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Laryngomalacia: The role of gender and ethnicity
International Journal of Pediatric Otorhinolaryngology 75 (2011) 1562–1564
Contents lists available at SciVerse ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Laryngomalacia: The role of gender and ethnicity Natalie E. Edmondson *, John P. Bent III, Christine Chan Department of Otorhinolaryngology – Head and Neck Surgery, Children’s Hospital at Montefiore, Albert Einstein College of Medicine, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 1 August 2011 Accepted 7 September 2011 Available online 28 September 2011
Introduction: The vast majority of infants in published studies regarding laryngomalacia are Caucasian. These studies suggest affected infants are likely to be male, of term pregnancies, and not of low birth weight. Our study seeks to identify possible associations among different genders and ethnicities with laryngomalacia as well as differences between our diverse population and published Caucasianpredominant studies. Methods: Data was collected by chart review for seventy-eight children diagnosed with laryngomalacia before the age of 5, prospectively entered into a database at our academic, tertiary-care hospital from 1/16/2008 to 10/15/2010. Data was compared to expected values from the 2009 census data for the Bronx as well as published data in the literature for multiple factors, including gender, ethnicity, low birth weight, and prematurity. Results: There were 45 male and 33 female infants in our study. Twenty-five children were AfricanAmerican, 17 Hispanic, 12 multi-racial, and 4 Caucasian. Eighteen infants were premature, and 21 were of low birth weight. Twenty-eight percent of our infants had neurologic comorbidity at diagnosis and 30% received surgical treatment. Discussion: Although larger studies are required, we have preliminary evidence that information learned from Caucasian-predominant studies may not apply to all patients of laryngomalacia. When ethnically diverse infants are represented, a strong association between male gender and laryngomalacia does not appear to exist. Our data suggests that premature African-American and Hispanic infants are at greater risk for laryngomalacia. In addition, African-American infants of all gestational ages may be at greater risk. Low birth weight may be a strong predictor of laryngomalacia regardless of the patient’s gender or ethnicity. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Laryngomalacia Pediatric Airway Gender Ethnicity
1. Introduction and background Laryngomalacia is the most common congenital laryngeal abnormality, accounting for over 60% of all cases of congenital stridor [1]. Literally meaning ‘‘soft larynx,’’ this condition refers to a dynamic prolapse of the supraglottic structures during inspiration with endoscopic findings such as shortened aryepiglottic folds and redundant soft tissue, with or without supraglottic edema [3,7]. Affected infants often present with inspiratory stridor within the first 10 days of life that typically resolves spontaneously by 12– 18 months of age. Approximately 10% of cases warrant surgical intervention due to failure to thrive, cyanosis, cor pulmonale, or apnea [5,6]. As disease etiology is still largely unknown, multiple theories of etiology exist including neurologic and anatomic. Gastroesophageal reflux (GERD) is a well-known, strongly associated comorbidity [6,7].
Prior published studies in the literature largely consist of Caucasian infants, ranging from 77 to 93% of subjects. These Caucasian-predominant studies noted a male predilection in their laryngomalacia study groups (58–76% male) and also suggested that affected infants are likely to be of term pregnancies and not of low birth weight [6–8]. No literature exists describing laryngomalacia in ethnically diverse infants. Our study seeks to identify possible associations and patterns of disease in patients diagnosed with laryngomalacia among different genders and ethnicities. We will attempt to find associations between different genders and ethnicities with prematurity, low birth weight, neurologic comorbidity, and surgical treatments received. Lastly we will look to identify differences between our ethnically diverse Bronx population and published Caucasianpredominant studies.
2. Methods * Corresponding author at: Albert Einstein College of Medicine, Children’s Hospital at Montefiore, 3400 Bainbridge Ave, 3rd Floor, Bronx, NY 10467, United States. Tel.: +1 718 920 4267; fax: +1 718 405 9014. E-mail address: [email protected] (N.E. Edmondson). 0165-5876/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2011.09.008
Seventy-eight children prospectively entered into a database by three collaborating Pediatric Otolaryngologists at our academic, tertiary-care children’s hospital from 1/16/2008 to 10/15/2010
N.E. Edmondson et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 1562–1564 Table 1 Gender.
Males Females Total p-Value
1563
Table 2 Laryngomalacia among ethnicities. Observed
Expected
Ethnicity
Observed
Expected
p-Value
45 33 78 0.08
36.58 39.55
African-American Caucasian Hispanic Multi-racial
25 4 17 12
20.06 13.51 30.20 1.65
0.17 0.0032 0.00058 3.18 10
were identified. Data was collected by chart review. Data points included gender, ethnicity, age at diagnosis, birth weight, gestational age at birth, presence of neurologic comorbidity, and treatments received. Ethnicity data was available for 58 children, and all children were diagnosed with laryngomalacia before the age of five. Children in the database presented as both inpatient and emergency room consults as well as outpatient referrals. The ethnicity of each child was designated by the parents or legal guardians and was subsequently recorded in the patient’s chart. The selection of ‘‘multi-racial’’ was not further specified by parents, but this probably represents a combination of the three ethnicities most prevalent in our population: Hispanic, African-American, and to a less degree, Caucasian. Data was compared to expected values for our population from the 2009 census data for the Bronx community by using chi-square tests to generate p values for all factors, including gender, ethnicity, low birth weight, prematurity, neurologic comorbidity, and surgical treatment received. Our rates of low birth weight and prematurity were compared to expected rates published in the literature [2,4]. 3. Results When compared to census data for the Bronx population, there was no significant difference in the number of male and female laryngomalacia patients in our study (Table 1). When the data was analyzed for gender according to ethnicity, there was still no significant difference between genders (Fig. 1). In order to examine the ethnic distribution of the laryngomalacia patients in our study, our data was compared to the expected values based on the ethnic distribution of children in the Bronx community. There were fewer Caucasian and Hispanic children than expected in our study group of laryngomalacia patients. There were more multi-racial children than expected (Table 2). When compared to expected rates of prematurity (defined as less than 37 weeks [4]), there were significantly more premature Hispanic and African-American babies in our laryngomalacia study group. There was no significant difference between the number of premature Caucasian or multi-racial infants and expected rates of prematurity for these groups. Similarly, when compared to expected rates of low birth weight (defined as less than
Fig. 1. Gender by ethnicity.
16
2500 g2), there were more babies of low birth weight than expected in all ethnic groups. There was no significant difference in gender distribution in the low birth weight or premature groups (Fig. 2). Twenty-eight percent of our babies had known neurologic disease at diagnosis. There were no significant differences among genders or ethnic groups in neurologic comorbidity at diagnosis. Thirty percent of the children in our study ultimately underwent supraglottoplasty without apparent selection bias between ethnicities or genders. 4. Discussion Previously published studies that include the demographics of laryngomalacia patients document a male predominance. In our cohort of ethnically diverse babies, male gender was less strongly associated with laryngomalacia and did not reach statistical significance even when comparing by ethnic group. These calculations include an insufficient number of Caucasian infants (4) to meaningfully impact the results, so we can neither refute nor support the accepted relationship between male gender and laryngomalacia in Caucasians. The inclusion of non-Caucasian children may have diluted the high percentage of males previously described in studies such as this. When analyzing the ethnic distribution of this diverse cohort, there were fewer Caucasian and Hispanic children and more multiracial children than expected. Given that many of our multi-racial children are likely to have some African ancestry, our findings give preliminary indication that laryngomalacia may occur with greater frequency among African-Americans than other racial groups. A substantially larger series with specific breakdown of each ‘‘multiracial’’ designation would be required to substantiate this observation. Compared to the expected rates of prematurity, there were significantly more premature Hispanic and African-American babies in our laryngomalacia study group. Given the association between neurologic disease and laryngomalacia, it is intuitive that premature babies would have higher rates of laryngomalacia. This, however, has not been previously observed in Caucasianpredominant studies. Perhaps the Caucasian larynx is less vulnerable to prematurity or other co-factors, such as laryngopharyngeal reflux, which may result in a more symptomatic nonCaucasian larynx.
Fig. 2. Prematurity and low birth weight.
1564
N.E. Edmondson et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 1562–1564
In addition, when comparing to expected rates of low birth weight, there were more babies of low birth weight than expected in all ethnic groups. Neither the low birth weight nor premature groups showed significant differences in gender distribution. The association of laryngomalacia and low birth weight may also be explained by impaired neurologic development, but this trend has not been shown in Caucasian populations. There were no significant differences among genders or ethnic groups in neurologic comorbidity at diagnosis or in surgical treatment received in our study. The 30% rate of supraglottoplasty may be inflated in our database accrual method in which surgical patients may be more likely to gain attention than less affected children. Furthermore, at our medical center, sleep studies are often obtained early in the laryngomalacia work-up, providing more tangible evidence to proceed with surgical treatment. Finally, we believe that supraglottoplasty is a very effective operation, and consequently, this treatment option is often recommended for patients at our institution who might otherwise be considered borderline surgical candidates. 5. Conclusion The demographic evidence presented herein suggests premature African-American and Hispanic infants are at increased risk for laryngomalacia. The disease process may also occur more often in the African-American population regardless of gestational age at birth. Furthermore, low birth weight may be a stronger predictor of
laryngomalacia than previously appreciated in both genders and all ethnicities. In ethnically diverse populations, male gender appears less strongly associated with laryngomalacia than in previously reported studies of Caucasian populations. Information learned from previously published Caucasianpredominant studies in the literature might not fully apply to diverse populations. Better knowledge of these patients will aid in understanding disease etiology, making accurate diagnoses, choosing optimal treatment plans, and predicting future prognoses for all infants affected. References [1] S.J. Daniel, The upper airway: congenital malformations, Paediatr. Respir. Rev. 7S (2006) S260–S263. [2] B.E. Hamilton, J.A. Martin, S.J. Ventura, Births: Preliminary Data for 2009. National Vital Statistics Reports Web Release, vol. 59, no. 3, National Center for Health Statistics, Hyattsville, MD, 2010. [3] L.D. Holinger, R.J. Konior, Surgical management of severe laryngomalacia, Laryngoscope 99 (1989) 136–142. [4] R.L. Goldenberg, J.F. Culhane, J.D. Iams, R. Romero, Epidemiology and causes of preterm birth, Lancet 371 (2008) 75–84. [5] J.C. Groblewski, R.K. Shah, G.H. Zalzal, Microdebrider-assisted supraglottoplasty for laryngomalacia, Ann. Otol. Rhinol. Laryngol. 118 (2009) 592–597. [6] D.R. Olney, J.H. Greinwald, J.R. Smith, R.J.N.M. Bauman, Laryngomalacia and its treatment, Laryngoscope 109 (11) (1999) 1770–1775. [7] D.M. Thompson, Abnormal sensorimotor integrative function of the larynx in congenital laryngomalacia: a new theory of etiology, Laryngoscope 117 (2007) 1–33. [8] J. Bent, Pediatric laryngotracheal obstruction: current perspectives on stridor, Laryngoscope 116 (7) (2006) 1059–1070.
Contents lists available at SciVerse ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Laryngomalacia: The role of gender and ethnicity Natalie E. Edmondson *, John P. Bent III, Christine Chan Department of Otorhinolaryngology – Head and Neck Surgery, Children’s Hospital at Montefiore, Albert Einstein College of Medicine, United States
A R T I C L E I N F O
A B S T R A C T
Article history: Received 1 August 2011 Accepted 7 September 2011 Available online 28 September 2011
Introduction: The vast majority of infants in published studies regarding laryngomalacia are Caucasian. These studies suggest affected infants are likely to be male, of term pregnancies, and not of low birth weight. Our study seeks to identify possible associations among different genders and ethnicities with laryngomalacia as well as differences between our diverse population and published Caucasianpredominant studies. Methods: Data was collected by chart review for seventy-eight children diagnosed with laryngomalacia before the age of 5, prospectively entered into a database at our academic, tertiary-care hospital from 1/16/2008 to 10/15/2010. Data was compared to expected values from the 2009 census data for the Bronx as well as published data in the literature for multiple factors, including gender, ethnicity, low birth weight, and prematurity. Results: There were 45 male and 33 female infants in our study. Twenty-five children were AfricanAmerican, 17 Hispanic, 12 multi-racial, and 4 Caucasian. Eighteen infants were premature, and 21 were of low birth weight. Twenty-eight percent of our infants had neurologic comorbidity at diagnosis and 30% received surgical treatment. Discussion: Although larger studies are required, we have preliminary evidence that information learned from Caucasian-predominant studies may not apply to all patients of laryngomalacia. When ethnically diverse infants are represented, a strong association between male gender and laryngomalacia does not appear to exist. Our data suggests that premature African-American and Hispanic infants are at greater risk for laryngomalacia. In addition, African-American infants of all gestational ages may be at greater risk. Low birth weight may be a strong predictor of laryngomalacia regardless of the patient’s gender or ethnicity. ß 2011 Elsevier Ireland Ltd. All rights reserved.
Keywords: Laryngomalacia Pediatric Airway Gender Ethnicity
1. Introduction and background Laryngomalacia is the most common congenital laryngeal abnormality, accounting for over 60% of all cases of congenital stridor [1]. Literally meaning ‘‘soft larynx,’’ this condition refers to a dynamic prolapse of the supraglottic structures during inspiration with endoscopic findings such as shortened aryepiglottic folds and redundant soft tissue, with or without supraglottic edema [3,7]. Affected infants often present with inspiratory stridor within the first 10 days of life that typically resolves spontaneously by 12– 18 months of age. Approximately 10% of cases warrant surgical intervention due to failure to thrive, cyanosis, cor pulmonale, or apnea [5,6]. As disease etiology is still largely unknown, multiple theories of etiology exist including neurologic and anatomic. Gastroesophageal reflux (GERD) is a well-known, strongly associated comorbidity [6,7].
Prior published studies in the literature largely consist of Caucasian infants, ranging from 77 to 93% of subjects. These Caucasian-predominant studies noted a male predilection in their laryngomalacia study groups (58–76% male) and also suggested that affected infants are likely to be of term pregnancies and not of low birth weight [6–8]. No literature exists describing laryngomalacia in ethnically diverse infants. Our study seeks to identify possible associations and patterns of disease in patients diagnosed with laryngomalacia among different genders and ethnicities. We will attempt to find associations between different genders and ethnicities with prematurity, low birth weight, neurologic comorbidity, and surgical treatments received. Lastly we will look to identify differences between our ethnically diverse Bronx population and published Caucasianpredominant studies.
2. Methods * Corresponding author at: Albert Einstein College of Medicine, Children’s Hospital at Montefiore, 3400 Bainbridge Ave, 3rd Floor, Bronx, NY 10467, United States. Tel.: +1 718 920 4267; fax: +1 718 405 9014. E-mail address: [email protected] (N.E. Edmondson). 0165-5876/$ – see front matter ß 2011 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2011.09.008
Seventy-eight children prospectively entered into a database by three collaborating Pediatric Otolaryngologists at our academic, tertiary-care children’s hospital from 1/16/2008 to 10/15/2010
N.E. Edmondson et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 1562–1564 Table 1 Gender.
Males Females Total p-Value
1563
Table 2 Laryngomalacia among ethnicities. Observed
Expected
Ethnicity
Observed
Expected
p-Value
45 33 78 0.08
36.58 39.55
African-American Caucasian Hispanic Multi-racial
25 4 17 12
20.06 13.51 30.20 1.65
0.17 0.0032 0.00058 3.18 10
were identified. Data was collected by chart review. Data points included gender, ethnicity, age at diagnosis, birth weight, gestational age at birth, presence of neurologic comorbidity, and treatments received. Ethnicity data was available for 58 children, and all children were diagnosed with laryngomalacia before the age of five. Children in the database presented as both inpatient and emergency room consults as well as outpatient referrals. The ethnicity of each child was designated by the parents or legal guardians and was subsequently recorded in the patient’s chart. The selection of ‘‘multi-racial’’ was not further specified by parents, but this probably represents a combination of the three ethnicities most prevalent in our population: Hispanic, African-American, and to a less degree, Caucasian. Data was compared to expected values for our population from the 2009 census data for the Bronx community by using chi-square tests to generate p values for all factors, including gender, ethnicity, low birth weight, prematurity, neurologic comorbidity, and surgical treatment received. Our rates of low birth weight and prematurity were compared to expected rates published in the literature [2,4]. 3. Results When compared to census data for the Bronx population, there was no significant difference in the number of male and female laryngomalacia patients in our study (Table 1). When the data was analyzed for gender according to ethnicity, there was still no significant difference between genders (Fig. 1). In order to examine the ethnic distribution of the laryngomalacia patients in our study, our data was compared to the expected values based on the ethnic distribution of children in the Bronx community. There were fewer Caucasian and Hispanic children than expected in our study group of laryngomalacia patients. There were more multi-racial children than expected (Table 2). When compared to expected rates of prematurity (defined as less than 37 weeks [4]), there were significantly more premature Hispanic and African-American babies in our laryngomalacia study group. There was no significant difference between the number of premature Caucasian or multi-racial infants and expected rates of prematurity for these groups. Similarly, when compared to expected rates of low birth weight (defined as less than
Fig. 1. Gender by ethnicity.
16
2500 g2), there were more babies of low birth weight than expected in all ethnic groups. There was no significant difference in gender distribution in the low birth weight or premature groups (Fig. 2). Twenty-eight percent of our babies had known neurologic disease at diagnosis. There were no significant differences among genders or ethnic groups in neurologic comorbidity at diagnosis. Thirty percent of the children in our study ultimately underwent supraglottoplasty without apparent selection bias between ethnicities or genders. 4. Discussion Previously published studies that include the demographics of laryngomalacia patients document a male predominance. In our cohort of ethnically diverse babies, male gender was less strongly associated with laryngomalacia and did not reach statistical significance even when comparing by ethnic group. These calculations include an insufficient number of Caucasian infants (4) to meaningfully impact the results, so we can neither refute nor support the accepted relationship between male gender and laryngomalacia in Caucasians. The inclusion of non-Caucasian children may have diluted the high percentage of males previously described in studies such as this. When analyzing the ethnic distribution of this diverse cohort, there were fewer Caucasian and Hispanic children and more multiracial children than expected. Given that many of our multi-racial children are likely to have some African ancestry, our findings give preliminary indication that laryngomalacia may occur with greater frequency among African-Americans than other racial groups. A substantially larger series with specific breakdown of each ‘‘multiracial’’ designation would be required to substantiate this observation. Compared to the expected rates of prematurity, there were significantly more premature Hispanic and African-American babies in our laryngomalacia study group. Given the association between neurologic disease and laryngomalacia, it is intuitive that premature babies would have higher rates of laryngomalacia. This, however, has not been previously observed in Caucasianpredominant studies. Perhaps the Caucasian larynx is less vulnerable to prematurity or other co-factors, such as laryngopharyngeal reflux, which may result in a more symptomatic nonCaucasian larynx.
Fig. 2. Prematurity and low birth weight.
1564
N.E. Edmondson et al. / International Journal of Pediatric Otorhinolaryngology 75 (2011) 1562–1564
In addition, when comparing to expected rates of low birth weight, there were more babies of low birth weight than expected in all ethnic groups. Neither the low birth weight nor premature groups showed significant differences in gender distribution. The association of laryngomalacia and low birth weight may also be explained by impaired neurologic development, but this trend has not been shown in Caucasian populations. There were no significant differences among genders or ethnic groups in neurologic comorbidity at diagnosis or in surgical treatment received in our study. The 30% rate of supraglottoplasty may be inflated in our database accrual method in which surgical patients may be more likely to gain attention than less affected children. Furthermore, at our medical center, sleep studies are often obtained early in the laryngomalacia work-up, providing more tangible evidence to proceed with surgical treatment. Finally, we believe that supraglottoplasty is a very effective operation, and consequently, this treatment option is often recommended for patients at our institution who might otherwise be considered borderline surgical candidates. 5. Conclusion The demographic evidence presented herein suggests premature African-American and Hispanic infants are at increased risk for laryngomalacia. The disease process may also occur more often in the African-American population regardless of gestational age at birth. Furthermore, low birth weight may be a stronger predictor of
laryngomalacia than previously appreciated in both genders and all ethnicities. In ethnically diverse populations, male gender appears less strongly associated with laryngomalacia than in previously reported studies of Caucasian populations. Information learned from previously published Caucasianpredominant studies in the literature might not fully apply to diverse populations. Better knowledge of these patients will aid in understanding disease etiology, making accurate diagnoses, choosing optimal treatment plans, and predicting future prognoses for all infants affected. References [1] S.J. Daniel, The upper airway: congenital malformations, Paediatr. Respir. Rev. 7S (2006) S260–S263. [2] B.E. Hamilton, J.A. Martin, S.J. Ventura, Births: Preliminary Data for 2009. National Vital Statistics Reports Web Release, vol. 59, no. 3, National Center for Health Statistics, Hyattsville, MD, 2010. [3] L.D. Holinger, R.J. Konior, Surgical management of severe laryngomalacia, Laryngoscope 99 (1989) 136–142. [4] R.L. Goldenberg, J.F. Culhane, J.D. Iams, R. Romero, Epidemiology and causes of preterm birth, Lancet 371 (2008) 75–84. [5] J.C. Groblewski, R.K. Shah, G.H. Zalzal, Microdebrider-assisted supraglottoplasty for laryngomalacia, Ann. Otol. Rhinol. Laryngol. 118 (2009) 592–597. [6] D.R. Olney, J.H. Greinwald, J.R. Smith, R.J.N.M. Bauman, Laryngomalacia and its treatment, Laryngoscope 109 (11) (1999) 1770–1775. [7] D.M. Thompson, Abnormal sensorimotor integrative function of the larynx in congenital laryngomalacia: a new theory of etiology, Laryngoscope 117 (2007) 1–33. [8] J. Bent, Pediatric laryngotracheal obstruction: current perspectives on stridor, Laryngoscope 116 (7) (2006) 1059–1070.