"Long term speech outcomes after using the Sommerlad technique for primary palatoplasty: a retrospective study in the Wilhelmina Children's Hospital, Utrecht."

OBJECTIVES: After cleft lip and/or palate (CL/P) repair, children may develop velopharyngeal insufficiency (VPI) leading to speech imperfections, necessitating additional speech correcting surgery. This study examines the incidence of VPI and speech correcting surgery after Sommerlad's palatoplasty for CL/P, and its association with various clinical features. MATERIALS AND METHODS: A retrospective cohort study was performed in the Wilhelmina Children's Hospital in Utrecht and child records from 380 individuals with CL/P registered from 2008 to 2017 were retrospectively reviewed. Inclusion criteria comprised the diagnosis of CL/P, primary palatoplasty according to Sommerlad's technique, and speech assessment at five years or older. Association between cleft type and width, presence of additional genetic disorders and postoperative complications (palatal dehiscence, fistula) were assessed using odds ratios and chi squared tests. RESULTS: A total of 239 patients were included. The VPI rate was 52.7% (n = 126) and in 119 patients (49.8%) a speech correcting surgery was performed. Severe cleft type, as indicated by a higher Veau classification, was associated with a significant higher rate of speech correcting surgeries (p = 0.033). Significantly more speech correcting surgeries were performed in patients with a cleft width >10 mm, compared to patients with a cleft width ≤10 mm (p < 0.001). Patients with oronasal fistula underwent significantly more speech correcting surgeries than those without fistula (p = 0.004). No statistically significant difference was found in the incidence of speech correcting surgery between patients with and without genetic disorders (p = 0.890). CONCLUSIONS/CLINICAL RELEVANCE: Variations in cleft morphology, cleft width and complications like oronasal fistula are associated with different speech outcomes. Future research should focus on creating a multivariable prediction model for speech correcting surgery in CL/P patients.

Speech correcting surgery after primary palatoplasty: a systematic literature review and meta-analysis.

OBJECTIVES: In cleft palate patients, the soft palate is commonly closed using straight-line palatoplasty, Z-palatoplasty, or palatoplasty with buccal flaps. Currently, it is unknown which surgical technique is superior regarding speech outcomes. The aim of this review is to study the incidence of speech correcting surgery (SCS) per soft palatoplasty technique and to identify variables which are associated with this outcome. MATERIALS AND METHODS: A systematic literature search was carried out according to the PRISMA guidelines. Inclusion and exclusion criteria were applied to focus on the incidence of SCS after soft palatoplasty. Additional variables like surgical modification, cleft morphology, syndrome, age at palatoplasty, fistula and assessment of velopharyngeal function were reported. A modified New-Ottawa Scale (NOS) was used for quality appraisal. Pooled estimates from the meta-analysis were calculated using a random-effects model. RESULTS: One thousand twenty-nine studies were found of which 54 were included in the analysis. The pooled estimate proportion of SCS after straight-line palatoplasty was 19% (95% CI 15-24), after Z-palatoplasty 6% (95% CI 4-9), and after palatoplasty with buccal flaps 7% (95% CI 4-11). CONCLUSIONS: A lower SCS rate was found in patients receiving Z-palatoplasty when compared to straight-line palatoplasty. We propose a minimum set of outcome parameters which ideally should be included in future studies regarding speech outcomes after cleft palate repair. CLINICAL RELEVANCE: Current literature reports highly heterogenous data regarding cleft palate repair. Our recommended set of parameters may address this inconsistency and could make intercenter comparison possible and of better quality.

Bulging after DIEP Breast Reconstruction: New Insights Concerning Rectus Diastasis and Medial Perforator Harvest.

The aim of this study was to evaluate the association between flap harvest technique and occurrence of abdominal bulging. Methods: A retrospective analysis of 159 patients undergoing DIEP flap breast reconstruction between 2014 and 2021 in the University Medical Center Utrecht was conducted. Outcomes measured were preoperative rectus diastasis, flap weight, laterality of flap harvest (unilateral or bilateral), timing of the harvest (immediate or delayed), number of perforators harvested (single or multiple), and location of the harvested perforator (medial, lateral, or both). Results: In 159 patients, 244 DIEP flaps were performed, 16 of these donor-sites (6.6%) developed a clinically evident abdominal bulge. When preoperative rectus abdominis diastasis was found (n = 97), postoperative bulging occurred significantly more often (P < 0.01). Patients in whom the medial perforator artery was harvested for reconstruction (n = 114) showed less abdominal bulging than patients in whom the lateral (n = 92) was harvested (P = 0.02). Using single versus multiple perforators for the DIEP flap, bilateral versus unilateral reconstruction or timing of the operation showed no significant difference in outcome of bulging (P = 1.00, P = 0.78, P = 0.59, respectively). Conclusions: The incidence of bulging in our study cohort is comparable to the literature. Harvesting the medial perforator artery for the DIEP flap showed less abdominal bulging than using the lateral perforator artery in a DIEP flap breast reconstruction. Also, preoperative rectus diastasis was found to be an important risk factor for the occurrence of bulging.

Reconstructive Strategies in Pediatric Patients after Oncological Chest Wall Resection: A Systematic Review.

An appropriate reconstruction strategy after surgical resection of chest wall tumors in children is important to optimize outcomes, but there is no consensus on the ideal approach. The aim of this study was to provide an up-to-date systematic review of the literature for different reconstruction strategies for chest wall defects in patients less than 18 years old. A systematic literature search of the complete available literature was performed and results were analyzed. A total of 22 articles were included in the analysis, which described a total of 130 chest wall reconstructions. All were retrospective analyses, including eight case reports. Reconstructive options were divided into primary closure (n = 21 [16.2%]), use of nonautologous materials (n = 83 [63.8%]), autologous tissue repair (n = 2 [1.5%]), or a combination of the latter two (n = 24 [18.5%]). Quality of evidence was poor, and the results mostly heterogeneous. Reconstruction of chest wall defects can be divided into four major categories, with each category including its own advantages and disadvantages. There is a need for higher quality evidence and guidelines, to be able to report uniformly on treatment outcomes and assess the appropriate reconstruction strategy.

Assessment of Health-Related Quality of Life in Robin Sequence: A Comparison of Mandibular Distraction Osteogenesis and Tongue-Lip Adhesion.

BACKGROUND: Numerous studies have proven the efficacy of mandibular distraction osteogenesis or tongue-lip adhesion in Robin sequence infants with upper airway obstruction. However, none has compared health-related quality of life outcomes. METHODS: In the present retrospective study, Robin sequence infants younger than 1 year, who underwent mandibular distraction osteogenesis or tongue-lip adhesion, were included (2006 to 2016). The infants' caregivers were asked to complete a questionnaire based on the Glasgow Children's Benefit Inventory. RESULTS: The response rate was 71 percent (22 of the 31 questionnaires; mandibular distraction osteogenesis, 12 of 15; and tongue-lip adhesion, 10 of 16) and median age at surgery was 24 days (range, 5 to 131 days). Median total Glasgow Children's Benefit Inventory scores after mandibular distraction osteogenesis and after tongue-lip adhesion were 21.9 (interquartile range, 9.4) and 26.0 (interquartile range, 37.5), respectively (p = 0.716), indicating an overall benefit from both procedures. Positive changes were observed in all subgroups emotion, physical health, learning, and vitality. In syndromic Robin sequence, both procedures demonstrated a lower positive change in health-related quality of life compared with isolated Robin sequence (p = 0.303). CONCLUSIONS: Both surgical procedures demonstrated an overall benefit in health-related quality-of-life outcomes, with no significant differences. The authors' findings contribute to the debate regarding the use of mandibular distraction osteogenesis versus tongue-lip adhesion in the surgical treatment of Robin sequence; however, studies evaluating health-related quality of life in larger Robin sequence cohorts are necessary to identify which procedure is likely to be best in each individual Robin sequence infant. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Long-Term Results of Mandibular Distraction Osteogenesis with a Resorbable Device in Infants with Robin Sequence: Effects on Developing Molars and Mandibular Growth.

BACKGROUND: Mandibular distraction osteogenesis with a unidirectional resorbable device is an effective treatment option for severe upper airway obstruction in infants with Robin sequence. Long-term effects, especially with regard to tooth development and mandibular outgrowth, are not known. METHODS: Robin sequence infants with a follow-up of greater than or equal to 5 years were included. Baseline characteristics were extracted from medical records. Panoramic and lateral cephalometric radiographs were analyzed and patients were recalled for physical examination. RESULTS: Ten infants underwent mandibular distraction osteogenesis at a mean age of 3.7 months (median, 19 months; range, 11 days to 27 months). Mean length of follow-up was 6.8 years (range, 5.0 to 7.9 years). Ten Robin sequence infants without mandibular distraction osteogenesis (mean length of follow-up, 7.4 years; range, 6.7 to 8.9 years) were the controls. Shape anomalies, positional changes, and root malformations of molars were seen significantly more often than in the control group (p = 0.007, p = 0.009, and p = 0.043, respectively). Mandibular length was shorter (p = 0.030), but mandibular ramus height was comparable (p = 0.838) with that of the non-mandibular distraction osteogenesis group. Compared with healthy controls, all Robin sequence infants had a significantly shorter mandible. CONCLUSIONS: Mandibular distraction osteogenesis with a resorbable system reveals overall good short- and long-term results, but the effects on developing molars and mandibular outgrowth likely necessitate secondary procedures. This factor should be considered when deciding on treatment options and counseling of parents. CLINICAL QUESTION/LEVEL OF EVIDENCE: Therapeutic, III.

Etiology and pathogenesis of robin sequence in a large Dutch cohort.

Robin sequence (RS) can be defined as the combination of micrognathia and upper airway obstruction/glossoptosis causing neonatal respiratory problems, with or without a cleft palate and either isolated or non-isolated. Pathogenesis varies widely. We hypothesize that optimal treatment depends on pathogenesis and therefore patients should be stratified according to diagnosis. Here, we evaluate diagnoses and (presumed) pathogeneses in an RS cohort. Medical records of all RS patients presenting between 1995-2013 in three academic hospitals were evaluated. Four clinical geneticists re-evaluated all information, including initial diagnosis. Diagnoses were either confirmed, considered uncertain, or rejected. If uncertain or rejected, patients were re-evaluated. Subsequent results were re-discussed and a final conclusion was drawn. We included 191 RS patients. After re-evaluation and changing initial diagnoses in 48 of the 191 patients (25.1%), 37.7% of the cohort had isolated RS, 8.9% a chromosome anomaly, 29.3% a Mendelian disorder, and 24.1% no detectable cause. Twenty-two different Mendelian disorders were diagnosed, of which Stickler syndrome was most frequent. Stratification of diagnoses according to (presumed) pathogenic mechanism in 73 non-isolated patients with reliable diagnoses showed 43.9% to have a connective tissue dysplasia, 5.5% a neuromuscular disorder, 47.9% a multisystem disorder, and 2.7% an unknown mechanism. We diagnosed more non-isolated RS patients compared to other studies. Re-evaluation changed initial diagnosis in a quarter of patients. We suggest standardized re-evaluation of all RS patients. Despite the relatively high diagnostic yield pathogenesis could be determined in only 59.7% (71/119), due to limited insight in pathogenesis in diagnosed entities. Further studies into pathogenesis of entities causing RS are indicated.

Tracheostomy versus mandibular distraction osteogenesis in infants with Robin sequence: a comparative cost analysis.

Many treatments have been described for infants with Robin sequence and severe respiratory distress, but there have not been many comparative studies of outcome and cost-effectiveness. The aim of this study was to compare the cost and complications of two common interventions - mandibular distraction osteogenesis and tracheostomy. Nine patients with isolated Robin sequence (mandibular distraction osteogenesis, n=5, and tracheostomy, n=4) were included in the analyses. Predetermined costs and complications were obtained retrospectively from medical records and by questionnaires to the parents over a 12-month period. Overall direct costs (admission to hospital, diagnostics, surgery, and homecare) were 3 times higher for tracheostomy (€105.523 compared with €33.482, p=0.02). Overall indirect costs (absence from work) were almost 5 times higher (€2.543 compared with €543, p=0.02). There was a threefold increase in overall total cost/patient (both direct and indirect) for tracheostomy (€108.057 compared with 34.016, p=0.02) and 4 times more complications were encountered. This study shows that mandibular distraction osteogenesis in infants diagnosed with Robin sequence costs significantly less and results in fewer complications than tracheostomy, and this contributes to our current knowledge about the ideal approach for infants with Robin sequence and might provide a basis for institutional protocols in the future.

A cadaver study of the vascular territories of dominant and nondominant internal mammary artery perforators.

The perfusion territory of the pedicled internal mammary artery perforator flap has been described, but the number of perforators to be included in the flap's pedicle is controversial. We studied the vascular territory of the dominant perforator and the contribution of additional nondominant perforators to it. Therefore, the dominant perforators in 9 fresh cadavers and the nondominant perforators in 4 of these, were injected with water-based ink. The dominant perforator vascularized a territory extending from the superior border of the clavicle to the xiphoid, and from midsternal to the anterior axial fold, with a mean craniocaudal length of 19.4 cm (range, 17.0-24.0) and a mean mediolateral width of 18.6 cm (range, 16.0-22.5). Additional injection of nondominant perforators did not lead to any substantial enlargement of this territory. One single dominant perforator vascularizes a large part of the hemithorax, allowing for various flap designs. Nondominant perforators do not have to be included in the vascular pedicle of the internal mammary artery perforator flap, which leads to less donor-site morbidity.

Anatomy of the vascular pedicle of the internal mammary artery perforator (IMAP) flap as applied for head and neck reconstruction.

INTRODUCTION: The vascular pedicle of the internal mammary artery perforator (IMAP) flap and its enhancement by inclusion of the internal mammary vessels up to the level of the first rib have not been systematically assessed anatomically, to date. This study assesses these features of this pedicle in light of the flap's application in head and neck reconstruction. MATERIAL AND METHODS: The length of the dominant perforator and the enhanced vascular pedicle of the IMAP flap were measured in 27 fresh cadaveric hemi-thoraxes. RESULTS: In 18 out of 27 cadaveric cases (0.67) the second perforator was dominant. The mean length of the 27 dominant perforators was 47 mm (range: 30-66 mm). By enhancement up to the level of the first rib, the mean length of the vascular pedicle could be doubled to 92 mm when based on the second perforator (0.67). In the five cases (0.19), where the third perforator was dominant, the mean length of the enhanced pedicle was 104 mm, whereas it was 61 mm in the four cases (0.15) where the first perforator was dominant. CONCLUSION: This is the first study that assessed the enhanced length of the vascular pedicle of the IMAP flap. Our results show that the mean length of the dominant second perforator may be doubled by such enhancement and that the arc of rotation to the head and neck region may, then, be doubled again. Hereby, even high-located defects in the head and neck region can be reached. This provides a better understanding of the feasibility of the pedicled IMAP flap in head and neck reconstruction.

Clinical application and outcome of the internal mammary artery perforator (IMAP) free flap for soft tissue reconstructions of the upper head and neck region in three patients.

BACKGROUND: The fasciocutaneous internal mammary artery perforator (IMAP) island flap allows for superior esthetical and functional skin cover in the head and neck region in combination with limited donor site morbidity. Its modification as a free flap allows reconstruction of more cranial defects. PATIENTS AND METHODS: Three IMAP free flaps varying from 7 × 4 cm² to 10 × 6 cm² were transplanted in three patients with a mean age of 59 years (range, 54-69 years). Enhancement of the flap's vascular pedicle at least doubles the diameter of the internal mammary vessels to be anastomosed. RESULTS: Coverage with excellent texture and color match was uneventfully obtained and the flaps' donor sites were primarily closed in all three cases. CONCLUSIONS: Our experience proves the consistent feasibility of successful transplantation of the IMAP free flap. Because of its characteristics, we suggest contemplating the use of this flap in the upper head and neck region.

Perioral wrinkles: histologic differences between men and women.

BACKGROUND: Women tend to develop more and deeper wrinkles in the perioral region than men. Although much is known about the complex mechanisms involved in skin aging, previous studies have described histologic differences between men and women with respect to skin aging only incidentally and have not investigated the perioral region. OBJECTIVE: The purpose of this study was to investigate gender-specific differences in the perioral skin. METHODS: To determine wrinkle severity, skin surface replicas of the upper lip region in 10 male and 10 female fresh cadavers were analyzed by using the dermaTOP blue three-dimensional digitizing system (Breuckmann, Meersburg, Germany). In 30 fresh male and female cadavers, three full-thickness lip resections were investigated in a blinded fashion for specific histologic features. All results were statistically analyzed in a linear regression model with SPSS software (version 15.0; SPSS, Chicago, IL). RESULTS: The female replicas showed more and deeper wrinkles than the male replicas (P < .01). Histologic analysis revealed that the perioral skin of men displayed a significantly higher number of sebaceous glands (P = .000; 95% confidence interval [CI] 23.6-53.2), sweat glands (P = .002; 95% CI 2.1-8.1), and a higher ratio between vessel area and connective tissue area in the dermis (P = .009; 95% CI 0.003-0.021). The amount of hair follicles did not significantly differ between men and women, although the average number of sebaceous glands per hair follicle was greater in men (P = .002; 95% CI 0.33-1.28). CONCLUSIONS: Women exhibit more and deeper wrinkles in the perioral region and their skin contains a significantly smaller number of appendages than men, which could be a feasible explanation for why women are more susceptible to development of perioral wrinkles.