Congenital laryngomalacia is related to exercise-induced laryngeal obstruction in adolescence

Magnus Hilland, Ola Drange Røksund, Lorentz Sandvik, Øystein Haaland, Hans Jørgen Aarstad, Thomas Halvorsen, John-Helge Heimdal, Magnus Hilland, Ola Drange Røksund, Lorentz Sandvik, Øystein Haaland, Hans Jørgen Aarstad, Thomas Halvorsen, John-Helge Heimdal

Abstract

Objectives: Congenital laryngomalacia (CLM) is the major cause of stridor in infants. Most cases are expected to resolve before 2 years of age, but long-term respiratory prospects are poorly described. We aimed to investigate if CLM was associated with altered laryngeal structure or function in later life.

Methods: Twenty of 23 (87%) infants hospitalised at Haukeland University Hospital during 1990-2000 for CLM without comorbidities and matched controls were assessed at mean age 13 years. Past and current respiratory morbidity was recorded in a questionnaire, and spirometry performed according to standard quality criteria. Laryngoscopy was performed at rest and continuously throughout a maximal treadmill exercise test (continuous laryngoscopy exercise test (CLE-test)), and scored and classified in a blinded fashion according to preset criteria.

Results: In the CLM group, laryngeal anatomy supporting CLM in infancy was described at rest in nine (45%) adolescents. Eleven (55%) reported breathing difficulties in relation to exercise, of whom 7 had similarities to CLM at rest and 10 had supraglottic obstruction during CLE-test. Overall, 6/20 had symptoms during exercise and similarities to CLM at rest and obstruction during CLE-test. In the control group, one adolescent reported breathing difficulty during exercise and two had laryngeal obstruction during CLE-test. The two groups differed significantly from each other regarding laryngoscopy scores, obtained at rest and during exercise (p=0.001 or less).

Conclusions: CLM had left footprints that increased the risk of later exercise-induced symptoms and laryngeal obstruction. The findings underline the heterogeneity of childhood respiratory disease and the importance of considering early life factors.

Keywords: Congenital laryngomalacia; Exercise induced inspiratory symptoms; Follow-up study; Respiratory Disorders; Vocal cord dysfunction.

Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/

Figures

Figure 1
Figure 1
The score system (Maat score) used to describe findings during continuous laryngoscopy exercise test (CLE-test). *The scores at each level (glottic (A and C) and supraglottic (B and D)) were assessed at moderate (A and B) (when subject started to run) and at maximal effort (C and D) (just before the subject stopped running at the treadmill); all four numbers (A–D) were noted together with a sum score (E) for each test/subject (Adapted from Eur Arch Othorhinolaryngol30).
Figure 2
Figure 2
Scores of laryngeal movements obtained during a continuous laryngoscopy exercise test (CLE-test) in 20 adolescents hospitalised for congenital laryngomalacia (CLM) and 20 healthy control subjects with no such history. The left graph (Maat-D) depicts scores for supraglottic movements during maximal exercise. The right graph (Maat-E) depicts a total score from the CLE-test. The boxes show IQRs, with bold lines denoting medians. Vertical bars denote minimum and maximum scores.
Figure 3
Figure 3
Venn diagrams depicting occurrence of self-reported dyspnoea during exercise, and laryngeal findings at rest and during exercise. Findings, exercise: Maat-D score of 2 or 3 at maximum exercise during the continuous laryngoscopy exercise test (CLE-test). Findings, rest: Prominent arytaenoids and epiglottis at rest. Symptoms: Self-reported dyspnoea during exercise. The areas of the circles are proportional to the number of subjects (n) with the condition in question. Overlapping areas indicate subjects with two or three findings.

References

    1. Olney DR, Greinwald JH Jr, Smith RJ, et al. . Laryngomalacia and its treatment. Laryngoscope 1999;109:1770–5. 10.1097/00005537-199911000-00009
    1. Ayari S, Aubertin G, Girschig H, et al. . Pathophysiology and diagnostic approach to laryngomalacia in infants. Eur Ann Otorhinolaryngol Head Neck Dis 2012;129:257–63. 10.1016/j.anorl.2012.03.005
    1. Macfarlane PI, Olinsky A, Phelan PD. Proximal airway function 8 to 16 years after laryngomalacia: follow-up using flow-volume loop studies. J Pediatr 1985;107:216–18. 10.1016/S0022-3476(85)80128-1
    1. Thompson DM. Abnormal sensorimotor integrative function of the larynx in congenital laryngomalacia: a new theory of etiology. Laryngoscope 2007;117(Pt 2 Suppl 114):1–33. 10.1097/MLG.0b013e31804a5750
    1. Thomson J, Turner AL. On the Causation of the Congenital Stridor of Infants. Br Med J 1900;2:1561–3. 10.1136/bmj.2.2083.1561-a
    1. Awan S, Saleheen D, Ahmad Z. Laryngomalacia: an atypical case and review of the literature. Ear Nose Throat J 2004;83:334, 36–8.
    1. McSwiney PF, Cavanagh NP, Languth P. Outcome in congenital stridor (laryngomalacia). Arch Dis Child 1977;52:215–18. 10.1136/adc.52.3.215
    1. Richter GT, Thompson DM. The surgical management of laryngomalacia. Otolaryngol Clin North Am 2008;41:837–64, vii 10.1016/j.otc.2008.04.011
    1. Sesterhenn AM, Zimmermann AP, Bernhard M, et al. . Polysomnography outcomes following transoral CO2 laser microsurgery in pediatric patients with laryngomalacia. Int J Pediatr Otorhinolaryngol 2009;73:1339–43. 10.1016/j.ijporl.2009.06.002
    1. Smith GJ, Cooper DM. Laryngomalacia and inspiratory obstruction in later childhood. Arch Dis Child 1981;56:345–9. 10.1136/adc.56.5.345
    1. Zalzal GH, Anon JB, Cotton RT. Epiglottoplasty for the treatment of laryngomalacia. Ann Otol Rhinol Laryngol 1987;96(1 Pt 1):72–6. 10.1177/000348948709600118
    1. Friedman EM, Vastola AP, McGill TJ, et al. . Chronic pediatric stridor: etiology and outcome. Laryngoscope 1990;100:277–80. 10.1288/00005537-199003000-00013
    1. Christopher KL, Morris MJ. Vocal cord dysfunction, paradoxic vocal fold motion, or laryngomalacia? Our understanding requires an interdisciplinary approach. Otolaryngol Clin North Am 2010;43:43–66, viii 10.1016/j.otc.2009.12.002
    1. Røksund OD, Maat RC, Heimdal JH, et al. . Exercise induced dyspnea in the young. Larynx as the bottleneck of the airways. Respir Med 2009;103:1911–18. 10.1016/j.rmed.2009.05.024
    1. Rundell KW, Spiering BA. Inspiratory stridor in elite athletes. Chest 2003;123:468–74. 10.1378/chest.123.2.468
    1. Beaty MM, Wilson JS, Smith RJ. Laryngeal motion during exercise. Laryngoscope 1999;109:136–9. 10.1097/00005537-199901000-00026
    1. Bent JP III, Miller DA, Kim JW, et al. . Pediatric exercise-induced laryngomalacia. Ann Otol Rhinol Laryngol 1996;105:169–75. 10.1177/000348949610500301
    1. Bittleman DB, Smith RJ, Weiler JM. Abnormal movement of the arytenoid region during exercise presenting as exercise-induced asthma in an adolescent athlete. Chest 1994;106:615 10.1378/chest.106.2.615
    1. Christensen PM, Thomsen SF, Rasmussen N, et al. . Exercise-induced laryngeal obstructions: prevalence and symptoms in the general public. Eur Arch Otorhinolaryngol 2011;268:1313–19. 10.1007/s00405-011-1612-0
    1. Dion GR, Eller RL, Thomas RF. Diagnosing aerodynamic supraglottic collapse with rest and exercise flexible laryngoscopy. J Voice 2012;26:779–84. 10.1016/j.jvoice.2012.01.004
    1. Hull JH, Selby J, Sandhu G. “You Say Potato, I Say Potato”: Time for Consensus in Exercise-induced Laryngeal Obstruction? Otolaryngol Head Neck Surg 2014;151:891–2. 10.1177/0194599814549529
    1. Johansson H, Norlander K, Berglund L, et al. . Prevalence of exercise-induced bronchoconstriction and exercise-induced laryngeal obstruction in a general adolescent population. Thorax 2015;70:57–63. 10.1136/thoraxjnl-2014-205738
    1. Maat RC, Hilland M, Røksund OD, et al. . Exercise-induced laryngeal obstruction: natural history and effect of surgical treatment. Eur Arch Otorhinolaryngol 2011;268:1485–92. 10.1007/s00405-011-1656-1
    1. Smith RJ, Bauman NM, Bent JP, et al. . Exercise-induced laryngomalacia. Ann Otol Rhinol Laryngol 1995;104:537–41. 10.1177/000348949510400707
    1. Masters IB, Chang AB, Patterson L, et al. . Series of laryngomalacia, tracheomalacia, and bronchomalacia disorders and their associations with other conditions in children. Pediatr Pulmonol 2002;34:189–95. 10.1002/ppul.10156
    1. Quanjer PH, Tammeling GJ, Cotes JE, et al. . [Lung volumes and forced ventilatory flows. Work Group on Standardization of Respiratory Function Tests. European Community for Coal and Steel. Official position of the European Respiratory Society]. Rev Mal Respir 1994;11(Suppl 3):5–40.
    1. Quanjer PH, Stanojevic S, Cole TJ, et al. . Multi-ethnic reference values for spirometry for the 3–95-yr age range: the global lung function 2012 equations. Eur Respir J 2012;40:1324–43. 10.1183/09031936.00080312
    1. Boris M, Goldblatt A, Krigsman A. Laryngeal dysfunction: a common cause of respiratory distress, often misdiagnosed as asthma and responsive to antireflux therapy. Allergy Asthma Proc 2002;23:133–9.
    1. Heimdal JH, Roksund OD, Halvorsen T, et al. . Continuous laryngoscopy exercise test: a method for visualizing laryngeal dysfunction during exercise. Laryngoscope 2006;116:52–7. 10.1097/
    1. Maat RC, Roksund OD, Halvorsen T, et al. . Audiovisual assessment of exercise-induced laryngeal obstruction: reliability and validity of observations. Eur Arch Otorhinolaryngol 2009;266:1929–36. 10.1007/s00405-009-1030-8
    1. Richter GT, Rutter MJ, deAlarcon A, et al. . Late-onset laryngomalacia: a variant of disease. Arch Otolaryngol Head Neck Surg 2008;134:75–80. 10.1001/archoto.2007.17
    1. Reidenbach MM. Aryepiglottic fold: normal topography and clinical implications. Clinical anatomy (New York, NY) 1998;11:223–35. 10.1002/(SICI)1098-2353(1998)11:4<223::AID-CA1>;2-S
    1. Mandell DL, Arjmand EM. Laryngomalacia induced by exercise in a pediatric patient. Int J Pediatr Otorhinolaryngol 2003;67:999–1003. 10.1016/S0165-5876(03)00178-2
    1. Gessler EM, Simko EJ, Greinwald JH. Adult laryngomalacia: An uncommon clinical entity. Am J Otolaryngol 2002;23:386–9. 10.1053/ajot.2002.126322

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