and Filippo Murina2
(1)
Center of Gynecology and Medical Sexology, San Raffaele Resnati Hospital, Milan, Italy
(2)
Lower Genital Tract Disease Unit V. Buzzi Hospital, University of Milan, Milan, Italy
4.1 Introduction
Vulvar pain is an “orphan” symptom in prepubertal children. When a child complains of acute or chronic pain in that area, pediatricians/clinicians refer to “genital pain”: certainly a comprehensive, yet not a specific nor a sensitive word.
In this chapter, the focus is on pain precisely referred to the entire vulva or part of it: the clitoris, the labia minora and/or majora, the vestibulum, and the fourchette. The term vulvar pain (VP) will be preferred and used here for the sake of clarity, precision, quality of care, detection of vulnerabilities, and prevention of risks to become clinically relevant facts: a must, even more compelling in children and young patients.
Childhood acute vulvar pain is usually found to have a clear biological cause (Clare and Yeh 2011). Accurate national studies have been carried out on unintentional, accidental genital lesions in children, with specific focus on product safety, such as the U.S. Consumer Product Safety Commission (CPSC) National Electronic Injury Surveillance System (NEISS) (Casey et al. 2013; Tasian et al. 2013).
However, systematic studies specifically focused on vulvar pain in children up to the age of puberty are still missing. The literature reports essentially single clinical cases, more focused on acute vulvar pain.
Pain characteristics in this group are similar to adults. The study of the psychological component of vulvar pain is limited due to small numbers of patients available for review. Children who are victims of sexual abuse reported feelings about their genital pain (Sham et al. 2013). Studies on long-term outcome of acute vulvar lesions, and specifically on chronic vulva pain/vulvodynia, are still lacking.
Neglect of childhood vulvar pain may then lead pain to evolve to vestibulodynia/vulvodynia in vulnerable subjects, with a progressive central component in their pain perception.
The goal of this chapter is twofold: first, analyze the existing literature to give a perspective on current issues raised by vulvar pain in children; second, increase the clinical awareness for the need of an early diagnosis and appropriate treatment to avoid the shift to chronic and neuropathic vulvar pain.
4.2 Etiology of Vulvar Pain in Children
Different etiologies may contribute to vulvar pain in prepubertal girls (Table 4.1).
Table 4.1
Leading etiologies of vulvar pain in children
Traumatic: as a consequence of mechanical injuries on the external genitalia |
After a fall |
Riding toy-related genital injuries (cane bicycle (crossbar) |
Play chutes in playground/children garden |
On the coccyx, with a later compression of the pudendal nerve at its source at S2,S2,S4 level |
After injuries while playing/masturbating with foreign objects |
After burns |
Sexual abuse, during and after childhood abuse |
Ritual, after genital mutilation/cutting/ritual modification |
Iatrogenic |
When performing invasive maneuvers such as bladder catheterization or vaginal swab, without caring attention |
When suturing traumatic unintentional vulvar lesions without proper care |
After genital surgery for vulvar cysts (anecdotal case reports) |
After chemotherapy and/or radiotherapy, with peripheral neuropathic genital pain |
Autoimmune, when lichen sclerosus causes intense itching/pruritus |
Neuropathic: vestibulodynia and/or vulvodynia, spontaneous or provoked |
4.2.1 Traumatic Unintentional Accidental Lesions of the Vulva
Traumatic unintentional accidental lesions of the vulva in childhood usually cause acute, intense, excruciating vulvar pain, given the extremely rich innervation of the area.
Intensity and duration of vulvar pain depends on:
Type of injury: vulvar cut, hematoma, compression, burn, chemical, iatrogenic, and ritual damages. Injury diagnoses are usually classified as contusion/abrasion, dermatitis, foreign body, hematoma, laceration, strain/sprain, and others (including avulsion, burns, crushing, fracture, hemorrhage, associated internal organ injury, and puncture) (Casey et al. 2013).
Pediatric external genital trauma because of sports, playground equipment, toys, or furniture does not seem to be uncommon in everyday life. However, the rate of accidental, unintentional, and nonsexual pediatric genital injury is unknown, as most related literature is focused on the association of sexual abuse with genital trauma. Studies focusing on nonsexual pediatric genital trauma consist of case reports or small series and focus on either female or male patients exclusively.
From these studies, it seems that genital injury occurs in 0.4–8 % of reported childhood trauma, and the majority of accidental pediatric genital injury is minor, not requiring surgical or intensive medical treatment.
A retrospective cohort study utilizing the U.S. Consumer Product Safety Commission (CPSC) National Electronic Injury Surveillance System (NEISS) from 1991 to 2010 was performed (Casey et al. 2013) to evaluate pediatric genital injuries.
Pediatric genital injuries represented 0.6 % of all pediatric injuries with the incidence of injuries rising through the period studied, 1991–2010. The incidence is raising steadily from 1991 to 2010, in spite of increasing awareness and attention to safety issues for children.
The mean age at injury was 7.1 years and was distributed 56.6 % girls and 43.4 % boys. A total of 43.3 % had lacerations and 42.2 % had contusions/abrasions. The majority of injuries occurred at home (65.9 %), and the majority of patients (94.7 %) were treated and released from the hospital. The most common consumer products associated with pediatric genital trauma were bicycles (14.7 % of all pediatric genital injuries), bathtubs (5.8 %), daywear (5.6 %), monkey bars (5.4 %), and toilets (4.0 %) (Casey et al. 2013).
Another ample study within NEISS indicates that children 4–7 years old were most frequently injured (36.8 % of all injuries), followed by those 8–11 years old (20.6 %). Girls comprised 55 % of the injured children. The most commonly injured organs were female external genitalia (vulva) (37.7 %), penises (21.6 %), and testicles (12 %). Genitourinary injuries were most commonly associated with sporting and exercise equipment (35.7 %), furniture, (15.5 %) and clothing items (11.9 %). Of the patients 91 % were treated at the emergency department and discharged home (Tasian et al. 2013). Bicycle use is responsible for the highest percentage of sport-related genitourinary traumas in children (Bagga et al. 2015).
Blunt perineal injuries that require surgical repair occur predominantly in patients less than 10 years of age who sustain blunt perineal trauma from a variety of causes but rarely motor vehicle crashes. Thus, such patients should undergo aggressive evaluation, including examination under anesthesia (EUA), especially if they present with perineal bleeding, hematoma, or swelling. Furthermore, perineal injuries in children under 4 years should raise the suspicion of abuse (Scheidler et al. 2000).
Lower genitourinary injury may have more serious implication when associated to pelvic injuries. Studies indicate that 2.8 % of children with pelvic fractures (6/212) reported bladder and urethral trauma (Tarman et al. 2002). Shared innervation and proximity may increase the vulnerability of the vulvar tissues to short- and long-term consequences. However, this is not specifically reported/quoted in available studies on genitourinary and pelvic injuries.
Extension, severity, and complexity of vulvar tissue damage.
Involvement of the urethra and/or anal area.
Number of traumatic events: single versus multiple, for example, repeated fall at the swimming pool or on the cane bicycle.
Adequacy of the treatment, both in terms of quality of primary care, medical and/or surgical, including attention to quality and duration of analgesia and antalgic treatment, while suturing a genital cut, and psychological support to the child and, when indicated, to the family.
Quality of the follow–up and pertinent medical/psychosexual intervention and support.
4.2.2 Sexual Abuse
Every year, about 4–16 % of children are physically abused and one in ten is neglected or psychologically abused. During childhood, between 5 and 10 % of girls and up to 5 % of boys are exposed to penetrative sexual abuse, and up to three times this number are exposed to any type of sexual abuse. However, official rates for substantiated child maltreatment indicate less than a tenth of this burden (Gilbert et al. 2009; Bailhache et al. 2013). Diagnosis can be challenging (Berkoff et al. 2008). Criteria for the clinical diagnosis of children sexual abuse are summarized in Boxes 4.1 and 4.2. A dramatically high percentage of children have increased vulnerabilities to further emotional and physical abuse, neglect, and long-term negative consequences. Severity variables include:
- 1.
Type of abuse (nonpenetrative vs. vaginal penetrative sexual abuse): the vestibulum is an extremely vulnerable area for potential long-term consequences in terms of vulvar pain/vestibulodynia, more so as it involves an extremely emotionally charged area such as the vulva.
- 2.
- 3.
Associated abuses: neglect and/or physical abuse.
- 4.
Vulnerable social context: poverty, low education, single and/or unemployed mother, and no family/relatives/social support.
- 5.
Presence of sexually transmitted infections (STI), such as papillomavirus-induced condylomata (Fig. 4.1). This finding must activate a careful investigation of a potential sexual abuse and the presence of other STI, including gonorrhea and HIV.
Fig 4.1
Perineal warts in children: healthcare providers must always consider a potential sexual abuse (Picture: courtesy of Metella Dei, MD)
Box 4.1. How to Diagnose a Sexual Penetrative Abuse
Method
The transhymenal diameters and the amount of tissue present between the hymenal edge and vestibule inferiorly at 6 o’clock and laterally at 3 o’clock and 9 o’clock were measured from photographs of 189 prepubertal children with a validated history of digital or penile penetration and 197 children who denied previous sexual abuse. Statistical analyses were conducted to compare the mean values and hymenal symmetry between groups as well as to determine the sensitivity and specificity of various cutoff points.
Results
Comparison of the mean diameters demonstrated that children with a penetration history had a significantly larger transverse opening than nonabused children when examined in the knee–chest position (5.6 vs. 4.6 mm). However, there was extensive overlap in measurements between the 2 groups. No significant differences were noted between groups in the size of the vertical diameter, the amount of tissue present inferiorly or laterally, or the symmetry of the hymen in either position.
Children with previous penetration were more likely than nonabused children to have a horizontal opening measuring >6.5 mm in the knee–chest position, but the sensitivity and specificity of this test were low (29 % and 86 %, respectively).
Higher values had better specificity but very low sensitivity. Less than 1.0 mm of hymenal tissue was detected at 6 o’clock only in those with a history of penetration (100 % specificity), but the sensitivity was low (1–2 %) (Berenson et al. 2002).
Conclusion
Most hymenal measurements lack adequate sensitivity or specificity to be used to confirm previous penetration. Less than 1.0 mm of hymenal tissue at 6 o’clock was detected only in victims of penetrative abuse, but the usefulness of this test is limited by the rarity of this finding.
Box 4.2. Classification of Genital Injuries Caused by Sexual Assault*
To ease the clinician’s evaluation of the damage and type of intervention, or immediate referral at a tertiary care Pediatric Surgical Unit (Sham et al. 2013):
- 1.
First-degree tear: laceration is limited to the fourchette and superficial perineal skin or vaginal mucosa. Perineal body intact.
- 2.
Second-degree tear: laceration extends beyond the fourchette, perineal skin, and vaginal mucosa to perineal muscles and fascia, but not the anal sphincter. Perineal body involved.
- 3.
Third-degree tear: the fourchette, perineal skin, vaginal mucosa, muscles, and anal sphincter are torn:
- (a)
Partial tear of the external anal sphincter involving less than 50 % thickness
- (b)
>50 % of external anal sphincter thickness torn
- (c)
Internal anal sphincter torn
- (a)
- 4.
Fourth-degree tear: the fourchette, perineal skin, vaginal mucosa, muscles, anal sphincter, and anorectal mucosa are torn.
* It is worth noting that the word “vulva” is not mentioned in spite of the fact that the vulva is frequently, if not always, involved during sexual assaults and that the fourchette is by definition part of the vulva.
Key Learning Points
Vulvar and genital STI in children should always raise the question of sexual abuse.
When one infection is diagnosed, screening for other STI with different incubation time and less obvious clinical signs must be done.
Immediate accurate video recording of the vulvar/genital lesions and audio recording of the child wording (everybody has a mobile phone!) at the very first visit is essential to:
- 1.
Objectively document the findings
- 2.
Avoid repeated examination and questioning that may further traumatize the unfortunate child
- 3.
Prepare a solid, impeccable material for the legal investigation
- 1.
Figure 4.2 presents a typical case of hymenal damage/change after sexual abuse.
Fig 4.2
Hymen modifications: reduced posterior hymenal ring with “rolling.” A potential sexual abuse is to be considered. Gentle and respectful clinical examination is essential. A complete immediate photographic documentation with audio recording of the child wording is mandatory at the very first visit (Picture: courtesy of Metella Dei, MD)
Prospective studies with careful follow–up of girls who underwent penetrative abuse focusing on vulvar pain/vestibulodynia and vulvodynia are lacking.
Retrospective studies show conflicting results. Some research groups failed to show an association between sexual abuse and later vulvodynia. The lack of association between sexual abuse and vulvodynia, or vulvar pruritus, was confirmed in more recent studies (Cohen-Sacher et al. 2015).
Other groups, probably because of methodologically more accurate retrospective studies, do instead strongly support the association between sexual abuse and subsequent vulvodynia (Harlow and Stewart 2005; Khandker et al. 2014).
In 2000–2003, Harlow and Stewart identified 125 women experiencing symptoms of vulvar pain consistent with vulvodynia and 125 age- and community-matched controls from the Boston, Massachusetts, area general population. Telephone-administered questionnaires were used to obtain medical, psychiatric, and reproductive histories. Self-administered surveys assessed childhood exposure (age <12 years) to physical and sexual abuse and too poor family support. After authors’ adjustment for socioeconomic position, women with vulvar pain versus controls were 2.6 times more likely to report never/rarely receiving childhood family support, such as comfort, encouragement, and love [95 % confidence interval (CI): 1.3, 5.1].
Adult–onset vulvodynia was strongly associated with abuse as a child more than a few times physically [odds ratio (OR) = 4.1, 95 % CI: 1.7, 10.0] or sexually (OR = 6.5, 95 % CI: 1.2, 35.1). When abused women were compared with those with no history of abuse, the association was largely confined to those harmed by a primary family member (OR = 3.6, 95 % CI: 1.6, 8.0 for physical abuse; OR = 4.4, 95 % CI: 0.9, 22.9 for sexual abuse). The association is impressive and alarming.
Khandker et al. (2014) found that, among women with a history of severe childhood abuse, those with vulvodynia had three times the odds of living in fear of any abuse compared to women without vulvodynia (95 % CI: 1.0, 11.0), after adjustment for childhood poverty. Among women with no history of childhood abuse, those with vulvodynia had over six times the odds of antecedent mood disorder compared to women without vulvodynia (95 % CI: 1.9,19.6) (Khandker et al. 2014).
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