Persistent sexual arousal syndrome

The concept of a sexual pain disorder is both intriguing and confusing. It is intriguing because the basic premise underlying this idea appears to be that there is a special type of pain which is sexual in nature. If this premise is true, then the study of sexual pain that becomes pleasurable or sexual pleasure that becomes painful might provide us with crucial information concerning the relationship of pain and pleasure. The nature of this relationship has intrigued philosophers, psychologists, and neuroscientists for many generations. On the other hand, the sexual pain concept is confusing because acceptance of the notion of sexual pain implies that there may be numerous other special types of pain, such as work pain and sports pain. This seems unlikely and raises doubt about whether there is a special type of sexual pain. The term, sexual pain, is also confusing because it is vague and can be understood in at least two distinct ways. Does it mean pain induced by sexual activities, thoughts, and feelings, or does it mean sexual pleasure induced by painful activities, thoughts, or feelings?

Current psychiatric thinking seems to have opted for the former. In DSM III-R (American Psychiatric Association, 1987) the concept of sexual pain disorder was introduced to encompass vaginismus and dyspareunia; this category was retained in the DSM IV (American Psychiatric Association, 1994). The sexual pain disorders are the only pain problems in the DSM IV outside of "pain disorder." This appears to reflect the idea that there is a special type of pain associated with sexual activities and that this pain is different from other types of pain. Our alternative view is that there is little that is uniquely sexual about vaginismus and dyspareunia other than the activity which typically induces the pain. These opposing views reflect the central issue in this field and our title: "Is the pain sexual or is the sex painful?"

Although we will be critically examining the concept of sexual pain disorder, we will retain the use of the term in this article in order to maintain consistency with other literature. Because little is known about this problem (cf. Wesselmann, Burnett, & Heinberg, 1997, for a review), sexual pain in men will not be discussed.

Background

Medical descriptions of pain associated with sexual intercourse are not new. The first mention of dyspareunia probably dates back at least 3,500 years to the Ramesseum papyri of ancient Egypt (Barns, 1956; Costa Talens & Colorado Vicente, 1971). This fragmentary papyrus appears to link dyspareunia to abnormal menstruation and reads as follows: "A woman who has pain in her KNS [probably vulva] and has pain in her coition, and things do not stop [descending from it]." Our first record of vaginismus may appear in the writings of Trotula of Salerno (di Ruggiero) (1547/1940), a female physician who lived in Salerno during the 11th century AD. Chapter 25 of her work, The Diseases of Women is entitled "On the manner of tightening of the vulva so that even a woman who has been seduced may appear a virgin."

Although there are numerous descriptions of vaginismus and dyspareunia in gynecology texts of the 19th or early 20th century, there is no published historical work detailing the history of the development of the concept of sexual pain. It appears that by the early 20th century the basic clinical phenomena were fairly well described. For example, many of the standard "gynecology" texts of the 19th century included descriptions such as "hyperaesthesia of the vulva" (Thomas, 1880) or "sensitive points about the mouth of the vagina" which rendered intercourse painful (Kellogg, 1889). These descriptions appear quite similar to a type of dyspareunia which is called vulvar vestibulitis syndrome today (cf. below). Deep dyspareunia is described by Byford (1902, p. 576) in the following way: "Hyperesthesia of the pelvic organs consists of an abnormal sensitiveness of the pelvic viscera, lower abdomen and sometimes of the tissues about the coccyx." Vaginismus, on the other hand, is typically described as resulting from muscular contractions or spasms accompanied by heightened physiological arousal. Sims (1861) provided us with the following classic description:

But the most remarkable thing in her history was the fact that she had remained a virgin notwithstanding a married state of a quarter of a century ... Amongst other investigations of her case, I attempted to make a vaginal examination but failed completely. The slightest touch at the mouth of the vagina producing most intense suffering. Her nervous system was thrown into great commotion; there was a general muscular agitation; her whole frame was shivering as if with the rigors of an intermittent [sic]. She shrieked aloud, her eyes glaring wildly, while tears rolled down her cheeks and she presented the most pitiable appearance of terror and agony. Notwithstanding all these outward involuntary evidences of physical suffering, she had the moral fortitude to hold herself on the couch, and implored me not to desist from any efforts if there was the least hope of finding out anything about her inexplicable condition. After pressing with all my strength for some minutes, I succeeded in introducing the index finger into the vagina up to the second joint, but not further. The resistance to its passage was so great, and the vaginal contraction so firm, as to deaden the sensation of the finger, and thus the examination revealed only an insuperable spasm of the sphincter vaginae. (pp. 358-359)

Although it is clear from these descriptions that vaginismus and dyspareunia interfere with intercourse, the writers do not attribute a sexual etiology to the problem nor do they describe the problem as a sexual one. Many texts mention possible "emotional" or "temperamental" causes (e.g., Byford, 1902; Thorburn, 1885), but the idea of a sexual pain or a sexual etiology for these problems was not common. This appears to change sometime in the middle of the 20th century when gynecology texts begin to conceptualize and classify vaginismus and dyspareunia as sexual problems. For example, Greenhill (1940, p. 103) writes the following: "In most cases of dyspareunia there is a large psychic factor. Nearly always the patient abhors sexual intercourse, and because of this, involuntarily makes an effort to keep out the penis." Our hypothesis, as amateur historians, is that this conceptual shift occurred as a result of the Freudian revolution. Although the influence of psychodynamic theories of sexual pain has waned, the conceptualization of vaginismus and dyspareunia as a sexual problem has been preserved.

Early gynecological writings also foreshadow many other current issues. For example, one German physician questions the centrality of reflexive vaginal spasm as the defining characteristic of vaginismus (Walthard, 1909). Although 19th century gynecologists had separate terms for vaginismus and dyspareunia, they pointed out there was significant symptomatic overlap and that the two problems often coexisted (e.g., Thorburn, 1885). Several describe or imply that there are different dyspareunic syndromes which may be unrelated (e.g., Thorburn, 1885). Others suggest that vaginal dilation alone is not an adequate treatment for vaginismus but must be accompanied with other forms of medical and psychosocial intervention (Martin, 1890; Walthard, 1909).

Description and Classification

In the 20th century there have been numerous attempts to formally describe or classify vaginismus and dyspareunia (cf. Bergeron, Binik, Khalifa, & Pagidas, 1997; Meana & Binik, 1994; Reissing, Binik, & Khalife, 1999). Currently, the term vaginismus appears in the DSM IV (American Psychiatric Association, 1994) the International Statistical Classification of Diseases and Related Health problems-ICD-10 (World Health Organization, 1992), the American College of Obstetricians and Gynecologists recommendations (ACOG Technical Bulletin, 1995), and the Classification of Chronic Pain (Merskey & Bogduk, 1994). The first three nosologies are quite similar in that they are categorical systems that conceptualize vaginismus as a sexual dysfunction defined by vaginal muscle spasm. The Classification of Chronic Pain nosology (Merskey & Bogduk, 1994) lists vaginismus as a genitourinary pain syndrome but does not provide any additional information.

Because vaginal muscle spasm is the defining diagnostic characteristic of vaginismus for the most influential current nosologies, it is crucial to examine critically what is meant by the term muscle spasm. Vaginismus is formally defined by DSM IV as a "recurrent and persistent involuntary spasm of the musculature of the outer third of the vagina that interferes with sexual intercourse" (p. 515). This definition is problematic in numerous ways. The most basic problem is that no one has ever reliably demonstrated the existence of such a spasm. This is remarkable considering the 150 year history of such reports. It is also remarkable that vaginismus is never mentioned in the vast literature concerning muscle activity, spasms, and cramps (e.g., Jansen, Joosten, & Vingerhoets, 1990; McGee, 1990). Moreover, from an anatomical point of view, it is not clear which muscles make up the "outer third of the vagina." Virtually every pelvic muscle and most adjoining muscle groups, including the adductors of the thighs, have been included in clinical descriptions of those muscles which spasm (cf. Reissing, Binik, & Khalifa, 1999). Finally, recent empirical attempts to demonstrate such spasms have not been successful. For example, Van der Velde and Everaerd (1996, 1999) were not able to demonstrate that surface electromyography (sEMG) measured pelvic muscular tension differences between vaginismic women and normal controls. In an ongoing study, Reissing, Binik, Khalife, and Cohen (1999) are examining the reliability of the muscular spasm defined diagnosis of vaginismus based on gynecological and pelvic floor physical therapist examinations, sEMG, and interview data. Preliminary results suggest that muscular spasms are not prevalent and that the levels of diagnostic reliability may not be sufficiently high for clinical practice or research.

Another important descriptive/classificatory issue is that the DSM IV diagnosis does not require the experience of pain even though vaginismus is classified as a sexual pain disorder. This classification strategy seems illogical but is presumably based on clinical reports from vaginismic women who report no pain. Unfortunately, there are no quantitative empirical studies of pain in vaginismic women, making it difficult to estimate what percentage of these women actually experience pain, The result is a diagnosis based on an as of yet unverified criterion of vaginal spasm which may or may not cause pain. This raises the question of what actually prevents intercourse for vaginismic women. Even in the presence of spasm and pain, a stoic woman or an insensitive partner should be able to achieve penetration. Thus, we currently have a circular definition based on presumed spasm, possible pain, and failure to achieve intercourse.

All of the current diagnostic systems differentiate between vaginismus and dyspareunia. Their specific approaches to dyspareunia, however, are somewhat different. In the gynecological literature, dyspareunia is seen more as a symptom of underlying physical or, in some cases, psychological pathology than as a separate diagnostic category (cf. Steege, Metzger, & Levy, 1998). The ICD-10 has two different diagnostic categories, one for organic and one for psychogenic dyspareunia. Neither category is explicitly defined. The Classification of Chronic Pain (Merskey & Bogduk, 1994) simply lists dyspareunia in its classification of genitourinary pain syndromes without additional information. Dyspareunia is currently defined in the DSM IV as "recurrent or persistent genital pain associated with sexual intercourse in either a male or a female" (P. 513). Diagnostic exclusion criteria include lack of lubrication and vaginismus. This definition is striking in that dyspareunia may be the only pain defined by the activity with which it interferes. As a result, there is no suggestion in the DSM IV that there is an important diagnostic difference between vulvar pain experienced during attempted penile penetration and deeper pain experienced during thrusting.

In addition, there is no justification given as to why lack of lubrication is an exclusion criterion. We presume that the DSM IV authors were attempting to exclude dyspareunic postmenopausal women experiencing decreased lubrication resulting from reduced estrogen levels associated with vulvo/vaginal atrophy. Although it appears to be relatively well established that reduced estrogen can result in vulvo/vaginal atrophy and decreased lubrication, there is no empirical evidence that these factors are closely associated with reports of pain. In fact, Laan and van Lunsen (1997) have shown that vaginal atrophy is related to estrogen levels but not to vaginal dryness and dyspareunia. Lack of lubrication may also not be a useful exclusion criterion for younger women. In a recent study with premenopausal women Wouda et al. (1998) suggested that reduced arousal or lack of lubrication may not differentiate dyspareunic women from controls. Finally, there is no reason to assume that vaginismus and dyspareunia cannot co-exist. In fact, there are now numerous clinical reports suggesting there is a significant rate of co-morbidity (e.g., Basson, 1994; Van Lankveld, Brewaeys, Ter Kuile, & Weijenborg, 1995).

The current definitions of dyspareunia and vaginismus also reflect many of the nosological difficulties shared by almost all classifications of sexual dysfunction. How is recurrent and persistent to be defined? Is it possible or useful to differentiate between psychological and organic etiologies? When a women has experienced a month of pain-free intercourse followed by years of pain is this to be classified as lifelong or acquired? The most basic problem, however, is that the classification of dyspareunia as a sexual dysfunction appears to have discouraged the study of its major symptom-pain.

Meana, Binik, Khalife, and Cohen (1997b) were the first to investigate the nosology of the dyspareunia from a pain research perspective. In their study, they examined 112 women with chronic coital pain, using a structured gynecological and psychosocial protocol. The gynecological protocol included a manual/visual pelvic examination, a vaginal ultrasound, and a colposcopy. The psychosocial protocol included the McGill/Melzack pain questionnaire (Melzack, 1975; Melzack & Katz, 1992), a structured pain interview, and several other psychometric instruments examining sexual and psychological functioning. Meana et al. (1997b) were able to demonstrate that the pain of dyspareunia was measurable quantitatively and qualitatively and was comparable in intensity to pain experienced in pain syndromes such as phantom limb pain, back pain, or arthritis (Meana, 1996). Using discriminant function analyses, Meana et al. (1997b) were able to describe four different possible subgroups of dyspareunia. The first two were identified as vulvar vestibulitis and vulvo/vaginal atrophy. A third group of women experienced recurrent acute pain during intercourse, which was not easily characterized by either psychosocial or gynecological markers. The fourth group included women with a variety of known gynecological problems or pathologies (e.g., pelvic adhesions, endometriosis, fibroids, cysts, etc.), which seemed closely linked to the dyspareunia. The location and temporal pattern of the pain were found to be the major predictors of diagnostic subtypes of dyspareunia. These factors are commonly used in pain classifications (cf. Merskey & Bogduk, 1994) and differ from DSM taxa (e.g., lifelong/acquired or generalized/situational), which were not statistically useful in these analyses.

Bergeron (1999) has continued to investigate dyspareunic pain by focussing on the vulvar vestibulitis subtype. Although this type of coital pain was probably first described over a century ago, it has only recently become known to gynecologists and sexologists (Bergeron, Binik, et al., 1997). Friedrich (1988) proposed the following criteria to diagnose vulvar vestibulitis syndrome (VVS): (a) severe pain on vestibular touch or attempted vaginal entry, (b) tenderness to pressure localized within the vulvar vestibule, and (c) physical findings confined to vestibular erythema. In typical gynecological practice, VVS is diagnosed by the "cotton swab test" in which the vulvar vestibule is lightly palpated using a Q tip or similar object. Women with vulvar vestibulitis report a burning or incisive pain during this examination, during intercourse, and in response to activities (e.g., sports) affecting this area. The pain is highly localized to the vulvar vestibule with the result that similar palpation of surrounding structures elicits no pain. Bergeron et al. (1999b) has demonstrated that gynecologists can reliably diagnose vulvar vestibulitis syndrome through the use of the cotton swab test and quantitative patient reports of pain.

Overall, these studies have strongly suggested that our current diagnostic conceptualizations of vaginismus and dyspareunia as a sexual dysfunction may not be adequate to deal with the complexity of the phenomena. Our current view is that we should attempt to classify the sexual pain disorders in the same way that we conceptualize headache or low back pain. In this view, there are a number of vulvo/vaginal/pelvic pain syndromes resulting in pain during intercourse and other activities. These different syndromes may not be closely related and will probably have different diagnostic characteristics, etiologies, and treatments. Vulvar vestibulitis appears to be such a syndrome. The defining characteristics of such subtypes of dyspareunia are likely to involve quantitative and qualitative aspects of the pain, not the activity (sexual intercourse) with which the pain interferes. It is not currently clear whether heightened chronic or acute pelvic muscle tension or spasm characterizes vaginismus or differentiates it from other forms of coital pain. We suspect that the role of pelvic muscle tension in vaginismus may be similar to the role of the pericranial muscles in tension headache or the role of the external oblique muscle in kidney stone induced hyperalgesia (i.e., an important symptom but not a defining characteristic; see more below, Giamberardino & Vecchiet, 1996; Simons & Mense, 1998).

Prevalence

The literature concerning the sexual pain disorders is filled with widely differing prevalence estimates. Some researchers have concluded that vaginismus is commonly underreported or overlooked, whereas others have reached the opposite conclusion (Reissing, Binik, & Khalifa, 1999). Prevalence estimates for dyspareunia have ranged from a low of 3% to over 50% (cf. Meana & Binik, 1994). The variance in these estimates probably reflects the poor methodological standards of many of these studies in addition to the widely differing populations that were sampled. In practice, it would be very difficult to carry out epidemiological surveys of vaginismus or vulvar vestibulitis syndrome because a gynecological examination is necessary to make the diagnosis.

The best prevalence estimates we currently have are from recent large-scale epidemiological studies in which the primary goal was to investigate sexually transmitted disease. Two of these studies, one carried out in France and one in the U.S., provided information relevant for our purposes by including a question asking respondents to report about the experience of pain during intercourse. The French study (Spira, Bajos, & Le Groupe ACSF, 1993) used a telephone interview methodology in which 20,055 respondents (11,104 women) between the ages of 18-69 were randomly chosen from the list of French residential telephone numbers for a brief telephone interview focussing on demographics and risk factors for sexually transmitted disease. From this group, 5,000 respondents were selected for a longer (30 minute) telephone interview which also asked additional questions about relationships and sexual practices. Three thousand of the participants for the "long interview" were chosen on the basis of being "high risk" for sexually transmitted disease; the remaining 2000 "controls" were chosen on the basis of a birth date representative of the original sample. Four thousand eight hundred twenty respondents (2,178 women) completed the long interview and were asked the following question: Have you ever experienced the following symptoms? (often, some of the time, very infrequently, never, no response) 1. You have painful intercourse; 2. You have an absence of or insufficient sexual desire; 3. You have an excess of sexual desire; and 4. You don't have orgasm (authors' translation). Five percent of the sexually active respondents indicated that they had often experienced pain during sexual relations. An additional 19% reported pain some of the time. There were no further reported analyses of this question.

The U.S. study (Laumann, Gagnon, Michael, & Michaels, 1994) was carried out at approximately the same time using a mixed personal interview/questionnaire methodology. In this study, a national probability sample of 1,511 men and 1,921 women between the ages of 18-59 living in U.S. households were interviewed for 90 minutes in their homes. All interviewees were asked the following question: "Sometimes people go through periods in which they are not interested in sex or are having trouble achieving sexual gratification .... During the past 12 months, has there been a period of several months or more when you. . ." Respondents were asked to circle yes or no next to each of the following items: (1) lacking desire for sex; (2) arousal difficulties; (3) inability to achieve climax or ejaculation; (4) climaxing or ejaculating too rapidly; (5) physical pain during intercourse; (6) not finding sex pleasurable; and (7) anxiety about sexual performance. Items 4, 5, and 6, including pain during intercourse, were only asked of those respondents sexually active during the last 12 months, whereas the other items were asked of everybody. Overall, 15% of women responded yes to the item concerning pain during intercourse.

The authors of this study have recently reported on numerous correlates of the prevalence findings related to sexual dysfunction (Laumann, Paik, & Rosen, 1999). With respect to sexual pain disorder there was a significant negative correlation with age. Women in the 18-29 age range were three times more likely to report pain during intercourse than those in the 50-59 year old range. Similarly women in poor health are about three and a third times more likely to be afflicted with sexual pain. Additional correlates included lower educational attainment, decreased family income, increased stress/more frequent emotional problems, and the presence of urinary tract symptoms. On the other hand, the presence of sexual pain was not significantly related to the experience of sexual trauma or abuse, race, marital status, religion, alcohol consumption, ever having an STD, number of lifetime partners, sexual frequency, masturbation rates, and same-sex sexual activity. Finally, experiencing pain during intercourse was significantly related to decreased quality of life reports, including reduced general happiness and lowered satisfaction with the emotional and physical aspects of one's relationships.

The large discrepancy between the 5% and 15% prevalence rates reported in the French and U.S. studies is likely to result, in part, from different data gathering techniques, different questions and response options, and different sampling techniques (Gribble, Miller, Rogers, & Turner, 1999). This discrepancy may also reflect cultural differences between the U.S. and France. Unfortunately, there is no way to determine the "true" prevalence of sexual pain disorders from available data. Nonetheless, even the use of the conservative 5% figure suggests that the report of pain during intercourse is a very frequent women's health problem.

Etiology

Physiological Mechanisms

Sensory innervation of the female reproductive organs. The innervation of the vulva, vagina, cervix, uterus, oviduct, and ovaries is supplied by the pudendal, pelvic, and hypogastric nerves. However, the innervation characteristics of the different regions differ considerably (Berkley & Hubscher, 1995a; Bonica, 1994; Papka & Traurig, 1993; Wesselmann et al., 1997). The vulva is innervated solely and densely by branches of the pudendal nerve (ilioinguinal and genitofemoral), which are somatic nerves that contain mostly heavily myelinated nerve fibers along with some thinly myelinated (AS) and unmyelinated (C) fibers. The heavily myelinated fibers are continually active and very rapidly convey precise information about gentle and intense mechanical stimulation of the vulvar regions to sacral segments (S2-S4) of the spinal cord. The vagina is innervated solely by the pelvic nerve, which is mainly a parasympathetic nerve that contains mostly C and some AS fibers. A substantive proportion of C-fibers are often silent but can be activated by either gentle or intense mechanical and chemical stimulation to convey this information more slowly than the pudendal afferents to the sacral spinal cord. The cervix and adjacent fornix region of the vagina is innervated more densely than the rest of the vagina by AS and C-fibers in both the pelvic splanchnic nerve (parasympathetic) and hypogastric (sympathetic) nerves. The C-fibers, like those that supply the vagina, are often silent but can be activated by either gentle or intense mechanical and chemical stimulation to convey the information to sacral (via pelvic nerve) segments, as well as to lower thoracic/upper lumbar (via hypogastric nerve) segments (TIO-L2) of the spinal cord.

The body of the uterus is supplied mainly by C-fibers (often silent) and some A8 in the hypogastric nerve, sending input mainly to T10-L2 spinal segments. Less is known about the innervation of the oviduct and ovary, but they appear to be innervated mainly by C (often silent) and some A8 fibers in two branches of the superior hypogastric plexus (ovarian plexus nerve and superior ovarian nerves) traveling via hypogastric nerve to lower thoracic segments. These two regions are also probably innervated by branches of the vagus nerve, the input being sent to the solitary nucleus of the medulla. Of importance may be recent evidence supporting the fact that the vagus may actually innervate all components of all pelvic organs (Cueva-Rolon et al., 1996; Komisaruk, Gerdes, & Whipple., 1997).

Processing of information from the female reproductive organs within the Central Nervous System (CNS). Information from the vulva, vagina, and cervix arrives in the sacral spinal cord by way of afferents in the pudendal and pelvic nerves, with additional input from the cervix via the hypogastric nerve, which conveys information as well from the rest of the uterus, the oviduct, and ovaries to the upper lumbar spinal cord. However, in patterns that are not well understood, animal research has shown that the spinal neurons that receive this information also receive converging input from afferent fibers that supply skin and muscles of the lower limbs, back, abdomen, and perineum, as well as fibers that supply other internal pelvic organs such as portions of the urinary and lower GI tracts (Berkley, Hubscher, & Wall, 1993). These recipient spinal neurons are also subject to influences originating from neurons located in other segments of the spinal cord, as well as from neurons in widespread areas throughout the brain (Watkins, Faris, Komisaruk, & Mayer, 1984; Wall, Hubscher, & Berkley, 1993). Furthermore, the spinal neurons receiving input from reproductive tract structures can convey their modified information to widespread regions throughout the spinal cord as well as to numerous targets in the brain, including (among other regions) the dorsal column and the solitary nuclei of the medulla, the parabrachial nucleus and central grey of the pons and midbrain, and the intralaminar and ventroposterior nuclei of the thalamus (Berkley, Guilbaud, Benoit, & Gautron, 1993; Berkley & Hubscher, 1995b; Komisaruk & Wallman, 1977). Each of these target regions is, in turn, subject to modifying influences and conveys the modified information to many other targets throughout the CNS. Added to this picture is additional input directly to the solitary nucleus of the medulla from vaginocervix sensory afferents that travel in the vagus nerve. The implications of this newly discovered innervation are only just beginning to be studied, but it is clear they contribute significantly to some of the analgesic and autonomic effects of vaginal stimulation (Cueva-Rolon et al., 1996).

Although the details of this complex situation remain poorly understood (reviewed in Berkley, 1997; Berkley & Hubscher, 1995b; and Vahle-Hinz, Bruggemann, & Kiniffki, 1995), its general features are of considerable significance to considerations of pain associated with sexual intercourse. First, the converging influence on CNS neurons of input from the vulva/vagina/cervix and input from perineal skin and muscle afferents is a likely substrate for referred pain and referred hyperalgesia (tenderness) of muscles (and sometimes skin) that can be induced by intense stimulation of internal organs (Giamberardino, Berkley, lezzi, de Bigontina, & Vecchiet, 1997; Giamberardino & Vecchiet, 1996). Second, the fact that information arriving from the vulva, vagina, and cervix is conveyed to widespread regions of the CNS implies that stimulation of these regions, such as that occurring during sexual intercourse, can affect a wide range of physiological and perceptual functions (see Komisaruk & Whipple, 1998). Third, the fact that the information arriving in the spinal cord is subject to complex modification from widespread portions of the entire neuroaxis immediately upon its entry, as well as within rostral targets in the brain, implies that a neural substrate exists for perceptual consequences of such stimulation that is highly vulnerable to modification.

Plasticity of information processing in the periphery and within the CNS. It has become clear in recent years that intense stimulation of peripheral tissues such as that due to injury or the threat of injury can give rise to changes within both the peripheral and CNS that long outlast the initial events. In the periphery, such stimulation can sensitize the silent afferent fibers, (i.e., the C-fibers), so that they continue to send information to the spinal cord even in the absence of stimulation or with much less stimulation than was necessary previously (McMahon & Koltzenburg, 1990). Within the spinal cord and perhaps elsewhere in the CNS, such stimulation can sensitize the neurons that originally received the information so that in the future they are more likely to be activated by previously subthreshold influences from any source, a phenomenon known as central sensitization (Coderre, Katz, Vaccarino, & Melzack, 1993; McMahon, Lewin, & Wall, 1993). In addition, evidence is accumulating that the vulnerability to such sensitizing influences on both peripheral afferent fibers and central neurons may depend on the natural plasticity of neurons such as those associated with stress and reproductive status (McEwen, Alves, Bulloch, & Weiland,1998).

This conceptualization is consistent with observations that dyspareunia and particularly vulvar vestibulitis are associated with a history of vulvo/vaginal infection (particularly candidiasis) and early contraceptive use (cf. Bergeron, Binik, Khalifa, & Pagidas, 1997; Bergeron, Binik, & Khalifa, 1999a, 1999b for reviews). These events may initiate a process of central sensitization which becomes a major pain mechanism long after the infection has disappeared. The conflicting (cf. Foster & Hasday, 1997; Nylander Lundqvist, Hofer, Olofsson, & Sj6berg, 1997) reports of nonspecific vestibular inflammation in vulvar vestibulitis are also consistent with this view. Central sensitization initiated by even one damaging vaginal penetrative incident could also underlie vaginismus. Vaginismus would then be conceptualized as a form of long-term referred hyperalgesia in perineal muscles. Such long-term referred hyperalgesia has been demonstrated to continue in flank muscles as long as 1 year following passage of a single kidney stone (Giamberardino & Vecchiet, 1996).

The complex situation described above, together with these plastic features of neural processing, indicate the huge potential that exists for events occurring in the reproductive organs to have long-lasting consequences on neural function and on resultant perception in ways that may depend considerably upon both past history and current circumstances, Suc plasticity also, however, that what has been done can in turn be modified.

Psychosocial Mechanisms

A review of the psychosocial mechanisms implicated in the sexual pain disorders is necessarily speculative, as little methodologically sound etiologic research has been conducted (Bergeron, Binik, et al., 1997; Meana & Binik, 1994; Reissing, Binik, & Khalifa, 1999). The empirical research that does exist has been in the form of either case or correlational studies, precluding cause and effect conclusions. Furthermore, considering the increasing empirical support for the multidimensional causality of chronic pain in general, it is unlikely that further investigation will yield simple linear relationships between psychosocial factors and sexual pain. It is, thus, impossible now and unlikely in the near future that we will be able to distinguish among psychosocial etiologic factors (factors that initiate the pain), mediating factors (factors that influence the experience of pain), or effects (factors that result from the pain). Nonetheless, the existing literature can be usefully summarized under the categories of affective, cognitive, and social mechanisms.

Affective. The most well-developed early etiological conceptualizations of dyspareunia and vaginismus are found in the psychoanalytic literature. Within this theoretical perspective, dyspareunia and vaginismus were generally considered to be hysterical or conversion symptoms symbolizing an unconscious intrapsychic conflict (Fenichel, 1945; Kaplan, 1974). With only a handful of case studies, the psychoanalytically oriented literature interpreted the sexual pain disorders to be the result of phobic reactions, major anxiety conflicts, hostility, or aversion to sexuality, often linked to abuse or trauma in infancy. The emphasis was clearly on the unconscious nature of the conflicts. Although this aspect of the conceptualization has not survived the scrutiny of more controlled research, this early invocation of fear and anxiety as important affective factors seems not to have been completely misplaced.

With the behavioral revolution of the mid-20th century, classical conditioning replaced unconscious conflict as the proposed mechanism by which the fear and anxiety of traumatic events such as abuse or assault became associated with sexuality (Lazarus, 1989). It was hypothesized that this conditioned anxiety/fear response interfered with both subjective and physiological arousal (e.g., lubrication), creating conditions that could make penetrative sex painful. Although this is a reasonable hypothesis, there is no empirical support for the classical conditioning mechanism. Moreover, two controlled studies (Laumann et al.,1999; Meana, Binik, Khalifa, & Cohen, 1997a) failed to find any association between abuse or trauma and sexual pain.

Independent of the hypothesized mechanism or causal direction, anxiety and fear have been extensively investigated with respect to different types of pain (Gamsa, 1994). Although some studies have found anxiety to be related to pain ratings (Litt, 1996; Velikova, Selby, Snaith, & Kirby 1995), the majority of the studies find a relationship between anxiety and pain behaviors and function rather than pain intensity (Arntz & deJong, 1993; Asmundson & Taylor, 1996; Biederman & Schefft, 1994; McCracken & Gross, 1998). In terms of the sexual pain disorders, fear of pain has been conceptualized as both a symptom and a cause of vaginismus in the literature. Several surveys suggest that many women with vaginismus report fear of pain as the primary reason underlying their condition and their sexual abstinence (Reissing, Binik, & Khalifa, 1999). This result is consistent with other research findings generally linking fear of pain to pain-related escape and avoidance (Asmundsen & Taylor, 1996). Fear of penetration, independently of pain, has not been documented, although this may be a factor. The only study to examine the relationship of pain intensity ratings and anxiety in dyspareunia found that anxiety was independently predictive of pain intensity ratings. (Meana, Binik, Khalifa, & Cohen, 1998). In this same matched control study, the undifferentiated group of women with dyspareunia reported more phobic anxiety and more erotophobia than controls (Meana. et al., 1997a). These findings, however, were not true of each diagnostic subgroup. Van Lankveld, Weijenborg, and Ter Kuile (1996) and Bergeron et al. (1999b) found that women with vulvar vestibulitis reported unremarkable levels of fear/anxiety as compared with norms.

Depression and chronic pain share a long association in the empirical literature and their comorbidity seems to be particularly marked in women (Meana, 1998). Attempts to determine the direction of the relationship, however, have ended primarily in confusion. The only fact that seems relatively clear is that where there is pain, there are higher than normal rates of depressive symptomatology, and where there is depression, there are more pain complaints than in the general population (Meana & Stewart, in press). Prior to any controlled studies investigating the specific link between depression and the sexual pain disorders, certain researchers speculated that depression was a potent cause of dyspareunia (Jarvis, 1984), whereas others argued that depressive symptoms in women with dyspareunia were reactive rather than etiologic (Fordney, 1978). More recently, in preliminary research, Meana et al. (1998) have shown that depression, unlike anxiety, is not predictive of pain ratings in dyspareunia. Although Meana et al. (1998) found that depressive symptomatology was elevated in their sample of women with dyspareunia, a closer analysis showed that the elevated depressive symptoms were confined to women who suffered from dyspareunia with no obvious physical pathology (Meana, 1997a). Women with vulvar vestibulitis and women with dyspareunia medically attributed to postmenopausal vaginal atrophy did not report more depressive symptoms than controls. Van Lankveld et al. (1996) and Bergeron (1999) also failed to find elevated depression in their samples of women with vulvar vestibulitis.

Two additional affective factors, hostility and somatization, have been hypothesized as etiological factors in the sexual pain disorders. Hostility was posited primarily by psychoanalytic theorists (Meana & Binik, 1994) who interpreted sexual pain as a possible displacement of anger. There is no strong empirical support for this position. The explanation of sexual pain as a type of somatization also has its roots in psychoanalytic theory and also has no empirical support. In the Meana et al. study (1997b), there was no difference between women with dyspareunia and matched controls either on the 1983 Brief Symptom Inventory (Derogatis & Melisaratos) somatization scale nor on the number of other regular pains reported.

Cognitive. Over 20 years ago researchers with a learning theory perspective hypothesized that the sexual pain disorders may be the result of negative scripts and expectations of sexual intercourse. The theory was that negative early experiences, and lack of or faulty learning, led to maladaptive schemas of sexual intercourse (Lazarus, 1989; Sotile & Kilmann, 1977). These maladaptive schemas were characterized primarily by negative expectations that then colored any sexual encounter as, at least, undesirable and, at worst, painful. Strict religious upbringing was one of the possible schemas proposed to potentially result in sexual pain but religiosity has failed to receive consistent support (Laumann et al., 1999; Reissing, Binik, & Khalife, 1999). More recently researchers have focussed on specific factors such as cognitive distortion, causal attribution, and attentional focus in the experience of pain. Pain-specific cognitive distortions such as catastrophizing have been found to be important factors in how individuals cope with pain (Sullivan, Stanish, Waite, Sullivan, & Tripp, 1998; Unruh, 1996). Preliminary data (Binik & Koerner, 1998) show that increased catastrophizing as measured by Sullivan's scale (Sullivan et al., 1998) is positively correlated with elevated pain reports during the cotton swab test in women with vulvar vestibulitis. Meana, Binik, Khalife, and Cohen (in press) elicited from women with dyspareunia their lay theories about the cause of their pain. Interestingly, women who made psychosocial causal attributions for their pain reported higher pain intensity, more psychological distress, lower levels of marital adjustment, and more sexual dysfunction than women who made physical causal attributions. In fact, it was perceived etiology rather than the medically determined etiology that predicted pain intensity and psychosocial adjustment.

Another intriguing finding is that attentional focus (distraction) seems to have a significant impact on pain experience (Arntz & deJong, 1993; Arntz, Dreessen, & Merckelbach, 1991). Although no research has been carried out specifically with respect to sexual pain, it is tempting to speculate that distraction would reduce sexual pain but possibly interfere with desire and arousal. After all, distraction is a common arousal-dampening strategy of men with premature ejaculation.

Social. More than any other pain syndrome, sexual pain is an interpersonal pain, It occurs primarily in the presence of another individual and as an immediate result of sexual contact with that individual. This makes sexual pain unique and particularly vulnerable to the influence of social factors. Unfortunately, there is very limited research investigating this issue.

In his discussion of potential psychosocial etiologic factors, Lazarus (1989) suggests that dyspareunia may be attributable to pervasive negative feelings between partners (hostility or lack of attraction). There is some evidence to support this view. Meana et al., (1997a) found that as compared with controls an undifferentiated group of women with dyspareunia exhibited more marital distress and scored significantly higher on a measure of interpersonal sensitivity. These differences did not extend to every diagnostic subgroup, as only the group with no obvious organic findings showed higher levels of marital distress compared to controls. Meana et al., (1998) also found that decreased relationship satisfaction was predictive of increased pain in women with vulvar vestibulitis. A promising area of study in this regard is the relationship between the sexual pain experience and the reactions of significant others to the problem. Pain relevant responses (e.g., solicitous behavior) from significant others have been linked to pain behavior and disability (Kerns & Rosenberg, 1995; Romano et al., 1992). The impact of significant others on the experience of sexual pain has not been studied.

Relationships with health-care practitioners may also play an important role in the sexual pain disorders. There is a recognized propensity on the part of health-care practitioners to attribute both idiopathic and women's pain to psychological causes (Bernstein & Kane, 1981; Bilkey 1996; Colmeco, Becker, & Simpson, 1983). This type of psychological attribution may have a negative impact on women who suffer from these disorders (Meana et al., in press). The traditional psychogenic default position when no tissue damage is found (cf. Binik, Bergeron, & Khalife, in press) is often interpreted by the patient as a lack of validation of the pain in turn adding to iatrogenically worsened feelings of hopelessness and depression and increased pain.

Therapy

In general, treatment strategies for the two sexual pain disorders have developed quite independently. Vaginismus has been typically conceptualized as a psychogenic disorder. As a result, mental health professionals have been the primary therapists and have used interventions revolving around vaginal dilation (Reissing, Binik, & Khalif6, 1999). Dyspareunia has usually been approached from an organic perspective with physicians using numerous medical and surgical interventions directed at the presumed underlying biological cause (Bergeron, Binik, et al., 1997; Meana and Binik, 1994).

Vaginismus

Despite the lack of controlled outcome studies, most sex therapists appear to feel confident about current methods of treating vaginismus (cf. Heiman & Meston, 1997). This confidence appears to be based on the 100% success rate reported by Masters and Johnson (1970) and Helen Singer Kaplan's comment that treatment is "incredibly simple" (1974, p. 417). The traditional treatment of vaginismus usually involves the following elements: (a) progressive vaginal dilation, (b) sex education and information, and (c) emotional desensitization to negative affect associated with sexuality. In principle, before such treatment is initiated, attempts are made to exclude biological causes such as rigid or imperforate hymens, vaginal septa, and so forth (cf. Stuntz, 1986, for a review). It is not clear, however, how often such screenings are actually carried out in clinical practice since gynecological examinations are often impossible or very difficult. Other types of interventions have also been reported including the following: hypnotherapy, biofeedback, local injections of acetylcholine blockers, controlled enlargement of the vaginal introitus, topical application of anesthetics, and abreaction interviews (Reissing, Binik, & Khalifa, 1999).

Even if one were to rely on the existing but methodologically flawed studies, there is little to suggest simple treatment and uniformly high success rates (cf. Leiblum, Pervin, & Campbell, 1989; Reissing, Binik, & Khalife, 1999). Therapy can often last many months and may involve complex interventions. Reported success rates range from 100% to below 60%. Treatment drop-out rates are significant when they are reported, and the few long-term follow-ups often show substantial relapse rates. To the best of our knowledge, there are no studies that examine the hypothesized vaginal muscle spasm before and after successful treatment. Although it appears that vaginal penetration is possible after successful treatment, it is often not pleasurable. Numerous authors have questioned the outcome criterion of successful penile-vaginal penetration by pointing out that having children is the major goal of many couples who seem to lose interest in further treatment once they conceive (e.g., Drenth et al., 1996; Leiblum et al., 1989).

It is interesting that, despite the presumed vaginal muscle spasm etiology of vaginismus, physical therapy has rarely, if ever, been recommended. Because physical therapy is the standard treatment for muscular spasms in every other part of the body, this seems unusual. Vaginal dilation appears to be a poor version of what an experienced pelvic floor physical therapist is able to achieve. Whether this type of intervention might be more successful than dilation, however, has not been investigated. Overall, Reissing, Binik, and Ehaliff's, (1999) conclusion seems apt: "It is not clear whether researchers have failed to empirically demonstrate the clinically reported positive treatment outcome, or whether the presumed positive outcome is an artifact of selfselection and selective reporting."

Dyspareunia

There is a very long list of medical treatments that have been applied to dyspareunia (cf. Bergeron, Binik, et al., 1997; Meana and Binik, 1994; Steege et al., 1998). In most cases, these treatments were directed at some underlying disease or abnormality presumed to cause the coital pain. Because standardized criteria are not available, the evidence linking the pathological state with the coital pain often depended heavily on a particular clinician's judgment. If the underlying disease or condition were successfully treated and the pain persisted then the dyspareunia was termed psychogenic or functional and often referred to a mental health clinician.

The mental health clinicians or sex therapists who treated these women typically used the available therapy models of their time whether psychodynamic, behavioral, cognitive, systemic, or mixed. Independent of their theoretical therapy model, many clinicians in the last 50 years have also used a variety of behavioral techniques. The most common technique, vaginal dilation, appears to be borrowed from the standard treatment of vaginismus based on the untested assumption that heightened muscular tension is also present in women with dyspareunia. Other reported techniques include systematic desensitization, vaginal muscle exercises (e.g., Kegels), hypnotherapy, sex education, changes in sexual repertoire, and couple and individual psychotherapy.

More specific types of therapy have been developed for the presumed subtypes of dyspareunia such as vulvo/vaginal atrophy or vulvar vestibulitis. Postmenopausal women experiencing dyspareunia are typically presumed to suffer from vulvo/vaginal atrophy leading to reduced lubrication and coital pain. They are often treated with vaginal creams or lubricants to compensate for the reduced lubrication. Alternatively, hormone replacement therapy is recommended to reduce or prevent vulvo/vaginal atrophy. With respect to vulvar vestibulitis, Bergeron, Binik, et al. (1997) and Bergeron, Binik, & Kbalife (1999a, 1999b) have recently reviewed the reported treatments under the categories of medical management, surgery, and cognitive-behavioral intervention. The most frequently investigated intervention is a surgical one (vestibulectomy), consisting of an excision of the hymen and of all the sensitive areas of the vestibule to a depth of approximately 2 mm (cf. Bergeron, Bouchard, Fortier, Binik, & Khalif6, 1997). This procedure is typically carried out as day surgery under general or epidural anesthesia and is usually recommended following the failure of medical management. Heiman and Meston (1997) have concluded that the available data are not sufficient to characterize any of the medical, surgical, or psychological therapies as even probably efficacious.

Recently, Bergeron et al., (1999a) have completed the first randomized treatment outcome study for dyspareunia. In this randomized controlled study, cognitive-behavioral therapy, surface electromyographic biofeedback, and vestibulectomy were compared in a carefully selected sample of 78 women with vulvar vestibulitis syndrome. All women were assessed at pretreatment, posttreatment, and 6-month follow-up via gynecological examinations, structured interviews, and standard questionnaires pertaining to pain, sexual function, and psychosocial adjustment. As compared with pretreatment, all treatment groups reported statistically significant reductions on pain measures at posttreatment and 6-month follow-up, although the vestibulectomy reported the most improvement. Based on a general measure of self-reported improvement, 68% of vestibulectomy participants reported a successful outcome at 6-month follow-up though 9% were worse. As for behavioral treatments, 35% of biofeedback participants and 39% of cognitive-behavioral therapy participants had a successful outcome at 6-month follow-up. Depending on the pain outcome measure, the average percentage of pain reduction from pretreatment to 6-month follow-up ranged from 47% to 70% for the vestibulectomy participants, 19% to 35% for the biofeedback participants, and 21% to 38% for the cognitive-behavioral therapy participants. All three groups significantly improved on measures of psychological adjustment and sexual function. No predictors of outcome were identified. A 2 1/2-year follow-up is now underway.

This study is important for a variety of reasons. First, it strongly suggests that vulvar vestibulitis can be treated successfully. Second, although vestibulectomy was the most successful treatment, psychosocial interventions were also clearly useful. This suggests that a biopsychosocial perspective may be the most appropriate. Third, the pain outcome measures were the most important predictors of patient-rated improvement. This supports a pain-centered conceptualization of vulvar vestibulitis.

Conclusion: Is the Pain Sexual or is the Sex Painful?

Meana, Binik, Khalife, Bergeron, et al. (1997) have pointed out that the clinical and scientific community's response to the question of "Is the pain sexual or is the sex painful" is more than a semantic exercise. The nature of one's response has, in fact, important implications for (a) how and which scientists go about studying pain, (b) how and which health professionals treat it, (c) how individuals react to their own pain, (d) the reactions of significant others, and (e) the way larger social institutions address the problem.

Although the clinical and societal implications of the answer to this question are important, they have not been a major emphasis of this review (cf. Binik et al., in press for a review of the clinical implications). Our focus has been a critical review of the scientific conceptualization of and empirical data related to sexual pain. Based on this review, there seems little justification for continuing to conceptualize vaginismus and dyspareunia as sexual problems. This conclusion is based on the following considerations: (a) The major symptom of both problems is pain and the traditional quantitative and qualitative measures of pain and pain disorders appear to be very useful in describing and classifying vaginismus and dyspareunia. (b) The pain (or spasm) of vaginismus and dyspareunia can be induced in nonsexual situations and the typical taxa of the sexual dysfunctions have not been useful in describing or classifying the sexual pain disorders; (c) From an epidemiological or taxonomic point of view neither vaginismus nor dyspareunia are closely related to other female sexual dysfunctions. Dyspareunia, on the other hand, appears to be closely related to many other pelvic floor or urinary dysfunctions or disorders. (d) Explanatory biopsychosocial mechanisms drawn from the pain and health psychology literatures (e.g., central sensitization, catastrophization, illness attribution) are opening promising new avenues for research; the traditional sexual dysfunction explanations (e.g., spasm, faulty sexual scripts, underlying couple, or medical pathology) have not resulted in significant recent progress. Finally, treatments for dyspareunia based on sexual dysfunction models have not been useful. Recent therapy outcome data based on multidisciplinary pain interventions including, surgery, biofeedback and cognitivebehavioral interventions show great promise. Vaginal dilation may be useful for the treatment of vaginismus but can be seen as a method of physical therapy rather than sex therapy.

In our view, sexual pain conceptualizations of vaginismus and dyspareunia have confused the nature of these problems with the major activity with which they interfere. Although this interference with intercourse is the major concern that brings women with pain during intercourse to clinical attention, it is not the only one. Interference with conception, interference with gynecological screening exams, and interference with sports are also important complaints. Our pain conceptualization, however, does not diminish the necessity for a therapeutic focus on sexuality because the reduction or removal of pain will not insure a pleasurable sex life. In this respect, vaginismus and dyspareunia are also similar to other chronic pain problems in that the reduction or removal of chronic pain often does not lead to a return to work or normal activities.

One direct implication of our approach is a recommendation for a change in nomenclature. The term dyspareunia currently encompasses diverse and probably unrelated pains in different organs. The term vaginismus is based on an unsubstantiated mechanism. A more reasonable approach would be to attempt to characterize pains that occur during intercourse according to the multiaxial pain classification of the International Association of the Study of Pain (Merskey & Bogduk, 1994). This system suggests that pain syndromes should be described according to bodily region, psychophysiological system, temporal characteristics of the pain, patient's statement of intensity, and etiology.

Following this general framework, we would likely reclassify "sexual pain" as a type of pelvic pain. The different subtypes of pelvic pain which are closely linked to intercourse would then be differentiated by more specific locations (e.g., vulvar vestibule), involvement of particular psychophysiological systems (e.g., muscular, autonomic, etc.), the temporal characteristics and description of the pain, and ultimately by etiology. What we know of vulvar vestibulitis syndrome fits nicely into this framework, and it is likely that other possible syndromes such as vaginismus or dyspareunia due to vaginal atrophy can be usefully described in this way. It seems likely to us that further research will justify possible diagnostic categories such as vaginal hyperalgesia. There will, of course, be pelvic pain syndromes not closely linked to intercourse that will affect the same reproductive organs. The issue of pain induced interference with activities then becomes an important clinical/rehabilitation issue but not a basic characteristic of the pain syndrome. We believe that this "de-sexualized" approach to sexual pain will lead to improved understanding and treatment of important and currently neglected women's health problems.

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