CED
Clinical dermatology • Original article
Clinical and Experimental Dermatology
Dysaesthetic penoscrotodynia may be a somatoform disorder: results from a two-centre retrospective case series M. C. Anyasodor,1 R. E. Taylor,2 A. Bewley1 and J. M. R. Goulding3 1 Department of Dermatology, Barts Health NHS Trust, London, UK; 2Barts and The London School of Medicine and Dentistry Centre for Psychiatry, London, UK; and 3Department of Dermatology, Heart of England NHS Foundation Trust, Birmingham, UK
doi:10.1111/ced.12824
Summary
Background. Dysaesthetic penoscrotodynia (DPSD) is a poorly understood disorder, in which men experience distressing symptoms such as burning pain in their genital skin. Drugs for neuropathic pain are often used, but with little success. Aim. To review a series of patients with DPSD to highlight common themes and response to treatment. Methods. Ten consecutive patients with DPSD were identified from specialist male genital dermatology and psychodermatology clinics at two centres. Clinical details, including psychiatric history, were reviewed retrospectively. Patients with no previously diagnosed psychiatric illness completed either the Generalized Anxiety Disorder (GAD)-7 scale and the Patient Health Questionnaire (PHQ)-9 depression scale, or the Hospital Anxiety and Depression Scale (HADS) and the Dermatology Life Quality Index (DLQI). Results. Of the 10 patients, 9 had known or newly diagnosed psychopathology. All patients were offered psychodermatological treatment, of which 7 of 10 accepted. All of those who accepted psychodermatological treatment experienced an improvement in their genital symptoms. When post-treatment scores were collected, improvement in psychiatric symptoms accompanied improvement in genital symptoms. Conclusions. Psychopathology is almost invariably present in individuals with DPSD, yet these patients rarely volunteer such information. DPSD is most likely to constitute a functional somatic symptom disorder, hence psychodermatological treatment is indicated for its management. This concept reflects a significant change in the approach to this condition. [Corrections added on 24 March 2016: In the results section the text in the second line was changed to ‘of which 7 of 10 accepted’ from ‘was accepted by seven patients’].
Introduction Dysaesthetic penoscrotodynia (DPSD) is a chronic debilitating condition in which men experience distressing sensations in their genital skin, such as burning pain or dysaesthesia.1–3 Symptoms commonly affect the glans penis, but can spread to involve the scrotum, groin, perineum, natal cleft and Correspondence: Dr Jon Goulding, Dermatology Department, Solihull Hospital, Lode Lane, Birmingham B91 2JL, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 25 August 2015
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Clinical and Experimental Dermatology (2016) 41, pp474–479
thighs. Patients often report redness or other visible abnormalities of the affected areas, but when examined clinically, the genitals have a normal appearance. The differential diagnosis for DPSD should include atopic eczema, psoriasis, seborrhoeic dermatitis, sexually transmitted infection, allergic contact dermatitis, chronic urticaria, necrolytic migratory erythema, penile intraepithelial neoplasia, pudendal neuralgia and other entities. There is no demonstrable evidence of underlying organic pathology in DPSD, and it is considered a diagnosis of exclusion. DPSD is a poorly understood disorder, with remarkably few papers on the subject published in the literature. DPSD shares several overlapping features with
ª 2016 British Association of Dermatologists
Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
red scrotum syndrome (RSS),4 and in many cases these terms may be referring to the same condition. It has been suggested that RSS is caused or exacerbated by overuse of topical corticosteroids,4–6 or that it may represent a localized form of erythromelalgia.7 Some authors have reported an improvement in RSS symptoms with use of doxycycline, implicating a rosacealike pathogenesis.6,8,9 Most practitioners subscribe to the theory that DPSD reflects a neuropathic pain disorder,1,3 hence the range of topical or systemic treatments usually employed, which includes lidocaine gel or doxepin cream, or oral amitriptyline, gabapentin, pregabalin or duloxetine. Practical experience with these drugs suggests that they are rarely successful in resolving symptoms, which can lead to a sense of despair for both patients and clinicians alike. We present a series of 10 patients diagnosed with DPSD, highlighting the almost invariable background psychopathology found in such cases, and the integral role for psychodermatological treatment in successfully managing the condition. We posit the theory that DPSD, rather than reflecting a neuropathic pain disorder, constitutes a functional somatic symptom disorder.
Methods Ethics approval was not required, as this was a retrospective, anonymized, descriptive case series. Patients
The study enrolled consecutive patients with DPSD from among men referred to specialist male genital dermatology and psychodermatology clinics in two hospital trusts over an 18-month period. Men diagnosed with DPSD by the two lead clinicians during this period were identified from the respective clinic databases. Patients were seen by the same two lead clinicians throughout, both of whom have a specialist interest and expertise in psychodermatology. Case notes were reviewed retrospectively for the following: demographics, disease duration, clinical details including psychiatric history, previous consultations and previous treatments. Validated questionnaires screening for anxiety, depression and/or impact on quality of life were administered in clinic to the majority of patients before further treatment, and to a minority afterwards. The Generalized Anxiety Disorder (GAD)-7 scale10 and the Patient Health Questionnaire (PHQ)-9 depression scale11 were used preferentially in one clinic (male genital dermatology clinic in Birmingham). The Hospital Anxiety and Depression Scale (HADS)12 and the
ª 2016 British Association of Dermatologists
Dermatology Life Quality Index (DLQI)13 were used in the other centre (psychodermatology clinic in London). The treatments offered and the response of genital symptoms to the treatments was recorded. Outcomes were determined from patients’ self-reported assessment of their genital symptoms and post-treatment screening questionnaire scores where available.
Results Ten consecutive patients with DPSD were identified, of whom eight were white British and two were British Asian (Table 1). Mean age at referral was 49.6 years (range 34–86) and mean duration of symptoms at referral was 34.2 months (range 4–96). A range of diagnoses had been postulated for the patients before referral to a specialist clinic. Prior to referral, seven men had already consulted general dermatologists and three had seen urologists, while two patients had been seen by both general dermatologists and urologists. All of the men had previously been offered multiple topical and oral treatments for DPSD, including drugs for neuropathic pain, with little or no relief in their symptoms (Table 2). Four patients (patients 3, 4, 5 and 10) had previously been diagnosed with psychiatric disorders. One (patient 3) had recently diagnosed depression, two (patients 5 and 10) had both depression and anxiety, and the fourth (patient 4) was under the care of a psychiatrist for bipolar disorder and generalized anxiety disorder. Five further patients with no prior diagnosis of psychiatric illness had evidence of depression or anxiety detected after validated screening questionnaires were administered (patients 1, 2, 6, 7 and 8). In all, 9 of the 10 patients with DPSD had evidence of psychopathology. Only one patient (patient 9) did not meet the criteria for diagnosis of anxiety or depression, although he was very close to the diagnostic threshold. All patients were offered psychiatric and/or psychological treatment. The patient with bipolar disorder had his existing drugs altered under the care of his psychiatrist (see Table 2). The other nine patients were offered selective serotonin reuptake inhibitors (SSRIs) at standard doses, and one of these patients was also offered low-intensity cognitive behavioural therapy (CBT) via his local Improving Access to Psychological Therapies (IAPT) team. Seven of the 10 patients accepted psychodermatological treatment, all of whom reported a noticeable improvement in their genital symptoms. One patient’s symptoms resolved completely, while another’s resolved almost com-
Clinical and Experimental Dermatology (2016) 41, pp474–479
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Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
Table 1 Patient demographics, disease duration and past psychiatric/medical history. Age at referral, years
Ethnicity
Clinic type
1
43
British Asian
2
39
White British
Male genital dermatology, Birmingham
3
34
4
Duration of symptoms at referral
Initial skin diagnosis
Past psychiatric history
Other medical history
Previous consultations
8 months
PD
None
None
2 years
Balanitis
None
White British
4 months
PD
57
White British
4 years
NP
5
38
White British
8 years
PD
None (but brother has bipolar diisorder and mother committed suicide) Depression (recently diagnosed in primary care) Bipolar disorder, generalized anxiety disorder Anxiety, depression
1 dermatology, 1 urology 1 dermatology
6
86
White British
3 years
None
None
7 8
46 59
White British White British
3 years 2.5 years
None None
9
37
White British
1 year
Uncertain Balanitis, LS RSS
10
57
British Asian
4 years
RSS
Anxiety, depression
Patient
Psychodermatology, London
None
None
Primary care only
Hypertension, dyslipidaemia, gout, asthma Atopic eczema, irritant contact dermatitis, tinnitus Prostate carcinoma, laryngeal carcinoma, peptic ulcer disease, erectile dysfunction Globus hystericus Post-viral fatigue, genital thrush Ichthyosis vulgaris, genital warts Chronic pain, venous eczema, hyperhidrosis
2 dermatology, 2 urology 1 urology
Several dermatology
1 dermatology 1 neurology 1 dermatology Several dermatology
LS, lichen sclerosus; PD, penile dysaesthesia; NP, neuropathic pain; RSS, red scrotum syndrome.
pletely. Post-treatment questionnaires were not previously offered routinely to all patients, although this now forms part of our standard practice. However, these questionnaires were administered at intervals to three of the seven patients who accepted treatment, all of whom showed evidence of concurrent improvement in psychiatric symptoms. The mean follow-up period was 21.7 months (range 3–76).
Discussion Although the literature on DPSD is remarkably scant, the condition has traditionally been viewed as a neuropathic pain disorder,1,3 or marginalized as ‘dermatological nondisease’.14 In our clinical experience, treating DPSD with multiple drugs for neuropathic pain is rarely beneficial, as well as being fraught with potential side-effects and very costly. Patients with chronic pain syndromes affecting any anatomical site, including the genitals, often have significant levels of associated psychological distress.1,3,14 A high frequency of overt psychiatric comorbidity has also been demonstrated in those diagnosed with RSS,9 and a few
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scattered case reports have reported response of patients with DPSD to SSRIs.2,3 However, the intrinsic role of psychiatric illness as an aetiopathogenetic factor in DPSD has been overlooked. We have demonstrated the almost invariable presence of psychopathology in our patients with DPSD. Psychodermatological treatment led to improvements in genital symptoms in all patients who accepted it, with accompanying improvement in their psychiatric symptoms where tested. This is no mean feat when it is considered that the patients had had their distressing symptoms for a mean of 2.5 years, and for a condition that is currently viewed as largely untreatable. It is notable that psychopathology was discovered for the first time in half of our patients only after the administration of validated screening questionnaires. Men find it difficult enough to approach healthcare professionals about genital problems, and this is compounded by potentially awkward conversations about mood issues. Thus, it is crucial not to make assumptions about patients, and to use screening questionnaires as a matter of routine to facilitate detection and discussion of possible underlying psychopathology.
ª 2016 British Association of Dermatologists
Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
Table 2 Previous treatments, screening results, psychiatric treatments offered and outcomes.
Patient
Previous treatments
1
Lidocaine 5% plasters, oral amitriptyline Emollients, soap substitutes, doxepin cream, Daktacort cream, amitriptyline Soap substitute, emollients, lidocaine 5% plasters, lubricant for intercourse Lidocaine ointment, duloxetine, lidocaine 4% cream
2
3
4
5
6 7
8
9
10
Soap substitutes, emollients, lidocaine plasters Amitriptyline, clindamycin E45 cream, menthol in aqueous, amitriptyline, carbamazepine, gabapentin, pregabalin, physiotherapy, acupuncture, duloxetine, sodium valproate, antifungal cream, doxepin cream, Balneum Plus cream Trimovate cream, Amitriptyline, doxepin cream, Balneum Plus bath oil, Dermovate ointment, Lotriderm cream, Eumovate ointment E45, Calmurid cream, anti-depressant, pregabalin, doxepin EMLA, doxepin, Elocon, Dermovate, Fucidin cream, Balneum; refused gabapentin
Pretreatment questionnaires
Psychiatric treatments offered
Genital symptoms
GAD-7 = 11 (moderate), PHQ-9 = 7 (mild) GAD-7 = 4 (nil), PHQ-9 = 9 (mild)
SSRI – declined
Lost to follow-up
Citalopram
Near-complete resolution
Not administered (recent diagnosis of depression)
Sertraline
Not administered (known bipolar disorder)
Already on lithium, quetiapine, diazepam and mirtazapine; short trial of duloxetine not tolerated; quetiapine reduced and mirtazapine changed to dosulepin Escitalopram + lowintensity cognitive behavioural therapy Citalopram
Follow-up, months 3
Post-treatment questionnaires NA
27
GAD-7 = 3 (nil), PHQ-9 = 3 (nil)
Resolved
4
Not administered
Improved
9
Not administered
Improved
18
Improved
11
GAD-7 = 5 (mild), PHQ-9 = 7 (mild) Not administered
Citalopram
Improved
18
HADS = 17, A = 7 (mild), D = 10 (mild), DLQI = 11
HADS = 12 (case), A = 4 (normal), D = 8 (mild), DLQI = 0
Citalopram
Improved
76
Not administered
HADS = 7 (normal), DLQI = 2 (small)
SSRI – declined
No improvement
24
NA
Declined (known depression and anxiety)
SSRI – declined
No improvement
27
NA
GAD-7 = 13 (moderate), PHQ-9 = 11 (moderate) GAD-7 = 1 (nil), PHQ9 = 11 (moderate) HADS = 24 (case), A = 10 (mild), D = 14 (moderate), DLQI = 22 (extremely large)
A, anxiety component of HADS (score range 0–21); D, depression component of HADS (score range 0–21); DLQI, Dermatology Life Quality Index (score range 0–30);13 GAD-7, Generalized Anxiety Disorder scale (score range 0–21);10 HADS, Hospital Anxiety and Depression Scale;12 NA, not applicable; PHQ-9, Patient Health Questionnaire depression scale (score range 0–27);11 SSRI, selective serotonin reuptake inhibitor.
We posit the theory that DPSD is most likely to represent a functional somatic symptom disorder, which reflects a significant change in the approach to this
ª 2016 British Association of Dermatologists
condition. DPSD could be classified as a ‘persistent somatoform pain disorder’ or ‘other somatoform disorder’ in the International Classification of Diseases and
Clinical and Experimental Dermatology (2016) 41, pp474–479
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Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
Related Health Problems, 10th Revision (ICD-10)15 or within the ‘somatic symptom and related disorders’ in the Diagnostic and Statistical Manual of Mental Disorders (DSM-5) of the Fifth Edition.16 Somatoform disorders are frequently comorbid with anxiety and depression, which probably accounts for the high prevalence of these disorders in our cohort of patients. Thus, the existence of anxiety and/or depression in such patients does not constitute a final diagnosis in its own right, but rather it points to the possibility of an underlying somatoform disorder. The question of cause and effect is of course difficult to disentangle; does the altered mood state lead to somatoform pain, or vice versa? Fortunately, appropriate treatment helps both. Although the large majority of our patients were treated with oral psychiatric medications, there is an equally valid case for instituting psychological treatment. A recent report illustrates the potential benefit of so-called ‘journey psychotherapy’ in a series of men with refractory genital pain.17 DPSD in men is analogous to vulvodynia in women, and there have been calls to use a similar classification system.3 Women with vulvodynia experience chronic burning vulval pain and frequently have psychosocial comorbidities.1,18,19 It has already been suggested that vulvodynia may also reflect a somatization disorder,18 yet dermatologists still seem to persist with largely useless treatments directed against neuropathic pain for this condition too. We encourage further investigation, similar to the present study but involving patients with vulvodynia, to test and refine this hypothesis. We acknowledge several shortcomings in our study. The sample was small, and patients were drawn from specialist settings that may not be representative of the wider population. It is possible that some patients with DPSD were not captured in the study timeframe, although this is unlikely given the detailed clinic databases maintained at both centres. The retrospective nature of the study prevented the acquisition of a complete dataset and introduced the possibility of bias. There is no available validated scale for the measurement of DPSD symptom severity, so we were reliant on subjective patient-reported outcomes. However, we would argue that this adds to, rather than detracts from, our findings, as the majority of patients with DPSD are somewhat entrenched in their symptom perceptions, hence any improvement is not volunteered willingly. There was a variable response to treatment, with one patient experiencing complete resolution in his genital symptoms while others had some residual symptoms. However, average follow-up was short, and
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Clinical and Experimental Dermatology (2016) 41, pp474–479
we would expect further improvement to be attained over longer periods. Our data suggest that larger, multicentre prospective studies of this type would be worthwhile, perhaps also incorporating a specific somatoform disorder screening tool such as the PHQ-15.20 In the meantime, it seems appropriate to actively screen patients with DPSD for underlying psychopathology, and to institute psychiatric or psychological treatment as indicated. Given patients’ frequent reluctance to be seen by psychiatrists, this may well need to be initiated by the treating dermatologist. Having said this, psychiatric referral is mandated if patients display evidence of severe psychiatric illness and/or present a high risk for suicide. Patients may be served best by being referred to, and treated in, an integrated multidisciplinary psychodermatology clinic.
What’s already known about this topic? Dysaesthetic
penoscrotodynia is a poorly understood debilitating condition, in which men experience burning pain in their genital skin. Drugs for neuropathic pain are usually used in the treatment of DPSD, although these are rarely effective.
What does this study add? Psychopathology is almost invariably present
in individuals with DPSD, and those who accept psychodermatological treatment may experience an improvement in their genital symptoms. We propose that DPSD constitutes a functional somatic symptom disorder, a concept that reflects a significant change in the approach to this condition.
References 1 Lynch PJ, Edwards L. Chronic idiopathic mucocutaneous pain syndromes: vulvodynia, penodynia, and scrotodynia. In: Practical Psychodermatology (Bewley A, Taylor RE, Reichenberg JS, Magid M, eds), 1st edn. Oxford: John Wiley & Sons Ltd, 2014: 176–9.
ª 2016 British Association of Dermatologists
Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
2 Markos AR. The male genital skin burning syndrome (dysaesthetic peno/scroto-dynia). Int J STD AIDS 2002; 13: 271–2. 3 Markos AR. Dysaesthetic penoscrotodynia: nomenclature, classification, diagnosis and treatment. Int J STD AIDS 2011; 22: 483–7. 4 Fisher BK. The red scrotum syndrome. Cutis 1997; 60: 139–41. 5 Rapaport MJ, Rapaport V. The red skin syndromes: corticosteroid addiction and withdrawal. Exp Rev Dermatol 2006; 1: 547–61. 6 Abbas O, Kibbi AG, Chedraoui A, Ghosn S. Red scrotum syndrome: successful treatment with oral doxycycline. J Dermatol Treat 2008; 19: 371–2. 7 Prevost N, English JC. Red scrotal syndrome: a localized phenotypical expression of erythromelalgia. J Drugs Dermatol 2007; 6: 935–6. 8 Byun JW, Hong WK, Han SH et al. Red scrotum syndrome: successful treatment with oral doxycycline. Int J Dermatol 2012; 51: 362–3. 9 Narang T, Dogra S, Kumaran MS, Saikia UN. Red scrotum syndrome: a clinicopathological study. Br J Dermatol 2013; 169(Suppl.): 42. 10 Spitzer RL, Kroenke K, Williams JW, L€ owe B. A brief measure for assessing generalized anxiety disorder: the GAD-7. Arch Intern Med 2006; 166: 1092–7. 11 Kroenke K, Spitzer RL, Williams JBW. The PHQ-9: validity of a brief depression severity measure. J Gen Intern Med 2001; 16: 606–13. 12 Snaith RP. The hospital anxiety and depression scale. Health Qual Life Outcomes 2003; 1: 29.
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13 Finlay AY, Khan GK. Dermatology Life Quality Index (DLQI) – a simple practical measure for routine clinical use. Clin Exp Dermatol 1994; 19: 210–16. 14 Bunker CB, Neill SM. Dermatological non-disease, dysaesthesia and chronic pain syndromes. In: Rook’s Textbook of Dermatology (Burns T, Breathnach S, Cox N, Griffiths C, eds), 8th edn. Oxford: Wiley-Blackwell, 2010: 71.51–2. 15 World Health Organisation. The ICD-10 classification of mental and behavioural disorders: clinical descriptions and diagnostic guidelines, 2014. Available at: http:// www.who.int/classifications/icd/en/bluebook.pdf (accessed 22 April 2015). 16 American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders (DSM), 5th edn. Arlington, VA: American Psychiatric Publishing, 2013: 74–85. 17 Naim M, Ende D. A new approach to the treatment of non-specific male genital pain. BJU Int 2011; 107 (Suppl.): 34–7. 18 Mascherpa F, Bogliatto F, Lynch PJ et al. Vulvodynia as a possible somatization disorder. More than just an opinion. J Reprod Med 2007; 52: 107–10. 19 Iglesias-Rios L, Harlow SD, Reed BD. Depression and posttraumatic stress disorder among women with vulvodynia: evidence from the population-based woman to woman health study. J Womens Health (Larchmt) 2015; 24: 557–62. 20 Kroenke K, Spitzer RL, Williams JBW. The PHQ-15: validity of a new measure for evaluating the severity of somatic symptoms. Psychosom Med 2002; 64: 258–66.
Clinical and Experimental Dermatology (2016) 41, pp474–479
479
Clinical dermatology • Original article
Clinical and Experimental Dermatology
Dysaesthetic penoscrotodynia may be a somatoform disorder: results from a two-centre retrospective case series M. C. Anyasodor,1 R. E. Taylor,2 A. Bewley1 and J. M. R. Goulding3 1 Department of Dermatology, Barts Health NHS Trust, London, UK; 2Barts and The London School of Medicine and Dentistry Centre for Psychiatry, London, UK; and 3Department of Dermatology, Heart of England NHS Foundation Trust, Birmingham, UK
doi:10.1111/ced.12824
Summary
Background. Dysaesthetic penoscrotodynia (DPSD) is a poorly understood disorder, in which men experience distressing symptoms such as burning pain in their genital skin. Drugs for neuropathic pain are often used, but with little success. Aim. To review a series of patients with DPSD to highlight common themes and response to treatment. Methods. Ten consecutive patients with DPSD were identified from specialist male genital dermatology and psychodermatology clinics at two centres. Clinical details, including psychiatric history, were reviewed retrospectively. Patients with no previously diagnosed psychiatric illness completed either the Generalized Anxiety Disorder (GAD)-7 scale and the Patient Health Questionnaire (PHQ)-9 depression scale, or the Hospital Anxiety and Depression Scale (HADS) and the Dermatology Life Quality Index (DLQI). Results. Of the 10 patients, 9 had known or newly diagnosed psychopathology. All patients were offered psychodermatological treatment, of which 7 of 10 accepted. All of those who accepted psychodermatological treatment experienced an improvement in their genital symptoms. When post-treatment scores were collected, improvement in psychiatric symptoms accompanied improvement in genital symptoms. Conclusions. Psychopathology is almost invariably present in individuals with DPSD, yet these patients rarely volunteer such information. DPSD is most likely to constitute a functional somatic symptom disorder, hence psychodermatological treatment is indicated for its management. This concept reflects a significant change in the approach to this condition. [Corrections added on 24 March 2016: In the results section the text in the second line was changed to ‘of which 7 of 10 accepted’ from ‘was accepted by seven patients’].
Introduction Dysaesthetic penoscrotodynia (DPSD) is a chronic debilitating condition in which men experience distressing sensations in their genital skin, such as burning pain or dysaesthesia.1–3 Symptoms commonly affect the glans penis, but can spread to involve the scrotum, groin, perineum, natal cleft and Correspondence: Dr Jon Goulding, Dermatology Department, Solihull Hospital, Lode Lane, Birmingham B91 2JL, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 25 August 2015
474
Clinical and Experimental Dermatology (2016) 41, pp474–479
thighs. Patients often report redness or other visible abnormalities of the affected areas, but when examined clinically, the genitals have a normal appearance. The differential diagnosis for DPSD should include atopic eczema, psoriasis, seborrhoeic dermatitis, sexually transmitted infection, allergic contact dermatitis, chronic urticaria, necrolytic migratory erythema, penile intraepithelial neoplasia, pudendal neuralgia and other entities. There is no demonstrable evidence of underlying organic pathology in DPSD, and it is considered a diagnosis of exclusion. DPSD is a poorly understood disorder, with remarkably few papers on the subject published in the literature. DPSD shares several overlapping features with
ª 2016 British Association of Dermatologists
Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
red scrotum syndrome (RSS),4 and in many cases these terms may be referring to the same condition. It has been suggested that RSS is caused or exacerbated by overuse of topical corticosteroids,4–6 or that it may represent a localized form of erythromelalgia.7 Some authors have reported an improvement in RSS symptoms with use of doxycycline, implicating a rosacealike pathogenesis.6,8,9 Most practitioners subscribe to the theory that DPSD reflects a neuropathic pain disorder,1,3 hence the range of topical or systemic treatments usually employed, which includes lidocaine gel or doxepin cream, or oral amitriptyline, gabapentin, pregabalin or duloxetine. Practical experience with these drugs suggests that they are rarely successful in resolving symptoms, which can lead to a sense of despair for both patients and clinicians alike. We present a series of 10 patients diagnosed with DPSD, highlighting the almost invariable background psychopathology found in such cases, and the integral role for psychodermatological treatment in successfully managing the condition. We posit the theory that DPSD, rather than reflecting a neuropathic pain disorder, constitutes a functional somatic symptom disorder.
Methods Ethics approval was not required, as this was a retrospective, anonymized, descriptive case series. Patients
The study enrolled consecutive patients with DPSD from among men referred to specialist male genital dermatology and psychodermatology clinics in two hospital trusts over an 18-month period. Men diagnosed with DPSD by the two lead clinicians during this period were identified from the respective clinic databases. Patients were seen by the same two lead clinicians throughout, both of whom have a specialist interest and expertise in psychodermatology. Case notes were reviewed retrospectively for the following: demographics, disease duration, clinical details including psychiatric history, previous consultations and previous treatments. Validated questionnaires screening for anxiety, depression and/or impact on quality of life were administered in clinic to the majority of patients before further treatment, and to a minority afterwards. The Generalized Anxiety Disorder (GAD)-7 scale10 and the Patient Health Questionnaire (PHQ)-9 depression scale11 were used preferentially in one clinic (male genital dermatology clinic in Birmingham). The Hospital Anxiety and Depression Scale (HADS)12 and the
ª 2016 British Association of Dermatologists
Dermatology Life Quality Index (DLQI)13 were used in the other centre (psychodermatology clinic in London). The treatments offered and the response of genital symptoms to the treatments was recorded. Outcomes were determined from patients’ self-reported assessment of their genital symptoms and post-treatment screening questionnaire scores where available.
Results Ten consecutive patients with DPSD were identified, of whom eight were white British and two were British Asian (Table 1). Mean age at referral was 49.6 years (range 34–86) and mean duration of symptoms at referral was 34.2 months (range 4–96). A range of diagnoses had been postulated for the patients before referral to a specialist clinic. Prior to referral, seven men had already consulted general dermatologists and three had seen urologists, while two patients had been seen by both general dermatologists and urologists. All of the men had previously been offered multiple topical and oral treatments for DPSD, including drugs for neuropathic pain, with little or no relief in their symptoms (Table 2). Four patients (patients 3, 4, 5 and 10) had previously been diagnosed with psychiatric disorders. One (patient 3) had recently diagnosed depression, two (patients 5 and 10) had both depression and anxiety, and the fourth (patient 4) was under the care of a psychiatrist for bipolar disorder and generalized anxiety disorder. Five further patients with no prior diagnosis of psychiatric illness had evidence of depression or anxiety detected after validated screening questionnaires were administered (patients 1, 2, 6, 7 and 8). In all, 9 of the 10 patients with DPSD had evidence of psychopathology. Only one patient (patient 9) did not meet the criteria for diagnosis of anxiety or depression, although he was very close to the diagnostic threshold. All patients were offered psychiatric and/or psychological treatment. The patient with bipolar disorder had his existing drugs altered under the care of his psychiatrist (see Table 2). The other nine patients were offered selective serotonin reuptake inhibitors (SSRIs) at standard doses, and one of these patients was also offered low-intensity cognitive behavioural therapy (CBT) via his local Improving Access to Psychological Therapies (IAPT) team. Seven of the 10 patients accepted psychodermatological treatment, all of whom reported a noticeable improvement in their genital symptoms. One patient’s symptoms resolved completely, while another’s resolved almost com-
Clinical and Experimental Dermatology (2016) 41, pp474–479
475
Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
Table 1 Patient demographics, disease duration and past psychiatric/medical history. Age at referral, years
Ethnicity
Clinic type
1
43
British Asian
2
39
White British
Male genital dermatology, Birmingham
3
34
4
Duration of symptoms at referral
Initial skin diagnosis
Past psychiatric history
Other medical history
Previous consultations
8 months
PD
None
None
2 years
Balanitis
None
White British
4 months
PD
57
White British
4 years
NP
5
38
White British
8 years
PD
None (but brother has bipolar diisorder and mother committed suicide) Depression (recently diagnosed in primary care) Bipolar disorder, generalized anxiety disorder Anxiety, depression
1 dermatology, 1 urology 1 dermatology
6
86
White British
3 years
None
None
7 8
46 59
White British White British
3 years 2.5 years
None None
9
37
White British
1 year
Uncertain Balanitis, LS RSS
10
57
British Asian
4 years
RSS
Anxiety, depression
Patient
Psychodermatology, London
None
None
Primary care only
Hypertension, dyslipidaemia, gout, asthma Atopic eczema, irritant contact dermatitis, tinnitus Prostate carcinoma, laryngeal carcinoma, peptic ulcer disease, erectile dysfunction Globus hystericus Post-viral fatigue, genital thrush Ichthyosis vulgaris, genital warts Chronic pain, venous eczema, hyperhidrosis
2 dermatology, 2 urology 1 urology
Several dermatology
1 dermatology 1 neurology 1 dermatology Several dermatology
LS, lichen sclerosus; PD, penile dysaesthesia; NP, neuropathic pain; RSS, red scrotum syndrome.
pletely. Post-treatment questionnaires were not previously offered routinely to all patients, although this now forms part of our standard practice. However, these questionnaires were administered at intervals to three of the seven patients who accepted treatment, all of whom showed evidence of concurrent improvement in psychiatric symptoms. The mean follow-up period was 21.7 months (range 3–76).
Discussion Although the literature on DPSD is remarkably scant, the condition has traditionally been viewed as a neuropathic pain disorder,1,3 or marginalized as ‘dermatological nondisease’.14 In our clinical experience, treating DPSD with multiple drugs for neuropathic pain is rarely beneficial, as well as being fraught with potential side-effects and very costly. Patients with chronic pain syndromes affecting any anatomical site, including the genitals, often have significant levels of associated psychological distress.1,3,14 A high frequency of overt psychiatric comorbidity has also been demonstrated in those diagnosed with RSS,9 and a few
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scattered case reports have reported response of patients with DPSD to SSRIs.2,3 However, the intrinsic role of psychiatric illness as an aetiopathogenetic factor in DPSD has been overlooked. We have demonstrated the almost invariable presence of psychopathology in our patients with DPSD. Psychodermatological treatment led to improvements in genital symptoms in all patients who accepted it, with accompanying improvement in their psychiatric symptoms where tested. This is no mean feat when it is considered that the patients had had their distressing symptoms for a mean of 2.5 years, and for a condition that is currently viewed as largely untreatable. It is notable that psychopathology was discovered for the first time in half of our patients only after the administration of validated screening questionnaires. Men find it difficult enough to approach healthcare professionals about genital problems, and this is compounded by potentially awkward conversations about mood issues. Thus, it is crucial not to make assumptions about patients, and to use screening questionnaires as a matter of routine to facilitate detection and discussion of possible underlying psychopathology.
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Dysaesthetic penoscrotodynia may be a somatoform disorder M. C. Anyasodor et al.
Table 2 Previous treatments, screening results, psychiatric treatments offered and outcomes.
Patient
Previous treatments
1
Lidocaine 5% plasters, oral amitriptyline Emollients, soap substitutes, doxepin cream, Daktacort cream, amitriptyline Soap substitute, emollients, lidocaine 5% plasters, lubricant for intercourse Lidocaine ointment, duloxetine, lidocaine 4% cream
2
3
4
5
6 7
8
9
10
Soap substitutes, emollients, lidocaine plasters Amitriptyline, clindamycin E45 cream, menthol in aqueous, amitriptyline, carbamazepine, gabapentin, pregabalin, physiotherapy, acupuncture, duloxetine, sodium valproate, antifungal cream, doxepin cream, Balneum Plus cream Trimovate cream, Amitriptyline, doxepin cream, Balneum Plus bath oil, Dermovate ointment, Lotriderm cream, Eumovate ointment E45, Calmurid cream, anti-depressant, pregabalin, doxepin EMLA, doxepin, Elocon, Dermovate, Fucidin cream, Balneum; refused gabapentin
Pretreatment questionnaires
Psychiatric treatments offered
Genital symptoms
GAD-7 = 11 (moderate), PHQ-9 = 7 (mild) GAD-7 = 4 (nil), PHQ-9 = 9 (mild)
SSRI – declined
Lost to follow-up
Citalopram
Near-complete resolution
Not administered (recent diagnosis of depression)
Sertraline
Not administered (known bipolar disorder)
Already on lithium, quetiapine, diazepam and mirtazapine; short trial of duloxetine not tolerated; quetiapine reduced and mirtazapine changed to dosulepin Escitalopram + lowintensity cognitive behavioural therapy Citalopram
Follow-up, months 3
Post-treatment questionnaires NA
27
GAD-7 = 3 (nil), PHQ-9 = 3 (nil)
Resolved
4
Not administered
Improved
9
Not administered
Improved
18
Improved
11
GAD-7 = 5 (mild), PHQ-9 = 7 (mild) Not administered
Citalopram
Improved
18
HADS = 17, A = 7 (mild), D = 10 (mild), DLQI = 11
HADS = 12 (case), A = 4 (normal), D = 8 (mild), DLQI = 0
Citalopram
Improved
76
Not administered
HADS = 7 (normal), DLQI = 2 (small)
SSRI – declined
No improvement
24
NA
Declined (known depression and anxiety)
SSRI – declined
No improvement
27
NA
GAD-7 = 13 (moderate), PHQ-9 = 11 (moderate) GAD-7 = 1 (nil), PHQ9 = 11 (moderate) HADS = 24 (case), A = 10 (mild), D = 14 (moderate), DLQI = 22 (extremely large)
A, anxiety component of HADS (score range 0–21); D, depression component of HADS (score range 0–21); DLQI, Dermatology Life Quality Index (score range 0–30);13 GAD-7, Generalized Anxiety Disorder scale (score range 0–21);10 HADS, Hospital Anxiety and Depression Scale;12 NA, not applicable; PHQ-9, Patient Health Questionnaire depression scale (score range 0–27);11 SSRI, selective serotonin reuptake inhibitor.
We posit the theory that DPSD is most likely to represent a functional somatic symptom disorder, which reflects a significant change in the approach to this
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condition. DPSD could be classified as a ‘persistent somatoform pain disorder’ or ‘other somatoform disorder’ in the International Classification of Diseases and
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Related Health Problems, 10th Revision (ICD-10)15 or within the ‘somatic symptom and related disorders’ in the Diagnostic and Statistical Manual of Mental Disorders (DSM-5) of the Fifth Edition.16 Somatoform disorders are frequently comorbid with anxiety and depression, which probably accounts for the high prevalence of these disorders in our cohort of patients. Thus, the existence of anxiety and/or depression in such patients does not constitute a final diagnosis in its own right, but rather it points to the possibility of an underlying somatoform disorder. The question of cause and effect is of course difficult to disentangle; does the altered mood state lead to somatoform pain, or vice versa? Fortunately, appropriate treatment helps both. Although the large majority of our patients were treated with oral psychiatric medications, there is an equally valid case for instituting psychological treatment. A recent report illustrates the potential benefit of so-called ‘journey psychotherapy’ in a series of men with refractory genital pain.17 DPSD in men is analogous to vulvodynia in women, and there have been calls to use a similar classification system.3 Women with vulvodynia experience chronic burning vulval pain and frequently have psychosocial comorbidities.1,18,19 It has already been suggested that vulvodynia may also reflect a somatization disorder,18 yet dermatologists still seem to persist with largely useless treatments directed against neuropathic pain for this condition too. We encourage further investigation, similar to the present study but involving patients with vulvodynia, to test and refine this hypothesis. We acknowledge several shortcomings in our study. The sample was small, and patients were drawn from specialist settings that may not be representative of the wider population. It is possible that some patients with DPSD were not captured in the study timeframe, although this is unlikely given the detailed clinic databases maintained at both centres. The retrospective nature of the study prevented the acquisition of a complete dataset and introduced the possibility of bias. There is no available validated scale for the measurement of DPSD symptom severity, so we were reliant on subjective patient-reported outcomes. However, we would argue that this adds to, rather than detracts from, our findings, as the majority of patients with DPSD are somewhat entrenched in their symptom perceptions, hence any improvement is not volunteered willingly. There was a variable response to treatment, with one patient experiencing complete resolution in his genital symptoms while others had some residual symptoms. However, average follow-up was short, and
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we would expect further improvement to be attained over longer periods. Our data suggest that larger, multicentre prospective studies of this type would be worthwhile, perhaps also incorporating a specific somatoform disorder screening tool such as the PHQ-15.20 In the meantime, it seems appropriate to actively screen patients with DPSD for underlying psychopathology, and to institute psychiatric or psychological treatment as indicated. Given patients’ frequent reluctance to be seen by psychiatrists, this may well need to be initiated by the treating dermatologist. Having said this, psychiatric referral is mandated if patients display evidence of severe psychiatric illness and/or present a high risk for suicide. Patients may be served best by being referred to, and treated in, an integrated multidisciplinary psychodermatology clinic.
What’s already known about this topic? Dysaesthetic
penoscrotodynia is a poorly understood debilitating condition, in which men experience burning pain in their genital skin. Drugs for neuropathic pain are usually used in the treatment of DPSD, although these are rarely effective.
What does this study add? Psychopathology is almost invariably present
in individuals with DPSD, and those who accept psychodermatological treatment may experience an improvement in their genital symptoms. We propose that DPSD constitutes a functional somatic symptom disorder, a concept that reflects a significant change in the approach to this condition.
References 1 Lynch PJ, Edwards L. Chronic idiopathic mucocutaneous pain syndromes: vulvodynia, penodynia, and scrotodynia. In: Practical Psychodermatology (Bewley A, Taylor RE, Reichenberg JS, Magid M, eds), 1st edn. Oxford: John Wiley & Sons Ltd, 2014: 176–9.
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2 Markos AR. The male genital skin burning syndrome (dysaesthetic peno/scroto-dynia). Int J STD AIDS 2002; 13: 271–2. 3 Markos AR. Dysaesthetic penoscrotodynia: nomenclature, classification, diagnosis and treatment. Int J STD AIDS 2011; 22: 483–7. 4 Fisher BK. The red scrotum syndrome. Cutis 1997; 60: 139–41. 5 Rapaport MJ, Rapaport V. The red skin syndromes: corticosteroid addiction and withdrawal. Exp Rev Dermatol 2006; 1: 547–61. 6 Abbas O, Kibbi AG, Chedraoui A, Ghosn S. Red scrotum syndrome: successful treatment with oral doxycycline. J Dermatol Treat 2008; 19: 371–2. 7 Prevost N, English JC. Red scrotal syndrome: a localized phenotypical expression of erythromelalgia. J Drugs Dermatol 2007; 6: 935–6. 8 Byun JW, Hong WK, Han SH et al. Red scrotum syndrome: successful treatment with oral doxycycline. Int J Dermatol 2012; 51: 362–3. 9 Narang T, Dogra S, Kumaran MS, Saikia UN. Red scrotum syndrome: a clinicopathological study. Br J Dermatol 2013; 169(Suppl.): 42. 10 Spitzer RL, Kroenke K, Williams JW, L€ owe B. A brief measure for assessing generalized anxiety disorder: the GAD-7. Arch Intern Med 2006; 166: 1092–7. 11 Kroenke K, Spitzer RL, Williams JBW. The PHQ-9: validity of a brief depression severity measure. J Gen Intern Med 2001; 16: 606–13. 12 Snaith RP. The hospital anxiety and depression scale. Health Qual Life Outcomes 2003; 1: 29.
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13 Finlay AY, Khan GK. Dermatology Life Quality Index (DLQI) – a simple practical measure for routine clinical use. Clin Exp Dermatol 1994; 19: 210–16. 14 Bunker CB, Neill SM. Dermatological non-disease, dysaesthesia and chronic pain syndromes. In: Rook’s Textbook of Dermatology (Burns T, Breathnach S, Cox N, Griffiths C, eds), 8th edn. Oxford: Wiley-Blackwell, 2010: 71.51–2. 15 World Health Organisation. The ICD-10 classification of mental and behavioural disorders: clinical descriptions and diagnostic guidelines, 2014. Available at: http:// www.who.int/classifications/icd/en/bluebook.pdf (accessed 22 April 2015). 16 American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders (DSM), 5th edn. Arlington, VA: American Psychiatric Publishing, 2013: 74–85. 17 Naim M, Ende D. A new approach to the treatment of non-specific male genital pain. BJU Int 2011; 107 (Suppl.): 34–7. 18 Mascherpa F, Bogliatto F, Lynch PJ et al. Vulvodynia as a possible somatization disorder. More than just an opinion. J Reprod Med 2007; 52: 107–10. 19 Iglesias-Rios L, Harlow SD, Reed BD. Depression and posttraumatic stress disorder among women with vulvodynia: evidence from the population-based woman to woman health study. J Womens Health (Larchmt) 2015; 24: 557–62. 20 Kroenke K, Spitzer RL, Williams JBW. The PHQ-15: validity of a new measure for evaluating the severity of somatic symptoms. Psychosom Med 2002; 64: 258–66.
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