Maithilee Menezes, MD, presents on if there is an underlying "excuse" for feedings to be difficult for the infant and is there a need for continuation of care with outpatient feeding clinic?
As you all know, I'm a pediatric otolaryngologist here at Children's and I've had the pleasure to interact with most of you on a regular basis. I have No financial disclosures, and as we had just gone over, and I'll do again just a little bit of background to help set the stage for what we want to go through next. The tongue, the frenulum, as we know, is a fold of mucous membrane and extends from the floor of the mouth to the midline of the underside of the tongue. It helps to stabilize the base of the tongue, and it does not normally interfere with tongue tip movement. As a partner of mine said recently, anytime you fold a mucose over, you can have a band or a tie. That's like having a pleat in the skirt. We've talked about how it can have challenges from a feeding standpoint, and I also want to think about the longer late term later effects that people have often come to and discussed about this, including speech and articulation, ability to sweep or keep the teeth clean, or other challenges. This has been recognized as far back as the time of Aristotle from 350 years before the common era. Techniques regarding tongue tie released were published as early as the 1600s in textbooks, and reportedly midwives had intentionally overgrown a fingernail at one point to help lice those lingual attachments. Thinking a little bit about that embryology. Yay. By the 1st 4 weeks, that tongue formation begins, and it arises from the 1st, the 2nd, the 3rd, and the 4th arches, pharyngeal arches, and it starts from the tuberculum impart um to form the anterior tongue, the median swellings then form the posterior tongue, and ankyloglacia then results from the failure in the apoptosis or the degeneration of the rela. But there's a whole lot of stuff that's been talking about tongue tying, and if you think about this from before and now. The incidents or the discussion of tongue tie has gone up tremendously. So In a systematic review and a meta-analysis from 2021, the overall prevalence, when they're looking at over 24,000 patients was about 8%, though it has been cited anywhere from 0.3% to 16%. Less than 50% though of the patients had had difficulty with breastfeeding, at least from the older literature. So, as I mentioned, there's been a significant amount of increase in what we know. So tenfold increase from 1997 to 2012 in the reported studies, or 866% increase in the treatment recommended over the last two decades. Most frequently noted are um due to maternal symptoms when it first comes up, especially with issues with breastfeeding, and hence we've had some of that wonderful discussion already. So how should we assess it? Well, I think you've heard a really good overview of how we could have an assessment. Um, how about just a grading system we're discussing that. So several systems have been put forth. Kotlow in 1999, Carlos in 2004, a hatlif assessment, which is A larger assessment of the child rather than just a structural, and then there's also like the latch assessment too. To go through a few of them, the cutler system focuses on the free tongue, specifically the distance between the frenulum and the tip of the tongue, and it really only is an anatomic um evaluation. Carlos does look at anatomy, so it incorporates the frenulum attachment point onto the tongue. It grades the firmness and the shape of the lingual attachment, and it recommends that this assessment is being done with the breastfeeding assessment as well. The hat lifts also looks for anklo glossia with both appearance and function. It has a 12 point assessment in there, and it judges that a tongue tie is present if the appearance is less than 8 on their grading scale and the function is less than 11. And then the latch system is latch, audible swallowing, nipple type comfort hold, and I think we're going through a number of that already, as we had talked about and identifies difficulty in the breastfeeding process and the breastfeeding competence. So there are a number of different grading systems, and when we look at this from a like a motor standpoint. There's not always great correlation. The hat lift system in this review from 2021 looked at it, and as I mentioned, there's 12 items in total. It does correlate well when you're looking at it from a breastfeeding assessment tool, and there's a good correlation between function and appearance. The cola system not quite so much, and the corals there wasn't quite as much of an ability for that um correlation between function and appearance. There are other systems as well, and not that we're going to be extensive and exhausted with everything, but just to point out that there could be the Bristol tongue assessment tool, the lingual frenulum protocol, the breastfeeding assessment tool, the phrenotomy decision tool for breastfeeding dyads, and it goes on. You'll see a lot of this if you ever do a deep dive like I did, um, you'll see a lot of these different systems and tools being put forth and just so that you're aware of them. By no means, as I mentioned, this is exhaustive. There are controversies as we have brought up. Is there a posterior tongue tie? Is it only an anterior tongue tie? What is a posterior tongue tie? It's a band of tissue with a perhaps a thick fibrous band that's inserting posteriorly into the ventral tongue, but not extending to the tip of the tongue as we more classically might see. When we look at it from an ENT perspective in 2020 with the consensus practice guidelines, Experts couldn't come to consensus regarding a posterior tongue tie. Some feel it's not to be a true clinical entity, and there were a lot of challenges with this surrounding the definition, including how do we assess it. The literature is not great in a small sample size and varying methodology. All right, those are different systems. Is there anything else that we might know? Well, let's talk about our assessment. When we first assess a child, at least from the ENT clinic, you can guess, we're looking for ABCs. We're looking for any structural problem. The only signs early on might be a poor latch or challenges with feeding. These could be the tip of the iceberg, and we may see much more nine months later, as you well know, when there could be more developmental issues, and now we could identify those challenges. So, as I'm looking at this kid, and I hope you're beginning to identify a few things that are already making me say, mm, I'm a little bit concerned right here from what I'm seeing, looking at again, as we mentioned, the jaw, the tone. How long is the feeding going on, length of feeds, are they tiring? What is the sleep state and the hunger state regulation? What is the latch itself? What is mom assessing? What is mom going through? Is she pumping? Is that what's the volume or the rate of the flow? Do they have lactation support? What are their concerns? What are their anxiousness? What is the pain? What is discomfort? So there's a lot going on in here. May also be thinking about sounds. Is the child noisy? Is there sturter? Is there stridor? And while I might not have time to do the full assessment, it really makes me think about other members of the team and how can we support this diet. What am I looking for? really quickly, and this will be in the slides that will come out. I'm looking at the head and neck evaluation, the tongue, the mandible, the maxilla, the palate is a relative glossoptosis and then key. Are there signs and symptoms of relative airway obstruction? What is their tone? Did they have a feeding tube before? Is there neuromuscular disorders? I'm looking at other head and neck sources. Is there signs that make me get concerned about nasal obstruction? As I mentioned, is there stridor? Is there stor? Should be thinking about other things like laryngomalacia? Should I be thinking about what else in the past medical history? Do I need to be thinking about like vocal fold immobility? Do I, do I see any craniofacial anomalies coming up? Remember, and you know this, there are unique features of the neonatal airway. They're an obligate breather until the 1st 6 to 8 weeks of life. There is a superior relative position of the larynx. The epiglottis may be a little bit more flaccid and collapsible. Remember that the subglottis is the narrowest portion of the airway, and it is sensitive to small changes in airway and dimension. In comparison, in that upper airway in that newborn, the tongue is larger in proportion, the pharynx is smaller, we talked about the epiglottis, the larynx is more anterior and superior, and the trachea is narrow and less rigid. So OK. Now we've gone through all of that background. Let's go through some literature. When is it of benefit? So, let's first think about breastfeeding. Going through some of the best articles that I could find, and these are the ones that are often cited. And so hopefully this will be a good resource for you later on. Um, Dahlberg back in 2006, looked at patients undergoing phrenotomy versus sham procedures. They didn't really notice a big difference in latch scores, but they did notice an improvement in the pain that mom may feel. 2012, Barry and all noted an immediate improvement in maternal pain and perceived feeding, though the objective observer didn't note any real significant improvement. Edmunds in 2014 did a randomized control trial, and I'm sorry about the abbreviations there, but you'll see RCTU in the future for these slides. Looking at those assessments, the hatless score did improve after phrenotomy. But the functional scores didn't differ significantly. So Cochrane review, looking at this in 2017, doing a very large analysis said, well, as we're looking at this, 5 of these randomized control trials met inclusion. And then they had an N of 302. 3 of them did objectively measure breastfeeding. 4 of them objectively measured and assessed for maternal pain. Where were the challenges with all of this? There were methodological issues. These were small trials. Most controls in these eventually underwent procedure. None could report increased long-term successful breastfeeding. What they did find was that there was a significant improvement for the family though, for reduced nipple pain, and unfortunately, there was no consistent positive effect on feeding. That sounds like it might be minimizing this a little bit. It's not. As we had already said, if there is a significant amount of pain, we need to think about this as a diet. We need to think about what's the system itself. If the family, if mom feels like this is really, really uncomfortable, it's going to affect the whole process and the whole system, and it will affect them in terms of their enthusiasm for breastfeeding, their enthusiasm for trying to do this, um, and will affect how baby interacts as well. So there is some evidence to say that this can be helpful. What about a posterior tongue tie? Should we do a release? So some have tried to do a more rigorous assessment and to be systematic about it. So like Harry in 2017 tried to do an assessment of that from a perspective cohort study. In 2021, they set up a prospective randomized control trial, and they had 47 patients. They examined using a bottle feeding system measuring tongue function, and they also used the breastfeeding self-assessment uh survey. So this is what the mother felt afterwards. And what they noted was that there was some improvement of feeding after the release. There were challenges though that have come up with this, including was there concern about how it was set up for the trial, as in every child was assessed and they did have a malfunctional and an occupational therapy, um sorry, speech assess uh speech feeding assessment. And so all of them did have stretching maneuvers. And if this was going through, did that potentially affect the outcome? It depends on how you set up the trial. It is a good trial. It was well it was for all that we have. It was well um set up, but there is also selection bias. All of these were families who were very motivated to come on in and wanted to be able to um. Have an improvement in breastfeeding, and they actively sought the clinic. So should we just go ahead then and offer a release for it from a breastfeeding standpoint? I think that there's a significant amount of evidence to say that if a family is motivated and they want to try to do more and they're going to be working uh to try to increase flow and increase uh the amount for breastfeeding, I think that there's a lot for, yes, especially early in life. Are there any other options? And what is it about in general or in for the longer term? Are there any other options? So, this paper was published in 2019, and it came from a quality improvement project at Boston Children's, and their goal was to develop a sustainable and reproducible multidisciplinary, uh sorry, reproducible program. They did a multidisciplinary assessment and they developed a therapeutic strategy program. They assessed how often procedures were required both before and then after a comprehensive feeding evaluation. And as I mentioned, all of them were evaluated through a multidisciplinary feeding consultation. So, after assessment, they trialed several different interventions. Moms learned different techniques if they were having challenges to address defeating challenges. Prenotomy was only recommended if their functional impairments appeared to be related to the tongue. In this case, it also included a lip tie. Of those 115 patients, 72 did not choose to undergo a surgical procedure for tongue tie release. 63% of them essentially did not. The rates of phrenotomy overall, prior to this quality intervention in the same clinic, about 95% of them were having a release. After going through this, the rates of phrenotomy decreased from 95% to 37% after introducing lactation support. And the phrenotomy was associated with higher maternal worries with feeding, and reduced scores on breastfeeding self-efficient efficacy. We often hear, I'm gonna change hats a little bit. We often hear about, well, but if I have a tongue tie, isn't this gonna cause challenges for my child in the long term? Is it gonna cause problems for longer term feeding? It's gonna cause problems with speech, right? What about with reflex? And it's gonna cause challenges with sleep apnea. So let's go through some of the literature of what we do have. Does it, is there a benefit for longer term feeding? A study out of the Netherlands in 2021 said, yeah, we think that there is going to be some improvement. 60% were still nursing at 6 months. But this was a very educated group who are all highly motivated and they had time to feed and time to spend with breastfeeding. This is a study done in the Netherlands. The family is given a year off to be able to care for the newborn, not quite what we have in the United States. From San Diego, they did a prospective study with 343 patients, and they did a routine follow-up at 1 week and 3 months. 220 patients were able to do follow-up. 56% had mild to moderate improvement in longer term feeding, but At 3 months. Only 20% were exclusively breastfeeding, and 17% had stopped completely. At 3 months. All of these studies have challenges. They're limited in their follow up. All of these are really essentially to infancy, so it's not with the, um, so not looking at that longer term after a year. There is no mention of lactation involvement in these two prior studies, nor of other therapies such as speech or occupational therapy assessment or ongoing teaching, and some they did have high loss to follow up. What they do come up with though, is emphasizing a need for holistic multidisciplinary evaluation, looking at factors involved in breastfeeding before referring on to phrenotomy. What about speech? We hear that a lot. 2015 Journal of Pediatrics, they did a systematic review that tried to be as inclusive as they could because the data wasn't great. So they looked at 1600 studies, of which only 15 could be assessed. Two were randomized control trials, 2 were cohort studies, 11 were case series. One had a poor quality and it compared the surgical techniques, but they did not include any untreated control groups. They did know that there was improvement in articulation, but not in word. Sentence or fluent speech. All were challenged with small size, incomplete, and poor characterization of the children. In the cohort studies, there was a risk of ascertainment bias, or kids, parents of kids with tongue tie would have higher index of concern for speech issues than those who never had any tongue mobility restriction. The uh cohort studies, as I mentioned, they compared a control group. No ankle glossia treated children with one with ankle glossian untreated, and the outcomes were assessed by parent reported survey only, which was non-validated. Is there anything else? 2019, a retrospective study, a phone survey, looking at kids who had been treated or assessed for tongue tie. These parents did not notice any difference in the speech difficulty or tongue mobility, but they did notice potential improvement in tongue tasks. Visconti, so I'm gonna go away. It seems like I'm only looking at ENT literature. I'm trying to be as inclusive as I can, so looking at speech pathology, they also did a systematic review. They saw that there was research supported to release decreased nipple pain, but it was inconclusive for speech benefits regarding tongue tie release and again setting the same challenges of data and quality of the research. How about from the dental reviews? Systematic review. They tried to do as much as they could with a wide inclusion criteria, cohort studies, case control studies, case series, randomized control trials. The self-controlled trials, or studies I should say, showed improvement. The cohort studies with untreated controls didn't show any difference. And the randomized control trials compared technique, but they didn't have any controls of any treated versus untreated patients. They all had the same challenges of small sample sizes and varying occurrence rates and outcomes. But none could find a connection between tongue tie and speech disorders. So the summary. General challenges in research, lack of control groups, poor randomization or blinding. Small sample sizes or variance in sizes between study and control. Use of non-validated assessments, failure to adequately describe or standardize utilization of therapy, including even did they get speech therapy or not, with patients. So the true incidence of speech difficulties is unknown. There are no clear guidelines except that they strongly evaluate recommend evaluation with the speech therapist before phonotomy and renioplasty. And ongoing care and treatment afterwards. When I have a child who's coming in, I'm gonna be looking for a couple of other things too. But I'm gonna put on my ENT hat bias, which is, uh, how well do they hear? Have they had a hearing assessment? What's been going on from that standpoint? What have been their overall developmental milestones? What's going on in their life? So I'm gonna switch hats. What about reflux? Because you may have been beginning to hear, oh, you know, if we do a tongue tie release, it's really gonna help from reflex overall. So let's go through what we could find. Similar study 2021. A longitudinal cohort study, 175 diets mom baby diet. They were assessed with the breast um feeding self efficacy form. They also did an IGERQ reflex questionnaire. Which doesn't really have a formal assessment. We don't know if they did have ongoing therapy or if they had speech or OT or lactation involvement from that standpoint. What did they come up with? These were kids who were followed for 20 days after randomization. They advocated for tongue tie release after exhausting all non-operative interventions. There are concerns though with this study. There was no formal routine assessment. They relied on patient questionnaires. There was a lack of control arm without intervention. I don't think there's a lot that could say one way or the other. Does it help from a reflex standpoint? What about sleep apnea? Because I'm sure you've been hearing this more and more. If we do not assess the tongue, if we do not address this, if the tongue goes up into a position where it sits higher in the mouth and it continues to hit the palate, we're gonna have more challenges. We're gonna cause more sleep apnea challenges from this standpoint. So let's talk about it. Looking back at that um large review from 2015, as I mentioned, it has a number of randomized control trials. Their summation on this. The data was poor and insufficient, and there was no studies including data related to sleep disordered breathing in the non-breastfeeding child. Here's a paper that was published in 2019 from a sleep center and that was in an academic medical center. They looked at their children who had been referred over a 20 month period of time. These children were 3 to 12 months of age. All of them did have a systematic sleep medicine evaluation, a history and a physical. They also included standardized maneuvers. They did measurements, and they did a sleep study, as I mentioned. They tried to use a systematic grading system combining some of the ones that we talked about before, looking at length of the tongue, if it was short or abnormal. They also, and they came up with a decision that based on putting all those studies together, that the normal length was less than 16 millimeters in terms of distance between the tongue tie and the tip of the tongue. After they looked at all of their patients, they said, well, we're gonna divide you into two groups. You had a normal frenulum and you had large tonsils and adenoids. We're gonna keep you in one group. You had a short frenulum, not big tonsils and adenoids. You did have sleep apnea. So that was about an end of 63. They looked at the apnea hypopnea indexes to say, what did that mean in terms of sleep apnea? So they divided them between the two groups. Group A, as I mentioned, is the normal frenulum. They had tonsiller. Um, anotons or hypertrophy. Group B is the short frenulum. Well, we think that there's a difference. Group A, their AI indices were 11.36, plus or minus. Confidence unfolds of minus 5.39. Group B is 13.06 plus or minus intervals of 4.17. Based on this, There was the short frenulum. Has a direct association with sleep apnea. When I read this, I have a little bit of a challenge, and I should say I'm sorry that the excluded children who are obese or they had a chronic medical condition, if they had other syndromes associated with this. My challenge is here. Confidence, the AI indices are 11.36 versus 13.06. The intervals. Sure seem really close and I'm having challenges with being able to separate out the two and say that there's not a significant amount of overlap potentially there. But as I mentioned, based on this, they said that the short frenulum has a direct association with sleep apnea. Why do I bring that up? Because these are papers, including the ones that we've gone through, that are sometimes used to promote. Or to discount. Discount, you shouldn't read what's there. Yeah, no, it really causes sleep apnea. Yes, it really causes speech challenges and used to promote we should do a tongue tie release. At best, the data is inconclusive. You should know that there are some more things talking about malfunctional therapy. Used in the scope of treatment. I think it'd be hard pressed to say that's something that we would do, especially for our younger children. Is tongue tie release all completely easy to do? Sure, it should seem like that. Let's talk about complications. Cost This is $500 to $800. Depending on where you go, that may be an out of pocket. If it is a different child, meaning if I have a child who's over like 4 months of 34 months of age, it's not an easy thing to do in clinic. If I have a child who's coming to me at 10 months of age, I'm not gonna do it in clinic, which means that there is a cost for sedation. The anesthesia cost could be an extra $500 to $900. The hospital fee might be $500 to $8000. Most complications are thought to be minor. Maybe some minor bleeding, maybe there's some pain. Maybe there's scarring of the surgical site. There are more challenges than we think. In a systematic review looking at what was reported, and these are highly, highly underreported. They noted that there was poor feeding. Some had hypovolemic shock. There was acute airway obstruction. A prospective study from New Zealand, they noted that a mean complication rate of 13.9 out of 100,000 infants, that complication rate, depending on the province could be anywhere from 0 to 85 out of 100,000 children. Could there be other complications? This is a busy slide, but to point out, and this is from the breast um International Breastfeeding Journal surveying members of the Academy of Breastfeeding Medicine, 130 out of 211 reported a complication or misdiagnosis. 100 out of 211 reported in a separate way, misdiagnosis, and the most common. Was a neuromuscular dysfunction that had not been adequately assessed. Let's also put into here that we are seeing that there was inadequate breastfeeding support and non-recognition of abnormal or facial anatomy. I should note that there was only one ENT in this group. Most were pediatrics, neonatology, dentistry, and these were all people with expertise in breastfeeding and clinical practices. So, if I was gonna give a summary, I'd say our take home points, position and airway will always influence the examination. We need to think about if there's multiple lesions that can be present. We need to think about other factors that are played when we're thinking about the long term, including hearing and the environment. We ask, try to be consistent. Use a standardized form. Is there a perfect system? No, but you can look at that and say, what do I assess every single time. Critically praised the literature. The studies that I pointed out are the ones that are often used to be talking about um tongue tie and if it's potential complications in long term. Look at the methodology. You are very smart, educated people. Take the time to look at that. I took a lot of time to look at this and I have to admit. I was surprised at what was being used to site for support. There is significant variability in the measurement, the length of the follow up, many use varying outcomes and measures and varying grading systems. And most important, find your village. Pediatrics, lactation, make sure that they're certified. Someone like Mr. Doherty is IBLCC certified. A lot of questions come up from dentistry. Do you have a pediatric dentist or somebody that you might trust well? Think about the people who will be able to help you along the way, occupational therapy, speech therapy, especially for the older patient. If we had a goal, Our goal would be at one point, could we have a shared pre-visit? Could we do comprehensive visits and multi-specialty evaluations? This is my dream. It doesn't mean that it's there yet. And then importantly, coordinated decision making and communication with parents and providers, and that we, if we are going to advocate for this, we are also committed to our outcomes and assessments. And these are my references, and you're more than welcome to all of this. Um, and this is by no means exhaustive, as I said.
