The Laryngoscope C 2013 The American Laryngological, V
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Hoarseness Evaluation: A Transatlantic Survey Of Laryngeal Experts Babak Sadoughi, MD; Marvin P. Fried, MD; Lucian Sulica, MD; Andrew Blitzer, MD, DDS Objectives/Hypothesis: Hoarseness is a symptom of laryngeal dysfunction, without an existing consensus regarding its appropriate evaluation. A survey of laryngeal specialists is proposed to establish expert opinion on the methodology for evaluation of hoarseness, and to identify divergence of opinion regarding appropriate management. Study Design: Cross-sectional survey. Methods: A 13-item questionnaire was submitted electronically to the membership of the American Laryngological Association, the American Broncho-Esophagological Association, and the European Laryngological Society. Responses were collated anonymously and subjected to cross-tabulated data analysis. Results: A total of 195 responses were included for review. The majority of respondents identified themselves as laryngologists/phoniatricians (54.9%). Two-thirds (64.1%) of the providers dedicated more than 25% of their practice to voice management, and 48.8% managed more than 10 dysphonic patients weekly. Most respondents defined hoarseness and dysphonia as symptoms and not diseases. The panel recommended a mandated time to laryngoscopy of 1 week to 1 month from the onset of symptoms for most acutely dysphonic patients, regardless of risk factors for malignancy, while it was not advised to defer laryngoscopy beyond 2 months of symptom persistence in any situation. A majority (96.2%) felt that an otolaryngologist ought to perform the initial laryngoscopy of a newly hoarse patient. Conclusion: This survey demonstrates an agreement to expedite specialized laryngeal visualization for cases of hoarseness not subsiding within 1 month, and exemplifies controversies stemming from a recently published clinical practice guideline. Ongoing research and practice evaluation will contribute to set forth improved standards of care and to appropriately counsel dysphonic patients. Key Words: Hoarseness, dysphonia, laryngoscopy, guideline. Level of Evidence: 5. Laryngoscope, 124:221–226, 2014
INTRODUCTION Hoarseness is a common otolaryngologic complaint that, similarly to any other symptom, attains full significance only in its unique clinical context—either in combination with other concordant symptoms or in correlation with corroborative physical findings. Largely falling within the prerogatives of the otolaryngologist, the symptom-to-disease diagnostic investigations have traditionally relied on timely laryngoscopy. Though it is rarely objectionable to inspect the organ-exhibiting dysfunction, to visually examine the larynx requires specific training and equipment not necessarily available at the
From the New York Center for Voice and Swallowing Disorders (B.S., A.B.), St. Luke’s-Roosevelt Hospital Center; the Department of Otolaryngology–Head and Neck Surgery (L.S.), Weill Cornell Medical College, New York, New York; and the Department of Otorhinolaryngology–Head and Neck Surgery (M.P.F.), Albert Einstein College of Medicine, Montefiore Medical Center, Bronx, New York, U.S.A. Editor’s Note: This Manuscript was accepted for publication on April 4, 2013. This article has been accepted for oral presentation at the American Laryngological Association Annual Meeting in Orlando, Florida, on April 10–11, 2013. The authors have no funding, financial relationships, or conflicts of interest to disclose. Send correspondence to Babak Sadoughi, MD, New York Center for Voice and Swallowing Disorders 425 W 59th Street, 10th Floor, New York, NY 10019. E-mail:
[email protected] DOI: 10.1002/lary.24178
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time of the initial provider encounter. These limitations highlight the need to ensure best possible outcomes while optimizing costs in the management of a patient presenting with new-onset hoarseness. In an effort to address the paucity of existing recommendations, the American Academy of Otolaryngology–Head and Neck Surgery (AAO-HNS) published the Clinical Practice Guideline: Hoarseness (Dysphonia) (CPG) in 2009,1 elaborated by assembling a multidisciplinary panel of clinicians. In spite of its laudable intent, the document generated significant controversy among otolaryngologists, explained by the lack of evidence support to some of the proposed recommendations, such as the acceptance of a 3-month time lapse from the initiation of hoarseness to laryngoscopy, which is viewed by many as potentially harmful to patients. Despite the lack of pertinent scientific evidence and notwithstanding existing empirical practice trends, the CPG claimed to set a standard of care based on the collegial opinion of a group formed by a majority of nonotolaryngologists. We sought to offer a balancing view to that approach by questioning an array of laryngeal experts and gathering their opinions on the proper initial evaluation of hoarseness, with the aim of identifying contemporary practice patterns and discrepancies with the CPG proposals. Three professional societies representing Sadoughi et al.: Hoarseness Evaluation: A Transatlantic Survey
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Fig. 1. Survey questionnaire.
specialists proficient in the study and care of laryngeal disorders were queried for this purpose: the American Broncho–Esophagological Association (ABEA), the American Laryngological Association (ALA), and the European Laryngological Society (ELS), all of which impose substantial clinical or scholarly achievement in the field of laryngology as a prerequisite for membership.
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MATERIALS AND METHODS An online questionnaire consisting of 13 items (Fig. 1) was designed and submitted electronically to the membership of the ABEA, the ALA, and the ELS, after obtaining prior approval from the Executive Board and the Research and Education Committee of the ABEA, the ALA Council, and the Presidential Council of the ELS. Institutional Review Board submission was
Sadoughi et al.: Hoarseness Evaluation: A Transatlantic Survey
TABLE II. Portion of Practice Dedicated to Voice. Number of Respondents (%)
Fig. 2. Membership profiles of survey respondents. not required as this study is an anonymous opinion survey of providers involving neither human subjects nor protected health information. Anticipating the possibility of individuals holding multiple society memberships, the questionnaire included a disqualifying item designed to exclude duplicate entries, as well as a set of normative questions pertaining to membership and practice demographics. The questions were distributed to members using a web-based survey software program. All responses were collected anonymously. Data were collated and retrieved via the online data management module of the survey software. Statistical analysis using descriptive frequencies, cross-tabulations, and chi-squared (v2) testing was performed using IBM SPSS Statistics (Version 20.0).
RESULTS The questionnaire was sent electronically to the 423 members of the ABEA, 149 active fellows of the ALA, and 339 active members of the ELS with viable email addresses. A total of 241 responses was received. After elimination of duplicate entries from existing respondents (23 entries) and partially completed questionnaires (23 entries), a total of 195 entries was included for data analysis.
Demographics The membership profiles of selected respondents are summarized in Figure 2. The response rates were TABLE I. Field of Practice.
0%–25% 26%–50%
70 (35.9) 53 (27.2)
51%–75%
37 (19.0)
76%–100% Total
35 (17.9) 195 (100)
23% (98/423) from the ABEA, 42% (63/149) from the ALA, and 26% (88/339) from the ELS. The majority of respondents identified themselves as clinical laryngologists or phoniatricians (54.9%) and general otolaryngologists (24.1%). The remainder included head and neck surgeons (9.2%), pediatric otolaryngologists (7.2%), speech and language pathologists (2.6%), one anesthesiologist, one gastroenterologist, and one rhinologist (1.5%), as well as one voice-based basic science researcher (0.5%). Two-thirds (64.1%) of the surveyed providers dedicated more than 25% of their practice to the management of voice disorders, with 48.8% of the survey population managing more than 10 patients with a chief complaint of hoarseness per week in their practices (Tables I, II, and III).
Nomenclature, Timing to Laryngoscopy, and Referral Adequacy With reference to their understanding of the definition of hoarseness and dysphonia, most respondents viewed both merely as symptoms (86.2% and 67.7%, respectively), while some viewed them as both symptoms and diseases (13.8%; 19.0%). A small subset of respondents rejected dysphonia as a symptom and only recognized it as a disease entity (12.8%) or neither a symptom nor a disease (0.5%) (Tables IV and V). The time period that low-risk patients with newonset hoarseness were advised to wait before seeking any form of medical care was 1 week (31.8%) to 1 month (62.6%) from the initiation of symptoms. Low-risk patients were defined as denying a history of tobacco or alcohol abuse, hemoptysis, head and neck malignancy, or radiation therapy. Primary-care providers seeing patients complaining of new-onset hoarseness were advised to defer otolaryngologic referral for 1 week (12.8%) to 1 month (74.9%), and less frequently 2 months (8.7%) and 3 months (3.6%) in a low-risk setting. For high-risk patients, that time period was compressed
Number of Respondents (%)
Clinical laryngology
107 (54.9)
Voice-based basic science research Speech-language pathology
1 (0.5) 5 (2.6)
TABLE III. Average Number of Dysphonic Patients Managed per Week. Number of Respondents (%)
General otolaryngology
47 (24.1)
0–2
19 (9.7)
Head and neck surgery Pediatric otolaryngology
18 (9.2) 14 (7.2)
3–10 11–20
81 (41.5) 43 (22.1)
Anesthesia/Gastroenterology/Rhinology
3 (1.5)
> 20
52 (26.7)
Total
195 (100)
Total
195 (100)
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TABLE IV. Perceived Definition of Hoarseness. Number of Respondents (%)
Symptom Disease
168 (86.2) 0 (0)
Both
27 (13.8)
Neither Total
0 (0) 195 (100)
to between 1 week (77.9%) and 1 month (21.0%), while 1.0% of respondents suggested 2 months, and none agreed with a 3-month wait. For low-risk patients presenting to the otolaryngologist with an inaugural complaint of hoarseness, the vast majority of respondents felt that it would not be appropriate to delay laryngoscopy beyond 1 week (69.7%) or 1 month (24.6%) since the initiation of symptoms. Few were in agreement with a 2- to 3-month delay (3.6% and 2.1%, respectively). Among the advocates of a 2-month mark, a statistically significant majority (57.1%; Pearson v2 value 5 24.9; degrees of freedom 5 9; P 5 0.003) were specialists reporting a volume of only two or fewer dysphonic patients managed per week (Fig. 3). Overall, a significant majority was of the opinion that foregoing laryngeal visualization after a first visit to the primary care provider based on elements of history and physical examination was occasionally (19.5%) to never (4.1%) justified, while 76.4% felt it was often or always necessary to consider laryngoscopy, regardless of the clinical risk assessment performed upon primary care screening. An overwhelming majority (96.2%) believed that the initial laryngoscopy of a newly hoarse patient should be the responsibility of the otolaryngology specialist. A comprehensive cross-tabulated review of the remainder of the dataset did not reveal additional statistically significant discrepancies in response patterns according to society membership, field of practice, and weekly volume of dysphonic patients seen.
DISCUSSION The appropriate steps in the evaluation of a patient with new-onset hoarseness remain to be consensually validated. For clinical presentations not entailing noteworthy risk factors for malignancy, most respondents in our study agreed with self-management by the patient, TABLE V. Perceived Definition of Dysphonia. Number of Respondents (%)
Symptom
132 (67.7)
Disease Both
25 (12.8) 37 (19)
Neither
1 (0.5)
Total
195 (100)
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or initial empirical management by the primary care provider for a limited period of time, ranging approximately from 1 week to 1 month. On the other hand, a high-risk history reduces that period to 1 week from the initiation of symptoms before a referral to an otolaryngologist is recommended. Similar responses were observed with respect to the time from symptom onset to laryngoscopy in the otolaryngologist’s office. Despite marginal support for a 2-month waiting time, our study results reflect an indisputable rejection of a 3-month mark as an acceptable duration of empirical management by any provider in any clinical scenario of inaugural hoarseness. Although our questionnaire did not include the scenario of a high-risk patient presenting to the otolaryngologist, it is certainly safe to assume that such circumstances would only incite respondents to greater caution and an even shorter time to laryngoscopy. These expert views directly challenge the current controversial CPG recommendation to defer laryngoscopy for no longer than 3 months when hoarseness symptoms fail to resolve. Although the CPG also stated in a conflicting assertion that clinicians might perform laryngoscopy at any time in a patient with hoarseness, that mitigating statement was labeled with a lower policy level (“option” rather than “recommendation”). Moreover, intentional vagueness was admittedly introduced in the recommendation “to allow for clinical judgment in the expediency of the evaluation.” While we commend the objective of maintaining sensible safeguards and flexibility in the elaboration of an evaluation algorithm, we also regard the language used in the CPG as somewhat equivocal, creating potential opportunities for misinterpretation. This defeats the purpose of a practice guideline, which should ideally aim to foster more coherent and consistent practices, rather than be a vector of further uncertainty in a territory where reliable evidence is scarce. Improper nomenclature may also be a source of ambiguity. Hoarseness, as a symptom of laryngeal dysfunction, appears frequently in the review of systems portion of an otolaryngologist’s patient encounter record. Though often translated into its semiological counterpart “dysphonia,” which may also designate a physical sign rather than a symptom, both terms are routinely used interchangeably. In our study, hoarseness and dysphonia are viewed by most as symptoms, yet a surprisingly significant number of respondents still misconstrue them as diseases. Some confusion may have arisen from the fact that the International Classification of Diseases published by the World Health Organization has assigned diagnosis codes for “dysphonia” and “other voice disturbances.” However, this effort to establish uniform international terminology for medical communication, coding, billing, and research purposes should not be viewed as an endorsement of hoarseness/dysphonia as true nosological entities, since dysphonia indeed is a sign that can correlate with multiple underlying etiologies. The CPG’s opening action statement recommends that “clinicians should diagnose hoarseness (dysphonia) in a patient with altered voice. …” We postulate that dysphonia remains merely one element of a comprehensive medical evaluation rather than a final assessment. Sadoughi et al.: Hoarseness Evaluation: A Transatlantic Survey
Fig. 3. Mandated time to laryngoscopy for low-risk patient presenting to the otolaryngologist with new onset hoarseness (cross-tabulated activity volume).
This study reports opinions emanating from members of well-established national and international authorities in the study of laryngeal disorders, with longstanding histories as professional societies. Most respondents’ declared focus of clinical practice mainly encompasses laryngology. That otolaryngologists be the ones performing diagnostic laryngoscopy was perhaps a recommendation that had to be expected from this group. Nevertheless, the CPG did not explicitly make specialty training a prerequisite for laryngoscopy in a position document geared toward any clinician managing dysphonic patients. From an otolaryngologist’s perspective, performing laryngoscopy is not necessarily a standard component of every physical examination, but few would argue against it in the evaluation of any patient with new-onset hoarseness; the risk-benefit ratio of office laryngoscopy is almost invariably favorable. Furthermore, the information gathered from the dysphonic patient’s history and “blinded” physical exam appears to provide poor diagnostic accuracy when compared to laryngoscopy and stroboscopy findings, as suggested by a clinical vignette-based survey of laryngologists by Paul et al.2 CPG publications deserve the same critical eye expected from the reader of any other medical literature. Ransohoff et al. described in a recent health policy editorial the lack of trustworthiness of many of the myriad practice guidelines that have recently emerged in the medical literature, and emphasized that CPG production has its own methodology guidelines. Poor general adherence to those methodological standards has generated enough concern to prompt a charge by Congress of the Institute of Medicine of the National Academies to address the issue.3 In an earlier commentary, Johns et al. described the shortcomings of the hoarseness CPG and their potentially negative impact on patient care and outcomes.4 For instance, the 3-month “safety net” mandate Laryngoscope 124: January 2014
suggested before performing laryngoscopy was criticized as a regression from the previously published AAO-HNS educational material, which recommended performing laryngoscopy for hoarseness not resolving within 2 to 4 weeks of onset. In another recent survey of the membership of the ABEA on the management of dysphonia, Paul et al. outlined further discrepancies between existing clinical practice schemas and positions expressed in the CPG, and acknowledged the paradoxical role that the CPG has played in stimulating scholarly commentary, debate, and research to improve our understanding of what represents proper management of acute onset dysphonia.5 Our study constitutes another element of this work in progress, which is expected to lead to broader consensus prior to the elaboration of viable guidelines for the medical community. Similar undertakings, such as Cohen et al.’s efforts to identify practice patterns in the evaluation6 and management7 of the dysphonic patient by the otolaryngologist, shall also contribute to further our knowledge. We recognize some limitations of our work, mainly relating to the very nature of the survey method and its limited response rate—albeit quite typical for an online query. The lack of available evidence only highlights the magnitude of the task at hand to substantiate future guidelines through collaborative scientific research.
CONCLUSION Hoarseness is a common occurrence with various causes. This international survey of laryngeal clinicians demonstrates a broad agreement to expedite laryngoscopy by an otolaryngologist for most cases of new-onset hoarseness not subsiding within 1 month, and reemphasizes existing controversies regarding the appropriateness of the hoarseness evaluation methods advocated by the CPG. An all-encompassing effort needs to be Sadoughi et al.: Hoarseness Evaluation: A Transatlantic Survey
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undertaken to produce the scientific evidence needed to propose clinically sound and safe practice guidelines.
Acknowledgement The authors are grateful to the ABEA, the ALA, and the ELS for their cooperation with this work, and express their gratitude to Drs. Seth Dailey (ABEA), Gady Har-El (ALA), and Marc Remacle (ELS) for their logistical assistance.
BIBLIOGRAPHY 1. Schwartz SR, Cohen SM, Dailey SH, et al. Clinical practice guideline: hoarseness (dysphonia). Otolaryngol Head Neck Surg 2009;141:S1–S31.
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2. Paul BC, Chen S, Sridharan S, Fang Y, Amin MR, Branski RC. Diagnostic accuracy of history, laryngoscopy, and stroboscopy. Laryngoscope 2013;123:215–219. 3. Ransohoff DF, Pignone M, Sox HC. How to decide whether a clinical practice guideline is trustworthy. JAMA 2013;309:139–140. 4. Johns MM, 3rd, Sataloff RT, Merati AL, Rosen CA. Shortfalls of the American Academy of Otolaryngology–Head and Neck Surgery’s clinical practice guideline: hoarseness (dysphonia). Otolaryngol Head Neck Surg 2010;143:175–177; discussion 175–180. 5. Paul BC, Branski RC, Amin MR. Diagnosis and management of new-onset hoarseness: a survey of the American Broncho–Esophagological Association. Ann Otol Rhinol Laryngol 2012;121:629–634. 6. Cohen SM, Pitman MJ, Noordzij JP, Courey M. Evaluation of dysphonic patients by general otolaryngologists. J Voice 2012;26:772–778. 7. Cohen SM, Pitman MJ, Noordzij JP, Courey M. Management of dysphonic patients by otolaryngologists. Otolaryngol Head Neck Surg 2012;147: 289–294.
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