Post-orgasm pain associated with endometriosis and complete resolution of symptoms after laparoscopic en-bloc peritonectomy, a case report

A 36-year-old nulliparous woman presented with severe disabling pain (visual analog scale of pain 9/10) on the left side of the pelvis, abdomen, flank, and lower chest immediately following orgasm. She had been experiencing symptoms of constant bladder fullness and urinary urgency for six months prior. Urinary infections were ruled out through laboratory tests. Importantly, the reported symptoms were not indicative of simple dyspareunia, as sexual intercourse was normal. The pain occurred exclusively upon reaching orgasm. The patient had menarche at the age of 12 and experienced secondary dysmenorrhea since the age of 23, which improved with the administration of combined oral contraceptives (COCs). She discontinued the use of COCs 18 months before the onset of orgasm-related pain symptoms. Her menstrual cycles were regular. The patient reported a progressive worsening of menstrual cramps coinciding with the onset of orgasm-related symptoms. At this time, there were no significant dyspareunia or gastrointestinal alterations. The patient had previously attempted two alternative treatments for her symptoms under the care of other medical services. She underwent a six-month treatment with serotonin receptor inhibitor antidepressants and one year of pelvic physiotherapy, both of which failed to alleviate her condition. Endometriosis had never been diagnosed in the patient, who had regular gynecological visits since adolescence without any obvious clinical changes and had a history of normal Pap smears throughout her life. Physical examination revealed a normal, visible uterine cervix with no abnormalities. The vaginal touch maneuver revealed thickening of the left USL, accompanied by severe localized pain. No abnormalities were identified on the right side. Focal thickening of approximately 0.5 cm was observed in the retrocervical posterior vaginal wall. Deep infiltrative endometriosis of the left USL, involving the left hypogastric nerve, was suspected. Pelvic MRI confirmed irregular thickening of the left USL, with possible left hypogastric nerve retraction, common soft signs ( Fig. 1 ). Fig. 1 Sagittal and coronal sections of the pelvic MRI revealing a white area (T2) in the retrocervical region in the posterior vaginal fornix. MRI, magnetic resonance imaging. Fig. 1 Sagittal and coronal sections of the pelvic MRI revealing a white area (T2) in the retrocervical region in the posterior vaginal fornix. MRI, magnetic resonance imaging. The surgical procedure was performed in a tertiary hospital by a gynecological surgeon with expertise in video surgery and 15 years of experience in non-oncological peritonectomy and en-bloc excision for endometriosis in Brazil. Videolaparoscopic surgery revealed diffuse peritoneal endometriosis in the posterior compartment and an infiltrating nodular lesion over the hypogastric nerve bundle and left USL, causing retraction and deviation ( Fig. 2 ). A nodular lesion was observed in the posterior vaginal fornix. Increased vascularization in the posterior pelvic compartment, indicating diffuse peritoneal disease, was also noted. Fig. 2 Laparoscopic view showing the DIE (Deep infiltrating endometriosis) in the region of posterior vaginal fornix and hypogastric nerve and uterossacral ligament (USL) with local retraction. DIE, deep infiltrating endometriosis USL, uterossacral ligament. Fig. 2 Laparoscopic view showing the DIE (Deep infiltrating endometriosis) in the region of posterior vaginal fornix and hypogastric nerve and uterossacral ligament (USL) with local retraction. DIE, deep infiltrating endometriosis USL, uterossacral ligament. The surgery involved en bloc resection and complete peritonectomy of the posterior compartment, with bilateral ureter dissection, resection of hypogastric nerve endometriosis, and preservation of the hypogastric and pre sacral fascias and nerve sparing. Cold scissors were used for the procedure, and bipolar cauterization was performed for hemostasis ( Fig. 3 ). Follow-ups at 30 days, 6 months, and 1 year showed complete resolution of orgasm-related pain and urinary symptoms. Post-operative physical examination indicated the disappearance of clinical signs of endometriosis prior to surgery. The patient expressed no future desire for reproduction. Fig. 3 Laparoscopy result of the final en-bloc peritonectomy of the posterior compartment and nerve sparing. Fig. 3 Laparoscopy result of the final en-bloc peritonectomy of the posterior compartment and nerve sparing.

Igor Chiminacio: study concept or design, data collection, data analysis or interpretation, writer Carolina Obrzut: study concept or design, data collection, data analysis or interpretation, revision Samanta Saggin: data collection, review of the literature. Igor Chiminacio: study concept or design, data collection, data analysis or interpretation, writer Carolina Obrzut: study concept or design, data collection, data analysis or interpretation, revision Samanta Saggin: data collection, review of the literature.

Written informed consent was obtained from the patient for the publication of this case report and the accompanying images. A copy of the written consent form is available for review by the journal editor.

Ethical approval for this study (Ethical Committee No. 03/2022) was provided by the Ethical Committee CHIMINACIO MEDICINA DA MULHER, Pato Branco, Paraná, Brasil, on September 15, 2022.

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Name of the registry: n/a.

Igor Chiminacio.

This case, along with previous literature, demonstrates that endometriosis can manifest in a variety of ways, including pain associated with orgasm, and not just the usual dyspareunia or pain of sexual intercourse. A comprehensive understanding of the anatomic locations, development, the embryologic pattern of involvement, the natural history and clinical presentation of endometriosis is essential for effective management of these patients. The successful treatment of this patient highlights the efficacy of en-bloc excision and peritonectomy for endometriosis. Endometriosis involving the hypogastric nerve and inferior hypogastric plexus may present as post-orgasmic pain. Extensive en-bloc excision and peritonectomy with adequate nerve sparing are safe and effective treatment options resulting in long-term resolution of symptoms.

Endometriosis is a common, benign, chronic gynecological disorder affecting 10 % of women of reproductive age. The involvement of the posterior compartment accounts for 90 % of deep endometriosis cases, with the USL being the most frequent location of deep infiltrating endometriosis (DIE) [ 1 , 3 ]. While painful sexual dysfunction, particularly dyspareunia, is a typical symptom of endometriosis, orgasm-associated pain has rarely been reported and no specific causes have been identified [ 3 ]. The PubMed database search using the terms “endometriosis,” “orgasmic pain,” “hypogastric nerve,” and “orgasmic pain and endometriosis” yielded only one case report suggesting a causal association between this disorder and endometriosis [ 4 ]. No randomized controlled trials or meta-analyses on this topic were found. A case report by Yong et al. in 2020 had an association between post-orgasm pain and endometriosis, with symptom improvement with gonadotropin-releasing hormone analog use, suggesting causality to endometriosis [ 5 ]. The clinical presentation of deep endometriosis depends on the anatomical location and extent of the lesions, as well as the immunologic response during reproductive life. It may cause dysmenorrhea or chronic pelvic pain. The etiology and pathogenesis of endometriosis are multifactorial. Recent studies suggest a role for embryologic Müllerian mesodermal migration in the development of this disease [ 6 ]. Therefore, it is recommended to understand the relationship between the mesodermal layers and the peritoneum and pelvic endofascias, hypogastric fascia and pre sacral fascia, as well as the location of the hypogastric nerve and the inferior hypogastric plexus in this connective tissue into this fascias, often known as the USL, and the role of entrapment and fibrosis caused by endometriosis in the natural history of the disease [ 7 ]. The embryological concept proves to be much more plausible and interesting than the concept established almost 100 years ago with the classic description of endometriosis by John Sampson in 1927, characterizing it as retrograde menstruation. The Mullerian concept shows the real infiltration of endometriosis between the layers of the pelvis, and this is of fundamental importance when thinking about improving the results of surgical techniques that effectively remove endometriosis, following the embryological pattern for surgical systematization, it may be possible to restore the anatomy and physiology of the female pelvis [ 8 ]. Zhang et al. reported persistent clinical symptoms of endometriosis in the uterosacral branch of the inferior hypogastric plexus. The USL and hypogastric nerves were the most affected sites in DIE. Endometriosis extending along the neurovascular bundle in the USL is a common and significant cause of persistent or recurrent symptoms. DIE of the hypogastric nerve and hypogastric plexus has been frequently reported in or around the rectovaginal septum and USL. Excision of endometriotic nodules involves surgical procedures that can potentially injure the pelvic autonomic nerves in the pre sacral and pararectal spaces, which control bladder, rectal, and sexual function. The goal of the nerve-sparing approach to DIE is to preserve the inferior hypogastric nerve, inferior hypogastric plexus, and pelvic splanchnic nerves [ 9 , 10 ]. Endometriosis usually causes hematomas in the embryological patches of ectopic endometrium each cycle in the collagen permeation of the uterine parametria, this process repeatedly during reproductive life will lead to fibrosis and traction of the somatic nerve bundles below the pre-sacral fascia and consequent appearance of the symptom of dyspareunia, which is common pain during sexual intercourse. In the case presented, the patient did not have this symptom, but had pain shortly after orgasm, in which case its appearance was due to irritation or neuropathy of the hypogastric nerve, responsible for the afferent reflex of orgasm, which was in the path of the areas of endometriosis. The inferior hypogastric nerve contains fibers responsible for bladder detrusor relaxation, urethral sphincter contraction, and urinary continence and arises from the superior hypogastric plexus arising from the paraaortic sympathetic trunk. It follows an anterior and distal course lateral to the hypogastric fascia and merges with the pelvic splanchnic nerves in the pararectal space to form the inferior hypogastric plexus. The pelvic splanchnic nerves, thin parasympathetic branches arising from the sacral nerve roots (S2-S4), stimulate bladder contractions. Involvement of these anatomical sites in endometriosis results in symptoms related to plexus dysfunction as reported by the patient in the case presented [ 11 , 12 ]. In this case, COC administration masked the symptoms of endometriosis and the onset of symptoms coincided with discontinuation of the medication. COCs have been shown to reduce and control endometriosis symptoms in previous studies [ 13 ]. Therefore, clinical suspicion and correct diagnosis of endometriosis are crucial. Physical examinations play an important role in screening patients and facilitate efficient referral systems for imaging and specialized services. Extensive non-oncologic en-bloc peritonectomy of the posterior pelvic compartment, typically performed laparoscopically or robotically, is an emerging technique for removing endometriosis [ 9 ]. This procedure involves extensive removal of the peritoneum and underlying fascia to ensure optimal resection while preserving nerves. Similar techniques have been proposed by Dückelmann, who achieved favorable results in terms of symptom relief and improved fertility rates [ 14 , 15 ]. Another previous study reported significant improvement in pain symptoms in patients with deep endometriosis after systematic peritonectomy with long-term follow-up, showing reduced recurrence rates, alleviated pain symptoms, and increased fertility [ 15 , 16 ].

Endometriosis refers to the presence of endometrial glands and stroma outside the uterus. Its estimated prevalence ranges from 10 to 20 % among women of reproductive age and up to 40 % among infertile women [ 1 ]. Although endometriosis can cause various symptoms, orgasm-related pain is rarely reported or associated with the condition. Endometriosis of the uterosacral ligament (USL) is common in women with deep endometriosis and is often associated with dyspareunia [ 1 ]. The hypogastric nerve pathway, located at this anatomical site, plays a role in female orgasm, making it an interesting area to investigate in cases of post-orgasm pain associated with endometriosis. This case report describes an unusual presentation of post-orgasm pain caused by endometriosis involving the hypogastric nerve. The case was treated at a private referral center for endometriosis excision surgery in southern Brazil and has been reported following the SCARE criteria [ 2 ].

The authors declare that they have no competing interests.