LATE SURGICAL TREATMENT FOR SPONTANEOUS RUPTURE OF HEPATOCELLULAR ADENOMA: CASE REPORT

The cryptorchidism results from abnormalities in the formation and testicular descent during the embryonic period4. It is present in 6% of newborns at term and in 0.8% of infants under one year of age. Can be bilateral in up to 10% of cases, and sometimes is associated with other defects in genitourinary tract5. The most feared complication of undescended testicle is cancer, ranging from 3.5-14.5% among patients with cryptorchidism5. The testis are intra-abdominal in 10% of cases and at risk 200 times greater in malignant transformation3. Malignant degeneration has the peak incidence in third and fourth decade of life2,3. They are usually asymptomatic and are identified incidentally by imaging tests. When symptomatic, diagnosis is difficult and the symptoms may mimic acute appendicitis, urinary calculus and mass effects, compressive symptoms of the gastrointestinal and genitourinary tracts3. Imaging tests, US, CT and MRI show pelvic or retroperitoneal mass, well defined, homogeneous, without obvious evidence of necrosis or calcification2,3,5. These findings have as main differential diagnoses lymphadenopathy and sarcoma, which are more common situations. The predominant histological type is pure seminoma (43%), followed by embryonic carcinoma (28%), teratocarcinoma (27%) and choriocarcinoma (2%)3. Surgical treatment is mandatory, with resection of intra-abdominal mass and chemotherapy may be an alternative, depending on the stage and histological type of malignant transformation2.


INTRODUCTION
H epatocellular adenoma (HA) is a rare benign neoplasm of the liver. It is strongly associated with oral contraceptive used by woman in childbearing age, by men receiving anabolic steroids and metabolic diseases. HA incidence has raised over the years from 5 per 1 milion in 1964 9 to 4 per 100.000 3 , for unknown reasons.
HA is important because of its risk of complications such as life threatening rupture of the tumor and malignant transformation. Surgical treatment in ruptured HA has a high mortality rate but it is still the standard procedure in those cases 5 . Emergency liver resection in ruptured HA has a mortality rate from 5-10%, but in elective resections the mortality rate is under 1%.
Different procedures have been suggested to decrease mortality rates and spare liver parenchyma, such as arterial embolization of ruptured adenomas, although it is not an available procedure in most centers.

CASE REPORT
A 44-year-old woman admitted in the emergency department of another hospital with complaints of sudden upper quadrant abdominal pain. She had been using oral contraceptives for 31 years. She presented with acute mild abdominal pain in epigastrium as well as right hypochondrium pain followed by light dyspnea and dizziness. She was treated with analgesics and ordered an abdominal ultrasound made only three days after the pain. The ultrasound showed a solid liver mass in the right hepatic lobe measuring 150x100x100 mm 3. She was discharged without additional treatment.
After almost five months after the occurrence, she was referred to our surgical department for diagnostic investigation after another episode of abdominal pain just like the last time, followed by tachycardia and cold sweating. Physical examination revealed mild anemia and a diffuse abdominal pain, without peritonitis or palpable masses.
Abdominal CT and MRI showed an enlarged liver, with a contrast enhanced liver mass involving segments VI and VII. The liver mass measured 118x70mm 2 associated with a perilesional voluminous hematoma measuring 8cm and haemoperitoneum. The radiologic findings suggested a ruputured hepatocellular adenoma ( Figure 1) Patient was admitted and sent to observation room to be prepared for surgical procedure after clinical improvement. She was transfused with two bags of packed red bloods cells and intensive electrolytes control. Patient was sent to the operation room and had an open laparotomy with Chevron incision. During operative exploration was found a voluminous liver lesion involving segments VI and VII, large subcapsular hematoma over the right hepatic lobe and mild hemoperitoneum. The right lobe of the liver had strong adhesions to the right diaphragm. It was decided to make an anterior approach with inflow control due to the risk of bleeding, by ligation of the right portal vein and the right hepatic artery. The procedure continued with parenchyma right transection in the cantlie´s line, showed by isquemic demarcation, using bipolar forceps, argon coagulator device and kelly-crush technique. The liver parenchyma was dissected by the hematoma in some parts, disarranging the liver architecture. In the last part of the procedure, was mobilized all the right liver lobe followed by outflow control, by ligation of the right hepatic vein. During the liver mobilization, was able to see a large rupture in the posterior part of the liver and multiple adhesions between liver parenchyma and diaphragm, but without any sign of active bleeding. The surgery was completed with an anatomic right hepatectomy (Figures 2 and 3) The surgical specimen weighted 1.170 g, measured 18,0x14,0x12,0 cm, presenting with a 12 cm rupture, externalizing an irregular and hemorrhagic mass. The tumor measured 1,0x6,0x6,0 cm, friable, along with a large subcapsular hematoma and clear margins.
Microscopy showed a neoplasm filled with hepatocytes cells showing enlarged plasmatic volume, eosinophils, macrovesicular steatosis, regular nucleus with minimum atypia and rare nucleolus. There was sinusoidal dilatation with spots of necrosis and hemorrhage. All these data corroborate with the hepatocellular adenoma ( Figures  4 and 5).
Patient was kept in the intensive care unit for a day. There were no blood transfusions and the patient was discharged in the 6 th operative day without any complications.

DISCUSSION
HA is a rare condition, and is commonly associated with oral contraceptive use 6 . The longer women uses oral contraceptives with increased estrogen level, the higher is the chance to develop hepatocellular adenomas. It´s frequently seen in woman in childbearing age, as well as in men and diseases like hemochromatosis and type 1 glycogen storage disease 4 . It has clinical importance because of the risk of complications. Spontaneous rupture is the most important complication and usually happens in adenomas greater than 5 cm, in 20-40% of the cases 4 .
Approximately 10% of patients with HA present with acute abdominal pain due to rupture and hemoperitoneum, in some cases followed by hipovolemic shock. Patients might also refer nauseas, vomiting, anorexia and fever. Mortality in ruptured HA has been associated with late diagnosis, coagulopathy and post-operative complications 8 .
Conservative treatment is used to small adenomas, mainly the ones related to oral contraceptives and anabolic steroids. The follow up in these cases includes abdominal TC or ultrasound each sux months. Although, when the adenoma is higher than 5cm or show symptoms, the surgical treatment is recommended because of the increased risk of hemorrhage and malignant transformation 6 .
The surgical treatment vary on the case, as well as the different approaches of liver resection. The standard treatment for ruptured HA must be local or segmental resections, to spare as much liver parenchyma as possible. Laparoscopic hepatectomy is a feasible option for benign liver lesions in elective cases 1,2 , but when it comes to ruptured, the open laparotomy is preferred due to bleeding and close contact to important liver vessels. Patients with ruptured HA must have elective resections when possible. There are several procedures to avoid emergency treatment but surgery still remains the main approach. If patient is stable and the tumor is localized in one segment, partial hepatectomy should be promptly made, but if there is important blood loss, volemic reposition with fluids and transfusions of hemocomponents should be attempted as well as arterial embolization 10 , with late resection when patient is stable. The selective arterial embolization have been playing an important role in the treatment of ruptured and non-ruptured HA, with low complication rates when compared with emergency surgery 7 . Arterial embolization is a very expensive procedure and is not available in most centers.

INTRODUCTION
T he congenital diaphragmatic hernia (CDH) is defined as an anatomical defect on diaphragm, which permits the herniation of abdominal viscera into the thorax 4 . The hernia occurs due to an incomplete occlusion of the pleuroperitoneal channel during the embrionary period. The main cause of the incomplete closure can be a genetic mutation, a teratogen or both.
In terms of anatomic location, the CDH can be classified as Bochdalek type when an incomplete pleuroperitoneal channel occlusion is found posterolaterally; as Morganni type, while the defect is seen retrosternally; and yet as a congenital transhiatal esophagic type hernia. Among them, the Bochdalek type is the most common, found in 78-90% of patients; the Morganni type, in 1,5-6% of cases; and transhiatal, 14-24% 11 .
In most cases, the clinical impact occurs in the neonatal period, since only 10% of hernias are diagnosed after this period 7 . In neonates, the clinical presentation is acute, providing a higher morbidity and mortality. In adulthood, symptoms, if any, are more insidious, vague and intermittent, affecting not