BACKGROUND



To report a case of metastatic hepatocholangiocarcinoma to the vitreous and retina.



CASE PRESENTATION



A 70-year-old male, who was recently diagnosed with hepatocholangiocarcinoma, was complaining of floaters in his right eye over the past 5 months and was referred to the Liverpool Ocular Oncology Centre. On presentation, his visual acuity in the right eye was 6/24. Fundus exam revealed a whitish, unilateral, full-thickness retinal lesion at the inferotemporal arcade of his right eye, with vitreous infiltration and subretinal fluid. The patient underwent 25G pars plana vitrectomy with biopsy, resection of the lesion and intravitreal bevacizumab injection. Histopathology testing of the surgical specimens confirmed the diagnosis of metastatic carcinoma to the eye. Two months postoperatively his visual acuity had improved to 6/7.5 and there was no sign of active disease in his right eye, while 9 months postoperatively his visual acuity decreased to 6/9.5 due to developing nuclear sclerotic cataract in his right eye.



CONCLUSION



The current report presents the first case of a hepatocholangiocarcinoma metastasis to the vitreous and retina

Coupland, Sarah E

Groenewald, Carl

Heimann, Heinrich

Irion, Luciane

Jacob, Sarita

Praidou, Anna

Sivaraj, Ramesh R

BMC Cancer

Abstract Background To report a case of metastatic hepatocholangiocarcinoma to the vitreous and retina. Case presentation A 70-year-old male, who was recently diagnosed with hepatocholangiocarcinoma, was complaining of floaters in his right eye over the past 5 months and was referred to the Liverpool Ocular Oncology Centre. On presentation, his visual acuity in the right eye was 6/24. Fundus exam revealed a whitish, unilateral, full-thickness retinal lesion at the inferotemporal arcade of his right eye, with vitreous infiltration and subretinal fluid. The patient underwent 25G pars plana vitrectomy with biopsy, resection of the lesion and intravitreal bevacizumab injection. Histopathology testing of the surgical specimens confirmed the diagnosis of metastatic carcinoma to the eye. Two months postoperatively his visual acuity had improved to 6/7.5 and there was no sign of active disease in his right eye, while 9 months postoperatively his visual acuity decreased to 6/9.5 due to developing nuclear sclerotic cataract in his right eye. Conclusion The current report presents the first case of a hepatocholangiocarcinoma metastasis to the vitreous and retina

Anna Praidou

Sarita Jacob

Luciane Irion

Ramesh Sivaraj

Carl Groenewald

Sarah E. Coupland

Heinrich Heimann

Directory of Open Access Journals

Retinal and vitreous metastases from hepatocholangiocarcinoma

Crossref

Heart of England: HEFT Repository

Retinal and vitreous metastases from hepatocholangiocarcinoma.

HEFT Repository

BackgroundTo report a case of metastatic hepatocholangiocarcinoma to the vitreous and retina.Case presentationA 70-year-old male, who was recently diagnosed with hepatocholangiocarcinoma, was complaining of floaters in his right eye over the past 5 months and was referred to the Liverpool Ocular Oncology Centre. On presentation, his visual acuity in the right eye was 6/24. Fundus exam revealed a whitish, unilateral, full-thickness retinal lesion at the inferotemporal arcade of his right eye, with vitreous infiltration and subretinal fluid. The patient underwent 25G pars plana vitrectomy with biopsy, resection of the lesion and intravitreal bevacizumab injection. Histopathology testing of the surgical specimens confirmed the diagnosis of metastatic carcinoma to the eye. Two months postoperatively his visual acuity had improved to 6/7.5 and there was no sign of active disease in his right eye, while 9 months postoperatively his visual acuity decreased to 6/9.5 due to developing nuclear sclerotic cataract in his right eye.ConclusionThe current report presents the first case of a hepatocholangiocarcinoma metastasis to the vitreous and retina

Sivaraj, Ramesh

University of Liverpool Repository

CASE REPORT Open AccessRetinal and vitreous metastases fromhepatocholangiocarcinomaAnna Praidou1* , Sarita Jacob2, Luciane Irion3, Ramesh Sivaraj2, Carl Groenewald1, Sarah E. Coupland3and Heinrich Heimann1AbstractBackground: To report a case of metastatic hepatocholangiocarcinoma to the vitreous and retina.Case presentation: A 70-year-old male, who was recently diagnosed with hepatocholangiocarcinoma, wascomplaining of floaters in his right eye over the past 5 months and was referred to the Liverpool Ocular OncologyCentre. On presentation, his visual acuity in the right eye was 6/24. Fundus exam revealed a whitish, unilateral, full-thickness retinal lesion at the inferotemporal arcade of his right eye, with vitreous infiltration and subretinal fluid.The patient underwent 25G pars plana vitrectomy with biopsy, resection of the lesion and intravitreal bevacizumabinjection. Histopathology testing of the surgical specimens confirmed the diagnosis of metastatic carcinoma to theeye. Two months postoperatively his visual acuity had improved to 6/7.5 and there was no sign of active disease inhis right eye, while 9 months postoperatively his visual acuity decreased to 6/9.5 due to developing nuclearsclerotic cataract in his right eye.Conclusion: The current report presents the first case of a hepatocholangiocarcinoma metastasis to the vitreous and retina.Keywords: Metastasis, Hepatocholangiocarcinoma, Vitreous, Retina, Eye, MetastasesBackgroundHepatocellularcarcinoma (HCC) and cholangiocellularcar-cinoma (CCC) within the same liver, is designated as hepa-tocholangiocarcinoma (HCC/CCC) [1]. Uveal metastasis isthe most common intraocular malignancy, [2] while retinalmetastasis is rare [3]. Retinal metastasis from HCC/CCCwas not previously reported in the literature. We presentthe first case of metastatic spread of HCC/CCC to theretina and vitreous.Case presentationA 70-year-old male was referred to Liverpool OcularOncology Centre (LOOC), complaining of floaters in hisright eye (RE) over the past 5 months. One month beforepresentation to LOOC, the patient presented to OncologyDepartment with abdominal pain, lack of appetite andweight loss. The patient was diagnosed by MedicalOncologists with HCC/CCC with multiple liver metasta-ses. The diagnosis of HCC/CCC was made via endoscopicultrasound (EUS) and fine-needle aspiration (FNA) with19G needle of the primary tumour which sent for hist-ology and confirmed the diagnosis. Computed tomog-raphy (CT) scan confirmed the presence of livermetastases. The staging of the tumour was T3N1M1 and/or stage IV B (liver metastases).On presentation to LOOC, best corrected visual acuity(BCVA) was 6/24 in RE and 6/4.8 in left eye. Fundusexamination of his RE showed a single whitish elevatedlesion 3–4 DD in size at the infero-temporal arcade asso-ciated with vitreous infiltration and intra- and sub-retinalfluid. (Fig. 1a-c) The patient underwent a 25G pars planavitrectomy with excisional biopsy of the affected tissue, re-section of the lesion, peeling of the epiretinal membrane,air tamponade and intravitreal bevacizumab (anti-VEGF)injection. Four surgical specimens were sent for histopath-ology test which confirmed the diagnosis of metastaticcarcinoma to the vitreous and retina (Fig. 2). The patientwas started on cycles of palliative chemotherapy intraven-ously with gemcitabine 1000 mg/m2 on days 1 and 8 alongwith cisplatin 70 mg/m2 on day 1, after intraocularmetastatic disease was diagnosed.* Correspondence: praidou2003@yahoo.co.uk1Ocular Oncology Service, St Paul’s Eye Unit, Royal Liverpool UniversityHospital, L7 8XP, Liverpool, UKFull list of author information is available at the end of the article© The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.Praidou et al. BMC Cancer  (2017) 17:430 DOI 10.1186/s12885-017-3429-8One month postoperatively, his BCVA has improved to 6/9.5 in his RE and fundoscopy revealed peripheral retinalhaemorrhage and subretinal haemorrhage near the resolvingmetastatic lesion. Two months postoperatively the BCVAhad improved to 6/7.5 and there was no sign of active diseasein his RE (Fig. 1d-f). Four months postoperatively fundos-copy showed telengiectatic vessels on thickened retina withno vitreous infiltration. Six months postoperatively therewere pigmentary changes at the site of metastatic lesion, thevitreous was clear and there were no new lesions. Ninemonths postoperatively his BCVA decreased to 6/9.5 due todeveloping nuclear sclerotic cataract in his RE, while the vit-reous was clear, with no reactivation and no new ophthalmiclesions. Metastatic intraocular lesion had stable regressionafter treatment with vitrectomy, excisional biopsy and sys-temic chemotherapy for the duration of ocular follow-up.The patient underwent in total 8 cycles of palliativechemotherapy with gemcitabine along with cisplatin ap-proximately every 4 weeks but unfortunately succumbed towidespread metastatic disease 5 weeks after completion oflast chemo cycle and one year after the initial diagnosis ofthe primary disease.DiscussionHCC/CCC has emerged as a distinct subtype; it representsless than 1% of primary liver cancers [4]. There are no clearlydefined diagnostic criteria, and no guidelines regarding ther-apy [5]. Known risk factors include cholelithiasis, cirrhosisand viral hepatitis [4, 5]. HCC/CCC metastasizes through thevenous or lymphatic system or along the biliary lumen with apredilection for the liver [4]. Its distant metastases are rare;however, bony metastases were noted in one patient [5].There is only one case in the literature of a solitary lacrimalgland tumor from metastatic HCC [6].Metastatic lesions are the most common intraocular ma-lignancies. The majority of intraocular metastases affect thechoroid, while the ciliary body, the iris, the retina and thevitreous are less frequently affected. The current caseillustrates the first case of a rare site of metastasis to the vit-reous and retina for HCC/CCC. Breast and lung cancermost commonly metastasises to the uvea [2, 3]. Patientswith ocular metastasis most often present with blurring vi-sion, metamorphopsia, floaters, photopsia, and a visual fielddefect [2]. Our patient was complaining of floaters in hisRE over the past 5 months. Retinal metastases are usuallyunilateral, appear white, yellow, or brown; are located inthe inner retina or full-thickness retina; and have vitreousinfiltrates, vitreous hemorrhage, retinal hemorrhage, subret-inal fluid, intraretinal exudation [3] and are initially oftenmisdiagnosed as retinitis, hemangioma, choroidal neovas-cular membrane, or nerve fiber layer infarction [3]. In thepresent case it was a unilateral white, full-thickness retinallesion with vitreous infiltration and subretinal fluid, whichresembled the clinical picture of a localized retinitis.As the patient presented with a recently diagnosedsystemic malignancy and some primary tumours tend toa b c d e f Fig. 1 a Colour wide angle fundus picture of the right eye of the patient at presentation showed a single whitish elevated lesion at the infero-temporalarcade associated with vitreous infiltration and retinal fluid, b Spectral Domain OCT of the right macula of the patient at presentation showing intraretinaland subretinal fluid, c Spectral Domain OCT through the lesion at presentation showing full thickness retinal lesion with vitreous involvement d Colourwide angle fundus picture of the right eye of the patient two months postoperatively showed clear vitreous, no new retinal lesion and no signs of anyactive eye disease, e Spectral Domain OCT of the right macula of the patient two months postoperatively showing complete regression of the retinal fluid,f Spectral Domain OCT through the lesion two months postoperatively showing total anatomical restoration of the retinal layersPraidou et al. BMC Cancer  (2017) 17:430 Page 2 of 3metastasize to the retina and vitreous, the suspecteddiagnosis was that of retinal and vitreous metastatic lesion.The differential diagnosis included fungal endophthalmitisan infectious retinitis of unknown origin. The biopsy wasperformed to establish the suspected diagnosis of an intra-ocular metastasis against the background of the unusuallocation of the lesion [2, 3] as the treatment for these en-tities varies significantly and severe and permanent visualloss was to be expected in case of progressive, untreateddisease. Our patient was diagnosed with a metastaticHCC/CCC to the vitreous and retina which was confirmedby biopsy. Treatment of ocular metastasis includes radio-therapy, resection with/out intravitreal anti-VEGF injec-tion, enucleation, observation along with/or systemicchemotherapy or biologic therapy [2, 3].ConclusionIn conclusion, the current report presents the first case ofunusual HCC/CCC metastasis to the vitreous and retina. Aretinal and vitreous biopsy was required to establish thediagnosis, while the therapeutic options for this caseincluded local treatment for the intraocular metastasis andsystemic chemotherapy for the primary lesion and itsmetastatic disease.AbbreviationsBCVA: Best corrected visual acuity; CT: Computed tomography; EUS: Endoscopicultrasound; FNA: Fine-needle aspiration; HCC/CCC: Hepatocholangiocarcinoma;LOOC: Liverpool Ocular Oncology Centre; RE: Right eyeAcknowledgementsThe authors thank Dr. Medy Tsalic, Consultant Medical Oncologist atBirmingham Heartlands Hospital, Birmingham, UK for the specialist opinionand important contribution on the oncological information for this rare case.FundingThere was no funding for this paper.Availability of data and materialsPatient’s data are confidential patient information which are kept at the sites ofcare as part of the official medical record and are available on reasonable request.Authors’ contributionsAP was involved in acquisition of data; reviewing of the literature; analysis andinterpretation of data; drafting the manuscript; critical revision of themanuscript for important intellectual content. SJ was involved in acquisition ofdata; analysis of data; critical revision of the manuscript for importantintellectual content. LI was involved in acquisition, analysis and interpretation ofdata; critical revision of the manuscript for important intellectual content. RSwas involved in acquisition of data; interpretation of data; critical revision of themanuscript for important intellectual content. CG was involved in acquisition ofdata; analysis of data; critical revision of the manuscript for importantintellectual content. SC was involved in analysis and interpretation of data;critical revision of the manuscript for important intellectual content. HH wasinvolved in acquisition of data; analysis and interpretation of data; drafting themanuscript; critical revision of the manuscript for important intellectual content.All authors have read and approved the final version of this manuscriptCompeting interestsThe authors declare that they have no competing interests.Consent for publicationWritten consent for publication was obtained from the patient described.Ethics approval and consent to participateNot applicable as this is not a study.Author details1Ocular Oncology Service, St Paul’s Eye Unit, Royal Liverpool UniversityHospital, L7 8XP, Liverpool, UK. 2Department of Ophthalmology, Heart ofEngland NHS Foundation Trust, Birmingham, UK. 3Department of Molecularand Clinical Cancer Medicine, Institute of Translational Medicine, University ofLiverpool, Liverpool, UK.Received: 19 January 2017 Accepted: 13 June 2017References1. Piscaglia AC, Shupe TD, Pani G, Tesori V, Gasbarrini A, Petersen BE.Establishment of cancer cell lines from rat hepatocholangiocarcinoma andassessment of the role of granulocyte-colony stimulating factor andhepatocyte growth factor in their growth, motility and survival. J Hepatol.2009;51(1):77–92.2. Konstantinidis L, Rospond-Kubiak I, Zeolite I, Heimann H, Groenewald C,Coupland SE, et al. Management of patients with uveal metastases at theLiverpool ocular Oncology Centre. Br J Ophthalmol. 2014;98:92–8.3. Shields CL, McMahon JF, Atalay HT, Hasanreisoglu M, Shields JA. Retinal metastasisfrom systemic cancer in 8 cases. JAMA Ophthalmol. 2014;132(11):1303–8.4. Razumilava N, Gores GJ. Cholangiocarcinoma. Lancet. 2014;383(9935):2168–79.5. Bhagat V, Javle M, Yu J, Agrawal A, Gibbs JF, Kuvshinoff B, et al. Combinedhepatocholangiocarcinoma: case-series and review of literature. Int JGastrointest Cancer. 2006;37(1):27–34.6. Chen H, Li J, Wang L, Cong N, Shi C, Song J, et al. Hepatocellular carcinomametastasis to the lacrimal gland: a case report. Oncol Lett. 2014;8:911–3.CEA AE1/AE3 b a MGG MGG Fig. 2 Histopathology examination of retinal and vitreous specimensestablished the diagnosis of metastatic carcinoma; a Cytologypreparation stained with May-Grunwald Giemsa stain (MGG) showinggroups of atypical epithelioid cells, some of which with cytoplasmicvacuolation; b Immunocytochemistry demonstrating cytoplasmic andmembranous expression of tumour cells by pan-epithelial marker AE1/AE3 and carcinoembryonic antigen (CEA)Praidou et al. BMC Cancer  (2017) 17:430 Page 3 of 3

Retinal and vitreous metastases from hepatocholangiocarcinoma.

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