Appearance of benign mass lesions is varied within and across pathology types. Top row: normal; symmetric nodules; right sessile polyp; right sessile polyp with left reactive nodule. Middle row: left sessile hemorrhagic polyp; asymmetric nodules; asymmetric nodules; right pseudocyst. Bottom row: left cyst with right sulcus; right nodule with left reactive fibrosis; right dermoid cyst
Nodules are bilateral thickenings of the membranous true vocal folds, often at the junction of the anterior and middle thirds [2, 3]. They can be symmetric or asymmetric. They are comprised of thick fibronectin deposits in the superficial lamina propria which can be accompanied by basement membrane injury [4]. There is typically minimal impairment of the mucosal wave, and nodules tend to improve with voice therapy [2]. They represent the most common benign lesion in children [5, 6].
Polyps are exophytic masses that are often unilateral but can be bilateral, with a thin overlying epithelium [2]. They can be further classified as pedunculated or sessile according to the nature of attachment to the native vocal fold, as well as hemorrhagic or nonhemorrhagic [7]. The mucosal wave is typically minimally reduced. Surgery is often required for polyps, though voice therapy may be adequate for smaller lesions [8–10]. Polyps are rare in children.
Cysts are subepidermal epithelial-lined sacs within the lamina propria [7]. They may be further divided into those within the subepithelial space versus the vocal ligament [2]. They can be either mucus retention or epidermoid in origin and can be either congenital or acquired. Cysts are typically unilateral but can be bilateral.
Pseudocysts are superficial, subepithelial lesions with a clear, vesicle-like appearance [2]. They are sometimes associated with glottic insufficiency [2, 11]. The lesion is composed of a semisolid fluid or localized edema within Reinke’s space [2, 12] deep to thinned epithelium [2]. Importantly, in contrast to cysts, there is no encapsulation.
Epidemiology
Pediatric dysphonia is common. Prevalence estimates vary depending on study, location, methodology, and definitions. A rate of 1% in the United States was reported based on the 2012 National Health Interview Survey [13]. Carding et al. reported a prevalence of 11% based on parental report and 6% based on clinician report in a cohort of 7389 8-year-olds in the United Kingdom [14]. A smaller study in Finland found a rate of 12%, with higher rates for boys (15.8%) compared to girls (7.8%) [15]. A significant portion of children with persistent dysphonia have vocal fold nodules. The estimated prevalence of nodules among children presenting with dysphonia also varies significantly, from estimates of 5–35% [16, 17] to 38–78% [5]. Data on the prevalence of cysts, pseudocysts, and polyps are less widely reported but are relatively uncommon [18].
Pathophysiology
During vocal fold vibration, the junction of the anterior and middle thirds is exposed to maximal shearing and collision forces, resulting in vascular congestion and edema, with eventual hyalinization of Reinke’s space with hyperplasia of the overlying epithelium [7]. Nodules are acellular and composed of thickened epithelium over a dense fibrous stroma with increased type IV collagen and fibronectin [19].
Polyps are caused by impaired circulation followed by thrombosis, exudate, and edema in the lamina propria, with secondary inflammation and atrophy of the epithelium [20]. Hemorrhagic polyps may have a feeding varix [7]. Compared to nodules, they are typically more vascular with less organized collagen, though the distinction can sometimes be challenging [7].
Cysts can be either mucus retention or epidermoid in origin. Mucus retention cysts form secondary to an obstructed mucous gland and expand secondary to retained secretions. Epidermoid cysts can be congenital, developing from cell nests in the subepithelium of the fourth and sixth branchial arches, or acquired, developing from buried mucosa within injured, healing epithelium [7]. Pseudocysts typically develop secondary to phonotrauma in the setting of glottic insufficiency [2].
Presentation
Patients with benign mass lesions will typically present with dysphonia, which can be described as intermittent. If the patient is a singer, there may be inability to maintain phonation in the falsetto register. Voice may be breathy and require additional effort to produce. Vocal fatigue is commonly reported. Dysphagia is uncommon, and the presence should prompt consideration of an alternative or comorbid etiology. In rare cases with large lesions occupying a clinically significant portion of the airway, there may be history of stridor or change in respiration.
We are learning more about the psychosocial impact of dysphonia on children. If questioned, children may report anger, sadness, and frustration, with negative impacts on quality of life [21]. Further, children with dysphonia can be viewed negatively by others [22, 23].
Predisposing factors to vocal fold inflammation should be identified and treated if present. These can include vocal overuse or misuse, secondary or primary smoke exposure, laryngopharyngeal reflux, and allergic rhinitis. A recent study by D’Alatri and colleagues also identified attention-deficit hyperactivity disorder (ADHD) as a potential risk factor [24]. ADHD is one of the most common psychiatric disorders in children [25] and can include loud, impulsive vocalizations as a feature [26], predisposing to phonotraumatic vocal fold lesions.
Speech-Language Pathologist Approach
Benign vocal fold lesions are the most common cause of dysphonia in children, with nodules being the most frequently identified of these. Collaboration between speech pathologist and laryngologist in the identification and treatment of benign lesions is vital. The speech pathologist’s (SLP) role in evaluation involves careful evaluation of the child’s history, voice use, perceptual and instrumental assessment, and visualization of the larynx. The SLP can assess the impact of both the lesion(s) and the patient’s use of adaptive or maladaptive compensation and stimulability for improvement with voice therapy. The SLP’s role in treatment is primarily behavioral therapy in the treatment of nonsurgical lesions and perioperative therapy in the treatment of lesions more suited to surgical excision.
History
- 1.
Duration and onset of dysphonia
- 2.
Impact of dysphonia on emotional, social, and academic function
- 3.
Changes in behavior due to voice
- 4.
Typical daily voice use (e.g., does the child talk nonstop, do they scream and throw tantrums, and do they have heavy daily voice needs with sports or theater?)
- 5.
Other health factors that could be contributing to dysphonia, including allergies, asthma, acid reflux, and pulmonary compromise
Quality of Life Measures
These are detailed in Chap. 13 and can be helpful in determining the impact of the voice disorder on the child and their family, as well as their interest in pursuing treatment. Children with benign vocal fold lesions were found to have elevated scores on the Pediatric Voice Handicap Index, and there was no statistically significant difference by lesion type [27].
Perceptual Evaluation
The use of a perceptual instrument such as the consensus auditory perceptual evaluation of voice (CAPE-V) or the grade, roughness, breathiness, asthenia, and strain (GRBAS) scale is necessary to quantify the severity of dysphonia, characterize the dysphonia, and measure change over time. There are no studies indicating that severity of dysphonia differs by lesion type. There is a weak correlation between perceived severity of dysphonia by the clinician and the patient’s perception of impact on quality of life [28–30], so these measures should be taken as complementary.
Acoustic and Aerodynamic Evaluation
Detailed descriptions of acoustic and aerodynamic measures and how they are obtained are found in Chaps. 8, 9, 10, and 11. Children with dysphonia due to benign lesions exhibit abnormal values on acoustic parameters such as jitter, shimmer, and noise-to-harmonic ratio [6] compared to peers without dysphonia. Mean values of aerodynamic parameters in children with and without benign mass lesions are lacking, but serial measurements can be used to monitor changes with treatment.
Laryngeal Visualization
The best visualization of vocal fold lesions and their impact on vocal fold vibration and function are obtained using videostroboscopy or high-speed videoendoscopy. 70-degree rigid endoscopes or distal chip flexible endoscopes provide the best images, but it can be difficult for very young children to tolerate and participate in the exam. In these patients, it can be challenging to characterize the stroboscopic parameters, and a combination of history, perceptual, acoustic, and aerodynamic evaluation will be critical to arriving at a correct diagnosis and appropriate treatment plan.
When evaluating stroboscopic video of benign lesions, the SLP does not diagnose the lesion; however, we do describe its presence or absence, appearance, location, and impact on glottic closure and vibration. Careful observation of the primary effects of the lesions on stroboscopic parameters, as well as any compensatory behaviors in reaction to the lesion, is necessary to plan treatment. Closure pattern, pliability or stiffness, and mucosal wave symmetry all influence clinical decisions for how to proceed with therapy.
Treatment
Voice therapy is the gold standard treatment for dysphonia secondary to nodules and either with or without surgery for cyst and polyp [31]. Voice therapy has well-established effectiveness in treatment of nodules in adults [32–36]. There is a growing body of evidence that voice therapy is also effective in treating nodules in children [37–41]. Studies have included a mix of direct and indirect therapy approaches and have examined different outcome measures including quality of life, perceptual voice quality, acoustic and aerodynamic evaluation of voice, and lesion resolution. As such, it is difficult to compare findings across studies. In general, though, these studies have shown that children who undergo voice therapy achieve improvement or resolution of their dysphonia.
Nodules are frequently cited as being the result of “vocal abuse” or “vocal misuse,” terms that are falling out of favor and being replaced by “phonotrauma.” Both the quantity and quality of voice use influence development of lesions, but it is not clear why some children who clearly are both heavy and strained voice users do not develop lesions, while some with less exuberant vocal use do. However, while the research is still developing on this, the treatment of dysphonia related to nodules is focused on changing the way that children use their voices, specifically the manner in which the vocal folds contact, the coordination of subsystems of voice, and the use of unnecessary muscle activation. As nodules are assumed to form from excessive repetitive force on the vocal folds, voice therapy focuses on changing the duration, frequency, force, and manner in which the vocal folds contact.
In our practice, children with nodules typically undergo six to eight sessions of voice therapy, once per week. We recommend a combination of approaches tailored to the individual child and their needs. Voice therapy can be characterized as direct or indirect, and a combination of these is often used.
Indirect therapy includes education on vocal health, reduction and replacement of presumed unhealthy voice behaviors, parent education, and implementation of any needed behavioral changes. This is typically addressed at the first therapy session and then briefly discussed in subsequent sessions. Indirect therapy might consist of helping parents tame tantrums, giving children alternatives to yelling, and identifying play noises that may be vocally traumatic. However, simply giving children and parents lists of what not to do is neither practical nor effective. If voice therapy is punitive and children have negative associations with it, they are less likely to be adherent. As we tell toddlers to use their “walking feet” instead of running, we have to give children functional and useful ways of using their voices. Direct therapy teaches more effective and efficient ways of using the voice to achieve a functional sound with lower shearing stresses and impact and without as much perceived effort. In our practice, the majority of time spent in therapy is focused on direct therapy, training healthier voice production. There are a variety of effective ways to address this, but strategies should always be adapted based on knowledge of the anatomy and physiology, impact of the lesion, the child’s compensatory behaviors, and the child’s motivational factors and developmental level. An overview of voice therapy approaches and ways to adapt to the child’s developmental level are provided in section “Overview” of this text. Semi-occluded vocal tract exercises, resonant voice, vocal function exercises, and flow mode phonation, and often a combination of several of these, can be used to address benign vocal fold lesions.
Perioperative Voice Therapy
While voice therapy is the primary treatment modality for most children with nodules, lesions such as cysts and polyps as well as refractory nodules may require surgical excision to achieve optimal voice quality and efficiency. In these cases, we recommend pre- and postoperative voice therapy. Often, preoperative voice therapy is part of the decision-making process in pursuing surgery. If children are able to make the desired changes in voice with therapy alone, surgery is not recommended. However, if they are unable to meet vocal needs despite optimal adherence to voice therapy, excision may be recommended. When surgery is planned, one preoperative voice therapy session should focus on preparation for voice rest and return to voice use after surgery. The amount of voice rest recommended varies greatly across institutions [42]. A review of wound healing and orthopedic literature indicates that voice rest is preferable to uncontrolled voice use [43], but optimal duration of voice rest is not known. In vitro research has shown that low-amplitude vibrations may have an antiinflammatory effect on healing tissues [44], and Verdolini and colleagues found that resonant voice activities after a vocal loading task resulted in lower biomarkers of inflammation in laryngeal secretions than voice rest or uncontrolled phonation [45]. While it is not possible to generalize this to postoperative conditions, it does suggest that the large amplitude, low impact vibrations associated with resonant voice may play a role in return to vocal health after surgery. Based on the available evidence, but also on the feasibility of voice rest in children, we typically recommend 3 days of complete voice rest followed by gradual return to full voice use over the course of 2 weeks, with use of resonant voice as able, combined with multiple daily practices of semi-occluded vocal tract exercises. Following surgery, the postoperative course of therapy is typically four to eight sessions, once per week, focused on semi-occluded vocal tract exercises, resonant voice, and avoidance of any maladaptive compensatory behaviors that may arise following surgery.
Otolaryngologist Approach
History
Both the patient and parent should be included when eliciting the history, if possible. Critical elements include the nature of dysphonia (e.g., breathy, rough, weak, intermittent, or constant), alleviating and exacerbating factors, and a temporal description. Time of onset and any progression since onset should be noted. Effects of the voice disorder on patient quality of life, functioning in school, home life, and interactions with peers should be evaluated. Presence of any associated breathing or swallowing impairment is questioned. Whether any prior nonsurgical (including observation or voice rest) or surgical therapies have been tried is asked, as well as the results from them.
Potential sources of laryngeal inflammation should be sought, including symptoms of laryngopharyngeal reflux, sleep-disordered breathing, chronic cough, asthma, allergic rhinitis, smoke exposure, and vocal abuse. Extracurricular activities with heavy voice load, including singing, should be noted.
Patients with nodules may report repeated episodes of voice loss, decreased ability to sing softly, breathiness, vocal fatigue, and voice breaks [7]. Patients with polyps can report breathiness, vocal fatigue, diplophonia, and roughness. With cysts, there is often less vocal limitation than may be expected based on lesion size [7]; there can be pitch instability, diplophonia, and compensatory supraglottic hyperfunction. Pseudocysts typically cause minimal impacts on vocal fold vibration, but patients may experience breathiness secondary to impaired glottic closure [46].
Examination
A general head and neck exam is performed. In addition, several aspects are focused on in the patient presenting with dysphonia with concern for underlying benign mass lesion. Any stridor or increased respiratory effort should be noted, though infrequently encountered. The voice should be described, paying attention to presence of breathiness (implying impaired glottic closure), roughness (implying impaired vibration), pressed quality (implying hyperadduction at level of the glottis or supraglottis), projection, and range. The strap muscles are palpated during voice production to evaluate for increased tension, which may indicate primary or secondary muscle tension dysphonia.
Instrumented Assessment
Central to the accurate diagnosis of benign vocal fold mass lesions is careful laryngoscopy and videostroboscopy. In young children, this is typically accomplished with a flexible transnasal endoscope, although children as young as 5 years old may be able to participate in rigid stroboscopy. Image quality is improved with distal chip technology. Older children may tolerate a rigid transoral 70-degree endoscope which will allow for superior image quality. Exams are recorded, which allows for improved visualization of the glottis in the uncooperative patient and provides a baseline for reference during treatment.
