- Email: [email protected]
Congenital inferior turbinate hypertrophy: An unusual cause of neonatal nasal obstruction
International Journal of Pediatric Otorhinolaryngology Extra (2007) 2, 26—30
www.elsevier.com/locate/ijporl
CASE REPORT
Congenital inferior turbinate hypertrophy: An unusual cause of neonatal nasal obstruction Jeff Kwok *, Man-Kit Leung, Peter Koltai Touro University College of Osteopathic Medicine, United States Received 19 October 2006; accepted 23 November 2006
KEYWORDS Inferior turbinate hypertrophy; Neonatal nasal obstruction; Obstructive sleep apnea; Endoscopic microdebrider-assisted turbinoplasty
Summary Inferior turbinate hypertrophy (ITH) in the pediatric population is generally an acquired problem whose etiology includes septal deviation, allergic rhinitis, gastroesophageal reflux disease, and enlarged adenoids. Congenital ITH is rarely seen, but requires full evaluation since these babies can exhibit respiratory and feeding difficulties. A review of the English language citations in the PubMed database (1965— 2006) failed to reveal any references that specifically address congenital ITH. We report a case of a newborn boy with ITH and resulting respiratory distress, obstructive sleep apnea, and altered external nasal development who failed medical management but was successfully treated by performing an endoscopic inferior turbinoplasty utilizing a microdebrider. # 2006 Elsevier Ireland Ltd. All rights reserved.
Introduction
Case report
Neonatal nasal obstruction (NNO) results in respiratory distress for a newborn infant and therefore requires conscientious evaluation and management. Inferior turbinate hypertrophy (ITH) in the pediatric population is generally an acquired problem and congenital ITH is rarely seen. A review of English language citations in the PubMed database (1965— 2006) failed to reveal any references to congenital ITH. However, this disorder manifests with similar respiratory and feeding difficulties as other causes of NNO. We present the case of an infant with congenital ITH who failed medical management, and subsequently underwent bilateral, powered inferior turbinoplasty with successful resolution of his symptoms.
A 1-month-old male infant was referred to the pediatric otolaryngology service for evaluation of persistent nasal obstruction, recurrent oxygen desaturations, and feeding difficulties. Born via caesarean section at 33 weeks gestational age, he immediately experienced respiratory distress and was noted to have hypertrophied inferior turbinates. He was initially managed with CPAP and ultimately weaned to room air by his second day of life. The patient was then started on oxymetazoline and mometasone which temporarily reduced the frequency of his desaturations and choking episodes. Nevertheless, he continued to demonstrate poor feeding endurance and slow development of nippling skills. At the time of the consultation, he was found to have bilateral nasal obstruction secondary to enlarged, pale, pink inferior turbinates which failed
* Corresponding author. Tel.: +1 650 270 7609. E-mail address: [email protected] (J. Kwok).
1871-4048/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2006.11.004
Congenital inferior turbinate hypertrophy
27 growth, a decision was made to intervene surgically. Based on our positive experience with partial microdebrider-assisted endoscopic inferior turbinoplasty in children, and the lack of literary guidance of similar problems, we planned sequential unilateral inferior turbinate reductions spaced 2 months apart. Starting on the left side and using standard technique of pediatric nasal endoscopy, the markedly thickened mucosa was shaved off of the lateral, inferior and medial aspects of the turbinal bone with a 3.5 mm straight-shafted microdebrider (Figs. 2—4). The inferior portion of the turbinal bone
Fig. 1 Coronal computed tomography shows bilateral opacification of nasal cavities and maxillary sinuses.
to vasoconstrict with topical oxymetazoline. A fiberoptic laryngoscope was passed with difficulty around the hypertrophic turbinates and revealed normal laryngeal anatomy. A CT scan demonstrated bilateral ITH with near-complete transnasal obstruction (Fig. 1). Topical and systemic steroids provided minimal relief and despite clinical evidence of gastroesophageal reflux disease, treatment with metoclopramide and ranitidine did not improve his obstructive symptoms. Due to failure of medical management, surgical options were discussed. However, over the period of his first 6 weeks of life, with intermittent readmissions for respiratory insufficiency, the patient developed the ability to mouth breathe and temporarily stabilized. During subsequent follow-up clinic visits over the next year, his breathing and feeding problems continued, and he was noted to have progressive widening of the dorsum of the nose. His weight for age hovered at the 10th percentile, and his pediatrician expressed concern about failure to thrive. A chloride sweat test was negative. His parents observed chronic mouth-breathing, snoring and apneic periods lasting for several seconds which resolved without intervention. A sleep study at 11 months of age demonstrated apneic periods lasting 35 s with multiple episodes of paradoxical breathing and a minimum desaturation of 84% (ranging from 84 to 90%). A repeat sinus CT demonstrated obliteration of the nasal cavity bilaterally by the inferior turbinates and opacification of the maxillary and ethmoid sinuses. Given the failure of medical management and persistent respiratory distress, failure to thrive, obstructive sleep apnea, and alteration of facial
Fig. 2 16 November 2005 endoscopic view of left nasal cavity demonstrating significant obstruction by a hypertrophied inferior turbinate.
Fig. 3 H&E at 100 magnification of left inferior turbinate shavings. This slide depicts submucosal gland hypertrophy and stromal proliferation. The aggregate volume of the left inferior turbinate shavings was also greater than that of the right, leading to a pathologic diagnosis of ITH more significant on the left than the right turbinate.
28
Fig. 4 H&E at 100 magnification of right inferior turbinate shavings. This slide depicts submucosal gland hypertrophy and stromal proliferation, consistent with ITH.
J. Kwok et al.
Fig. 6 21 November 2005 endoscopic view of left nasal cavity at 1-week postoperative follow-up, demonstrating a healing inferior turbinate remnant and patent left nasal airway passage.
was also burred down by about 1—2 mm with the same microdebrider blade. Care was taken not to injure the posterior portion of the inferior turbinate to avoid post operative hemorrhage (Fig. 5). He had an uneventful recovery from the initial procedure with minimal blood loss and no complications. Two weeks later, he was taken back to the OR for endoscopic debridement of the healing left inferior turbinate remnant and application of mitomycin-C (0.4 mg/ml) (Fig. 6). One month later, the right side was treated identically. Postoperatively, the child was treated systemically with amoxicillin (40 mg/kg) and ciprofloxa-
cin/dexamethasone drops to his nose on a TID basis for 2 months. He was followed closely in the outpatient clinic during the healing phase of his recuperation. His obstructive symptoms resolved after the initial procedure on the left side and he has been symptom free for a year postoperatively. The turbinates have remucosalized and appear to be functioning normally with no crusting or rhinorrhea (Fig. 7). He is now in the normal interval for weight and height.
Fig. 5 16 November 2005 endoscopic view of left nasal cavity following inferior turbinate reduction with powered microdebrider.
Fig. 7 21 December 2005 endoscopic view of left nasal cavity at 1-month postoperative follow-up, showing an incompletely healed inferior turbinate remnant, and continued patency of the left nasal airway passage.
Congenital inferior turbinate hypertrophy
Discussion As obligate nasal breathers, neonates with nasal obstruction suffer from respiratory distress, feeding difficulties, sleep apnea, and failure to thrive. While congenital non-inflammatory inferior turbinate hypertrophy (ITH) is a rare cause of neonatal nasal obstruction (NNO), it should be considered in the differential diagnosis of an infant presenting with NNO. In our experience with children, ITH is generally an acquired problem and its congenital non-inflammatory variant is one we have not previously seen. However, as this case illustrates, congenital ITH can be the cause of significant respiratory compromise and morbidity in a newborn infant. A review of the English language resources in the PubMed database (1965—2006) failed to yield articles that specifically address the presentation or treatment of congenital ITH. Without literary guidance, our management of this patient was based on experience with pediatric cases of acquired ITH. In the neonate, ITH is generally seen in response to mucosal irritation from problems such as meconium rhinitis or gastroesophageal reflux, GER. Neonates with acquired ITH from chemical irritation of the mucosa can be initially treated pharmacologically with systemic and intranasal steroids or short-term decongestants. Systemic antibiotics and topical antibiotic drops, usually used for the eye or ear, can be utilized for infectious rhinitis. Anti-reflux medications should be given if GER is suspected. Our patient’s inferior turbinates failed to demonstrate appreciable diminution in size, despite intense medical intervention. The decision to intervene surgically was derived from our experience with older children who have persistent ITH refractory to medical therapy. Surgical treatment for inferior turbinate hypertrophy aims to maximize nasal airflow while simultaneously preserving physiologic mucosal function. While various surgical techniques have been employed in children to treat ITH, our experience has been primarily with radio-frequency turbinate cautery and with partial endoscopic resection with a microdebrider. Much controversy exists regarding any type of surgery on the pediatric inferior turbinate. Complete resection of the inferior turbinates is reported to be associated with a high incidence of postoperative nasal dryness, crusting, and malodorous secretions [1]. Cryosurgery, laser, and electrocautery are related to impaired mucociliary function secondary to mucosal damage. Partial endoscopic resection with a microdebrider has been reported in children as being effective
29 without the problems associated with more radical resection [2—4]. Submucosal turbinoplasty with a special microdebrider blade, which resects the turbinate tissue from the inside out, has been reported to optimally increase airflow and preserve mucosal function [5—8]. However, there are no reports of this technique being used in children. While one study on rabbits demonstrated that middle turbinate resection can affect middle face growth [9], we found no references which suggest a deleterious impact of pediatric turbinoplasty on facial growth. Interestingly, Shapiro argues that equally dysmorphic features that may also arise from untreated nasal obstruction [10]. A few longterm studies have followed surgical treatment of ITH, many reporting successful outcomes with two year follow-up. Although some children have now been followed-up for as many as fourteen years, definitive conclusions cannot be established since atrophic changes may take many years to develop [11].
Conclusion Congenital ITH is an unusual cause of neonatal nasal obstruction. When recognized, the problem warrants thorough medical evaluation, with a focus on respiratory compromise, impairment of feeding, obstructive sleep apnea, and impairment of growth and development. When intensive medical management fails, endoscopic microdebrider-assisted turbinoplasty is an effective surgical intervention. While our patient has had resolution of his symptoms without complication or recurrence after 1 year of follow-up, the long-term outcomes of pediatric inferior turbinate surgery have yet to be established.
References [1] M.K. Hol, E.H. Huizing, Treatment of inferior turbinate pathology: a review and critical evaluation of the different techniques, Rhinology 38 (4) (2000) 157—166. [2] C.F. Lee, T.A. Chen, Powered microdebrider-assisted modification of endoscopic inferior turbinoplasty: a preliminary report, Chang Gung Med. J. 27 (5) (2004) 359—365. [3] B.S. Gendeh, Conventional versus endoscopic inferior turbinate reduction: technique and results, Med. J. Malaysia 55 (3) (2000) 357—362. [4] M.R. Van delden, P.R. Cook, W.E. Davis, Otolaryngol. Head Neck Surg. 121 (4) (1999) 406—409. [5] R. Rohrich, J.K. Kreuger, W.P. Adams, B.F. Marple, Rationale for submucous resection of hypertrophied inferior turbinates in rhinoplasty: an evolution, Plastic Reconstruct. Surg. 108 (2) (2001) 536—544.
30 [6] L.E. Jackson, R.J. Koch, Controversies in the management of inferior turbinate hypertrophy: a comprehensive review, Plastic Reconstruct. Surg. 103 (1) (1999) 300—312. [7] B.M. Lippert, J.A. Werner, Treatment of the hypertrophic inferior turbinate, 1 HNO, 2000 Mar, 48(3), 170—181. [8] D. Passali, F.M. Passali, V. Damiani, G.C. Passali, L. Bellussi, Treatment of inferior turbinate hypertrophy: a randomized clinical trial, Ann. Otolol., Rhinol., Laryngol. 112 (8) (2003) 683—688.
J. Kwok et al. [9] M. Egrilmez, C. Mutlu, H.H. Unlu, O. Celik, Facial growth after middle turbinate resection: an experimental study in the rabbit, Am. J. Rhinol. 17 (5) (2003) 275—281. [10] P.A. Shapiro, Effects of nasal obstruction on facial development, J. Aller. Clin. Immunol. 81 (5 Pt. 2) (1988) 967—971. [11] J.Y. Lee, J.D. Lee, Comparative study on the long-term effectiveness between coblation- and microdebriderassisted partial turbinoplasty, Laryngoscope 116 (5) (2006) 729—734.
www.elsevier.com/locate/ijporl
CASE REPORT
Congenital inferior turbinate hypertrophy: An unusual cause of neonatal nasal obstruction Jeff Kwok *, Man-Kit Leung, Peter Koltai Touro University College of Osteopathic Medicine, United States Received 19 October 2006; accepted 23 November 2006
KEYWORDS Inferior turbinate hypertrophy; Neonatal nasal obstruction; Obstructive sleep apnea; Endoscopic microdebrider-assisted turbinoplasty
Summary Inferior turbinate hypertrophy (ITH) in the pediatric population is generally an acquired problem whose etiology includes septal deviation, allergic rhinitis, gastroesophageal reflux disease, and enlarged adenoids. Congenital ITH is rarely seen, but requires full evaluation since these babies can exhibit respiratory and feeding difficulties. A review of the English language citations in the PubMed database (1965— 2006) failed to reveal any references that specifically address congenital ITH. We report a case of a newborn boy with ITH and resulting respiratory distress, obstructive sleep apnea, and altered external nasal development who failed medical management but was successfully treated by performing an endoscopic inferior turbinoplasty utilizing a microdebrider. # 2006 Elsevier Ireland Ltd. All rights reserved.
Introduction
Case report
Neonatal nasal obstruction (NNO) results in respiratory distress for a newborn infant and therefore requires conscientious evaluation and management. Inferior turbinate hypertrophy (ITH) in the pediatric population is generally an acquired problem and congenital ITH is rarely seen. A review of English language citations in the PubMed database (1965— 2006) failed to reveal any references to congenital ITH. However, this disorder manifests with similar respiratory and feeding difficulties as other causes of NNO. We present the case of an infant with congenital ITH who failed medical management, and subsequently underwent bilateral, powered inferior turbinoplasty with successful resolution of his symptoms.
A 1-month-old male infant was referred to the pediatric otolaryngology service for evaluation of persistent nasal obstruction, recurrent oxygen desaturations, and feeding difficulties. Born via caesarean section at 33 weeks gestational age, he immediately experienced respiratory distress and was noted to have hypertrophied inferior turbinates. He was initially managed with CPAP and ultimately weaned to room air by his second day of life. The patient was then started on oxymetazoline and mometasone which temporarily reduced the frequency of his desaturations and choking episodes. Nevertheless, he continued to demonstrate poor feeding endurance and slow development of nippling skills. At the time of the consultation, he was found to have bilateral nasal obstruction secondary to enlarged, pale, pink inferior turbinates which failed
* Corresponding author. Tel.: +1 650 270 7609. E-mail address: [email protected] (J. Kwok).
1871-4048/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.pedex.2006.11.004
Congenital inferior turbinate hypertrophy
27 growth, a decision was made to intervene surgically. Based on our positive experience with partial microdebrider-assisted endoscopic inferior turbinoplasty in children, and the lack of literary guidance of similar problems, we planned sequential unilateral inferior turbinate reductions spaced 2 months apart. Starting on the left side and using standard technique of pediatric nasal endoscopy, the markedly thickened mucosa was shaved off of the lateral, inferior and medial aspects of the turbinal bone with a 3.5 mm straight-shafted microdebrider (Figs. 2—4). The inferior portion of the turbinal bone
Fig. 1 Coronal computed tomography shows bilateral opacification of nasal cavities and maxillary sinuses.
to vasoconstrict with topical oxymetazoline. A fiberoptic laryngoscope was passed with difficulty around the hypertrophic turbinates and revealed normal laryngeal anatomy. A CT scan demonstrated bilateral ITH with near-complete transnasal obstruction (Fig. 1). Topical and systemic steroids provided minimal relief and despite clinical evidence of gastroesophageal reflux disease, treatment with metoclopramide and ranitidine did not improve his obstructive symptoms. Due to failure of medical management, surgical options were discussed. However, over the period of his first 6 weeks of life, with intermittent readmissions for respiratory insufficiency, the patient developed the ability to mouth breathe and temporarily stabilized. During subsequent follow-up clinic visits over the next year, his breathing and feeding problems continued, and he was noted to have progressive widening of the dorsum of the nose. His weight for age hovered at the 10th percentile, and his pediatrician expressed concern about failure to thrive. A chloride sweat test was negative. His parents observed chronic mouth-breathing, snoring and apneic periods lasting for several seconds which resolved without intervention. A sleep study at 11 months of age demonstrated apneic periods lasting 35 s with multiple episodes of paradoxical breathing and a minimum desaturation of 84% (ranging from 84 to 90%). A repeat sinus CT demonstrated obliteration of the nasal cavity bilaterally by the inferior turbinates and opacification of the maxillary and ethmoid sinuses. Given the failure of medical management and persistent respiratory distress, failure to thrive, obstructive sleep apnea, and alteration of facial
Fig. 2 16 November 2005 endoscopic view of left nasal cavity demonstrating significant obstruction by a hypertrophied inferior turbinate.
Fig. 3 H&E at 100 magnification of left inferior turbinate shavings. This slide depicts submucosal gland hypertrophy and stromal proliferation. The aggregate volume of the left inferior turbinate shavings was also greater than that of the right, leading to a pathologic diagnosis of ITH more significant on the left than the right turbinate.
28
Fig. 4 H&E at 100 magnification of right inferior turbinate shavings. This slide depicts submucosal gland hypertrophy and stromal proliferation, consistent with ITH.
J. Kwok et al.
Fig. 6 21 November 2005 endoscopic view of left nasal cavity at 1-week postoperative follow-up, demonstrating a healing inferior turbinate remnant and patent left nasal airway passage.
was also burred down by about 1—2 mm with the same microdebrider blade. Care was taken not to injure the posterior portion of the inferior turbinate to avoid post operative hemorrhage (Fig. 5). He had an uneventful recovery from the initial procedure with minimal blood loss and no complications. Two weeks later, he was taken back to the OR for endoscopic debridement of the healing left inferior turbinate remnant and application of mitomycin-C (0.4 mg/ml) (Fig. 6). One month later, the right side was treated identically. Postoperatively, the child was treated systemically with amoxicillin (40 mg/kg) and ciprofloxa-
cin/dexamethasone drops to his nose on a TID basis for 2 months. He was followed closely in the outpatient clinic during the healing phase of his recuperation. His obstructive symptoms resolved after the initial procedure on the left side and he has been symptom free for a year postoperatively. The turbinates have remucosalized and appear to be functioning normally with no crusting or rhinorrhea (Fig. 7). He is now in the normal interval for weight and height.
Fig. 5 16 November 2005 endoscopic view of left nasal cavity following inferior turbinate reduction with powered microdebrider.
Fig. 7 21 December 2005 endoscopic view of left nasal cavity at 1-month postoperative follow-up, showing an incompletely healed inferior turbinate remnant, and continued patency of the left nasal airway passage.
Congenital inferior turbinate hypertrophy
Discussion As obligate nasal breathers, neonates with nasal obstruction suffer from respiratory distress, feeding difficulties, sleep apnea, and failure to thrive. While congenital non-inflammatory inferior turbinate hypertrophy (ITH) is a rare cause of neonatal nasal obstruction (NNO), it should be considered in the differential diagnosis of an infant presenting with NNO. In our experience with children, ITH is generally an acquired problem and its congenital non-inflammatory variant is one we have not previously seen. However, as this case illustrates, congenital ITH can be the cause of significant respiratory compromise and morbidity in a newborn infant. A review of the English language resources in the PubMed database (1965—2006) failed to yield articles that specifically address the presentation or treatment of congenital ITH. Without literary guidance, our management of this patient was based on experience with pediatric cases of acquired ITH. In the neonate, ITH is generally seen in response to mucosal irritation from problems such as meconium rhinitis or gastroesophageal reflux, GER. Neonates with acquired ITH from chemical irritation of the mucosa can be initially treated pharmacologically with systemic and intranasal steroids or short-term decongestants. Systemic antibiotics and topical antibiotic drops, usually used for the eye or ear, can be utilized for infectious rhinitis. Anti-reflux medications should be given if GER is suspected. Our patient’s inferior turbinates failed to demonstrate appreciable diminution in size, despite intense medical intervention. The decision to intervene surgically was derived from our experience with older children who have persistent ITH refractory to medical therapy. Surgical treatment for inferior turbinate hypertrophy aims to maximize nasal airflow while simultaneously preserving physiologic mucosal function. While various surgical techniques have been employed in children to treat ITH, our experience has been primarily with radio-frequency turbinate cautery and with partial endoscopic resection with a microdebrider. Much controversy exists regarding any type of surgery on the pediatric inferior turbinate. Complete resection of the inferior turbinates is reported to be associated with a high incidence of postoperative nasal dryness, crusting, and malodorous secretions [1]. Cryosurgery, laser, and electrocautery are related to impaired mucociliary function secondary to mucosal damage. Partial endoscopic resection with a microdebrider has been reported in children as being effective
29 without the problems associated with more radical resection [2—4]. Submucosal turbinoplasty with a special microdebrider blade, which resects the turbinate tissue from the inside out, has been reported to optimally increase airflow and preserve mucosal function [5—8]. However, there are no reports of this technique being used in children. While one study on rabbits demonstrated that middle turbinate resection can affect middle face growth [9], we found no references which suggest a deleterious impact of pediatric turbinoplasty on facial growth. Interestingly, Shapiro argues that equally dysmorphic features that may also arise from untreated nasal obstruction [10]. A few longterm studies have followed surgical treatment of ITH, many reporting successful outcomes with two year follow-up. Although some children have now been followed-up for as many as fourteen years, definitive conclusions cannot be established since atrophic changes may take many years to develop [11].
Conclusion Congenital ITH is an unusual cause of neonatal nasal obstruction. When recognized, the problem warrants thorough medical evaluation, with a focus on respiratory compromise, impairment of feeding, obstructive sleep apnea, and impairment of growth and development. When intensive medical management fails, endoscopic microdebrider-assisted turbinoplasty is an effective surgical intervention. While our patient has had resolution of his symptoms without complication or recurrence after 1 year of follow-up, the long-term outcomes of pediatric inferior turbinate surgery have yet to be established.
References [1] M.K. Hol, E.H. Huizing, Treatment of inferior turbinate pathology: a review and critical evaluation of the different techniques, Rhinology 38 (4) (2000) 157—166. [2] C.F. Lee, T.A. Chen, Powered microdebrider-assisted modification of endoscopic inferior turbinoplasty: a preliminary report, Chang Gung Med. J. 27 (5) (2004) 359—365. [3] B.S. Gendeh, Conventional versus endoscopic inferior turbinate reduction: technique and results, Med. J. Malaysia 55 (3) (2000) 357—362. [4] M.R. Van delden, P.R. Cook, W.E. Davis, Otolaryngol. Head Neck Surg. 121 (4) (1999) 406—409. [5] R. Rohrich, J.K. Kreuger, W.P. Adams, B.F. Marple, Rationale for submucous resection of hypertrophied inferior turbinates in rhinoplasty: an evolution, Plastic Reconstruct. Surg. 108 (2) (2001) 536—544.
30 [6] L.E. Jackson, R.J. Koch, Controversies in the management of inferior turbinate hypertrophy: a comprehensive review, Plastic Reconstruct. Surg. 103 (1) (1999) 300—312. [7] B.M. Lippert, J.A. Werner, Treatment of the hypertrophic inferior turbinate, 1 HNO, 2000 Mar, 48(3), 170—181. [8] D. Passali, F.M. Passali, V. Damiani, G.C. Passali, L. Bellussi, Treatment of inferior turbinate hypertrophy: a randomized clinical trial, Ann. Otolol., Rhinol., Laryngol. 112 (8) (2003) 683—688.
J. Kwok et al. [9] M. Egrilmez, C. Mutlu, H.H. Unlu, O. Celik, Facial growth after middle turbinate resection: an experimental study in the rabbit, Am. J. Rhinol. 17 (5) (2003) 275—281. [10] P.A. Shapiro, Effects of nasal obstruction on facial development, J. Aller. Clin. Immunol. 81 (5 Pt. 2) (1988) 967—971. [11] J.Y. Lee, J.D. Lee, Comparative study on the long-term effectiveness between coblation- and microdebriderassisted partial turbinoplasty, Laryngoscope 116 (5) (2006) 729—734.