Reducing delayed detection of isolated cleft palate-related deformity: a call for routine intraoral examination of newborns

Denadai, Rafael; Lo, Lun-Jou

doi:10.1016/j.jped.2023.12.005

Rafael Denadai ^** Corresponding author. E-mail: denadai.rafael@hotmail.com (R. Denadai).

conceived the idea for the manuscript

reviewed the literature

drafted the manuscript

Centro Avançado de Cirurgia Plástica Facial, A&D DermePlastique, São Paulo, SP, Brazil

Centro Universitário Max Planck (UniMAX), Faculdade de Medicina, Indaiatuba, SP, Brazil

Chang Gung Memorial Hospital, Craniofacial Research Center, Taoyuan, Taiwan

https://orcid.org/0000-0003-0525-3480

Lun-Jou Lo

conceived the idea for the manuscript

reviewed the literature

drafted the manuscript

Chang Gung Memorial Hospital, Craniofacial Research Center, Taoyuan, Taiwan

Chang Gung University, Chang Gung Memorial Hospital, Department of Plastic and Reconstructive Surgery, Taoyuan, Taiwan

https://orcid.org/0000-0003-1606-7325

* Corresponding author. E-mail: denadai.rafael@hotmail.com (R. Denadai).

Conflicts of interest

The authors declare no conflicts of interest.

Figures (3)
Tables (1)

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Figure. 1
(Left) Schematic drawings illustrating cleft palate deformities, encompassing instances featuring (Left, top) solely the soft palate, (Left, bottom) both the soft and hard palates, and (Right) an overt submucous cleft palate deformity. These deformities are categorized using Noordhoff’s modified double-numbered Y classification system. Courtesy of Rafael Denadai, M.D.

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Figure. 2
Schematic drawings illustrating the (Left) normal anatomy of the palate and (Right) the abnormal anatomy in an isolated cleft palate deformity. (Right) The cleft muscle mass (including the levator muscle) runs almost parallel with the cleft margin before it inserts aberrantly into the posterior border of the hard palate. The anterior tendinous fibers of the tensor attach to the lateral aspect of the posterior edge of the hard palate. The aberrant positioning of the cleft muscle mass, along with an abnormal fusion with the tendon of the tensor muscle, is believed to impair the function of the tensor muscle in assisting with Eustachian tube function. The aberrantly inserted cleft muscle mass results in ineffective contraction and an inability to close the palate against the posterior pharyngeal wall. Courtesy of Rafael Denadai, M.D.

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Figure. 3
Intraoral images displaying the spectrum of isolated cleft palate deformity, highlighted by a gray arrow to indicate the escalating severity of abnormalities. Tongue depressors assist in gently retracting the tongue, providing a broader intraoral view. Simultaneously, direct lighting helps to illuminate the intraoral space, ensuring adequate brightness and contrast to observe the specific details of the palatal structures. Courtesy of Rafael Denadai, M.D.

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Table 1
Studies reporting delayed diagnosis of isolated cleft palate. ¹²12 Hanny KH, de Vries IA, Haverkamp SJ, Oomen KP, Penris WM, Eijkemans MJ, et al. Late detection of cleft palate. Eur J Pediatr. 2016;175:71–80., ¹³13 Tierney S, Blackhurst M, Scahill R, Callery P. Loss and rebuilding: a qualitative study of late diagnosis of cleft palate. J Spec Pediatr Nurs. 2015;20:280–9., ¹⁴14 Van Veen-van der Hoek M, Jedeloo S, Bredero-Boelhouwer H, Balk-Leurs I, Ongkosuwito E. Effects of delayed diagnosis in cleft palate: a preliminary qualitive study on the experience of parents. Verpleegkd Spec. 2014;9:30–5., ¹⁵15 Amstalden-Mendes LG, Xavier AC, Antunes DK, Ferreira AC, Tonocchi R, Fett-Conte AC, et al. Time of diagnosis of oral clefts: a multicenter study. J Pediatr. 2011;87:225–30., ¹⁶16 Reiter R, Brosch S, Wefel H, Schlömer G, Haase S. The submucous cleft palate: diagnosis and therapy. Int J Pediatr Otorhinolaryngol. 2011;75:85–8., ¹⁷17 Habel A, Elhadi N, Sommerlad B, Powell J. Delayed detection of cleft palate: an audit of newborn examination. Arch Dis Child. 2006;91:238–40.

Parameters	Author (country)
Parameters	Habel et al. (United Kingdom)	Hanny et al. (Netherlands)	Tierney et al. (United Kingdom)	Van Veen-van der Hoek et al. (Netherlands)	Amstalden-Mendes et al. (Brazil)	Reiter et al. (Germany)
Total sample	n = 316	n = 271	n = 17	n = 18	n = 214	n = 439
Age at diagnosis after birth	^– –, data unavailability.	mean age of 502 days	2 days to 7 months of age	Up to 6 months of age	^– –, data unavailability.	mean age of 4.9 years
Time of diagnosis
1st versus 2nd day	n = 96 (28 %) versus n = 70 (20 %)	^– –, data unavailability.	n =0 (0%) versus n =4 (23.5%)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
3rd day to 60th day	n = 48 (15%)	^– –, data unavailability.	n = 12 (70.6 %)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
≤ 14 days versus > 14 days	^– –, data unavailability.	n = 151 (55.7%) versus n = 120 (44.3%)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
≤ 30 days versus > 30 days	^– –, data unavailability.	n = 170 (62.7%) versus n = 101 (37.3%)	n = 14(82.4%) versus n = 3(17.6)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
≤ 1 year versus > 1 year	n = 314(98 %) versus n = 5 (2 %)	n = 204 (75.27 %)versus n = 67 (24.7%)	n = 17 (100 %) versus n =0(0%)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
Type of cleft palate deformity						^ , 68.1 % had a bony notch in the hard palate; 58.9 % had a bifid uvula; 45.1 % had a zona pellucida; and 23.9 % had all 3 signs.
Isolated cleft palate	n = 316(100%)	n = 271 (100%)	n = 75 (27.7%) = 17(100%)	n = 18(100%)	n =45 (21 %)	n = 439(100%)
Submucous cleft palate	^– –, data unavailability.	n = 75 (27.7%)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	n = 439(100%)
Soft palate only	^– –, data unavailability.	n = 63 (23.2 %)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
Soft palate and hard palate	^– –, data unavailability.	n = 133 (49.1 %)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
Nonsyndromic versus syndromic	n = 168 (53.2%) versus n = 148 (46.8%)	n = 145 (53.5%) versus n = 126 (46.5%)	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.
Diagnosis of cleft lip versus isolated cleft palate					^* *, Significant differences between cleft types (p < 0.0001).
Prenatal care	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	n = 30(17.8%) versus n =0(0%)	^– –, data unavailability.
Maternity ward	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	n = 137 (81 %) versus n = 25 (55.6 %)	^– –, data unavailability.
After hospital discharge	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	^– –, data unavailability.	n = 2 (1.2) versus n = 20(44.4%)	^– –, data unavailability.
Additional comments	Main symptoms: feeding problems and nasal regurgitation	Main symptoms: feeding difficulties, hearing and speech/language problems	Mothers had seen a psychologist for issues to do with the late diagnosis	All children underwent neonatal screening, during which the palate was checked by palpation. However, in all cases, the cleft palate was later detected through visual inspection	Physician, nurse, family members, or friends were responsible for disclosing the presence of clefts to the parents	Main symptoms: hypernasality, Eustachian tube dysfunctions, and nasal reflux

[1] * Corresponding author. E-mail: denadai.rafael@hotmail.com (R. Denadai).

Brasil

Brasil

Reducing delayed detection of isolated cleft palate-related deformity: a call for routine intraoral examination of newborns

Abstract

Objective:

Source of data:

Summary of the findings:

Conclusions: