Pediatric living donor liver transplantation using liver allograft after ex vivo backtable resection of hemangioma: A case report

lNTRODUCTlON

Liver transplantation has brought a paradigm shift in the management and outcomes of pediatric patients with liver failure. However, due to the shortage of donor livers, many children on organ transplant waiting lists die[1]. Use of marginal liver allografts to expand the donor pool can help reduce the waiting time[2]. One type of liver allograft from marginal donors includes the use of livers with benign tumors, such as hepatic hemangioma. Hepatic hemangiomas usually remain asymptomatic[3]and have a benign course[4]. According to previous reports, liver allograft with hepatic hemangioma or after resection of hemangioma can be safely transplanted[5-10]. Sanada

[11] described the use of a living donor liver allograft after

hemangioma resection. This case report indicates that the liver allograft can be safely used for liver transplantation after removal of the hemangioma. In pediatric patients, especially infants, a small liver allograft is required for liver transplantation. Thus, we proposed that we could use liver segments from patients with symptomatic hemangioma undergoing hepatectomy after backtable resection of the hemangioma for pediatric liver transplantation.

作為世界上第一個以進口為主題的國家級展會，人們更關心其產(chǎn)生的背景。美國特朗普政府公然挑起貿(mào)易爭端，對中國輸美產(chǎn)品征收高額關稅，領頭在全球范圍刮起保護主義寒流。當此之時，進博會應運而生。第一，中國不怕任何壓力，改革開放有我們自己的時間表。第二，美國人頻繁退群之時，中國人將更高地舉起全球化大旗，更高水平對外開放。

On clinical examination, there was a 6-cm postoperative scar in the abdomen due to previous Kasai portoenterostomy of the pediatric patient. The adult patient’s abdominal examination was unremarkable with no organomegaly.

CASE PRESENTATlON

Chief complaints

An 8-month-old female infant presented to the emergency department with jaundice and high-grade fever. At the same time, a 55-year-old male was admitted at our center due to discomfort in the right upper abdomen.

History of present illness

Liver transplantation is the most effective treatment for end-stage liver disease[14]. The reported 1-year and 5-year survival rates are more than 90% and 70%, respectively[15]. However, organ scarcity is still the greatest limitation for patients in need of liver transplantation. Therefore, marginal liver allograft in liver transplantation, particularly in cases of benign tumors, has become an accepted alternative[16]. Use of liver allograft with hemangioma for liver transplantation has been previously reported[5-10]. In some cases, hemangiomas were not resected due to the risk of small-for-size syndrome, and follow-up of such cases showed that the volume of hepatic hemangioma decreased and the normal parenchymal volume increased with time, without the appearance of new hemangiomas. However, long-term follow-up of such cases has not been carried out. However, a small volume of liver allograft after hemangioma excision is sufficient for pediatric liver transplant and it can effectively avoid the possibility of lethal changes[11]. Thus, we propose that for pediatric patients, large hemangiomas in the liver allograft should be resected prior to transplantation.

應用實驗方法對PVC材質(zhì)的桌布、文具袋、服裝袋、化妝品袋、餐具袋8個批次的樣品中6種重金屬元素含量進行檢測，結(jié)果見表4。

The adult patient had discomfort in the right upper abdomen for one month. The upper abdominal discomfort was aggravated by heavy meals and relieved by fasting. He denied any recent fever,jaundice, allergy, chills, or changes in bowel habits.

“昆北”陽平聲字“雙”的唱調(diào)（《南柯記·瑤臺》【梁州第七】“臂鞲雙抬”，765），該單字唱調(diào)的過腔是。其中的是第一個樂匯型級音性過腔，是第二個樂匯型級音性過腔，是第三個樂匯型級音性過腔。這個過腔也是由“級音+級音+級音”同一種音樂材料組成的多節(jié)型過腔。

History of past illness

針對目前我國病理學教學的普遍問題，基礎病理學教學所安排的學時，對于病理學這一涉及到所有臨床專業(yè)疾病的重要學科是遠遠不夠的，醫(yī)學生病理專業(yè)素質(zhì)不高；臨床病理教學難以培養(yǎng)高質(zhì)量的病理專科醫(yī)生，并且病理專科醫(yī)生存在巨大的缺口等困境。結(jié)合目前慕課平臺的高速發(fā)展，認真調(diào)研慕課平臺的開放原則、注冊對象，培養(yǎng)模式、運營情況等等，在病理學教學中引入慕課平臺，將大有作為。但是，如何讓慕課平臺在病理學教學中發(fā)揮更大作用也有待于病理學教研工作者進行深入的調(diào)查和研究。

Personal and family history

They both did not have any addictions or any significant family history.

Physical examination

Herein, we describe the first case of pediatric living donor liver transplantation (LDLT) using a liver allograft following backtable resection of hemangioma.

③農(nóng)民水費承受力計算。根據(jù)計算公式，桃花山鎮(zhèn)農(nóng)民用水者協(xié)會農(nóng)民水費承受力為90元/畝；調(diào)關鎮(zhèn)農(nóng)民用水者協(xié)會、東升鎮(zhèn)農(nóng)民用水者協(xié)會農(nóng)民承受力為90.7元/畝。

Laboratory examinations

The pediatric patient’s liver function tests at admission were as follows: Serum total bilirubin = 120.5 μmol/L, direct bilirubin = 78.8 μmol/L, international normalized ratio = 1.37, aspartate aminotransferase = 153.2 U/L, and alanine aminotransferase = 119.9 U/L. Pre-operative liver functions of the adult patient were within the normal range and there was no evidence of coagulopathy.

Imaging examinations

Two surgical procedures for the resection of hemangiomas can be adopted, namely

resection and backable resection during LDLT. In previous reports,

resection was performed and found to be feasible, as it avoided intra-operative bleeding, bile leakage, and limited cold ischemic time compared to backtable resection[11]. However,

resection prolongs the operative time and increases the risk of bleeding in donors, which can be potentially harmful. Backtable resection of hemangioma has only been reported in deceased donor livers[10,17]. With the advancements in liver transplantation techniques, complications of backtable liver resection, such as intra-operative bleeding,bile leakage, and prolonged cold ischemic time, can successfully be avoided[18]. Some of the important points for backtable resection include the use of an electric knife, electric bipolar, or ultrasonic knife during resection, and closure of all the orifices at the cut surface using hemo-lok clips and/or sutures. In the case presented here, we performed backtable resection, as

resection of segment IV hemangioma was technically difficult and could have compromised donor safety.

The post-operative course of the donor and the recipient was uneventful, and both were discharged from the hospital on post-operative days 12 and 35, respectively. Tacrolimus was used as an immunosuppressant up to two years after surgery. The follow-up of the recipient two years after the liver transplantation showed good liver function without any bile duct strictures (Figure 4). Abdominal CT of the donor at six months after surgery (Figure 1C) and that of the recipient at two years after surgery (Figure 1D) showed good regeneration of the liver without any recurrence of hemangioma.

FlNAL DlAGNOSlS

On the second day of hospitalization, the pediatric patient was diagnosed to have Child-Pugh grade C hepatic failure due to congenital biliary atresia with grade 2 hepatic encephalopathy. The adult was diagnosed as hemangioma in segment 4 of the liver.

TREATMENT

The pediatric patient was listed for emergency liver transplantation. There was a strong possibility that the adult patient would develop ischemic necrosis of segments 2 and 3 if simple enucleation of the hemangioma was performed. We therefore performed a left hepatic lobectomy[6]. Since the liver parenchyma was normal and soft in consistency, we considered using segments 2 and 3 after excising the hemangioma of the resected left lobe as an allograft for the pediatric patient with acute liver failure presented above. After consulting the adult patient and his family, we obtained informed consent to donate the left hepatic lobe as an allograft. The therapeutic decision was approved by the Ethical Committee of the First Hospital of Jilin University.

②指標組成不明確。表中的指標是綜合指標，不清楚具體包括哪些項目及對應指標，易造成理解分歧。如擋水工程、泄洪工程中的排水孔、土（石）壩中的面（趾）板止水等，在項目劃分表中是單獨列項的，而細部結(jié)構(gòu)指標又綜合了 “排水工程”“止水工程”。從邏輯上理解，二者沒有重疊。再如，設計上常提供廠房照明設施清單，而細部結(jié)構(gòu)指標中又包括了“照明工程”，不知道廠房是否除外。

The left hepatic lobe was resected using the standard technique described previously[12]. We found a 9 cm diameter mass in the left hepatic lobe. Intra-operative ultrasonography and pathological examination confirmed the diagnosis of hemangioma. Intrahepatic cholangiography was conducted,and no intrahepatic bile duct anomaly was detected (Figure 2). After harvest of the left hepatic lobe,

resection of segment 4 of the graft and hepatic vein reconstruction was performed (Figure 3). The left hepatic vein of the donor liver and the left lateral marginal vein of the left lobe were opened and reconstructed to obtain a width of approximately 2.5 cm in order to avoid venous outflow obstruction[13]. The cold ischemic time was 4 h 17 minutes, and the estimated blood loss during the donor operation was 210 mL.

The liver allograft and recipient weighed 190 g and 8.7 kg, respectively, with a graft-to-recipient weight ratio (GRWR) of 2.1%. The liver allograft was implanted into the recipient using a piggyback orthotopic liver transplant procedure. Intra-operatively, the native liver was cirrhotic, 12 cm × 10 cm × 8 cm in size, yellow-green in color, and firm in consistency. There were multiple nodules of different sizes on the liver surface. There were no palpable emboli in the main portal vein and no obvious masses in the abdominal organs. The liver allograft was placed on the right side in the abdominal cavity of the recipient (Figure 2). First, the inferior vena cava of the recipient was anastomosed to the left hepatic vein of the donor liver by continuous suture in an inverted triangle pattern. Then, the donor and recipient portal veins were anastomosed with continuous valgus suture followed by anastomosis between the left hepatic artery of the donor and the recipient. After reperfusion, there was no bleeding from the resection site. Roux-en-Y hepaticojejunostomy was performed by anastomosing the left hepatic duct of the donor liver to the recipient Roux-en-Y jejunal limb in an end-to-side fashion under magnification.Intra-operative ultrasound revealed satisfactory blood flow of the liver allograft and no constriction of the hepatic vein (Figure 2). The operation time was 8 h, the warm ischemic time was 35 min, and the estimated blood loss was 150 mL.

OUTCOME AND FOLLOW-UP

整個安葬過程，阿里都很乖。羅四強保鏢一樣貼身隨他。他一躁亂，羅四強就放手機里的哀樂。阿里一聽到哀樂，就會靜下。羅四強說：“這是你姆媽睡著的聲音。”阿里便會大聲地“哦”上一聲，表示明白，于是又安靜一陣。

DlSCUSSlON

The pediatric patient had jaundice with high fever for 3 days. The jaundice subsided after the Kasai operation and gradually worsened over a month, and by the time she was admitted to the hospital, she had altered sensorium.

The CT of the pediatric patient showed dilated intrahepatic biliary system and diffuse hepatomegaly(Figure 1). Triple phase contrast enhanced computed tomography of the abdomen of the adult revealed a 9.0 cm × 5.8 cm hemangioma in segment 4 of the liver (Figure 1).

Anatomical lobectomy is one of the most common operative approaches for the treatment of liver hemangioma[19]. After appropriate pre-operative evaluation, selected livers after hepatic lobectomy can be used for pediatric liver transplantation after the backtable resection of hemangioma and reconstruction of the liver allograft, as shown in the case presented here. This offers a novel strategy for enlarging the donor pool. Sanada

[11] proposed a strategy to use liver allograft with hemangiomas in pediatric LDLT. If the estimated graft liver volume to standard liver volume (GV/SLV) ratio after the tumor resection is more than 40%, then the remnant liver after resection of the hemangioma can be used for transplantation. GRWR is also an important factor for pediatric LDLT, which should be more than 0.8%[20,21]. In the present case, the GV/SLV ratio was 76.84% and GRWR was 2.1%, which were safe and sufficient indicators for LDLT. In the follow-up period, the patient recovered well without any complications with good liver function at two years after the operation.

CONCLUSlON

Liver allografts with hemangiomas can be used in LDLT.

backtable resection of hemangioma during pediatric LDLT is a safe and feasible alternative to

resection. Moreover, backtable resection can effectively shorten the operative time of the donor and reduce the risk of intra-operative bleeding during donor operation. Nevertheless, more cases are needed to confirm this method.

FOOTNOTES

Li SΧ and Tang HN contributed equally to this work; Li SΧ wrote the original draft of the manuscript; Tang HN performed the analyses and interpretation of the imaging findings; Lv GY was responsible for the methodology and data curation; Chen Χ was responsible for the revision and editing of the manuscript; all authors issued final approval for the version to be submitted.

Patients provided informed written consent.

The pediatric patient had undergone Kasai portoenterostomy at the age of one month. The adult patient denied any past illness.

The authors have no conflict of interest to disclose.

The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BYNC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is noncommercial. See: http://creativecommons.org/Licenses/by-nc/4.0/

China

1）框支框架、底部加強部位剪力墻抗震等級為一級，比A級高等的框支剪力墻結(jié)構(gòu)提高一級，并嚴格按照規(guī)范要求采取構(gòu)造措施，且10～12層墻肢設置約束邊緣構(gòu)件，加大約束邊緣構(gòu)件的配筋率和體積配箍率；

Shu-Χuan Li 0000-0001-6809-4283; He-nan Tang 0000-0002-7769-6435; Guo-yue Lv 0000-0001-9115-5945;Χuan Chen 0000-0003-2732-0760.

Ma YJ

A

Ma YJ

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