Case Report  病例報告

Case report  病例報告
Hepatolithiasis  肝內膽管結石
Hepatic duct  肝管
Intrahepatic stones  肝內結石

Hepatolithiasis is characterized by the presence of gallstones in all bile ducts peripheral to the confluence of the left and the right hepatic ducts, regardless of the existence of gallstones in other regions of the biliary system, such as the extrahepatic bile duct and the gallbladder [1]. The factors that can lead to hepatolithiasis include the history of recurrent cholangitis, biliary stricture, hepatic abscess, liver atrophy, liver cirrhosis, and a poor prognosis in intrahepatic cholangiocarcinoma [2]. In East Asia, hepatolithiasis is a frequent condition [1,3]. Though symptoms of the intrahepatic kind are more common in younger age groups, the incidence is more in the fifth and sixth decades [4]. Hepatic resection and percutaneous transhepatic cholangioscopic lithotomy (PTCSL) are the primary therapeutic options for hepatolithiasis [5,6]. This case report follows all the SCARE and PROCESS criteria [7,8].
肝內膽管結石症的特徵是左、右肝管匯合處周圍的所有膽管中均存在膽結石，無論膽道系統其他區域（例如肝外膽管和膽囊）是否存在膽結石 [1]。導致肝內膽管結石症的因素包括復發性膽管炎病史、膽道狹窄、肝膿瘍、肝萎縮、肝硬化以及預後不良的肝內膽管癌 [2]。在東亞，肝內膽管結石症是一種常見疾病 [1,3]。雖然肝內膽管結石症的症狀在年輕人中更為常見，但五、六十歲的人發生率更高 [4]。肝切除術和經皮經肝膽管鏡取石術 (PTCSL) 是肝內膽管結石症的主要治療選擇 [5,6]。本病例報告遵循所有 SCARE 和 PROCESS 標準 [7,8]。

A 38-year-old female with a history of Autoimmune hepatitis and open cholecystectomy 19 years back for symptomatic gallstone was presented with a recurrent episode of right upper abdominal pain. The pain was insidious on the onset, dull, aching, intermittent, and nonradiating. She didn’t give any history of fever, nausea, vomiting, diarrhea or jaundice. She had normal bowel and bladder habits. She had
一名38歲女性，過去有自體免疫性肝炎病史，19年前因膽結石症狀而接受開腹膽囊切除術，現因右上腹部反覆疼痛就診。疼痛起病隱匿，呈鈍痛、隱隱作痛、間歇性疼痛，且無放射徵。患者無發燒、噁心、嘔吐、腹瀉或黃疸病史。排便習慣正常。
similar abdominal pain six months back, and endoscopic ultrasound (EUS) showed a common bile duct (CBD) stone (Fig. 1). There is a family history of gallstones. She has been under corticosteroid for 14 years for Autoimmune Hepatitis. She subsequently underwent endoscopic retrograde cholangiopancreatography (ERCP) for CBD stone removal (Fig. 2).
六個月前出現類似腹痛，內視鏡超音波檢查 (EUS) 顯示總膽管 (CBD) 結石（圖 1）。有膽結石家族史。患者因自體免疫性肝炎接受皮質類固醇治療 14 年。隨後，她接受了內視鏡逆行性胰膽管攝影 (ERCP) 以取出總膽管結石（圖 2）。

On examination, she was moderately built and oriented to time, place, and person, and her vitals were stable. There were no signs of pallor, icterus, clubbing, cyanosis, or dehydration. On Abdominal examination, there was around 3 cm of a surgical scar on the right upper quadrant, soft, non-distended, with mild tenderness present in the right upper quadrant. Blood analysis revealed an increase in alanine aminotransferase (ALT/SGPT) 189U/L, aspartate aminotransferase (AST/ SGOT) $219\mathrm{U}/\mathrm{L}$$219\mathrm{U}/\mathrm{L}$219U//L219 \mathrm{U} / \mathrm{L}, and alkaline phosphatase (ALP) $407\mathrm{U}/\mathrm{L}$$407\mathrm{U}/\mathrm{L}$407U//L407 \mathrm{U} / \mathrm{L}. Magnetic Resonance Cholangiopancreatography (MRCP) which was a multislice two-dimensional (2D) fast-spin-echo showed findings suggestive of hepatolithiasis in the left hepatic duct with dilatation of the left hepatic duct distal to hepatolithiasis (Fig. 3).
身體檢查時，患者身體中等，對時間、地點、人物的定向力良好，生命徵象穩定。沒有蒼白、黃疸、杵狀指、發紺或脫水的徵兆。腹部檢查時，右上腹部有約 3 cm 的手術疤痕，質地柔軟，無膨脹，右上腹部有輕微壓痛。血液分析顯示丙胺酸轉氨酶 (ALT/SGPT) 升高 189U/L，天門冬胺酸氨基轉移酶 (AST/SGOT) $219\mathrm{U}/\mathrm{L}$$219\mathrm{U}/\mathrm{L}$219U//L219 \mathrm{U} / \mathrm{L} ，鹼性磷酸酶 (ALP) $407\mathrm{U}/\mathrm{L}$$407\mathrm{U}/\mathrm{L}$407U//L407 \mathrm{U} / \mathrm{L} 。磁振造影胰膽管攝影 (MRCP) 是一種多層二維 (2D) 快速自旋回波，顯示提示左肝管肝內膽管結石的發現，伴隨肝內膽管遠端左肝管擴張（圖 3）。

She was operated on for a left hepatectomy (left lateral segmentectomy) by a GI surgeon at a tertiary level hospital. The intraoperative assessment showed coarse liver adhesion from previous surgery. Intraoperative contact ultrasonography was done, which did not reveal any calculi in the right hepatic duct and the CBD. The postoperative period was uneventful, and she was discharged on the 4th postoperative day. The postoperative histopathology report showed autoimmune hepatitis
患者在一家三級醫院接受胃腸外科手術，進行左肝切除術（左側肝段切除術）。術中評估顯示，患者先前手術留下的肝臟沾黏較為粗糙。術中接觸性超音波檢查未發現右肝管及肝膽管結石。術後情況順利，患者於術後第四天出院。術後組織病理學報告顯示患者為自體免疫性肝炎。

Fig. 1. A stone of 7.7 mm seen in the distal part of CBD. The CBD measures 6.1 mm distally and the CHD is mildly dilated at 9.1 mm .
圖1. 總膽管遠端可見一顆7.7毫米的結石。總膽管遠端長6.1毫米，總膽管輕度擴張，長度為9.1毫米。

Fig. 2. Cholangiogram showing a 15 mm dilated CBD with a 7 mm stone in the mid CBD . The intrahepatic ducts are mildly dilated.
圖2. 膽管攝影顯示膽總管擴張15毫米，總膽管中部有7毫米結石。肝內膽管輕度擴張。

Fig. 3. MRCP showing hepatolithiasis in the left hepatic duct with dilatation of the left hepatic duct distal to hepatolithiasis
圖 3. MRCP 顯示左肝管肝內膽管結石，肝內膽管遠端左肝管擴張
with liver cirrhosis and sclerosing cholangitis.
患有肝硬化和硬化性膽管炎。

There are two forms of intrahepatic stones: primary stones, which are generated in the intrahepatic bile duct, which are more frequent in East Asian countries, and secondary stones developed in the gallbladder, which are common in Western countries [4]. Primary intrahepatic stones have complicated pathogenesis. Intrahepatic stone formation is most commonly associated with bile stasis caused by postoperative strictures, sclerosing cholangitis, Caroli’s disease, or neoplasms that result in biliary stenosis and stasis [9,10]. The combination of bile stasis, bile infection, malnutrition, and parasite infestation is likely to cause these stones [11,12]. Proliferative cholangitis (linked to ~ 75% of hepatolithiasis cases in Asia) plays an essential role in the pathogenesis of hepatolithiasis and contributes to high rates of biliary restenosis that cause chronic inflammation necessary to produce intramural and extramural peribiliary gland proliferation [13,14]. Notably, hepatolithiasis is more common in the left lobe
肝內結石有兩種：原發性結石，產生於肝內膽管，在東亞國家較常見；繼發性結石，發生於膽囊，在西方國家較常見 [4]。原發性肝內結石的發病機轉複雜。肝內結石的形成最常與術後狹窄、硬化性膽管炎、卡羅利症或腫瘤引起的膽道狹窄和淤滯所致的膽汁淤積有關 [9,10]。膽汁淤積、膽汁感染、營養不良和寄生蟲感染等因素綜合作用，都可能導致肝內結石 [11,12]。增生性膽管炎（與亞洲約75%的肝內膽管結石病例相關）在肝內膽管結石的發病機制中起著至關重要的作用，並導致膽道再狹窄率高，而再狹窄又會引發慢性炎症，從而導致膽管壁內和壁外膽管周圍腺體增生[13,14]。值得注意的是，肝內膽管結石在左葉較常見。
because the left hepatic duct coalesces with the CBD at an acute angle which tends to induce bile stasis when associated with a biliary stricture [4]. Recurrent cholangitis, biliary stricture, hepatic abscess, liver atrophy, liver cirrhosis, and a poor prognosis in intrahepatic cholangiocarcinoma are all related to hepatolithiasis [2].
由於左肝管與總膽管以銳角匯合，當合併膽道狹窄時，容易誘發膽汁淤積[4]。復發性膽管炎、膽道狹窄、肝膿瘍、肝萎縮、肝硬化、肝內膽管癌的不良預後均與肝內膽管結石有關[2]。

Asymptomatic patients exist in whom hepatolithiasis is an incidental finding in abdominal imaging [15]. Typical presenting symptoms involve abdominal discomfort, fever, and jaundice, but cholangiocarcinoma is a long-term adverse consequence of the condition. Primary stones are more prevalent in Eastern patients than secondary stones, and recurring pyogenic cholangitis is the most well-known clinical symptom of the disease [16].
存在一些無症狀患者，肝內膽管結石是腹部影像檢查偶然發現的[15]。典型的就診症狀包括腹部不適、發燒和黃疸，但膽管癌是此疾病的長期不良後果。原發性結石在東方患者中比繼發性結石更常見，復發性化膿性膽管炎是此病最常見的臨床症狀[16]。

Efforts at diagnosing hepatolithiasis should be aimed at accurately locating stones, biliary strictures, and segments of the liver involved and excluding concomitant cholangiocarcinoma [15]. Computed tomography (CT) and abdominal ultrasonography (USG) scans are the principal imaging modalities for hepatolithiasis. Magnetic resonance imaging (MRI) and magnetic resonance cholangiopancreatography (MRCP) can
肝內膽管結石的診斷重點應在於準確定位結石、膽道狹窄及受累肝段，並排除合併的膽管癌[15]。電腦斷層掃描 (CT) 和腹部超音波 (USG) 掃描是肝內膽管結石的主要影像檢查方法。磁振造影 (MRI) 和磁振造影胰膽管攝影 (MRCP) 可以
produce more detailed pictures of the bile duct and detect stones without exposing the patient to radiation [17]. In obstructive jaundice, magnetic resonance cholangiopancreatography (MRCP) has accuracies of 96-100% and 90% for the level and cause of obstruction, respectively, as well as sensitivity, specificity, and accuracy of $97\mathrm{\%}$$97\mathrm{\%}$97%97 \%, $99\mathrm{\%}$$99\mathrm{\%}$99%99 \%, and $98\mathrm{\%}$$98\mathrm{\%}$98%98 \%, respectively, in detecting and locating intrahepatic stones [18].
產生更詳細的膽管影像並檢測結石，而無需患者暴露於輻射 [17]。對於阻塞性黃疸，磁振造影胰膽管攝影 (MRCP) 對阻塞部位和原因的準確率分別為 96%-100% 和 90%，檢測和定位肝內結石的敏感度、特異性和準確率分別為 $97\mathrm{\%}$$97\mathrm{\%}$97%97 \% 、 $99\mathrm{\%}$$99\mathrm{\%}$99%99 \% 和 $98\mathrm{\%}$$98\mathrm{\%}$98%98 \% [18]。

Hepatolithiasis care is complex, including interdisciplinary treatment to remove stones and bile stasis. The best management for intrahepatic stones is not yet known; however, various surgical and noninvasive methods are available. Nonetheless, the primary aim should be the total removal of stones and eradication of biliary stasis and infection [19]. Non-surgical therapy options for hepatolithiasis include percutaneous transhepatic cholangioscopy lithotripsy, and surgical management includes hepatectomy [14,17,20].
肝內膽管結石的治療非常複雜，包括跨學科治療以去除結石和膽汁淤積。目前尚不清楚肝內結石的最佳治療方法；然而，目前已有多種手術和非侵入性治療方法。儘管如此，首要目標仍應是徹底清除結石並消除膽汁淤積和感染 [19]。肝內膽管結石的非手術治療包括經皮肝穿刺膽管鏡碎石手術，手術治療包括肝切除術 [14,17,20]。

Regardless of treatment modality, these individuals have a high rate of residual stones [5]. The surgical approach to hepatolithiasis involves the removal of the affected segment(s). Hepatectomy removes stones, eliminates strictures and the consequent bile stasis, which causes stone formation, and eradicates the risk of cholangiocarcinoma; in rare cases, it removes a known intraductal tumor [15].
無論採用何種治療方式，這些患者的殘留結石率都很高 [5]。肝內膽管結石的手術治療包括切除受影響的節段。肝切除術可以去除結石，消除狹窄及由此導致的膽汁淤積（導致結石形成），並消除膽管癌的風險；在極少數情況下，肝切除術還可以切除已知的膽管內腫瘤 [15]。

Hepatolithiasis is quite common in the 5th and 6th decade, primarily seen in South East Asia; however, in our case, the presentation is in the 3rd decade with a known case of autoimmune hepatitis and sclerosing cholangitis with a history of open cholecystectomy and CBD stone removal. It is essential to practice an interdisciplinary approach to best treat and manage these patients. We start with appropriate imaging and a diligent review of imaging studies with radiologists and surgeons before choosing the optimal treatment path. Early diagnosis is crucial; treatment should be multidisciplinary and complete to avoid recurrence and complications.
肝膽管結石在五、六十歲族群中較為常見，主要見於東南亞；然而，本例患者為三十多歲，已知患有自體免疫性肝炎和硬化性膽管炎，並曾接受過開腹膽囊切除術和總膽管結石摘除術。採用跨學科方法對此類患者進行最佳治療和管理至關重要。我們首先會進行適當的影像學檢查，並與放射科醫生和外科醫生仔細回顧影像學研究，然後再選擇最佳治療方案。早期診斷至關重要；治療應採用多學科綜合治療，以避免復發和併發症。

Exempted by our institution.
經我機構豁免。

None declared.  沒有申明。

Lukash Adhikari: involved in patient management, data collection, design of study, manuscript writing and revision.
Lukash Adhikari：參與病患管理、資料收集、研究設計、手稿撰寫和修改。

Eliz Achhami: design of study, data collection, evidence collection, manuscript writing and revision, corresponding author.
Eliz Achhami：研究設計、資料收集、證據收集、手稿撰寫和修改、通訊作者。

Nabin Bhattarai: design of study, evidence collection, manuscript revision.
Nabin Bhattarai：研究設計、證據收集、手稿修改。

Ashim Kandel: design of study, data collection, manuscript revision Abhigan Babu Shrestha: design of study, manuscript revision.
Ashim Kandel：研究設計、資料收集、手稿修改 Abhigan Babu Shrestha：研究設計、手稿修改。

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.
患者已獲得書面知情同意，同意發表本病例報告及附圖。本期刊主編可應要求提供書面同意書副本以供審閱。

The Guarantor is the one or more people who accept full responsibility for the work and/or the conduct of the study, had access to the data, and controlled the decision to publish Eliz Achhami.
擔保人是承擔本研究工作和/或實施的全部責任、有權存取資料並控制發布 Eliz Achhami 的決定的一人或多人。

Not commissioned, externally peer reviewed.
未委託，外部同儕審查。

The authors declare that they have no conflicts of interest.
作者聲明他們沒有利益衝突。

Supplementary data to this article can be found online at https://doi. org/10.1016/j.amsu.2022.104788.
本文的補充資料可在網路上找到 ：https://doi.org/10.1016/j.amsu.2022.104788 。

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