Abstract
Past investigations of chronic testicular pain provide a sparse representation of the men with this condition and lack key details to aid our understanding of this important men’s health condition. As a chronic pain syndrome, more research is necessary to understand the phenomenon of chronic testicular pain and the pain experience of these men. This integrative literature review provides a summary of the current state of the science of chronic testicular pain in men, identifies the gaps in our knowledge, and provides recommendations to address this knowledge gap.
Introduction
Approximately 116 million American adults suffer from some form of chronic pain, costing over $635 billion each year in lost productivity and medical care costs (Institute of Medicine [IOM], 2011). The IOM recognizes chronic pain as a public health challenge that causes considerable loss of productivity and places a financial drain on the health care system (IOM, 2011). Men with chronic testicular pain (CTP) represent a small number of adults evaluated in primary care, urology (Ciftci, Savas, Yeni, Verit, & Topal, 2010), and pain clinics. Although this represents a small number of men, the impact of CTP on quality of life, social, sexual, and economic circumstances is high. In an attempt to address their chronic pain, these men are evaluated by multiple providers from different disciplines. This is congruent with the health-seeking behaviors of many other chronic pain populations, such as chronic low back pain, fibromyalgia, and interstitial cystitis (Gatchel, Peng, Peters, Fuchs, & Turk, 2007; IOM, 2011; Wesselmann, Burnett, & Heinberg, 1997). CTP represents a challenging clinical entity for providers to successfully treat and manage because of its varying etiology. Oftentimes, there can be straightforward explanations for the CTP (varicocele or referred pain from an inguinal hernia), but equally often the etiology remains unexplained. In fact, from 18.6% (Ciftci et al., 2010) to 25% of CTP has no known cause (Davis, Noble, Weigel, Foret, & Mebust, 1990).
Men with CTP experience loss of economic productivity (Doubleday, Kulig, & Landel, 2003), alterations in social and family roles (Davis et al., 1990; Hong, Corcoran, & Adams, 2009), and compromises in their sexual function (Ciftci et al., 2011). Men seek evaluation from multiple providers in an attempt to uncover an explanation and treatment for their CTP (Costabile, Hahn, & McLeod, 1991; de Oliveira et al., 2009; Heidenreich, Olbert, & Engelmann, 2002; Planken, Voorham-van der Zalm, Lycklama, Nijeholt, & Elzevier, 2010).
This condition also represents a gender gap and a disparity in the knowledge for a subpopulation of men with chronic pain. There is an established body of literature that evaluates chronic pelvic pain in women (e.g., dyspareunia, interstitial cystitis, vulvodynia, vaginismus) and the sexual and social issues of these complaints (Bergeron, Rosen, & Morin, 2011; Johansen & Weidner, 2002; Wesselmann et al., 1997). As such, chronic sexual and genital pain is well detailed in women (Haefner et al., 2005). However, less is known about similar issues in the men. Findings in women with chronic genital pain recognize the need to place this pain within a psychosocial framework to evaluate not only the characteristics of the pain but also its status within the larger social and sexual context of women (Bergeron et al., 2011; Haefner et al., 2005; Mandal et al., 2010). Reports of chronic genital pain conditions in men fail to evaluate their experience in this same social, sexual, and self-esteem context. There is a paucity of research regarding the experience of men with CTP. Examination of this clinical entity is limited to its manifestation as a component of chronic prostatitis/chronic pelvic pain syndrome or attributed to acute conditions (e.g., epididymitis; Johansen & Weidner, 2002; Luzzi, 2003; Tripp et al., 2006).
CTP is gaining increasing awareness among men and providers, due in part to the opportunity for men to self-refer to providers, widespread availability of Internet information, and articles in gender-specific magazines (such as
Background
Chronic orchialgia is defined as an “intermittent or constant unilateral or bilateral testicular pain three months or longer in duration that significantly interferes with the daily activities of a patient so as to prompt him to seek medical attention” (Davis et al., 1990, p. 936). This frequently cited definition for CTP is consistent with the IOM (2011) description of chronic pain as pain lasting more than 3 to 6 months outside the realm of normal healing.
Potential causes for CTP include postvasectomy pain, epididymitis, varicocele, prostatitis, hydrocele, testicular tumor, radicular pain, and referred pain from various sources (Basal et al., 2012; Keoghane & Sullivan, 2010; Levine, 2010; Luzzi, 2003; Masarani & Cox, 2003). Once identifiable causes are ruled out, a diagnosis of CTP of unknown etiology remains. Many men submit to invasive procedures to alleviate their chronic pain (vasectomy reversal, spermatic cord block, epididymectomy, denervation of the spermatic cord) with varying degrees of success (Costabile et al., 1991; Strom & Levine, 2008).
Innervation of the testes is reasonably well explained. Autonomic supply to the male genitals originates with sympathetic fibers from the T10 and L1 vertebral segments. An additional 10% of this autonomic supply is parasympathetic and originates from the sacral S2-S4 level (Reynolds & Sills, 2007). These nerves converge, forming the spermatic plexus, which innervates the testes, epididymis, and vas deferens (Wesselmann et al., 1997). Somatic supply to the scrotum and testes originates from the lumbar spine L1 and L2 levels. The ilioinguinal nerve supplies sensory innervation to the root of the penis and upper scrotum, while the genitofemoral nerve innervates the cremaster muscle and parietal and visceral tunica vaginalis. S2 to S4 nerve roots innervate the posterior and inferior scrotum by way of the pudendal nerve (Keoghane & Sullivan, 2010). This overlap in nerve supply to the testes, as well as their embryologic origin within the viscera, may contribute to poor localization of pain to a specific scrotal structure.
Theoretical Model and Purpose
The biopsychosocial model (BPS) is frequently used to examine pain. Its origins are in general systems theory (Engel, 1982) and include the interrelatedness of systems and processes, incorporating social and psychosocial conditions within the context of medical care. Within the BPS model, an individual is seen as part of a hierarchy that recognizes distinct components as parts of a larger system (e.g., community); nothing exists alone, but has influence as a component of something larger (Engel, 1982). An advantage of this approach, especially within the context of chronic pain, is the emphasis that a particular patient complaint, although pertinent on its own, exists within a larger context of the person’s history, experiences, and sensations. Furthermore, the BPS model underscores the importance that patient presentation is subject to a variety of lesser influences that might not be initially apparent (Borrell-Carrio, Suchman, & Epstein, 2004) such as social, sexual, and cultural norms. It further emphasizes the unique, subjective experience of the person. This becomes especially evident as a condition progresses from an acute to chronic state and potential economic, social, and medicolegal aspects of it become increasingly influential (Gatchel & Mayer, 2008).
The IOM (2011) recommends several parameters drawn directly from the BPS model for examination of chronic pain. These include incidence and prevalence data, a description of the characteristics of the chronic pain, its impact on daily activities, any disability related to pain, an account of utilization of services, and the costs of pain and pain care. Therefore, the purposes of this integrated literature review are to describe what is currently known about men with CTP, to identify the gaps in the state of the science, and to provide recommendations for future research. To this end, we will describe the current definition of CTP, the scope of the problem, and present a review of studies addressing this important aspect of men’s health.
Method
We conducted a search of the published literature from January 1970 to September 30, 2012, using the following databases: Google Scholar, MEDLINE, CINAHL, ProQuest, PSYCHinfo, Web of Science, and SCOPUS. The search was limited to articles with the following key words in their titles: testicular pain, chronic orchalgia, chronic testicular pain, chronic testalgia, chronic scrotal pain, chronic male genital pain, chronic orchidynia, and chronic orchialgia. These terms were further narrowed by excluding children and selecting subjects at least 18 years of age. Only articles available in English were reviewed. Review articles, published abstracts, citations, letters to the editor, commentaries, patents, book chapters, and animal studies were excluded. Articles that included discussion of CTP with an identified cause, such as malignancy or postvasectomy pain, were also excluded. This resulted in a total number of 15 research-based articles and 11 case reports (Figure 1).
Strategic search for studies addressing unexplained chronic testicular pain.
Exclusion and inclusion criteria used in this review resulted in the purposeful exclusion of one of the most frequently cited articles discussing CTP (Davis et al., 1990). We chose to exclude this article for two reasons: it includes children as young as 11 years (range = 11-69) and because it contains no details regarding the number of participants below the age of 18 years. It is likely that the etiology and experience of CTP in adolescent and preadolescent boys is different from that of adult men. Its interpretation and evaluation requires an understanding and appreciation of their chronological age and development. We also excluded two other articles (Perimenis, Speakman, & Higgins, 1994; van Haarst, van Andel, Pels Rijcken, Schatmann, & Taconis, 1999) because of the young age of subjects (14 and 10 years, respectively).
Results
A total of 26 articles were reviewed (15 research articles, 11 case studies). A consistent definition of CTP is noted among the articles reviewed, based on the Davis et al. (1990) definition. The research articles are predominantly retrospective chart reviews, with the largest cohort evaluating 74 men (Benson, Abern, Larsen, & Levine, 2012). A summary of the research studies (Table 1) and case studies (Table 2) is presented.
Research Studies to Date Investigating Chronic Testicular Pain.
Note. DIPFF = standard Dutch evaluation of pelvic floor function that includes the PelFIs history tool and electromyography; IIEF = International Index of Erectile Function; WHOQOL-BRIEF = World Health Organization Quality of Life questionnaire–Brief; VAS = visual analog scale.
Includes results from 1996 and 2001.
Includes results from 1996, 2001, and 2008.
Case Studies/Series Evaluating Chronic Testicular Pain.
For some articles, total
Biological Aspects
Pain ratings for article are by self-report, and only one article describes an intervention that included a control group (Ciftci et al., 2011). Little is known about demographic characteristics of these populations beyond age. Although age is consistently reported, other demographic information is not. Only Costabile et al. (1991) reports the racial composition of their sample (89% Caucasian; 10% African American). A complete workup to rule out identifiable causes for CTP is mentioned, but the precise nature of any workup is not detailed, and the focus is primarily on success or failure of invasive treatments or surgical interventions. Pain description is limited to its location in the scrotum (left, right, bilateral) without localizing it distinctly to the testis. A visual analog scale is used to measure intensity. Only three of the research articles (Ciftci et al., 2011; de Oliveira et al., 2009; Misra, Ward, & Coker, 2009) and four of the case reports (McJunkin, Wuollet, & Lynch, 2009; Nouri & Brish, 2011; Rosendal, Moir, de Pennington, Green, & Aziz, 2012; Rowell & Rylander, 2012) include the quality of CTP.
Sinclair, Miller, and Lee (2007) offer support for a neuropathic origin of CTP and are the first to show that neuromodulating medications decrease pain in chronic orchialgia. This is consistent with reports that describe the pain as
Many of the descriptors within the case studies are consistent with descriptions of neuropathic and radicular-type referred pain. The pain is described as “sharp, shooting” (McJunkin et al., 2009), “burning, stabbing, with radiation” (Nouri & Brish, 2011), “continuous, sharp, with radiation and hyperesthesia” (Rosendal et al., 2012), “incapacitating” (Choa & Swami, 1992), and as a “constant dull ache” (Rowell & Rylander, 2012). Central vertebral disc protrusion, thoracolumbar dysfunction, muscle spasms, or sacroiliac joint dysfunction (Doubleday et al., 2003; Jemelik, Penickova, & Vyborny, 1992) is documented in men with CTP, offering a potential pathophysiological etiology for their complaints of neurologic pain sensations.
Psychological Aspects
Additional aspects of the impact of CTP are poorly reported in the articles reviewed. Ciftci et al. (2011) used standardized tools to establish that CTP reduces quality of life, sexual satisfaction, frequency of sexual activity, and overall libido when compared with controls.
CTP may be a unique manifestation of chronic pelvic floor dysfunction, a result of possible sexual abuse, or may be related to chronic pelvic pain syndrome (Planken et al., 2010). Costabile et al. (1991) suspect a precipitating event prior to a diagnosis of CTP, although no specifics are provided. When this aspect is explored, men become “angry and incredulous when it is suggested they may benefit from referral to a mental health professional” (Cadeddu et al., 1999, p. 734).
The case studies in this review offer insight into a possible psychogenic component of CTP. In the original article describing microsurgical denervation of the spermatic cord, Devine and Schellhammer (1978) report that one of their patients had “recurrent pain . . . due to an overriding psychogenic component” (p. 151). Naim and Ende (2011) report the identification of deep-seated emotions resulting in chronic genital and testicular pain.
Social Aspects
Only 3 of the 15 research studies address the issue of work status, educational level, or the potential economic impact of CTP. Costabile et al. (1991) note that 30% of the Army population in their study completed some level of college, 20% had professional-level jobs, 31% were skilled laborers, and 42% were manual laborers, but the impact of CTP on their ability to function at their jobs remains unclear. However, 31% (15/48) were eventually granted medical discharge due to their chronic orchialgia. Treatment with pulsed radiofrequency of the spermatic cord allowed 10 men to resume normal activities (Misra et al., 2009). Cadeddu et al. (1999) speculate that men resort to surgery in part due to limited insurance coverage for physical therapy and mental health services.
The economic impact of CTP on the work status of these men is unclear; not all men were working when evaluated as subjects of the case studies. However, after treatment many are able to return to work (Doubleday et al., 2003; McJunkin et al., 2009; Nouri & Brish, 2011; Rosendal et al., 2012).
Discussion
Based on the 26 articles in the review, the characteristics of the average man with CTP is a 45-year-old, Caucasian of European descent, and has suffered CTP for 3 to 366 months (Benson et al., 2012; Nariculam et al., 2007). The pain is more likely to be unilateral and slightly more likely to be on the right side of the scrotum. Various and numerous conservative treatment measures have failed; he has seen on average 4.5 urologists (Heidenreich et al., 2002), has undergone 4.7 to 7.2 procedures (Costabile et al., 1991; Heidenreich et al., 2002), and has had 1.6 surgeries (Costabile et al., 1991).
The estimated prevalence of chronic
Biologic Considerations
A precise cause for complaints of chronic scrotal or testicular pain is difficult to ascertain due to the inconsistent use of descriptors throughout the literature. The terms
The reviewed studies of CTP fall short of the current IOM recommendations regarding assessment of pain. Many fail to use standardized tools to capture characteristics of pain, with the exception of a pain scale. When incorporated, pain scales are inconsistently used among these studies. Some studies use a 0 to 10 scale, whereas others use a 0 to 5 scale, making comparisons difficult. Assessment of pain should include investigation of its multiple dimensions (intensity, quality, location, duration, and sites other than a primary site) (IOM, 2011). The case studies provide a much richer and detailed description of the chronic pain experience of these men, including a much better description of pain quality and precise location(s) of the pain.
The review of the literature reveals that little is known about the demographics of men with CTP beyond age. This lack of demographic data significantly limits our ability to understand the potential influence of sexual orientation, ethnic, social, and cultural variables on CTP and hampers our ability to recognize at-risk men for developing CTP. Understanding the social and cultural norms that influence men and their response to CTP, while establishing a potential pain phenotype for this condition, is very difficult given the limited information.
Psychological Considerations
To date, very little attention is focused on the psychosocial aspects of the chronic pain experience for men with CTP, despite the existing model available for parallel conditions in women (Haefner et al., 2005; Mandal et al., 2010). The intensity of pain alone provides inadequate insight into the effects of pain on an individual. This limited knowledge hinders comparison across the studies as well as among other groups of chronic pain patients. It also ignores the extent to which pain and anticipation of pain may result in avoidance and other maladaptive behaviors.
Furthermore, the literature review reveals a lack of research on predicting factors that contribute to CTP and disability in men. This may represent a failure of current medical treatment or a lack of recognition of the emotional suffering and poor quality of life that bring men back to providers or specialty clinics for evaluation and treatment (Kumar, Mehta, & Nargund, 2010). Psychological or psychiatric evaluation is not a priority within current recommendations for treatment. Psychological evaluation may be considered prior to certain invasive surgical procedures (Davis et al., 1990), such as orchiectomy, but this option is not reflected in contemporary algorithms (Benson et al., 2012; Benson & Levine, 2012). Within the parameters of the BPS, it would be prudent to evaluate psychological status prior to less invasive treatments, as psychological factors can influence the overall experience of pain (Gatchel, 2004; IOM, 2011).
Men with CTP demonstrate characteristics found in other chronic pain syndrome patients, such as reporting pain as their chief complaint, low correspondence between symptoms and objective medical findings, an association with psychosocial dysfunction, and a history of multiple, unsuccessful treatments (IOM, 2011; Kumar et al., 2010; Planken et al., 2010). This includes a “try anything” attitude that results in “an extraordinary number of interventions” (Egan & Krieger, 1997, p. 215) usually with a low success rate. This is consistent with somatization disorders. Standardized instruments for general assessment of somatization disorders may not be reliable and valid in men with CTP, supporting the premise that psychological and psychiatric evaluation be conducted prior to invasive procedures (Hong et al., 2009).
Some men with CTP have pain secondary to sexual abuse. Four articles suggest there may be a component of sexual abuse in some of these men (Costabile et al., 1991; Davis et al., 1990; Planken et al., 2010; Schover, 1990). The Schover (1990) pilot study describes 48 men who underwent a psychological interview after being referred by urologists who could not establish an organic cause for their
Social Considerations
Despite the known impact of chronic pain on lost productivity and medical care costs, the economic impact of CTP is rarely cited in the literature. There is almost no mention of it as it relates to work status prior to treatment or as it relates to the ability to return to work posttreatment. This creates a challenge in drawing conclusions regarding the true economic impact of this condition. There are ample data indicating that the economic impact of chronic pain is significant, regardless of the site of pain. IOM (2011) data indicates that the annual cost to the health care system is $4,516 more per individual with moderate pain and $7,726 more per individual with severe pain when compared with an individual without pain.
Limitations
There are two limitations of this review. First, it did not include children, which may limit our overall understanding of CTP. However, as CTP in children is most likely a distinct clinical entity, limiting our review to adult men helps establish the parameters unencumbered by issues related to growth and development. Second, only chronic unexplained testicular pain was examined. Although the inclusion of CTP with identified causes would have increased the overall pool of studies, we chose to focus on CTP without identifiable causes to shed light on this elusive area of study.
Future Directions
Given the paucity of studies describing men with CTP, there is a desperate need for data describing this area of men’s health and its impact on men and their families. Demographic data are needed to characterize men affected by CTP, including socioeconomic status, sexual orientation, ethnicity, relationship status, and past medical and surgical history. Information detailing types of providers who are associated with various treatment options is also needed and may include primary care providers (including nurse practitioners and physician assistants), urologists, neurologists, psychologists, and anesthesiologists.
Because contemporary literature lacks psychological description of the experience of CTP, qualitative research is needed to uncover the key aspects of what it is like to live with this debilitating chronic pain condition. An increasing body of literature demonstrates that individuals living with chronic tend to catastrophize their pain experience, resulting in specific bodily complaints (Edwards, Bingham, Bathon, & Haythornthwaite, 2006; Gatchel, 2004; IOM 2011; Seminowicz & Davis, 2006). It is reasonable to conclude that some men with CTP may also catastrophize their pain. If so, this has implications in terms of their overall CTP experience, as the cognitive interpretation of pain can give rise to physiological and behavioral responses that influences an individual’s performance outcomes and create a more intense pain experience and increase emotional distress (Gatchel et al., 2007; Jensen, 2010). Catastrophizers have difficulty directing their attention away from pain and succumb to rumination, feelings of helplessness, and pessimism regarding outcomes, all of which predict higher self-reported disability (Edwards et al., 2006; Seminowicz & Davis, 2006). The IOM (2011) specifically recommends screening for this characteristic as a predictor of treatment success, as it predicts future disability scores after controlling for variables such as gender and type of pain syndrome (Karoly & Ruehlman, 2007; Sullivan et al., 2001).
In addition, catastrophizing may also represent a broader dimension of maladaptive coping for men with CPT, providing dysfunctional feedback and increasing the pain experience. This method of coping may represent an attempt to ensure that pain is dealt externally rather than addressing the individual’s internal cognitive environment (Sullivan et al., 2001) and is consistent with the findings of Schover (1990), who concluded that pain was a strategy to create attention. To evaluate catastrophizing, the Pain Catastrophizing Scale should be administered and considered a primary screening tool. It may become possible to predict who is less likely to respond to traditional medical and surgical treatments, if there is strong evidence for catastrophizing. Further investigation of this particular aspect of chronic pain may make it possible to propose treatments for men with CTP that have been successful with other chronic pain groups that catastrophize.
Additionally, standardized tools should be incorporated into the study of CTP to capture its multiple facets, as noted in the BPS, to direct future interventions to manage, control, and treat CTP. These measures include overall coping, symptom perception, and pain-related disability and not simply questions regarding the presence or absence of pain or genitourinary symptoms. Instruments such as the Somatization subscale of the Symptom Checklist-90-Revised, McGill Pain Questionnaire, the Perceived Stress Scale, the Brief Sexual Functioning Questionnaire, the Survey of Pain Attitudes, the Short Form-36, the Multidimensional Scale of Perceived Social Support, and the Chronic Pain Coping Inventory may be beneficial. A “pain map” (schematized picture of the male genitalia) may also be helpful in determining the precise anatomic location of pain. These tools have been widely used with other chronic pain populations (Edwards et al., 2006; IOM, 2011) and would allow for comparison between men with CRTP and other chronic pain patients.
Conclusions
The current state of the science regarding CTP is underdeveloped and incomplete; the characteristics of men affected by CTP are poorly described, as are consistently effective treatments and interventions. The three most cited studies (Costabile et al., 1991; Davis et al., 1990; Schover, 1990) are more than 20 years old and consist of a combination of retrospective and prospective chart review without the inclusion of standardized psychometric tools. Additional research is urgently needed to address this poorly studied condition affecting the quality of life for men and their families.
Footnotes
Declaration of Conflicting Interests
The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.
Funding
The authors received no financial support for the research, authorship, and/or publication of this article.