Editorial Type:
Article Category: Case Report
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Online Publication Date: 01 Sept 2016

A Case of Cavernous Hemangioma of the Gallbladder Treated With Single-Incision Laparoscopic Cholecystectomy

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Page Range: 431 – 436
DOI: 10.9738/INTSURG-D-15-00015.1
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Although hepatic hemangiomas are common, gallbladder (GB) hemangiomas are extremely rare. We present a case of a cavernous GB hemangioma, which was detected during routine ultrasonography screening. The 51-year-old female patient was asymptomatic and had no relevant medical history. The preoperative imaging findings, including those of endoscopic ultrasonography, suggested that malignancy was highly unlikely in this submucosal tumor. Thus, we performed a single-incision laparoscopic cholecystectomy as a diagnostic and curative procedure. The postoperative pathological examination confirmed a GB hemangioma. In this case, the preoperative diagnosis was quite difficult, especially because of this rare presentation of a GB hemangioma. This is probably why the definitive diagnosis is established postoperatively in most previously reported cases of GB hemangioma.

Keywords: Cholecystectomy; Endoscopic ultrasonography; Gallbladder; Hemangioma; Laparoscopic surgery; Single incision

Hemangiomas occur commonly in several organs, including the liver, brain, lung, and skeletal muscle. However, gallbladder (GB) hemangiomas are extremely rare.1,2 In a PubMed search, we found only 8 other cases of GB hemangioma.1,3–9 In general, it is quite difficult to make a definitive diagnosis of this rare condition. In addition, the definitive diagnosis of GB tumor can be quite difficult without pathological examinations, such as bile cytology. All of the previously reported cases of GB hemangiomas underwent surgery and a definitive diagnosis was made only after pathological examination of the specimen. Open laparotomy was performed in all cases because the possibility of malignancy could not be excluded based on preoperative imaging evaluations alone. In this study, we report a case of a cavernous GB hemangioma where the preoperative imaging findings, including those obtained by endoscopic ultrasonography (EUS) with Sonazoid (Daiichi Sankyo, Tokyo, Japan), excluded the possibility of malignancy, thereby allowing us to perform a single-incision laparoscopic cholecystectomy.

Case Report

A 51-year-old woman was referred to our hospital for further evaluation because abdominal screening by ultrasonography (US) detected a GB mass. The patient was asymptomatic and had no medical history, except for cervical conization to treat cytological atypia.

The physical examination on admission was unremarkable. Laboratory data, including the hepatobiliary function and tumor marker levels, were within the normal limits: hemoglobin = 9.9 g/dL; total bilirubin level = 1.4 mg/dL; aspartate aminotransferase = 15 IU/L; alanine aminotransferase = 10 IU/L; and alkaline phosphatase = 143 IU/L. The tumor markers were also within the normal limits: carcinoembryonic antigen = 1.6 ng/mL; carbohydrate antigen 19-9 = 12.1 U/dL; and cancer antigen 125 = 24.1 U/mL.

Abdominal computed tomography (CT) detected a slightly hyperdense mass with a diameter of 17 mm at the GB fundus and a few dots in the marginal area, which were enhanced by the contrast medium, were likely to be Rokitansky–Ashoff sinuses (Fig. 1A, 1B). Transabdominal US identified a mass adjoining the GB fundus. Moreover, EUS detected the precise structure of a well-defined, round, and homogeneous mass arising from the GB fundus. The mucosal surface was smooth, intact, and homogeneous without thickened areas (Fig. 2). Endoscopic US with color Doppler and Sonazoid showed that blood flow was almost entirely absent from the mass, thereby indicating that the mass was not malignant. However, it was difficult to establish a definitive diagnosis based on the imaging findings alone, and thus adenomyomatosis or a submucosal tumor (e.g., gastrointestinal stromal tumor) was suspected. Therefore, we decided to perform a diagnostic operation by single-incision laparoscopic cholecystectomy. Intraoperatively, a dark-brown nodular lesion was observed protruding from the GB fundus, which appeared to be covered with the GB serosa in a continuous manner. Part of it also adhered to the liver surface (Fig. 3). The mass and GB were resected with a portion of liver parenchyma. The operative specimen was a mass that measured 18 × 17 mm, which was located at the GB fundus and it originated from the GB wall (Fig. 4). The mucosal surface of the GB was intact, according to EUS. The pathological examination found large dilated submucosal vascular channels covered by a single layer of endothelium (Fig. 5). Because of its continuity with GB, the mass was diagnosed as a cavernous GB hemangioma and malignancy was excluded. The patient's postoperative course was uneventful. She was discharged on postoperative day 3 and her progress was favorable at the 1-year postoperative follow-up.

Fig. 1 . Abdominal CT. (A) A slightly hyperdense lesion at the gallbladder fundus was visible by plain CT. (B) A few dots in the marginal area, which were enhanced by the contrast medium, were visible by contrast-enhanced CT.Fig. 1 . Abdominal CT. (A) A slightly hyperdense lesion at the gallbladder fundus was visible by plain CT. (B) A few dots in the marginal area, which were enhanced by the contrast medium, were visible by contrast-enhanced CT.Fig. 1 . Abdominal CT. (A) A slightly hyperdense lesion at the gallbladder fundus was visible by plain CT. (B) A few dots in the marginal area, which were enhanced by the contrast medium, were visible by contrast-enhanced CT.
Fig. 1 Abdominal CT. (A) A slightly hyperdense lesion at the gallbladder fundus was visible by plain CT. (B) A few dots in the marginal area, which were enhanced by the contrast medium, were visible by contrast-enhanced CT.

Citation: International Surgery 101, 9-10; 10.9738/INTSURG-D-15-00015.1

Fig. 2 . Endoscopic US detected a well-defined, round, and homogeneous mass arising from the fundus of the gallbladder wall (upper). The surface of the mucosa was smooth and homogeneous without any thickened areas (lower).Fig. 2 . Endoscopic US detected a well-defined, round, and homogeneous mass arising from the fundus of the gallbladder wall (upper). The surface of the mucosa was smooth and homogeneous without any thickened areas (lower).Fig. 2 . Endoscopic US detected a well-defined, round, and homogeneous mass arising from the fundus of the gallbladder wall (upper). The surface of the mucosa was smooth and homogeneous without any thickened areas (lower).
Fig. 2 Endoscopic US detected a well-defined, round, and homogeneous mass arising from the fundus of the gallbladder wall (upper). The surface of the mucosa was smooth and homogeneous without any thickened areas (lower).

Citation: International Surgery 101, 9-10; 10.9738/INTSURG-D-15-00015.1

Fig. 3 . Intraoperative findings. Laparoscopy detected a dark-brown nodular lesion protruding from the gallbladder fundus and the mass was covered with the gallbladder serosa in a continuous manner.Fig. 3 . Intraoperative findings. Laparoscopy detected a dark-brown nodular lesion protruding from the gallbladder fundus and the mass was covered with the gallbladder serosa in a continuous manner.Fig. 3 . Intraoperative findings. Laparoscopy detected a dark-brown nodular lesion protruding from the gallbladder fundus and the mass was covered with the gallbladder serosa in a continuous manner.
Fig. 3 Intraoperative findings. Laparoscopy detected a dark-brown nodular lesion protruding from the gallbladder fundus and the mass was covered with the gallbladder serosa in a continuous manner.

Citation: International Surgery 101, 9-10; 10.9738/INTSURG-D-15-00015.1

Fig. 4 . Macroscopic findings based on the operative specimen. The surgical specimen was a mass measuring 18 × 17 mm, which was located at the gallbladder fundus and originated from the gallbladder wall. The serosa of the mass was continuous with that of the gallbladder.Fig. 4 . Macroscopic findings based on the operative specimen. The surgical specimen was a mass measuring 18 × 17 mm, which was located at the gallbladder fundus and originated from the gallbladder wall. The serosa of the mass was continuous with that of the gallbladder.Fig. 4 . Macroscopic findings based on the operative specimen. The surgical specimen was a mass measuring 18 × 17 mm, which was located at the gallbladder fundus and originated from the gallbladder wall. The serosa of the mass was continuous with that of the gallbladder.
Fig. 4 Macroscopic findings based on the operative specimen. The surgical specimen was a mass measuring 18 × 17 mm, which was located at the gallbladder fundus and originated from the gallbladder wall. The serosa of the mass was continuous with that of the gallbladder.

Citation: International Surgery 101, 9-10; 10.9738/INTSURG-D-15-00015.1

Fig. 5 . Microscopy. The pathological examination detected large dilated submucosal vascular channels covered by a single layer of endothelium.Fig. 5 . Microscopy. The pathological examination detected large dilated submucosal vascular channels covered by a single layer of endothelium.Fig. 5 . Microscopy. The pathological examination detected large dilated submucosal vascular channels covered by a single layer of endothelium.
Fig. 5 Microscopy. The pathological examination detected large dilated submucosal vascular channels covered by a single layer of endothelium.

Citation: International Surgery 101, 9-10; 10.9738/INTSURG-D-15-00015.1

Discussion

In this study, we presented a case of GB hemangioma. Hemangioma is the most common benign mesenchymal tumor of the liver and its estimated prevalence ranges from 5 to 7%.10 However, to the best of our knowledge, only 8 cases of GB hemangioma have been reported in the medical literature. The details of the 9 cases, including the present case, are listed in Table 1.1,3–9 Arbab1 reported the first case of a GB hemangioma in 1967. According to a review of 8 cases of GB hemangioma by Crucitti,9 open laparotomy was performed in all cases, mainly because the possibility of malignancy could not be excluded based on the preoperative imaging findings alone.7,9 As mentioned above, the very low possibility of malignancy according to our preoperative evaluation allowed us to perform a single-incision laparoscopic surgery in the present case. As far as we know, no other case has been reported in the medical literature where a GB hemangioma was excised by single-incision laparoscopic surgery. We consider that the assessment of the GB tumor by EUS was important in our case because the detection of a well-defined, round, and homogeneous mass without blood flow can exclude malignancy. None of the other case reports mentioned the use of EUS in the preoperative diagnosis. However, we obtained no findings to indicate malignancy in this case (e.g., infiltration of adjacent organ or heterogenic macroscopic appearance), but the operative strategy that we employed could be converted to multiple port laparoscopic surgery or open laparotomy. In addition, if a diagnosis of a GB malignant lesion is established postoperatively based on pathological examination, the patient might have had to undergo a second procedure with curative intent.11

Table 1 Overview of previously reported cases of gallbladder hemangioma
Table 1

Computed tomography might not be very useful for the definitive diagnosis of focal or diffuse thickenings of the GB,12 while it may not be helpful for distinguishing benign and malignant tumors.13 Although we were misled by the enhanced dots in the marginal area of the tumor according to contrast-enhanced CT, this could be a feature that is indicative of hemangiomas. Ultrasound is a noninvasive and relatively safe procedure. However, compared with transabdominal US, EUS has been reported to be an accurate imaging modality for evaluating GB,14 and it may be very useful for assessing the mucosal surface and the depth of tumor invasion in the GB.15,16 In this case, according to EUS, we considered that the mass was unlikely to be malignant based on our assessment of the mucosal surface and the properties of the GB mucosa. Color Doppler examination was not helpful for establishing a definitive diagnosis in this case because it did not detect blood flow within the hemangioma. Similar to our experience, a previous study reported that approximately 50% blood flow within a hemangioma could not be detected by color Doppler,17 which might indicate that color Doppler cannot improve the accuracy of US. However, some previous reports suggest that the presence/absence of blood flow can be a useful sign for differentiating between benign and malignant tumors.18,19 In addition, some reports state that contrast-enhanced US is a highly useful tool that can potentially help differentiate between benign and malignant tumors.19–21 However, it is difficult to establish a preoperative diagnosis when such rare conditions affect the GB, including hemangiomas. In our case, EUS was helpful for excluding malignancy, which allowed us to perform a noninvasive and cosmetically appropriate diagnostic and curative procedure.

Conclusions

Although GB hemangiomas are benign tumors, a definitive preoperative diagnosis is difficult, probably because of their rarity. Endoscopic US is a potentially useful tool that can aid in their diagnosis and it may sometimes help exclude malignancies. However, further studies with a large number of patients are necessary to reach any definite conclusions.

Acknowledgments

Patient care, operation, and manuscript writing: Y. Mizuguch and Y. Akama. Manuscript revision and final article approval: E. Uchida, Y. Nakamura, and N. Taniai. Pathological examination: H. Takata and Z. Naito. Figure and table creation: T. Shimizu and T. Kanda. All authors discussed the results and commented on the manuscript. This case report was not funded. The authors declare no conflict of interest associated with this manuscript.

References

  • 1
    Arbab AA,
    Brasfield R.
    Benign tumors of the gallbladder. Surgery1967;61(
    4
    ):535–540
  • 2
    Christensen AH,
    Ishak KG.
    Benign tumors and pseudotumors of the gallbladder. Report of 180 cases. Arch Pathol1970;90(
    5
    ):423–432
  • 3
    Sewell JH,
    Miron MA.
    Benign cavernous hemangioma of the gallbladder. Arch Pathol1969;88(
    1
    ):30–31
  • 4
    Moffat JH.
    Cavernous hemangioma of the gallbladder and liver. Can J Surg1973;16(
    2
    ):172–175
  • 5
    Cabrera OR,
    Hernandez Viruel JA, Colinabarranco M, Villa Fernandez A. [Visceral multiple angiomatosis. Clinical case and review of the literature]. Prensa Med Mex1977;42(
    7–8
    ):325–330
  • 6
    Jones WP,
    Keller FS,
    Odrezin GT,
    Kelly DR.
    Venous hemangioma of the gallbladder. Gastrointest Radiol1987;12(
    4
    ):319–321
  • 7
    Furukawa H,
    Kanai Y,
    Mukai K,
    Yamasaki S.
    Arteriovenous hemangioma of the gallbladder: CT and pathologic findings. AJR Am J Roentgenol1997;168(
    5
    ):1383
  • 8
    Mayorga M,
    Hernando M,
    Val-Bernal JF.
    Diffuse expansive cavernous hemangioma of the gallbladder. Gen Diagn Pathol1997;142(
    3–4
    ):211–215
  • 9
    Crucitti A,
    La Greca A,
    Antinori A,
    Antonacci V,
    Magistrelli P.
    Cavernous hemangioma of the gallbladder. Case report and review of the literature. Tumori2005;91(
    5
    ):432–435
  • 10
    Farges O,
    Daradkeh S,
    Bismuth H.
    Cavernous hemangiomas of the liver: Are there any indications for resection? World J Surg 1995;19(
    1
    ):19–24
  • 11
    Goetze TO,
    Paolucci V.
    Benefits of reoperation of T2 and more advanced incidental gallbladder carcinoma: analysis of the German registry. Ann Surg2008;247(
    1
    ):104–108
  • 12
    Kumar A,
    Aggarwal S.
    Carcinoma of the gallbladder: CT findings in 50 cases. Abdom Imaging1994;19(
    4
    ):304–308
  • 13
    Furukawa H,
    Kosuge T,
    Shimada K,
    Yamamoto J,
    Kanai Y,
    Mukai K,
    et al.     Small polypoid lesions of the gallbladder: differential diagnosis and surgical indications by helical computed tomography. Arch Surg1998;133(
    7
    ):735–739
  • 14
    Sugiyama M,
    Atomi Y,
    Yamato T.
    Endoscopic ultrasonography for differential diagnosis of polypoid gall bladder lesions: analysis in surgical and follow up series. Gut2000;46(
    2
    ):250–254
  • 15
    Fujita N,
    Noda Y,
    Kobayashi G,
    Kimura K,
    Yago A.
    Diagnosis of the depth of invasion of gallbladder carcinoma by EUS. YMGE1999;50(
    5
    ):659–663
  • 16
    Sadamoto Y,
    Kubo H,
    Harada N,
    Tanaka M,
    Eguchi T,
    Nawata H.
    Preoperative diagnosis and staging of gallbladder carcinoma by EUS. Gastrointest Endosc2003;58(
    4
    ):536–541
  • 17
    Perkins AB,
    Imam K,
    Smith WJ,
    Cronan JJ.
    Color and power Doppler sonography of liver hemangiomas: a dream unfulfilled? J Clin Ultrasound 2000;28(
    4
    ):159–165
  • 18
    Komatsuda T,
    Ishida H,
    Konno K,
    Hamashima Y,
    Naganuma H,
    Sato M
    et al.     Gallbladder carcinoma: color Doppler sonography. Abdom Imaging2000;25(
    2
    ):194–197
  • 19
    Sato M,
    Ishida H,
    Konno K,
    Naganuma H,
    Komatsuda T,
    Watanabe S
    et al.     Localized gallbladder carcinoma: sonographic findings. Abdom Imaging2001;26(
    6
    ):619–622
  • 20
    Liu L-N,
    Xu H-X,
    Lu M-D,
    Xie X-Y,
    Wang W-P,
    Hu B
    et al.     Contrast-enhanced ultrasound in the diagnosis of gallbladder diseases: a multi-center experience. PLoS One2012;7(
    10
    ):e48371
  • 21
    Xie X-H,
    Xu H-X,
    Xie X-Y,
    Lu M-D,
    Kuang M,
    Xu Z-F
    et al.     Differential diagnosis between benign and malignant gallbladder diseases with real-time contrast-enhanced ultrasound. Eur Radiol2010;20(
    1
    ):239–248
Copyright: © 2017 Akama et al.; licensee The International College of Surgeons. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-commercial License which permits use, distribution, and reproduction in any medium, provided the original work is properly cited, the use is non-commercial and is otherwise in compliance with the license. See:
<bold>Fig. 1</bold>
Fig. 1

Abdominal CT. (A) A slightly hyperdense lesion at the gallbladder fundus was visible by plain CT. (B) A few dots in the marginal area, which were enhanced by the contrast medium, were visible by contrast-enhanced CT.

<bold>Fig. 2</bold>
Fig. 2

Endoscopic US detected a well-defined, round, and homogeneous mass arising from the fundus of the gallbladder wall (upper). The surface of the mucosa was smooth and homogeneous without any thickened areas (lower).

<bold>Fig. 3</bold>
Fig. 3

Intraoperative findings. Laparoscopy detected a dark-brown nodular lesion protruding from the gallbladder fundus and the mass was covered with the gallbladder serosa in a continuous manner.

<bold>Fig. 4</bold>
Fig. 4

Macroscopic findings based on the operative specimen. The surgical specimen was a mass measuring 18 × 17 mm, which was located at the gallbladder fundus and originated from the gallbladder wall. The serosa of the mass was continuous with that of the gallbladder.

<bold>Fig. 5</bold>
Fig. 5

Microscopy. The pathological examination detected large dilated submucosal vascular channels covered by a single layer of endothelium.

Contributor Notes

Corresponding author: Yoshiaki Mizuguchi, 1-1-5 Sendagi, Bunkyo-Ku, Tokyo, Japan. Tel.: +81 3 3822 2131; Fax: +81 3 5685 0989; E-mail: yoshi1224@gmail.com
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