Journal of Perinatology (2009) 29, 585–587 r 2009 Nature Publishing Group All rights reserved. 0743-8346/09 $32 www.nature.com/jp PERINATAL/NEONATAL CASE PRESENTATION Meconium periorchitis: intrauterine diagnosis and neonatal outcome: case reports and review of the literature

RH Regev1,3, O Markovich2,3, S Arnon1,3, S Bauer1,3, T Dolfin1,3 and I Litmanovitz1,3 1Neonatal Department, Meir Medical Center, Kfar Saba, Israel; 2Ultrasound Unit, Department of Obstetrics and Gynecology, Meir Medical Center, Kfar Saba, Israel and 3Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel

Case 1 Meconium periorchitis (MP) is a rare disorder caused by fetal meconium A routine ultrasound screening for anomalies performed at 22 peritonitis with subsequent spillage of meconium into the scrotal sac. The weeks of gestation was described as normal. Because of suspected condition is seldom diagnosed correctly during fetal life and the fetal growth restriction, a second ultrasound evaluation was ultrasonographic diagnoses reported vary from no diagnosis to hematoma performed at 39 weeks of gestation, detecting bilateral hydroceles, or hydrocele. It is usually diagnosed clinically during the first year of life and at the base of the right testis, echogenic reflections of when a scrotal mass is an incidental finding. Here, we describe two cases of 19 Â 11 Â 17 mm, creating acoustic shadows (Figure 1). MP that were diagnosed during routine intrauterine ultrasound Following the birth of a 3080 g, full-term male infant through a examination for fetal growth assessment, and confirmed after birth. One spontaneous vaginal delivery, bilateral hydroceles and right infant underwent a surgical excision of the scrotal mass, confirming the paratesticular masses, extending to the internal orifice of the histological diagnosis of meconium periorchitis. The other was managed inguinal canal were palpable. Testicular palpation was normal. conservatively. Neither had cystic fibrosis. Thus, we believe that a diagnosis Abdominal examination was normal. There were no feeding or of MP should be considered when prenatal ultrasonographic findings are stooling problems. Ultrasound of the scrotum showed normal suspicious for the problem. The awareness of the ultrasonographer and the testicles and hyperechoic particles within the scrotum and inguinal neonatologist are important for immediate postnatal management, as canal in the right extratesticular space. No abdominal congenital scrotal masses may have other etiologies. calcifications or bowel dilatation were found on abdominal Journal of Perinatology (2009) 29, 585–587; doi:10.1038/jp.2009.15 X-ray. Testing for cystic fibrosis (CF) was negative. Excision Keywords: intrauterine diagnosis; meconium periorchitis; scrotal of the right paratesticular mass was performed at the age of calcifications; newborn infants five months. The histological report was consistent with the diagnosis of MP.

Case 2 Ultrasound screening for anomalies performed at 15 and 23 Introduction weeks of gestation revealed no abnormal findings. Due to suspected Neonatal meconium periorchitis (MP) is caused by intrauterine intrauterine growth restriction, another ultrasound scan was spillage of meconium from a bowel perforation into the scrotal sac. performed at 36 weeks gestation that detected abdominal The incidence of this condition is unknown, although it occurs calcifications and an enlarged scrotum with echogenic reflections. infrequently. MP is seldom diagnosed accurately during fetal life. The scrotum measured 46 Â 39 mm. A full-term, male infant, with Previous intrauterine diagnoses have usually incorrectly included birth weight of 2860 g was delivered vaginally after prolonged hydrocele with inguinal hernia1 and intrascrotal hematoma.2 Very rupture of membranes. Following birth, bilateral hydroceles with few cases of the disorder have been described in the literature, and normal testicular consistency were observed. Abdominal only four were diagnosed prenatally.3,4 We present two cases of examination was normal, as were feeding and meconium passage. infants with an intrauterine diagnosis of MP and their postnatal Scrotal ultrasonography confirmed the diagnosis of bilateral outcomes. periorchitis. The testicles were normal. Scattered abdominal intraperitoneal calcifications were also identified, with normal Correspondence: Dr RH Regev, Neonatal Department, Meir Medical Center, 59 Tchernicovsky bowel. The parents declined further evaluation. At 24 months of Street, Kfar Saba 44821, Israel. E-mail: [email protected] age, the child was well and thriving, without any medical Received 1 September 2008; revised 4 January 2009; accepted 12 January 2009 problems. Intrauterine diagnosis of meconium periorchitis RH Regev et al 586

Discussion not clearly separable from the testis.10,11 Calcified plaques scattered We present two cases of MP that were accurately diagnosed in the peritoneal cavity are the most common radiographic feature, prenatally in otherwise healthy infants. It is usually diagnosed and are the result of healed meconium peritonitis. Scrotal and from a few months to five years of age.5 In most infants, MP abdominal ultrasonography contribute to the diagnosis of MP after presents after birth as a soft hydrocele6–8 or swelling.9 Of the four birth. cases with a prenatal diagnosis that were previously reported,1–4 On clinical examination, a soft extratesticular mass may be two were initially diagnosed as a scrotal mass1,4 and two as a present, or the condition may present as scrotal swelling and 12 hydrocele, only subsequently identified as MP (Table 1). One case discoloration mistakenly attributed to birth trauma, or a soft 6 12 of intrauterine diagnosis in a fetus with CF was recently published.2 hydrocele. Often no scrotal abnormality is detected at birth. The In the cases presented here, MP was diagnosed prenatally and scrotal mass hardens over subsequent weeks to months and confirmed after birth in two infants without other health problems. becomes partially calcified. Such events may delay the diagnosis 7,12,14 5 Meconium periorchitis is a rare cause of benign scrotal mass, until the age of a few months, or even a few years. resulting from a bowel perforation that occurred during fetal life. When both scrotal and abdominal calcifications are seen on X- Meconium spreading in the peritoneal space causes a foreign body ray and ultrasonography, no diagnostic dilemma arises, as they are 13,14 inflammatory reaction. Meconium penetrates the paratesticular soft virtually diagnostic of MP. However, when calcifications are tissues through a patent processus vaginalis, causing sterile present in the scrotum only, the differential diagnosis should alert inflammation of the paratesticular tissue. The presence of the physician to the possibility of neoplasms, such as teratoma, meconium in the scrotum for a prolonged period causes rhabdomyosarcoma, metastatic neuroblastoma, lymphoma and subsequent fibrosis and calcification that may mimic a testicular or pseudotumors, which have similar ultrasonographic 10,11 paratesticular tumor, particularly if it feels firm, nontender, and characteristics. CF was diagnosed in three published cases of infants with MP.2,13,16 Thus, infants with MP should be evaluated for CF, as meconium peritonitis has been reported to occur in 15% of infants with CF.6 Meconium periorchitis can be been managed with either surgical or conservative treatment. Most authors recommend that surgery should be performed when a neoplasm is suspected. Others advocate a conservative approach, especially in the presence of abdominal calcifications, as the natural history in an asymptomatic infant is self-limiting, with spontaneous resolution and resorption of the calcifications.6–8,13,17,18 The presence of both abdominal and scrotal calcifications on abdominal X-ray in our second case directed us toward a conservative approach, whereas the absence of abdominal calcifications in case 1 resulted in the evaluation for other causes and eventual excision of a paratesticular mass. Meconium periorchitis is a rare entity, with a relatively large spectrum of differential diagnoses. An accurate prenatal diagnosis Figure 1 Case 1FUltrasound of scrotum showing hydrocele with a has an important impact on the choice of treatment modalities, paratesticular echogenic mass. leading either to surgery or expectant management.

Table 1 Summary of cases of meconium peritonitis diagnosed prenatally

Author Gestational age at diagnosis (weeks) Presentation after birth Treatment Cystic fibrosis

Gililland et al.1 29 Hydrocele Surgery À Wax et al.2 33–34 Hydrocele Not described + Kenney et al.3 28 Scrotal mass Surgerya À Sukcharoen et al.4 38 Scrotal mass Surgery À Present cases 39 Hydrocele+scrotal mass Surgery À 36 Hydrocele Conservative management À aBecause of the presence of intestinal perforation at birth in a preterm, 30-week infant.

Journal of Perinatology Intrauterine diagnosis of meconium periorchitis RH Regev et al 587

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