Abdominoscrotal hydrocele: when one sac becomes bissac

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A 5‐month‐old male infant was referred to our department with a history of left scrotal hydrocele since birth. His parents related a progressive enlargement of the hydrocele accompanied by the development of a mass in the ipsilateral lower quadrant of the abdomen over the month before admission. On bimanual palpation, both abdominal and scrotal swellings showed cross‐fluctuation. A sonography was arranged revealing a dumbbell‐shaped hypoechoic lesion continuing from the left scrotum to the abdomen suggesting the provisional diagnosis of abdominoscrotal hydrocele (ASH) (Fig. 1). Additionally, a right scrotal hydrocele and an undescended testis within the left inguinal canal were noticed. Surgical treatment consisted of left inguinal skin crease incision, resection of ASH after its complete exteriorization from the abdominoscrotal cavity, identification with ligation of the processus vaginalis and left orchiopexy (Fig. 2). The patient did well post‐operatively and no recurrent hydrocele developed after 7 years of follow‐up.
Hydrocele en bissac, as coined by Dupuytren in 1834 and later renamed abdominoscrotal, bilocular or hourglass hydrocele (ASH) by Bickle in 1919, is a rare congenital condition in which a hydrocele of the tunica vaginalis develops two large components, one in the scrotum and one in the abdomen, communicating in an hourglass fashion through the inguinal canal.1 Unique in its kind, this pathological entity can manifest in the newborns and children (only 130 cases so far) as well as in the adults (some 100 cases) with a predilection for male sex.2 The aetiopathogenesis is under debate and several interesting hypotheses have been proposed.3 The most accepted speculation is the cephalad extension of ASH espoused by Dupuytren: when the intracystic pressure within the scrotal hydrocele exceeds the intraperitoneal one, the tunica vaginalis becomes overdistended and, protruding into a locus minoris resistentiae, the inguinal canal ascends to the abdomen forming a new sac.3 Other theories implicate the herniation of a peritoneal diverticulum into the inguinoscrotal space (caudad extension of ASH) or the existence of a one‐way valve‐like mechanism within a patent processus vaginalis (PPV) at the internal inguinal ring.4 Regarding the last topic, however, most authors deem ASH a non‐communicating hydrocele explaining that a PPV is practically impossible to find in such a condition.1 Usually, the abdominal component is unilateral and larger than the scrotal counterpart.6 The abdominal component has been described properitoneal, retroperitoneal and also between the two layers of the broad ligament; in our patient, it was mainly properitoneal but also expanded into the left retroperitoneum.1 Basically, the diagnosis of ASH is clinically obtained through the ‘springing back ball’ sign: the manual reduction of the scrotal hydrocele implicates an initial enlargement of the abdominal lesion which is followed by the subsequent restoration of the scrotal component.7 Generally asymptomatic, ASH can manifest with exquisite abdominal or scrotal pain and has also been described in association with acute appendicitis, malignant mesothelioma of the tunica vaginalis, cryptorchidism and abnormal testicular findings.1 Abdominal ultrasound examination is the most practical, informative, innocuous and inexpensive imaging study corroborating the provisional diagnosis of ASH: characteristically, it shows an hourglass‐shaped cystic collection extending from the scrotal to the abdominal region with the narrowest portion passing through the inguinal canal.2 In 1991, Sasidharan et al. reported the first and so far only the case of ASH was detected antenatally during a maternal ultrasonography performed at 22 weeks of gestation.8 Computed tomography and magnetic resonance imaging of the abdomen represent sophisticated radiological resources to deploy when diagnostic uncertainty remains following clinical and ultrasound examination. Differential diagnosis of the abdominal component of ASH must take into account several conditions such as indirect inguinal hernia, bladder diverticulum, hydronephrosis and mesenteric, pseudopancreatic, urachal, hydatid or lymphogenous cysts.
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