Background Patients with therapy-resistant benign esophageal strictures (TRBES) suffer from chronic dysphagia and gener - ally require repeated endoscopic dilations. For selected patients, esophageal self-dilation may improve patient’s autonomy and reduce the number of endoscopic dilations. We evaluated the clinical course and outcomes of patients who started esophageal self-dilation at our institution. Methods This study was a retrospective case series of patients with TRBES who started esophageal self-dilation between 2012 and 2016 at the Academic Medical Center Amsterdam. To learn self-dilation using Savary-Gilliard bougie dilators, patients visited the outpatient clinic on a weekly basis where they were trained by a dedicated nurse. Endoscopic dilation was continued until patients were able to perform self-bougienage adequately. The primary outcome was the number of endoscopic dilation procedures before and after initiation of self-dilation. Secondary outcomes were technical success, final bougie size, dysphagia scores, and adverse events. Results Seventeen patients started with esophageal self-dilation mainly because of therapy-resistant post-surgical (41%) and caustic (35%) strictures. The technical success rate of learning self-bougienage was 94% (16/17). The median number of endoscopic dilation procedures dropped from 17 [interquartile range (IQR) 11–27] procedures during a median period of 9 (IQR 6–36) months to 1.5 (IQR 0–3) procedures after the start of self-dilation (p < 0.001). The median follow-up after initiation of self-dilation was 17.6 (IQR 11.5–33.3) months. The final bougie size achieved with self-bougienage had a median diameter of 14 (IQR 13–15) mm. All patients could tolerate solid foods (Ogilvie dysphagia score ≤ 1), making the clinical success rate 94% (16/17). One patient (6%) developed a single episode of hematemesis related to self-bougienage. Conclusions In this small case series, esophageal self-dilation was found to be successful 94% of patients when conducted under strict guidance. All patients performing self-bougienage achieved a stable situation where they could tolerate solid foods without the need for endoscopic dilation. Keywords Benign esophageal strictures · Esophageal dysphagia · Self-dilation · Endoscopic dilation · Esophagus · Endoscopic therapy Abbreviations Benign esophageal strictures can have various causes such TRBES Ther apy-resistant benign esophageal strictures as post-surgical ischemic strictures, radiotherapy-induced, IQR Interquartile range post-endoscopic dissection, ingestion of caustic substances, reflux-induced, and other rarer causes [1 –6]. They can be divided in the simple (short, not angulated, allow passage of endoscope) and complex (angulated, > 2 cm, severely nar- This research was presented at the Digestive Disease Week, 6–9 rowed luminal diameter) strictures . Over 80% of patients May 2017, Chicago, IL, USA (Gastrointest Endosc, May 2017 Volume 85, Issue 5, Supplement, Page AB205). with benign esophageal strictures are successfully treated with repeated endoscopic bougie or balloon dilation [5, 8, * Jacques J. Bergman 9]. However, a subgroup of patients suffer from therapy- email@example.com resistant benign esophageal strictures (TRBES). Koch- Department of Gastroenterology & Hepatology, Academic man et al. proposed a definition to distinguish two types of Medical Center, Meibergdreef 9, 1105 AZ Amsterdam, resistant strictures: (1) the refractory stricture, that cannot The Netherlands Vol:.(1234567890) 1 3 Surgical Endoscopy (2018) 32:3200–3207 3201 be remediated to a diameter of 14 mm over five endoscopic a bridge too far because patients were unfit for or refused sessions at 2-week intervals, and (2) the recurrent stricture, major surgery, or because of extensive fibrosis involving in which case a satisfactory luminal diameter cannot be the stomach after a chemical burn. The primary outcome of maintained for 4 weeks once the target diameter of 14 mm the study was the number of endoscopic dilation procedures has been reached . Complex and nonsurgical strictures after the start of esophageal self-dilation. Secondary out- are more prone to meet this definition [5 , 11, 12]. When comes of interest were technical success, time to technical patients fail to respond to standard repeated dilations, other success, clinical success, final bougie diameter, and adverse endoscopic options include the addition of steroid injections events related to self-dilation. Technical success was den fi ed and, in case of a suitable morphology, incision of the stric- as introduction of the bougie on a daily basis below the level ture . Another option is temporary placement of a self- of the stricture, as indicated by a taped marker on the bougie. expandable stent, which is effective in approximately 40% We defined clinical success as patients being able to man- of cases . Besides the high risk of recurrent dysphagia, age their dysphagia themselves at home without the need of stent migration (29%) and adverse events (21%) are common repeated endoscopic dilations and having an Ogilvie dyspha- problems with the use of self-expandable stents for the treat- gia score of 0 or 1 (Table 1) . ment of benign esophageal strictures . Although reports of patients performing esophageal self-dilation have already been published in the early 60s Procedures [15–17], this treatment option is rarely reported in mod- ern literature and only consists of some small case series Patients with therapy-resistant strictures were invited to [18–21]. For selected patients, self-dilation with dilation the outpatient clinic of our specialized nurses, where they bougies may allow them to regain autonomy and reduce were seen on a weekly basis to learn self-dilation using the need for endoscopic dilations. Case series have reported Savary-Gilliard bougie dilators according to the technique excellent outcomes with 90% clinical success rates, includ- as described by Dzeletovic and Fleischer . The first ing 90–100% tolerability and intake of solids in 90–100% of consultation included education about the rationale of patients without any dilation-related adverse events [18–20]. self-bougienage, a demonstration video from the Mayo These results suggest that self-dilation is a valid alternative Clinic Arizona, U.S. , and contact with another expe- to repeated endoscopic dilations for a subgroup of patients rienced self-dilation patient who could answer to potential with TRBES. At our institution, we offer self-dilation to questions and concerns. The patient received an 8–10 mm patients with TRBES since 2012. In this study, we aim to Savary-Gilliard bougie, which was smaller in size than the evaluate the clinical course and outcomes of our self-dilation diameter achieved at the previous endoscopic dilation, to patients. practice self-dilation at home. When the patient was able to demonstrate adequate self-dilation (i.e., technical suc- cess) during the next consultation, the bougie was upsized Materials and methods in consecutive steps to a diameter that allowed the patient to tolerate solid foods (Fig. 1). Endoscopic dilations were In this case series, we retrospectively analyzed the clinical continued until patients were able to perform self-dilation course and outcomes of all patients who started self-dilation adequately. Once patients reached a stable bougie diameter at our institution. This study was reviewed by our Medical by which they could tolerate solid food, the self-dilation Ethics Review Committee and did not apply to the Dutch frequency was reduced, usually to a frequency of once a Act ‘Medical Research Involving Human Subjects’ (date of week. Whenever dysphagia recurred or patients encoun- review: June 22, 2016). Since 2012, we offer self-dilation to tered resistance with self-dilation, an endoscopic dilation patients with TRBES. All patients performing esophageal was scheduled to relieve complaints and to re-facilitate self-dilation are prospectively registered in a database by self-bougienage. See also Fig. 2 for our self-dilation two specialized nurses (CtH and PdK) involved in the train- protocol. ing of these patients. Suitable candidates for self-dilation included patients motivated to learn self-bougienage who had (1) chronic dysphagia because of a benign esophageal Table 1 Dysphagia grading according Ogilvie  stricture requiring multiple endoscopic dilations and (2) 0 No dysphagia a stricture morphology that allowed safe self-bougienage. 1 Normal diet avoiding certain foods The presence of a diverticulum, an excentric lumen, tortuous 2 Semi-solid diet strictures, and strictures within 2 cm of the upper esopha- 3 Fluids only geal sphincter were considered relative contraindications for 4 Complete dysphagia for even liquids esophageal self-bougienage. Salvage surgery was considered 1 3 3202 Surgical Endoscopy (2018) 32:3200–3207 Fig. 1 A–D. Patient with caustic stricture performing esophageal self-bougienage using a 16 mm Savary bougie dilator Statistical analysis Results Data were retrieved from the electronic medical records. Between January 2012 and December 2016, a total of 17 We mainly used descriptive statistics. Analyses were per- patients started esophageal self-dilation because of TRBES. formed on intention-to-treat basis. For the comparison of The stricture etiology was mainly post-surgical (41%) and the number of endoscopic dilations before and after the caustic injury (35%). Strictures were present at multiple start of self-dilation, we used the nonparametric Wilcoxon levels in the esophagus in 47% of patients with the most signed-rank test for paired data. Two-sided p values < 0.05 dominant stricture mainly located in the proximal esophagus were considered statistically significant. We used the sta- (71%). Before the start of self-dilation, patients underwent a tistical software SPSS Statistics version 24 (IBM corp., median of 17 endoscopic dilation procedures (IQR 11–27) Armonk, New York, USA). during a median period of 9 months (IQR 6–36 months). 1 3 Surgical Endoscopy (2018) 32:3200–3207 3203 Fig. 2 Self-dilation protocol Besides endoscopic bougie or balloon dilations, 47% of dilation procedures, failed to learn adequate self-dilation patients received additional endoscopic treatments such as because of anxiety and motivational problems. This patient steroid injections, incision therapy, or stent placement. The received three additional endoscopic dilations before he was largest bougie size reached with endoscopic dilation had a diagnosed with metastasized esophageal carcinoma and died median diameter of 15 mm (IQR 13–17 mm). Endoscopic 8 months after the start of self-dilation. The remaining 16 treatment was complicated by an iatrogenic perforation in patients were able to perform adequate self-bougienage 24% (4/17) of patients, which was managed conservatively after a median duration of 16 days (IQR 10–52 days). The in all cases. Further details are summarized in Table 2. median follow-up period from the start of self-dilation was 17.6 months (IQR 11.5–33.3 months). Self‑dilation During the period in which the bougie was upsized to a satisfactory target diameter, 59% (10/17) of patients under- The median time from the first endoscopic dilation pro- went endoscopic dilation to facilitate the self-bougienage cedure to the start of self-dilation was 9 months (IQR with a median of 1 (IQR 0–2) endoscopic procedure per 6–36 months). The technical success rate of learning self- patient. Once a stable situation was reached with a satis- bougienage was 94% (16/17). A 52-year-old male patient factory bougie size, 29% (5/17) of patients required addi- with a 2–3-cm-long post-radiation stricture in the proximal tional endoscopic dilation with a median of 0 (IQR 0–1) esophagus, who started self-dilation after 24 endoscopic procedures per patient. The overall number of endoscopic 1 3 3204 Surgical Endoscopy (2018) 32:3200–3207 Table 2 Baseline characteristics No. (%) (N = 17) Gender (male) 10 (59) Age [median (range)] 65 (32–76) years Etiology of stricture Post-surgical 7 (41) Caustic 6 (35) Other 4 (24) History of esophageal cancer (yes) 8 (47) Stricture at multiple levels in esophagus (yes) 8 (47) Stricture longer than 2 cm (yes) 11 (65) Location of dominant stricture Proximal esophagus (< 25 cm from incisors) 12 (71) Mid esophagus (25–30 cm from incisors) 3 (18) Distal esophagus (> 30 cm from incisors) 2 (12) Number of previous endoscopic dilations; median (IQR) 17 (11–27) Previous endoscopic treatment in addition to bougie/balloon dilation None 9 (53) + Steroid injections 2 (12) + Incision with steroid injections 4 (24) + Stent placement 1 (6) + Incision and stent placement 1 (6) Maximum diameter reached with endoscopic dilation ; median (IQR) 15 (13–17) mm IQR interquartile range Peptic (n = 1), radiation-induced (n = 1) and chronic inflammation of unknown origin (n = 2) Largest bougie size that was passed endoscopically through the stricture dilation procedures dropped from a median of 17 procedures signs of bleeding. There were no perforations or other seri- (IQR 11–27) before the start of self-dilation to a median ous adverse events related to self-bougienage. of 1.5 procedures (IQR 0–3) after the start of self-dilation (p < 0.001), see also Fig. 3. The final bougie size achieved with self-bougienage had a median diameter of 14 mm (IQR Discussion 13–15 mm). All patients reported that they could tolerate solid foods with a median Ogilvie dysphagia score of 0 (IQR In this small case series of 17 patients with TRBES, 94% 0–1), making the clinical success rate 94% (16/17). At the of patients were able to learn and perform esophageal self- end of follow-up, 76% (13/17) of patients were still actively dilation using bougie dilators. Esophageal self-bougienage performing self-bougienage, two patients (12%) had stopped led to a statistically significant and, most of all, clinically self-dilation for a period of 1.5 years and 1.5 months, and relevant reduction in the need for endoscopic dilation pro- two patients (12%) had died because of metastasized esopha- cedures. All patients performing esophageal self-bougienage geal carcinoma. The outcomes are summarized in Table 3. reported excellent outcomes regarding dysphagia with all Regarding the safety of self-bougienage, one patient (6%) being able to eat and swallow solid foods (Ogilvie dysphagia presented at the emergency department with hematemesis, score ≤ 1). Although the literature on this topic is scarce, no signs of hemodynamic instability and a hemoglobin level other series also support self-dilation as alternative treatment of 6.9 mmol/L. Upper endoscopy revealed a small mucosal option in patients with TRBES [18, 19, 25]. A case series tear at the gastric cardia (Fig. 4), most likely caused by the of 32 patients from the Mayo Clinics, U.S., reported com- tip of the bougie due to too deep insertion with self-boug- parable results with a technical success rate of 94% (30/32) ienage. After careful instructions and marking the bougie and a reduction in the average number of endoscopic dila- with a piece of tape to indicate the appropriate depth of tion procedures from 22 to 1 before and after initiation of self-bougienage, the patient was discharged on the same day. self-dilation, respectively, with a median follow-up of 32 Another patient was referred to the emergency department months . There was a significant improvement in dys- because of melena, but upper endoscopy did not show any phagia symptoms, as well as in stricture diameter and weight 1 3 Surgical Endoscopy (2018) 32:3200–3207 3205 Fig. 3 Endoscopic dilation procedures before and after the start of self-dilation* Table 3 Outcomes of Total esophageal self-dilation (N = 17) Technical success [no. (%)] 16 (94) Time to achieve technical success [median (IQR)] 16 (10–52) days Final bougie size [median (IQR)] 14 (13–15) mm Duration of follow-up [median (IQR)] 17.6 (11.5–33.3) months No. of endoscopic dilations after start self-dilation [median (IQR)] 1.5 (0–3) Able to eat solid foods (Ogilvie dysphagia score ≤ 1) [no. (%)] 16 (94) Adverse events Hematemesis [no. (%)] 1 (6) IQR interquartile range after initiation of self-dilation. No adverse events related to were satisfied or very satisfied with their overall ability to eat self-bougienage occurred . and all patients indicated that they would use self-bougien- Besides endoscopic outcomes and dysphagia symptoms, age again under similar circumstances . Patients did not esophageal self-dilation also positively impacts on patient- report any adverse events related to self-bougienage using reported quality of life scores. A study from the University a Maloney dilator with the median duration of self-dilation of Michigan, U.S., reported that during a 33-year period 158 being almost 10 years . Another series from the Mayo patients with cervical esophagogastric anastomotic strictures Clinic Arizona found that, when retrospectively assessed by were taught self-dilation, which was 8% of all patients who a self-designed questionnaire, global scores for dysphagia underwent a transhiatal esophagectomy during that period and overall quality of life significantly improved under self- . Out of the 78 survivors, 34 patients responded to an dilation compared to the period of endoscopically performed esophageal-specific survey, showing that 85% of patients dilations . These results emphasize the positive effect of 1 3 3206 Surgical Endoscopy (2018) 32:3200–3207 cervical esophagogastric anastomotic stricture had finally stopped self-dilation and the remaining 53% were still performing self-bougienage with an average frequency of once every 2 months . In the series by Dzeletovic et al., consisting of a more heterogeneous population, 10% (3/30) of patients were able to stop self-dilation and 27% (8/30) had decreased the self-bougienage frequency to a maximum of twice weekly . So when a satisfactory bougie size is reached, the self-bougienage frequency can gradually be reduced and thereby patient burden can fur- ther be alleviated. Adverse events related to self-bougienage are rare and particularly perforations have not been reported in the afore- mentioned case series [18–21]. However, there is a report of a pneumomediastinum related to Eder–Puestow self-dilation Fig. 4 Self-dilation induced small mucosal tear in the gastric cardia . Other rare complications reported are complete swal- in a patient with a hiatal hernia lowing of a Maloney dilator requiring surgical removal from the stomach , and repeated unintentional insertion of a Maloney dilator into the right bronchus in a patient with a self-dilation on the patient’s quality of life, including emo- hypopharyngeal stenosis . So patients should be care- tional and social well-being compared to hospital bound fully selected based on a suitable anatomy and stricture endoscopic dilations. morphology, they should be well-informed about the poten- Teaching patients how to perform self-bougienage tial risks and considerable resistance with self-bougienage requires strict guidance to overcome anxiety and moti- should always be avoided. vational problems. In our series, one patient who started This small case series is limited by its retrospective self-bougienage was not able to insert the bougie below nature, the small sample size, lack of controls, and the the level of the stricture and stopped further attempts selected and heterogeneous population from a single ter- because of anxiety and lack of motivation to continue the tiary care center. Nevertheless, this retrospective evalua- training with our nurses. Dzeletovic et al. also reported tion shows that self-dilation may be a valid alternative for two failure cases (6%) because of anxiety and in addition selected patients with TRBES who require repeated endo- three patients (9%) who stopped self-dilation because of scopic dilations. This analysis is the first step to develop a intolerance due to throat and/or chest pain . To over- systematic approach for future patients who will start self- come anxiety, we invited patients on a weekly basis at dilation at our institution. Future prospective evaluation of the outpatient clinic of our dedicated nurses to monitor clinical and patient-reported outcomes will learn more about the progress and answer to questions and concerns. For the efficacy of self-dilation on the physical, psychological, further motivational support, we occasionally invited other and social well-being of patients with strictures that hardly self-dilation patients to the training session so that patients respond to endoscopy therapy. could share their experience. Lidocaine spray or gargle in the bottom of the throat may also be helpful to get through Funding This work was supported by the Dutch Digestive Foundation, the first phase of self-bougienage . Grant I 16-04. Self-dilation as treatment option in patients with benign esophageal strictures is often unknown or being ignored Compliance with ethical standards because of insufficient experience with teaching this technique. However, as demonstrated by our results, self- Disclosures Jeanin E. van Hooft received research grants from Cook Medical and Abbott, and consulting fees from Boston Scientific and dilation can be a real solution for a subgroup of patients Medtronic. Emo E. van Halsema, Chantal A.’t Hoen, Patricia S. de that do not respond to endoscopic dilation. In our series, Koning, Wilda D. Rosmolen, and Jacques J. Bergman have no conflicts 88% (15/17) of patients had a history of at least ten endo- of interest or financial ties to disclose. scopic dilation procedures. With a target bougie size of 14 mm all patients will be able to eat solid foods and, if Open Access This article is distributed under the terms of the Creative desired, patients can even upsize their bougie over 14 mm Commons Attribution 4.0 International License (http://creativecom- to achieve a satisfactory situation. The study from the mons.org/licenses/by/4.0/), which permits unrestricted use, distribu- tion, and reproduction in any medium, provided you give appropriate University of Michigan, U.S., showed that after a median credit to the original author(s) and the source, provide a link to the duration of almost 10 years 47% (16/34) of patients with a Creative Commons license, and indicate if changes were made. 1 3 Surgical Endoscopy (2018) 32:3200–3207 3207 Options Gastroenterol 13(1):47–58. https://doi.org/10.1007/ References s11938-014-0043-6 14. Fuccio L, Hassan C, Frazzoni L, Miglio R, Repici A (2016) 1. Repici A, Small AJ, Mendelson A, Jovani M, Correale L, Has- Clinical outcomes following stent placement in refractory san C, Ridola L, Anderloni A, Ferrara EC, Kochman ML (2016) benign esophageal stricture: a systematic review and meta- Natural history and management of refractory benign esopha- analysis. Endoscopy 48(2):141–148. https://doi.org/10.105 geal strictures. 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