The role of a parental history of Balkan endemic nephropathy in the occurrence of BEN: a prospective study

Hanjangsit, Kesinee; Karmaus, Wilfried; Dimitrov, Plamen; Zhang, Hongmei; Burch, Jim; Tzolova, Svetla; Batuman, Vecihi

doi:10.2147/ijnrd.s30615

Cited by 6 publications

(5 citation statements)

References 24 publications

(30 reference statements)

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“…94 In addition, parental history of BEN had direct and indirect effects on offspring, which also predicted the onset of BEN. 95 and genetic predisposition may also interact with environmental determinants to produce the associated tumors. 20 Thus, there is likely an underlying genetic condition that predisposes individuals to susceptibility by another agent to cause BEN.…”

Section: 33mentioning

confidence: 99%

Limitations and plausibility of the Pliocene lignite hypothesis in explaining the etiology of Balkan endemic nephropathy

Maharaj¹

2013

International Journal of Occupational and Environmental Health

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Background: Balkan endemic nephropathy (BEN) is a chronic, tubulointerstitial renal disease often accompanied by urothelial cancer that has a lethality of nearly 100%. Introduction: One of the many factors that have been proposed to play an etiological role in BEN is exposure to organic compounds from Pliocene lignite coal deposits via the drinking water in endemic areas. Objectives: The objective of this study was to systematically evaluate the role of the tenets of the Pliocene lignite hypothesis in the etiology of BEN in order to provide an improved understanding of the hypothesis for colleagues and patients alike. Methods: A comprehensive compilation of the possible limitations of the hypothesis, with each limitation addressed in turn is presented. Results: The Pliocene lignite hypothesis can best account for, is consistent with, or has the potential to explain the evidence associated with the myriad of factors related to BEN. Conclusions: Residents of endemic areas are exposed to complex mixtures containing hundreds of organic compounds at varying doses and their potentially more toxic (including nephrotoxic) and/or carcinogenic metabolites; however, a multifactorial etiology of BEN appears most likely.

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Section: 33mentioning

confidence: 99%

Limitations and plausibility of the Pliocene lignite hypothesis in explaining the etiology of Balkan endemic nephropathy

Maharaj¹

2013

International Journal of Occupational and Environmental Health

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“…In the offspring of a mother with BEN, ultrasound measurements of the kidney cortex thickness seem to portend a prognostic value [ 19 ]. In conclusion, adult offspring in BEN families can be characterized by shorter kidney length and an increased excretion of albumin, total protein and β2-microglobulin, in particular, when the mother had BEN [ 19 , 20 ].…”

Section: Introductionmentioning

confidence: 99%

Balkan endemic nephropathy--current status and future perspectives

Pavlović¹

2013

Clinical Kidney Journal

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Balkan endemic nephropathy (BEN), originally described in 1956, is a unique familial, chronic renal disease encountered with a high-prevalence rate in Serbia, Bulgaria, Romania, Croatia and Bosnia and Herzegovina. The most prominent features of the disease are its endemic nature, long-incubation period, familial clustering of the disease and an unusually high incidence of associated upper urothelial cancer (UUC). There are no clear-cut data on BEN incidence and prevalence, since the studies carried out in different endemic areas yielded contradictory information. In spite of intermittent variations, the incidence of new cases has remained stable over time. It has been estimated that almost 100 000 people are at risk of BEN, whereas 25 000 have the disease. The clinical signs and symptoms of BEN are non-specific and often remain unrecognized for years. There are no pathognomonic diagnostic features of BEN, but the set of epidemiological, clinical and biochemical data along with the pattern of pathologic injury in the absence of any other renal diseases are highly suggestive of this entity. Although the aetiology has been extensively studied, fostering the publication of various hypotheses, only one of them has provided conclusive evidence related to the aetiology of BEN. Studies conducted over the past decade have provided particularly strong arguments that BEN and UUC are caused by chronic poisoning with aristolochic acids (AAs). In light of these later studies, one can raise the question whether AAs could be responsible for previously and currently widespread unrecognized global renal disease and UUC.

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“…On the other hand, it is well known that cadmium, a natural element and a toxic metal found in cigarettes, exhibits nephrotoxic properties [36]. Finally, the potential role of smoking as a risk factor for the development of BEN has also been described [37].…”

Section: Discussionmentioning

confidence: 99%

Assessment of Retinal Microangiopathy in Patients with Balkan Endemic Nephropathy Using Optical Coherence Tomography Angiography—A Pilot Study

Djordjevic-Jocic,

Kokoris,

Mitic

et al. 2024

Medicina

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Background and Objectives: It is well known that alterations in microvascular structure and function contribute to the development of ocular, renal, and cardiovascular diseases. Accordingly, the presence of fundus vascular changes in patients suffering from chronic kidney disease (CKD) and Balkan endemic nephropathy (BEN) may provide information of prognostic value regarding the progression of renal disease. This study aimed to examine the associations between clinical characteristics and retinal optical coherence tomography angiography (OCTA) parameters in patients with BEN and compare them with those in CKD. Materials and Methods: This pilot study, conducted from March 2021 to April 2022, included 63 patients who were divided into two groups: the first group consisted of 29 patients suffering from BEN, and the second was a control group of 34 patients with CKD. Demographic, laboratory, clinical, and medication data were noted for all the patients included in this study. Each eye underwent OCT angiography, and the results were interpreted in accordance with the practical guide for the interpretation of OCTA findings. Results: Statistically significantly higher levels of total serum protein and triglycerides were recorded in the BEN group than in the CKD group, while the level of HDL cholesterol was lower. Based on the performed urinalysis, statistically significantly higher values of total protein and creatinine were detected in patients with CKD compared to the BEN group. It was demonstrated that the OCTA vascular plexus density of certain parts of the retina was in significant association with systolic and diastolic blood pressure, creatinine clearance, urinary creatinine, total cholesterol, diabetes mellitus type 2, age, body mass index, total serum and urinary protein, sCRP, and diuretic and antihypertensive treatment. Conclusions: In comparison with CKD, BEN leads to more significant disturbances in retinal vasculature density.

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The role of a parental history of Balkan endemic nephropathy in the occurrence of BEN: a prospective study

Cited by 6 publications

References 24 publications

Limitations and plausibility of the Pliocene lignite hypothesis in explaining the etiology of Balkan endemic nephropathy

Limitations and plausibility of the Pliocene lignite hypothesis in explaining the etiology of Balkan endemic nephropathy

Balkan endemic nephropathy--current status and future perspectives

Assessment of Retinal Microangiopathy in Patients with Balkan Endemic Nephropathy Using Optical Coherence Tomography Angiography—A Pilot Study

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