An assessment of orofacial clefts in Tanzania

Manyama, Mange; Rolian, Campbell; Gilyoma, Japhet M; Magori, Cassian; Mjema, Kilalo; Mazyala, Erick; Kimwaga, Emmanuel; Hallgrímsson, Benedikt

doi:10.1186/1472-6831-11-5

Cited by 43 publications

(62 citation statements)

References 35 publications

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“…[11] Both the syndromic and asyndromal forms of CL/P may have genetic basis, as according to Stainer and Moore; [12] Ghassibe, et al, [13] several causative genes for the syndromic forms have been identified, and there is evidence that these genes also contribute to the occurrence of isolated (asyndromal) forms. However, some syndromal cases of CL/P may be non chromosomal in etiology.…”

Section: Discussionmentioning

confidence: 97%

“…In previous Nigerian and Tanzanian reports, CL/P occurring in association with other anomalies such as Van der Woude's syndrome, Down's syndrome and congenital heart defects, neural tube defects, Talipes and persistent ductus arteriosus have been recorded. [3,4,11] Zimpfer, et al, [15] reported on a male fetus with bilateral brachial Amelia, complex facial clefts, frontal craniosynostosis and hypoplasia, thoracic kyphoscoliosis, and holoprosencephaly. In addition, an interrupted aortic arch, a muscular ventricular septal defect, and a localized non-compaction of the septal myocardium were present.…”

Section: Discussionmentioning

confidence: 97%

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Facial cleft with Amelia: A Nigerian case report

Benjamin

Adebayo²,

Mohammed

et al. 2013

Ann Afr Med

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Clefts of the lip and palate are the most common serious congenital anomalies in orofacial region. However, the occurrence of associated defects in the limbs is quite rare. This report presents a 12-week-old male child born with unilateral left cleft lip and palate, absence of the upper left arm and forearm, and fusion of both big and middle toes and absence of the entire small toe on the left foot. The repairs of lip and palate defects were done at 6 and 19 months of age while parents were referred for rehabilitation to ameliorate absence of upper left arm. We discuss the challenges of management of defects in a developing economy, issues of acceptance of children born with defects amenable to surgery and the benefit of parental education on the care of the child.

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Section: Discussionmentioning

confidence: 97%

Section: Discussionmentioning

confidence: 97%

Facial cleft with Amelia: A Nigerian case report

Benjamin

Adebayo²,

Mohammed

et al. 2013

Ann Afr Med

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“…Several studies have addressed facial shape variation associated with CL/P in Caucasian populations [6,7,15,16], in which the prevalence of clefts is 0.91 to 2.69 per 1000 live births [17,18]. However, there have been no reported studies of facial shape in individuals with CL/P in African populations, in which the prevalence of CL/P is considerably lower, 0.3 per 1,000 to 1.65 per 1,000 live births [19,20], and the proportions of cleft types is also somewhat different [21], perhaps reflecting different underlying genetic and environmental aetiologies.…”

Section: Introductionmentioning

confidence: 99%

Facial morphometrics of children with non-syndromic orofacial clefts in Tanzania

et al. 2014

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BackgroundOrofacial clefts (cleft lip/palate; CL/P) are among the most common congenital anomalies, with prevalence that varies among different ethnic groups. Craniofacial shape differences between individuals with CL/P and healthy controls have been widely reported in non-African populations. Knowledge of craniofacial shape among individuals with non-syndromic CL/P in African populations will provide further understanding of the ethnic and phenotypic variation present in non-syndromic orofacial clefts.MethodsA descriptive cross-sectional study was carried out at Bugando Medical Centre, Tanzania, comparing individuals with unrepaired non-syndromic CL/P and normal individuals without orofacial clefts. Three-dimensional (3D) facial surfaces were captured using a non-invasive 3D camera. The corresponding 3D coordinates for 26 soft tissue landmarks were used to characterize facial shape. Facial shape variation within and between groups, based on Procrustes superimposed data, was studied using geometric morphometric methods.ResultsFacial shape of children with cleft lip differed significantly from the control group, beyond the cleft itself. The CL/P group exhibited increased nasal and mouth width, increased interorbital distance, and more prognathic premaxillary region. Within the CL/P group, PCA showed that facial shape variation is associated with facial height, nasal cavity width, interorbital distance and midfacial prognathism. The isolated cleft lip (CL) and combined cleft lip and palate (CLP) groups did not differ significantly from one another (Procrustes distance = 0.0416, p = 0.50). Procrustes distance permutation tests within the CL/P group showed a significant shape difference between unilateral clefts and bilateral clefts (Procrustes distance = 0.0728, p = 0.0001). Our findings indicate the morphological variation is similar to those of studies of CL/P patients and their unaffected close relatives in non-African populations.ConclusionThe mean facial shape in African children with non-syndromic CL/P differs significantly from children without orofacial clefts. The main differences involve interorbital width, facial width and midface prognathism. The axes of facial shape differences we observed are similar to the patterns seen in Caucasian populations, despite apparent differences in cleft prevalence and cleft type distribution. Similar facial morphology in individuals with CL/P in African and Caucasian populations suggests a similar aetiology.

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“…8 BCLP is seen less commonly than unilateral cleft lip and palate. 9,10 Few studies pertain to BCLP, and changes in upper-airway dimensions in subjects with BCLP have not been investigated comprehensively. Studies on airways have focused on either unilateral [11][12][13] or mixed samples of patients with cleft lip and palate [14][15][16] or adult BCLP patients.…”

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confidence: 99%