Tongue-lip Adhesion Research Articles

The tongue-lip adhesion

The tongue-lip adhesion (TLA) has long been a part of the management algorithm for glossoptosis associated with the Pierre Robin sequence (PRS). Advancement of the base of the tongue has been shown to relieve airway obstruction in some infants. TLA has few complications or long-term sequelae. The procedure has also been shown to ameliorate the feeding difficulties associated with PRS and therefore, reduces hospital stay, simplifies nursing care, and makes home care less demanding.

Management of Micrognathia

Micrognathia and the associated retroposition of the tongue into the oropharynx (glossoptosis) can obstruct the upper airway, producing obstructive apnea. The primary management of respiratory compromise in patients who have micrognathia is controversial. Numerous modalities have been used, including prone positioning, nasopharyngeal airways, tongue-lip adhesion, mandibular distraction, and tracheostomy. The goal of any intervention is to relieve airway obstruction, with secondary goals reported in the literature including avoidance or early removal of tracheostomy, improvement in feeding, and accelerated growth. This review examines nonsurgical and surgical therapeutic options and their outcomes. The diagnostic and treatment algorithm employed at the senior author's institution is presented.

Tongue-Lip Adhesion in the Treatment of Pierre Robin Sequence

Pierre Robin sequence (PRS) is a congenital malformation in which micrognathia causes retroposition of the tongue. This results in feeding and breathing difficulties that can lead to severe complications. If conservative treatment is not sufficient, a surgical procedure such as a tongue-lip adhesion can be performed. The objective of this study was to evaluate our benefits and complications with this operation. All patients under the care of the Department of Plastic and Reconstructive Surgery of the VU University Medical Center, Amsterdam, The Netherlands, in the period 1993 to 2002. A consecutive series of 22 patients who needed operative intervention for PRS. All underwent a tongue-lip adhesion. Retrospective analysis of the patient charts. Follow-up has been performed for a minimum of 1 year. In 16 (73%) of the patients operated on, feeding and breathing have improved. In 6 children, the results were limited because of concomitant congenital anomalies. Complications, all without lasting effect, were observed in 12 (55%) of the patients: 4 of 5 children with dehiscence of the adhesion needed reoperation, 6 developed small chin abscess that resolved after removal of the supporting suture, and 1 patient was reintubated for bronchospasm. Tongue-lip adhesion is a good surgical treatment for most children with PRS who have an isolated tongue-base airway obstruction. More invasive procedures such as mandibular distraction can be reserved for patients where a tongue-lip adhesion has not been successful.

Subglossopalatal Synechia in Association with Cardiac and Digital Anomalies

A 1-day-old boy in respiratory distress had a midline soft tissue band between the floor of the mouth and the posterior edge of the hard palate. There was also a soft palatal cleft, cardiac anomalies, and a hypoplastic right fifth finger and toe. Although his airway initially improved following urgent excision of the subglossopalatal band, he continued to have episodic desaturations. A tongue-lip adhesion opened his airway, and he subsequently underwent resection of juxtaductal aortic coarctation and ligation of patent ductus arteriosus and left superior vena cava. Congenital oral synechiae are uncommon. Affected infants often require prompt intervention secondary to respiratory distress and feeding difficulty. Review of the literature indicates that midline subglossopalatal synechia with cardiac and digital anomalies may be in the oromandibular-limb hypogenesis spectrum.

32: Tongue-Lip Adhesion for Severe Robin Sequence: Determinants of Outcome

32: Tongue-Lip Adhesion for Severe Robin Sequence: Determinants of Outcome

Using a Modified Nutrition Plate for Early Intervention in a Newborn Infant with Pierre Robin Sequence: A Case Report

Pierre Robin sequence is a triad of micrognathia, glossoptosis, and cleft palate that results in upper airway obstruction and feeding problems. It is a serious condition with potentially severe, life-threatening airway obstruction. Approaches for treatment of airway obstruction among neonates with Pierre Robin sequence include prone positioning of the infant, tongue-lip adhesion, mandibular distraction, and tracheostomy. This case report describes a modified nutrition plate and a baby treated using this appliance.

Robin sequence: A retrospective review of 115 patients

Review a large series of patients with Robin sequence to document the incidence of (1) associated syndromic diagnoses; (2) co-morbid conditions; (3) frequency and type of operative management for airway compromise and feeding difficulties; and (4) possible differences in treatment between syndromic and nonsyndromic infants. Retrospective case-review of 115 patients with Robin sequence managed between 1962 and 2002 at two tertiary-care teaching hospitals for evaluation of demographic information, clinical findings, and treatment interventions. Fifty-four percent (N=63) of patients were nonsyndromic. Syndromic patients included: Stickler syndrome (18%), velocardiofacial syndrome (7%), Treacher-Collins (5%), facial and hemifacial microsomia (3%), and other defined (3.5%) and undefined (9%) disorders. There was no statistical difference between the syndromic and nonsyndromic patients with regard to need for operative airway management (Fisher's exact test, p=0.264). Forty-two percent of patients required a feeding gastrostomy tube to correct feeding difficulties. Patients with a syndromic diagnosis were more likely to be developmentally delayed. Fifty-one (44%) patients underwent operative airway management: 61% underwent tongue-lip adhesion and 39% underwent tracheotomy. Fifteen percent of patients initially had tongue-lip adhesion subsequently required tracheotomy. While the preferred treatment for respiratory compromise differed between the two institutions, the percentage of patients requiring operative intervention was similar. The pathogenesis of Robin sequence is multifactorial and syndromic in nearly half of the patients. Operative treatment of respiratory failure was required in 44% of infants; the rate was similar in both hospitals. The operative approach differed significantly between the institutions, however, based on the philosophy and training of the managing surgical specialty. Co-morbid factors such as baseline cardiopulmonary and neurologic status did not play a significant role in surgical decision making.

To Distract or Not to Distract: An Algorithm for Airway Management in Isolated Pierre Robin Sequence

Approaches advocated for treatment of airway obstruction among neonates with Pierre Robin sequence include positioning, tongue-lip adhesion, mandibular distraction, and tracheostomy, with no established guidelines regarding which modality is appropriate for a specific patient. This report proposes an algorithm for the management of neonatal upper airway obstruction among patients with isolated Pierre Robin sequence. Data for 21 patients with isolated Pierre Robin sequence who were treated by one surgeon during a 9-year period were reviewed. Eighteen patients presented during the first 1 week of life and three patients presented late, between 12 and 33 months of age. Follow-up periods ranged from 9 to 70 months (median, 33 months). Successful airway management was achieved with positioning alone for 10 patients, with tongue-lip adhesion for seven of nine patients, with tracheostomy for two patients, and with mandibular distraction for three patients. Changes in the maxillary-mandibular discrepancy were significant with natural mandibular growth during the first 1 year of life (p < 0.0001). Oromotor studies performed 3 months or more after tongue-lip adhesion reversal (n = 9) demonstrated no appreciable deficits in tongue function, relative to other children with cleft lips/palates. A multidisciplinary team should evaluate all patients with isolated Pierre Robin sequence, to fully assess the maxillary-mandibular relationship, anatomically define the site of airway obstruction, and identify feeding difficulties. Patients should be evaluated for episodes of desaturation occurring spontaneously, during feeding, or during sleeping. Patients with desaturation should be further evaluated with double endoscopy (nasoendoscopy and bronchoscopy). If the airway obstruction is localized to the tongue base alone and cannot be controlled with positioning, then tongue-lip adhesion is the initial treatment of choice, because such patients demonstrate significant mandibular growth during the first 1 year of life. Mandibular distraction among neonates is reserved for failures of tongue-lip adhesion in which isolated tongue-base airway obstruction is documented. Neither of the patients who experienced failure of tongue-lip adhesion in this series would have been a candidate for distraction with the algorithm presented. Avoiding routine neonatal distraction serves to avoid facial scarring, nerve and tooth bud injury, and potential disturbances of intrinsic mandibular growth. Patients with persistent respiratory difficulties beyond age 9 months require reevaluation for multiple sites of airway obstruction. Mandibular distraction may be one of several modalities required to avoid tracheostomy for such patients.

Airway management in patients with isolated Pierre Robin sequence during the first year of life.

Managing the airway of patients with Pierre Robin sequence is diagnostically and therapeutically challenging. Like many other pathologies, Pierre Robin sequence is best managed with a multidisciplinary team. Providing a comprehensive evaluation is important to detect patients who may have silent events during activities of early life. Children with airway obstruction should have a complete assessment to anatomically define the site of airway obstruction. Therefore, management options can be targeted to the specific deficiency. The present article outlines a treatment protocol for patients who present with isolated Pierre Robin sequence. The authors propose that initial diagnostic and therapeutic interventions remain relatively noninvasive. Using this protocol, more than 80% of patients with isolated Pierre Robin sequence who the authors have examined for airway obstruction in the neonatal period have been effectively treated with positioning or tongue-lip adhesion. While more aggressive and successful techniques have been reported, the authors have not found them necessary for the majority of patients with isolated Pierre Robin sequence. The following review details their approach to patients with Pierre Robin sequence.

Surgical airway management in Pierre Robin sequence: is there a role for tongue-lip adhesion?

The purpose of this study was to examine the efficacy of tongue-lip adhesion (TLA) in the management of clinically significant airway obstruction associated with Pierre Robin sequence. The records of all children admitted to The Children's Hospital of Philadelphia with a diagnosis of Pierre Robin sequence were reviewed. Charts were reviewed for birth data, diagnosis, preoperative airway management methods, and surgical intervention. Records of infants undergoing TLA were analyzed for timing of surgery, operative technique, postoperative complications, length of hospital stay, and treatment outcome. Over the 28-year period 1971 to 1999, 107 patients (47 boys, 60 girls) meeting the criteria for Pierre Robin sequence were admitted for treatment. Of these, 74 (69.2%) were successfully managed by positioning alone. Surgical management of the airway was performed in the remaining 33 (30.8%) patients, 29 of whom underwent TLA and 4 of whom underwent tracheostomy. Dehiscence of the adhesion occurred in five patients (17.2%), two of whom subsequently required tracheostomy. Within the group of patients who underwent mucosal adhesion alone, the dehiscence rate was 41.6%. When the adhesion included muscular sutures, however, dehiscence was not observed in any patient. Of the 24 patients in whom primary TLA healed uneventfully, airway obstruction was successfully relieved in 20 (83.3%). Failure of a healed TLA to relieve the airway obstruction resulted in conversion to a tracheostomy in four patients. Six patients who underwent TLA (20.7%) ultimately required a tracheostomy; five of these patients (83.3%) were syndromic. Of patients requiring preoperative intubation, 42.9% ultimately required tracheostomy. TLA successfully relieves airway obstruction that is unresponsive to positioning alone in the majority of patients with Pierre Robin sequence and should therefore play an important role in the management of these infants.

Surgical Airway Management in Pierre Robin Sequence: Is There a Role for Tongue-Lip Adhesion?

Surgical Airway Management in Pierre Robin Sequence: Is There a Role for Tongue-Lip Adhesion?

口蓋裂を伴う先天性上下顎癒合症の１例

Congenital maxillomandibular fusion (syngnathia) is a rare anomaly associated with soft tissue (synechia) or bony adhesions (synostosis).We describe a girl with synechia who had two mucosal bands between the margins of the cleft palate and the floor of the mouth. We separated the bands surgically at the age of 2 months. Subsequently, some respiratory problems remained. We performed anterior traction of the tongue with a tongue-lip adhesion procedure. Her respiration improved. Seven months after the tongue-lip adhesion procedure, the adhesion was released. At the age of 18 months, the cleft palate was repaired by a push-back method. We describe our treatment and review the literature concerning the origin of the mucosal bands.

Pierre Robin Sequence: Secondary Respiratory Difficulties and Intrinsic Feeding Abnormalities

There is considerable variation in opinion regarding the optimal management of patients with Pierre Robin sequence (PRS). No single method of airway intervention or feeding strategy is universally appropriate and effective. This study was performed to examine methods used for airway and feeding management and to identify specific problems encountered. A retrospective study of 252 patient charts between 1989 and 1997 at Children's Hospital of Wisconsin. Patient information was collected regarding perinatal history, genetics evaluation, and airway and feeding evaluations and intervention. A group of 47 patients was determined as having PRS. Secondary respiratory difficulties, defined as respiratory abnormalities in addition to the expected PRS obstruction, were identified in 23% of patients. Also, intrinsic feeding abnormalities not associated with airway obstruction were identified in 11% of patients. Analysis by Fisher's Exact Test revealed patients with a syndromic diagnosis to have a significantly higher rate for tracheotomies and gastrostomy tube placement (P = .041, and P = .0004, respectively). Syndromic patients were also found to have significantly lower Apgar scores and longer hospital stays. Positioning techniques, tongue-lip adhesion, and tracheotomy were also employed effectively with specific indications and specific difficulties that need to be considered. Patients with PRS require thorough airway and feeding evaluation. Those with additional syndromic diagnoses demonstrate higher rates of more invasive interventions. Patients with PRS must undergo individualized approaches with consideration of multiple factors for successful management.

Bedside polysomnography as an adjunct in the management of infants with Robin sequence.

Objective indications for tongue-lip adhesion in infants with Robin sequence have been lacking because of the difficulty in assessing the clinical significance of airway obstruction. In 1988, we began to use 20-hour, four-channel polysomnography to assist us in the management of infants with Robin sequence. The four channels included electrocardiogram, respiratory motion, airflow, and oxygen saturation. Infants demonstrating significant episodes of airway obstruction during sleep were recommended for tongue-lip adhesion. Fifteen infants with Robin sequence were evaluated during a 3-year period. Two babies were having severe, clinically obvious events on admission and underwent tongue-lip adhesion without polysomnography. Polysomnography was done on the other 13 infants. No significant events were seen in 7 infants, and they were discharged after a mean hospitalization of 8 days. Six studies documented significant airway obstruction, and tongue-lip adhesion was recommended. Follow-up polysomnography performed after successful tongue-lip adhesion failed to show any significant events, and the infants only required hospitalization for a mean of 12 days. We conclude that polysomnography is a useful adjunct in the management of infants with Robin sequence, providing objective indications for surgical intervention and shortening hospitalization.

Management of infants with Robin anomaly.

Congenital micrognathia and secondary glossoptosis, with or without cleft palate, constitute the Robin anomaly. Neonates with this condition are usually at great risk for life-threatening respiratory and feeding problems. The approach to the management of infants with this condition has included, in order of increasing complexity, positioning of the patient, surgical tongue-lip adhesion and tracheostomy. Because of dissatisfaction with the effectiveness of surgical tongue-lip adhesion, and a desire to avoid performing a tracheostomy, a trial of intense non-surgical management was instituted. Ten consecutive patients admitted to the Boston Floating Hospital with Robin anomaly were treated successfully by positioning, without requiring tongue-lip adhesion or tracheostomy. Medical management procedures are outlined and discussed.

Pitfalls in the treatment of the Pierre Robin Syndrome

Five possibly preventable deaths occurred in a series of 28 children with the Pierre Robin Syndrome. The primary goal in management is to move the base of the tongue from the pharynx. If this cannot be done easily, the reason, such as a tight lingular frenulum, should be found. A tongue traction stitch may be adequate to relieve the obstruction, but depending upon it for long periods of time is dangerous. The most adequate tongue-lip adhesion includes a mattress suture from the base of the tongue to beneath the chin. Patients with severe micrognathia who can be maintained in a prone position should have tongue-lip adhesion before discharge from the hospital to prevent acute airway obstruction subsequently. The Pierre Robin Syndrome is not incompatible with life, but does require good clinical judgement and careful attention to detail if treatment is to be successful.