Stronger evidence for small fiber sensory neuropathy in restless genital syndrome: two case reports in males.
BACKGROUND
Restless genital syndrome (ReGS) is characterized by unwanted, unpleasant genital sensations, restless legs, and/or overactive bladder, as well as neuropathy of the dorsal nerve of the clitoris. So far, ReGS has only been reported in females.


OBJECTIVE
To report the occurrence of ReGS in two males.


METHODS
Two males with unwanted genital sensations presented in our clinic. In-depth interview, routine and hormonal investigations, electro-encephalography, magnetic resonance imaging of brain and pelvis, manual examination of the pubic bone, and sensory testing of genital dermatomes were performed. In both males, conventional transcutaneous electrical nerve stimulation was applied bilaterally at the pudendal dermatome.


METHODS
Oral report, questionnaire on frequency and intensity of restless genital feelings, restless legs, overactive bladder, and satisfaction with the transcutaneous electrical nerve stimulation (TENS) treatment.


RESULTS
ReGS in a 74-year-old male manifested as unpleasant genital sensations of being on the edge of an orgasm with overactive bladder, in the absence of erection and ejaculation. Genital sensory testing elicited bilateral points of static mechanical hyperesthesia in the pudendal dermatome. Manual examination of the dorsal nerve of the penis (DNP) elicited the genital sensations. TENS application resulted in a 90% reduction of genital sensations and complaints of overactive bladder syndrome (OAB). ReGS in a 38-year-old male manifested as unwanted and unpleasant spontaneous ejaculations and complaints of OAB. Genital sensory testing elicited bilateral points of static mechanical hyperesthesia in the pudendal dermatome. Manual examination of the DNP elicited the genital sensations. TENS application had no effect on genital complaints and complaints of OAB.


CONCLUSIONS
ReGS is not a typical female disorder as it also affects males. This notion and the finding of typical sensory abnormalities of the genital end branches of the pudendal nerve in males and females--as previously reported--provides strong evidence for Small Fiber Sensory Neuropathy as a common cause of ReGS.
Waldinger
Marcel D
MD
Department of Psychiatry and Neurosexology, HagaHospital Leyenburg, The Hague, the Netherlands. md@waldinger.demon.nl
Venema
Pieter L
PL
van Gils
Ad P G
AP
de Lint
Govert J
GJ
Schweitzer
Dave H
DH
D000328:Adult; D000368:Aged; D006801:Humans; D006941:Hyperesthesia / Q000175:diagnosis / Q000628:therapy; D008297:Male; D010523:Peripheral Nervous System Diseases / Q000175:diagnosis / Q000628:therapy; D011446:Prospective Studies; D012148:Restless Legs Syndrome / Q000175:diagnosis / Q000628:therapy; D012735:Sexual Dysfunction, Physiological / Q000175:diagnosis / Q000628:therapy; D013577:Syndrome; D004561:Transcutaneous Electric Nerve Stimulation; D053201:Urinary Bladder, Overactive / Q000175:diagnosis / Q000628:therapy