Evaluation of the reproductive and developmental safety of cysteamine in the rat: Effects on female reproduction and early embryonic development

Assadi, Farahnak; Mullin, J.J.; Beckman, David A.

doi:10.1002/(sici)1096-9926(199809/10)58:3/4<88::aid-tera4>3.0.co;2-5

Cited by 9 publications

(1 citation statement)

References 24 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…In accordance, in a previous study investigating the effect of oral cysteamine on female reproduction in wild-type rats, the authors showed that cysteamine had no adverse effects on conception and early embryonic development [ 49 ]. Hence, it is believed that cysteamine would not have a negative impact on reproduction in both male and female cystinosis patients.…”

Section: Effect Of Cysteamine On Fertilitysupporting

confidence: 70%

Fertility in Cystinosis

Reda

Veys

Besouw

2021

Cells

View full text Add to dashboard Cite

Cystinosis is a rare inheritable lysosomal storage disorder characterized by cystine accumulation throughout the body, chronic kidney disease necessitating renal replacement therapy mostly during adolescence, and multiple extra-renal complications. The majority of male cystinosis patients are infertile due to azoospermia, in contrast to female patients who are fertile. Over recent decades, the fertility status of male patients has evolved from a primary hypogonadism in the era before the systematic treatment with cysteamine to azoospermia in the majority of cysteamine-treated infantile cystinosis patients. In this review, we provide a state-of-the-art overview on the available clinical, histopathological, animal, and in vitro data. We summarize current insights on both cystinosis males and females, and their clinical implications including the potential effect of cysteamine on fertility. In addition, we identify the remaining challenges and areas for future research.

show abstract

Section: Effect Of Cysteamine On Fertilitysupporting

confidence: 70%

Fertility in Cystinosis

Reda

Veys

Besouw

2021

Cells

View full text Add to dashboard Cite

show abstract

Developmental toxicity of cysteamine in the rat: Effects on embryo–fetal development

1998

View full text Add to dashboard Cite

Pregnancy in cystinosis patients with chronic kidney disease: A European case series

Janssen

Blakey

Greco

et al. 2022

J of Inher Metab Disea

View full text Add to dashboard Cite

Cystinosis is a rare autosomal recessive disease leading to end-stage renal disease within the second or third decade of life. Since the era of specific treatment with cysteamine, prognosis has substantially improved and pregnancy becomes an increasing concern. Pregnancy data in patients with cystinosis were collected through an anonymized survey. We collected data for 19 pregnancies in 12 women. Seventeen patients were transplanted, 1 was on hemodialysis and 1 had chronic kidney disease (CKD) stage 4. These 19 pregnancies resulted in 13 live births (68.4%): 3 spontaneous early miscarriages, 1 ectopic pregnancy, 1 early pre-eclampsia (at 21 weeks), and 1 preterm birth with neonatal death at 24 weeks were reported. After exclusion of early miscarriage or termination, pregnancy success rate was 86.7%. In successful pregnancies, median gestational age at delivery was 34 weeks (24-37). Preeclampsia occurred in seven pregnancies (7/15, 46.7%). A cesarean section was performed in all pregnancies. Median baby weight at delivery was 2175 g (620-3374 g). After pregnancy, one patient reached end-stage renal disease, but she already had advanced CKD before pregnancy (creatinine 239 μmol/L, eGFR 23 ml/min/1.73 m 2 ). In three other patients, there was a decrease of eGFR of 8, 20, and 53 ml/min/1.73 m 2 , respectively. The majority of pregnancies were

show abstract

Evaluation of the reproductive and developmental safety of cysteamine in the rat: Effects on female reproduction and early embryonic development

Cited by 9 publications

References 24 publications

Fertility in Cystinosis

Fertility in Cystinosis

Developmental toxicity of cysteamine in the rat: Effects on embryo–fetal development

Pregnancy in cystinosis patients with chronic kidney disease: A European case series

Contact Info

Product

Resources

About