Research productivity in catamenial epilepsy: A bibliometric analysis of worldwide scientific literature (1956-2022).

Objectives: To perform a bibliometric analysis as a comprehensive review of publications associated with catamenial epilepsy and discuss the current state of knowledge in the field. Methods: Publications published between 1956 and 2022 were retrieved from the Scopus database. Bibliometric analysis was performed using the R package and VOSviewer to show the data and network of journals, organizations, authors, countries, and keywords. The analysis conducted on October 15, 2022, yielded a total of 320 refinement studies. Results: The number of publications has escalated significantly, particularly in the last 20 years. Catamenial epilepsy-related publications originated mostly from medicine and other subject areas, with the United States having the largest publication output. Collaboration is low at the author, organizational, and national levels, especially in the Asian continent. Publications remain scarce, particularly on practice guidelines, risk assessment, and medication-related research. Based on a keyword analysis, a bibliometric analysis identified possible themes for future investigation. Conclusion: Catamenial epilepsy-related literature is crucial but still insufficient, and further studies are required.

Ictal activity is sustained by the estrogen receptor ß during the estrous cycle.

Catamenial epilepsy, defined as a periodicity of seizure exacerbation during the menstrual cycle, affects up to 70 % of epileptic women. Seizures in these patients are often non-responsive to medication; however, our understanding of the relation between menstrual cycle and seizure generation (i.e. ictogenesis) remains limited. We employed here field potential recordings in the in vitro 4-aminopyridine model of epileptiform synchronization in female mice (P60-P130) and found that: (i) the estrous phase favors ictal activity in the entorhinal cortex; (ii) these ictal discharges display an onset pattern characterised by the presence of chirps that are thought to mirror synchronous interneuron firing; and (iii) blocking estrogen receptor ß-mediated signaling reduces ictal discharge duration. Our findings indicate that the duration of 4AP-induced ictal discharges, in vitro, increases during the estrous phase, which corresponds to the human peri-ovulatory period. We propose that these effects are caused by the presumptive enhancement of interneuron excitability due to increased estrogen receptor ß-mediated signaling.

A surgical series on endometriosis-related diaphragmatic hernia.

BACKGROUND: Thoracic endometriosis syndrome gives rise to various clinical and radiological manifestations. We reviewed the records of patients operated for intrathoracic migration of abdominal viscera through a diaphragmatic hernia secondary to thoracic endometriosis. METHODS: We retrospectively reviewed the single-center prospective collected database of all patients operated for thoracic endometriosis during the twenty years. All cases in which an abdominal organ was found to be herniated into the thoracic cavity were retrieved. Clinical and pathological data are presented and analyzed. RESULTS: Twenty women of median age 36 (range 25-58) years were operated for endometriosis-related diaphragmatic hernia. The hernia was diagnosed concomitantly with endometriosis-related pneumothorax in 13 cases and during the exploration of catamenial thoracic pain in seven cases. There were 18 cases on the right side and two cases on the left side. The median diameter of the hernia was 8 cm (2.5-20 cm). In seventeen cases, the hernia was repaired by direct suture, and in three cases a heterologous prosthesis was positioned. At follow-up, two patients had an episode of recurrent pneumothorax. CONCLUSIONS: Diaphragmatic hernia should be ruled out in the presence of endometriosis-related pneumothorax or catamenial thoracic pain. Surgery is indicated to make a pathological diagnosis, restore anatomy, and prevent recurrence in patients presenting with pneumothorax.

Case report: catamenial hyperglycemia: the trigger of recurrent DKA in a female patient with three-year follow-up.

Background: Catamenial hyperglycemia is a rare type of spontaneous, recurring Diabetic Ketoacidosis(DKA) in females during the luteal phase, most commonly observed in type 1 diabetes mellitus. Even with controlled serum glucose levels, adherence to a diabetic diet, medications, and in the absence of other common influencing factors such as infection, glucose levels tend to increase during the premenstrual period. This uncommon issue related to the menstrual cycle phase has not been extensively researched. Therefore, this study aims to diagnose catamenial hyperglycemia promptly and initiate early treatment to prevent complications. Case report: We presented a case of a 19-year-old girl who experienced recurrent DKA during the premenstrual period, without an apparent cause. She was admitted multiple times to various hospitals and sought consultations, undergoing numerous laboratory and imaging examinations, yet the etiology remained elusive. Ultimately, she received a diagnosis of catamenial diabetic hyperglycemia. To prevent recurrence of complications associated with catamenial hyperglycemia, we initiated a comprehensive approach which included continuous glucose monitoring, adherence to a strict diabetic diet, diabetic health education, regular exercise, timely medication administration, and increase in insulin dosage during the premenstrual period based on glucose levels. Conclusions: Although catamenial hyperglycemia is rare, it should be considered a cause of recurrent hyperglycemia in any postpubertal female to prevent complications. The specific underlying mechanisms responsible for catamenial hyperglycemia or DKA remain unidentified.

Parietal endometriosis: An uncommon cause of c-section scar pain - A case report.

INTRODUCTION: Parietal endometriosis is the most common form of extra-pelvic endometriosis. It develops on the surgical scar of c-section or hysterectomy. It is one of the causes of scar pain. CASE PRESENTATION: A 26 years old patient presents with recurring pain and swelling of a Pfannenstiel scar 6 years after a caesarean section. Physical examination revealed a firm tender subcutaneous nodule that appeared at MRI as a heterogenous parietal mass infiltrating the rectus abdominis muscles. The patient underwent a wide excision of the nodule. DISCUSSION: Parietal endometriosis can be the cause of debilitating scar pain even in patients with no history of deep endometriosis. It presents as firm parietal nodule that can become large and infiltrative if left untreated. Diagnosis is purely histological. Surgery remains the treatment of choice and requires a wide excision. CONCLUSION: Parietal endometriosis, potentially more common due to rising number of caesarean sections, should be considered with persistent scar pain. Surgery is the preferred treatment, offering a conclusive diagnosis.

Refractory tonic-myoclonic status epilepticus with catamenial recurrence in epilepsy with myoclonic atonic seizures: A case report.

In epilepsy with myoclonic-atonic seizures (EMA), status epilepticus (SE) may occur during the onset phase, uncommonly in post-puberal patients. We report a post-puberal patient with EMA who presented SE with insidious onset and catamenial recurrence. She had a stormy epilepsy onset at 4 years, with tonic seizures, atypical absences, and myoclonic-atonic seizures, in the absence of SE. After the onset phase, sporadic nocturnal tonic seizures persisted and a mild intellectual disability appeared. At the age of 7, after gonadotropin-releasing hormone analog administration due to central precocious puberty, she presented with SE characterized by recurrent atypical absences, tonic seizures, and awareness impairment, which was successfully treated in 4 days. At 11 years, one week before menstruation, the patient presented with analogous SE that lasted 8 days. One week before the subsequent menstruation, she presented again with SE, initially characterized by atypical absences alternating with phases of awareness and motor impairment related to fast low-voltage EEG activity in the central regions; later, tonic and myoclonic seizures occurring even in the awake state increased, and the "atonic-akinetic status" related to fast EEG activity worsened. After conventional antiepileptic drugs had failed to control the seizures, a progestin was added, with subsequent gradual complete recovery.

Spontaneous recurrent menstrual pneumothorax: a case report.

Introduction and importance: Endometriosis is most commonly found in the pelvic area, ~12% of people have it in other areas or organs, which is known as extrapelvic endometriosis. Thoracic endometriosis, which is also classified as extrapelvic endometriosis, manifests with four distinct forms: catamenial pneumothorax, catamenial hemothorax, catamenial hemoptysis, or lung nodules. Catamenial pneumothorax is the most common clinical symptom of these; however, it is frequently neglected by clinicians and goes undiagnosed and untreated. As a result, it is critical to raise awareness of this medical condition among clinicians. Case presentation: The authors present a case report of a 34-year-old woman of reproductive age who had recurrent episodes of spontaneous pneumothorax during menstruation and underwent treatment with thoracoscopic surgery as well as gynaecological hormonal drugs including oral progesterone and dienogest throughout this time. Based on her symptoms, a catamenial pneumothorax caused by thoracic endometriosis was suspected. Clinical discussion: The clinical symptoms, pathogenesis, diagnosis, and treatment of Catamenial Pneumothorax are analyzed. Furthermore, the usage of gynaecological hormone medications in this condition has been discussed. The mechanisms of oral contraceptives and progestin-based medications are evaluated by comparing the patient's treatment process, highlighting their pros and cons. Conclusions: Thoracoscopic surgery combined with postoperative gynaecological hormonal medications may be the most effective treatment for this issue. Several gynaecological hormonal medicines are available, each of which has its own set of pros and cons, and must be thoroughly evaluated as well as correctly tailored to the patient's specific circumstances to have a positive therapeutic outcome.

Neumotórax catamenial con fuga aérea persistente / Catamenial pneumothorax with persistent air leak

El neumotórax catamenial (NC) es aquel neumotórax espontáneo y recurrente que se presenta en mujeres en edad reproductiva y en relación temporal con la menstruación. Se han descrito múltiples variaciones en cuanto a la relación temporal, aunque suele producirse 24 horas antes del inicio de la menstruación o 72 horas después. Su consideración de patología poco frecuente podría deberse a que sea infradiagnosticada debido a la falta de conocimiento. El diagnóstico de NC no suele ser fácil; depende principalmente de la historia clínica pero también puede ser un diagnóstico quirúrgico o histopatológico. Las estrategias de manejo del NC pueden incluir cualquier combinación de terapia hormonal, pleurodesis, resección de parénquima pulmonar y resección/reparación del diafragma. Se presenta este caso de neumotórax catamenial de manifestación atípica para resaltar la importancia de tener un adecuado conocimiento de esta enfermedad que, por su aparente baja incidencia, puede pasar desapercibida. (AU)

Catamenial pneumothorax is a spontaneous recurrent pneumothorax that occurs in women of reproductive age and in temporal relationship with the menses. It usually occurs within 24 hours before or 72 hours after the start of the menstrual cycle; however, multiple variations have been described in terms of the temporal relationship. To date, it is considered a rare condition; this may be justified by the limited available knowledge on this disease leading to mis-diagnosis. Often, making a diagnosis of catamenial pneumo-thorax is complicated; it mainly depends on the medical history; also, it may be a surgical or histopathological diagnosis. Management strategies for catamenial pneumothorax may include combinations of hormonal therapy, pleurodesis, lung parenchyma resection, and diaphragm resection/repair.The aim of reporting this atypical case of catamenial pneumothorax is to highlight the relevance of having the adequate knowledge on this entity so it does not go unnoticed despite its apparent low incidence. (AU)

Female sex steroids and epilepsy: Part 2. A practical and human focus on catamenial epilepsy.

Catamenial epilepsy is the best described and most researched sex steroid-specific seizure exacerbation. Yet despite this there are no current evidence-based treatments, nor an accepted diagnostic tool. The best tool we currently have is tracking seizures over menstrual cycles; however, the reality of tracking seizures and menstrual cycles is fraught with challenges. In Part 1 of this two-part review, we outlined the often complex and reciprocal relationship between seizures and sex steroids. An adaptable means of tracking is required. In this review, we outline the extent and limitations of current knowledge on catamenial epilepsy. We use sample data to show how seizure exacerbations can be tracked in short/long and even irregular menstrual cycles. We describe how seizure severity, an often overlooked and underresearched form of catamenial seizure exacerbation, can also be tracked. Finally, given the lack of treatment options for females profoundly affected by catamenial epilepsy, Section 3 focuses on current methods and models for researching sex steroids and seizures as well as limitations and future directions. To permit more informative, mechanism-focused research in humans, the need for both a consistent classification of catamenial epilepsy and an objective biomarker is highlighted.

Female sex steroids and epilepsy: Part 1. A review of reciprocal changes in reproductive systems, cycles, and seizures.

Seizures, antiseizure medications, and the reproductive systems are reciprocally entwined. In Section 2 of this review, we outline how seizures may affect the hypothalamic-pituitary-gonadal axis, thereby altering sex steroids, and changes in sex steroids across the menstrual cycle and changes in pharmacokinetics during pregnancy may alter seizure susceptibility. The literature indicates that females with epilepsy experience increased rates of menstrual disturbances and reproductive endocrine disorders. The latter include polycystic ovary syndrome, especially for females on valproate. Studies of fertility have yielded mixed results. We aim to summarize and attempt to detangle the existing knowledge on these reciprocal interactions. The menstrual cycle causes changes in seizure intensity and frequency for many females. When this occurs perimenstrually, during ovulation, or in association with an inadequate luteal phase, it is termed catamenial epilepsy. There is a clear biophysiological rationale for how the key female reproductive neurosteroids interact with the brain to alter the seizure threshold, and Section 3 outlines this important relationship. Critically, what remains unknown is the specific pathophysiology of catamenial epilepsy that describes why not all females are affected. There is a need for mechanism-focused investigations in humans to uncover the complexity of the relationship between reproductive hormones, menstrual cycles, and the brain.

Catamenial haemothorax-A rare cause of pleural effusion.

Common causes of haemorrhagic pleural effusions include malignancy (primary or metastatic), tuberculosis, pulmonary embolism, collagen vascular diseases, trauma and iatrogenic causes. Clinical history along with pathologic, microbiologic and biochemical evaluation of pleural fluid confirms the diagnosis in most cases. However, if there is recurrent haemorrhagic effusion without corroborative history or mass lesion in lung, or evidence of microorganisms, then we should think of uncommon causes. Catamenial haemothorax (CHt) is a rare cause of haemorrhagic pleural effusion, which recurs during each menstrual cycle. This is a manifestation of thoracic endometriosis syndrome (TES) caused by ectopic endometrial tissue in the thoracic cavity in women of child-bearing age. This extremely rare condition is difficult to diagnose, unless direct correlation with the menstrual cycle is established. TES consists of pleural forms such as catamenial pneumothorax, non-catamenial endometriosis-related pneumothorax and haemothorax; and parenchymal forms such as catamenial haemoptysis and lung nodules. Here we report a case of CHt in a 43-year-old female whose diagnosis was established by thoracoscopic pleural biopsy.

Scar Endometriosis vs Hemangioma: A Diagnostic Conundrum.

Scar endometriosis refers to the presence of endometrial glands and stroma at the site of a scar. Hemangiomas, on the other hand, are benign vascular tumors. In this case report, we unravel the clinical enigma around a patient who presented with a painful mass at the previous cesarean section scar site. Initially, we were confident that this was ectopic endometrium presenting as scar endometriosis. However, our journey took an unexpected turn when histopathological findings contradicted our clinical suspicions. Here, we delve into the intricate details of this captivating case, shedding light on the complexities of the diagnosis we faced.

Navigating Nasal Surges: Understanding Catamenial Epistaxis.

Catamenial epistaxis is a rare form of epistaxis (nosebleed) that occurs in women during menstruation due to hormonal changes. There are numerous hypotheses on the cellular mechanisms and pathophysiology of endometriosis. Endometriosis may present a wide range of symptoms depending on where endometrial tissue was implanted. This entity's diagnosis is neither simple nor difficult. There are numerous clinical and laboratory diagnostic techniques in use, but none of them is considered to be the best. Every woman who experiences recurrent symptoms (such as epistaxis and hemoptysis) of extrapelvic organs should be clinically suspicious of endometriosis because of its multipotent location and the variety of clinical manifestations of the condition. This case report demonstrates that periodic epistaxis may infrequently be the root cause of the extra pelvic endometrium in the nasal septum in a woman who has had treatment for recurrent pelvic discomfort and dysmenorrhea.

Catamenial Pneumothorax: A Rare Diagnosis Among Menstruating Women.

Catamenial Pneumothorax is a rare condition often associated with endometriosis in menstruating women. Due to the rarity of this condition, its etiology is not well studied and, thus, effective treatment regimens have not been well established. We present a case of a 21-year-old female with no significant past medical history who developed recurrent episodes of spontaneous pneumothorax, chronologically associated with her menstrual cycle. This pattern is known as the sine qua non criteria and is one of the only established criteria in current literature for diagnosing catamenial pneumothorax. Our aim with this case report is to expand the current collection of published knowledge about this rare condition and to bring awareness so that those affected by catamenial pneumothorax can be diagnosed and treated more efficiently. Additional research on the pathophysiology of this disease needs to be done to aid in the development of effective treatment regimens.

Diaphragmatic Endometriosis Presenting as Recurrent Catamenial Pneumothorax: A Case Report.

Catamenial pneumothorax is one of the most common extra-pelvic presentations of endometriosis, with the gastrointestinal tract being the most common location. Catamenial pneumothorax is defined as spontaneous recurrent pneumothorax occurring in women of reproductive age in a temporal relationship with menses. Symptoms include dyspnea, sharp chest pain, and hypoxemia. A much rarer presentation is the involvement of endometriosis with the diaphragm. In this case, we present a 31-year-old female who presented with signs of pneumothorax. She has had multiple episodes leading to suspicion of catamenial pneumothorax. However, it wasn't until her surgery that the extent of diaphragmatic involvement, characterized by numerous holes secondary to endometriosis, was discovered. She was surgically treated, which led to a drastic improvement in symptoms and a reduction in subsequent episodes. We hope that this case can add to the current limited literature on diaphragmatic endometriosis cases. Since this patient presented with mainly catamenial pneumothorax symptoms, we urge clinicians to still consider diaphragmatic involvement as a primary cause in patients with recurrent episodes of pneumothorax.

Thoracic Endometriosis Syndrome (TES) in Martinique, a French West Indies Island.

INTRODUCTION: Endometriosis is a female disease that affects 5-10% of women of childbearing age, with predominantly pelvic manifestations. It is currently declared as a public health priority in France. Thoracic endometriosis syndrome (TES) is the most common extra-pelvic manifestation. OBJECTIVE: The objective of this study was to describe the epidemiological and clinical characteristics, and outcomes of patients with TES in Martinique. PATIENTS AND METHODS: We performed a descriptive, retrospective study including all patients managed at the University Hospital of Martinique for TES between 1 January 2004 and 31 December 2020. RESULTS: During the study period, we identified 479 cases of pneumothorax, of which 212 were women (44%). Sixty-three patients (30% of all female pneumothorax) were catamenial pneumothorax (CP) including 49 pneumothoraxes alone (78% of catamenial pneumothorax) and 14 hemopneumothorax (22% of catamenial pneumothorax). There were 71 cases of TES, including 49 pneumothoraxes (69%), 14 hemopneumothoraxes (20%) and 8 hemothorax (11%). The annual incidence of TES was 1.1 cases/100,000 inhabitants. The prevalence of TES was 1.2/1000 women aged from 15 to 45 years and the annual incidence of TES for this group was 6.9/100,000. The annual incidence of CP was 1 case/100,000 inhabitants. The average age at diagnosis was 36 ± 6 years. Eight patients (11%) had no prior diagnosis of pelvic endometriosis (PE). The mean age at pelvic endometriosis diagnosis was 29 ± 6 years. The mean time from symptom onset to diagnosis was 24 ± 50 weeks, and 53 ± 123 days from diagnosis to surgery. Thirty-two patients (47%) had prior abdominopelvic surgery. Seventeen patients (24%) presented other extra-pelvic localizations. When it came to management, 69/71 patients (97%) underwent surgery. Diaphragmatic nodules or perforations were found in 68/69 patients (98.5%). Histological confirmation was obtained in 55/65 patients who underwent resection (84.6%). Forty-four patients (62%) experienced recurrence. The mean time from the initial treatment to recurrence was 20 ± 33 months. The recurrence rate was 16/19 (84.2%) in patients who received medical therapy only, 11/17 (64.7%) in patients treated by surgery alone, and 17/31 (51.8%) in patients treated with surgery and medical therapy (p = 0.03). CONCLUSIONS: We observed a very high incidence of TES in Martinique. The factors associated with this high incidence in this specific geographical area remain to be elucidated. The frequency of recurrence was lower in patients who received both hormone therapy and surgery.

Postoperative hormonal therapies reduce the recurrence of thoracic endometriosis-related pneumothorax.

OBJECTIVES: Thoracic endometriosis-related pneumothorax (TERP) frequently recurs even after surgery. Meanwhile, postoperative hormonal therapies (HTx) are believed to be effective for pelvic endometriosis. Therefore, we evaluated the relationship between postoperative TERP recurrence and postoperative HTx in a retrospective observational study. METHODS: We retrospectively reviewed the data of patients with TERP who underwent the first video-assisted thoracoscopic surgery between January 2011 and February 2022. RESULTS: Of the 248 patients eligible for this study, 67 (27.0%) experienced postoperative TERP recurrence. Postoperative HTx were administered to 70 patients (28.2%). Dienogest was the most frequently administered drug, given to 56.7% of patients. Following univariable analysis, postoperative hormonal therapies was closely related to reduce postoperative recurrence (P = 0.003). Likewise, the multivariable analysis revealed postoperative hormonal therapies were significantly associated with the risk reduction of recurrence (hazard ratio 0.28, P < 0.001). CONCLUSIONS: Postoperative HTx reduced TERP recurrence. We hypothesize that HTx may control residual endometrial tissues to avoid TERP if pleural endometrial tissues are resected as much as possible.

A scoping review of hormonal clinical trials in menstrual cycle-related brain disorders: Studies in premenstrual mood disorder, menstrual migraine, and catamenial epilepsy.

Cyclic variations in hormones during the normal menstrual cycle underlie multiple central nervous system (CNS)-linked disorders, including premenstrual mood disorder (PMD), menstrual migraine (MM), and catamenial epilepsy (CE). Despite this foundational mechanistic link, these three fields operate independently of each other. In this scoping review (N = 85 studies), we survey existing human research studies in PMD, MM, and CE to outline the exogenous experimental hormone manipulation trials conducted in these fields. We examine a broad range of literature across these disorders in order to summarize existing diagnostic practices and research methods, highlight gaps in the experimental human literature, and elucidate future research opportunities within each field. While no individual treatment or study design can fit every disease, there is immense overlap in study design and established neuroendocrine-based hormone sensitivity among the menstrual cycle-related disorders PMD, MM, and CE. SCOPING REVIEW STRUCTURED SUMMARY: Background. The menstrual cycle can be a biological trigger of symptoms in certain brain disorders, leading to specific, menstrual cycle-linked phenomena such as premenstrual mood disorders (PMD), menstrual migraine (MM), and catamenial epilepsy (CE). Despite the overlap in chronicity and hormonal provocation, these fields have historically operated independently, without any systematic communication about methods or mechanisms. OBJECTIVE: Online databases were used to identify articles published between 1950 and 2021 that studied hormonal manipulations in reproductive-aged females with either PMD, MM, or CE. We selected N = 85 studies that met the following criteria: 1) included a study population of females with natural menstrual cycles (e.g., not perimenopausal, pregnant, or using hormonal medications that were not the primary study variable); 2) involved an exogenous hormone manipulation; 3) involved a repeated measurement across at least two cycle phases as the primary outcome variable. CHARTING METHODS: After exporting online database query results, authors extracted sample size, clinical diagnosis of sample population, study design, experimental hormone manipulation, cyclical outcome measure, and results from each trial. Charting was completed manually, with two authors reviewing each trial. RESULTS: Exogenous hormone manipulations have been tested as treatment options for PMD (N = 56 trials) more frequently than MM (N = 21) or CE (N = 8). Combined oral contraceptive (COC) trials, specifically those containing drospirenone as the progestin, are a well-studied area with promising results for treating both PMDD and MM. We found no trials of COCs in CE. Many trials test ovulation suppression using gonadotropin-releasing hormone agonists (GnRHa), and a meta-analysis supports their efficacy in PMD; GnRHa have been tested in two MM-related trials, and one CE open-label case series. Finally, we found that non-contraceptive hormone manipulations, including but not limited to short-term transdermal estradiol, progesterone supplementation, and progesterone antagonism, have been used across all three disorders. CONCLUSIONS: Research in PMD, MM, and CE commonly have overlapping study design and research methods, and similar effects of some interventions suggest the possibility of overlapping mechanisms contributing to their cyclical symptom presentation. Our scoping review is the first to summarize existing clinical trials in these three brain disorders, specifically focusing on hormonal treatment trials. We find that PMD has a stronger body of literature for ovulation-suppressing COC and GnRHa trials; the field of MM consists of extensive estrogen-based studies; and current consensus in CE focuses on progesterone supplementation during the luteal phase, with limited estrogen manipulations due to concerns about seizure provocation. We argue that researchers in any of these respective disciplines would benefit from greater communication regarding methods for assessment, diagnosis, subtyping, and experimental manipulation. With this scoping review, we hope to increase collaboration and communication among researchers to ultimately improve diagnosis and treatment for menstrual-cycle-linked brain disorders.