Blastopathies and microcephaly in a Chornobyl impacted regionof Ukraine
INVITED REVIEW ARTICLE: September 2014
Blastopathies and microcephaly in a Chornobyl impacted region of Ukraine
Wladimir Wertelecki1,2, Lyubov Yevtushok2,3, Natalia Zymak-Zakutnia2,4, Bin Wang5, Zoriana Sosyniuk2,3, Serhiy Lapchenko2, and Holly H. Hobart6
1Department of Medical Genetics, College of Medicine, and 5Department of Mathematics and Statisitcs, University of South Alabama, Mobile, Alabama, USA; 2OMNI-Net for Children International Charitable Fund, 3Rivne Regional Medical Diagnostic Center, Rivne, Rivne Province, 4Khmelnytsky Perinatal Center, Khmelnytsky, Khmelnytsky Province, Ukraine; and 6Cyto-Genetics Laboratory, Department of Pathology, University of Mississippi Medical Center, Jackson, Mississippi, USA
ABSTRACT This population-based descriptive epidemiol- ogy study demonstrates that rates of conjoined twins, teratomas, neural tube defects, microcephaly, and microphthalmia in the Rivne province of Ukraine are among the highest in Europe. The province is 200 km distant from the Chornobyl site and its northern half, a region known as Polissia, is significantly pol- luted by ionizing radiation. The rates of neural tube defects, microcephaly and microphthalmia in Polissia are statistically significantly higher than in the rest of the province. A survey of at-birth head size showed that values were statistically smaller in males and females born in one Polissia county than among neonates born in the capital city. These observations provide clues for confirmatory and cause-effect prospective investi- gations. The strength of this study stems from a reliance on international standards prevalent in Europe and a decade-long population-based surveillance of congenital malformations in two distinct large populations. The limitations of this study, as those of other descriptive epidemiology investigations, is that identified cause-effect associations require further assessment by specific prospective investigations designed to address specific teratogenic factors.
Key Words: blastopathies, Chornobyl, congenital malformations, ionizing radiation, sex ratio
The 1986 Chornobyl disaster in Ukraine (Chernobyl in Russian) is among the largest man-caused disasters and has impacted and con- tinues to impact human health, ecologic integrity, and the social welfare of multiple generations of culturally and ethnically diverse large populations. Some have called the Chornobyl tragedy a “natural experiment” and as pointed out by Garruto et al. (1999) such events represent unique opportunities for studying biomedical processes, disease etiology, and pathogenesis in populations with diverse ethnic and genetic structures, living under special circum- stances. This report summarizes the results of an investigation aimed at determining population-based rates and patterns of congenital malformations (CM) in the Rivne province of Ukraine during the 2000–2009 decade. Although two concurrent identi- cal CM population-based surveillance programs are ongoing in Khmelnytsky and Volyn provinces, which adjoin Rivne, this report is focused on observations in the latter province. The term “blastopathies” appears in the title of this report to stress that the CM reported are present prior to the embryonic implantation and organogenesis, a notion elaborated upon later. To our knowledge, there are no other population-based long term investigations of CM rates and patterns relying on international methods, focusing on an area relatively proximal and heavily impacted by ionizing radia- tion (IR) from the 1986 Chornobyl disaster (Fig. 1). The northern half of the Rivne province is a region of forested wetlands known as Polissia, which is inhabited by a native population known as Polishchuks. Coincidentally, the fallout of Chornobyl IR impacted mostly the Rivne-Polissia zone henceforth referred to as Rivne-P or simply Polissia (note that there are also Polissia regions in Volyn, Zhytomyr and Kyiv provinces). The non-Polissia regions hence- forth referred to as Rivne-nP were less impacted by Chornobyl IR. Noteworthy is that reports on the subject rarely point out contrasts between Rivne-P and Rivne-nP regions (Likhtarev et al. 1996, 2000; Zamostian et al. 2002). Polissia may be referred to as the Prypiat Marshlands or in older medical literature as Polisie, Poliesia, Polessky, and Polesie.
In this report, unless indicated otherwise, Polissia solely refers to Rivne-P, which is described in some detail in the Data Supplement. It is sufficient to underscore here that the native people of Rivne-P are known as Polishchuks and that they represent a population isolate surviving mostly by consumption of locally grown products, foods and fuels inherently contaminated by nuclides. Polishchuks continue to inhale and ingest nuclides and among whom, a growing proportion of individuals are exposed since birth. Furthermore, a growing proportion of pregnant Polishchuk women have them- selves incorporated nuclides to which all of their conceived children are exposed prenatally. The large size and well defined nature of the Polishchuk population facilitates long term studies of the health and teratogenic impacts of protracted exposures to low levels of IR.
In this report, we confirm and expand previous studies in Rivne that demonstrated elevated population-based rates of CM and include initial results of a series of surveys that may reflect impacts of IR among other causes (Yuskiv et al. 2004; Wertelecki 2010). To sustain in Rivne and two adjoining provinces (Volyn and Khmelnytsky) ongoing population-based CM monitoring systems, which uphold international standards and international partnership, we established OMNI-Net, a not for profit international organization registered in Kyiv, Ukraine (Wertelecki 2006). This report primarily concerns observations in Rivne, which are occasionally expanded by observations in the two adjoining provinces. Among the goals of the OMNI-Net is to promote and maintain international research and humanitarian partnerships intended to define causes, and to promote treatments to minimize and prevent CM. In Rivne, the OMNI-Net center is co-located with the Clinical Genetics and Prenatal Fetal Ultrasonography Services of the Provincial Diagnostic Center of the Rivne province, henceforth referred to as OMNI-Net or Diagnostic Center. Our confidence in the significance of the observations we report not only rests in the confirmation of two previous analyses but also on a concurrence of favorable circumstances in Rivne that foster and sustain the ongoing population-based CM surveillance process integrated with clinical services and public health programs.
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© 2014 The Authors. Congenital Anomalies published by Wiley Publishing Asia Pty Ltd on behalf of Japanese Teratology Society.
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