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Transoral endoscopic radical resection for a recurrent pyriform sinus fistula
Journal of Pediatric Surgery Case Reports 37 (2018) 30–32
Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc
Transoral endoscopic radical resection for a recurrent pyriform sinus fistula ∗
T
Keiichi Morita , Kosaku Maeda, Hiroaki Fukuzawa Department of Pediatric Surgery, Kobe Children's Hospital, Kobe, Japan
A R T I C LE I N FO
A B S T R A C T
Keywords: Endoscopic surgery Pyriform sinus fistula Recurrence Revision surgery Transoral surgery
In revision surgery for a recurrent pyriform sinus fistula (PSF), complete resection of the fistula via a transcervical approach is difficult because of the surrounding scar tissue that results from previous surgery and infections. We successfully performed transoral endoscopic radical resection of the fistula for a recurrent PSF in two patients. Approaching the fistula from the orifice is reasonable and radical resection of the fistula is a reliable procedure in revision surgery. Transoral endoscopic resection is a feasible surgical option for a recurrent PSF.
1. Introduction
was injected into the fistula via a 4-Fr feeding tube (Fig. 1a). A circular mucosal incision around the orifice of the fistula was made using 3-mm endoscopic scissors with an electrocautery instrument (Fig. 1b). A 5-0 absorbable polydioxanone monofilament traction suture with an 11mm needle was placed at the orifice of the fistula using 3-mm endoscopic needle forceps. Taking the traction suture, the fistula wall was dissected from the surrounding connective tissue using endoscopic scissors or a cotton-constructed dissector (Fig. 1c). The electrocautery instrument was not used for dissection of the fistula to avoid heat injury to the recurrent laryngeal nerve. The fistula was completely resected en bloc (Fig. 1d). Mucosa and submucosal tissue defects after resection of the fistula were closed with interrupted 5-0 absorbable polydioxanone monofilament sutures with an 11-mm needle (Fig. 1e and f).
A pyriform sinus fistula (PSF) is a relatively rare third or fourth brachial pouch anomaly [1]. PSFs often cause repeated infections of the neck or acute suppurative thyroiditis [2]. The most common surgical procedure for a PSF has been complete resection of the fistula via a transcervical approach. However, identification and complete resection of the fistula are not necessarily easy with a transcervical approach. Incomplete resection can lead to recurrence of the fistula, and the fistula recurs after resection by a transcervical approach in 3.0–4.0% of cases [3,4]. In revision surgery for a recurrent PSF, complete resection of the fistula is more difficult than primary surgery because of the surrounding scar tissue that results from previous surgery and infections. A novel transoral endoscopic radical fistula resection procedure for a recurrent PSF using pediatric endoscopic surgical techniques is described. With the present procedure, fistula resection can be performed easily and completely even in revision surgery. 2. Operative technique Under general anesthesia with endotracheal intubation, the patient was placed in the supine position with neck extension. A cuffed-endotracheal tube with as small a diameter as possible was selected and fixed at the opposite-sided corner of the mouth from the pyriform sinus fistula. A nasogastric tube was indwelled to identify the inlet of the esophagus during surgery. A WEERDA distending video operating laryngoscope (Karl Storz, Tuttlingen, Germany) was inserted orally to visualize the PSF from the front. Indigocarmine aqueous solution, as guidance for identifying the appropriate dissection plane of the fistula, ∗
3. Case reports 3.1. Case 1 A 17-year old boy with a recurrent left-sided PSF was admitted. He had undergone fistula resection via a transcervical approach twice. However, left-sided neck infections recurred after the surgeries, and video fluorography showed a tapered narrowing, left-sided PSF (Fig. 2a). The fistula was 15 mm long on videofluorography. Transoral endoscopic resection of the fistula was performed as the third surgery. The orifice of the fistula was clearly identified on the video operative laryngoscopic view. Despite it being the third surgery, dissection of the fistula was easy, and the fistula was completely resected en bloc. Mucosa and submucosal tissue defects were closed with two interrupted sutures. Operative time was 125 min. Videofluorography on postoperative day 2 showed absence of leakage at the left-sided pyriform
Corresponding author. Department of Pediatric Surgery, Kobe Children's Hospital, 1-6-7 Minatojimaminamimachi, Chuo-ku, Kobe, 650-0047, Japan. E-mail addresses: [email protected], [email protected] (K. Morita).
https://doi.org/10.1016/j.epsc.2018.07.020 Received 14 July 2018; Accepted 21 July 2018 Available online 24 July 2018 2213-5766/ © 2018 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
Journal of Pediatric Surgery Case Reports 37 (2018) 30–32
K. Morita et al.
Fig. 1. Transoral endoscopic resection procedure for a recurrent pyriform sinus fistula (PSF). a: Indigocarmine-stained orifice of the PSF (arrow). b: Circular mucosal incision around the orifice of the PSF. c: Dissection of the PSF wall (arrow). d: Resected specimen of the PSF. e: Mucosa and submucosal tissue defects after resection of the PSF. f: Completion of the repair of the mucosa and submucosal tissue defects.
Fig. 2. Videofluorographic and endoscopic findings of the pyriform sinus in case 1. a: Preoperative video fluorography showing a 15-mm-long, tapered narrowing, left-sided pyriform sinus fistula (arrow). b: Postoperative videofluorography on postoperative day 2 showing absence of leakage at the left-sided pyriform sinus. c: Postoperative pharyngeal endoscopy 12 months after surgery demonstrating closure of the orifice of the fistula.
nasogastric tube. Pharyngeal endoscopy on postoperative day 4 demonstrated complete closure of the orifice of the fistula (Fig. 3b). Videofluorography on postoperative day 7 showed a smooth suture site and absence of leakage at the left-sided pyriform sinus (Fig. 3c), and free oral feeding was started. The patient was discharged on postoperative day 9. Over the follow-up period of 12 months, no left-sided neck infection occurred.
sinus, and free oral feeding was started. There were no perioperative complications, and the patient was discharged on postoperative day 5 (Fig. 2b). Pharyngeal endoscopy 12 months after surgery demonstrated complete closure of the orifice of the fistula (Fig. 2c). Over the followup period of 24 months, no left-sided neck infection had occurred. 3.2. Case 2
4. Discussion
A 13-year old boy who had undergone left-sided PSF resection via a transcervical approach had recurrence of the fistula. Videofluorography showed a 17-mm-long, tapered narrowing, left-sided PSF (Fig. 3a). Transoral endoscopic resection of the fistula was performed as the second surgery, and the fistula was completely resected en bloc. Since the circular mucosal incision around the orifice of the fistula was large, mucosa and submucosal tissue defects were closed with 4 interrupted sutures. Operative time was 115 min. Since the suture site was large, oral feeding was not started, and enteral feeding was performed via a
The present transoral endoscopic resection for a recurrent PSF has two advantages compared with other procedures. First, this procedure can approach the fistula directly from the orifice of the fistula. In the revision surgery for a recurrent PSF, the fistula is often embedded in dense scar tissues from previous surgery and infections. It is more difficult to expose and pursue the fistula than in primary surgery with the transcervical approach. On the other hand, in this procedure, dissection 31
Journal of Pediatric Surgery Case Reports 37 (2018) 30–32
K. Morita et al.
Fig. 3. Videofluorographic and pharyngoscopic findings of the pyriform sinus in case 2. a: Preoperative videofluorography showing a 17-mm-long, tapered narrowing, left-sided pyriform sinus fistula (arrow). b: Postoperative pharyngeal endoscopy on postoperative day 4 demonstrating closure of the orifice of the fistula. c: Postoperative video fluorography on postoperative day 7 showing absence of leakage at the left-sided pyriform sinus.
along the fistula can be performed from the beginning of the surgery, and the effect of the surrounding scar tissues is minimal. Transoral resection of a PSF has been reported in two adult patients as a primary surgery [5,6], and the ease of fistula dissection was similarly noted. We consider that this advantage is greater in revision surgery. Second, radical resection can be performed in this procedure as with the conventional transcervical approach. In last two decades, transoral trichloroacetic acid cauterization [7] or CO2 laser cauterization [8] of the fistula's internal opening has been reported as a minimally invasive treatment for a PSF. Although these treatments are less painful and need no skin incisions, there is an important problem resulting from not resecting the fistula. Closure of the fistula depends on adhesion of the cauterized fistula epithelium, and it is difficult to control the degree of adhesion. The success rate of one cauterization treatment for a PSF was 55.4–77.3% [7,8], and it often requires multiple treatments. This fact is by no means satisfactory since revision surgery requires reliability. Since one can perform radical resection of the fistula with the present procedure, only one procedure is required. The present operative procedure can be performed using the common techniques and instruments of pediatric endoscopic surgery, and it is not complicated. There are two important technical points in the present procedure. First, the circular mucosal incision should be made as small as possible. If the circular incision becomes large, as in case 2, closure of the mucosa and submucosal tissue defects is difficult. Second, a complete circular mucosal incision around the orifice of the fistula and a traction suture facilitate subsequent dissection of the fistula. Recurrent laryngeal nerve injury during dissection is a major concern. Taking the traction suture, dissection staying in close contact with the fistula wall using cold instruments prevents recurrent laryngeal nerve injury, and no recurrent laryngeal nerve palsy occurred in the present cases. The present procedure has two limitations. First, the distending video operating laryngoscope is too large to insert orally in small children. Therefore, we suggest that the present procedure is indicated for patients from later childhood. Second, the present procedure cannot be performed for all types of PSFs. Radical resection for a cystic PSF by the present procedure is technically difficult. A tapered narrowing PSF, as in cases 1 and 2, is feasible for the present procedure. The form of a PSF should be confirmed by videofluorography or computed tomography before surgery.
surgical option for a recurrent PSF. Conflict of interest The authors declare that they have no conflicts of interest. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors. Patient consent Consent to publish the case report was not obtained. This report does not contain any personal information that could lead to the identification of the patient. Authorship All authors attest that they meet the current ICMJE criteria for Authorship. Appendix A. Supplementary data Supplementary data related to this article can be found at https:// doi.org/10.1016/j.epsc.2018.07.020. References [1] Franciosi JP, Sell LL, Conley SF, Bolender DL. Pyriform sinus malformations: a cadaveric representation. J Pediatr Surg 2002;37:533–8. [2] Takai SI, Miyauchi A, Matsuzuka F, Kuma K, Kosaki G. Internal fistula as a route of infection in acute suppurative thyroiditis. Lancet 1979;313:751–2. [3] Xiao X, Zheng S, Zheng J, Zhu L, Dong K, Shen C, et al. Endoscopic-assisted surgery for pyriform sinus fistula in children: experience of 165 cases from a single institution. J Pediatr Surg 2014;49:618–21. [4] Sheng Q, Lv Z, Xiao X, Zheng S, Huang Y, Huang X, et al. Diagnosis and management of pyriform sinus fistula: experience in 48 cases. J Pediatr Surg 2014;49:455–9. [5] Kamide D, Tomifuji M, Maeda M, Utsunomiya K, Yamashita T, Araki K, et al. Minimally invasive surgery for pyriform sinus fistula by transoral videolaryngoscopic surgery. Am J Otolaryngol 2015;36:601–5. [6] Koyama S, Fujiwara K, Morisaki T, Fukuhara T, Kawamoto K, Kitano H, et al. Submucosal abscess of the esophagus caused by piriform sinus fistula treated with transoral video laryngoscopic surgery. ORL J Otorhinolaryngol Relat Spec 2016;78:252–8. [7] Cha W, Cho SW, Hah JH, Kwon TK, Sung MW, Kim KH. Chemocauterization of the internal opening with trichloroacetic acid as first-line treatment for pyriform sinus fistula. Head Neck 2013;35:431–5. [8] Wang S, He Y, Zhang Y, Zhang J, Shah R, Feng G, et al. CO2 laser cauterization approach to congenital pyriform sinus fistula. J Pediatr Surg 201; 53:1313-1317.
5. Conclusion Approaching the fistula from the orifice is reasonable, and radical resection of the fistula is a reliable procedure in revision surgery for a recurrent PSF. Therefore, transoral endoscopic resection is a feasible
32
Contents lists available at ScienceDirect
Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc
Transoral endoscopic radical resection for a recurrent pyriform sinus fistula ∗
T
Keiichi Morita , Kosaku Maeda, Hiroaki Fukuzawa Department of Pediatric Surgery, Kobe Children's Hospital, Kobe, Japan
A R T I C LE I N FO
A B S T R A C T
Keywords: Endoscopic surgery Pyriform sinus fistula Recurrence Revision surgery Transoral surgery
In revision surgery for a recurrent pyriform sinus fistula (PSF), complete resection of the fistula via a transcervical approach is difficult because of the surrounding scar tissue that results from previous surgery and infections. We successfully performed transoral endoscopic radical resection of the fistula for a recurrent PSF in two patients. Approaching the fistula from the orifice is reasonable and radical resection of the fistula is a reliable procedure in revision surgery. Transoral endoscopic resection is a feasible surgical option for a recurrent PSF.
1. Introduction
was injected into the fistula via a 4-Fr feeding tube (Fig. 1a). A circular mucosal incision around the orifice of the fistula was made using 3-mm endoscopic scissors with an electrocautery instrument (Fig. 1b). A 5-0 absorbable polydioxanone monofilament traction suture with an 11mm needle was placed at the orifice of the fistula using 3-mm endoscopic needle forceps. Taking the traction suture, the fistula wall was dissected from the surrounding connective tissue using endoscopic scissors or a cotton-constructed dissector (Fig. 1c). The electrocautery instrument was not used for dissection of the fistula to avoid heat injury to the recurrent laryngeal nerve. The fistula was completely resected en bloc (Fig. 1d). Mucosa and submucosal tissue defects after resection of the fistula were closed with interrupted 5-0 absorbable polydioxanone monofilament sutures with an 11-mm needle (Fig. 1e and f).
A pyriform sinus fistula (PSF) is a relatively rare third or fourth brachial pouch anomaly [1]. PSFs often cause repeated infections of the neck or acute suppurative thyroiditis [2]. The most common surgical procedure for a PSF has been complete resection of the fistula via a transcervical approach. However, identification and complete resection of the fistula are not necessarily easy with a transcervical approach. Incomplete resection can lead to recurrence of the fistula, and the fistula recurs after resection by a transcervical approach in 3.0–4.0% of cases [3,4]. In revision surgery for a recurrent PSF, complete resection of the fistula is more difficult than primary surgery because of the surrounding scar tissue that results from previous surgery and infections. A novel transoral endoscopic radical fistula resection procedure for a recurrent PSF using pediatric endoscopic surgical techniques is described. With the present procedure, fistula resection can be performed easily and completely even in revision surgery. 2. Operative technique Under general anesthesia with endotracheal intubation, the patient was placed in the supine position with neck extension. A cuffed-endotracheal tube with as small a diameter as possible was selected and fixed at the opposite-sided corner of the mouth from the pyriform sinus fistula. A nasogastric tube was indwelled to identify the inlet of the esophagus during surgery. A WEERDA distending video operating laryngoscope (Karl Storz, Tuttlingen, Germany) was inserted orally to visualize the PSF from the front. Indigocarmine aqueous solution, as guidance for identifying the appropriate dissection plane of the fistula, ∗
3. Case reports 3.1. Case 1 A 17-year old boy with a recurrent left-sided PSF was admitted. He had undergone fistula resection via a transcervical approach twice. However, left-sided neck infections recurred after the surgeries, and video fluorography showed a tapered narrowing, left-sided PSF (Fig. 2a). The fistula was 15 mm long on videofluorography. Transoral endoscopic resection of the fistula was performed as the third surgery. The orifice of the fistula was clearly identified on the video operative laryngoscopic view. Despite it being the third surgery, dissection of the fistula was easy, and the fistula was completely resected en bloc. Mucosa and submucosal tissue defects were closed with two interrupted sutures. Operative time was 125 min. Videofluorography on postoperative day 2 showed absence of leakage at the left-sided pyriform
Corresponding author. Department of Pediatric Surgery, Kobe Children's Hospital, 1-6-7 Minatojimaminamimachi, Chuo-ku, Kobe, 650-0047, Japan. E-mail addresses: [email protected], [email protected] (K. Morita).
https://doi.org/10.1016/j.epsc.2018.07.020 Received 14 July 2018; Accepted 21 July 2018 Available online 24 July 2018 2213-5766/ © 2018 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).
Journal of Pediatric Surgery Case Reports 37 (2018) 30–32
K. Morita et al.
Fig. 1. Transoral endoscopic resection procedure for a recurrent pyriform sinus fistula (PSF). a: Indigocarmine-stained orifice of the PSF (arrow). b: Circular mucosal incision around the orifice of the PSF. c: Dissection of the PSF wall (arrow). d: Resected specimen of the PSF. e: Mucosa and submucosal tissue defects after resection of the PSF. f: Completion of the repair of the mucosa and submucosal tissue defects.
Fig. 2. Videofluorographic and endoscopic findings of the pyriform sinus in case 1. a: Preoperative video fluorography showing a 15-mm-long, tapered narrowing, left-sided pyriform sinus fistula (arrow). b: Postoperative videofluorography on postoperative day 2 showing absence of leakage at the left-sided pyriform sinus. c: Postoperative pharyngeal endoscopy 12 months after surgery demonstrating closure of the orifice of the fistula.
nasogastric tube. Pharyngeal endoscopy on postoperative day 4 demonstrated complete closure of the orifice of the fistula (Fig. 3b). Videofluorography on postoperative day 7 showed a smooth suture site and absence of leakage at the left-sided pyriform sinus (Fig. 3c), and free oral feeding was started. The patient was discharged on postoperative day 9. Over the follow-up period of 12 months, no left-sided neck infection occurred.
sinus, and free oral feeding was started. There were no perioperative complications, and the patient was discharged on postoperative day 5 (Fig. 2b). Pharyngeal endoscopy 12 months after surgery demonstrated complete closure of the orifice of the fistula (Fig. 2c). Over the followup period of 24 months, no left-sided neck infection had occurred. 3.2. Case 2
4. Discussion
A 13-year old boy who had undergone left-sided PSF resection via a transcervical approach had recurrence of the fistula. Videofluorography showed a 17-mm-long, tapered narrowing, left-sided PSF (Fig. 3a). Transoral endoscopic resection of the fistula was performed as the second surgery, and the fistula was completely resected en bloc. Since the circular mucosal incision around the orifice of the fistula was large, mucosa and submucosal tissue defects were closed with 4 interrupted sutures. Operative time was 115 min. Since the suture site was large, oral feeding was not started, and enteral feeding was performed via a
The present transoral endoscopic resection for a recurrent PSF has two advantages compared with other procedures. First, this procedure can approach the fistula directly from the orifice of the fistula. In the revision surgery for a recurrent PSF, the fistula is often embedded in dense scar tissues from previous surgery and infections. It is more difficult to expose and pursue the fistula than in primary surgery with the transcervical approach. On the other hand, in this procedure, dissection 31
Journal of Pediatric Surgery Case Reports 37 (2018) 30–32
K. Morita et al.
Fig. 3. Videofluorographic and pharyngoscopic findings of the pyriform sinus in case 2. a: Preoperative videofluorography showing a 17-mm-long, tapered narrowing, left-sided pyriform sinus fistula (arrow). b: Postoperative pharyngeal endoscopy on postoperative day 4 demonstrating closure of the orifice of the fistula. c: Postoperative video fluorography on postoperative day 7 showing absence of leakage at the left-sided pyriform sinus.
along the fistula can be performed from the beginning of the surgery, and the effect of the surrounding scar tissues is minimal. Transoral resection of a PSF has been reported in two adult patients as a primary surgery [5,6], and the ease of fistula dissection was similarly noted. We consider that this advantage is greater in revision surgery. Second, radical resection can be performed in this procedure as with the conventional transcervical approach. In last two decades, transoral trichloroacetic acid cauterization [7] or CO2 laser cauterization [8] of the fistula's internal opening has been reported as a minimally invasive treatment for a PSF. Although these treatments are less painful and need no skin incisions, there is an important problem resulting from not resecting the fistula. Closure of the fistula depends on adhesion of the cauterized fistula epithelium, and it is difficult to control the degree of adhesion. The success rate of one cauterization treatment for a PSF was 55.4–77.3% [7,8], and it often requires multiple treatments. This fact is by no means satisfactory since revision surgery requires reliability. Since one can perform radical resection of the fistula with the present procedure, only one procedure is required. The present operative procedure can be performed using the common techniques and instruments of pediatric endoscopic surgery, and it is not complicated. There are two important technical points in the present procedure. First, the circular mucosal incision should be made as small as possible. If the circular incision becomes large, as in case 2, closure of the mucosa and submucosal tissue defects is difficult. Second, a complete circular mucosal incision around the orifice of the fistula and a traction suture facilitate subsequent dissection of the fistula. Recurrent laryngeal nerve injury during dissection is a major concern. Taking the traction suture, dissection staying in close contact with the fistula wall using cold instruments prevents recurrent laryngeal nerve injury, and no recurrent laryngeal nerve palsy occurred in the present cases. The present procedure has two limitations. First, the distending video operating laryngoscope is too large to insert orally in small children. Therefore, we suggest that the present procedure is indicated for patients from later childhood. Second, the present procedure cannot be performed for all types of PSFs. Radical resection for a cystic PSF by the present procedure is technically difficult. A tapered narrowing PSF, as in cases 1 and 2, is feasible for the present procedure. The form of a PSF should be confirmed by videofluorography or computed tomography before surgery.
surgical option for a recurrent PSF. Conflict of interest The authors declare that they have no conflicts of interest. Funding This research received no specific grant from any funding agency in the public, commercial, or not-for-profit sectors. Patient consent Consent to publish the case report was not obtained. This report does not contain any personal information that could lead to the identification of the patient. Authorship All authors attest that they meet the current ICMJE criteria for Authorship. Appendix A. Supplementary data Supplementary data related to this article can be found at https:// doi.org/10.1016/j.epsc.2018.07.020. References [1] Franciosi JP, Sell LL, Conley SF, Bolender DL. Pyriform sinus malformations: a cadaveric representation. J Pediatr Surg 2002;37:533–8. [2] Takai SI, Miyauchi A, Matsuzuka F, Kuma K, Kosaki G. Internal fistula as a route of infection in acute suppurative thyroiditis. Lancet 1979;313:751–2. [3] Xiao X, Zheng S, Zheng J, Zhu L, Dong K, Shen C, et al. Endoscopic-assisted surgery for pyriform sinus fistula in children: experience of 165 cases from a single institution. J Pediatr Surg 2014;49:618–21. [4] Sheng Q, Lv Z, Xiao X, Zheng S, Huang Y, Huang X, et al. Diagnosis and management of pyriform sinus fistula: experience in 48 cases. J Pediatr Surg 2014;49:455–9. [5] Kamide D, Tomifuji M, Maeda M, Utsunomiya K, Yamashita T, Araki K, et al. Minimally invasive surgery for pyriform sinus fistula by transoral videolaryngoscopic surgery. Am J Otolaryngol 2015;36:601–5. [6] Koyama S, Fujiwara K, Morisaki T, Fukuhara T, Kawamoto K, Kitano H, et al. Submucosal abscess of the esophagus caused by piriform sinus fistula treated with transoral video laryngoscopic surgery. ORL J Otorhinolaryngol Relat Spec 2016;78:252–8. [7] Cha W, Cho SW, Hah JH, Kwon TK, Sung MW, Kim KH. Chemocauterization of the internal opening with trichloroacetic acid as first-line treatment for pyriform sinus fistula. Head Neck 2013;35:431–5. [8] Wang S, He Y, Zhang Y, Zhang J, Shah R, Feng G, et al. CO2 laser cauterization approach to congenital pyriform sinus fistula. J Pediatr Surg 201; 53:1313-1317.
5. Conclusion Approaching the fistula from the orifice is reasonable, and radical resection of the fistula is a reliable procedure in revision surgery for a recurrent PSF. Therefore, transoral endoscopic resection is a feasible
32