The Cleft Palate–Craniofacial Journal 51(4) pp. 378–380 July 2014 Ó Copyright 2014 American Cleft Palate–Craniofacial Association

EDITORIAL CPCJ 50th Anniversary Editorial Board Commentary: Otolaryngology—Then and Now Stephen F. Conley, M.D., M.S. This review continues the series of articles by the Editorial Board reflecting upon the first volume of the journal published in 1964 and celebrating the 50th anniversary of The Cleft Palate– Craniofacial Journal. This editorial examines the contributions of Otolaryngology–Head and Neck Surgery to interdisciplinary cleft care. The aerodigestive tract has many functions that are impacted by the cleft anomaly. Progress in the development of selected therapies will be reviewed.

The development of The Cleft Palate–Craniofacial Journal from its humble beginnings as a newsletter into a specialty journal has already been reviewed (Sharp, 2014). Of note in the inaugural edition of The Cleft Palate Journal was the president’s address to the American Cleft Palate Association membership reviewing the development of care for patients with cleft anomalies over the prior 20 years (Webster, 1964). In his address, Webster noted that tonsil and adenoid removal had been performed over the prior 20 years in children with former clefts with no awareness of the inherent special hazards that existed. He also described the association between cleft palate and chronic otitis media not being appreciated and stated that although progressive hearing loss in cleft patients was becoming recognized, the frequency was unknown because so few had audiograms at the time. There were no otolaryngology articles addressing care of the cleft anomaly in the first volume of The Cleft Palate Journal, but there was an article describing the prevalence of cleft uvula with ramifications for the otolaryngologist (Meskin et al., 1964). This editorial will broadly review developments in management of chronic otitis media in the presence of cleft palate, postadenoidectomy velopharyngeal insufficiency (VPI), and iatrogenic upper airway obstruction from 1964 to the present.

had been recognized for 85 years but still had not been well publicized (Alt, 1879). This changed with the 1969 publication of Paradise et al. describing the universality of otitis media with hearing loss associated with cleft palate, which was confirmed by others (Bluestone, 1971; Tasaka et al., 1990). A critical evaluation of the disease process yielded a reported 70% to 94% incidence of middle ear effusion in other studies of cleft palate patients (Young, 1968; Soudijin et al., 1975). Cleft palate repair was reported early on to improve hearing (Thornington, 1892). However, it is now apparent that cleft palate repair in combination with anatomic development and ventilation tube placement leads to normalization of eustachian tube function by an average of 6 years after palatoplasty in nearly 90% of patients (Smith et al., 1994). Ventilation tubes were reintroduced as a modern treatment for chronic otitis media in 1954 by Armstrong after two prior eras of myringotomy and grommet advocacy to aerate the middle ear (Rimmer et al., 2007). Placement of ventilation tubes is recommended for most patients with cleft palate. There is now a controversy as to whether to continue with ventilation tube use for otitis media with effusion in a normal population based upon a Cochrane review (Lous, 2005). Early ventilation tube placement for treatment of chronic otitis media in cleft palate patients would appear beneficial, given that up to 60% of children with no cleft management demonstrate hearing loss and abnormal tympanometry in the critical language-learning stage of life (Zheng et al., 2009). Although a recent systematic literature review showed some or no benefit of early tube placement in children with cleft palate, this appears to reflect the quality of the studies to date (Ponduri et al., 2009). In contrast to 1964, the incidence of hearing loss persistence without cleft palate repair is now known to be 30% (Zheng et al., 2009). Five decades after Dr. Webster’s observations, hearing loss and chronic otitis media associated with cleft palate are much better understood, but optimal treatment remains elusive.

CHRONIC OTITIS MEDIA In 1964, the association of chronic otitis media, hearing loss, and ear disease in cleft palate patients

Dr. Conley is Professor, Departments of Otolaryngology and Communication Sciences, and Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin. Submitted April 2014; Accepted April 2014. Address correspondence to: Dr. Stephen F. Conley, Pediatric Otolaryngology, Department of Otolaryngology and Communication Sciences, MS 782, CHW Clinics Building, P.O. Box 1997, 9000 West Wisconsin Avenue, Milwaukee, WI 53201. E-mail [email protected]. DOI: 10.1597/14-134 378

Conley, OTOLARYNGOLOGY—THEN AND NOW

POSTADENOIDECTOMY VELOPHARYNGEAL INSUFFICIENCY The risks of VPI and its avoidance are of critical importance when contemplating an adenoidectomy. The article published by Meskin et al. in the 1964 issue of The Cleft Palate Journal contributed to the knowledge of the prevalence of cleft uvula (1.47%) in a large patient cohort and its relation to more gross clefts of the secondary palate. Identification of a classic submucous cleft palate is most often heralded by a bifid uvula, which was progressively recognized to such a degree that the incidence of VPI acquired postadenoidectomy is currently reported as 1 per 1200 to 1 per 1450 procedures (Stewart et al., 2002; Witzel et al., 1986). This led to advocacy for using the cleft uvula as a ‘‘red flag’’ for other subtle palatal anomalies not immediately identifiable in the operative suite that could lead to postadenoidectomy VPI (Conley et al., 1997). Avoidance of postadenoidectomy VPI in patients with cleft uvula or repaired cleft palate is accomplished by a removal of the superior one half of the adenoid pad to maintain uninterrupted velopharyngeal closure (Tweedie et al., 2009). It is possible that the age-related involution of the inferior adenoid pad may still lead to late-onset VPI if the velopharyngeal apparatus has some compromise in function. Among postadenoidectomy VPI patients, speech therapy was sufficient intervention in 50 (37%) of the 137 reported; whereas, 50% needed pharyngeal surgery and 13% self-resolved (Witzel et al., 1986). Compared with the state of the art in 1964, adenoidectomy in patients at risk for postoperative VPI has significantly improved in technique and patient identification. IATROGENIC UPPER AIRWAY OBSTRUCTION In 1964, speech prostheses were the primary form of treatment for VPI. Upon removal of the prosthesis at the end of the day, all concerns for airway obstruction were also removed. Over the past 50 years, improvement of velopharyngeal closure has evolved to using a palatal procedure (Furlow, 1995; Sommerlad et al., 2002) or a local tissue flap obturator of the nasopharyngeal airway in the form of a sphincter pharyngoplasty or pharyngeal flap. Palatal procedures to reposition levator musculature for improved velopharyngeal function are preferred because nasopharyngeal airway patency is not compromised. Secondary speech management procedures in the pharynx risk development of upper airway obstruction or obstructive sleep apnea (OSA) (Shprintzen, 1988; Ysunza et al., 1993; Lesavoy et al., 1996; Lin et al., 1999; Wells et al., 1999; Liao et al., 2009; Ettinger et al., 2012). Transient postoperative OSA was reported as 38% in one study (Agarwal et al., 2003). Prolonged OSA (.6 months) may be present in up to 3% of patients after pharyngeal flap placement (Cole et al., 2008). Nasal continuous positive airway pressure therapy is sometimes an option if the pharyngeal flap is not too obstructive, but division of the

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flap can be performed without loss of velopharyngeal function (Agarwal et al., 2003). Another, more subtle airway obstruction is significant reduction of nasal airway function in the absence of OSA by polysomnography but demonstrated using posterior rhinomanometry (Yamashita et al., 2008). These patients have impaired nasal respiration and so lack the natural positive end-expiratory pressure provided during expiratory nasal airflow to maintain lung alveolar integrity. The result can be a breathless feeling for the patient and possible weight loss due to absence of the sense of smell and reduction in taste. In contrast with VPI treatment using speech prostheses 50 years ago, surgical therapies offer greater convenience for the patient but with the potential for significant airway disruption. LOOKING FORWARD Looking back on the articles in The Cleft Palate– Craniofacial Journal, one is struck by the tremendous strides accomplished in care for patients with cleft palate within the discipline of Otolaryngology–Head and Neck Surgery. A perusal of more recent The Cleft Palate–Craniofacial Journal issues reveals a vigorous contribution to cleft care. Management of the aerodigestive tract to maintain its optimal functions while trying to accommodate the impediments caused by the tissue deficiencies inherent with the cleft anomaly will remain a great challenge. One of the great challenges to surgeons is prediction of healing. The absence of control can conclude in less than desired results from the surgical procedure. This can be manifested as a tympanic membrane perforation from a ventilation tube or postadenoidectomy VPI, both of which likely lead to additional surgery. The advent of individual genomic arrays heralds the possibility of future control of healing to achieve the optimal surgical results as planned. The Cleft Palate–Craniofacial Journal is poised to continue to disseminate cutting-edge research on cleft palate and associated deformities of the mouth and face ‘‘where it counts’’: to the members of the American Cleft Palate–Craniofacial Association. REFERENCES Agarwal T, Sloan GM, Zajac D, Uhrich KS, Meadows W, Lewchalermwong JA. Speech benefits of posterior pharyngeal flap are preserved after surgical flap division for obstructive sleep apnea: experience with division of 12 flaps. J Craniofac Surg. 2003;14:630– 636. Alt A. Heilunger taustummheit erzielt durch beseitigung einer otorrhoe und einer angebornen gaumenspalte. Schmidt’s Jahrbuecher. 1879;183:277. Cole PC, Banerji S, Hollier L, Stal S. Two hundred twenty-two consecutive pharyngeal flaps: an analysis of postoperative complications. J Oral Maxillofac Surg. 2008;66:745–748.

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Conley SF, Gosain AK, Marks SM, Larson DL. Identification and assessment of velopharyngeal inadequacy. Am J Otolaryngol. 1997;18:38–46. Ettinger RE, Oppenheimer AJ, Lau D, Hassan F, Newman MH, Buchman SR, Kasten SJ. Obstructive sleep apnea after sphincter pharyngoplasty. J Craniofac Surg. 2012;23:1974–1976. Furlow LT Jr. Cleft palate repair by double-opposing Z-plasty. Plast Reconstr Surg. 1995;2:223–232. Lesavoy MA, Borud LJ, Thorson T, Riegelhuth ME, Berkowitz CD. Upper airway obstruction after pharyngeal flap surgery. Ann Plast Surg. 1996;36:26–32. Liao YF, Chuang ML, Chen PKT, Chen NH, Yun C, Huang CS. Incidence and severity of obstructive sleep apnea following pharyngeal flap surgery in patient with cleft palate. Cleft Palate Craniofac J. 2002;39:312–316. Lin KY, Goldberg D, Williams C, Borowitz K, Persing J, Edgerton M. Long-term outcome analysis of two treatment methods for cleft palate: combined levator retropositioning and pharyngeal flap versus double-opposing Z-plasty. Cleft Palate Craniofac J. 1999;36:73–78. Lous J, Burton MJ, Felding JU, Ovesen T, Rovers MM, Williamson I. Grommets (ventilation tubes) for hearing loss associated with otitis media with effusion in children. Cochrane Database Syst Rev. 2005;(1):CD001801. Meskin LH, Gorlin RJ, Isaacson RJ. Abnormal morphology of the soft palate: 1. The prevalence of cleft uvula. Cleft Palate J. 1964;1: 342–346. Paradise JL, Bluestone CD, Felder H. The universality of otitis media in fifty infants with cleft palate. Pediatrics. 1969;44:35–42. Ponduri S, Bradley R, Ellis PE, Brookes ST, Sandy JR, Ness AR. The management of otitis media with early routine insertion of grommets in children with cleft palate—a systematic review. Cleft Palate Craniofac J. 2009;46:30–38. Rimmer J, Giddings CEB, Wier N. J Laryngol Otol. 2007;121:911–916. Shprintzen RJ. Pharyngeal flap surgery and the pediatric upper airway. Int Anesthesiol Clin. 1988;26:79–88. Sharp HM. CPCJ 50th anniversary editorial board commentary: ethics and health policy—then and now. Cleft Palate Craniofac J. 2014;51(1):2–4.

Smith TL, Diruggerio DC, Jones KR. Recovery of eustachian tube function and hearing outcome in patients with cleft palate. Otolaryngol Head Neck Surg. 1994;111:423–429. Sommerlad BC, Mehendale FV, Birch MJ, Sell DA, Hattee C, Harland K. Palate re-repair revisited. Cleft Palate Craniofac J. 2002;39:295–307. Soudijin ER, Huffstadt AJC. Cleft palates and middle ear effusions in babies. Cleft Palate J. 1975;12:229–231. Stewart KJ, Ahmad T, Razzell RE, Watson AC. Altered speech following adenoidectomy: a 20-year experience. Br J Plast Surg. 2002;55:469–473. Thornington J. Almost total destruction of the velum palati corrected by an artificial soft palate, producing not only greatly improved speech, but an immediate increase in audition. Med News. 1892;61:269. Tweedie DJ, Skilbeck CJ, Wyatt ME, Cochrane LA. Partial adenoidectomy by suction diathermy in children with cleft palate, to avoid velopharyngeal insufficiency. Int J Pediatr Otorhinolaryngol. 2009;73:1594–1597. Webster RC. Advances in therapy in cleft palate—our first twenty years. Cleft Palate J. 1964;1:5–15. Wells MD, Vu TA, Luce EA. Incidence and sequelae of nocturnal respiratory obstruction following posterior pharyngeal flap operation. Ann Plast Surg. 1999;43:252–257. Witzel MA, Rich RH, Margar-Bacal F, Cox C. Velopharyngeal insufficiency after adenoidectomy: an 8-year review. Int J Pediatr Otorhinolaryngol. 1986;11:15–20. Yamashita RP, Trindade IE. Long-term effects of pharyngeal flaps on the upper airways of subjects with velopharyngeal insufficiency. Cleft Palate Craniofac J. 2008;45:364–370. Young A. The state of ears in children with a cleft palate deformity. J Laryngol. 1968;82:707–715. Ysunza A, Garcia-Velasco M, Garcia-Garcia M, Haro R, Valencia M. Obstructive sleep apnea secondary to surgery for velopharyngeal insufficiency. Cleft Palate Craniofac J. 1993;30:387–390. Zheng W, Smith JD, Shi B, Li Y, Wang Y, Li S, Meng Z, Zheng Q. The natural history of audiologic and tympanometric findings in patients with an unrepaired cleft palate. Cleft Palate Craniofac J. 2009;46:24–29.

CPCJ 50th Anniversary Editorial Board Commentary: Otolaryngology--then and now.

This review continues the series of articles by the Editorial Board reflecting upon the first volume of the journal published in 1964 and celebrating ...
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