Cleft-Related Infanticide and Abandonment: A Systematic Review of the Academic and Lay Literature.

OBJECTIVE: We aimed to describe the scope of cleft-related infanticide and identify issues that might inform prevention strategies. DESIGN: Systematic reviews of both academic (eg, PubMed, EBSCOhost) and lay literature (eg, LexisNexis Academic, Google) databases were performed to identify all primary reports of cleft-related infanticide. All languages were included. Records before 1985 were excluded. Reference lists of all included reports were screened for potentially relevant records. MAIN OUTCOME MEASURES: Country of origin and excerpts that pertained to the concepts surrounding cleft-related infanticide were extracted. Extracted excerpts were examined using a content analysis framework. RESULTS: Of the 1,151 records retrieved, 70 reports documented cleft-related infanticide from 27 countries. The largest number of reports was from China (14 reports; 48% of reports), followed by India (4; 14%) and Nigeria (4; 14%). However, 2 countries had 3 reports, 5 countries had 2 reports, and 17 countries had 1 report. Themes that emerged from excerpt analysis included stigma, lack of affordable cleft care, abandonment, orphanage overcrowding, and abuse and slavery. CONCLUSIONS: Cleft-related infanticide is a global problem. Initiatives to sensitize communities to cleft lip and/or cleft palate, provide timely and affordable cleft care, and build support systems for affected families may prove beneficial. Cleft care organizations have the opportunity to advocate for these initiatives, reduce the incidence of infanticide by providing or supporting timely and affordable cleft care, and demonstrate that children with successful cleft repairs reassimilate well into their communities.

Estimate of Unmet Need for Cleft Lip and/or Palate Surgery in India.

IMPORTANCE: The unmet need for cleft lip and/or palate (CL/P) care in India is significant. However, estimates required for CL/P care program planning are lacking. OBJECTIVE: To estimate the unmet need for CL/P surgery in India at the state level. DESIGN, SETTING, AND PARTICIPANTS: To determine the proportion of individuals with CL/P who presented for care in India, data were used from patients who received care at Operation Smile programs in 12 low- and middle-income countries from June 1, 2013, to May 31, 2014. The resulting model describes the prevalent unmet need for cleft surgery in India by state and includes patients older than the surgery target ages of 1 and 2 years for cleft lip and cleft palate repair, respectively. Next, the total number of unrepaired CL/P cases in each state was estimated using state-level economic and health system indicators. MAIN OUTCOMES AND MEASURES: Prevalent unmet need for CL/P repair. RESULTS: In the 28 states with available data, an estimated 72 637 cases of unrepaired CL/P (uncertainty interval, 58 644-97 870 cases) were detected. The percentage of individuals with unrepaired CL/P who were older than the respective target ages ranged from 37.0% (95% CI, 30.6%-43.8%) in Goa to 65.8% (95% CI, 60.3%-70.9%) in Bihar (median, 57.9%; interquartile range, 52.6%-63.4%). The rate of unrepaired CL/Ps ranged from less than 3.5 per 100 000 population in Kerala and Goa to 10.9 per 100 000 population in Bihar (median rate, 5.9 [interquartile range, 4.6-7.3] per 100 000 population). CONCLUSIONS AND RELEVANCE: An estimated 72 000 cases of unrepaired CL/P are found in India. Poor states with less health care infrastructure have exceptionally high rates (eg, Bihar). These estimates are useful for informing international and national CL/P care strategies, allocating resources, and advocating for individuals and families affected by CL/P more broadly. LEVEL OF EVIDENCE: NA.

A Health Systems Perspective on the Mission Model for Cleft Lip and Palate Surgery: A Matter of Sustainability or Responsibility?

One in 700 children around the world are born with cleft lip and/or palate (CL/P). Although reconstructive surgery is widely available in high-income settings, over 2 billion people in low- and middle-income countries lack access to essential surgical care. The mission model has been demonstrated to be highly effective in responding to the global surgical workforce crisis, but has been questioned in regard to its sustainability, value, and overall impact. Through effective health systems integration, the mission model presents abundant opportunities for streamlined delivery and horizontal impact. Still, the primary goal of the mission model is direct care delivery; and although the value of sustainability is indisputably vital, we contend that the mission model, when executed responsibly, creates high-value, sustained impact on the individual lives of those presently in need. We furthermore advocate for the sustained commitment of implementing organizations, patient safety, local integration, and a new focus on patient centeredness as key elements of the responsible mission model.

Elevated infant mortality rates among oral cleft and isolated oral cleft cases: a meta-analysis of studies from 1943 to 2010.

Objective : To review available published literature in order to obtain a more comprehensive assessment of the association between infant mortality and oral clefts, overall and isolated. Design : A wide-ranging search of published studies relating infant mortality rates to oral clefts and congenital malformations was conducted, yielding applicable data sets. Settings of the included studies varied to a limited extent, although all were conducted within high-income countries with superior health indicators. These results were tabulated and meta-analyzed. Random effects odds ratios comparing each data set with its respective population have been used to account for differing settings and specific infant mortality rates. Results : Nine articles with relatable data were included in these meta-analyses. The calculated odds ratio of infant mortality associated with oral cleft cases, including those with associated malformations, was substantial: 9.466 (95% confidence interval, 6.153 to 14.560). Excluding oral cleft cases with associated malformations, the odds ratio, although lower, was still significant: 2.073 (95% confidence interval, 1.390 to 3.092). Conclusions : Compared with the overall population, infants with oral clefts have nine times the odds of dying within the first year of life. Furthermore, even without additional malformations, infants with oral clefts are still twice as likely to die before 1 year of age. Current research points to a substantially higher relative risk of infant mortality among oral cleft cases in developing countries. Additional research is essential to determine the sources of these raised infant mortality rates and possible interventions to decrease them.

Cleft lip and palate as a cost-effective health care treatment in the developing world.

BACKGROUND: The considerable number of surgical procedures performed each year, as well as the significant burden of surgical disease globally, have brought surgery into the arena of public health concerns. Several articles have shown the cost-effectiveness of surgical interventions in the developing world. The authors suggest that surgery has an important role in global public health, specifically through international volunteer surgical missions as a vehicle for delivery. METHODS: Eight Operation Smile medical missions from 2008 are analyzed using the disability-adjusted life year (DALY) framework and disability weights from the life tables of the first Disease Control Priorities Project (DCP1). The DCP1 life tables suggest that the entire burden of disease from a cleft lip and palate is incurred within the first 4 years of life. The same group of missions is analyzed using life tables modified by the authors to reflect the disability associated with cleft lip and palate that goes beyond the first 5 years of life. RESULTS: Using the DCP1 life tables, the cost per patient during the eight Operation Smile missions analyzed was $278 and $1827 with an average of $796. Using the life tables modified by the authors, the cost per patient was between $7.36 and $96.04 (average $33.94). CONCLUSIONS: The absence of disability weights for untreated cleft lip and palate after the fifth year of life does not fit with the reality of living with an unrepaired cleft lip or palate. The authors conclude that secondary interventions alleviate disability and should enable an individual to approach a zero residual disability weight.