We describe two cases where patients undergoing hepatic resection for metastatic disease of colorectal origin were found to have concomitant sclerosing haemangiomas. The typical radiological and histological appearances of these lesions are discussed

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C. Lauder

G. Garcea

G. J. Maddern

H. Kanhere

HPB Surgery

English

PubMed

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Sclerosing Haemangiomas of the Liver: Two Cases of Mistaken Identity

We describe two cases where patients undergoing hepatic resection for metastatic disease of colorectal origin were found to have concomitant sclerosing haemangiomas. The typical radiological and histological appearances of these lesions are discussed.C. Lauder, G. Garcea, H. Kanhere, and G. J. Madder

Lauder, C.

Garcea, G.

Kanhere, H.

Maddern, G.

Adelaide Research & Scholarship

Hindawi Publishing CorporationHPB SurgeryVolume 2009, Article ID 473591, 3 pagesdoi:10.1155/2009/473591Case ReportSclerosing Haemangiomas of the Liver:Two Cases of Mistaken IdentityC. Lauder, G. Garcea, H. Kanhere, and G. J. MaddernDepartment of Hepatobiliary and Upper Gastrointestinal Surgery, The Queen Elizabeth Hospital,28 Woodville Road, Adelaide, SA 5011, AustraliaCorrespondence should be addressed to G. J. Maddern, guy.maddern@adelaide.edu.auReceived 4 May 2009; Revised 26 August 2009; Accepted 9 November 2009Recommended by Hobart W. HarrisWe describe two cases where patients undergoing hepatic resection for metastatic disease of colorectal origin were found to haveconcomitant sclerosing haemangiomas. The typical radiological and histological appearances of these lesions are discussed.Copyright © 2009 C. Lauder et al. This is an open access article distributed under the Creative Commons Attribution License,which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.1. IntroductionHaemangiomas constitute the largest group of noncystic liverlesions [1], as a result they frequently present as confoundinglesions when staging and investigating intra-abdominalmalignancies. The sclerosed variant of liver haemangiomasis considerably less common. This describes two patientswith metastatic colorectal malignancy in whom sclerosedhaemangiomas were also identified.2. Case 1The first patient, a 72-year-old male, presented with largebowel obstruction secondary to a rectal cancer which wastreated with a defunctioning colostomy and adjuvantchemoradiotherapy. Having completed the courseof chemotherapy a staging CT was performed whichdemonstrated an ill-defined low density lesion in segment4a. Dynamically this had enlarged from a CT scan 2 monthspreviously. There was mild contrast enhancement of thelesion between arterial and venous phases with an increaseof approx 5HU in density between the two phases. Anotherlesion was noted in segment 7, again low density, but stablein size when compared to the previous study. This showedless enhancement when compared to the lesion in 4a. Boththese lesions were thought to be consistent with metastaticdeposits.The patient underwent a routine ultra-low anteriorresection with covering loop ileostomy. It was noted atthe time of the laparotomy that there was a palpablemetastasis adjacent to the falciform ligament. Furtherimaging for planning of the liver resection showed bothlesions to have enlarged in size although the lesion insegment VII showed a smaller increase and was indeter-minate in nature. At operation two surface liver lesionswere seen and wedge excision was performed. The his-tology revealed a metastatic deposit of adenocarcinomawithin segment 4a whilst segment VII showed a sclerosedhaemangioma.3. Case 2The second patient, an 84-year-old male, had previouslyundergone a right hemicolectomy for adenocarcinoma andwas noted at the time of the resection to have hepaticmetastases. Having received chemotherapy postoperativelyhe was restaged to assess the feasibility of hepatic resection.The CT demonstrated four lesions in total, with subcentimetre hypodense lesions in segments 2 and 5, andlarger lesions in segments 4b and 6 (Figure 2). The twolarger lesions were thought to be consistent with malignantdisease whilst the smaller segment 2 lesion was indeterminateand the abnormality in segment 5 was in keeping with asimple cyst. At laparotomy the lesions were further evaluatedwith intraoperative ultrasound. This confirmed the lesionsin segments 2 and 5 to be cystic in nature and the patienttherefore proceeded to a partial right hepatectomy withoutincident. Upon histological sectioning of the specimen only2 HPB Surgeryone metastatic deposit was found in segment 6, with thesegment 4b lesion being a sclerosing haemangioma.4. DiscussionThese cases highlight the diﬃculty in diﬀerentiating betweenbenign and malignant diseases in staging colorectal hepaticmetastases. Furthermore we demonstrate the occurrence ofrare hepatic lesions in mimicking malignant disease. Someauthorities consider haemangiomas to be benign congenitalhamartomas. They are composed of masses of blood vesselsthat are atypical or irregular in arrangement and size. Aeti-ology remains unknown with no definite familial or geneticmode of inheritance being described. When fibrosis occursin an existing haemangioma then it may progress to thesclerosed variant. It is diﬃcult to ascertain the precipitatingevent for this to occur. There are numerous reports ofhaemangiomas rupturing spontaneously or in associationwith trauma. It would therefore seem plausible that minorhaemorrhage and thrombosis within a haemangioma mayinstigate fibrotic progression to a sclerosed haemangioma.Makhlouf et al. suggested that mast cells play a pivotal rolein the development of a sclerosed haemangioma, perhapsrepresenting a distinct histological subtype of liver lesion[2].Not all haemangiomas remain stable in size or displaytypical appearances on imaging (due to the development offibrous change) and this may lead to diﬃculty in accuratelypredicting the true nature of these incidental lesions whenthey occur. CT features suggestive of sclerosed haeman-giomas include geographic outline, capsular retraction,decrease in size over time, and loss of previously seen regionsof enhancement. Additional features include the presence oftransient hepatic attenuation diﬀerence, rim enhancement,and nodular regions of intense enhancement as seen intypical haemangiomas [3]. Alternate imaging modalitiessuch as MRI should be considered for further evaluation ofindeterminate lesions. Radiological appearances must also becorrelated to the treatment regimen specific to the patient.Adjuvant chemotherapy may lead to the change of a lesionon serial imaging, adding further weight to a diagnosis ofmalignancy.The common histological appearances of sclerosed hae-mangiomas consist of multiple thin walled vessels withina hypocellular stroma demonstrating varying degrees offibrosis and sclerosis (Figure 1). Macroscopically a haeman-gioma is usually reddish-blue and well demarcated fromsurrounding tissue. However the sclerosed variant is seen asa pale nodule if there is significant fibrosis present. The celltype of origin is mesenchymal and as such the lesions canoccur almost anywhere [4].Whilst the prevalence of haemangioma has been reportedas high as 20% when found incidentally at autopsy [5], thesclerosed variant is considered much rarer. Sclerosis mayoccur at varying degrees to mask the appearance of thehaemangioma. When complete the lesion takes on a muchmore fibrotic appearance exhibiting few of the characteristicsof the lesion from which it originated. In patients whomhave metastatic liver lesions any indeterminate lesion mustFigure 1: Sclerosed haemangioma with feeding vessel.Figure 2: CT Scan showing segment 4b sclerosing haemangioma.be treated with extreme caution to ensure that there is noresidual disease.One of the key questions in the management of patientsin this scenario is whether to biopsy the lesion in question atthe time of the operation or to proceed straight to formalresection. Whilst thorough preoperative investigation willguide the surgeon in their management, this decision canonly be made at the time of operation. We would certainlynot condone preoperative biopsy of any liver lesions inthe context of planning definitive treatment for suspectedcolorectal hepatic metastases. However, it is conceivablethat if excision of an indeterminate lesion may significantlyincrease the length or risk of surgery (especially in high-risk patients) then an intraoperative biopsy with frozenhistology could be considered. Patients should always beinformed of the potential for benign pathology when surgeryis recommended for indeterminate lesions. Hepatic resectionfor benign conditions is only indicated where there arepersistent symptoms, uncertainty regarding the diagnosisor the risk of malignant transformation [6]. However asystematic review failed to demonstrate any significantHPB Surgery 3evidence to support the elective resection of benign livertumours, although the number of patients was limited andno randomised control trial data is available [7].Cross-sectional imaging, whilst continually improvingin detail, can still not always diﬀerentiate between certainbenign lesions and malignant disease. This is particularlytrue for small lesions less than 5mm. In the context ofmultiple mixed lesions tumour makers will not help quantifyor localise metastatic deposits. Whilst a PET scan may helpdistinguish between benign and malignant disease thereare still instances where benign pathologies can mimicmalignancy.5. ConclusionIn both cases the clinical index of suspicion for metastaticspread was high given the advanced nature of the colorectalcancers. Despite accurate preoperative investigation it maynot always be possible to delineate between a benign lesionsuch as a sclerosing haemangioma and a true metastaticdeposit, especially when the two coexist. The typical imagingfeatures of sclerosed haemangioma have recently been pub-lished as discussed earlier. However no imaging techniquecan be 100% accurate. Tumour markers will not diﬀerentiatebetween the two pathologies in this scenario whilst bothfalse positives and negatives can occur with PET scans [8].If a lesion is not readily resectable, an intraoperative biopsycan provide a tissue diagnosis to negate the risk of a majorresection. When there is persistent doubt regarding thediagnosis it would seem prudent to plan for resection of thelesion with the patient prepared for the eventuality of benignpathology. Given the relatively common occurrence ofhaemangiomas combined with a high incidence of colorectalcancer, cases such as these may become a more commonevent.References[1] J. P. Arnoletti and J. Brodsky, “Surgical treatment of benignhepatic mass lesions,” The American Surgeon, vol. 65, no. 5, pp.413–433, 1999.[2] H. R. Makhlouf and K. G. Ishak, “Sclerosed hemangioma andsclerosing cavernous hemangioma of the liver: a compara-tive clinicopathologic and immunohistochemical study withemphasis on the role of mast cells in their histogenesis,” Liver,vol. 22, no. 1, pp. 70–78, 2002.[3] D. J. Doyle, K. Khalili, M. Guindi, et al., “Imaging featuresof sclerosed hemangioma,” American Journal of Roentgenology,vol. 189, pp. 67–72, 2007.[4] F. C. Nichols, J. A. van Heerden, and L. H. Weiland, “Benignliver tumours,” Surgical Clinics of North America, vol. 69, pp.297–314, 1989.[5] P. J. Karhunen, “Benign hepatic tumours and tumour likeconditions in men,” Journal of Clinical Pathology, vol. 39, no.2, pp. 183–188, 1986.[6] D. Clarke, E. Currie, K. Madhavan, R. Parks, and O. Garden,“Hepatic resection for benign non-cystic liver lesions,” HolidayProperty Bond, vol. 6, no. 2, pp. 115–119, 2004.[7] A. Colli, M. Fraquelli, S. Massironi, A. Colucci, S. Paggi, andD. Conte, “Elective surgery for benign liver tumours,” CochraneDatabase of Systematic Reviews, vol. 24, no. 1, Article IDCD005164, 2007.[8] J. M. Chang, H. J. Lee, J. M. Goo, et al., “False positive and falsenegative FDG-PET scans in various thoracic diseases,” KoreanJournal of Radiology, vol. 7, no. 1, pp. 57–69, 2006.

Sclerosing haemangiomas of the liver: Two cases of mistaken identity

Benign hepatic tumours and tumour like conditions in men,”

Benign liver tumours,”

C h a n g

Elective surgery for benign liver tumours,”

Hepatic resection for benign non-cystic liver lesions,” Holiday Property Bond,

Imaging features of sclerosed hemangioma,”

Sclerosed hemangioma and sclerosing cavernous hemangioma of the liver: a comparative clinicopathologic and immunohistochemical study with emphasis on the role of mast cells in their histogenesis,”

Surgical treatment of benign hepatic mass lesions,”

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Sclerosing Haemangiomas of the Liver: Two Cases of Mistaken Identity

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