Summary

Ankyloglossia is a congenital alteration of the base of the tongue responsible for some alterations in the development of speech, dentition and other aspects of adult life. For the implementation of the Ankyloglossia Outpatient Intervention Unit (UDIADEAN) in Primary Care, the prevalence of this pathology in the paediatric age of 0 to 14 years was determined. Method: During the first month of work, one of the authors of the study assessed the presence of ankyloglossia in all patients in their quota, attending the Health Center using the Hazelbaker morphological method in children under 6 months and Carmen Fernando in the elderly. Results: During the 17 working days of the month, a total of 443 patients of all age groups were attended. The reasons for consultation in 57 cases were due to a scheduled consultation at the “Control del Nen Sa”, in 76 controls were followed to previous visits with pathology and the rest were medical visits for frequent medical problems: cough, mucus, diarrhoea, vomiting and fever, among others. Of all the patients attended, 295 patients belonged to the paediatricians’ quota and in them an ankyloglossia prevalence of 31% was found.

Ankyloglossia, Tongue tie, Prevalence, Primary Health Care, Screening test, Diagnosis, Breastfeeding.

Complet text

Introduction

Ankyloglossia is a genetic malformation¹ of the floor of the mouth that causes sucking difficulties in infants² and causes various disorders in childhood, adolescence and adults: dyslalia, snoring, sleep apnea, atypical swallowing, skeletal disorders, dental disorders, among others³ .
It has been known since antiquity, although the health interest has been lost with the rise of bottle feeding. At the beginning of the 20th century, this malformation was habitually treated by midwives⁴in newborns. With the increase in breastfeeding has resurfaced this situation in infants, thus producing a significant increase in the demand for health interventions and, as a consequence, an increase in medical publications developing arguments for and against this intervention⁵ .

The prevalence of tongue tie has not been studied as such and the existing discrepancies in some studies in nearby areas show the lack of unanimity in the definition of ankyloglossia as well as for the diagnostic criteria of this malformation.
After one of the authors of the study joined a Primary Care Center as a pediatrician, during the first month of work, the presence of ankyloglossia or disorders related to this difficulty was assessed in all children aged 0 to 14 who attended the the consultation and that they belonged to the quota of patients assigned to him.
The objective of studying the prevalence of ankyloglossia in the area was to calculate an estimate of the situation prior to the creation in the Primary Care Center of an Intervention Unit in Primary Care of Ankyloglossia. (UDIDEAN).

Methods

The existence of ankyloglossia was assessed in all attendees for any reason during the month of November 2015. Hazelbaker’s morphological criteria⁶ were followed in children under 6 months of age and in older adults the existence of ankyloglossia was assessed when tongue mobility was impaired. and following the criteria of Carmen Fernando ⁷.
The medical records of the patients visited with a diagnosis of Ankyloglossia were subsequently obtained and reviewed, and the following variables were recorded: reason for visit, diagnoses other than ankyloglossia, and presence of pathologies related to them and the presence of ankyloglossia.
The exit diagnoses of the children were studied at the end of the visit.

Results:

17 days were worked in the assignment CAP. In another four, the pediatrician was assigned to another health center and the data was not recorded because it was a population with another quota.
The total number of registered children was 443, of which 295 belonged to the main researcher’s quota, 106 were from the same Health Center, but from other quotas. 38 did not show up and 11 were from other clinics. The age range of the children visited with the highest attendance is from 0 to 4 years, in which there are 211, 71.5% of all those visited. Figure 1 shows the distribution by age groups.
Data was collected from 386 of those visited.
The reason for the visit for which the majority of patients with previously scheduled consultations attended was for control and follow-up of pathologies already diagnosed (76 cases), followed by well-child care visits (57 cases) and in 4 cases they were eating problems. The rest registered on the day as medical visits for pathologies of various types with greater or lesser urgency in the presentation: cough, runny nose, diarrhea, vomiting and fever, among others.
The diagnoses obtained at the end of the visit, in addition to ankyloglossia, were as shown in Table 1.
In fig. 2 you can see the number of visits per day, the ankyloglossia found and the daily percentage of this pathology.
According to these data, the prevalence of ankyloglossia in patients attending this health center with an age range between 0 and 14 years is 31.8% (95% CI: 29.8 – 33.76).

Discussion:

In Newborns we do not know the real incidence of this situation. In a study carried out in Israel, they found an incidence of lingual frenulum in 99% of children, although not all had ankyloglossia ⁸. In Brazil, since the mandatory implementation of the Martinelli Tongue test in all newborns, the incidence found in this age group is around 20% (Personal communication).
The data we have from our population does not show selection bias, since the pediatrician’s Curriculum was not known in the population of the area prior to their incorporation and the intention to create an Ankyloglossia Primary Care Intervention Unit (UDIADEAN) as stated in your contract. The individuals who attended the consultation did so for the usual causes in a health center unrelated to the ankyloglossia. The age distribution data is similar to other primary care centers ¹⁰. The ages studied are similar to the population pyramid of the Municipality.
The results of the prevalence of ankyloglossia in our population are higher than those usually found in the literature. In most studies, the figure is 5%, based on and referencing the Messner ¹¹ study carried out in the year 2000, which diagnosed ankyloglossia subjectively.
Other subsequent studies that give prevalence figures were not planned with this objective.
In the study carried out in Asturias where they tried to evaluate the prevalence, the data from the Province studied gave an average of 12%, with areas with a prevalence of 50% and others less than 5%¹² . Prevalence data in older children show a prevalence similar to ours when the prevalence of this situation is specifically studied ¹³.
Our study is in the total child population attending the CAP. The power of the study does not allow comparison with other studies of specific incidences for different ages, although the observed trend is that it confirms those that show a higher prevalence than currently considered.
At UDIADEAN we are studying the prevalence, assessing all newborns with the Martinelli’s tongue test ⁹ and the Hazelbaker Morphology test ⁶, which make it possible to identify ankyloglossia that generates sucking disorders and determines the intervention of this pathological fixation at an early age.
The lack of a unified and consensual diagnostic method is one of the reasons for the variability in the prevalence of this alteration appreciated in the diagnostic opinions of health workers in the different studies carried out.
When one of the four most well-known diagnostic methods is systematically used, the health comment ceases to be a medical opinion ¹⁴, it is a diagnosis and we could compare it scientifically.
The opinion that some health professionals make about the non-existence of ankyloglossia in the consultation, sometimes without palpating the frenulum to assess elasticity or without determining the exit of the tongue outside the lip simply by looking at the baby’s mouth is not enough to determine the existence of this alteration and obviously the felt incidence will be much less than the real one.
In the work of the UDIADEAN Group carried out since 2016, in the frenectomies performed both in the health center and those carried out in the reference hospital with the previous assessment of the Hazelbaker test and giving this positive, we have noticed an improvement in the sucking of babies and decrease in discomfort reported by the mother as seen in other previous studies ¹⁵.
We believe that using some form of diagnosis is a condition prior to concretely determining the incidence and prevalence of this disorder ¹⁶. Unifying the diagnostic criteria for Ankyloglossia will allow us to carry out comparative studies on the prevalence of this pathology in the general population in different areas, which we believe is underdiagnosed.

REFERENCES

[1] Ruiz Guzmán L, Cueva Quiroz T, Rodríguez Bailón N, Rubira Felices L, Peña C, Gabarrell Guiu C. Herencia de la anquiloglosia: de tal palo, tal astilla. Rev Pediatr Aten Primaria. 2019;21.

[2] Campanha SMA, Martinelli RLC, Palhares, DB. Association between ankyloglossia and breastfeeding. CoDAS, 31(1), e20170264. Epub February 25, 2019.https://doi.org/10.1590/2317-1782/20182018264

[3] Hong P, Lago D, Seargeant J, Pellman L, Magit AE, Pransky SM. Defining ankyloglossia: a case series of anterior and posterior tongue ties. Int J Pediatr Otorhinolaryngol. 2010;74:1003-6.

[4] Cullun IM. An old wives’ tale. Br Med J. 1959;2:497-8.

[5] Bin-Nun A, Kasirer YM, Mimouni FB A Dramatic Increase in Tongue Tie-Related Articles: A 67 Years Systematic Review. Breastfeed Med. 2017 Sep;12(7):410-414. doi: 10.1089/bfm.2017.0044.

[6] Hazelbaker AK. The assessment tool for lingual frenulum function (ATLFF). Use in a lactation consultant private practice [Thesis].  Los Angeles: Pacific Oaks College; 1993.

[7] Fernando C. Tongue tie: From confussion to clarity. A guide to the diagnosis and treatment of Ankiloglossia (tongue tie). Sidney: Tandem publications; 1998.

[8] Haham A, Marom R, Mangel L, Botzer E, Dollberg S. Prevalence of breastfeeding difficulties in newborns with a lingual frenulum: a prospective cohort series. Breastfeed Med. 2014 Nov;9(9):438-41. doi: 10.1089/bfm.2014.0040. Epub 2014 Sep 19.

[9] Martinelli RLC, Marchesan IQ, Berretin-Felix G. Protocolo de avaliação do frênulo lingual para bebês: relação entre aspectos anatômicos e funcionais. Rev Cefac. 2013;15:599-610  [en línea] [consultado el 04/05/2019]. Disponible en http://www.scielo.br/pdf/rcefac/v15n3/162-11.pdf

[10] L. A. García Llop, A. Asensi Alcoverro. C. Grafiá Juan, P. Coll Mas. Estudio de la demanda en Atención Primaria pediátrica. An Esp Pediatr 1996;44:469-474. https://www.aeped.es/sites/default/files/anales/44-5-15.pdf

[11] Messner AH¹, Lalakea ML, Aby J, Macmahon J, Bair E. Ankyloglossia: incidence and associated feeding difficulties. Arch Otolaryngol Head Neck Surg. 2000 Jan;126(1):36-9.

[12]González Jiménez D, Costa Romero M, Riaño Galán I, González Martínez MT, Rodríguez Pando MC, Lobete Prieto C. Prevalencia de anquiloglosia en recién nacidos en el Principado de Asturias. An Pediatr (Barc). 2014 Aug;81(2):115-9.

https://www.analesdepediatria.org/es-prevalencia-anquiloglosia-recien-nacidos-el-articulo-S1695403313004347

[13] Gutiérrez Centeno LY. Prevalencia de anquiloglosia en escolares de 6 a 12 años del nivel primario de las escuelas públicas de los municipios de Zacualpa y San Miguel. Tesis de Grado de Cirugía Dental. Universidad de San Carlos. Guatemala 2006. http://biblioteca.usac.edu.gt/tesis/09/09_1833.pdf

[14] Diez V, Ruiz MM, Peña C, Calderón J, González C, Ruiz L. Estudio comparativo de métodos de diagnóstico de anquiloglosia en el lactante. Poster presentado en el XIV Congreso Fedalma. Villafranca del Penedes 2017.  http://www.elfrenillolingual.com

[15]Francis DO, Krishnaswami S, McPheeters M. Treatment of Ankyloglossia and Breastfeeding Outcomes: A Systematic Review. PEDIATRICS Volume 135, number 6, 1-10. 2015. www.pediatrics.org/cgi/doi/10.1542/peds.2015-0658

[16]Hill R¹.Implications of Ankyloglossia on Breastfeeding. Am J Matern Child Nurs. 2019 Mar/Apr;44(2):73-79. doi: 10.1097/NMC.0000000000000501

Este articulo ha sido publicado en “Pediatria Catalana” en  Julio 2021. Referencia:

Ruiz-Guzmán L, Sadino-Vallvé S, Almeda-Ortega J, Basulto-Marset J, Terrazas-Mejía V, Maya-Enero S. Anquiloglòssia com a causa de deshidratació hipernatrèmica neonatal. Pediatr Catalana. 2021;81(2):81-3.

Traducción del catalán por los autrores. Autorizado por lo editores. Original:http://webs.academia.cat/revistes_elect/view_document.php?tpd=2&i=17228

RESUMEN

Introducción. La anquiloglosia es una malformación lingual que puede causar dificultad en los movimientos de la lengua y de la mandíbula de los bebés, y que provoca dolor en los pezones de la madre, una succión débil y una inadecuada transferencia de leche durante las tomas en el pecho. Esto puede ocasionar una pérdida de peso y un destete precoz si no se detecta y no se  trata. Presentamos el caso de dos bebés con anquiloglosia y dificultades en la lactancia materna que se complicaron con una deshidratación hipernatrèmica.

Observación clínica. Ambos bebés sufrieron una deshidratación hipernatrèmica sin signos de infección a los diez días de vida. Ambos tenían anquiloglosia, no tratada en el primer caso, y no  diagnosticada en el segundo. La anquiloglosia del segundo caso se diagnosticó posteriormente, a  raíz de la presentación del primero. La Frenectomía hecha al primer caso permitió la relactació  completa, lo que no se consiguió en el segundo.

Comentarios. Las dificultades que el frenillo sublingual corto puede provocar la lactancia pueden  ocasionar una deshidratación hipernatrèmica grave. Es importante detectar la anquiloglosia para  poder hacer un seguimiento estrecho de estos bebés y tratar los casos sintomáticos. La  exploración del frenillo sublingual debería ser sistemática en la exploración física inicial de todos los recién nacidos. 

Palabras clave: anquiloglosia. Lactancia materna. Hipogalactia. Deshidratación. Hipernatremia. Recién nacido.

Introducción

La pérdida de hasta el 10% del peso de nacimiento se considera normal los primeros días de vida y se debe, entre otras causas, a la adaptación al ambiente aéreo viniendo de un ambiente hídrico (lo  que aumenta las pérdidas por evaporación), la eliminación del exceso de agua del feto y en una  mala técnica de lactancia o en una técnica deficiente durante los primeros tres días de vida¹. El  establecimiento de una adecuada producción de leche puede tardar unos días, aunque este tiempo se acorta si se permite el contacto precoz entre madre y bebé, y se hacen tomas frecuentes y con  una succión adecuada². Los primeros días posparto, la madre produce calostro en pequeñas cantidades, adecuadas a la capacidad gástrica del bebé y en su necesidad de nutrientes. La  lactogènesis II, conocida como «subida de leche», suele comenzar al cabo de tres o cuatro días.  El dolor y el estrés materno pueden dificultar el establecimiento de la lactancia, así como la posición incorrecta en el pecho y un apego inadecuado².

El anquiloglosia (o frenillo sublingual corto) puede dificultar que el bebé se agarre al pecho, y provocar dolor a la madre, una succión débil y una inadecuada aportación de leche; esto afecta la nutrición del lactante, y le ocasiona una pérdida de peso excesiva y un destete precoz^3-4. Más  adelante, la anquiloglosia también puede provocar problemas dentales y del habla, problemas para la deglución de sólidos, ronquidos, apneas y dificultad para la respiración nasal debido a un paladar ojival ^3-4.

Presentamos dos casos de anquiloglosia neonatal que ilustran la posible relación entre las  dificultades para la alimentación y la deshidratación hipernatrèmica.

Observación clínica

Caso 1
Primera hija de una madre sana y sin antecedentes de interés, nacida a cabo mediante un parto  instrumentado con espátulas. Apgar 9-10-10, peso de 3.300 g. Después de un puerperio sin  incidencias, se dió de alta a los tres días de vida con un peso de 2.900 g (pérdida de peso> 10%) y lactancia materna exclusiva. A los diez días fue llevada al pediatra por una somnolencia marcada, por lo que fue derivada al servicio de urgencias hospitalario: presentaba una afectación del estado general (Hipoactiva aunque reactiva), pérdida de peso del 24% (pesaba 2.400 g) y una  deshidratación grave (Fontanela anterior hundida, ojerosa y con signo del pliegue). Los análisis de sangre mostraban una acidosis metabólica (pH 7,23, pCO2 45,8 mmHg, bicarbonato 17,2 mmol / L, exceso de base -7,5 mmol / L) y una hipernatremia grave (176 mmol / L), sin signos de infección.
Los cultivos de sangre, de líquido cefalorraquídeo (LCR) y de orina fueron negativos. Se inició una reposición intravenosa de líquidos, con buena respuesta clínica y normalización de la exploración física y neurológica. Se dió de alta cinco días después con un peso de 2.740 g y lactancia artificial. A los veinticinco días de vida y con 3.070 g, se derivó a una consulta de lactancia. Durante las tomas se sentían chasquidos, había obstrucción nasal y los pezones de la madre enrojecidos. Se
observó un frenillo sublingual de tipo 3 según la clasificación de Coryllos⁵, con anquiloglosia y con una puntuación del aspecto según Hazelbaker⁶ de 6, así como una retrognàtia y la presencia de un callo de succión. (Fig. 1).

Se propuso la relactación a la madre: se le dieron consejos para mejorar la posición en el pecho ayudando con sonda en el pecho. Se trató con domperidona y se empezó a sacar leche con un sacaleches y ponerse al bebé al pecho con pezoneras porque no se agarraba. Con todo, consiguió una relactación completa en un mes. Se practicó una Frenectomía al bebé a los dos meses, con lo cual mejoró la succión. La alimentación complementaria se inició a los seis meses, con una buena tolerancia a los sólidos. El desarrollo a los ocho meses era normal.

Caso 2

Primera hija de una madre sana y sin antecedentesde interés, fruto de una gestación a termino sin incidencias. Apgar 9-10-10, peso de 2.950 g. Después de un puerperio inmediato normal, se le dio el alta a los tres días de vida con un peso de 2.680 g (pérdida del 9,1%) y lactancia materna exclusiva a demanda. A los nueve días de vida la vio el pediatra. Pesaba 2.530 g (pérdida del 14% del peso), se veía francamente deshidratada y con un aspecto general regular, pero reactiva. Se derivo al  Hospital. Los análisis de sangre mostraban una hipernatremia (152 mmol / L), sin signos de infección. Los cultivos fueron negativos. Recibió fluidoterapia intravenosa con una buena respuesta clínica y neurológica. Se dio el alta los veintiún un días de vida, con lactancia artificial. El día posterior al alta su pediatra propuso a la madre la relactació, que comenzó a los veintiocho días con extracciones frecuentes y tratamiento con domperidona oral. Todo y que se corrigió la posición en el pecho, las molestias y el enrojecimiento en los pezones persistían. Un mes después el bebé recibía lactancia materna prácticamente exclusiva, con una toma nocturna de 120 mL de leche adaptada. Continuó así hasta que la madre decidió dejar la lactancia a los nueve meses.

A los seis meses se  introdujo la alimentación complementaria, con regurgitaciones frecuentes y problemas con la deglución.

A los cuatro años y, a raíz del diagnóstico del caso 1, se la citó para hacer una exploración. El desarrollo era normal. Se observaba una retrognàtia, una boca torcida al abrirla, un paladar ojival, una muesca en la punta de la lengua y una rotación de los incisivos centrales inferiores (Fig. 2). se
diagnosticó de un frenillo tipo 3 según Coryllos⁵ con componente submucoso. El logopeda detectó también trastornos del habla.

Discusión

Los dos casos presentados muestran muchas similitudes y apoyan la hipótesis de que la anquiloglosia puede condicionar una mala técnica de lactancia y una deshidratación hipernatrèmica neonatal¹. Un agarre pobre, la lactancia con horarios prefijados, las presiones familiares y sociales, y la anquiloglosia se han asociado con el fracaso de la lactancia y la deshidratación neonatal^1,4. El dolor durante las tomas y las dificultades para la extracción de leche que puede ocasionar la anquiloglosia pueden provocar una pérdida de peso considerable al recién nacido incluso cuando la técnica de lactancia es adecuada^3-4.

La Hipogalactia primaria es muy rara, y la producción de leche suele aumentar rápidamente mejorando la técnica de lactancia y la postura de agarre en el pecho, como demuestran los casos presentados.

Ambos casos eran primeros hijos de madres sin experiencia en lactancia, sin apoyo familiar cercano y con un gran deseo de amamantar. Ambos bebés ingresaron por una deshidratación hipernatrèmica los diez días de vida y las dos madres pudieron relactar.

Los dos casos tenían un frenillo sublingual corto y no tenían otras causas aparentes de deshidratación. La Frenectomía del primer caso permitió una relactación completa. En el segundo, las molestias maternas continuaron, lo que precipitó el destete a los nueve meses. En ambos casos, la presencia de anquiloglosia y el retraso de la ayuda a la lactancia tuvieron un papel importante en la deshidratación de los bebés.

El tipo de frenillo sublingual era similar en ambos casos según la tipificación de Coryllos⁵ y el diagnóstico del segundo caso fue más clínico al no poder utilizar el método de Hazelbaker⁶, destinado lactantes. La utilización de protocolos de diagnóstico más actuales, como el “Test de la Lengüita“⁷ o el “Bristol Tongue tie Assessment Tool (BTAT)“⁸ en recién nacidos y lactantes, y el “Test de la Lengua para niños mayores“⁹, permitirían identificar de manera objetiva la anquiloglosia, que posiblemente tiene una mayor incidencia de la supuesta previamente¹⁰.

Probablemente, si el hospital donde nacieron hubiera formado parte de la red Iniciativa Hospital Amigo los Niños, esta situación no se hubiera producido.

En resumen, se recomienda tener en cuenta la anquiloglosia como causa de una deshidratación neonatal grave en bebés con dificultades en la lactancia porque, además, el tratamiento (la Frenectomía) es sencillo. Hay que explorar todos los bebés buscando si tienen o no un frenillo  lingual corto y tratar los que sean sintomáticos según protocolos adecuados.

Agradecimientos

Queremos agradecer al Dr. Alejandro Allepuz, a la Dra. Natalie Garcia, a la Dra. Esther Grunnis y al DUE Norberto Murillo sus comentarios, aportaciones y revisiones.

REFERENCES

1. Mujawar NS, Jaiswal AN. Hypernatremia in the Neonate: Neonatal Hypernatremia and Hypernatremic Dehydration in Neonates Receiving Exclusive Breastfeeding. Indian J Crit Care Med. 2017;21(1):30-3.
2. Lactancia materna. Documento de Consenso. Sociedad Española de Ginecología y Obstetricia. Accessible a la xarxa [data de consulta: 6-11-2020]. Disponible a: https://www.aeped.es/sites/default/files/11-sego_doc_de_consenso.pdf.
3. Rowan-Legg A. Ankyloglossia and breastfeeding. Paediatr Child Health. 2015;20(4):209-18.
4. Kumar M, Kalke E. Tongue-tie, breastfeeding difficulties and the role of Frenotomy. Acta Paediatr. 2012;101(7):687-9.
5. Coryllos EW, Genna CW, Salloum AC. Congenital tongue-tie and its impact on breastfeeding. American Academy of Pediatrics. Section on breastfeeding; 2004. Accessible a la xarxa [data de consulta: 28-9-2020]. Disponible a: https://www.researchgate.net/publication/301346077_Congenital_tongue-tie_and_its_impact_on_breastfeeding.
6. Walsh J, Tunkel D. Diagnosis and Treatment of Ankyloglossia in Newborns and Infants: A Review. JAMA Otolaryngol Head Neck Surg. 2017;143(10):1032-9.
7. Lopes de Castro Martinelli R, Queiroz Marchesan I, Lauris JR, Marques Honório H, Jordão Gusmão R, Berretin-Felix G. Validade e confiabilidade da triagem: “teste da linguinha”. Rev CEFAC [revista electrónica]. 2016;18(6):1323-31.
8. Ingram J, Johnson D, Copeland M, Churchill C, Taylor H, Emond A. The development of a tongue assessment tool to assist with tongue-tie identification. Arch Dis Child Fetal Neonatal Ed. 2015;100(4):F344-8.
9. Araujo MDCM, Freitas RL, Lima MGS, Kozmhinsky VMDR, Guerra CA, Lima GMS, et al. Evaluation of the lingual frenulum in newborns using two protocols and its association with breastfeeding. J Pediatr (Rio J). 2020;96(3):379-85.
10. González Jiménez D, Costa Romero M, Riaño Galán I, González Martínez MT, Rodríguez Pando, Lobete Prieto C. Prevalencia de anquiloglosia en recién nacidos en el Principado de Asturias. An Pediatr (Barc). 2014;81(2):115-9.

This article has been published in “Pediatria Catalana” in July 2021. Reference:

Ruiz-Guzmán L, Sadino-Vallvé S, Almeda-Ortega J, Basulto-Marset J, Terrazas-Mejía V, Maya-Enero S. Anquiloglòssia com a causa de deshidratació hipernatrèmica neonatal. Pediatr Catalana. 2021;81(2):81-3.

English translation by the authors. Authorized by editors. Original:http://webs.academia.cat/revistes_elect/view_document.php?tpd=2&i=17228

ABSTRACT

Introduction.  Ankyloglossia as a cause of neonatal hypernatremic dehydration

Ankyloglossia is a lingual malformation that may cause difficulty in tongue and mandibular movements in the newborn. It results in nipple pain in the mother, weak suction and inadequate milk transfer during breastfeeding. , Ankyloglossia may lead to weight loss and an early cessation of breastfeeding if undetected and untreated.
We report the case of two tongue-tied neonates with breastfeeding difficulties that presented with neonatal hypernatremic dehydration.

Case report. Both infants experienced hypernatremic dehydration, with no signs of infection, at 10 days of life. Both had ankyloglossia, which was untreated in case 1 and undiagnosed in case 2. The undetected ankyloglossia of case 2 was diagnosed a posterior after the identification of case 1. The frenotomy eventually performed in case 1 enabled complete re-lactation, which was not achieved in case 2.

Comments. Breastfeeding difficulties caused by tongue-tie may lead to severe hypernatremic dehydration. It is important to detect
the presence of ankyloglossia in all affected newborns so that symptomatic cases can be closely monitored and treated. Screening for ankyloglossia should be routinely performed at the initial physical examination of all neonates.

Keywords: Ankyloglossia. Breastfeeding. Hypogalactia. Hypernatremia. Dehydration. Newborn.

Introduction

Losing up to 10% of birth weight is considered normal in newborn infants. Causes of such weight loss are associated with adaptation, elimination of the excess water of the foetus and insufficient or poor breastfeeding during the first 3 days of life¹. The establishment of adequate milk production can take time, although this time span decreases with immediate infant-breast contact as well as frequent feedings and an appropriate latch². In the first few days postpartum, the mother produces colostrum which is delivered to the infant in small amounts, suited to his stomach capacity and need for nutrients. Lactogenesis II, the copious milk supply, usually starts around day 3 or 4. Pain and maternal stress may hinder milk transfer to the infant, which in turn will hinder milk supply. Both are symptoms and causes of poor positioning on the breast and of an inadequate latch².

Ankyloglossia, or tongue-tie, can generate difficulties in latching, causing pain to the mother, a weak suction and an inadequate milk transfer, hence limiting the nourishment of the child and leading to weight loss and an early cessation of breastfeeding^3-4. It can also produce dental and speech problems, disorders swallowing solids, snoring, apnoea and difficult nasal breathing due to a V-shaped palate in childhood and adulthood^3-4.

We present two cases of ankyloglossia that illustrate the possible correlation between feeding difficulties and hypernatremic dehydration.

CASE 1:

A first-born daughter with no family history of illness was born at term with an instrumented delivery with an Apgar score of 9-10-10, birth weight of 3300 g and length of 50 cm. After a normal puerperal period, she was discharged at the age of three days with a weight of 2900 g (>10% weight loss). The infant was exclusively breastfed on demand. At 10 days of life, the infant was taken to her paediatrician presenting marked sleepiness. Was referred to the emergency room in a children’s hospital for admission due to dehydration. She weighed 2400 g (having lost 24% of the birth weight) was severely dehydrated, had a poor general condition and was hypoactive yet reactive. Blood tests showed: metabolic acidosis and hypernatremia 176 mmol/L, with no signs of infection. CSF, blood and urine cultures were negative. An intravenous hydration treatment was administered with a good clinical response. Physical and neurological courses were normal. She was discharged home 5 days later with a weight of 2740 g and the diagnoses of maternal hypogalactia and hypernatremic dehydration. Breastfeeding advice was given prior to discharge and at discharge, she was bottle-fed.

The infant was referred to an experienced breastfeeding consultant at 25 days of life. She started to use a milk pump and nipple shields because latching was difficult. She weighed 3070 g; Feeding was evaluated, and the mother was given advice on how to improve positioning. The consultant observed clicking while sucking; nasal obstruction and reddening of the nipple. Type 3 ankyloglossia (Coryllos⁵) with a submucosal component was observed, and Hazelbaker’s assessment for appearance was 6 (Image 1).

A re-lactation plan was proposed, consisting of frequent milk extraction and a catheter was placed on the breast for feeding. Moreover, the mother was treated with domperidone and was given instructions and support to uphold the correct latch positioning. She achieved complete re-lactation in one month’s time. The infant underwent frenotomy at two months, leading to an improvement in the feeding technique.

Complimentary feeding began at 6 months of age, and the infant displayed good tolerance to solid foods¹. At 8 months, the infant presented an adequate development.

CASE 2

A first-born daughter with no family history of illness was born at term after an uneventful pregnancy with an Apgar score of 9-10, a birth weight of 2950 g and a length of 50 cm. After a normal puerperal period, she was discharged three days later with a weight of 2680g  (9,1% weight lost). The infant was exclusively breastfed on demand, and the parents noted her very calm disposition. At 9 days of age, the paediatrician saw her and referred her to the children’s hospital for admission with the diagnosis of dehydration. She weighed 2530 g (14% weight loss from birth), appeared severely dehydrated, had a poor general condition yet was reactive. Blood tests showed metabolic acidosis and hypernatremia 152mmol/L with no signs of infection, and CSF, blood and urine cultures remained negative. An intravenous hydration treatment was administered with a good clinical response. The clinical course, including the neurological course, was normal. There was no mention of breastfeeding or of any health education in her medical report. The infant was discharged at the age of 21 days with bottle feeding.

Twenty-four hours after discharge, at the appointment with the paediatrician and lactation specialist, there was a proposal for re-lactation, which began at the age of 28 days with frequent extraction plus oral domperidone to the mother. The position was corrected frequently, and the mother continued to have nipple discomfort and redness, although there was no cracking and she was progressing appropriately. One month after beginning re-lactation, the infant was almost exclusively breastfed, receiving only one 120-cc bottle of formula at night. This continued until the mother decided to discontinue breastfeeding at 9 months. At 6 months of age, complementary feeding was introduced, which was difficult, due to frequent spitting and swallowing difficulty.

At the age of 4 years, the child was called in for a medical examination following case 1, due to a possible ankyloglossia that had not been detected initially. She had normal development, facial asymmetry, retrognathia, a twisted mouth when opening, a V-shaped palate, a groove on the tongue and rotated lower central incisors. This was a case of type 3 ankyloglossia (Coryllos²) with a submucosal component (Image 2). Speech disabilities were noted by a speech therapist.

DISCUSSION

The two cases described present a great deal of similarity. Both were treated at the same hospital. The undetected ankyloglossia of case 2 that was diagnosed at a later age, following the presentation of case 1, supports our hypothesis of ankyloglossia as a cause of neonatal hypernatremic dehydration¹.

An inadequate breastfeeding technique has been described as a cause of neonatal hypernatremic dehydration. Poor latching, set nursing times, family and social pressures and ankyloglossia have all been associated with breastfeeding failure and neonatal dehydration^1,4. Ankyloglossia is associated with pain during nursing, difficult milk extraction even when the technique is correct, and considerable weight loss in the neonate^3-4. Upon discharge from the university hospital to which both cases were referred, no causes were cited beyond the mother’s hypogalactia, which both discharge reports mention. Primary hypogalactia occurs rarely in nature. Moreover, milk production resumes with a correct positioning on the breast and appropriate support, as displayed in both cases. Both mothers re-lactated; however, in case 2, re-lactation was not complete despite an appropriate follow-up and support.

The symptoms and signs in the cases described are vastly similar: both were first-time parents with little breastfeeding experience, far from their families and had a keen desire to nurse their infants. Both neonates were diagnosed with hypernatremic dehydration at 10 days of life; both were re-lactated despite the previous experience; and both had ankyloglossia, undiagnosed in case 2 and treated in case 1. The frenotomy performed in case 1 enabled a complete re-lactation that was not achieved in case 2, in which nipple discomfort continued and contributed to the ending of breastfeeding at 9 months. We believe that in both cases, ankyloglossia, which was not detected in case 2 and was identified thanks to its similarity with case 1, as well as the delay in support for breastfeeding, played an important role in the dehydration that these infants suffered.

Tongue-tie was similar in both following Coryllos Calssification⁵. Case 2 diagnosis was clinic because Hazelbaker test is oriented to infants.

Use diagnosis protocols more actuals like “Test de la lenguita”⁷ or Bristol Tongue-tie Assessment Tool (BTAT)⁸ in new-borns or infants and the “Test de la Lengua”⁹ in infants and older people could allow us to identify in an objective form the ankyloglossia that probably could have a higher prevalence than previously supposed.

We believe that if the delivery hospital has had the Baby Friendly award this situation could not be presented.

In summary, we recommend that ankyloglossia be taken into account as a cause of severe neonatal dehydration due to breastfeeding problems, with a minor and easy surgical solution for the recovery of breastfeeding. Hence, from our point of view, ankyloglossia should be searched for in the initial physical examination in all neonates.

Acknowledgments

Thanks to Dr Alejandro Allepuz, Dra Natalie Garcia,  Dra Esther Grunnis and DUI Norberto Murillo for their comments and revision.

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