BJU International (1999), 84, 739–740

CASE RE PORT

Behcet’s disease and spontaneous haematocele: an unusualcomplicationI . ORHAN, R. ONUR, A. ARDICOGL U and Y. SALATANDepartment of Urology, University of Firat, School of Medicine, Elazig, Turkey

chronic multisystem inflammatory disorder of unknownCase report

aetiology, with urological manifestations, the commonestof which are recurrent genital ulcerations, especiallyA 37-year-old man with long-standing Behcet’s disease

(BD) presented with a painful, right scrotal swelling of aphthous ulcerations or scarring. Other urogenital symp-toms are prostatitis, orchitis, chronic sterile cystitis,4 h duration. He reported that the swelling developed

suddenly and reached maximum size within several epididymitis, relapsing salpingitis and renal failure fromamyloidosis or glomerulonephritis [2]. The urologicalhours. There had been no recent trauma but a history

revealed that the patient had experienced several attacks manifestation in the present case was the occurrence ofa spontaneous haematocele caused by a ruptured vein,of oral and genital aphthous ulcers which are character-

istic of BD. During the preceding 2 years the patient had with vascular damage. A spontaneous haematocele inBD has not been reported previously. Although this istaken no medication. A physical examination revealed

a tender and diCuse swelling of the scrotum. There was an unreported complication, there is a well-recognizedform of BD, ‘vasculo-BD’ which primarily aCects arteriesno evidence of trauma or loss of skin integrity except for

signs of scars in the scrotum from previous attacks of and veins, and has an increased morbidity. Vasculo-BDis characterized by a wide range of vascular manifes-ulcerative genital lesions. Laboratory data showed a

normal blood count with a mean (range) of 2.56 tations, especially superficial thrombophlebitis.Thrombotic complications have been reported and(1.4–4.0)×105 platelets/mL, a bleeding time of 1.5

(1–3) min and a coagulation time of 3.5 (4–5) min. The account for most of the deaths caused by BD [3].Vasculitic lesions are characterized by lymphocytic infil-patient underwent scrotal and Doppler ultrasonography

(US) for the diCerential diagnosis of acute scrotal patho- tration and immune-complex deposition in the vesselwall. Occasionally, granuloma formation, fibrosis andlogies and presence of testicular tumour. US showed an

echogenic mass of 5×4 cm within the scrotum and severe tissue disruption may occur [3]. In the presentcase, the patient had an unusual complication of BDnormal testicles; similarly, Doppler US revealed normal

testicular blood flow. The patient underwent emergency because of involvement of a visceral vessel, having ahaemorrhagic rather than a thrombotic event, andexploration via a right scrotal incision, which revealed

a haematocele that was removed completely. The bleed- finally spontaneous occurrence. A possible explanationfor such a complication is perivascular infiltration in theing originated from a ruptured vein of 3 mm in diameter

within the right pampiniform plexus. Macroscopically vessel wall, leading to degeneration of the endothelium.Moreover, fragmentation and splitting of the elastic fibresthe vessel was hypertrophied and the vessel wall seemed

to be oedematous. A sample was removed for histopathol- in the media, and mild to advanced fibrinization in theogical examination and the patient underwent simpleligation of the vein. The scrotal skin was closed over aPenrose drain. Subsequent histopathological examin-ation revealed lymphocytic infiltration characteristic ofBD (Fig. 1). On the first day after surgery the drain wasremoved; the patient was discharged on the second day,with the appropriate medication.

Comment

In 1937, Behcet, a Turkish dermatologist, first describedthe triple complex of symptoms (oral and genital ulcers, Fig. 1. Histological appearance of the vessel wall showing charac-

teristic lymphocytic infiltration. Haematoxylin and eosin×200.and uveitis) probably originated by a virus [1]. BD is a

739© 1999 BJU International

740 CASE REPORT

Genitalien. Dermatologische Wochenchirurgie 1937; 105:intima and adventitia, may enhance this particular event1152–7[3]. Although visceral vessel involvement is rare in BD,

2 Bradbury AW, Milne AA, Murie JA. Surgical aspects ofit is likely that a vein could rupture at any site in theBehcet’s disease. Br J Surg 1994; 81: 1712–21body [3]. The spontaneous haematocele in the present

3 Kuzu MA, Ozaslan C, Koksoy C et al. Vascular involvementcase occurred in the absence of trauma and with noin Behcet’s disease: 8-year audit. World J Surg 1994;

anticoagulation therapy. However, thrombotic or18: 948–54

haemorrhagic complications are not uncommon invasculo-BD, and with early recognition and treatment

Authorsof this disease, there should be a further reduction inmortality and morbidity. I. Orhan, MD, Assistant Professor of Urology.

R. Onur, MD, Chief Resident in Urology.A. Ardicoglu, MD, Assistant Professor of Urology.

References Y. Salatan, MD, Resident in Urology.Correspondence: Dr I. Orhan, Firat Tip merkezi, Uroloji ABD1 Behcet H. Uber rezidivierende apthose, durch ein Virus

verersachte Geschwure am Mund am Auge und an den ogretim uyesi, 23200 Elazig, Turkey.

© 1999 BJU International 84, 739–740