Bladder Dysfunction in Behcet's Disease/Behcet Hastaliginda Mesane Disfonksiyonu.
Behcet's disease (BD) is a systemic vasculitis usually seen in young adults, which affects both venous and arterial vessels of the vascular system. Its etiology is still unknown. The patognomic symptoms are recurrent oral aphtae accompanies with at least two of these; positive pathergy test, skin lesions, recurrent genital ulcers and lesions in the eye. Urogenital involvement usually consists of genital ulcers, urethritis, epididymitis and recurrent cystitis. Bladder involvement is extremely rare and only several cases have been reported. The findings suggest that neurogenic bladder due to neuro-Behcet disease is characterized uro-dynamically by overactive detrusor in storage phase and detrusor sphincter dyssynergia in voiding phase 12. We aimed to present several aspects of bladder involvement of Behcet disease in this case report.
Keywords: Behcet, neurogenic bladder, overactive detrusor, anticholinergic
Behcet Hastaligi, vaskuler sistemin hem arteryel hem de venoz damarlarini tutan, genellikle genc eriskinlerde gorulen sistemik bir vaskulittir. Etyolojisi halen bilinmemektedir. Patognomonik belirtileri; rekurren oral aft ile birlikte pozitif paterji testi, deri lezyonlari, rekurren genital ulserler ve goz lezyonlarindan en az ikisinin varligidir. Urogenital tutulum genellikle genital ulserler, uretrit, epididimit ve rekurren sistiti icerir. Mesane tutulumu oldukca nadirdir ve sadece birkac vaka rapor edilmistir. Urodinamik olarak noro Behcet hastaliginda gelisen norojenik mesane bulgulari; depolama fazinda asiri aktif mesane ve bosaltim fazinda detruor sfinkter dissinerjisidir. Biz bu vaka sunumunda mesane tutulumunun bazi acilarini sunmayi amacladik.
Anahtar Sozcukler: Behcet, norojen mesane, asiri aktif detrusor, antikolinerjik
Behcet's disease (BD) is a systemic vasculitis usually seen in young adults, which affects both venous and arterial vessels of the vascular system (1). Its etiology is still unknown (1). The patognomic symptoms are recurrent oral aphtae accompanies with at least two of these; positive pathergy test, skin lesions, recurrent genital ulcers and lesions in the eye (2).
Urogenital involvement usually consists of genital ulcers, urethritis, epididymitis and recurrent cystitis. Bladder involvement is extremely rare and only several cases have been reported (3). The findings suggest that neurogenic bladder due to neuro-Behcet disease is characterized urodynamically by overactive detrusor in storage phase and detrusor sphincter dyssynergia in voiding phase (4). We aimed to present several aspects of bladder involvement of Behcet disease in this case report.
Informed consent document was written and signed by the patient. A 27 year-old man admitted to our institution with chief complaint of urinary incontinence and erectile dysfunction in September 2014. He had a history of Behcet's disease since April 2013. The patient was under systemic cyclophosphamide 200mg/day, azathioprine 100mg/day and prednisolone 10mg/day therapy since the date of initial diagnosis. During this period patient had severe urge incontinence symptoms. Solifenasine succinate 5 mg/day tablet was started as anticholinergic therapy and symptoms partially recovered. On July 2014, patient was catheterized because of urinary retention then catheter was taken after 24 hours and the patient had spontaneous micturation. Bladder capacity of 365c and residual volume of 140 cc was reported by which urodynamic tests showed detrusor hyperreflexia (Figure 1). Patient was managed by clear intermittent catheterization (CIC) 6 times/day combined with oral solifenasin 5 mg/day regiment.
Bladder involvement in BD is usually seen in young patients (3, 5). Although the most common symptoms are storage symptoms, urge incontinence may be seen as well (5, 6). Our, 27 year old male, patient's chief complaint was urge urinary incontinence. Additionally, acute urinary retention and voiding symptoms may also be seen (7). Acute urinary retention after anticholinergic therapy was also seen in our case. According to Nakagawa et al. (4) report of three cases; two of them had a complaint of urinary frequency and urinary incontinence while one of them had a complaint of urinary retention. There can be any abnormality in sphincter function although sphincter deficiency or detrusor sphincter dyssynergia have been reported (5, 8). Meatal ulceration, sterile urethritis or cystitis can be the main reasons of dysuria (9). Gross hematuria is considered as a very rare symptom (10). Bladder cancer has been reported concomitant with BD as well. It can be sporadically or as a result of cyclophosphamide treatment. Our patient was under systemic cyclophosphamide treatment; but there was no evidence of a bladder malignancy yet. Urethrovaginal and vesicovaginal fistulas can also be seen rarely with BD (10). Increased intravesical pressure may cause hydroureteronephrosis or severe trabeculation of the bladder wall (3). Urodynamic tests may report detrusor hyperreflexia either sporadically or combined with impaired contractility, decreased bladder compliance or capacity, bladder hypersensitivity, hypoor acontractile bladder and increased postvoid residual urine concomitant to the detrusor hyperreflexia (3, 5- 8, 10). In our case bladder capacity of 365c and residual volume of 140 cc was reported by which urodynamic tests showed detrusor hyperreflexia.
Neurologic symptoms called as "Neuro-Behcet's Disease" has been reported in 5% to 10% of BD patients, and 5% of these patients had voiding symptoms (11).
Neurologic involvement usually affects the brain stem and can resemble multiple sclerosis, infection of central nervous system and stroke (5-7). Vasculitis inside the pontine micturition center can be the reason of high prevalence of urgency and frequency in BD patients (6). However ulceration or recurrent cystitis of the bladder may be seen as well.
Storage symptoms could be the result of direct involvement of bladder wall, neuro-Behcet's disease or combination of these two mechanisms (1).
A case whose bladder's pathology changed from areflexia to instability with impaired contractility was reported in a study by Porru and colleagues (7). Cetinel et al. (3) reported the incidence of bladder involvement in Behcet's Disease as 0.07% (n=8). The major findings were bladder wall trabeculation (5/8 cases) and bladder ulcer or hypervascularity in cystoscopy (2/8 cases), detrusor hyperactivity (7/8 cases) and low compliance of bladder (4/8 cases) according to that study (3). These findings suggest the main characteristics of neurogenic bladder due to Behcet's disease are detrusor hyperreflexia in the storage phase and detrusor-sphincter dyssynergia in the voiding phase. Voiding dysfunction can be treated by clean intermittent catheterization where the storage symptoms have been usually managed by anticholinergic agents. As final treatment, our patient was under anticholinergic therapy combined with clear intermittent catheterization (CIC) 6 times/day. Besides this Saito and Miyagawa (12) also suggest that intravesical oxybutynin therapy combined with clear intermittent catheterization has a highly successful rate of detrusor hyperreflexia with fewer side effects, i.e., dry mouth, flushing and obstipation, than oral medication. Recently, intravesical botulinum toxin injection therapy is being used to manage detrusor hyperreflexia. Denys et al. (13) suggest that botulinum toxin injected into the detrusor muscle seems to be an efficient treatment of bladder hyperreflexia for 6 months in patients resistant to anticholinergic drugs. According to Neugart et al. (14) report of 16 cases with refractory detrusor hyperactivity treated with intravesical botulinum-A-toxin injections; the urodynamic parameters of the bladder were improved clearly in all patients, subjective satisfaction was reached in 72.7% with no severe side effects. In addition, augmentation cystoplasty can be a final solution if needed in very rare situations. For example Theodorou et al. (15) reported their case which they had performed clam-type augmentation cystoplasty using sigmoid colon to avoid lower urinary tract symptoms and incontinence. After four years postoperatively; the patient was reported to be dry, and asymptomatic with the combination of clear intermittent catheterization.
Voiding dysfunction in BD patients may be the reason of bladder involvement. Urodynamic tests should be considered in these patients in order to classify the type of voiding dysfunction.
Informed Consent: Written informed consent was obtained from patient who participated in this case.
Peer-review: Externally peer-reviewed.
Author Contributions: Concept--M.D.; Design--M.D.; Supervision--M.D., O.O.C., O.F.; Resources--M.D., O.O.C., E.K.; Materials--M.D., O.O.C., E.K.; Data Collection and/or Processing--M.D., O.O.C., E.K.; Analysis and/or Interpretation--M.D., E.K.; Literature Search--M.D., O.O.C., O.F.; Writing Manuscript--M.D., O.O.C., E.K.; Critical Review--M.D., O.F.; Other--M.D., O.F.
Conflict of Interest: No conflict of interest was declared by the authors.
Financial Disclosure: The authors declared that this study has received no financial support.
Hasta Onami: Yazili hasta onami bu calismaya katilan hastadan alinmistir.
Hakem Degerlendirmesi: Dis bagimsiz.
Yazar Katkilari: Fikir--M.D.; Tasarim--M.D.; Denetleme--M.D., O.O.C., O.F.; Kaynaklar--M.D., O.O.C., E.K.; Malzemeler--M.D., O.O.C., E.K.; Veri Toplanmasi ve/veya Islemesi--M.D., O.O.C., E.K.; Analiz ve/veya Yorum--M.D., E.K.; Literatur Taramasi--M.D., O.O.C., O.F.; Yaziyi Yazan--M.D., O.O.C., E.K.; Elestirel Inceleme--M.D., O.F.; Diger--M.D., O.F.
Cikar Catismasi: Yazarlar cikar catismasi bildirmemislerdir.
Finansal Destek: Yazarlar bu calisma icin finansal destek almadiklarini beyan etmislerdir.
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(9.) Kirkali Z, Yigitbasi O, Sasmaz R. Urological aspects of Behcet's disease. Br J Urol 1991; 67: 638-9. [CrossRef]
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(12.) Saito M, Miyagawa I. Bladder dysfunction due to Behcet's Disease. Urol Int 2000; 65: 40-2. [CrossRef]
(13.) Denys P, Even-Schneider A, Thiry Escudie I, Ben Smail D, Ayoub N, Chartier-Castler E. Efficacy of botulinum toxin A for the treatment of detrusor hyperreflexia. Ann Readapt Med Phys 2003; 46: 326-8. [CrossRef]
(14.) Neugart F, Groh R, Gotz T, Horsch R. Injections of botulinum toxin a into the detrusor vesicae for treatment of refractory detrusor hyperactivity in non-neurological patients. Aktuelle Urol 2006; 37: 212-7. [CrossRef]
(15.) Theodorou C, Floratos D, Hatzinicolaou P, Vaiopoulos G. Neurogenic bladder dysfunction due to Behcet's disease. Int J Urol 1999; 6: 423-5. [CrossRef]
Murat Dincer (1), Omer Onur Cakir (1), Onur Fikri (2), Engin Kandirali (1)
(1) Clinic of Urology, University of Health Sciences Bagcilar Training and Research Hospital, Istanbul, Turkey
(2) Clinic of Urology, Edirne Sultan 1.Murat State Hospital, Edirne, Turkey
Address for Correspondence / Yazisma Adresi: Murat Dincer, E-mail: email@example.com
Received Date / Gelis Tarihi: 24.10.2015 Accepted Date / Kabul Tarihi: 01.01.2016
Cite this article as: Dincer M, Cakir OM, Fikri O, Kandirali E. Bladder Dysfunction in Behcet's Disease. JAREM 2017; 7: 86-8.
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|Title Annotation:||Case Report / Olgu Sunumu|
|Author:||Dincer, Murat; Cakir, Omer Onur; Fikri, Onur; Kandirali, Engin|
|Publication:||Journal of Academic Research in Medicine|
|Date:||Aug 1, 2017|
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