Is painful rectovaginal endometriosis an intermediate stage of rectal endometriosis?

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1 Is painful rectovaginal endometriosis an intermediate stage of rectal endometriosis? Horace Roman, M.D., a Alexis Gromez, M.D., a Patrick Hochain, M.D., b Nolwenn Marouteau-Pasquier, M.D., c Jean-Jacques Tuech, M.D., Ph.D., d Benoit Resch, M.D., a and Loic Marpeau, M.D., Ph.D. a a Department of Gynecology and Obstetrics, c Department of Imagery, and d Department of Digestive Surgery, University Hospital Rouen, France; and b Clinique du Cedre, Boisguillaume, France Objective: To compare the history of pain complaints of women presenting rectovaginal and rectal endometriosis to show that rectovaginal locations may progress to a rectal involvement of the disease. Design: Retrospective comparative study. Setting: Department of Gynecology and Obstetrics, University Hospital Rouen, France. Patient(s): Thirty-two patients with rectovaginal endometriosis and 16 patients with rectal involvement. Intervention(s): Standardized questionnaires recording the clinical history of painful deep endometriosis up to diagnosis. Main Outcome Measure(s): Length of time from onset of pain to diagnosis, types of pain, disability related to the pain, and number of physicians consulted before the diagnosis was made. Result(s): Women with rectal endometriosis had an earlier onset of dysmenorrhoea. The age of dysmenorrhoea and the length of time between the onset of the first pain to the first time that the endometriosis was suspected were significantly increased in women with rectal endometriosis. Pain during defecation was more frequent in patients with rectal endometriosis. Women consulted an average of three physicians before the endometriosis diagnosis was suggested. A nongynecologist physician made the diagnosis of rectovaginal and rectal endometriosis in respectively 26% and 31% of cases. Conclusion(s): Rectal endometriosis is associated with an earlier onset and a longer history of painful symptoms until the diagnosis was made when compared with rectovaginal endometriosis locations. These observations support the hypothesis that rectovaginal location may be an intermediate stage of rectal endometriosis. (Fertil Steril Ò 2008;90: Ó2008 by American Society for Reproductive Medicine.) Key Words: Pain, rectovaginal endometriosis, rectal endometriosis, diagnosis The statement No one is born with stage 4 endometriosis! (1), is reasonable, because it means that lower stages of endometriosis, whether or not treated, may develop into advanced stages. This statement appears to be quite obvious to physicians involved in gynecologic care of women presenting with endometriosis. However, there is little evidence concerning the manner of endometriosis development, especially in regard to deep endometriosis (2, 3). It has been proven that endometriosis development is unpredictable, and either aggravation, spontaneous regressions, or an unchanged status could be observed over a period of 6 months (4). However, these observations specifically concern mild stages of endometriosis, and they should not be extrapolated without paying close attention to advanced stages or deep locations. The recent study of Matsuzaki et al. (3) showed that fourth-stage deep endometriosis appears to have an earlier onset when compared with lower stages of the disease, suggesting that a progression might lead from lower to advanced stages. This data may support the option of an active management of endometriosis Received April 13, 2007; revised July 27, 2007; accepted July 30, Reprint requests: Horace Roman, M.D., Department of Gynecology and Obstetrics, Rouen University Hospital, 1 rue de Germont, Rouen, France (FAX : ; horace.roman@gmail.com). regardless of the patient s age to avoid the progression of the pathology (5). Our hypothesis presumes that deep endometriosis located in the rectovaginal space may progress to secondary rectal involvement, and that rectal endometriosis has an initial rectovaginal stage. If this is the case, it may have major consequences on the management of young women presenting with painful rectovaginal endometriosis. One of the consequences may be related to the option to delay surgical treatment because of the patient s age or nulliparity, which may appear risky. To verify this hypothesis, we sought to show that women presenting with rectal endometriosis experienced longer histories of painful symptoms until the diagnosis was made, and that they had an earlier onset of the painful disease when compared with women presenting with mere rectovaginal endometriosis. MATERIALS AND METHODS Women presenting with a suspected painful deep posterior endometriosis from March 2005 to January 2007 in the Department of Gynecology and Obstetrics of University Hospital Rouen, France, were asked to answer a specific questionnaire that recorded the clinical history of the painful disease. This questionnaire encompassed 72 queries and 1014 Fertility and Sterility â Vol. 90, No. 4, October /08/$34.00 Copyright ª2008 American Society for Reproductive Medicine, Published by Elsevier Inc. doi: /j.fertnstert

2 represented a share of the patient s medical folder. It allowed for the precise recording of the length of the painful history, the number of physicians involved in the gynecologic care during the period following the first signs of pain, the treatments previously administered, as well as previous surgical procedures. It also recorded types of pain (dysmenorrhoea, dyspareunia, noncyclic pain, defecation pain) and the age of each onset, the intensity (EVA score, disability related to the daily activities of women), and several consequences of the pain on the social, familial and sexual behaviors. The questionnaire was provided to women during their first visit to our department; it was then filled out at home and reviewed during the following gynecologic visit. At this time the surgical treatment was decided on the basis of clinical, endorectal ultrasound, and magnetic resonance imaging (MRI) examinations. The physician reviewed the answers during the second visit; consequently, the rate of missing answers was negligible. When the patient had a sole preoperative visit, the answers were discussed during the hospitalization period, usually before the surgical procedure. Each patient agreed to the potential use of these data in future clinical retrospective studies. In accordance with French laws, this retrospective study was exempt from institutional review board approval. We conducted a retrospective observational study whose inclusion criteria were the presence of painful rectovaginal or rectal endometriosis determined by both surgical exploration and histologic examination of excised tissues. We excluded women with previous resections of rectovaginal or rectal lesions, but we included those who only had a biopsy. All patients received GnRH analogs 4 to 6 weeks before surgical management. The type of pain (dysmenorrhoea, dyspareunia, noncyclic, and defecation) was recorded using specific questions: Have you ever experienced...? The question asked to gather information regarding age of onset was At what age did you begin to experience...? The age of their first menstruation was recorded using question number 8 ( What was the age of your first menstruation? ), while Q12 ( Was menstruation painful from the beginning? ) and Q13 recorded the age of first dysmenorrhoea ( If the answer to the previous question was no: at what age did your menstruation become painful? ). The dysmenorrhoea was the first type of pain experienced by all women in our series; consequently, the delay of the diagnosis was recorded by the difference in time between Q17 ( What was the length of time, in years, of the interval between your first pain [during menses or not] and the first time that the endometriosis was suspected? ). The suspicion of endometriosis means that a physician associated women s painful complaints to the hypothesis of endometriosis, and consequently, recommended either a treatment (contraceptive pill, progestin, analogs of GnRH, or other therapy) or an examination using ultrasound or MRI. The antiquity of pain was defined as the difference between the study inclusion age, corresponding in most of cases to the diagnosis of deep endometriosis and age of the onset of pain. Pain during adolescence was evaluated by six questions (Q14 16, 21, 22, and 24) that gathered information regarding pain-relief medication intake, the effect of hormonal therapy, and missing school classes because of pain. On the other hand, the questions regarding pain level evaluated by EVA score concerned only the period preceding the visit to our department. The statistical analysis was performed using Stata 9.2 (Stata Corporation, Lakeway Drive, TX.). The test Shapiro Wilk showed that continuous variables (age of first dysmenorrhoea and the length of the interval between the onset of pain and the first time that the endometriosis was suspected) did not have a normal distribution (P¼.02 and P<.001). Consequently, they were represented using median value (10th percentile; 90th percentile), and were compared using the test of Mann Whitney. Box plots graphically represented them. Qualitative variables were compared using chi square or Fischer s exact test. The values of P<.05 were considered as being statistically significant. RESULTS During the period of 22 months, 48 women meeting the inclusion criteria had filled out the questionnaire. In addition, 17 other patients received the questionnaire but were not included in the study. In 13 women, laparoscopic findings excluded the deep endometriosis and showed peritoneal or moderate and severe ovarian endometriosis (nine and two cases, respectively), and adenomyosis (two cases). Two other patients had a fortuitous clinical and MRI diagnosis of posterior deep endometriosis, but were free of pain, and only a gynecologic attending visit was proposed. Ultimately, two patients benefited from the surgical management of rectal endometriosis, but did not provide a complete questionnaire, and thus were excluded from the study. Among the 48 women included in the study, the principal location of endometriosis was rectal in 16 cases (33%) and rectovaginal in 32 cases (67%). One patient had had a biopsy of a rectovaginal nodule performed 5 months previously in another department, followed by 3 months of GnRH therapy, and was included in the cohort of women with rectovaginal disease. Four other patients (two women in each group) had a previous histologic diagnosis of peritoneal or ovarian endometriosis, and had benefited by GnRH treatments during 3 to 6 months after the first surgical procedure, with a transitory relief of painful complaints. The main characteristics of patients are presented in Table 1. The age of onset of dysmenorrhoea was significantly lower in women who developed rectal involvement than in women with mere rectovaginal location. The history of dysmenorrhoea was significantly increased in women with rectal endometriosis (Fig. 1). Neither the history of dyspareunia nor Fertility and Sterility â 1015

3 TABLE 1 Patients characteristics, gynecologic care, and diagnostic circumstances, stratified by principal location of deep endometriosis. Whole sample N [ 48 (100%) Women with rectovaginal endometriosis N [ 32 (67%) Women with rectal endometriosis N [ 16 (33%) Age (years) a 34.5 [24;43] 33.5 [23;41] 35.5 [27;43] Nulliparas 31 (60) 21 (66) 10 (62) Dysmenorrhoea 46 (96) 30 (94) 16 (100) Primary 28 (58) 16 (50) 12 (75) Secondary 20 (42) 16 (50) 4 (25) Age of the onset (years) a 14 [11;35] 14 [12;32] d 13 [11;18] d Antiquity of dysmenorrhoea (years) a 16 [4;30] 11 [3;26] e 20 [14;31] e Dysmenorrhoea in adolescence At least 1 day of missing school classes 22 (46) 14 (44) 8 (50) due to dysmenorrhoea Regular intake of medication for 31 (66) 19 (59) 12 (75) dysmenorrhoea Sexual intercourse b 44 (92) 30 (94) 14 (88) Dyspareunia c 37 (84) 26 (87) 11 (76) Age of onset (years) a 26 [10;33] 25 [17;32] 28 [20;33] Antiquity of dyspareunia (years) a 5 [1;17] 5 [1;16] 7 [2;17] Defecation pain 36 (75) 21 (66) f 15 (94) f Antiquity of defecation pain (years) a 6 [1;20] 6 [2;20] 7 [1;17] Time from the onset of pain until the first 10 [1;22] 9 [1;20] e 16 [1;26] e time that the endometriosis was suspected (years) Number of physicians whose advice was 3 [1;7] 3 [1;7] 3.5 [2;6] considered before the diagnosis was suspected Gynecologists 2 [1;4] 1 [1;4] 2 [1;4] General physicians 1 [0;3] 1 [0;3] 1 [0;3] Other specialists 0 [0;1] 0 [0;1] 0 [0;2] Endometriosis suspected by 13 (28) 8 (26) 5 (31) a nongynecologist physician Pill intake to relive the cyclic pain 19 (40) 11 (34) 8 (50) Pain relief by pill 16/34 (47) 10/21 (48) 6/13 (46) Pain increases after the end of pill intake 24/33 (73) 16/22 (73) 8/11 (73) a Median [10th percentile; 90th percentile]. b Women having previously had at least one sexual intercourse. c Dyspareunia rate concerns only women having had sexual intercourses. d P¼.05. e P¼.01. f P¼.03. Roman. Rectal and rectovaginal endometriosis. Fertil Steril that of defecation pain presented a significant association with endometriosis location. The time interval from the onset of the first pain to the first time that the endometriosis was suspected was significantly greater in women presenting with rectal endometriosis, even despite several medical visits (Table 1, Fig. 1). A nongynecologist physician performed the diagnosis in 26% of rectovaginal locations and in 31% of cases with rectal involvement. To relieve the pain, the contraceptive pill was recommended in <50% of adolescents, whereas stopping the hormonal therapy was followed by an increase in the pain in three women out of four Roman et al. Rectal and rectovaginal endometriosis Vol. 90, No. 4, October 2008

4 DISCUSSION The principal finding of our study was that there is an increase in the length of time of pain history in patients presenting with rectal endometriosis when compared with those presenting with rectovaginal location. The earlier onset of pain may correspond to a more aggressive disease, whereas the increased length in time from onset of pain to both diagnosis of endometriosis and surgical management may lead us to believe that rectal endometriosis is merely an advanced stage of the rectovaginal endometriosis. The aim of this study was not to focus on the circumstances of the delayed diagnosis. However, we found that the advice of three different physicians was required to suspect the endometriosis in young women presenting cyclic pain. Furthermore, a nongynecologist physician in more than one out of four women diagnosed deep endometriosis. This finding is in agreement with other studies that showed a significant delay in the diagnosis of endometriosis (3, 6 10), much greater when women present with painful symptoms than when they experience infertility (7, 9). Our study does not provide a control group to include women with mild to moderate endometriosis. In fact, in our department, the questionnaire was specifically made for and used only with women presenting deep endometriosis. However, we may roughly compare our results to those of other series that included women with less advanced stages of the disease. In a similar study, Arrunda et al. (7) reported a series of 200 women with endometriosis with a stages ranging from I to IV, whose median age at onset of symptoms was 20.5 years, much higher than that of our population. This suggests that earlier onsets of pain increase the likelihood of progression of endometriosis to more severe stages. One may find that there exists a long delay in the diagnosis in our series. It must be underlined that in most of cases, the history of pain experienced by our patients began during adolescence and, in this way our results are comparable to those of other investigators. Matsuzaki et al. (3) showed that in patients presenting with deep endometriosis, the delay of diagnosis averaged 15.2 years if the onset of pain was inferior to 20 years of age. Similarly, Arrunda et al. (7) found that younger women with pelvic pain because of endometriosis took a significantly longer time to report their symptoms to a doctor (2 years), and they also had to a longer period until a definitive diagnosis was made (12 years). Patients whose onset of symptoms began before the age of 15, consulted an average of 4.2 doctors (5). Otherwise, physicians are probably more likely to pay less attention to dysmenorrhoea in adolescents and will avoid performing thorough vaginal examination or recommending MRI examinations that may cause increased anxiety in these patients. Furthermore, vaginal examination may be difficult or impossible to perform in young women who have not had vaginal intercourse (5), and this fact may, without doubt, contribute to the delay of diagnosis. The rectal examination may detect a deep endometriosis (5), but it is often avoided because of its inconvenient nature. Data used in the analysis was provided by a specific questionnaire; however, we cannot guarantee the accuracy of the answers, especially in regard to events occurring in adolescence. We tried to avoid rough timing errors by associating the pain to the school or professional activity ( Did you miss courses? Did you miss an exam? ). Although all errors may not be completely avoided, it is less likely that they significantly modified our results. FIGURE 1 Length of time from the onset of first pain to the first time that the endometriosis was suspected (left) and to the inclusion in the study (right) box plot procedure. Interval from first pain to diagnosis (years) Duration of dysmenorrhoea (years) Rectovaginal Rectal Rectovaginal Rectal Roman. Rectal and rectovaginal endometriosis. Fertil Steril Fertility and Sterility â 1017

5 Because the design of our study is observational, we cannot come to a formal conclusion that women presenting with mere rectovaginal endometriosis will develop rectal involvement when the diagnosis is not performed in time. It is possible for an unknown rate of rectovaginal locations to remain static, improve, or progress laterally, without any rectal involvement. Our department is a tertiary care center, and the observed ratio 2:1 between rectovaginal and rectal locations is likely to differ from that of the general population. An interesting study by Fedele et al. (11) recently showed that <10% women presenting pain-free rectovaginal endometriosis are likely to experience a progression of their disease. Thus, it is probable that the pain related to rectovaginal nodule is a sign of an active disease and should be taken into account. Otherwise, on the bases of the conclusions of Fedele et al. (11), we did not propose surgical management in two women referred to us for a fortuitous diagnosis of rectovaginal endometriosis (excluded from the study), and we only recommended regular gynecologic care. Determining accurately which adolescent is at higher risk to develop deep endometriosis may be challenging. Contrarily, carrying out the diagnosis of deep posterior endometriosis before the progression toward rectal involvement should be a reasonable public health objective. This could be accomplished by adding careful queries to adolescent examinations regarding the characteristics of the cyclic pelvic pain, dyspareunia, and defecation pain, and by thorough vaginal examinations. Studies like ours should lead to two main conclusions. The first conclusion relates to the necessity and improvement of early endometriosis diagnosis in adolescents to avoid the occurrence of deep localizations. There is no evidence supporting the hypothesis that the early diagnosis of the endometriosis may decrease the risk of progressions toward deep forms, and it will not be available in the near future. However, it has been shown that endometriosis progresses by cyclic bleedings (12), sever stages are preceded by less advanced stages (1), and the length of the disease without adequate treatment is related to its severity (3, 6, 7). These arguments are strong enough to induce the choice to treat this pathology earlier an adolescence (5). The second conclusion is that painful deep endometriosis forms may sometimes progress toward rectal infiltrations. This statement is in agreement with that of Koninckx et al. (2), who showed that the depth of the endometriosis invasion increased with age. In the literature, there are several retrospective studies of patients who benefited from surgical treatment of both rectovaginal and rectal endometriosis. Despite the lack of randomized studies, it is obvious that rectovaginal and rectal surgical resections are not alike with regard to the difficulty of the procedure and postoperative outcomes. Whenever the rectal resection is required, the rate of postoperative complications inevitably increases (13, 14). Furthermore, even when postoperative complications do not occur, women may be temporarily embarrassed by unpleasant postoperative symptoms related to segmental rectal resection, such as an increased frequency in stools or constipation, difficult defecation, or dysuria (13, 14). These observations support the option of surgical removal of painful rectovaginal endometriosis to avoid the eventual progression toward rectal forms. Acknowledgments: We gratefully acknowledge the reviewers of this manuscript for their pertinent and useful remarks, which allowed the improvement of our article. REFERENCES 1. Donnez J, Pirard C, Smets M, Jadoul P, Squifflet J. 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