Fowler's syndrome: cause or consequence of insulin resistance?

Abstract

A 26-year-old woman with polycystic ovarian syndrome (PCOS) presented with recurrent urinary retention. A neurological examination, cystoscopy and upper tract imaging were normal. EMG of the urethral sphincter revealed a typical trace for Fowler's syndrome. She was not suitable for sacral neuromodulation because of her physique and high body mass index, so CISC was commenced. Further investigations showed the metabolic form of PCOS, with insulin resistance, compensatory hyperinsulinaemia and impaired glucose tolerance. Oral metformin (an insulin-sensitizing agent) was commenced, with a dramatic reduction in weight and return of spontaneous voiding. CommentFowler's syndrome comprises abnormal EMG activity of the urethral sphincter, voiding dysfunction and PCOS [1]. The abnormal EMG activity consists of an ephaptic spread of excitation between muscle ®bres, rather than from terminal branches of the motor axon. This leads to complex repetitive discharges and decelerating bursts, preventing a complete relaxation of the urethral sphincter. Until recently, the only effective management was CISC; sacral neuromodulation has shown promise [2]. In the present patient with Fowler's syndrome, treating the insulin resistance had direct (return of spontaneous voiding) and indirect bene®ts, including weight loss, correction of abnormal triglyceride/lipoprotein metabolism and the resultant reduction in cardiovascular risk factors [3]. A randomized controlled trial would con®rm the therapeutic potential of screening for and treating the insulin resistance in Fowler's syndrome. We recommend an endocrinological opinion in this unusual patient group.

Functional obstructed voiding in the neurologically normal patient

“…This is a complex condition of chronic anovulation associated with hyperandrogenism and hyperinsulinemia [15]. There is one case report of the successful treatment of dysfunctional voiding in an obese woman with an insulin-sensitizing agent [16]. There also are recent data suggesting that patients with Fowler's syndrome may have an underlying dysautonomia [17].…”