Images: Sleep-related painful erection with concomitant hypnic headache.

Sleep-related painful erection (SRPE) is a parasomnia defined by the repetition of painful erections during rapid eye movement (REM) sleep. Hypnic headache (HH) is a primary headache occurring exclusively at night, often during REM sleep. We report the observation of a 33-year-old man with simultaneous SRPE and HH. Physical examination was normal. Comprehensive urological and endocrine explorations excluded other organic differential diagnoses. Polysomnography revealed several awakenings in REM, due to SRPE and concurrent HH. Medication by baclofen at bedtime seemed to have resulted in a decrease in SRPE episodes, confirmed by polysomnography, but at the cost of excessive daytime sleepiness, and was discontinued by the patient. Caffeine intake at bedtime was proposed, but the patient was reluctant because he was concerned about worsening insomnia. At 9-month follow-up, the patient had accepted his medical condition and was coping with both SRPE and HH. He felt reassured and wished no "overmedicalization." To our knowledge, the coexistence of both conditions has not yet been reported, yet their frequencies might be underestimated. We hypothesize a common underlying pathophysiology with a possible dysfunction of the vascular control and/or the autonomic nervous system and that could involve the hypothalamus. Somnologists should be aware of SRPE, potentially overlapping with HHs. SRPE should be considered in case of sleep-maintenance insomnia. Patient reassurance seems to be central in the care process of SRPE. CITATION: Moreau A, Monnier L, Medde A, Bourgin P, Ruppert E. Images: sleep-related painful erection with concomitant hypnic headache. J Clin Sleep Med. 2024;20(5):837-839.

Improvement of associated symptoms using combined therapy in 44 patients with sleep-related painful erection during 1-year follow up.

The current study was aimed at analysing the clinical features and efficacy of combined treatments in patients with sleep-related painful erection (SRPE). Patients who presented with SRPE were continuously enrolled from the outpatient clinic of Peking Union Medical College Hospital from 2015 to 2021. Demographic data, medical history, diagnostics, treatment options and their effectiveness on SRPE in the short and long therapeutic term were recorded. Individually designed combined therapy aimed at controlling SRPE-related symptoms and comorbidities (general health, pain, psychological and sleeping disorders, late-onset hypogonadism, and lower urinary tract symptoms) was used, and the effectiveness was evaluated. In total, 44 patients with an average age of 44.66 ± 7.96 years were enrolled. The median length of the delay in diagnosis was 1.5 years (range, 1 month to 27 years). Combined treatment aimed at controlling symptoms was used, the mean GAD-7, PHQ-9, PSQI and VAS scores were significantly decreased to 4.25 ± 3.44, 4.55 ± 2.86, 7.65 ± 3.06, and 2.90 ± 1.89 after treatment for 3 months. Moreover, the VAS ratings were significantly decreased at 1 year of follow-up (p < 0.001). SRPE mainly occurred in middle aged males, 79.55% (35/44) patients were more than 40 years old. The prevalence of anxiety, depression, poor sleep and nocturia is high in patients with SRPE. Combined treatments aimed at controlling these symptoms can be more effective.

Sleep related painful erection: an algorithm for evaluation and management.

BACKGROUND: Sleep related painful erection (SRPE) is a rare parasomnia consisting of nocturnal penile tumescence accompanied by pain that awakens the individual. Normal non-painful erections are experienced when awake. No penile anatomic abnormalities are present. No conclusive randomized clinical trial is present in the literature about the management of this rare condition. The aim of this article is to review the current knowledge about the management of SRPE and to suggest an algorithm to help physicians evaluate and manage SRPE. MATERIAL AND METHODS: A literature review was conducted through PubMed database using the terms: sleep, pain, painful, penile, and erection. The reference lists of the articles were also reviewed. The search returned 23 references that were published between 1987 and 2019. Results were presented in a descriptive manner. RESULTS: Treatment decision for now is based on reports of the treatment success, the sustainability of remission, the tolerability by the patients and the potential side effects of each medication. From data available in literature, Baclofen is the mostly used medication with a tolerable profile of adverse effects. Phosphodiesterase type 5 inhibitors are considered potential treatments and are already widely used and tolerated for other indications, but so far only 2 successful trials have been reported for SRPE. Cinitapride is very promising, but only one case was studied and no side effects were reported. Clozapine can be very dangerous although highly effective. CONCLUSION: Based on the limited number of treatment trials and reported cases, the low level of evidence and the lack of randomized clinical trials, no treatment consensus for SRPE can be reached. We suggested a useful tool for clinicians: an algorithm for the management of SRPE to facilitate their access to the literature without exhaustive return to case reports and series upon each case faced.

CONTEXTE: L'érection nocturne douloureuse est. une parasomnie, caractérisée par des épisodes de tumescence douloureuse provenant exclusivement lors du sommeil, avec une anatomie normale et des érections indolores lorsque le patient est. réveillé. La prise en charge de cette maladie est. inconnue vue le petit nombre de cas et le manque d'études randomisées. Nous présentons dans cet article une revue de la littérature concentrée sur les stratégies thérapeutiques décrites. Un algorithme est. suggéré pour faciliter l'évaluation et la prise en charge des patients. MATÉRIEL ET MÉTHODES: La revue de la littérature a été réalisée en utilisant la base de donnée PubMed et les termes: « sleep, pain, painful, penile, erection ¼. Les références des articles ont été revues. En total 23 références ont été utiles pour notre revue. Les résultats ont été présentés d'une manière descriptive. RÉSULTATS: Les stratégies thérapeutiques actuelles se basent sur les essais décrits dans la littérature à travers un nombre limité de patients. La durée de la rémission et les effets secondaires minimes rendent certains traitements supérieurs. Le baclofen est. le plus utilisé grâce à son efficacité et à son faible potentiel d'effets secondaires. Le cinitapride a été très efficace lors d'un essai unique chez un seul patient, sans effets secondaires rapportés. Les inhibiteurs de la phosphodiesterase de type 5, largement utilisés en urologie, ont été trouvés utiles pour le traitement des érections nocturnes douloureuses chez 2 patients. La clozapine qui est. très efficace, présente un profil d'effets secondaires dangereux. CONCLUSION: A cause du faible nombre de cas et des essais thérapeutiques rapportés, le meilleur traitement des patients ayant des érections douloureuses nocturnes reste indéterminé. Nous suggérons un algorithme utile aux praticiens pour l'évaluation, le diagnostic et la prise en charge de ces patients.

[Considerations on priapism].

Priapism is a rare pathological penile erection, and there are some inadequacies in its definition, classification, diagnosis, and therapeutic strategies. In this article, we sum up our years of experience with priapism and put forward some new views and ideas about its definition, classification, pathophysiologic process, pathological change, diagnostic essentials, therapeutic measures, indications of successful treatment, and post-therapeutic rehabilitation of erectile function. We also describe the clinical features, diagnosis and treatment of some special types of priapism, such as intermittent seizure, sleep-related painful erection, and tumor-related priapism, hoping to help urologists and andrologists in the further understanding and management of priapism.

Sleep-Related Painful Erections: A Meta-Analysis on the Pathophysiology and Risks and Benefits of Medical Treatments.

BACKGROUND: Patients with sleep-related painful erections (SRPEs) have frequent awakenings from deep penile pain during nocturnal erections. This results in severe sleep deprivation. AIM: To review the current literature on SRPEs and discuss the pathophysiologic theories and risks and benefits of medical treatments. METHODS: PubMed was searched using the terms sleep-related painful erections, nocturnal priapism, treatment, and sleep-related erections. OUTCOME: Variables included patient demographics, medical history, diagnostics, hypotheses on pathophysiology, and treatment modalities and their effect on SRPE in the short and long term. RESULTS: The search yielded in 66 SRPE cases that were analyzed, including our mono-institutional series of 24 patients. The phenomenon of SRPEs is not well understood. Theories about pathophysiology concerned increased serum testosterone levels, altered autonomic function, compression of the lateral preoptic area, coexistent obstructive sleep apnea syndrome, the existence of a "compartment syndrome," and psychosomatic factors. Except for polysomnographic findings that showed sleep fragmentation and decreased sleep efficiency in all patients, other diagnostic results varied widely. Multiple agents were tried. Baclofen and, to lesser degree, clonazepam showed noticeable results, most likely due to their influence on the γ-aminobutyric acid system and, hence, suppression of glutamate release. In addition, baclofen relaxes the ischiocavernosus and bulbospongiosus muscles, which are involved in penile erection. CLINICAL IMPLICATIONS: By providing a critical analysis and complete overview on the limited literature about this overlooked and undermanaged condition, this review contributes to a better understanding of the pathophysiology and provides directions for future research on the treatment of SRPE. STRENGTHS AND LIMITATIONS: Because the literature on SRPEs includes only case reports and small case series, the level of evidence of treatment advice is limited. CONCLUSION: The pathophysiology of SRPEs is not yet clarified. Further diagnostic evaluation, including electromyography of the ischiocavernosus and bulbospongiosus muscles to elucidate the pathophysiology, is recommended. Prospective controlled investigations are warranted to assess the efficacy and safety of long-term use of baclofen and develop evidence-based treatment advice. Vreungdenhil S, Weidenaar AC, de Jong IJ, van Driel MF. Sleep-Related Painful Erections: A Meta-Analysis on the Pathophysiology and Risks and Benefits of Medical Treatments. J Sex Med 2018;15:5-19.

Considerations on priapism / 中华男科学杂志

Priapism is a rare pathological penile erection, and there are some inadequacies in its definition, classification, diagnosis, and therapeutic strategies. In this article, we sum up our years of experience with priapism and put forward some new views and ideas about its definition, classification, pathophysiologic process, pathological change, diagnostic essentials, therapeutic measures, indications of successful treatment, and post-therapeutic rehabilitation of erectile function. We also describe the clinical features, diagnosis and treatment of some special types of priapism, such as intermittent seizure, sleep-related painful erection, and tumor-related priapism, hoping to help urologists and andrologists in the further understanding and management of priapism.

Sleep-Related Painful Erections-A Case Series of 24 Patients Regarding Diagnostics and Treatment Options.

BACKGROUND: Patients with sleep-related painful erections (SRPEs) have deep penile pain during nocturnal erection that wakes them up and disturbs their nights of sleep. This rare parasomnia is poorly recognized by general practitioners and by urologists and sexologists. AIM: To gain more insight into diagnostics and therapeutic options. METHODS: Data from a series of 24 consecutive patients who presented with SRPEs at the outpatient clinic from 1996 to 2015 were retrospectively analyzed. Additional questionnaires were completed to complement data and to obtain information about follow-up. Long-term treatment efficacy of baclofen was assessed using the Wilcoxon signed rank test. OUTCOMES: SRPEs were not associated with urologic, surgical, or psychiatric history or with serum testosterone levels. The mean doctors' delay was 3.5 years. 14 of the 24 patients were treated with baclofen (10-75 mg). In 11 of them, complete remission was observed within a few weeks. 2 of the 3 remaining patients noticed a slight improvement of SPRE symptoms and only 1 patient experienced no effect at all. After an average follow-up of 4.5 years, only 41.6% of patients who had used baclofen were satisfied with their SRPEs. The others (58.4%) were dissatisfied, mostly owing to relapse of symptoms after the discontinuation of baclofen. Other treatment forms were applied sporadically, with strongly varying results. CLINICAL IMPLICATIONS: This overview of SRPE contributes to a better clinical understanding and recognition of the phenomenon and provides new, more constructed advice about therapeutic implications, especially concerning the use of baclofen. STRENGTHS AND LIMITATIONS: This study provides a systematic overview of a relatively large series of patients with SRPE, which provides substantiated treatment advice. However, treatment efficacy was based mainly on the patients' subjective perception and it was not possible to compare the results of baclofen with other forms of pharmacologic treatment, because these alternative drugs were applied only sporadically. Nevertheless, this study is directional for future research. CONCLUSIONS: This study confirmed a long doctors' delay in patients with SRPE. There was no association between SRPEs and comorbidity and total serum testosterone levels. Treatment with baclofen proved successful and safe in the short term. Long-term feasibility needs further investigation. Vreugdenhil S, Weidenaar AC, de Jong IJ, van Driel MF. Sleep-Related Painful Erections-A Case Series of 24 Patients Regarding Diagnostics and Treatment Options. Sex Med 2017;5:e237-e243.

Two Cases of Sleep-Related Painful Erection

Sleep-related painful erection (SRPE) is characterized by deep penile pain accompanied with erection occurring rapid eye movement (REM) movement period. Two (47-year-old and 40-year-old, respectively) male visited with the complaint of painful penile erection occurring during sleep. They had no problems with erection during daytime sexual activities except for mild premature ejaculation in one patient. Urologic inspections revealed no focal abnormalities. Polysomnography with simultaneous penile erection monitoring showed several episodes of awakening with painful erection which are time-locked to onset of REM sleep periods. According to the diagnostic criteria in international classification of sleep disorders, each patient was diagnosed to have chronic, severe SRPE. Despite of a low prevalence of SRPE, this condition should be considered in a patient who presents with nocturnal penile. A polysomnography accompanied with penile erection recording may help confirm diagnosis.