Quality-of-Life in Children with Orofacial Clefts and Caregiver Well-being

Sischo, Lacey; Wilson‐Genderson, Maureen; Broder, Hillary L.

doi:10.1177/0022034517725707

Cited by 37 publications

(41 citation statements)

References 56 publications

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“…Children born with OFCs suffer severe feeding problems, speech difficulties, frequent middle ear infections, and dental defects (Mossey et al, 2009). Long‐term multidisciplinary treatments for these afflictions are heavy medical, psychological, social, and economic burdens on OFC patients and their families (Cassell, Meyer, & Daniels, 2008; Christensen, Juel, Herskind, & Murray, 2004; Marcusson, Akerlind, & Paulin, 2001; Sischo, Wilson‐Genderson, & Broder, 2017; Wehby & Cassell, 2010). Although vertebrate faces are highly diversified, the basic mechanism driving orofacial development is relatively conserved at the cellular and molecular levels (Chai & Maxson Jr., 2006).…”

Section: Introductionmentioning

confidence: 99%

Cellular and developmental basis of orofacial clefts

Garland

Sun

et al. 2020

Birth Defects Research

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During craniofacial development, defective growth and fusion of the upper lip and/or palate can cause orofacial clefts (OFCs), which are among the most common structural birth defects in humans. The developmental basis of OFCs includes morphogenesis of the upper lip, primary palate, secondary palate, and other orofacial structures, each consisting of diverse cell types originating from all three germ layers: the ectoderm, mesoderm, and endoderm. Cranial neural crest cells and orofacial epithelial cells are two major cell types that interact with various cell lineages and play key roles in orofacial development. The cellular basis of OFCs involves defective execution in any one or several of the following processes: neural crest induction, epithelial‐mesenchymal transition, migration, proliferation, differentiation, apoptosis, primary cilia formation and its signaling transduction, epithelial seam formation and disappearance, periderm formation and peeling, convergence and extrusion of palatal epithelial seam cells, cell adhesion, cytoskeleton dynamics, and extracellular matrix function. The latest cellular and developmental findings may provide a basis for better understanding of the underlying genetic, epigenetic, environmental, and molecular mechanisms of OFCs.

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Section: Introductionmentioning

confidence: 99%

Cellular and developmental basis of orofacial clefts

Garland

Sun

et al. 2020

Birth Defects Research

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“…It had been shown in the literature that neither self-administration nor accompaniment by an investigator affect the results of questionnaires [34]. Thus, the fact that children responded with their parents, when they were not able to do it alone, did not limit the correct interpretation of the answers [35, 36].…”

Section: Discussionmentioning

confidence: 99%

Oral health related quality of life of children and adolescents affected by rare orofacial diseases: a questionnaire-based cohort study

Friedlander¹,

Berdal²,

Boizeau³

et al. 2019

Orphanet J Rare Dis

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Background Rare diseases affecting the teeth, the oral cavity and the face are numerous, each of them present specific characteristics, and is a life-long condition. The aim of the study was to assess the association between Oral health-related quality of life (OHRQoL), and demographic characteristics, clinical and dental factors, and psycho-social characteristics to investigate that oral symptoms are not the main factors underlying a decrease in OHRQoL. Material and methods We conducted a national cohort study in French centres for rare diseases (RD) specialized in orofacial diseases. The inclusion criteria were: to have received care in RD centres over the last 5 years (2012–2017) and to have been between 6 and 17 years of age on September 1, 2017. Patients were invited to answer a questionnaire composed of socio-demographic, clinical and dental questions, psychosocial questions and then fill in the Child-OIDP Index. At the end of the questionnaire, a free space was left for the patient to add a verbatim comment to provide qualitative data. Thematic analysis was used to analyze the verbatim answers. Results Complete data were available for 110 patients. The sample included 44.5% boys and 55.5% girls. Ages ranged from 6 to 17 years old and 68.2% were between 6 to 12 years old and 31.8% were between 13 and 17 years old. Factor associated with a lower OHRQoL were: being a girl ( p = 0.03), renouncement to dental care for financial reasons ( p = 0.01), having syndromic disease (p = 0.01), having a problem with tooth shape and color (p = 0.03), feeling isolated, alone and different from other children ( p = 0.003 and p = 0.02). Qualitative analysis highlighted very little recourse to psychological care and patients reported great anxiety and fear about the future. Conclusion OHRQoL of children suffering from these diseases is impaired, especially from the psychosocial point of view but also from that of the course of treatment and access to care. There is a need to improve the legibility of care pathways and the financial coverage of treatments. Electronic supplementary material The online version of this article (10.1186/s13023-019-1109-2) contains supplementary material, which is available to authorized users.

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“…Recent studies show that reports of children on OHRQoL are reliable and valid. Instruments developed to measure the quality of life of children must also assess the impact of these problems on the quality of life of the family, as they are inseparable factors [13][14][15][16].…”

Section: Quality Of Life Instrument Evaluationmentioning

confidence: 99%

Quality of Life in Adolescents with Cleft Lip and Palate

Elmouden¹,

Elgasmi²,

Ousehal³

2020

Current Treatment of Cleft Lip and Palate

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The cleft lip and palate (CLP) is the second most common congenital anomaly in the world; therefore, understanding the quality of life in children and adolescents with this malformation is extremely important. The study of the quality of life in this population is based on how the patient feels about his condition and how it affects his general well-being depending on the problems he may encounter concerning bone growth, phonation, speaking, facial appearance, and social relationship. As part of a descriptive survey, the aim was to assess and study the quality of life of patients with labio-palatal clefts operated and undergoing medical management, as well as the quality of life perceived by their parents to evaluate the family impact of this dysmorphia. According to the studies, teenagers had the lowest quality of life rates in the items: physical appearance and self-esteem, so the psychological support for patients and their families with multidisciplinary treatment that meets standards, are the keys to improving the quality of life of this category of patients.

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Quality-of-Life in Children with Orofacial Clefts and Caregiver Well-being

Cited by 37 publications

References 56 publications

Cellular and developmental basis of orofacial clefts

Cellular and developmental basis of orofacial clefts

Oral health related quality of life of children and adolescents affected by rare orofacial diseases: a questionnaire-based cohort study

Quality of Life in Adolescents with Cleft Lip and Palate

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