Abstract
This report highlights the long-term management of velopharyngeal insufficiency (VPI) and misarticulation in an 18-year-old male with unilateral cleft lip and palate. Despite primary and secondary surgeries, persistent VPI required a speech appliance and intensive therapy to address glottal stops and pharyngeal fricatives. While therapy eliminated these errors, residual VPI continued to impact speech intelligibility. Reassessment revealed that while some misarticulations persisted, the patient retained speech improvements from prior therapy, allowing for partial substitutions of target sounds. This case emphasizes the role of speech therapy in managing misarticulation despite unresolved VPI and highlights the need for ongoing multidisciplinary care.
Keywords
Introduction
Cleft lip and palate (CLP) is one of the most common congenital anomalies in approximately 1 in 700 live births worldwide. 1 This condition results from an incomplete fusion of the facial tissues during embryonic development, which can affect the upper lip, hard palate, soft palate, or all 3. Children born with CLP often face feeding, hearing, and speech development challenges due to structural deficits in the oral and nasal cavities. Early surgical interventions, such as cheiloplasty and palatoplasty, aim to correct these anatomical abnormalities and support typical development. Although some children go on to develop normal speech without additional intervention, a substantial proportion, approximately 50%, continue to exhibit speech deviations by the age of 5, despite surgical repair.2,3
One of the most persistent challenges in CLP patients is velopharyngeal insufficiency (VPI). Velopharyngeal insufficiency is a condition where the soft palate fails to close completely against the back wall of the throat during speech, resulting in air escaping through the nose, leading to hypernasality, nasal air emission, and distorted speech sounds.4-6 It is estimated that approximately 20% of patients who undergo cleft palate repair experience persistent speech impairments, primarily as a result of VPI. 7 The main methods used to diagnose VPI are auditory-perceptive evaluations and video nasoendoscopies. 6 Managing VPI is often complex and typically requires a multidisciplinary approach, combining surgery, speech therapy, and sometimes prosthetic interventions..8,9
Speech therapy plays a crucial role in the habilitation of CLP patients by addressing compensatory articulation errors, such as glottal stops and pharyngeal fricatives, as well as phonological errors and articulation distortions not caused by malocclusion. 10 However, speech issues resulting from structural deficits, such as hypernasality, nasal emissions, and weak pressure consonants, typically require surgical intervention or prosthetic management, as speech therapy alone is ineffective in resolving these resonance disorders. 11 Therefore, an interdisciplinary approach combining surgery, speech therapy, and in some cases, prosthetic devices is essential for optimal speech outcomes in CLP patients. 12
This study aimed to highlight the management of an 18-year-old male with left-side CLP, focusing on the integration of speech therapy and the use of speech prosthesis to address persistent VPI and compare the results before and after the treatment. This case is notable for its 4-year gap in speech therapy, which provides valuable insights into the long-term challenges of managing speech in CLP patients with persistent VPI.
Case Presentation
The patient is an 18-year-old male with left-side CLP. His surgical history includes cheiloplasty at 5 months, primary palatal surgery at the age of 1-year-old, secondary palatal surgery at the age of 3 years old, and lip revision with external nasal reconstruction at 5 years old, all performed at the same institution and by the same oral maxillofacial surgeon. He had regular follow-ups with an oral and maxillofacial surgeon every 6 months until age 4. In addition to these scheduled visits, he also had irregular consultations with the surgeon before and after surgeries or whenever necessary. Regarding speech and language therapy, he received regular follow-ups every 3 months after palatoplasty to monitor his developmental progress. Despite these surgical interventions, he developed VPI, which led to hypernasality, nasal air emission causing distortion of consonants, and persistent speech difficulties, including glottal stops and pharyngeal fricatives. Hypernasality was evaluated using the 4-point scale established by the Japanese Association of Communication Disorders, where 0 = good, 1 = mild, 2 = moderate, and 3 = severe.
First Phase of Speech Therapy (4-11 Years old)
The patient received his first speech therapy treatment, focusing on speech errors of glottal stops. Despite persistent VPI symptoms, speech therapy specifically targeting misarticulation yielded positive results. By focusing on articulation drills, the patient demonstrated improved precision in sound production, particularly under structured tasks such as syllable repetition and word-level practice, even though velopharyngeal closure remained incomplete.
Significant hypernasality and glottal stops were noted during an examination at the age of 4, indicating ongoing VPI. The velopharyngeal function (VPF) test was conducted following the “Cleft Palate Speech Assessment” guidelines issued by the Japanese Association of Communication Disorders. The assessment included an auditory assessment of speech, specifically a 4-point scale rating for hypernasality and consonant distortions caused by nasal emission. Additionally, the evaluation measured the amount of nasal emission during speech production and blowing tasks. The result of a follow-up VPF test revealed moderate VPI, which may affect the patient's ability to apply correct articulation in everyday conversation consistently. However, by the age of 11, after several years of speech therapy, the patient exhibited progress in correcting misarticulation patterns, showing reduced glottal stops in controlled settings. The progress in misarticulation was measured using a widely used articulation test for children in Japan. The evaluation was conducted by a native Japanese-speaking speech-language pathologist (SLP) with over 10 years of experience in cleft palate speech therapy. Additionally, VPF was evaluated at that time, and the results confirmed the presence of VPI. These findings suggest that speech therapy effectively improved misarticulation, including errors related to incorrect place and manner of articulation, despite the continued presence of VPI.
Therapy Gap (Age 11-15)
From age 11 to 15, the patient did not participate in speech therapy sessions or receive evaluations for VPI at our division. Instead of speech therapy by the experienced SLP treating cleft palate speech, he received sessions from the teacher at school.
Upon resuming therapy at age 15, a comprehensive reevaluation was conducted to assess the gap's impact on his speech. The examination indicated that VPI symptoms persisted, with moderate hypernasality. The patient's speech intelligibility was rated as “sometimes understandable but often unclear in conversational settings.” The result of an articulation test showed recurring glottal stops, a new sound error of pharyngeal fricatives, and moderate consonant distortion caused by nasal air emissions, impacting his speech. Although he retained knowledge of sound production techniques from previous therapies, the therapy gap has stalled his overall progress, and he developed a new error of pharyngeal fricative, which was not noted in the previous therapy sessions.
Postgap Management (Age of 15 and Beyond)
Following the reevaluation, the patient resumed speech therapy sessions to improve and maintain prior gains in misarticulation. Therapy focused on reducing articulation errors, such as glottal stops and pharyngeal fricatives, to improve sound clarity in structured tasks. Although these sessions helped maintain correct articulation in controlled conditions, challenges persisted with generalizing these corrections to spontaneous speech.
In the VPF test at 15 years old, hypernasality was rated as moderate. However, residual VPI symptoms, including nasal air emissions during speech and occasional glottal stops and pharyngeal fricatives, continued to affect conversational speech. The patient's speech intelligibility remained limited in everyday contexts, rating “sometimes understandable but often unclear in conversational settings.” While ongoing VPI continued to cause nasal air emission and hypernasality, speech therapy successfully reinforced correct articulation in structured tasks. In particular, the patient produced more precise speech sounds during controlled exercises, showing that speech therapy could manage misarticulation effectively, even without complete velopharyngeal closure.
During a routine SLP consultation at age 17, the patient stated: “I want to be able to speak more clearly. High school is not enjoyable. I do not communicate verbally with my classmates; I send messages via social media when I need to say something. My life up until high school is only half of my journey. I have faced many challenges, but my efforts are nothing significant. I have only one friend I talk to, and the friend also had the same surgery in childhood.” These statements were collected in the medical records as part of standard clinical observations by an experienced SLP. Previously, the patient and his family were not receptive to proactive treatment for VPI, so only speech therapy was provided. However, in response to the patient's desire for more explicit speech, proactive VPI treatment, which had not been previously implemented, was initiated.
The patient was fitted with a palatal prosthesis to aid with velopharyngeal closure and address ongoing hypernasality and consonant distortion caused by nasal air emission. The attending physician follows an approach in which prosthesis is used to activate the pharyngeal muscles before considering surgical intervention. This method aims to optimize muscle function and improve postsurgical outcomes. The prosthesis was created by the attending oral and maxillofacial surgeon, who has been treating cleft palate for over 30 years. When determining the type of prosthesis, an endoscopic examination was performed to observe dynamic movements, while lateral cephalometric radiographs were taken at rest and during phonation to assess static conditions. Additionally, the imaging was conducted in coordination with an SLP, who provided speech samples to distinguish compensatory errors. 13 The prosthesis was adjusted multiple times to improve its anatomical alignment and function.
Ongoing Management and Future Considerations
The patient attends regular speech therapy sessions focusing on articulation to support improved generalization of corrected sounds. His palatal prosthesis is periodically adjusted to optimize velopharyngeal closure. Given persistent VPI-related challenges, future treatment options, including potential surgical interventions, are being considered to achieve more reliable VPF, which may further support clear speech in spontaneous settings.
Discussion
This case highlights the complexity of managing speech disorders in patients with CLP, particularly when complicated by VPI. Despite multiple surgical interventions and early speech therapy, the patient continued to experience significant speech challenges, including hypernasality, consonant distortion from nasal air emission, and articulation errors. This discussion explores the implications of the patient's extended therapy gap, the psychosocial impact of his condition, and the approach of the palatal prosthesis in managing persistent VPI.
The patient experienced a 4-year gap in speech therapy from 11 to 15 years old. During this period, no structured interventions were conducted to address his speech difficulties. The reevaluation at age 15 indicated that the gap had led to stagnation in the patient's speech progress. Objective assessments revealed persistent hypernasality, nasal air emission, and VPI, with limited articulation and speech intelligibility improvement. The VPF and conversation intelligibility were unchanged from pregap evaluations, suggesting that the absence of regular speech therapy halted potential improvements.
Literature on therapy gaps’ impact on speech development among CLP patients emphasizes the importance of consistent, early intervention.10,14,15 Gaps in therapy, particularly over prolonged periods, can lead to stagnation or regression in progress, as seen in this case. The lack of speech improvement over the 4 years possibly exacerbated the patient's speech-related challenges, further complicating his ability to generalize speech corrections into spontaneous conversation. The findings from this case align with reports that ongoing speech therapy is crucial for managing articulation errors, especially in adolescence, when speech becomes more integrated into social and academic life, and long therapy gaps can significantly disrupt progress in managing VPI and articulation.10,14
While the prolonged gap in therapy may have hindered the patient's speech progress, it is important to consider additional factors that could have contributed to the persistence or recurrence of speech errors. One possible explanation is residual VPI, which may have limited the patient's ability to achieve stable articulation, particularly at the conversational level. Without sufficient velopharyngeal closure, speech therapy alone may not have been enough to ensure consistent improvements in articulation. An additional factor may have been inappropriate therapy in a school-based setting. Initially, the patient exhibited glottal stops, but later interventions inadvertently reinforced pharyngeal fricatives, introducing a new error pattern. A study highlights disparities in SLP training, particularly in nonspecialized settings where limited access to cleft/craniofacial teams can lead to inadequate intervention. 16 In this case, the patient developed a new compensatory error after receiving therapy from a teacher lacking expertise in cleft speech, emphasizing that SLPs unfamiliar with cleft speech disorders may unintentionally reinforce misarticulations rather than correct them. These findings underscore the need for specialized speech therapy, particularly for patients with compensatory articulation errors. Additionally, insufficient home-based practice may have contributed to the limited progress. 17 Consistent reinforcement outside therapy sessions is essential for generalizing correct articulation into spontaneous speech. In this case, the patient's family may have faced barriers to implementing home exercises, possibly due to geographic constraints or limited availability for frequent clinic visits. Without ongoing structured support, therapy gains may not have been fully maintained. 16 Taken together, these factors likely played a role in the patient's ongoing speech difficulties, emphasizing that successful speech intervention requires a combination of continuous therapy, proper intervention techniques, and adequate home reinforcement. This case underscores the complexity of managing speech disorders in children with cleft-related VPI and highlights the need for tailored, multidisciplinary treatment plans to prevent the development of new compensatory errors and ensure long-term speech success.
This case also illustrates the profound psychosocial impact of persistent speech difficulties on a patient's emotional well-being, especially during adolescence. Throughout his treatment journey, the patient expressed frustration with his speech difficulties, particularly during his evaluations at the age of 17. He stated a desire to “speak well” but reported that “school is not very fun.” While this does not necessarily indicate disengagement from school, his preference for written communication over verbal interactions may suggest challenges in social interactions, where clear speech is essential. Notably, the patient shared that he has only one close friend, who also underwent cleft surgery as a child. This may suggest a tendency to form social connections with individuals who have similar experiences, possibly due to a shared understanding of speech-related challenges and reduced self-consciousness in communication. Adolescents with speech disorders related to cleft palate speech often struggle with self-esteem and social isolation. Research has shown that speech impairments can lead to feelings of embarrassment, withdrawal, and reduced participation in social and academic activities.18-20 Children and young adults with CLP had more significant behavioral problems and increased depression symptoms than healthy ones. 21 The patient's comments about not being interested in school and his desire to perform well in interviews for an electrical academy may reflect a lack of confidence in social situations, especially where effective communication is crucial.
His expression of emotional exhaustion, seen in statements such as “two minutes feel like two thousand years,” further underscores the emotional burden of long-standing speech challenges.
In response to the patient's continued speech difficulties, a palatal prosthesis was introduced at age 17 to aid VPI, which might have impacted his speech. The prosthesis was adjusted multiple times to optimize its function, and the results showed modest improvements in increased accuracy of speech sound production. However, despite these improvements in structured settings, the patient struggled to generalize these gains to spontaneous conversation. Studies on the use of palatal prostheses in managing VPI have shown mixed outcomes, with some patients achieving significant speech improvements while others experience only marginal gains.22-24 While many centers prioritize surgical intervention for persistent VPI, the attending physician in this case follows an approach where the pharyngeal muscles are activated with a prosthesis before surgery to enhance VPF and optimize surgical outcomes. This approach has been reported in previous studies as a potential method to improve muscle coordination before surgical intervention.13,25 Given the patient's condition, this method was selected to maximize the chances of a successful postsurgical outcome while providing temporary improvement in VPF. In the case of delayed surgery, temporary use of the prosthesis can train the VPF and minimize speech mislearning. The effectiveness of preoperative prosthesis therapy has already been reported. 26 In this case, the prosthesis improved resonance and articulation during structured tasks, but challenges remained with speech intelligibility in everyday conversation. Research has shown that speech aid prostheses, such as palatal lifts and pharyngeal bulbs, can significantly improve resonance, with one study reporting that 70% of patients eliminated hypernasality after prosthetic intervention. 27 However, individual outcomes may vary, and some patients continue to experience persistent speech challenges despite prosthetic use. 28 These findings suggest that while the prosthesis was beneficial, it was not a complete solution for addressing the patient's VPI, and additional interventions may be needed.
In the management of VPI, treatment decisions must balance clinical recommendations, patient preferences, and long-term functional outcomes. 29 In this case, early treatment focused on speech therapy to target compensatory articulation errors, such as glottal stops and pharyngeal fricatives, which are commonly observed in individuals with cleft palate speech. However, speech therapy alone does not correct VPI, and despite improvements in articulation, hypernasality and nasal air emission persisted due to inadequate velopharyngeal closure. As the patient matured, they became increasingly aware of how persistent VPI affected not only their speech intelligibility but also their academic performance, social interactions, and future career opportunities. This shift in perspective reinforced the need to reassess the treatment strategy. Research has shown that adolescents with cleft lip and/or palate desire greater involvement in medical decision-making as they grow older, particularly in treatments that impact speech and appearance. 30 Shared decision-making in adolescence has been linked to increased autonomy and self-esteem, as well as greater satisfaction with treatment outcomes. 30 However, adolescents sometimes feel excluded from these discussions or pressured by parental preferences. 30 In this case, as the patient's awareness of their speech limitations increased, their role in decision-making became more central, leading to a shift in treatment goals. To align with the patient's wishes, the treatment approach was modified to include prosthetic intervention in addition to speech therapy, as the patient preferred a nonsurgical option. However, improvement was observed with the prosthesis but sufficient velopharyngeal closure was not achieved, further interventions are being considered, including pharyngeal flap surgery or sphincter pharyngoplasty, which can reach a normal resonance of 76 and 61%, respectively.8,31
In this case, due to the temporary interruption of speech therapy conducted by an SLP experienced in cleft palate treatment, the patient continued to exhibit persistent speech sound errors. However, the patient was capable of producing correct speech sounds during structured training sessions. Generalization of these improvements to spontaneous conversation was challenging due to residual VPI, and initially, active intervention for VPI was not pursued in accordance with the patient's and family's preferences. Nevertheless, long-term follow-up by a multidisciplinary team enabled the patient, as he matured, to participate actively in decision-making regarding his VPI treatment.
Key lessons learned from this case include the following: First, even in the presence of VPI, appropriate speech therapy can enable patients to produce accurate speech sounds in controlled settings. Second, as patients with cleft palate mature and progress through various life phases, their preferences regarding treatment can change. Long-term follow-up facilitates adapting the treatment strategy flexibly, in alignment with the patient's own decision-making. Furthermore, this case highlights that psychosocial challenges likely occurred throughout the patient's developmental process, emphasizing the necessity for more appropriate and timely intervention in the future.
In addressing VPI, which was the primary cause of unintelligible speech, the introduction of a palatal prosthesis proved partially effective. However, it was not a comprehensive solution to fully resolve the patient's VPI and associated articulation disorders. Moving forward, additional surgical interventions and ongoing multidisciplinary speech therapy will likely be required to achieve further functional improvements.
Conclusion
This case highlights that targeted speech therapy can effectively reduce misarticulation, even in the presence of VPI. However, the patient's ongoing speech difficulties likely resulted from multiple factors, including residual VPI, therapy limitations, and insufficient home reinforcement, rather than the therapy gap alone. This underscores the need for well-coordinated, individualized intervention strategies to ensure long-term speech stability. Future VPI treatments, including potential surgery, may further support the generalization of correct sounds in spontaneous speech, especially when combined with ongoing speech therapy, to maintain and enhance speech outcomes.
Footnotes
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Ethical Consideration
The patient was informed that their experiences and statements could be included in a clinical report and agreed to their inclusion in the case presentation. This study was conducted in accordance with ethical guidelines and received approval from the institutional review board.
Funding
The author(s) received no financial support for the research, authorship, and/or publication of this article.
Informed consent
Written informed consent was obtained from the patient’s parents to collect patient information and photographs to be published in this case report.
