Clinico-pathological factors associated with radioiodine refractory differentiated thyroid carcinoma status.

PURPOSE: Risk factors for developing radioiodine refractory thyroid cancer (RAIR-TC) have rarely been analyzed. The purpose of the present study was to find clinical and pathological features associated with the occurrence of RAIR-disease in differentiated thyroid cancers (DTC) and to establish an effective predictive risk score. METHODS: All cases of RAIR-DTC treated in our center from 1990 to 2020 were retrospectively reviewed. Each case was matched randomly with at least four RAI-avid DTC control patients based on histological and clinical criteria. Conditional logistic regression was used to examine the association between RAIR-disease and variables with univariate and multivariate analyses. A risk score was then developed from the multivariate conditional logistic regression model to predict the risk of refractory disease occurrence. The optimal cut-off value for predicting the occurrence of RAIR-TC was assessed by receiver operating characteristic (ROC) curves and Youden's statistic. RESULTS: We analyzed 159 RAIR-TC cases for a total of 759 controls and found 7 independent risk factors for predicting RAIR-TC occurrence: age at diagnosis ≥ 55, vascular invasion, synchronous cervical, pulmonary and bone metastases at initial work-up, cervical and pulmonary recurrence during follow-up. The predictive score of RAIR-disease showed a high discrimination power with a cut-off value of 8.9 out of 10 providing 86% sensitivity and 92% specificity with an area under the curve (AUC) of 0.95. CONCLUSION: Predicting the occurrence of RAIR-disease in DTC patients may allow clinicians to focus on systemic redifferentiating strategies and/or local treatments for metastatic lesions rather than pursuing with ineffective RAI-therapies.

Treatment of Obesity with Thyroid hormones in Europe. Data from the THESIS* Collaboration.

PURPOSE: The use of thyroid hormones (TH) to treat obesity is unsupported by evidence as reflected in international guidelines. We explored views about this practice, and associations with respondent characteristics among European thyroid specialists. METHODS: Specialists from 28 countries were invited to a survey via professional organisations. The relevant question was whether "Thyroid hormones may be indicated in biochemically euthyroid patients with obesity resistant to lifestyle interventions". RESULTS: Of 17,232 invitations 5695 responses were received (33% valid response rate; 65% women; 90% endocrinologists). Of these, 290 (5.1%) stated that TH may be indicated as treatment for obesity in euthyroid patients. This view was commoner among non-endocrinologists (8.7% vs. 4.7%, p < 0.01), private practice (6.5% vs. 4.5%, p < 0.01), and varied geographically (Eastern Europe, 7.3%; Southern Europe, 4.8%; Western Europe, 2.7%; and Northern Europe, 2.5%). Respondents from Northern and Western Europe were less likely to use TH than those from Eastern Europe (p < 0.01). Gross national income (GNI) correlated inversely with this view (OR 0.97, CI: 0.96-0.97; p < 0.001). Having national guidelines on hypothyroidism correlated negatively with treating obesity with TH (OR 0.71, CI: 0.55-0.91). CONCLUSIONS: Despite the lack of evidence, and contrary to guidelines' recommendations, about 5% of respondents stated that TH may be indicated as a treatment for obesity in euthyroid patients resistant to life-style interventions. This opinion was associated with (i) respondent characteristics: being non-endocrinologist, working in private practice, treating a small number of hypothyroid patients annually and (ii) national characteristics: prevalence of obesity, Eastern Europe, low GNI and lack of national hypothyroidism guidelines.

Anti-HBV DNA vaccination does not prevent relapse after discontinuation of analogues in the treatment of chronic hepatitis B: a randomised trial--ANRS HB02 VAC-ADN.

OBJECTIVE: The antiviral efficacy of nucleos(t)ide analogues whose main limitation is relapse after discontinuation requires long-term therapy. To overcome the risk of relapse and virological breakthrough during long-term therapy, we performed a phase I/II, open, prospective, multicentre trial using a HBV envelope-expressing DNA vaccine. DESIGN: 70 patients treated effectively with nucleos(t)ide analogues for a median of 3 years (HBV DNA <12 IU/mL for at least 12 months) were randomised into two groups: one received five intramuscular injections of vaccine (weeks 0, 8, 16, 40 and 44) and one did not receive the vaccine. Analogues were stopped after an additional 48 weeks of treatment in patients who maintained HBV DNA <12 IU/mL with no clinical progression and monthly HBV DNA for 6 months. The primary endpoint was defined as viral reactivation at week 72 (HBV DNA >120 IU/mL) or impossibility of stopping treatment at week 48. RESULTS: Reactivation occurred in 97% of each group after a median 28 days without liver failure but with an HBV DNA <2000 IU/mL in 33%; 99% of adverse reactions were mild to moderate. Immune responses were evaluated by enzyme-linked immunosorbent spot and proliferation assays: there was no difference in the percentage of patients with interferon-γ secreting cells and a specific T-cell proliferation to HBcAg but not to HBsAg after reactivation in each group. CONCLUSIONS: Although it is fairly well tolerated, the HBV DNA vaccine does not decrease the risk of relapse in HBV-treated patients or the rate of virological breakthrough, and does not restore the anti-HBV immune response despite effective viral suppression by analogues. TRIAL REGISTRATION NUMBER: NCT00536627.

Cerebro-retinal microangiopathy with calcifications and cysts due to recessive mutations in the CTC1 gene.

Cerebro-retinal microangiopathy with calcifications and cysts (CRMCC) or Coats plus syndrome is a pleiotropic disorder affecting the eyes, brain, bone and gastrointestinal tract. Its primary pathogenesis involves small vessel obliterative microangiopathy. Recently, autosomal recessively inherited mutations in CTC1 have been reported in CRMCC patients. We herein report an adolescent referred to our hospital following new seizures in a context of an undefined multisystem disorder. Cerebral imaging disclosed asymmetrical leukopathy, intracranial calcifications and cysts. In addition, he presented other typical CRMCC features i.e. a history of intrauterine growth retardation, skeletal demineralization and osteopenia, bilateral exudative vitreo-retinopathy reminiscent of Coats disease, recurrent gastrointestinal hemorrhages secondary to watermelon stomach and variceal bleeding of the esophagus due to idiopathic portal hypertension and telangiectatic and angiodysplasic changes in the small intestine and colon, and anemia due to recurrent bleeding and bone marrow abnormalities. The patient was diagnosed with Coats plus syndrome. CTC1 gene screening confirmed the diagnosis with the identification of heterozygous deleterious mutations. CRMCC due to CTC1 mutations has a broad clinical expressivity. Our case report illustrates the main possible associated phenotypes and their complications, demonstrating the need for a careful etiological search in order to initiate appropriate therapeutic and preventive measures.

Normal Parathyroid Glands Are Most Often Seen on Neck Ultrasound.

RATIONALE AND OBJECTIVES: The dogma is that normal parathyroid glands (PTGs) are not visible on ultrasound (US). Recently, several studies have shown that PTGs present these US features: ovoid structure, homogeneous and hyperechoic. The primary objective was to assess the detection rate, standard size and locations of normal PTGs in a population of patients consulting for thyroid US exam. The secondary objective was to determine if the presence of a goiter or a thyroiditis could modify the visualization of normal PTGs. METHOD: Single-center prospective study on 192 patients based on the typical US appearance previously described to identify one or more PTGs. RESULTS: One or more PTGs were visualized in 75% of patients (144/192). They were visualized preferentially at the lower pole of the thyroid gland and in the infra-thyroid region (66%). The mean (± SD) size of normal PTGs was 5.68 mm (± 1,42 mm)×4.05 mm (± 1,03 mm)×2,68 mm (± 0,61 mm) and mean volume was 33.3 mm3 (± 17.75 mm3). The presence of a goiter made the search for PTGs more difficult whereas the presence of thyroiditis facilitated it. CONCLUSION: The US detection rate of PTGs is high (75%). The identification of PTGs could be particularly useful in the preoperative assessment before total thyroidectomy or parathyroid surgery. It could reduce the risk of postoperative hypoparathyroidism and improve the accuracy of postoperative US surveillance of thyroid cancer. Better knowledge of the usual anatomical location of normal PTGs could also enable better detection of abnormal glands.

[Oncological patients' decision making processes concerning their pain medication at home: a qualitative secondary analysis]. / Entscheidungswege von onkologischen Patienten im Umgang mit Schmerzmedikamenten im häuslichen Umfeld: eine qualitative Sekundäranalyse.

Unrelieved pain affects up to 75 % of cancer patients. Possible reasons for the undertreatment of pain are, amongst others, patient-related barriers towards cancer pain management. However the way patients decide on the use of analgesics remains unclear. The purpose of this qualitative study was to explore decision-making processes of four women and four men with diverse cancers concerning their pain medications. Audiotaped protocols of the 10-week-intervention and interviews of the PEINCA-pilot study provided data for a secondary analysis. This pilot study was conducted at a comprehensive cancer centre in Germany to test the German version of a cancer pain self-management intervention to enhance oncology patients' pain self-management for the first time. The data of purposively selected patients were analysed using content analysis. The results showed that these patients were very ambivalent about their analgesic use. The need to relieve severe pain conflicted with the desire to avoid opioids at any price. Decisions were reconsidered and overturned even after good experiences with analgesics. This study seems to provide a first look into decision-making processes over 10 weeks during a self-management education. Individually tailored counselling by a professional within the education programme helped the patients adopt new attitudes towards analgesics and gradually reduce their pain levels. Previous experiences of the patients and their possible ambivalence towards analgesics should be considered in a pain therapy, and patients should be coached by professionals.

SFE-AFCE-SFMN 2022 consensus on the management of thyroid nodules: Synthesis and algorithms.

The SFE-AFCE-SFMN 2022 consensus deals with the management of thyroid nodules, a condition that is a frequent reason for consultation in endocrinology. In more than 90% of cases, patients are euthyroid with benign and non-progressive nodules that do not warrant specific treatment. The clinician's objective is to detect malignant thyroid nodules at risk of recurrence and death, toxic nodules responsible for hyperthyroidism or compressive nodules warranting treatment. The diagnosis and treatment of thyroid nodules requires close collaboration between endocrinologists, nuclear medicine physicians and surgeons but also involves other specialists. Therefore, this consensus statement was established jointly by 3 societies, the French Society of Endocrinology (SFE), the French Association of Endocrine Surgery (AFCE) and the French Society of Nuclear Medicine (SFMN); the various working groups included experts from other specialties (pathologists, radiologists, pediatricians, biologists, etc.). This specific text is a summary chapter taking up the recommendations from specific sections and presenting algorithms for the exploration and management of thyroid nodules.

Outcomes of active surveillance of EU-TIRADS 5 thyroid nodules.

OBJECTIVE: Active surveillance of cytologically proven microcarcinomas has been shown as a safe procedure. However, fine needle aspiration biopsy (FNAB) is not recommended by European Thyroid Association (ETA) and American Thyroid Association (ATA) guidelines for highly suspicious nodules ≤ 10 mm. The aim of the study was to assess the outcomes of active surveillance of EU-TIRADS 5 nodules ≤ 10 mm not initially submitted to FNAB. PATIENTS AND METHODS: 80 patients with at least one EU-TIRADS 5 nodule ≤ 10 mm and no suspicious lymph nodes, accepting active surveillance, were included. RESULTS: Mean baseline diameter and volume were 5.4 mm (±2.0) and 64.4 mm3 (±33.5), respectively. After a median follow-up of 36.1 months, a volumetric increase ≥ 50% occurred in 28 patients (35.0%) and a suspicious lymph node in 3 patients (3.8%). Twenty-four patients underwent an FNAB (30.0%) after at least a 1 year follow-up of which 45.8% were malignant, 8.3% benign, 33.3% undetermined and 8.3% nondiagnostic. Sixteen patients (20.0%) underwent conversion surgery after a median follow-up of 57.2 months, confirming the diagnosis of papillary carcinoma in 15/16 cases (not described in 1 histology report), all in remission at 6-12 months postoperative follow-up. CONCLUSION: Applying ETA and ATA guidelines to avoid FNA of EU-TIRADS 5 sub-centimeter nodules and proceeding to active surveillance of such nodules in selected patients is a safe procedure. Thus, US-FNAB could be postponed until the nodule shows signs of progression or a suspicious lymph node appears, with no added risk for the patient.

Impact of age on survival in radioiodine refractory differentiated thyroid cancer patients.

OBJECTIVE: The objectives of our study were to analyze the influence of age on the survival of patients with RAIR-DTC and to determine their prognostic factors according to age. METHODS: This single-center, retrospective study enrolled 155 patients diagnosed with RAIR-DTC. The primary end point was overall survival (OS) according to different cutoff (45, 55, 65, 75 years). Secondary endpoints were progression free survival (PFS) and prognostic factors in patients under and over 65 years. RESULTS: Median OS after RAIR diagnosis was 8.2 years (95% IC: 5.3-9.6). There was no difference according to age with a 65 (P = 0.47) and 55 years old cutoff (P = 0.28). Median OS improved significantly before 45 years old (P = 0.0043). After 75 years old, median OS significantly decreased (P = 0.0008). Median PFS was 2.1 years (95% CI: 0.8-3) in patients < 65 years old, and 1 year in patients ≥ 65 years old (95% CI: 0.8-1.55) with no statistical difference (P = 0.22). There was no impact of age on PFS with any cutoff. In both groups, progressive disease despite 131I treatment reduced OS. In patients < 65 years old, an interval of less than 3 years between the initial diagnosis and the diagnosis of RAIR metastatic disease was predictive of poor survival. In patients > 65 years old, the presence of a mediastinum metastasis was a significant factor for mortality (HR: 4.55, 95% CI: 2.27-9.09). CONCLUSION: In RAIR-DTC patients, a cut-off age of 65 years old was not a significant predictive factor of survival. Forty-five and 75-years-old cutoff were predictive for OS but not PFS.

[Poorly differentiated large cell endocrine carcinoma of the extrahepatic bile ducts]. / Carcinome endocrine peu différencié à grandes cellules des voies biliaires extrahépatiques.

We report a case of a poorly differentiated endocrine large cell carcinoma of the extrahepatic bile ducts in a 73-year-old man, revealed by abdominal pain, jaundice and weight loss. Computed tomography and endoscopic retrograde cholangiography found tumoral stenosis of the main bile duct. Brush cytology detected tumor cells. Pathological examination of the resected bile duct disclosed a high-grade large cell carcinoma with morphological endocrine features and positivity for chromogranin A. This tumor was associated with a minor component of adenocarcinomatous cells. Despite polychemotherapy, the patient had widely metastatic disease a few months later. We discuss here the histogenesis of this tumor as well as its nosological position among the endocrine and mixed tumors of bile ducts.

Liver cell adenomas and portosystemic shunt.

We report the case of a young man who developed multiple liver cell adenomas 13 years after a mesentericocaval shunt. Radiological findings did not provide diagnosis. Histological findings of two biopsied nodules were compatible with liver cell adenoma. Our patient had no known risk factors for liver cell adenomas. We discuss the hypothesis that disturbed hepatic vascularisation could promote the development of liver cell adenomas.

TIRADS score is of limited clinical value for risk stratification of indeterminate cytological results.

CONTEXT: Thyroid nodules with cytological indeterminate results represent a daily and recurrent issue for patient management. OBJECTIVE: The primary aim of our study was to determine if TIRADS (Thyroid Imaging Reporting and Data System) could be used to stratify the malignancy risk of these nodules and to help in their clinical management. Secondary objective was to estimate if this risk stratification would change after reclassification of encapsulated non-invasive follicular variant of papillary carcinomas (FVPTC) as non-invasive follicular thyroid neoplasm (NIFTP). PATIENTS AND METHODS: Single-center retrospective study of a cohort of 602 patients who were referred for ultrasound-guided fine-needle aspiration from January 2010 to December 2016 with an indeterminate cytological result and in whom histological results after surgery were available. TIRADS score was prospectively determined for all patients included. Nodules that had been classified as FVPTC were submitted to a rereading of histological report and reclassified as NIFTP when judged relevant. A table of malignancy risk crossing Bethesda and TIRADS results was built before and after this reclassification. RESULTS: The study included 602 cytologically indeterminate nodules. TIRADS score was positively correlated with the malignancy rate (P < 0.0001). Risk stratification with TIRADS was significant only in Bethesda V nodules (P = 0.0004). However, the risk of malignancy in this Bethesda V category was always above 45%, whatever the TIRADS score. CONCLUSION: For a clinician facing an indeterminate cytological result for a thyroid nodule, return to TIRADS score is of limited value in most conditions to rule in or rule out malignancy and to guide subsequent management of patients.

Molecular perspectives in differentiated thyroid cancer.

Progress in understanding the molecular genetics of thyroid cancer in the last 20 years has accelerated recently with the advent of high-throughput sequencing technologies known as Next-Generation Sequencing. Besides classical molecular abnormalities involving the MAPK (Mitogen Activated Protein Kinase) and PI3K (PhosphoInositide 3-Kinase) pathways that play a key role in follicular-derived thyroid tumorigenesis, new molecular abnormalities have been discovered. The major advances in recent years have been the discovery of new somatic driver gene point mutations (such as RASAL1 [RAS protein activator Like 1] mutations in follicular cancer) and/or mutations that have prognostic value (such as TERT [Telomerase reverse transcriptase] promoter mutations); new chromosomal rearrangements, usually having close connection with exposure to ionizing radiation (such as ALK [Anaplastic Lymphoma Kinase] rearrangements); and deregulation of some gene or microRNA expression representing a molecular signature. Progress made in understanding the molecular mechanisms of thyroid cancer offers new perspectives for the diagnosis of the benign or malignant status of a thyroid nodule, to refine prognosis and offer new perspectives of targeted therapy for radioiodine-refractory cancers.

Indications for radioiodine administration in follicular-derived thyroid cancer.

Indications for radioiodine administration after thyroid cancer surgery have shifted in recent years toward personalized management, adapted to the individual risk of tumor progression. The most recent guidelines and studies favor de-escalation in indications for administration, dosage and means of preparation with exogenous recombinant TSH stimulation as treatment of choice. Radioiodine administration has 3 possible objectives: • ablation of normal thyroid tissue remnants in patients with low risk of progression, using low radioiodine activity levels, with the advantage of completing disease staging on whole-body scintigraphy performed after administration of the radioiodine capsule, and of facilitating follow-up by thyroglobulin assay; • adjuvant treatment for suspected microscopic metastases in patients with intermediate or high risk of progression, using higher activity levels, with the theoretic aim of limiting recurrence and mortality; • curative treatment in high-risk patients with proven metastases, using exclusively high activity levels, with a view to improving specific survival. In future, indications for ablation and/or activity prescription may be governed by an algorithm incorporating individual baseline progression risk (essentially founded of pTNM staging) and postoperative data such as thyroglobulin level and neck ultrasound results.

Diagnosis and prognosis of AIDS-related cholangitis.

OBJECTIVE: To determine more precisely the clinical and biological characteristics of AIDS-related cholangitis, and to investigate prognostic variables of this disease. DESIGN: Retrospective clinical and prognostic study. SETTING: Biliary unit, Bicêtre Hospital, France. PATIENTS: HIV-positive patients (n = 52) referred to the unit between December 1986 and June 1993 for biliary symptoms leading to the suspicion of AIDS-related cholangitis, (42 men; 10 women; mean age, 37 +/- 8 years). INTERVENTION: Endoscopic retrograde cholangiopancreatography (ERCP) was performed in order to determine the cause of the biliary symptoms. MAIN OUTCOME MEASURE: Clinical features and evolution of the cholangitis. RESULTS: Among the 52 patients, 45 met the ERCP criteria of AIDS-related cholangitis (36 men; nine women). The diagnosis of cholangitis was strongly suggested by abdominal ultrasonography in 47% of the cases. ERCP showed papillary stenosis, diffuse cholangitis, extrahepatic cholangitis alone, and intrahepatic cholangitis alone in 60, 67, 7 and 27%, respectively. Endoscopic sphincterotomy was performed in 28 patients. Pain was relieved by sphincterotomy in nine patients, but the other clinical or biological features were not influenced. One-year and 2-year survival rates were 41 +/- 7% and 8 +/- 4%, respectively. Multidimensional analysis using a Cox model showed that a lymphocyte count > 500 x 10(6)/l was the only independent predictive factor of better survival. CONCLUSION: AIDS-related cholangitis is a disease which leads preferentially to papillary stenosis or diffuse abnormalities of the biliary tract. Prognostic factors depend on the stage of the HIV infection. Another diagnosis of cholestasis was found in approximately 15% of the patients who showed biliary symptoms.

Protein-losing gastropathy associated with cytomegalovirus: a rare and late complication of allogeneic bone marrow transplantation.

A 36-year-old women with chronic myelogenous leukemia in first chronic phase received a bone marrow transplant from her HLA-identical brother. The preparatory regimen consisted of total body irradiation (10 Gy) and cyclophosphamide (60 mg/kg for 2 days). Full engraftment was achieved and the woman was monitored as an outpatient after discharge from hospital on day 35. One year after BMT, while she was on cyclosporin A and steroids because of chronic graft-versus-host disease, the patient developed protein-losing gastropathy associated with cytomegalovirus infection (with no gastrointestinal symptoms), which regressed spontaneously in 4 weeks.

Bile levels of carcino-embryonic antigen in patients with hepatopancreatobiliary disease.

OBJECTIVE: To evaluate the value of biliary carcino-embryonic antigen (CEA) in the differential diagnosis of malignant and benign hepatopancreatobiliary disease. PATIENTS: One hundred patients were prospectively studied. Benign diseases were present in 39% of the patients while 61% had malignant diseases. METHODS: Samples of serum were taken from all patients just before endoscopic retrograde cholangiopancreatography (ERCP) and samples of biliary CEA were obtained during ERCP. RESULTS: The sensitivity of serum CEA and carbohydrate antigen 19-9 (CA 19-9) in detecting malignancy were 50% and 92%, respectively, while the respective specificities were 95% and 72%. The mean biliary CEA level of the benign group was significantly different from that of the malignant group (35.7 +/- 8.7 ng/ml vs 268 +/- 85.5 ng/ml), but there was considerable overlap between the two groups. With a cut-off level of 20 ng/ml, the sensitivity and specificity were 84% and 64% respectively. The mean bilirubinaemia value was significantly higher in malignant disease than in benign disease (57.4 +/- 13.9 mumol/l vs 235 +/- 19.8 mumol/l). Multidimensional analysis indicated that only bilirubinaemia (P < 109-3)) was independently predictive of malignant disease. CONCLUSION: Biliary CEA assessment seems useless in distinguished between benign and malignant causes of cholestasis.

Plasma deproteinization by precipitation and filtration in the 96-well format.

The need for fast bioanalytical methods within the pharmaceutical sector is rapidly growing. Sample preparation is often the bottleneck step. A new approach to increasing sample throughput involves precipitated protein removal by filtration in the 96-well format, thereby eliminating the need for centrifugation and manual handling of individual tubes. The potential for such a new technique has been investigated for the determination of an iron chelator, a highly protein-bound compound (> or =99.5%) in plasma. An analog was used as internal standard. Acetonitrile and plasma were sequentially aspirated, separated by an air gap, using a manual electronic pipettor. They were then dispensed into the channel of an Empore filter PPT plate above the filter, and a slight vacuum was applied. The eluate was collected and diluted prior to injection. The compounds were then separated by reversed-phase chromatography and detected by UV at 295 nm. The chromatographic run time was 6 min. The mean recovery following protein precipitation was 78%, which shows that the technique can apply to a highly protein-bound compound. Replicate quality control samples were prepared in drug-free normal human plasma at four different concentrations. The mean accuracy ranged from 87 to 108% with the CV ranging from 3 to 8%. The described procedure is simple, fast and reproducible. It requires minimal equipment. The time required to prepare a plate manually is only about 20 min. The use of 12-channel repeater pipettors reduces the risk of error and improves productivity. Automation should be an aid to further increasing sample throughput when more than one plate a day is to be prepared.

Celiac artery aneurysm.

Aneurysms of the celiac axis are rare, since only 50 cases were reported in 1976, 14 of which were operated on successfully. We report here the 15th case. This celiac aneurysm involved the origin of the splenic left gastric and common hepatic arteries. After controlling the aorta by a thoraco-phrenolaparotomy, the celiac artery was ligated at its origin, the anterior wall of the aneurysm was incised and the splenic and the left gastric arteries ligated by an obliterative endoaneurysmorrhaphy. The hepatic vascularization was restored from the gastroduodenal artery with a restorative endoaneurysmorrhaphy. The control arteriogram showed a good arterial revascularization.

[Intra-hepatic biliary cystadenocarcinoma]. / Cystadénocarcinome biliaire intra-hépatique.

Biliary cystadenocarcinoma is a rare tumor of the intrahepatic biliary tract, which frequently develops in a preexisting benign biliary cystadenoma. In the present case, diagnosis was difficult because of the lack of specificity of clinical, biological and radiological findings. The correct diagnosis was only achieved by histological examination of the resected lesion. Macroscopically, the right lobe of the liver showed evidence of a whitish, multilolobed, malignant mass of about 6 cm in diameter. Upon light microscopic analysis, cysts were found to be lined with papillary forms. In some areas, epithelial cells were clearly malignant contrasting with persistent non dysplasic areas, suggesting the presence of underlying cystadenomas. Eleven months after complete surgical resection, the patient is in good condition with no evidence of recurrence.