4
of the patient to the hospital, comorbidities, and surgeon preference.
Complications
Figure 3
Oropharyngeal view of tract.
tonsillar fossa is identified superiorly, the tract can be ligated and divided (Figure 3). One must avoid premature transection of the tract, as it can be extremely difficult to find again. To avoid the aforementioned situation in the case of a fistula or sinus tract, we have found it helpful to place a 5 Fr suction catheter through the skin opening into the tract and secure it in place with a 2-0 silk suture ligature. The catheter tends to provide a semirigid structure that aids in dissection. In the case of a fistula, the catheter can be seen entering the oropharynx, a small cuff of tissue around the catheter can be incised intraorally, and the catheter and entire fistulous tract can be delivered through the mouth. The oropharyngeal wound should then be closed with interrupted, absorbable sutures. Excision of most second branchial anomalies is relatively avascular, and use of a drain postoperatively is based on the extent of dissection and surgeon preference. We generally close the incision in multiple layers; the platysma and dermis are closed with absorbable monofilament suture and the skin with topical glue.
Surgical excision is an effective means of dealing with second branchial anomalies with recurrence rates noted later. Major complications are rare. Complications of second branchial anomaly surgery that do not require surgical intervention include minor wound infections, seromas, and stitch extrusion. Hypertrophic scars and keloids have been reported, as with any neck incision. Hypoglossal and superior laryngeal nerve injury, most often temporary, have also been reported.4 Although seemingly straightforward, misdiagnosis can occur in 15% of cases or more.13 Initial correct diagnosis is crucial because prior evidence shows that the recurrence rates after surgical excision of branchial anomalies are 14% and 22% with previous infection and surgery, respectively, whereas the recurrence rate for a primary lesion is 3%.14 Because sinuses and fistulae are more likely to become infected preoperatively than cysts (67% vs 21%, respectively),3 they are also more likely to recur. Additionally, absence of an epithelial lining identified at the time of original excision is associated with a much higher risk of recurrence.3
Conclusions Second branchial anomalies, including cysts, sinuses, and fistulae are relatively common congenital neck lesions, representing approximately 69%-95% of all branchial anomalies. Sinuses and fistulae are generally discovered earlier and are more likely to become infected and recur than cysts. Cysts most commonly present as painless, slowly enlarging lateral neck masses, whereas fistulae and sinuses often present with draining skin openings anterior to the sternocleidomastoid. Complete surgical excision is generally curative and carries a low risk of complications, though recurrence is more likely to occur in previously infected lesions.
Disclosure Postoperative care Postoperative care for most patients after second branchial anomaly excision is generally not complex. In patients with anomalies that extend to the pharynx requiring excision of tissue from the tonsillar fossae, oral feeding can resume safely within the immediate postoperative period, though a soft diet may be better tolerated during the first few days after surgery. Postoperative antibiotics are generally not recommended. Overnight monitoring is not required, assuming no injury to surrounding neurovascular structures; however, this decision is based on patient age, whether or not a neck drain was placed, caregiver reliability, proximity
The authors reported no proprietary or commercial interest in any product mentioned or concept discussed in this article.
References 1. Waldhausen JH: Branchial cleft and arch anomalies in children. Semin Pediatr Surg 15(2):64-69, 2006 2. Choi SS, Zalzal GH: Branchial anomalies: A review of 52 cases. Laryngoscope 105(9, pt 1):909-913, 1995 3. Schroeder JW Jr., Mohyuddin N, Maddalozzo J: Branchial anomalies in the pediatric population. Otolaryngol Head Neck Surg 137(2): 289-295, 2007
Grohmann and Herrington
Second Branchial Anomalies
4. 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 25(6):449-452, 1996 5. Prasad Sampath Chandra, Azeez Arun, Thada Nikhil Dinaker, et al: Branchial anomalies: Diagnosis and management. Int J Otolaryngol 2014, [9 pp, article ID 237015], 2014 6. Neel HB, Pemberton J: Lateral cervical cysts and fistulas. Surgery 18:267-286, 1945 7. Shedden WM: Branchial cysts and fistulas. N Engl J Med 205:800-811, 1931 8. Hu YJ, Li YD, Qu XZ, et al: Clinical analysis of branchial cleft cyst (fistula): Report of 284 cases. Shanghai Kou Qiang Yi Xue 17 (5):461-464, 2008 9. Black CJ, O'Hara JT, Berry J, et al: Magnetic resonance imaging of branchial cleft abnormalities: Illustrated cases and literature review. J Laryngol Otol 124(2):213-215, 2010
5 10. Kim MG, Lee NH, Ban JH, et al: Sclerotherapy of branchial cleft cysts using OK-432. Otolaryngol Head Neck Surg 141(3):329-334, 2009 11. Chen J, Chen W, Zhang J, et al: Endoscope-assisted second branchial cleft cyst resection via an incision along skin line on lateral neck. Eur Arch Otorhinolaryngol 271(10):2789-2793, 2014 12. Chen L, Huang X, Lou X, et al: A comparison between endoscopicassisted second branchial cleft cyst resection via retroauricular hairline approach and conventional second branchial cleft cyst resection. Lin Chung Er Bi Yan Hou Tou Jing Wai Ke Za Zhi 27(22):1258-1262, 2013 13. Kenealy JF, Torsiglieri AJ Jr., Tom LW: Branchial cleft anomalies: A five-year retrospective review. Trans Pa Acad Ophthalmol Otolaryngol 42:1022-1025, 1990 14. Reiter D: Third branchial cleft sinus: An unusual cause of neck abscess. Int J Pediatr Otorhinolaryngol 4(2):181-186, 1982