Case Report Open Access
Copyright ©The Author(s) 2015. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. May 28, 2015; 21(20): 6409-6416
Published online May 28, 2015. doi: 10.3748/wjg.v21.i20.6409
Liver transplantation for a giant mesenchymal hamartoma of the liver in an adult: Case report and review of the literature
Jiang Li, Jin-Zhen Cai, Qing-Jun Guo, Jun-Jie Li, Xiao-Ye Sun, Zhong-Yang Shen, Department of Transplant Surgery, Tianjin First Central Hospital, Tianjin 300192, China
Zhan-Dong Hu, Department of Pathology, Tianjin First Central Hospital, Tianjin 300192, China
David KC Cooper, Thomas E Starzl Transplantation Institute, University of Pittsburgh, Pittsburgh, PA 15260, United States
Author contributions: Shen ZY, Cai JZ and Guo QJ performed transplant surgery and provided the intellectual content; Li JJ summarized the clinical data; Sun XY performed the follow-up; Hu ZD contributed to the pathology; Li J performed a literature review and wrote the initial manuscript; Cooper DKC contributed critical comments and revised the manuscript; all authors read, contributed to, and approved the final manuscript.
Supported by National Natural Science Foundation of China, No. 81400680; and the National High Technology Research and Development Program of China, No. 2012 AA021001.
Ethics approval: Approval from the Ethics Committee of the Tianjin First Central Hospital was obtained for this study.
Informed consent: The patient gave her written informed consent before entering the study and gave consent to the study protocol.
Conflict-of-interest: We declare that we have no financial and personal relationships with other people or organizations that can inappropriately influence our work. There is no professional or other personal interest of any nature or kind in any product, service and/or company that could be construed as influencing the position presented in, or the review of, the manuscript.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 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 non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Zhong-Yang Shen, MD, Department of Transplant Surgery, Tianjin First Central Hospital, Nankai District of Tianjin Rehabilitation Road No. 24, Tianjin 300192, China. shenzy_009@sina.com
Telephone: +86-22-23626860 Fax: +86-22-23626199
Received: November 2, 2014
Peer-review started: November 3, 2014
First decision: December 26, 2014
Revised: January 26, 2015
Accepted: February 11, 2015
Article in press: February 11, 2015
Published online: May 28, 2015
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Abstract

Mesenchymal hamartomas of the liver (MHLs) in adults are rare and potentially premalignant lesions, which present as solid/cystic neoplasms. We report a rare case of orthotopic liver transplantation in a patient with a giant MHL. In 2013, a 34-year-old female sought medical advice after a 2-year history of progressive abdominal distention and respiratory distress. Physical examination revealed an extensive mass in the abdomen. Computed tomography (CT) of her abdomen revealed multiple liver cysts, with the diameter of largest cyst being 16 cm × 14 cm. The liver hilar structures were not clearly displayed. The adjacent organs were compressed and displaced. Initial laboratory tests, including biochemical investigations and coagulation profile, were unremarkable. Tumor markers, including levels of AFP, CEA and CA19-9, were within the normal ranges. The patient underwent orthotopic liver transplantation in November 2013, the liver being procured from a 40-year-old man after cardiac death following traumatic brain injury. Warm ischemic time was 7.5 min and cold ischemic time was 3 h. The recipient underwent classical orthotopic liver transplantation. The recipient operative procedure took 8.5 h, the anhepatic phase lasting for 1 h without the use of venovenous bypass. The immunosuppressive regimen included intraoperative induction with basiliximab and high-dose methylprednisolone, and postoperative maintenance with tacrolimus, mycophenolate mofetil, and prednisone. The recipient’s diseased liver weighed 21 kg (dry weight) and measured 41 cm × 32 cm × 31 cm. Histopathological examination confirmed the diagnosis of an MHL. The patient did not experience any acute rejection episode or other complication. All the laboratory tests returned to normal within one month after surgery. Three months after transplantation, the immunosuppressive therapy was reduced to tacrolimus monotherapy, and the T-tube was removed after cholangiography showed no abnormalities. Twelve months after transplantation, the patient remains well and is fulfilling all normal activities. Adult giant MHL is extremely rare. Symptoms, physical signs, laboratory results, and radiographic imaging are nonspecific and inconclusive. Surgical excision of the lesion is imperative to make a definite diagnosis and as a cure. Liver transplantation should be considered as an option in the treatment of a non-resectable MHL.

Key Words: Liver; Mesenchymal hamartoma; Adult; Organ donor; After cardiac death; Transplantation

Core tip: Mesenchymal hamartoma of the liver is a rare disease in adults. Only 45 patients with this condition have been reported worldwide. This report presents a rare case of adult giant mesenchymal hamartoma of the liver that could not be treated by partial hepatectomy. Orthotopic liver transplantation relieved compression of other organs and avoided the risk of malignant change. Liver transplantation should be considered as an option in the treatment of non-resectable benign hepatic tumors.



INTRODUCTION

Mesenchymal hamartoma of the liver (MHL) was first described by Edmondson in 1956[1]. It is a rare mesenchymal tumor affecting almost exclusively infants and children in the first two years of life, with a slight male predilection. Its occurrence in children older than five years is rare (about 5% of cases) and is extremely rare in adults[2-4]. MHL is a potentially premalignant lesion that presents as a solid/cystic neoplasm. The patient’s symptoms are typically nonspecific, though abdominal pain is the most common. Laboratory results are noncontributory and radiographic imaging is variable and inconclusive. Needle biopsy is rarely diagnostic and surgical excision of symptomatic or enlarging lesions is recommended to exclude the possibility of malignancy and to establish a diagnosis[5].

CASE REPORT

A 34-year-old, previously healthy, woman presented in 2011 with abdominal fullness and loss of appetite. She took no medications, had no history of liver disease, and denied alcohol and drug use, including the use of anabolic steroids. She presented to our hospital with increasing abdominal girth, abdominal pain, and vomiting. Physical examination revealed a grossly distended abdomen without evidence of ascites, a firm and massively enlarged liver extending below the umbilicus, and tenderness in the upper quadrant. Contrast enhanced computed tomography (CT) of the abdomen revealed near replacement of the liver with diffuse cystic masses of low density (Figure 1). Initial laboratory test results were unremarkable. Hematological, biochemical investigations and the coagulation profile were within normal limits. Tumor markers, including levels of α-fetoprotein, and carcinoembryonic antigen, carbohydrate antigen 19-9, were within the normal ranges. Serology for hepatitis B virus, hepatitis C virus and human immunodeficiency virus was negative. The extensive hepatic involvement precluded resection, and so she was evaluated and placed on the waiting list for liver transplantation.

Figure 1
Figure 1 Contrast enhanced computed tomography of the abdomen revealed the near replacement of the liver with diffuse cystic masses of low density. A: Enhanced computed tomography scan shows the near replacement of the liver with diffuse cystic masses, leaving only small amounts of liver parenchyma. The portal vein and inferior vena cava are obviously compressed; B: The massively enlarged liver essentially occupies the entire abdominal cavity, with other abdominal organs being compressed and displaced.

The patient underwent orthotopic liver transplantation in November 2013. Our techniques of organ procurement and preservation have been previously described[6,7]. The liver graft was procured from a 40-year-old male donor after cardiac death. The liver graft was preserved in 4  °C UW solution. The warm ischemia time was 7.5 min and cold ischemia time was 3 h.

The native diseased liver filled about 80% of the abdominal cavity and displaced the normal vascular anatomy. The excised diseased native liver weighed 20 kg (dry weight) and measured 41 cm × 32 cm × 31 cm (Figure 2). The recipient operation was conducted according to the classical orthotopic liver transplantation procedure[8]. The whole transplant procedure took 8.5 h and the total blood volume loss was 5500 mL. A blood reinfusion system replaced 3000 mL, and an additional 10 units of packed RBC and 1000ml of plasma were infused. The anhepatic phase lasted for 1 h without the use of venovenous bypass. After release of the vascular clamps, Doppler ultrasound demonstrated the liver graft to be well perfused (Figure 3). The patient was extubated on the second day after surgery.

Figure 2
Figure 2 Intraoperative view of the tumor mass (A) and the excised liver (B).
Figure 3
Figure 3 View of the operative field after liver transplantation, demonstrating the well-perfused liver graft.

The immunosuppressive regimen included intraoperative induction with basiliximab and high-dose methylprednisolone, and postoperative maintenance with tacrolimus, mycophenolate mofetil and prednisone. No acute rejection episode was documented. The patient was discharged home on postoperative day 20, at which time all laboratory tests were within normal limits. Three months after the operation, the immunosuppressive regimen was reduced to tacrolimus monotherapy, and the T-tube was removed after cholangiography showed no abnormalities. After 12 mo, the patient remains well and is carrying out all normal activities.

Pathologic examination of the excised diseased native liver was carried out. It contained multiple well-circumscribed masses, ranging in diameter from 2-16 cm. All masses were cystic in the central portion and contained 20-50 mL of muddy yellowish or bloody fluid. The liver mass contained dilated bile ducts with connective tissue forming multiple cysts. Histologically, corresponding to the cystic areas noted grossly, myxoid stroma and spindle cells showed smooth muscle differentiation, confirmed by positive staining for vimentin and smooth muscle actin. Benign dilated bile ducts were confirmed by positive staining for cytokeratin 7. In peripheral areas, only small amounts of liver tissue remained, with a lack of lobular architecture. There was a clear boundary between the liver parenchyma and proliferating connective tissue (Figure 4). The diagnosis of MHL was based on the typical morphological appearance, as described above.

Figure 4
Figure 4 Clear boundary between liver parenchyma and proliferating connective tissue. A: The mass consisted of loose connective tissue full of myxoid matrix forming visible cysts (upper arrow). Small amounts of remaining liver tissue, with a lack of lobular architecture, were located in peripheral areas (lower arrow) (HE, original magnification × 100); Myxoid stroma with spindle cells showing smooth muscle differentiation were confirmed by positive staining for vimentin (B) and smooth muscle actin (C) (original magnification × 100); Benign dilated bile ducts were confirmed by positive staining for cytokeratin 7 (D) (original magnification × 100).
DISCUSSION

MHL was first reported by Maresch in 1903[9]. Until relatively recently, this disease was known by different names, such as cavernous lymph adenomatoid tumor, bile cell fibroadenoma and benign mesenchymoma. The first definitive description of MHL was provided by Edmondson[1]. While the precise pathogenesis of MHL is uncertain, the most common theory relates to aberrant mesenchymal development in the portal tract, likely related to the bile ducts[10,11].

The clinical presentation of MHL appears to depend on the age of the patient. Most pediatric patients present with painless abdominal enlargement, normally appreciated by their parents[5]. However, in adult patients (age range: 19-87 years; females 62%, mean age 39 years; males 40%, mean age 60 years (Table 1), clinical features included hepatomegaly, and diffuse abdominal pain or pain in the right hypochondrium or left upper quadrant[12-14]. In severe cases, there may be compression of the diaphragm and lungs causing respiratory difficulties[4]. In the present case, the patient suffered from progressive abdominal distention and respiratory distress caused by the expanding multiple cystic masses distributed throughout the liver.

Table 1 Cases of adult mesenchymal hamartomas of the liver reported in the literature.
No.Ref.YearSexAge (yr)Clinical manifestationSize (cm)Gross appearance(cystic or solid)Liver lobe(s)affectedSurgical treatment
1Yamamura et al[26]1976F22NANACysticBothNA
2Grases et al[27]1979F19Abdominal pain, jaundice, hepatomegaly24 × 19 × 8CysticLeftLeft hepatic lobectomy
3Li et al[28]1983F21Asymptomatic17 × 10CysticRightHemihepatectomy
4Kawata et al[29]1984F43NA22 × 15 × 10SolidLeftNA
5Ishizuka et al[30]1985M59NA30 × 28 × 12CysticRightNA
6Kawakami et al[31]1986M67NANACysticRightNA
7Jennings et al[32]1987F32Asymptomatic14 × 11CysticLeftLeft hepatic lobectomy
8Kato et al[33]1988M66AsymptomaticNASolidLeftLeft hepatic lobectomy
9Gutierrez et al[34]1988F30NA18BothBothNon-resectable
10Gramlich et al[35]1988F28Abdominal distention, hepatomegaly30 × 20 × 14SolidRightRight hepatic trisegmentectomy
11Alanen et al[36]1989F20Asymptomatic6 × 8CysticLeftLeft hepatic lobectomy
12Ito et al[37]1989F43NA16 × 16 × 7.7CysticBothNA
13Urabe et al[38]1990F39Asymptomatic1.2SolidLeftLeft hepatic lobectomy
14Drachenb et al[39]1991F69Asymptomatic26 × 20 × 11.5CysticLeftNA
15Wada et al[40]1992M62Asymptomatic6 × 6 × 4.5SolidLeftHepatectomy
16Chau et al[41]1994M53Abdominal pain20 × 14 × 10CysticRightNA
17Megremis et al[42]1994F56Abdominal pain7.5CysticBothNA
18Yamamoto et al[43]1994M52Abdominal discomfort, weight loss6 × 4 × 3.5CysticLeftLateral segmentectomy
19Chung et al[44]1999F57Abdominal discomfort, weight loss6 × 4 × 3.5SolidRightRight hepatectomy
20Papastratis et al[45]2000F21Abdominal pain, abdominal mass17 × 10CysticRightRight hepatectomy
21Cook et al[13]2002F46Abdominal pain6 × 4 × 5CysticRightRight hepatectomy
22Cook et al[13]2002F66Cough and shortness of breath5 × 4 × 2CysticRightRight hepatectomy
23Cook et al[13]2002F63Abdominal pain11 × 16 × 24SolidLeftLeft hepatic lobectomy
24Mao et al[46]2002M44Abdominal discomfort2 × 2SolidLeftHepatectomy
25Mao et al[46]2002F43Asymptomatic3 × 4 × 4CysticRightRight hepatectomy
26Mao et al[46]2002M76Abdominal pain4 × 5 × 4CysticRightRight hepatectomy
27Brkic et al[47]2003M38Abdominal pain8 × 5SolidRightRight hepatectomy
28Kim et al[48]2003MNAAsymptomatic5BothRightNA
29Yesim et al[12]2005F54NA2.5 × 2.5 × 1.5CysticLeftTotal cystectomy
30Yesim et al[12]2005F51NA6 × 7 × 8CysticRightUnroofing procedure
31Kim et al[49]2006F40Asymptomatic5 × 5CysticRightRight hepatectomy
32Ayadi-Kaddour et al[50]2006F21NA11 × 5CysticLeftNA
33Hernández et al[25]2006M51NA19 × 13SolidRightLiver transplantation (4th reported1)
34Chang et al[51]2006M79Asymptomatic2 × 2NARightNA
35Chang et al[51]2006F39Asymptomatic5 × 5CysticNANA
36Li et al[17]2007F33Abdominal distention16BothBothNA
37Mori et al[52]2008F36Abdominal distention20 × 15 × 10CysticRightRight hemihepatectomy
38Giunippero et al[53]2009M87Abdominal distention20 × 20CysticRightHemihepatectomy
39Nakajo et al[54]2009M38Asymptomatic5 × 5SolidRightRight hepatectomy
40Klaassen et al[5]2010F53NA9 × 9 × 7.5CysticRightHepatectomy
41Kulkarni et al[55]2010F20Abdominal mass, abdominal pain14 × 11cysticRightMass resection
42Tucker et al[56]2012W74Abdominal distention, abdominal pain18 × 15 × 13cysticLeftLeft hepatectomy
43Liu et al[57]2013M42Asymptomatic1.5 × 1.0 × 1.0solidLeftHepatectomy
44Lakić et al[58]2014M44Asymptomatic2.9 × 3.1 × 3.5NALeftHepatectomy
45Sharma et al[59]2014M81Abdominal distention21.8 × 12.3 × 18.6cysticLeftHepatectomy
46Current case2014F34abdominal discomfort, dyspnea41 × 32 × 31cysticBothLiver transplantation (5th reported1)

Concerning the localization and structure of the tumor, pediatric and adult populations have different characteristics. MHLs are more common in the left liver lobe in children. In adults, 17 cases (38%) were localized to the left lobe, 22 (49%) to the right lobe, and in six (13%) extended into both lobes (Table 1). All six cases of MHLs involving both lobes occurred in females. Among 45 cases of MHLs, 30 (67%) presented with cystic lesion, 12 (26%) with solid lesions, and three (7%) with both types. Of 30 cases of cystic MHLs, 21 (70%) were reported in females and only nine (30%) in males (Table 1).

MHLs are difficult to diagnose by laboratory tests or other investigations because of its non-specificity. Liver function tests and AFP values for MHLs are usually within normal limits[15]. Additionally, all imaging methods, including ultrasonography, CT and magnetic resonance imaging (MRI), provide nonspecific findings. The differential diagnosis of a cystic MHL includes simple liver cysts, hydatid cysts, biliary cystadenocarcinoma, and cystic metastases. If a lesion consists of a solid mass, the differential diagnosis includes focal nodular hyperplasia, hepatic adenoma, cavernous hemangioma, angiomyolipoma and hepatocellular carcinoma. In the present case, the initial abdominal enhanced CT scan revealed multiple liver cysts, which could easily have been misdiagnosed as a polycystic liver.

The diagnosis of MHL often relies on histological examination of tissue obtained by biopsy or by tumor resection; however, the histological appearance of the stromal component of an MHL can be variable. Hematoxylin and eosin (HE) staining, as well as immunohistochemical studies, have indicated MHLs as having spindle cells positive for vimentin and smooth muscle actin and negative for CD31, CD34 and S100 proteins, while the ducts stain positive for cytokeratin 7 and negative for cytokeratin 20[13,16].

MHLs have premalignant potential, particularly in adult patients[17]. The potential malignant evolution of a subset of MHLs into embryonal sarcoma or angiosarcoma supports the necessity for complete surgical excision both in children and adults[4,18]. Incomplete resection or marsupialization must be avoided because of the possibility of recurrence[19-21]. Laparoscopic liver resection for MHLs has been reported with successful outcomes[22].

Very rarely an MHL is non-resectable, even in an experienced center, and liver transplantation may have to be considered. Tepetes et al[23] reported two children who underwent liver transplantation following partial resections for MHLs. One died from intraoperative bleeding and the other survived. Bejarano et al[24] described a neonate with a recurrent MHL (after resection) who underwent successful liver transplantation. Hernández et al[25] reported the first case of an MHL in an adult that was treated by liver transplantation.

In conclusion, giant MHLs in adults are extremely rare. Clinical features, laboratory results and radiographic imaging are often nonspecific and inconclusive. Surgical excision of the whole lesion is imperative for both definitive diagnosis and cure. Liver transplantation should be considered as an option in the treatment of non-resectable MHLs.

COMMENTS
Case characteristics

A 34-year-old female with a history of progressive abdominal distention and respiratory distress.

Clinical diagnosis

Physical examination revealed a grossly distended abdomen without evidence of ascites, a firm and massively enlarged liver extending below the umbilicus, and tenderness in the upper quadrant.

Differential diagnosis

Polycystic liver, hydatid cyst, biliary cystadenocarcinoma and cystic metastases.

Laboratory diagnosis

Laboratory test results were unremarkable and non-diagnostic.

Imaging diagnosis

Abdominal computed tomography scan showed multiple liver cysts, with the diameter of the largest cyst being 16 cm × 14 cm. The liver hilar structures were not clearly displayed. The adjacent organs were compressed and displaced.

Pathological diagnosis

Histological examination showed dilated bile ducts and extensive connective tissue in the liver mass, while immunohistochemical staining showed positivity for vimentin, smooth muscle actin and cytokeratin 7.

Treatment

The patient underwent orthotopic liver transplantation.

Related reports

Mesenchymal hamartoma of the liver is a rare disease in adults and only 45 patients with this condition have been reported; the references are cited.

Term explanation

Mesenchymal hamartoma of the liver is a rare and potentially premalignant lesion that presents as a solid/cystic neoplasm. The pathogenesis remains incompletely understood; however, these lesions have generally been considered to represent a developmental abnormality in bile duct plate formation.

Experiences and lessons

This case report represents a successful application of liver transplantation for adult giant mesenchymal hamartomas of the liver, which could not be treated by conventional partial hepatectomy. We recommend that liver transplantation should be considered as an option in the treatment of non-resectable benign hepatic tumors.

Peer-review

This paper is a case report of a 34-year-old woman with liver transplantation for a giant mesenchymal hamartoma of the liver. Mesenchymal hamartoma of liver is a rare disease in adults and only 31 patients have been reported to date worldwide.

Footnotes

P- Reviewer: Nakayama Y, Ohkohchi N S- Editor: Ma YJ L- Editor: Stewart G E- Editor: Wang CH

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