A rare case of accessory liver lobe torsion in a pediatric patient who showed recurrent epigastralgia and who was treated by elective laparoscopic resection

Abstract

Background Accessory liver lobe (ALL) is a rare liver malformation. An ALL develops due to malformation of the endodermal caudal foregut and segmentation of the hepatic bud in the third week of gestation. Most ALLs are asymptomatic and are detected incidentally during abdominal surgery. The incidence of ALL is < 1% in patients who undergo abdominal surgery. However, some ALLs twist and cause acute abdomen. We experienced a pediatric case of ALL torsion in a patient who underwent elective laparoscopic surgery. Case presentation The 5-year-old girl had a 3-month history of epigastralgia and vomiting, which occurred every 2 weeks. Abdominal ultrasonography with color Doppler imaging revealed an 11.8 × 13.6 mm nonvascular lesion with mixed echogenicity near the round ligament of the liver. Enhanced computed tomography confirmed a 14 × 16 × 20 mm low-attenuation mass surrounded by a hyperdense line and disproportionate fat stranding on the right side of the round ligament of the liver. There was no ascites or hemorrhage. These findings suggested an abscess of the round ligament of the liver. Her symptoms improved with the administration of oral antibiotics; thus, we planned to perform elective exploratory laparoscopy and subsequent resection. Two trocars (5 mm) were inserted through a multichannel port device at the umbilicus and one trocar (3 mm) was inserted at the right lateral abdomen. Upon observation of the abdominal cavity, the omentum was observed adhering to the round ligament of the liver. Macroscopic observation revealed no apparent mass lesions. We performed adhesiolysis of the omentum from the round ligament of the liver using a vessel sealing system. We performed resection at the site at which adhesion had formed between the round ligament of the liver with the surrounding tissue using a vessel sealing system and the resected specimen was extracted through the umbilical wound. The postoperative course was uneventful. A pathological examination revealed necrotic liver tissue. The resected tissue was founded to be an ALL with ischemic change. Conclusions The recurrent abdominal pain was induced by torsion of the ALL. Pediatric surgeons should consider ALL torsion as a differential diagnosis for epigastralgia of unknown etiology.

Case report: A case of giant accessory hepatic lobe torsion combined with left hepatic vein branch thrombosis in a child

“…Prompt management and emergency excision or laparoscopy should be recommended in cases of suspected AHL torsion (6,17). Even among individuals presenting with complications, most do well following surgery (4)(5)(6). As this disease is relatively rare, torsion of the giant pedunculated AHL was already evident at the onset of abdominal pain in this child, but the diagnosis on early imaging failed and emergent operation was not performed.…”

“…AHL is categorized on the basis of size and its attachment to the liver: (1) large AHL (weight >30 g), (2) small AHL (weight <30 g), (3) ectopic AHL without hepatic attachment, and (4) microscopic AHL in the gallbladder wall (8). Among the 23 pediatric AHL cases reported in the literature, the age ranged from 1 day to 15 years, the male to female ratio was 11:12, and laparoscopic resection of AHL torsion has been reported (4)(5)(6). AHL was typically an incidental finding on radiographic imaging or autopsy, and most cases were also associated with persistent defects of anterior abdominal wall (4,5).…”

“…Among the 23 pediatric AHL cases reported in the literature, the age ranged from 1 day to 15 years, the male to female ratio was 11:12, and laparoscopic resection of AHL torsion has been reported ( 4 – 6 ). AHL was typically an incidental finding on radiographic imaging or autopsy, and most cases were also associated with persistent defects of anterior abdominal wall ( 4 , 5 ). Corbitt et al ( 4 ) reported that 41% of children with AHL are associated with abdominal wall defects, including umbilical bulge, Beckwith–Wiedemann syndrome, umbilical hernia, and cloacal exstrophy, stemming from abnormal development of the AHL that may obstruct the closure of the umbilical ring ( 4 ).…”

“…In that case, most patients are presented with sudden severe right-sided abdominal pain, probably accompanied by nausea or vomiting. As for palpation, an enlarged tender mass might be detected ( 5 , 13 ). The AHL should not be reversed before ligation to prevent dislodgement of the thrombus ( 2 ).…”

“…Azmy (3) first described a neonatal case of Beckwith-Wiedemann syndrome combined with an accessory hepatic lobe torsion in 1980. Among 23 reported cases of pediatric AHL, only 3 have undergone laparoscopic resection of twisted AHL (4)(5)(6). We report the case of a rare pediatric giant AHL torsion combined 10.3389/fped.2022.970876 with left hepatic vein branch thrombosis which was successfully treated by laparoscopic lobectomy followed by excision of AHL.…”