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Reproductive function of long-term treated patients with hepatic onset of Wilson's disease: a prospective study
Reprod Biomed Online. 2021 Apr;42(4):835-841. doi: 10.1016/j.rbmo.2020.12.012.Epub 2020 Dec 27.
Giuseppe Gabriele Iorio1, Alessandro Conforti2, Roberta Vallone2, Luigi Carbone2, Margherita Matarazzo3, Anna De Rosa2, Pasquale De Rosa2, Silvia Picarelli2, Flora Fedele3, Giuseppe Perruolo3, Pietro Formisano3, Raffaele Iorio3, Carlo Alviggi2, Fabiola Di Dato3
1Department of Neuroscience, Reproductive Science and Odontostomatology, School of Medicine and Surgery, University of Naples Federico II, Naples, Italy. Electronic address: email@example.com.
2Department of Neuroscience, Reproductive Science and Odontostomatology, School of Medicine and Surgery, University of Naples Federico II, Naples, Italy.
3Department di Translational Medical Science, School of Medicine and Surgery, University of Naples Federico II, Naples, Italy.
Research question: Wilson's disease (WD) is a disorder of copper metabolism that can cause hormonal alterations. The impact of WD and its therapies on fertility is not well defined. The aim of this study was to evaluate ovarian reserve and sperm parameters in long-term treated WD patients with hepatic onset.
Design: WD patients with hepatic onset treated for at least 5 years were compared with healthy controls. Men underwent spermiogram and sperm DNA fragmentation (SDF) analysis. Women were tested for serum FSH, anti-Müllerian hormone (AMH) and sonographic antral follicle count (AFC) in the early follicular phase. Ovulation was monitored with ultrasound and progesterone serum concentrations in the luteal phase.
Results: The WD group included 26 patients (12 males), the control group 19 subjects (9 males). All patients apart from four (one male) were responders to WD treatment. Sperm count and morphology were comparable between cases and controls. Sperm motility (total and after 1 h) was significantly lower in cases (44.78 ± 21.65%; 47.85 ± 21.52%) than controls (61.88 ± 11.03; 69.44 ± 11.02%, P = 0.03 and 0.01, respectively). The only non-responder had severe oligo-astheno-teratozoospermia. SDF values were normal in cases and controls. AMH, AFC and FSH did not differ between cases and controls. LH was significantly lower in cases (3.36 ± 1.65 mIU/ml) than controls (6.25 ± 1.03 mIU/ml, P < 0.0001). A non-responder woman who developed neurological signs had a 7-year history of infertility.
Conclusions: WD patients with hepatic onset, diagnosed early and treated, have no impairment in fertility potential even if males show reduced sperm motility and females lower LH values. Only patients with poor disease control have some evidence of impaired fertility.