ORIGINAL ARTICLE

A Review of the Cleft Lip/Palate Literature Reveals That Differential Diagnosis of the Facial Skeleton and Musculature is Essential to Achieve All Treatment Goals Samuel Berkowitz, DDS, MS Background: After 40 years of monitoring cleft palate treatment results with extensive objective records of cephaloradiographs, dental casts, and photographs, it became apparent that patients with the same cleft type who received the same treatment at approximately the same age were obtaining different results. Method: An extensive review of cleft palate surgical, orthodontic, facial, and palatal longitudinal growth studies was undertaken to determine the critical physical difference between these patients that determined why some treatments succeeded while others failed. Results: Treatment should be based on performing staged palatal surgery between 18 and 24 months when the palatal surface area to cleft space size is approximately 15% to 20%. Presurgical orthopedics with a gingivoperiosteoplasty causes midfacial deformities. Conclusion: Even though patients have the same cleft type and have received the same surgical treatment, usually between 18 and 24 months, the ratio of cleft and palatal size of 15% to 20% is critical to obtain good palatal development. Key Words: Literature review, differential diagnosis, facial/ palatal growth, bone deficiency, cleft palate growth (J Craniofac Surg 2015;26: 1143–1150)

B

ased on the review of recent literature, one can conclude that for the foreseeable future, surgical treatment will continue to be managed by surgeons using a variety of modified surgical procedures, but many cases will still be accompanied by physical trauma. Early palatal surgery, ie, before patients reach 5 to 8 months of age, should be considered successful only when favorable outcomes of clear speech, satisfactory facial growth, dental occlusion, and normal psychosocial development have been achieved. These goals are sometimes but not always reached. Surgically induced impairment of facial growth and muscle function is often a negative outcome in early palatal closure cases. Despite long use of accepted presurgical orthopedic and dependent surgical procedures, much controversy remains about the significance of these techniques regarding timing and sequence, ancillary procedures, and surgical skill (Bardach et al,1 Berkowitz et al,2 Bongarts et al3). Due to certain clinics’ failure in reporting outcomes, there is still much doubt about the influences of From the University of Illinois School of Dentistry, Chicago, IL. Received August 28, 2014. Accepted for publication January 20, 2015. Address correspondence and reprint requests to Samuel Berkowitz, DDS, MS, 11035 Paradela Street, Coral Gables, FL 33156; E-mail: [email protected] The author reports no conflicts of interest. Copyright # 2015 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000001592

The Journal of Craniofacial Surgery



select palatal surgery protocols in response to surgery (Latham,4 Millard and Latham,5 Rosenstein and Kernaham,6 Mulliken,7 Cutting and Grayson,8 Grayson and Cutting9). Particular theories, considered but not proven useful, have led to the selection of some techniques over others. Such treatment uncertainties have been reviewed by concerned clinics with the use of longitudinal treatment objective records using serial lateral cephs, dental casts, and photographs (Pruzansky10). Patients have been followed from birth through adolescence to determine what their treatment had revealed about facial and palatal treatment in general and its long-term effects in unilateral and bilateral cleft lip and palate conditions. This article review’s time range extends from the mid-1900 s to the present.

Early Palatal Surgery The cleft literature advocates early palate repair and the use of presurgical orthopedics with or without lip surgery to obtain normal speech and facial aesthetics as early as possible (Brophy,11 Dorrance,12 Cronin,13 Crikelair,14 Robertson and Fish,15 Chapman16). Implementation of palatal obturators to cover palatal clefts was employed by some (Hotz17) but was soon abandoned by most orthodontists and pedodontists. Today, even with expert cleft palate team collaboration, surgeons, with the support of speech language pathologists, are generally still the sole decision makers in selecting the treatment plan to be used. Unfortunately, current treatment outcomes of longitudinal studies by the cleft palate teams’ orthodontists and reviewed by the teams’ clinicians are not well documented. This was the result with Monroe and Rosenstein,18 Rosenstein and Kernaham,6 Mulliken,7 Millard and Latham,5 and Cutting and Grayson.8 More recently, many knowledgeable clinicians, disappointed with poor results, have concluded that successful treatment, relative to the time and type of surgery, is contingent upon understanding the natural history of the palatal cleft defect and the face in which it exists (Pruzansky,19,20 Harvold,21 Pruzansky and Aduss,22 Ross,23,24 Kremenak,25 Aduss,26 Hanada and Krogman,27 Friede,28,29 Witzel et al,30 Vargervik,31 Shprintzen et al,32 Van Demark,33 Semb,34 Semb and Shaw,35,36 Berkowitz,2 Friede et al37). Thus, longitudinal objective studies, done by the above clinicians, helped explain many cause-andeffect relationships between palatal surgery and subsequent successful and failed facial development. These studies concluded that increased success requires understanding of differences in the palatal cleft deformity and its dependence on the facial growth pattern (Figs. 1 and 2A–C) and skeletal and muscular function among patients with the same cleft type. Understanding issues such as variations in palatal development (Moss,38 Krogman et al39) (Figs. 3A, B), palatal bone deficiencies (Fig. 3B), and nasopharyngeal architecture (Figs. 4A, B) and its muscle function, Berkowitz40 stated that combining these factors will promote improved habilitative procedures to achieve all treatment goals equally (Figs. 5A, B and 6A–E). The following are the questions that must be answered to increase the number of successfully treated cases: 1. Are the palatal shelves intrinsically deficient, adequate, or excessive in mass?

Volume 26, Number 4, June 2015

1143

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Berkowitz

The Journal of Craniofacial Surgery

FIGURE 1. Variations in facial forms. Bone remodeling during growth. Remodeling is a basic part of the growth process. The reason why a bone must remodel during growth is because its regional parts become moved: ‘‘drift’’ moves each part from one location to another as the whole bone enlarges. The maxilla grows downward and forward from the cranial base with growth occurring at the articulations with other bones. Furthermore, there is little variation in the upper face form between groups. Therefore, because of the small variation, it is likely that individual differences in facial form result from growth in other facial areas where there is more variation.

2. How are these parameters altered as a consequence of growth and surgical reconstruction? 3. How do different facial and palatal growth patterns affect surgical decision making? 4. Is there more than one preferred surgical treatment procedure based on cleft type?



Volume 26, Number 4, June 2015

FIGURE 3. A, Anatomic variations within each cleft type. In the upper left is a grouping of all different cleft types; however, when looking at a grouping of each cleft type, it becomes apparent that there are physical differences. For example, within the bilateral cleft lip and palate cases, there can be difference in size and shape of the premaxilla due to the number and position of the maxillary incisors. B, Variations in isolated cleft palate or in any other cleft type. The length and width of the cleft space is highly variable. The cleft extends anteriorly to various distances but not beyond the incisal canal.

5. Does success of a given surgical procedure depend on the size of the palatal cleft defect, relative to the size of the surrounding palatal soft tissue medial to the alveolar ridges at the time of surgery and timed with the resulting facial growth pattern?

Facial Growth Differences in UCLP, BCLP Semb34 stated that the growth pattern is only slightly different in one obvious respect between UCLP and BCLP groups. In comparison with UCLP patients, BCLP groups have greater maxillary prominence in early childhood (SNA is 5.3 degrees larger at 5 years), but this difference lessens with time, so that by the time the patient is 18 years old, the maxilla is only slightly more prominent (1.4 degrees), on average. In other respects, facial growth patterns were similar in both conditions, although the mandibular angle was somewhat greater

FIGURE 2. A–C, Various patterns of postnasal growth. Facial growth rotations resulting from differential vertical and horizontal growth. A, Hyperdivergent pattern with posterior growth rotation. B, Neutral growth pattern. C, Hypodivergent growth pattern with anterior growth. Based on the serial studies, 3 general patterns of postnasal growth have been demonstrated. In the Pierre Robin sequence, and in complete bilateral clefts of the lip and palate, most cases demonstrate substantial improvement through ‘‘catch-up’’ in the growth of the mandible. In the second pattern, mandibulofacial dysostosis, the pattern of growth is such that the deformity observed in infancy or early childhood is maintained throughout the growth period. The deformity of the mandible neither improves nor worsens in the course of time. The third pattern is one in which the growth process is so deranged that the severity of the deformity increases with age.

1144

FIGURE 4. A and B, The influence of the skeletal architecture on the pharyngeal form and size. The hard palate of the maxillary complex is anatomically associated with the anterior cranial base and can vary in its anteroposterior dimension. The importance of cranial base angle (Ba-S-N): A, obtuse cranial base angle. In cases with a severe obtuse cranial base angle, the pharyngeal space is usually deeper than normal even in the presence of a long hard palate because it must be associated with the basilar portion of the occipital bone. This condition is usually seen when hypernasality exists in the absence of an overt palatal cleft. The condition is called congenital palatal insufficiency (CPI). B, With an acute cranial base angle, the cervical spine is positioned close to the hard palate creating a shallow pharyngeal depth. These relationships do not change with growth. The palate descends from the anterior cranial base in a parallel fashion. Due to changing slope of the posterior pharyngeal wall, the pharyngeal depth increases with time.

#

2015 Mutaz B. Habal, MD

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery



Volume 26, Number 4, June 2015

Diagnosis of Facial Skeleton/Musculature

being associated with skeletal and muscular dysmorphology, beyond the cleft site (Figs. 1, 2). Facial form in adults with repaired UCLP and BCLP are similar in many respects, but in comparison with the non-cleft subjects, their faces are characterized by a progressive retrusion of the

FIGURE 5. A, CUCLP—conservative treatment. This case demonstrates good palatal and facial growth in CUCLP. Surgical treatment: no presurgical orthopedics. Lip adhesion at 4 months followed by Millard’s Rotation Advancement at 6 months. Soft palatal closure at 2 months. Palatal cleft closure at 15 months using modified von Langenbeck procedure with modified vomer flap. Secondary alveolar cranial bone graft at 6 years and 8 months. Photographs showing various treatment stages from birth to 17 years of age: 2 years 7 months palatal view showing fixed buccal expander. B, CUCLP— conservative treatment. 7 years 3 months: lateral incisor is erupting through cranial bone graft. Orthodontics in the adult dentition: lateral incisor is extracted due to poor root development—conventional orthodontics. Surgery to close the palatal fistula was unsuccessful. Facial photographs at 17 years: intraoral photographs, Hawley orthodontic retainer with lateral incisor pontic. Millard, after this case, chose to close the hard and soft palate cleft at the same time. The fistula closure surgery was done as an adult because there were no difficulties when eating or speaking.

(3 degrees) in BCLP throughout the period of observation. This means that in CUCLP (Figs. 5A, B) and CBCLP (Figs. 6A–E) after closing the cleft lip, the face will flatten if nothing else is done to the midface and the mandible.

Intrinsic Facial Differences Not Attributed to Surgery Rosenberger,41 King,42 Kremenak et al,25 Ross,23 Shprintzen et al,34 Friedman et al,43 Shifman et al,44 and Shaw and Semb36 proposed the possibility of the failed union of the facial processes #

2015 Mutaz B. Habal, MD

FIGURE 6. A and B, CBCLP—conservative treatment. Serial photographs demonstrate excellent facial and palatal growth in CBCLP. No presurgical orthopedics other than the use of external lateral elastics off a head bonnet for 10 days before lip surgery. The palatal cleft was closed using a modified von Langenbeck procedure because the oronasal space was physically closed by the medial borders of the palatal segments contacting the vomer. Secondary alveolar cranial bone grafting was done at 8 years of age. At birth an extremely protrusive asymmetrical premaxilla whose protrusiveness gradually diminished with growth. At 20 years of age, the occlusion is ideal. Facial photographs show a harmonious and pleasing soft tissue profile. No early retraction midfacial mechanics were used. Palatal closure was delayed because the palatal segments medial border made contact with the vomer blocking air flow to the nose. No VPI was present at any age. Today—soft palate and hard palate cleft closure is performed at the same time between 18 and 24 months in most cases, some earlier, others later, depending on the relative size of the cleft space to the size of the palatal segments. C, Cephalometric serial tracings. D, Superimposed serial tracings using the Coben Basion Horizontal method shows an excellent facial growth pattern with an attached muscular lip which straightens the skeletal profile. There is very little forward midfacial growth between 11 and 20 years of age. E, Palatal cast outlines laterally limited by the alveolar ridges, were superimposed using the rugae for registration. This series shows that the premaxilla’s position within the maxillary complex at 17 years of age is similar to that seen at birth. Excellent growth occurs in all dimensions and is similar to the growth pattern seen in non-cleft palates. Increased posterior palatal growth is necessary to accommodate the developing molars. Alveolar bone growth with tooth eruption increases anterior alveolar bone size. The position of the anterior premaxilla relative to the anterior cranial base N (Nasion) and the anterior position of Pog (Pogonion) at the mandibular synthesis shows changes in convexity due to changes in upper and lower facial growth from birth to 17 years of age. Midfacial forward growth is retarded by the united lip musculature force in these conservatively treated CBCLP cases. The early premaxilla is ventroflexed by lip adhesion and not bodily retruded as it would have been when using the POPLA or NAM þ GPP procedure.

1145

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Berkowitz

The Journal of Craniofacial Surgery

profile. Both the maxilla and mandible become shorter and relatively retrusive and the incisors in both jaws are retroclined. Their pattern of facial growth is different from that of non-cleft individuals. In patients between 5 and 18 years of age, Shaw and Semb36 found almost no increase in the length of the maxilla, measured to the anterior outline of the alveolar process, in a mixed longitudinal study in 257 cases of complete UCLP. They found that there was a concomitant reduction in maxillary prominence at the dentoalveolar level.

Stimulating Palatal Growth In the 1960 s, the controversial theory of stimulating palatal growth, introduced by James Scott45,46 to reduce cleft size, was well accepted. He stated that the maxilla normally grows downward and forward, as a result of a growth force generated by the attached nasal septum. He mistakenly concluded that the reason for the malformed and retropositioned maxilla was not the result of nonphysiological surgery but rather of the lack of growth-stimulating forces to the cleft palatal segments emanating from the nasal septum. Orthodontists and surgeons, accepting Scott’s hypothesis, began using removable palatal presurgical orthopedic (PO) appliances (Hotz,17 Graf-Pinthus and Bettex,47 Hotz and Gnoinski,48 Weil,49 Gnoinski50). This ultimately led to delaying cleft closure surgery in patients between 5 and 9 years old (mostly in Europe), enabling the use of smaller mucoperiosteal flaps from the relatively large palatal segments. The resulting small palatal cleft space would reduce excessive growth inhibiting palatal scarring. This technique was eventually rejected by surgeons in the U.S.A., due to pressure from speech language pathologists who chose to close the cleft space in patients between the ages of 5 and 8 months. This rejection was supported by the State of the Art report, sponsored by the American Cleft Palate Association (Berkowitz51). The review committee concluded that the literature failed to support the value of presurgical ‘‘growth stimulating’’ orthopedics (PO). In CUCLP and CBCLP treatment, anterior crossbite correction was successful as long as the lateral incisor space(s) was/were surgically left open. If closed by molding alone, the cleft space could be easily recovered, provided a primary bone graft, early alveolar bone graft, or periosteoplasty was not utilized. Unfortunately, the presence of a bony bridge in the cleft would prevent the recovery of the cleft space and the establishment of upper and lower arch congruency.

Alveolar Cleft Closure Rosenstein and Kernaham6 accepted the earlier rejection of palatal stimulation concepts and, thereby, moved even further in another direction. Using tooth supported presurgical orthopedics, they recovered the lateral incisor space(s) in patients of 4 to 6 years old. This was followed by a primary alveolar bone graft to be inserted at the repositioned palatal segment’s alveolar contact point. By replacing the missing alveolar bone, however, they believed the false assumption that the bone graft would stabilize the corrected arch relationship and permit normal palatal growth. After a longterm serial cast and ceph study, using a similar PO with primary bone grafts, Friede and Johanson52,53 reported severe midfacial recessiveness along with anterior crossbite problems. After reports of similar outcomes by other reconstructive surgeons and orthodontists, this procedure was eventually abandoned.

CBCLP Surgery Many different surgical and orthopedic procedures were utilized for the treatment of CBCLP, to correct the protruding premaxilla according to Millard54,55 in his 3-volume Cleft Craft, as well as by Friede et al,56 Berkowitz,2,51,57–59 and Shaw and Semb.36 Dorrance12

1146



Volume 26, Number 4, June 2015

reported on the failure of premaxillary surgical setbacks and various fixation pins placed through the premaxilla into the vomer in attempt to stabilize the premaxilla’s new position (Figs. 6A–E and 7B, C). Berkowitz,59 not wishing to bodily retrude the premaxilla, has successfully used a head bonnet with a tape or elastics extending from both cheeks across the premaxilla to ventroflex the premaxilla (Figs. 6A– E). It was followed by lip surgery, the force of which gradually retards midfacial growth (Bardach et al1). The facial profile aesthetics were well accepted by patients, by the time they were 6 to 7 years old. The degree of protrusion remained consistent until patients were 5 years old, by which time it steadily receded. Thus, by the time they reached 7 years old, it was similar to the size of the non-cleft subjects and by 18 years old, it was about 6% less. Throughout the period of observation, the mandible was less prominent in subjects with BCLP at 5 years of age. These findings of the gradual flattening of the convex facial profile are well documented by Berkowitz,40,60 Friede et al,56 and Vargervik.31 In most of these cases, the profile became aesthetically pleasing, and the child spoke acceptably at the start of school. These clinicians tried to reach all treatment goals by the patients’ sixth or seventh year, rather than by 2 to 3 years of age, as did those surgeons who used presurgical orthopedics, periosteoplasty, and lip adhesion (POPLA) introduced by Millard and Latham5 and accepted by Mulliken.7 Since then, the same midfacial growth retarding nasoalveolar molding forces and gingivoperiosteoplasty (NAM and GPP), introduced by Cutting and Grayson in 2000,8 has not been supported by long-term objective records. Although orthodontists are now joined by other specialists on a cleft palate team, only a few orthodontists have recognized the need to collect objective longitudinal records of casts, cephaloradiographs, and photographs to aid in future surgical decision making. In the past and even today, most surgeons and orthodontists possess insufficient knowledge of facial growth, in general, and of the palate’s natural history, specifically, to be able to anticipate the effects of palatal surgery, with or without presurgical orthopedics on postnatal facial growth and speech development (Lee et al61). To identify the causes of treatment success and failure, Berkowitz58 documented Millard’s 40-year treatment of CUCLP and CBCLP outcomes (Figs. 7A–C), using serial casts, cephs, and photos, from birth to mid-adolescence (Figs. 4A, B, 6A–E, 7A–C). During the first 20 years (1960–1980), the patients were treated without presurgical orthopedic appliances other than a head bonnet with a facial strap (exceptions were newborns with CUCLP), and at 4 to 5 years old with a Hawley-like plastic appliance in severely protruding premaxillary cases in CUCLP and CBCLP. The aim of this therapy was to ventroflex but not to bodily retrude the premaxilla. During the second 20 years (1980 to 2000), all CUCLP and CBCLP cases were treated with the Millard and Latham (1990) procedure of using a palatally pinned presurgical appliance, followed by periosteoplasty and lip adhesion (POPLA) (Figs. 7A–C). A von Langenbeck palatal surgical technique with a modified vomer flap, which helped maintain vault height, was used by Millard in all cases. During these 2 periods, Berkowitz59 recommended that cleft closure should take place between 18 and 24 months to allow for a reduction in cleft size. In both cleft types, cleft closure was sometimes performed even later if the relative cleft space size to surrounding soft tissue size was considered too large. It was advisable to leave time for additional palatal growth to avoid causing extensive growth-inhibiting scarring. This adjusted time schedule was later supported by a multicenter retrospective 3D serial longitudinal cast study (Berkowitz et al62) (Figs. 8A-D). The study was limited to serial casts of successfully treated speech, dental, and facial cases from 7 clinics: 3 in the U.S.A., which used palatal surgery between 1 and 2 years, and 4 in Europe, which delayed closure to 5 to 9 years and presented varying degrees of cleft size to that of the surrounding palatal mucoperiosteum medial to the alveolar ridge. The European speech therapists’ report of desired speech outcomes are disputed by many #

2015 Mutaz B. Habal, MD

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery



Volume 26, Number 4, June 2015

Diagnosis of Facial Skeleton/Musculature

and the palatal growth velocity curve usually plateaued during this age period of 18 to 24 months; therefore, there was less chance of palatal scarring. Berkowitz et al62 and Bzoch69 concluded that, to reach all treatment goals, delaying surgery not to 5 to 8 months but to an older age of the patient is necessary. Relevant physical factors, including but not limited to age, are the following: (a) the depth of the nasopharynx, which is influenced by the shape and relative position of the cervical spine and the angle of the cranial base (Figs. 4A, B). An obtuse cranial base positions the spine more posteriorly, thereby increasing pharyngeal depth. The reverse is problematic too. In many craniofacial syndromes, such as those of Cruzon and Apert, the shallow pharyngeal depth is due to an acute cranial base with the spine being brought anteriorly (Fig. 4B). (b) the velar mass, length and mobility, and the closure patterns of the lateral pharyngeal wall muscles. For uncontrolled air flow, the surgeon should consider attaching the pharyngeal flap at the level above the palatal plane (superior pharyngeal flap) or at the first cervical vertebrae because this is where the velum is anatomically related to the lateral pharyngeal muscle sphincter action (inferior pharyngeal flap) in non-cleft children. (c) the sensory functions, genotype, geometrics of the original deformity, neurological disorders, the facial growth pattern, and the adequacy of the surgical procedure.

Correlation and Causation A cross-sectional study by Dorf and Curtin70 stated that most children who have the palatal cleft closed at 6 months of age had developed better speech when compared to those who were treated at a later age, and, therefore, they strongly recommended early cleft and various surgical-dental protocols of presurgical orthopedics. Early speech and surgery advocates were mistaken for definitively associating correlation with causation. Such correlation between speech proficiency and age at surgical closure does not prove that causation exists between these 2 variables or that later age closure causes poor speech and early closure produces good speech.

FIGURE 7. A (a, b), Complete bilateral cleft lip and palate—POPLA treatment. a, Latham’s presurgical orthopedic appliance with a laterally placed pin in the premaxilla. The elastic chain creates the activating forces to retract the premaxilla while expanding the palatal segments. The posterior palatal appliance is pinned to the palate for approximately 2 weeks. The premaxillary pins, which are pulled by the elastic chain, are positioned anterior to the premaxillary vomerine suture. b, The premaxilla is bodily retracted while the cleft posterior segments are moved anteriorly approximately 2–3 mm to make contact with the retruded premaxillary segment. B (a), Newborn: a small asymmetric protruding premaxilla. b, After premaxillary retrusion. Lip adhesion followed by periosteoplasty and definitive lip surgery (POPLA). c, Midfacial deficiency due to retruded premaxilla. C, Retruded premaxilla showing some missing incisor teeth. Orthodontic appliances placed to correct anterior crossbite and missing incisor spaces. The bony bridge created by the periosteoplasty prevented premaxillary advancement and crossbite correction.

American speech-language pathologists (Chapman and Hardin,63 DeLuke et al,64 Chapman et al,65 Peterson-Falzone66–68). The conclusion of the study showed that surgery at 18 to 24 months, in most cases and sometimes even later, if the ratio of the cleft space to the amount of surrounding palatal soft tissue medial to the alveolar ridge, was approximately 15% to 20%. The findings show that the degree of palatal bone deficiency in similarly classified cleft types was variable, #

2015 Mutaz B. Habal, MD

FIGURE 8. A and B, Serial 3D cast growth studies. Two cases where the palatal segments, although they are of different sizes, the large and smaller cleft segments grew at the same velocity no matter the size of the cleft space and the palatal segments. This happened in CBCLP cases as well. C and D, Palatal growth and velocity changes of all clinic cases. The non-cleft control series, shown at top, shows total larger palatal size than any of the cleft cases. All cases grew more rapidly the first 2 years and then continued in similar growth but at a slower velocity.

1147

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Berkowitz

The Journal of Craniofacial Surgery

The Hunt for Good Facial Aesthetics and Palatal Closure Protocols To appreciate the creation of PO, one needs to return to the early 1900 s to view the surgical-orthopedic hunt that used POPLA and then NAM þ GPP with the continued modifications of previous unsuccessful surgeries and variations in presurgical orthopedics. An early example of this unsuccessful hunt for the best surgical protocol was exemplified in the 1920 s by Brophy,11 a wellrespected surgeon. He believed that in CUCLP/CBCLP, the newborn’s displaced palatal segments were of normal size, and when brought together into perfect alignment soon after birth by presurgical orthopedics, followed by surgical palatal closure, the palate and face would develop normally. The treatment concept failed and resulted in severely malformed faces. The search for the best surgical-orthopedic procedure continued, and in the 1980 s, Latham,71 an orthodontist influenced by Scott, developed a new orthopedic palatal appliance. As I stated, this device was pinned to the malpositioned hard palate segments in the 6- to 8month-old infant. In CUCLP and CBCLP cases, the alveolar segments were quickly brought into a butt contact. Despite the lack of concurring long-term follow-up reports from Canada, Latham’s PO procedure was quickly adopted by Georgiade and Latham72 at Duke and at many other American clinics. Eventually Dr. D. Ralph Millard Jr., at the South Florida Cleft Palate Clinic, joined with Latham to create a revised protocol, using presurgical orthopedics with periosteoplasty, followed by lip adhesion (POPLA) (Figs. 7A–C). Latham’s71 preliminary introductory report on what he hoped the procedure would accomplish suggested that, in both CUCLP and CBCLP, the forces created by the fixed palatal appliance would bring the newborn’s retropositioned cleft lateral segments forward to make contact with the protruding premaxilla to close the anterior cleft space, and in CUCLP, it would bring the palatal segment forward into alveolar contact with the larger segment. To date, Latham has failed to perform any follow-up cephs with dental cast studies to support his and Scott’s hypothesis that the retruded newborn lateral palatal segments needed to be advanced to correct a recessive midface. Instead, in these POPLA cases, the premaxilla was bodily retruded, resulting in extensive midfacial recessiveness, with anterior crossbites due to closed lateral incisor spaces bound by a boney bridge. In CBCLP non-POPLA cases, limiting the premaxillary facial elastic forces to premaxillary ventroflexation is similar to what occurs after lip adhesion, which allows the premaxilla to be later uprighted and properly aligned to the lateral palatal segments. Had the premaxilla been bodily retruded, the premaxillary vomerine suture (PVS) would have become synostatic. This predictable outcome occurs as well with the now-popular procedure, called nasoalveolar molding with gingivoperiosteoplasty (NAM þ GPP) (Grayson et al73). Unfortunately, in one serial ceph study performed up to now, Hsin-Yi Hsieh et al74 reported that NAM þ GPP caused severe midfacial recessiveness as seen in POPLA-treated CBCLP and some CUCLP cases. It is interesting to note that Cutting had used the POPLA procedure for 15 years and then switched to NAM þ GPP. He believed that the modification of the mechanical retrusive forces from a different palatal appliance would be more physiological and lead to good midfacial growth. Without objective supporting outcome studies, one can thus safely conclude that another treatment modification has failed.

The Need for Differential Diagnosis As previously mentioned regarding timing of palatal cleft closure, some cases showed that early closure of the palatal cleft space in nonPO cases and palatal closure performed before 8 months old might still result in good palatal growth, as long as the ratio of the cleft space size was relatively small, when compared to the size of the palatal

1148



Volume 26, Number 4, June 2015

segments, so that midfacial growth enabled the upper to lower anterior arches to achieve congruency after limited orthodontics. After the 40year comparative outcome 3D cast study, Millard and Berkowitz concluded that the surgeon must accept that the repeated modifications of past surgical procedures were not the sole answer in determining the best way to predict treatment success. Berkowitz concluded that because physical assets and deficits of each case vary greatly, consideration must be given to the diversity of facial growth patterns, level of palatal bone deficiencies, and nasopharyngeal skeletal/muscle variations that, when put together, are the critical physical differences that may exist within similar cleft types and faces. In contrast to early cleft palatal closure in all cases, Berkowitz et al61 found that surgery at the cleft space, performed at approximately 18 to 24 months and maintaining a normal PVS, has led to promising facial and palatal growth and speech in most cases. This is not to say that if undetermined sufficient palatal bone appears soon after birth, one could still achieve good palatal and facial growth. Unfortunately, the degree of palatal bone deficiency cannot be determined at that early age because palatal growth studies (Figs. 3A, B and 8A-D) have shown that all palatal clefts are not physically the same in palatal bone content despite their cleft size before and after molding. Time remains a useful ally.

CONCLUSION This review emphasizes the concept that all clefts within a particular cleft type, although similarly classified, still differ in the degree of palatal osteogenic deficiency, as well as in facial growth pattern and nasopharyngeal space. Achieving a delicate balance between surgery and normal facial growth entails discarding the immediate goals of excellent early facial aesthetics in favor of the long-term outcomes of good facial growth, clear speech, proper occlusion, and normal psychosocial development.

ACKNOWLEDGMENTS All photographs and graphs appear in ‘Cleft Lip and Palate: Diagnosis and Management’ 3rd Edit. Editor Samuel Berkowitz. Permission granted by Springer-Verlag, Heidelberg, Berlin, New York 20013.

REFERENCES 1. Bardach J. The influence of cleft lip repair on facial growth. Cleft Palate J 1990;27:76–78 2. Berkowitz S. A comparison of treatment results in complete bilateral cleft lip and palate using a conservative approach versus MillardLatham PSOT procedure. Semin Orthod 1996;1 (3):169–184 3. Bongaarts A, Kuijpers-Jagtman A, Van’t Hof M, et al. Effect of infant orthopedics in UCLP. Cleft Palate Craniofac J 2004;41:633–641 4. Latham RA. Development and structure of the premaxillary deformity in bilateral cleft lip and palate. Br J Plast Surg 1973;26:1–11 5. Millard DR Jr, Latham RA. Improved surgical and dental treatment of clefts. Plast Reconstr Surg 1990;86:856–871 6. Rosenstein SW, Kernaham DA. The case for early bone grafting in cleft lip and palate. Plast Reconstr Surg 1982;70:297–309 7. Mulliken JB. Bilateral complete cleft lip repair. Plast Reconstr Surg 1985;76:477–487 8. Cutting C, Grayson B. Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip and alveolus of infants born with unilateral and bilateral clefts. Cleft Palate J 2000;37:528–532 9. Grayson BH, Cutting CB. Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip and alveolus of infants born with unilateral and bilateral clefts. Cleft Palate Craniofac J 2001;38:193–198 10. Pruzansky S, Aduss H, Berkowitz S, et al. Monitoring growth of the infant with cleft lip and palate. Trans Eur Orthod Soc 1973:538–546 11. Brophy TW. Cleft Lip and Palate. Philadelphia, PA: Blakiston’s; 1923: 131–132 #

2015 Mutaz B. Habal, MD

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

The Journal of Craniofacial Surgery



Volume 26, Number 4, June 2015

12. Dorrance GM. Lengthening of the soft palate in cleft palate operations. Ann Surg 1925;82:208 13. Cronin TD. Method of preventing raw area on nasal surface of soft palate in pushback surgery. Plast Reconstr Surg 1957;20:474–484 14. Crikelair GF, Bom AF, Luban J, et al. Early orthodontic movement of cleft maxillary segments prior to cleft lip repair. Plast Reconstr Surg 1962;30:426–440 15. Robertson NRE, Fish J. Early dimensional changes in the arches of cleft palate children. Am J Orthod 1975;12:59–84 16. Chapman KL. Phonologic processes in children with cleft palate. Cleft Palate Craniofac J 1993;30:64–72 17. Hotz R. The indications for preoperative and postoperative orthopedic treatment of cleft lip and palate. In: Hotz R, ed. Early Treatment of Cleft Lip and Palate. Berne: Hans Guber; 1964:78–89 18. Monroe CW, Rosenstein SW. Maxillary orthopedics and bone grafting in cleft palate. In: Grabb WC, Rosenstein SW, Bzoch KR, eds. Cleft Lip and Palate.. Boston: Little Brown; 1971 19. Pruzansky S. Description, classification, and analysis of unoperated clefts of the lip and palate. Am J Orthod 1953;39:59 20. Pruzansky S. The growth of the premaxillary-vomerine complex in complete bilateral cleft lip and palate. Tandlaegebladet 1971;75:1157– 1169 21. Harvold E. Cleft lip and palate morphologic studies of the facial skeleton. Am J Orthod 1954;40:493–506 22. Pruzansky S, Aduss H. Prevalence of arch collapse and malocclusion in complete unilateral cleft lip and palate. Rep Congr Eur Orthod Soc 1967:365–382 23. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Parts I, V, VII. Cleft Palate J 1987;24:05 24. Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Part VII: an overview of treatment and facial growth. Cleft Palate J 1987;24:71–77 25. Kremenak CR, Huffman WC, Olin WM. Maxillary growth inhabitation by mucoperiosteal denudation of palatal shelf bone in non-cleft beagles. Cleft Palate J 1970;7:817–825 26. Aduss H. Craniofacial growth in complete unilateral cleft lip and palate. Cleft Palate J 1971;41:202–212 27. Hanada K, Krogman W. A longitudinal study of postoperative changes in cleft lip and palate from birth to 6 years. Am J Orthod 1975;57:363– 376 28. Friede H. Studies on facial morphology and growth in bilateral cleft lip and palate. Ph.D. Thesis, Goteborg, Sweden, University of Goteborg. 1977 29. Friede H. The vomero-premaxillary suture—a neglected growth site in mid-facial development of unilateral cleft lip and palate patients. Cleft Palate J 1978;15:398 30. Witzel MA, Salyer KE, Ross RB. Delayed hard palate closure: the philosophy revisited. Cleft Palate J 1980;21:263–269 31. Vargervik K. Growth characteristics of the premaxilla and orthodontic treatment principles in bilateral cleft lip and palate. Cleft Palate J 1983;20:289 32. Shprintzen RJ, Siegel-Sadewitz VL, Amato J, et al. Anomalies associated with cleft lip, cleft palate, or both. Am J Med Genet 1985;20:585–595 33. Van Demark DR, Gnoinski W, Hotz MM, et al. Speech results of the Zurich approach in the treatment of unilateral cleft lip and palate. Plast Reconstr Surg 1989;83:605–613 34. Semb G. A study of facial growth in patients with bilateral cleft lip and palate treated by the Oslo CLP team. Cleft Palate Craniofac J 1991;28:1–22 35. Semb G, Shaw W. Simonart’s band and facial growth in unilateral clefts of the lip and palate. Cleft Palate Craniofac J 1991;28:40–46 36. Shaw W, Semb G. Facial growth related to surgical methods. In: Berkowitz S, ed. Cleft Lip and Palate: Diagnosis and Management. 3rd ed. Berlin-Heidelberg: Springer-Verlag; 2013 37. Friede H, Lilja J, Lohmander A. Two stage palatal surgery with early veloplasty and delayed hard palate repair: a balanced view on speech and growth outcome. In: Berkowitz S, ed. Cleft Lip and Palate: Diagnosis and Management. 3rd ed. Berlin & Heidelberg: SpringerVerlag; 2013 38. Moss ML. The primary role of functional matrices in orofacial growth. Dent Pract 1964;8:65 #

2015 Mutaz B. Habal, MD

Diagnosis of Facial Skeleton/Musculature

39. Krogman WM, Mazaheri M, Harding RL. A longitudinal study of craniofacial growth in children with clefts as compared to normal birth to six years. Cleft Palate J 1975;12:59–84 40. Berkowitz S, ed. Cleft Lip and Palate: Diagnosis and Management. 3rd ed. Heidelberg, New York, Berlin: Springer Verlag, 2013 41. Rosenberger HC. Growth and development of the nasorespiratory area in childhood. Ann Otol Rhinol Laryngol 1934;43:495 42. King EW. A roentgenographic study of pharyngeal growth. Angle Orthod 1952;22:23 43. Friedman LM, Furberg CD, DeMets DL. Fundamentals of Clinical Trials. 3rd ed. St Louis: Mosby; 1996 44. Shifman A, Finkelstein Y, Nachmani A, et al. Speech-aid prostheses for neurogenic velopharyngeal incompetence. J Prosth Dent 2000;83:99–106 45. Scott JH. The analysis of facial growth. Part 1. The anteroposterior and vertical dimensions. Am J Orthod 1965;44:507 46. Scott JH. The analysis of facial growth. Part II. The horizontal and vertical dimensions. Am J Orthod 1965;44:585 47. Graf-Pinthus B, Bettex M. The narrowing of the palatal cleft following presurgical orthopedic treatment in complete clefts of the lip and palate. Cleft Palate J 1974;11:253–260 48. Hotz M, Gnoinski W. Comprehensive care of cleft lip and cleft palate children at Zurich University: a preliminary report. Am J Orthod 1976;70:481–504 49. Weil J. Orthopaedic growth guidance and stimulation for patients with cleft lip and palate. Scand J Plast Reconstr Surg 1987;21:57–64 50. Gnoinski WM. Early maxillary orthopedics as a supplement to conventional primary surgery in complete cleft lip and palate patients. J Maxillofac Surg 1982;10:165–171 51. Berkowitz S. State of the art in cleft palate, orofacial growth. Am J Orthod 1978;74:5564–576 52. Friede H, Johanson BA. Follow-up study of cleft children treated with primary bone grafting. Scand J Plast Reconstr Surg 1974;8:88–103 53. Friede H, Johanson B. Adolescent facial morphology of early bone grafted cleft lip and palate patients. Scand J Plast Reconstr Surg 1982;16:41–53 54. Millard DR Jr. Cleft Craft. Vol. 1: The Unilateral Deformity. Boston: Little Brown; 1976 55. Millard DR Jr. Cleft Craft: Part III—The Evolution of Its Surgery: Alveolar and Palatal Deformity. Boston: Little Brown; 1980 56. Friede H, Lilja J, Johanson B. Cleft lip and palate treatment with delayed closure of the hard palate. Scand J Plast Reconstr Surg 1980;14:49–53 57. Berkowitz S. Timing cleft palate closure—age should not be the sole determinant. J Craniofac Genet Dev Biol (Suppl) 1985;1:69–83 58. Berkowitz S, Mejia M, Bystrik A. A comparison of the effects of the Latham-Millard procedure with those of a conservative treatment approach for dental occlusion and facial aesthetics in unilateral and bilateral complete cleft lip and palate: part I. Dental occlusion. Plast Reconstr Surg 2004;113:1–18 59. Berkowitz S. Timing of cleft palate closure should be based on the ratio of the area of the cleft to that of the palatal segments and not on age alone Ch. 17. In: Berkowitz S, ed. Cleft Lip and Palate: Diagnosis and Management. 2nd ed. Heidelberg. New York, Berlin: Springer Verlag; 2006 60. Berkowitz S. Facial growth in BCLP from birth to eight years of age. Masters Thesis, University of Illinois 1959. 61. Lee CT, Garfinkle JS, Warren SM, et al. Nasoalveolar molding improves appearance of children with bilateral cleft-lip-cleft lip palate. Plast Reconstr Surg 2008;122:1131–1137 62. Berkowitz S, Duncan R, Evans C, et al. Timing of cleft palate closure should be based on the ratio of the area of the cleft to that of the palatal segments and not on age alone. Plast Reconstr Surg 2005;115:113–149 63. Chapman KL, Hardin MA. Phonetic and phonologic skills of two-yearolds with cleft palate. Cleft Palate Craniofac J 1992;29:435–443 64. DeLuke DM, Marchand A, Robles EC, et al. Facial growth and the need for orthognathic surgery after cleft palate repair. J Oral Maxillofac Surg 1997;55:694–697 65. Chapman KL, Hardin-Jones MA, Goldstein JA, et al. Timing of palatal surgery and speech outcomes. Cleft Palate Craniofac J 2008;45:297– 308 66. Peterson-Falzone SJ. A cross-sectional analysis of speech results following palatal closure. In: Bardach J, Morris HL, eds. Multidisciplinary Management of Cleft Lip and Palate. Philadelphia: WB Saunders; 1990:750–757

1149

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Berkowitz

The Journal of Craniofacial Surgery

67. Peterson-Falzone SJ. The relationship between timing of cleft palate surgery and speech outcome: what have we learned, and where do we stand in the 1990 s? Semin Orthodon 1996;2:185–191 68. Peterson-Falzone S. Optimal timing of palatoplasty to facilitate normal speech development. What is the evidence? In: Berkowitz S, ed. Cleft Lip and Palate: Diagnosis and Management. Berlin-Heidelberg: Springer-Verlag; 2013 69. Bzoch KR. Clinical studies of the efficacy of speech appliances compared to pharyngeal flap surgery. Cleft Palate J 1964;35:275 70. Dorf DS, Curtin J. Early cleft palate repair and speech outcome. Plast Reconstr Surg 1982;68:153–157

1150



Volume 26, Number 4, June 2015

71. Latham RA. Orthopedic advancement of the cleft maxillary segment: a preliminary report. Cleft Palate J 1980;17:227 72. Georgiade NG, Latham RA. Maxillary arch alignment in the bilateral cleft lip and palate infant using the pinned coaxial screw appliance. J Plast Reconstr Surg 1975;52:52–60 73. Grayson BH, Santiago PE, Brecht LE, et al. Presurgical nasoalveolar molding in infants with cleft lip and palate. Cleft Palate Craniofac J 1999;36:486–498 74. Hsin-Yi Hsieh C, Wen-Ching Ko E, Kuo-Ting Chin P, et al. The effect of gingivoperiosteoplasty on facial growth in patients with complete unilateral cleft lip and palate. Cleft Palate Craniofac J 2010;47:439–446

#

2015 Mutaz B. Habal, MD

Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.

Palate Literature Reveals That Differential Diagnosis of the Facial Skeleton and Musculature is Essential to Achieve All Treatment Goals.

After 40 years of monitoring cleft palate treatment results with extensive objective records of cephaloradiographs, dental casts, and photographs, it ...
865KB Sizes 0 Downloads 7 Views