Infantile Hepatic Hemangioma: A Novel Approach Using Propranolol and Transarterial Embolization.

Benign vascular tumors, or hemangiomas, are common in young children. The most frequent way to identify them on the skin is as bright red surface lesions, although they can also be detected deeper as subcutaneous lesions. Visceral involvement, particularly of the liver, is commonly observed in patients with multiple cutaneous hemangiomas. Since most hemangiomas are self-limited, they can be clinically monitored. Despite this, hepatic hemangiomas can result in significant consequences, such as severe hepatomegaly, which can induce abdominal compartment syndrome, inadequate ventilation, and renal vein compression, as well as significant arteriovenous shunts that compromise the functioning of the heart. Depending on the patient's findings, management may range from routine follow-up to liver transplantation. Here, we present a case of hypothyroidism, hepatomegaly, and cardiac failure in a two-month-old female newborn with infantile hepatic hemangioma. The patient's symptoms were managed with the use of levothyroxine, propranolol, and transcatheter arterial embolization (TAE).

A Case Report: Giant Liver Hemangioma Treated with Transcatheter Embolization.

When treating patients with a massive cavernous hemangioma of the liver that requires nonsurgical therapy, transcatheter arterial embolization has proven to be an effective technique. Significant advantages include the ability to obliterate the vascular supply of these lesions and the minimally invasive nature compared to surgery. A 65-year-old woman arrived at our hospital complaining primarily of stomach pain that had been there for six months. The patient had a hard lump in the right hypochondrium on clinical examination. Ultrasound showed a large, well-defined, heterogeneous lesion with central necrotic areas, with the rest of the liver parenchyma having normal echotexture and flow in the portal vein. The 65-year-old woman's primary complaint upon arrival at our hospital was a stomach ache that had been there for six months. The results of the liver function test were normal. Upon presumptive identification of a significant hepatic hemangioma, the patient was brought to the angio-suite for angiography and proper hepatic artery embolization. Considering the patient's age, the severity of the lesion, and its highly vascular character, endovascular embolization of the proper hepatic artery using lipidol and bleomycin was performed. The patient was discharged after two days in the hospital, administered antibiotics, and advised to follow up after 15 days. Liver function after embolization was within normal limits. The patient had no symptoms after a follow-up at three months. Therefore, endovascular embolization with lipidol and bleomycin is a safe and effective method to obliterate the vascular supply to the lesion, prevent catastrophic bleeding, and provide symptomatic relief to the patient.

Three-dimensional visualized technology-guided surgical resection for giant hepatic hemangioma with hemorrhagic necrosis: A case report and literature review.

Background: Hepatic hemangioma is the most common type of benign mesenchymal liver tumor and often has a good prognosis. However, giant hepatic hemangioma larger than 10 cm is an unusual event, and accompanying symptoms of internal hemorrhagic necrosis are extremely rare. There are only a few cases reported. Case summary: Herein, we report the case of a 52-year-old man with hemorrhagic necrosis of a giant hepatic hemangioma. The patient presented to the Department of Hepatobiliary Surgery with a complaint of distending pain on the right abdomen. The patient underwent hepatic artery embolization for giant hepatic hemangioma 2 weeks before presentation. During hospitalization, abdominal computed tomography revealed a mass (15.8 × 14.2 × 14.7 cm) with high density below the right lobe of the liver. The patient subsequently underwent irregular right hepatectomy with the guidance of three-dimensional visualization technology. The surgical anatomy confirmed the diagnosis of internal hemorrhagic necrosis. There was no recurrence or complications in a 4-month follow-up. Previous cases were reviewed to characterize the clinical features of giant hepatic hemangioma with internal hemorrhage necrosis. Conclusion: Cases of giant hepatic hemangioma with internal hemorrhagic necrosis are rare and usually only exhibit fever or epigastric pain. All patients in reviewed cases finally underwent surgical resection. Under these circumstances, the healing effect of transhepatic arterial treatment is not very satisfactory. Patients are deemed poor laparoscopic surgical candidates due to limited abdominal cavity. In order to standardize the diagnosis of these rare cares, the aggregation of existing and future case data is certainly warranted. If diagnosed, consideration should be given to implementing surgical resection according to patients' condition by three-dimensional visualized technology.

A unique intersection: Exploring an exophytic, pedunculated hepatic hemangioma with gastric connection in a case report.

Hepatic hemangiomas are commonly benign liver tumors, typically asymptomatic and predominantly located in the right lobe. This case report details an exceptional instance of a left-lobe hepatic hemangioma manifesting as an exophytic, pedunculated mass resembling a gastric tumor. A 77-year-old woman with a history of melanoma presented with a mass incidentally discovered during evaluations for chest pain. Advanced imaging techniques, including computed tomography (CT) and endoscopic ultrasound (EUS), identified this mass as a benign, pedunculated hemangioma extending from the left hepatic lobe toward the gastric fundus. Given the tumor's benign nature and the patient's lack of symptoms, a conservative management approach was adopted. This case emphasizes the importance of accurate imaging and diagnostic assessment in managing atypical hepatic hemangiomas, highlighting the need to carefully consider rare growth patterns and locations in differential diagnoses to avoid unnecessary interventions. Such cases reinforce the complexity of diagnosing and managing unusual presentations of common benign tumors.

Surgical Resection of a Giant Hepatic Hemangioma in a Hepatitis B-positive Patient: A Case Report.

This case report describes the successful surgical removal of a giant hemangioma in a 41-year-old female with hepatitis B. The patient came with stomach distension, right upper quadrant, and right lumbar region pain. Imaging studies showed a mass measuring 12x7.6x11 cm emerging from the left lobe of the liver, causing compression of surrounding structures. The patient has undergone a laparotomy with successful anatomical resection of the hemangioma. Postoperative recovery was uneventful, and the patient was discharged on the fifth postoperative day. This case highlights the significance of considering surgery for symptomatic giant hemangiomas and normal follow-up to screen for recurrence and aims to present the successful surgical management of a giant hemangioma.

Retrospective analysis based on a clinical grading system for patients with hepatic hemangioma: A single center experience.

BACKGROUND: The optimal approach for managing hepatic hemangioma is controversial. AIM: To evaluate a clinical grading system for management of hepatic hemangioma based on our 17-year of single institution experience. METHODS: A clinical grading system was retrospectively applied to 1171 patients with hepatic hemangioma from January 2002 to December 2018. Patients were classified into four groups based on the clinical grading system and treatment: (1) Observation group with score < 4 (Obs score < 4); (2) Surgical group with score < 4 (Sur score < 4); (3) Observation group with score ≥ 4 (Obs score ≥ 4); and (4) Surgical group with score ≥ 4 (Sur score ≥ 4). The clinico-pathological index and outcomes were evaluated. RESULTS: There were significantly fewer symptomatic patients in surgical groups (Sur score ≥ 4 vs Obs score ≥ 4, P < 0.001; Sur score < 4 vs Obs score < 4, χ² = 8.60, P = 0.004; Sur score ≥ 4 vs Obs score < 4, P < 0.001). The patients in Sur score ≥ 4 had a lower rate of in need for intervention and total patients with adverse event than in Obs score ≥ 4 (P < 0.001; P < 0.001). Nevertheless, there was no significant difference in need for intervention and total patients with adverse event between the Sur score < 4 and Obs score < 4 (P > 0.05; χ² = 1.68, P > 0.05). CONCLUSION: This clinical grading system appeared as a practical tool for hepatic hemangioma. Surgery can be suggested for patients with a score ≥ 4. For those with < 4, follow-up should be proposed.

Clinical impact and role of major vessels involvement in laparoscopic resection for hepatic hemangioma.

BACKGROUND: Severe bleeding remains a significant concern in laparoscopic resection for hepatic hemangioma. It is rarely reported that how the degree of major vessels involvement impacts on severe bleeding. The present study primarily aimed to analyze the impacts of the number of involved major vessels (NIMV) during laparoscopic surgery for hepatic hemangioma and evaluate the risk factors associated with increased bleeding. METHODS: A database search was carried out for consecutive patients who underwent laparoscopic resection for liver hemangiomas at our department from January 2018 to December 2023. The collected data included demographics, characteristics of the hemangiomas, laboratory data, operation method, surgical and postoperative variables. RESULTS: A total of 72 patients were enrolled in the study. 42 patients were categorized into the group with NIMV < 2, while 30 patients were divided into the group with NIMV ≥ 2. The group with NIMV ≥ 2 demonstrated a significant correlation with special segments, involved multiple segments and diameter of the hemangiomas (P < 0.01). And the perioperative variables including the extent of resection, operative time, blood loss, Pringle maneuver times, postoperative stay, drainage tube duration, and postoperative liver function (ALT, AST) also showed significant differences between the two groups (P < 0.05). Notably, NIMV ≥ 2 was identified as the most important independent risk factor for intraoperative blood loss ≥ 500 ml in laparoscopic surgery for hepatic hemangioma (P = 0.011). For NIMV ≥ 2, the independent risk factor was special segments in multivariate analysis (P = 0.000). CONCLUSION: The involvement of multiple major vessels (NIMV ≥ 2) was significantly associated with special segments, resulting in increased intraoperative blood loss, operation difficulty, and delayed postoperative recovery. Moreover, it was identified as the single independent risk factor with a considerable risk for increased blood loss during laparoscopic resection for hepatic hemangioma.

Fetal Hepatic Hemangioma Diagnosed in Utero Using Doppler Microvascular Imaging.

A 35-year-old pregnant woman was referred to our institution at 33 weeks' gestation for evaluation of a fetal abdominal tumor. B-mode ultrasonography demonstrated a massive lesion. Bidirectional power Doppler mode showed abundant blood flow surrounding the tumor. On superb micro-vascular imaging, various Doppler signal patterns were observed within the tumor, including diffuse fine dotted-like signals, linear flow, and internal shunt flow. Sequential observations of the tumor and cardiac cycles also revealed pulsatile flow beneath the edges of the tumor and continuous fine flow in the central area, resembling a 'centripetal fill-in' appearance on contrast computed tomography. Therefore, we assumed the fetal tumor to be a hepatic hemangioma. Fetal heart failure was detected at 37 weeks' gestation, and a 2,484-g female infant was delivered with 1- and 5-min Apgar scores of 7 and 8, respectively. A postnatal contrast computed tomography examination showed a progressive centripetal fill-in appearance, leading to a diagnosis of hepatic hemangioma. Kasabach-Merritt syndrome was also noted. Intensive treatment was performed, and the infant was discharged at 3 months after birth. In summary, we experienced a case of hepatic hemangioma diagnosed in utero using superb micro-vascular imaging. And basing seamless postnatal treatments on prenatal imaging findings may help to reduce the perinatal mortality.

Management of Neonatal Hepatic Hemangiomas: A Single-Center Experience Focused on Challenging Cases.

Background: Management of hepatic hemangioma (HH) in infancy ranges from close monitoring to surgical resection. We analyzed the clinical characteristics and outcomes of HH according to its treatment options, with particular focus on challenging cases. Methods: Data of patients diagnosed with HHs in their first year of life and followed up for at least 1 year were retrospectively reviewed and divided into treatment and observation groups. Serial imaging results, serum alpha-fetoprotein (AFP) levels, medications, and clinical outcomes were compared. The detailed clinical progress in the treatment group was reviewed separately. Results: A total of 87 patients (75 in the observation group and 12 in the treatment group) were included. The median HH size at the initial diagnosis and the maximum size were significantly larger in the treatment group than the observation group (2.2 [0.5-10.3] cm vs. 1.0 [0.4-4.0] cm and 2.1 [0.7-13.2] vs. 1.1 [0.4-4.0], respectively; all p < 0.05]. The median initial and last serum AFP levels were significantly higher in the treatment group than in the observation group (76,818.7 vs. 627.2 and 98.4 vs. 8.7, respectively; all p < 0.05). Serum AFP levels in both groups rapidly declined during the first 3 months of life and were almost undetectable after 6 months. Among the challenging cases, a large (14 × 10 × 6.5 cm sized) focal HH was successfully treated using stepwise medical-to-surgical treatment. Conclusions: Patients with large HH and mild symptoms can be treated using stepwise pharmacotherapy. More aggressive surgical treatment of tumors unresponsive to initial pharmacotherapy may help shorten the treatment period and improve outcomes.

Laparoscopic liver resection or enucleation for giant hepatic hemangioma: how to choose?

BACKGROUND: Laparoscopic treatment has been increasingly adopted for giant hepatic hemangioma (HH), but the role of liver resection or enucleation remains uncertain. The aim of this study is to compare the laparoscopic resection (LR) with laparoscopic enucleation (LE) for HH, and to provide evidence on how to choose the most suitable approach for HH. METHODS: A retrospective analysis of HH patients underwent laparoscopic treatment between March 2015 and August 2022 was performed. Perioperative outcomes were compared based on the surgical approaches, and risk factors for increased blood loss was calculated by logistic regression analysis. RESULTS: A total of 127 patients in LR group and 287 patients in LE group were enrolled in this study. The median blood loss (300 vs. 200 mL, P < 0.001) was higher in LE group than that in LR group. Independent risk factors for blood loss higher than 400 mL were tumor size ≥ 10 cm, tumor adjacent to major vessels, tumor occupying right liver or caudate lobe, and the portal phase enhancement ratio (PER) ≥ 38.9%, respectively. Subgroup analysis showed that LR was associated with less blood loss (155 vs. 400 mL, P < 0.001) than LE procedure in patients with high PER value. Both LR and LE approaches exhibited similar perioperative outcomes in patients with low PER value. CONCLUSIONS: Laparoscopic treatment for HH could be feasibly and safely performed by both LE and LR. For patients with PER higher than 38.9%, the LR approach is recommended.

Spontaneous rupture of neonatal hepatic hemangioma diagnosed by ultrasound: A case report.

Hepatic hemangioma is the most prevalent benign liver tumor during the fetal and neonatal period, and its rupture poses a severe threat to newborns' lives-this article presents a case involving the spontaneous rupture of a hepatic hemangioma in a neonate. Early diagnosis through ultrasound enabled prompt treatment, resulting in the patient's timely discharge.

Hepatic Hemangioma: Review of Imaging and Therapeutic Strategies.

Hepatic hemangiomas are the most common benign liver tumors. Typically, small- to medium-sized hemangiomas are asymptomatic and discovered incidentally through the widespread use of imaging techniques. Giant hemangiomas (>5 cm) have a higher risk of complications. A variety of imaging methods are used for diagnosis. Cavernous hemangioma is the most frequent type, but radiologists must be aware of other varieties. Conservative management is often adequate, but some cases necessitate targeted interventions. Although surgery was traditionally the main treatment, the evolution of minimally invasive procedures now often recommends transarterial chemoembolization as the treatment of choice.

Diffuse infantile hepatic hemangioma successfully treated with propranolol orally: a case report and literature review.

Background: Infantile hepatic hemangioma (IHH) is a common vascular, fast-growing hepatic tumor that is usually accompanied by multiple cutaneous hemangiomas. Diffuse IHH (DIHH) is a rare type of IHH that exhibits many tumors with nearly complete hepatic parenchymal replacement. At present, there is no specific standardized treatment plan for DIHH. Herein, we present the case of a 2-month-old girl with DIHH and without cutaneous hemangioma who achieved complete remission after undergoing propranolol monotherapy. Case presentation: The infant with low birth weight was presented to the pediatric department with a 2-month history of persistent vomiting and feeding difficulty. Ultrasonography and abdominal magnetic resonance imaging revealed hepatomegaly and diffused intrahepatic lesions. A computed tomography-guided percutaneous liver biopsy was performed, and the pathological examination suggested the diagnosis was DIHH. The patient exhibited remarkably response to an increasing dose of oral propranolol, from 0.5 mg/kg to 2 mg/kg every day. The intrahepatic lesions were almost completely regressed after one year of treatment and no distinct adverse reaction was observed. Conclusion: DIHH can induce life-threatening complications that require prompt interventions. Propranolol monotherapy can be an effective and safe first-line treatment strategy for DIHH.

Transarterial Bleomycin-Lipiodol Chemoembolization for the Treatment of Giant Hepatic Hemangiomas: An Assessment of Effectiveness.

This study evaluates the effectiveness of superselective transcatheter arterial chemoembolization (TACE) using a bleomycin-lipiodol emulsion in treating giant hepatic hemangiomas. A retrospective review included 31 patients with a mean age of 53 ± 10.42 years who underwent TACE from December 2014 to October 2022, with follow-up imaging examinations to assess outcomes. Technical success was defined as successful embolization of all feeding arteries, and clinical success was defined as a reduction in hemangioma volume by 50% or more on follow-up imaging. This study observed a 100% technical success rate. Post-embolization syndrome was common, and two cases of asymptomatic hepatic artery dissection were noted. Clinical success was achieved in 80.6% of patients, with significant volume reduction observed in the majority. Conclusively, superselective transcatheter arterial chemoembolization with bleomycin-lipiodol emulsions is presented as a viable and effective treatment option for giant hepatic hemangiomas. With no procedure-related mortality and significant volume reduction in most cases, this method offers a promising alternative to surgical intervention. This study's findings suggest a need for further exploration and validation in larger-scale prospective studies.

Postnatal treatment and evolution patterns of giant fetal hepatic hemangioma: a case series of 29 patients.

OBJECTIVES: To explore the clinical characteristics, postnatal treatment and prognosis of giant fetal hepatic hemangioma (GFHH). METHOD: Retrospective analysis was performed on children with giant fetal hepatic hemangioma (maximum tumor diameter > 40 mm) diagnosed by prenatal ultrasound and MRI from December 2016 to December 2020. These patients were observed and treated at the Children's Hospital of Fudan University after birth. The clinical data were collected to analyze the clinical characteristics, treatment, and prognosis of GFHH using independent sample t tests or Fisher's exact tests. RESULTS: Twenty-nine patients who were detected by routine ultrasound in the second and third trimester of pregnancy with giant fetal hepatic hemangiomas were included. The first prenatal ultrasound diagnosis of gestational age was 34.0 ± 4.3 weeks, ranging from 22 to 39 weeks. Of the patients, 28 had focal GFHHs and 1 had multifocal GFHHs. Surgery was performed, and the diagnosis was confirmed histopathologically in two patients. There were 8 cases with echocardiography-based evidence of pulmonary hypertension, 11 cases had a cardiothoracic ratio > 0.6, and 4 cases had hepatic arteriovenous fistula (AVF). The median follow-up time was 37 months (range: 14-70 months). During the follow-up, 12 patients received medical treatment with propranolol as the first-line therapy. The treatment group had a higher ratio of cardiothoracic ratio > 0.6 (P = 0.022) and lower albumin levels (P = 0.018). Four (14.8%) lesions showed postnatal growth before involuting. Complete response was observed in 13 (13/29) patients, and partial response was observed in 16 (16/29) patients. CONCLUSIONS: Fetal giant hepatic hemangioma is mainly localized, and its clinical outcome conforms to RICH (rapidly involuting) and PICH (partially involuting), but some fetal giant hepatic hemangiomas will continue to grow after birth and then gradually decrease. For uncomplicated giant fetal hepatic hemangioma, postnatal follow-up is the main concern, while those with complications require aggressive medical treatment. Propranolol may have no effect on the volume change of GFHH.

Beckwith-Wiedemann syndrome with multiple hepatic and cutaneous hemangiomas in a female patient of Albanian origin: Diagnostic and therapeutic considerations.

We describe a 2-month-old female infant with macroglossia, macrosomia, omphalocele, neonatal hypoglycemia, earlobe creases, low nasal bridge, midface retrusion, syndromic facies and multiple cutaneous and hepatic hemangiomas (HH). Genetic evaluation confirmed the diagnosis of Beckwith-Wiedemann Syndrome (BWS) with mosaic uniparental disomy 11 as the underlying genetic mechanism suggested by partial hypermethylation of H19/IGF2:IG-DMR and partial hypomethylation of KCNQ1OT1:TSS-DMR on chromosome 11p15.5. Pediatric endocrinology and cardiology assessments were normal. No malignant liver or renal tumors were detected during the follow-up period. Treatment with propranolol was started for the multiple HH, according to international recommendations. At 3-, 6-, and 9-month follow up, a gradual decrease in the size of the hemangiomas and AFP levels was observed, without side effects. This is the fifth case in the literature combining HH and BWS, and among these, the third case with this specific genetic defect suggesting a possible association between HH and BWS caused by 11 paternal uniparental disomy [upd(11)pat]. The case also highlights that if treatment is warranted, then oral propranolol can be used for the management of infantile HH in BWS patients similarly to non-BWS patients.

Patient with a novel syndrome with multiple benign hepatic lesions and extrahepatic neoplasms.

Simultaneous occurrence of benign hepatic lesions of different types is a sporadic phenomenon. To the best of our knowledge, we report the first clinical case of a syndrome with simultaneous manifestations of three different entities of benign liver tumors (hepatocellular adenoma, focal nodular hyperplasia and hemangioma) with a novel mutation detected in the liver adenoma and in the presence of a number of further extrahepatic organ neoplasms. Furthermore, we describe for the first time the presence of liver epithelial cells of hepatocytic phenotype expressing cytokeratin 7 (CK7) at the border of the adenoma. These findings may be important for explaining pathogenesis of benign as well as malignant tumors based on genetic and histopathological features.

A Liver-Derived Vascular Lesion: Hepatic Hemangioma or Hepatic Epithelioid Hemangioendothelioma?

Hepatic epithelioid hemangioendothelioma (EHE) is an uncommon vascular endothelial cell tumor of the liver with numerous symptoms and features. The median affected age is 41, and females are more frequently affected than men. In the following article, a 37-year-old nurse is presented who was referred to the hospital with severe right upper quadrant pain. She had been misdiagnosed with hepatic hemangioma for years, which finally turned out to be hepatic EHE. Liver transplantation has been recognized as the therapeutic method of choice due to the considerable extent of liver involvement and nonresponse to medications.