The Laryngoscope C 2014 The American Laryngological, V

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Vocal Fold Pseudocyst: A Prospective Study of Surgical Outcomes Christine Estes, MM, MA; Lucian Sulica, MD Objectives/Hypothesis: To examine rates of lesion recurrence and functional impairment after surgical removal of vocal fold pseudocysts and determine factors predictive of recurrence. Study Design: Prospective cohort study. Methods: Patients who underwent surgical removal of pseudocyst were recruited for 12 months of postoperative follow-up. A clinical consensus cohort of 10 laryngologists blindly reviewed pre- and postoperative videostroboscopic examinations to determine presence of pseudocyst and concurrent clinical variables (reactive lesion, varix, paresis). Patients completed a postoperative Voice Handicap Index-10 (VHI-10) and follow-up questionnaire. Results were analyzed to determine lesion recurrence rates, degree of postoperative functional impairment, and predictors of recurrence. Results: Eighteen surgeries on 17 patients (15F:2M) with pseudocyst were examined. All underwent perioperative behavioral treatment. Lesion recurrence rates were 22% (per treating laryngologist) to 33% (per clinical consensus cohort). No demographic variables proved predictive of lesion recurrence. No clinical variables predicted recurrence, although there was suggestion of paresis as a contributing factor. Postoperative VHI-10 scores were within normal limits for all patients, but 12% of patients experienced recurrent functional impairment requiring further treatment. Conclusion: Most individuals (15/17) returned to normal voice use without limitation after surgery. Some individuals appear predisposed to lesion recurrence, which occurs relatively swiftly. Demographic features and clinical variables are not predictive of recurrence. The role of glottic insufficiency related to recurrence warrants further study. Key Words: Pseudocyst, vocal fold pseudocyst, vocal fold lesion, voice disorder, phonotrauma, vocal fold paresis, glottic insufficiency, microlaryngoscopy, phonosurgery. Level of Evidence: 2b. Laryngoscope, 125:913–918, 2015

INTRODUCTION Pseudocysts are benign phonotraumatic lesions of the membranous vocal fold, characterized by presence of semisolid material without a capsule beneath a thinned epithelium, giving a translucent appearance akin to a blister.1 This blister-like nature allows for relative pliability, often with minimal-to-no effect on vocal fold vibratory properties. As vocal fold vibration is impacted relatively little, dysphonic characteristics are often less severe than with other types of phonotraumatic lesions such as hemorrhagic polyps. Pseudocysts are generally thought to be related to glottic insufficiency.1,2 Treatment recommendations vary, ranging from behavioral management3,4 to microsurgical removal of the lesion with or without treatment of underlying glottic insufficiency.1,2 A recent study suggests that behavioral intervention is sufficient for most patients; however, approximately one-third of patients pursue sur-

From the The Sean Parker Institute for the Voice, Department of Otolaryngology–Head and Neck Surgery, Weill Cornell Medical College, New York, New York, U.S.A. Editor’s Note: This Manuscript was accepted for publication October 7, 2014. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Christine Estes, MM, MA, The Sean Parker Institute for the Voice, Department of Otolaryngology–Head and Neck Surgery, Weill Cornell Medical College, 1305 York Avenue, New York, New York 10065. E-mail: [email protected] DOI: 10.1002/lary.25006

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gery for altered or inconsistent voice quality and continued vocal limitation.4 The objective of the current study is to prospectively examine a cohort of patients who underwent microsurgical removal of pseudocyst in order to: 1) establish the rate of lesion recurrence; 2) establish the rate of recurrent functional impairment; and 3) identify factors that may be predictive of recurrence.

MATERIALS AND METHODS The Weill Cornell Medical College Institutional Review Board approved this study, and informed consent was obtained at the time of decision for surgery. Participants were recruited from among those treated at the laryngology service of an urban university medical center between August 2007 and January 2011 and between November 2012 and January 2013 (hiatus due to administrative reasons). All patients were diagnosed based on laryngeal videostroboscopic examination. All were initially treated with behavioral management; those who chose to undergo surgery had not achieved satisfactory voice quality after treatment. Patients who chose to undergo surgery were offered participation and were advised that a commitment of 12 months of follow-up visits were required. All underwent surgical removal of vocal fold pseudocyst and were followed postoperatively at intervals of 1 week, 1 month, and 3 months— standard postoperative follow-up procedure at our center—and for additional study follow-up at 6, 9, and 12 months. Examination technique was consistent for each patient across all visits, including stroboscopy technique (transnasal flexible laryngoscopy or rigid transoral laryngoscopy) and vocal maneuvers during examination. Examinations included sustained modal phonation, sustained falsetto phonation, ascending pitch glides,

Estes and Sulica: Pseudocyst Surgical Outcomes


Fig. 1. Examples of unilateral and bilateral vocal fold pseudocysts visualized through rigid transoral laryngoscopy. (A) and (B) show the pseudocyst with vocal folds in adducted (A) and abducted (B) positions in a 24-year-old female performer. (C) and (D) show the bilateral pseudocysts with vocal folds in adducted (C) and abducted (D) positions in a 26-year-old female performer.

and inspiratory phonation. Some also included rapid alternating movement maneuvers. At least three of these maneuvers were included in each examination, although in many cases four or all were included. Demographic information and Voice Handicap Index-10 (VHI-10) scores were recorded prior to surgery. Surgery for all participants consisted of microlaryngoscopic removal of the pseudocyst(s) and, in some cases, removal of other mucosal pathology such as a contralateral reactive lesion. The process of removing the pseudocyst and other pathology, if applicable, involved epithelial incision at the superolateral border of the lesion, evacuation of pseudocyst contents, and epithelial redraping with removal of excess tissue. No other surgical procedures, such as measures to address glottic insufficiency, were performed. In addition to behavioral treatment prior to surgery, all patients received perioperative treatment by masters or doctorate-level certified speech-language pathologists (SLPs) specializing in voice disorders. Following 1 week of complete voice rest, patients resumed weekly voice therapy, which included education, vocal hygiene, reduction of maladaptive muscular/laryngeal tension and phonotraumatic behaviors, motor learning of healthy vocal production, and carryover activities. The amount of recommended voice use and contexts for voice use were gauged collaboratively by the treating pathologist and SLP based on stroboscopic examination and functional response to behavioral treatment. After completion of the 12-month follow-up period (or upon clear recurrence of a lesion, if this occurred prior to 12 months), anatomic and functional outcomes were assessed. To assess anatomic outcomes, 10 fellowship-trained laryngologists with a mean of 10.4 years in clinical practice were used as a clinical consensus cohort. They blindly reviewed DVDs containing 20second excerpts from preoperative and 12-month postoperative stroboscopic exams with audio in randomized order. The cohort

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indicated presence or absence of the following diagnostic clinical variables: pseudocyst (unilateral or bilateral), contralateral reactive lesion, varix, and paresis on a uniform rating sheet. The term pseudocyst was defined as a “subepithelial semisolid benign lesion without the presence of a capsule” in order to standardize diagnosis. Examples of pseudocysts are shown in Figure 1. Examples of pseudocysts with concurrent clinical variables specific to this study are shown in Figure 2. Interrater agreement among the members of the cohort was 0.773 per Fleiss’ v based on 1,800 clinical decisions; each clinical variable was deemed present based on majority consensus. Interrater agreement between the examining laryngologist and clinical consensus cohort was 0.742 per Krippendorff ’s a based on 360 clinical decisions. Intrarater reliability for the cohort, determined by four randomly repeated stroboscopic examinations, was 100%. Statistical analysis of demographic variables was calculated through Fisher’s exact test or paired t test, when appropriate. Fisher’s exact test was used to determine the relationship between concurrent clinical variables and recurrence rates and was performed with n 5 18 (number of surgeries). These were analyzed separately based on the diagnoses of the treating laryngologist and the clinical consensus cohort. The dependent variable/outcome measure was recurrent pseudocyst within 12 months. To assess functional outcome, participants completed a follow-up VHI-10 and questionnaire. Differences between presurgical and postsurgical VHI-10 scores were examined to determine functional impairment related to stroboscopic diagnosis. Paired 2-sample mean t tests were used to statistically examine pre- and postsurgical mean VHI-10 scores. The questionnaire responses were analyzed to further determine posttreatment functional voice impairment mainly in regard to vocation.

Estes and Sulica: Pseudocyst Surgical Outcomes

Fig. 2. Examples of vocal fold pseudocysts with concurrent clinical variables examined during this study, including: (A) contralateral reactive lesion (L-sided pseudocyst with Rsided reactive lesion); (B) a small varix/ectasia within the L-sided pseudocyst itself; (C) a linear varix on the side of the pseudocyst; and (D) left vocal fold atrophy with dilation of the ventricle, strongly suggestive of paresis.

RESULTS Seventeen patients met inclusion criteria, with one patient requiring a second surgery. Therefore, 18 surgeries (4 bilateral, 14 unilateral) were evaluated. The patient cohort consisted of 15 females (88.2%) and two males (11.8%) with mean age of 26 and age range of 18 to 46. Thirteen (76.5%) were performers (11 professional, 2 student). The remaining four required extensive voice use in professions including classroom education, exercise instruction, marketing, and customer service. Neither gender (Fisher’s exact test; P value 0.515), nor mean age (paired t test; P value 0.494) demonstrated a statistically significant difference between patients with recurrent pseudocyst and those without. Statistical significance was also not found between performers and nonperformers (Fisher’s exact test; P value 1.00). According to the treating laryngologist, four patients presented with bilateral pseudocysts and 14 with unilateral pseudocysts, for a total of 22 surgically excised pseudocysts; the findings of the clinical consensus cohort were in agreement. Four of 22 (18.2%) pseudocysts recurred, per the treating laryngologist; and six of 22 (27.3%) recurred, per the cohort (88.9% agreement; Krippendorff ’s a 0.769). There were no bilateral recurrences. In analyzing 18 surgeries, four (22.2%) experienced recurrence, per the treating laryngologist; and six of 18 (33.3%) experienced recurrence, per the cohort (90.9% agreement; Krippendorff ’s a 0.747). Laryngoscope 125: April 2015

The treating laryngologist diagnosed eight (44.4%) patients with a contralateral reactive lesion and the cohort diagnosed 10 (55.6%) with a reactive lesion (88.9% agreement; Krippendorff ’s a 0.784). Among those with a reactive lesion, the treating laryngologist noted two of eight (25%) recurrences of pseudocyst and the cohort diagnosed three of 10 (30%) recurrences (94.4% agreement; Krippendorf ’s a 0.774). It should be noted that one individual presented with a vocal fold cyst contralateral to pseudocyst, identified by the treating laryngologist and the cohort. This was removed surgically and was not associated with pseudocyst recurrence. This case was excluded from figures regarding reactive lesions. Four patients were diagnosed with varix by the treating laryngologist, of whom two (50%) experienced recurrence. The clinical consensus cohort also identified four patients with varix: three in agreement with the treating laryngologist and one in disagreement. Of these, one patient (25%) experienced a recurrent pseudocyst, indentified by both the treating laryngologist and the cohort. Several reviewers commented that they were not comfortable diagnosing paresis based on stroboscopic exam alone or that samples were too limited to allow diagnosis through clinical criteria. Paresis was not identified in any preoperative exams by the cohort, although diagnosed preoperatively in three patients by the treating laryngologist and postoperatively in one additional Estes and Sulica: Pseudocyst Surgical Outcomes


TABLE I. Statistical Findings Based on Presence of Pseudocyst With Concurrent Clinical Variables. Treating Laryngologist Variable

Unilateral pseudocyst Bilateral pseudocysts

Clinical Consensus Cohort

Diagnosed Pseudocyst recurrences P value

14 4

3 1


Diagnosed Pseudocyst recurrences P value


Unilateral pseudocyst Bilateral pseudocysts

14 4

Contralateral reactive lesion




4 5

1 2

1.00 1.00

Contralateral reactive lesion




Varix Paresis

4 4

2 3

0.197 Varix 0.019 Paresis

patient. Postoperative exams yielded stronger clinical findings of paresis and were diagnosed by the cohort in five patients, including the four diagnosed by the treating laryngologist (94.4% agreement, Krippendorff ’s a 0.856). Of these, two (40%) experienced recurrent pseudocyst. Per the treating laryngologist, three of four (75%) individuals with paresis experienced recurrence (94.4% agreement, Krippendorff ’s a 0.774). No significant statistical difference was found with regard to laterality, contralateral reactive lesion, or varix and recurrence—either from the standpoint of the treating laryngologist or the clinical consensus cohort. Paresis, based on the treating laryngologist’s diagnosis, in relation to pseudocyst recurrence was statistically significant, although significance was not found based on the diagnoses of the cohort. Statistical findings are summarized in Table I. The postsurgical VHI-10 was completed by 100% of study participants. Results are shown in Figure 3. For all, postoperative scores were within normal limits ( 11). The mean VHI-10 score decreased from 17.29 to 5.12 following surgery. For patients who experienced a recurrent pseudocyst, per the diagnosing laryngologist, mean VHI-10 scores decreased from 10.25 to 4.25, whereas scores decreased from 19.46 to 5.29 in those who did not. Based on the clinical consensus cohort, mean VHI-10 scores decreased from 16.67 to 5.33 in patients who experienced recurrence, whereas scores decreased from 18.17 to 4.68 in patients who did not.

4 2


Statistical significance was not found in relation to change in mean VHI-10 scores between patients who experienced recurrence and those who did not based on both the treating laryngologist’s diagnoses and the cohort, with P values of 0.57 and 0.97, respectively (paired 2-sample mean t tests). Along with the postsurgical VHI-10, a questionnaire related to functional voice ability was completed by all participants. Responses are summarized in Table II. The majority of patients (11/17, 64.7%) reported that they did not experience professional vocal limitation after surgery, and most (14/17, 82.4%) did not miss any work days during the past year due to a voice problem. Ten of 17 (58.8%) patients reported that they “never” experience voice problems, three (17.6%) “rarely” experience voice problems, three (17.6%) “sometimes” experience voice problems, and one (5.9%) reported “often” experiencing voice problems. None sought medical attention from another laryngologist and two (11.8%) sought voice therapy from another institution. There was minimal appreciable difference between those who experienced recurrent pseudocyst and those who did not. Two patients reported functional impairment following pseudocyst recurrence, despite behavioral treatment. Neither completed the 12-month follow-up period because functional limitation was clearly evident prior to that time. Their case studies are presented to illustrate the clinical course of recurrence.

Case Study 1

Fig. 3. Change in mean VHI-10 scores compared between patients who experienced recurrence and patients who did not experience recurrence. [Color figure can be viewed in the online issue, which is available at]

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A 29-year-old male musical theatre performer presented with complaint of voice change (VHI-10 5 25) after 10 weeks in the leading role of a play. He was diagnosed with bilateral pseudocysts. After an unsuccessful trial of voice therapy, he underwent surgery. Paresis was diagnosed by the laryngologist preoperatively and by the clinical consensus cohort postoperatively. He was compliant with voice rest and behavioral treatment and returned to normal vocal activities, including performance, by postoperative month 3. Unilateral pseudocyst recurrence was diagnosed by 6 months. The patient underwent a second surgery for excision of the recurrent pseudocyst 9 months later, with return to normal voice use by month 2 postoperatively. At the 3-month follow-up visit, irregularity at the site of the surgery along with a possible resolving hemorrhage Estes and Sulica: Pseudocyst Surgical Outcomes

TABLE II. Summary of Responses to Questionnaire Supplement for 17 Patients.

management. Paresis became evident following surgery. She adhered to voice rest, perioperative therapy, and recommendations, returning to her training program after one month. Three months after surgery, the patient returned with complaints of increased phonatory effort and vocal fatigue. A unilateral pseudocyst and contralateral reactive lesion were diagnosed. Although her complaints were not reflected in her VHI-10 score (8), she reported “often” experiencing voice problems, that she “somewhat disagreed” with the statement “I can participate in my profession without vocal limitation,” and that she missed 2 to 4 days of work due to voice problems. After discussion, she underwent a trial injection augmentation with equivocal benefit. She continued to perform, but still felt that she had significant functional impairment. Medialization laryngoplasty is being contemplated.


*No Recurrence (n 5 12)/recurrence (n 5 5) as determined by clinical consensus cohort. One patient underwent 2 surgeries; questionnaire was completed after the second surgery.

was noted. Midfold irregularity was observed consistently throughout follow-up exams, and eventually pseudocyst recurrence was diagnosed. He continued to perform, albeit with limitations, including unpredictable voice quality and pronounced pitch breaks between vocal registers. He reported a postoperative VHI-10 score of 11 (normal), but on the 5-item questionnaire he reported “sometimes” experiencing voice problems and that he “somewhat disagreed” with the statement “I can participate in my profession without vocal limitation, noting 1 missed work day within the year due to a voice problem. Beyond the 12-month follow-up period for this study, the patient underwent a third surgery because of continuing functional limitation. In addition to pseudocyst excision, underlying glottic insufficiency was treated with injection augmentation. He continues to be followed 5 months postoperatively with no lesion recurrence, reporting improved voice quality despite occasional minor instability in his falsetto.

Case Study 2 An 18-year-old female musical theatre performance student presented with complaints of hoarseness and decreased singing range (VHI-10 5 10). She was diagnosed with bilateral pseudocysts. She underwent bilateral surgery after failure to respond to behavioral Laryngoscope 125: April 2015

Vocal fold pseudocysts are a distinct category of benign lesions, differentiated from other lesions by demographic predisposition, characteristic features, and treatment recommendations. Clinical series in the literature indicate that they occur predominantly in females2,4 and performers.4 The patient cohort in this surgical series is consistent with prior literature in these respects. The fact that performers represented the majority of the patient cohort may be related to social and geographical aspects of our practice, but we believe it is more likely due to higher sensitivity to relatively mild vocal changes characteristic of pseudocyst leading them to seek treatment. Common clinical opinion is that development of pseudocyst is related to glottic insufficiency.1,2,4 This study provides further suggestion of a relationship between glottic insufficiency and pseudocyst. Based on the treating laryngologist’s diagnoses in this study, 17% of patients had paresis. This number rose to 22% on postoperative examination and rose from 0% preoperatively to 28% postoperatively by the clinical consensus cohort. This reflects our clinical impression that the presence of paresis is frequently not appreciated upon preoperative stroboscopic examination because glottic closure deficits are attributed to the lesion itself. Postoperatively, incomplete vocal fold closure can no longer be attributed to mass effect of the pseudocyst. According to the treating laryngologist, there was a statistically significant relationship between paresis and pseudocyst recurrence; no other concurrent clinical variables were predictive of recurrence. The clinical consensus cohort’s findings regarding paresis and recurrence did not achieve statistical significance. We were concerned that reviewers might be justifiably reluctant to diagnose paresis based on the limited stroboscopic exam samples provided. Samples were limited to 20 seconds with practical considerations for the number to be reviewed (40) by each member of the cohort. Although the treating laryngologist had the benefit of longer/multiple exams and case information, agreement between the laryngologist and the cohort was surprisingly good, Estes and Sulica: Pseudocyst Surgical Outcomes


with statistical analysis yielding 0.856 agreement (Krippendorff ’s a). Our concern that abridged exam excerpts might have lowered the number of patients diagnosed with paresis appears to have been misplaced; the cohort diagnosed paresis in more patients that the treating laryngologist. We have proposed elsewhere that the posterior glottic insufficiency found in a subset of normal female larynges functions as a pseudoparesis that predisposes females to phonotraumatic injury.4 This hypothesis is supported by extensive study of physiologically normal female vocal-fold closure patterns.5 Simulated threedimensional models of vocal fold edge shapes and closure patterns provide evidence of physiologic posterior glottic insufficiency, common to females, which leads to increased phonation threshold pressure and, in turn, increased phonotraumatic shearing and stress. Although study of this phenomenon has been limited to development of vocal fold nodules thus far, these models may mimic the pathophysiology of pseudocyst formation and explain why some neurologically normal female larynges are similarly predisposed to pseudocyst. Current clinical consensus is that failure to address glottic insufficiency correlates with a high rate of recurrence.1,2 However, the majority of patients in this study, including those with paresis, did not experience recurrence. These outcomes do not support blanket use of augmentation in patients undergoing surgery for pseudocyst. Further study is needed to clarify the role of glottic insufficiency in relation to pseudocyst formation and/ or recurrence and the efficacy of augmentation/medialization in preventing or treating recurrence. The two case studies presented shed light on the clinical course of pseudocyst recurrence. In both, concerning mucosal abnormalities were identified early and lesion recurrence, along with functional impairment, was within 3 to 6 months. We are not aware of any patients who experienced recurrence beyond the 12month study period. This suggests that certain individuals are predisposed to pseudocysts and will experience recurrence swiftly. In this representative sample of pseudocyst surgeries, 22% (based on the treating laryngologist) to 33% (based on the clinical consensus cohort) experience recurrence of a mucosal abnormality. Despite this relatively high recurrence rate compared to other benign vocal fold lesions, few patients experienced functionally significant recurrence and only 12% of patients returned to treatment with complaints of vocal limitation after surgery. Eighty-eight percent were able to return to normal voice use without further treatment, including per-

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formers. These findings show that lesion recurrence is not necessarily functionally limiting. This is consistent with our observations in an earlier article assessing voice therapy outcomes that the presence of a pseudocyst does not necessarily result in an impaired patient.4 This may be the most clinically relevant finding of our study. Limitations include a relatively small sample size and a 12-month term of follow-up. The small sample size is perhaps unavoidable in a prospective study of a specific pathology. Retrospective studies of pseudocyst2,3 presented similarly small patient cohorts. The 12-month follow-up period is somewhat arbitrary, but we have not seen patients presenting with recurrent pseudocyst beyond 12 months. Additionally, the Singing Voice Handicap Index-10 (sVHI-10) may have been more sensitive to voice change in the performers included in this study and could have provided more information to quantify patient complaints specific to singing voice impairment.

CONCLUSION These data show that postsurgical recurrence of pseudocyst occurs in 22% to 33% of cases; however, only 12% of patients experience recurrent functional voice limitations. Most patients who undergo surgical intervention for vocal fold pseudocyst are unlikely to experience recurrence, regardless of demographic characteristics, vocation, or concurrent clinical factors. When it occurs, recurrence appears to evolve relatively briskly in a matter of months. The majority of patients who undergo surgical intervention along with perioperative behavioral management have satisfactory functional outcomes with return to normal voice use without the need for ancillary procedures. The role of vocal fold augmentation or medialization laryngoplasty in preventing or treating recurrence remains to be studied. These findings can be used to counsel patients considering surgery for pseudocyst.

BIBLIOGRAPHY 1. Rosen C, Gartner-Schmidt J, Hathaway B, et al. A nomenclature paradigm for benign midmembranous vocal fold lesions. Laryngoscope 2012;122: 1335–1341. 2. Koufman J, Belafsky P. Unilateral or localized Reinke’s edema (pseudocyst) as a manifestation of vocal fold paresis: the paresis podule. Laryngoscope 2001;111:576–580. 3. Cohen S, Garrett CG. Utility of voice therapy in the management of vocal fold polyps and cysts. Otolaryngol Head Neck Surg 2007;136:742–746. 4. Estes C, Sulica, L. Vocal fold pseudocyst: result of 46 cases undergoing a uniform treatment algorithm. Laryngoscope 2014;124:1180–1186. doi: 10.1002/lary.24451. Epub 2013. 5. DeJonckere P, Kob M. Pathogenesis of vocal fold nodules: new insights from a modeling approach. Folia Phoniatr Logop 2009;61:171–179.

Estes and Sulica: Pseudocyst Surgical Outcomes

Vocal fold pseudocyst: a prospective study of surgical outcomes.

To examine rates of lesion recurrence and functional impairment after surgical removal of vocal fold pseudocysts and determine factors predictive of r...
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