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

Abstract

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.

1. Introduction

Hepatic hemangioma (HH) is the most common type of benign mesenchymal liver tumor and is often asymptomatic, small, slow-growing and has a good prognosis [1,2]. It is usually found occasionally during imagological examinations of upper abdominal organs, such as computed tomography (CT) or ultrasonography [3]. Space-occupying lesions larger than 10 cm have been defined as giant HH with high-risk or rare complications [4]. The incidence of ruptured bleeding, which is the most severe complication, is low (1–4%) [5,6], but the mortality is rather high (36–39 %) [7]. Meanwhile, the incidence of internal hemorrhage is extremely rarer, with only a few cases reported [8,9]. Therefore, there are limited treatment experiences that we can refer to for their management. Untimely or inappropriate treatment will contribute to a worse prognosis. Even today, surgical resection is still considered the most effective treatment, and three-dimensional visualized technology (3DVT) is an emerging and important method of individualized treatment [10].

Clear management of HH with internal hemorrhage is urgently needed. In this report, we report a case of giant HH with spontaneous internal hemorrhagic necrosis in a male patient and show the entire treatment process. He was managed by irregular right hepatectomy under the guidance of 3DVT, recovered well and was discharged from the hospital. Through a literature review of previous cases, we summarize the clinical features of giant HH with internal hemorrhage necrosis.

2. CASE presentation

2.1. Chief complaints

A 52-year-old Chinese man was admitted to the Department of Hepatobiliary Surgery I of Zhujiang Hospital of Southern Medical University with a complaint of distending pain on the right abdomen for 1 day.

2.2. History of present illness

Symptoms suddenly started 1 day before presentation with persistent distending pain on the right abdomen. There was no obvious inducement.

2.3. History of past illness

The patient had previously been diagnosed with giant HH at another hospital through CT and MRI on February 12, 2023. Due to insufficient surgical conditions at the other hospital, he subsequently underwent transhepatic arterial embolization (TAE) for giant HH 2 weeks before this presentation. The patient denied any medication history and underlying diseases.

2.4. Personal and family history

The patient denied any family history of tumors.

2.5. Physical examination

On physical examination, the vital signs were as follows: body temperature, 36.7 °C; blood pressure, 101/66 mmHg; heart rate, 75 beats per min; respiratory rate, 18 breaths per min. There was no jaundice or scleral icterus. Furthermore, a painful mass with a diameter of 15 cm was found on the right abdomen. Other abdominal physical examinations could not be performed due to the large painful mass.

3. Laboratory examinations

The red blood cell level was 2.99 T/L (normal, 4.3–5.8 T/L), the hemoglobin level was 83 g/L (normal, 130–175 g/L), liver function was Child‒Pugh A, coagulation function and α-fetoprotein were within the normal range, and no abnormalities were found in other laboratory results (Table 1).

Table 1.

Laboratory parameters upon admission to our hospital.

Testing Item	Result				Reference range		Testing Item	Result		Reference range
Hematology							Coagulation
WBC	9.54		G/L		3.5–9.5	G/L	TT-CON	19.0	s	–
Neut	7.29		G/L		1.8-6.3	G/L	APTT-CON	28.2	s	–
Lymph	1.32		G/L		1.1–3.2	G/L	PT-CON	11.6	s	–
Mono	0.82		G/L		0.1-0.6	G/L	PT	13.3	s	9.8–12.1	s
Eos	0.10		G/L		0.02-0.52	G/L	INR	1.15		0.88-1.08
Baso	0.02		G/L		0.0–0.06	G/L	PTA	80.1	%	70–130	%
RBC	2.99		T/L		4.3–5.8	T/L	APTT	29.8	s	25.0–31.3	s
Hb	83		g/L		130–175	g/L	TT	16.7	s	14.0–21.0	s
Hct	0.26		L/L		0.40-0.50	L/L	Fg	6.35	g/L	1.80-3.50	g/L
PLT	294		G/L		125–350	G/L	DDI	2.15	mg/L	0–0.55	mg/L
ESR	99		mm/h		0–28	mm/h	PLG	90.9	%	80.0–120.0	%
Biochemistry							Tumor markers
eGFR	90.4				>90		CA199	8.13	kU/L	0–35	kU/L
TP	57.4		g/L		65–85	g/L	NSE	6.81	μg/L	0–13	μg/L
ALb	31.5		g/L		35–50	g/L	CEA	1.15	μg/L	0–5	μg/L
TBil	23.8		μmol/L		3.4–20.5	μmol/L	CA242	2.38	kU/L	0–20	kU/L
DBil	14.7		μmol/L		0–8.6	μmol/L	Fer	595.01	μg/L	0–322	μg/L
AST	14		IU/L		≤40	IU/L	AFP	1.70	μg/L	0–20	μg/L
ALT	41		IU/L		≤41	IU/L	PSA	0.98	μg/L	0–5	μg/L
ALP	212		IU/L		40–130	IU/L	CA125	20.89	kU/L	0–35	kU/L
GGT	317		IU/L		10–60	IU/L	HGH	0.50	μg/L	0–7.5	μg/L
LDH	243		IU/L		120–250	IU/L	CA153	6.04	kU/L	0–35	kU/L
LAP	52.9		IU/L		12–37	IU/L
CK	55.6		IU/L		30–200	IU/L	Hepatitis virus
BUN/Cr	13.6				20.0–40.0		HBsAg	0.397	COI	<1.00	COI
chol	3.78		mmol/L		<5.18	mmol/L	HBsAb	<2.00	IU/L	<10	IU/L
TG	1.02		mmol/L		≤1.7	mmol/L	HBeAg	0.071	COI	<1.00	COI
Na	4.25		mmol/L		3.5–5.3	mmol/L	HBeAb	1.51	COI	>1.00	COI
K	136.4		mmol/L		136–145	mmol/L	HBcAb	1.96	COI	>1.00	COI
Cl	102.5		mmol/L		96–108	mmol/L	HCV-lgG	0.037	COI	<1.00	COI
Ca	1.99		mmol/L		2.10-2.70	mmol/L
UA	243		μmol/L		208–428	μmol/L	Urine
HSCRP	78.54		mg/L		≤3.0	mg/L	NIT	(−)		(−)
Glu	5.31		mmol/L		3.9-6.1	mmol/L	WBC	(−)		(−)
AMY	33.1		IU/L		25–125	IU/L	ERY	(−)		(−)

WBC: white blood cell, Neut: neutrophil, Lymph: lymphocyte, Mono: monocyte, Eos: eosinophil granulocyte, Baso: basophil leucocyte, RBC: red blood cell, Hb: hemoglobin, Hct: hematocrit, PLT: platelet, ESR: erythrocyte sedimentation rate, eGFR: evaluation of glomerular filtration rate, TP: total protein, ALb: albumin, TBiL: total bilirubin, DBiL: direct bilirubin, AST: aspartate aminotransferase, ALT: alanine aminotransferase, ALP: alkaline phosphatase, GGT: gamma-glutamyl transferase, LDH: lactate dehydrogenase, LAP: leucineaminopeptidase, CK: creatine kinase, BUN/Cr: blood urea nitrogen/creatinine, chol: cholesterol, TG: triglyceride, Na: sodium, K: potassium, Cl: chloride, Ca: Calcium, UA: uric acid, HSCRP: High-sensitivity C-reactive protein, Glu: glucose, AMY: amylase, CON: control, TT: thrombin time, PT: prothrombin time, APTT: activated partial thromboplastin time, INR: international normalized ratio, PTA: Prothrombin activity, Fg: fibrinogen, DDI: D-Dimer, PLG: plasminogen, NSE: neuron specific enolase, CEA: carcinoembryonic antigen, Fer: ferritin, AFP: α-fetoprotein, PSA: prostate specific antigen, HGH: human growth hormone, HBsAg: hepatitis B surface antigen, HBsAb: hepatitis B surface antibody, HBeAg: hepatitis B e antigen, HBeAb: hepatitis B e antibody, HBcAb: hepatitis B core antibody, HCV: hepatitis C virus, IgG: immunoglobulin G, NIT: nitrite, ERY: erythrocyte.

3.1. Imaging examinations

CT scans revealed a lumpy mass shadow protrusion with a nodular and patchy dense shadow in the right lobe of the liver (Fig. 1A) and a mass (15.8 × 14.2 × 14.7 cm) with high density exceeding the horizontal plane of the umbilicus by 6 cm (Fig. 1B).

Fig. 1.

Imaging findings of the patient. (A) Preoperative CT scan suggesting a mass (arrow) and postoperative change after arterial embolization (dotted line); (B) Preoperative CT scan suggesting a giant hepatic hemangioma (dotted line indicates the horizontal plane of the umbilicus); (C) Preoperative three-dimensional visualized model (H: hepatic hemangioma); (D) Giant hepatic hemangioma; (E) Cutting surface after resection of hemangioma (dotted line) (R, right liver lobe; G, gallbladder); (F) Surgical specimen; (G) Postoperative CT scan; (H) Postoperative three-dimensional visualized model transection.

4. Further diagnostic work-up

The self-developed 3D visualization system (MI-3DVS, software copyright: No.2008SR18798) was used for 3D reconstruction. A three-dimensional visualized model was made based on CT showing adhesion between the HH and hepatic segments of S5 and S6 (Fig. 1C). No vascular variation was found. After communication, the patient agreed to undergo surgical treatment. The surgical plan and auxiliary line of resection were decided based on the patient's condition. Before surgery, conservative medication for protecting liver was given to patient. The tumor was marked on the surface of the skin under the guidance of ultrasound. An exploratory laparotomy was performed under general anesthesia. A 30 cm reverse L-incision was made from the right side of the ventral midline, and the giant HH with poor mobility and tissue edema was exposed (Fig. 1D). During the dissociation process of hemangioma, its lower boundary extended to the right pelvic cavity, the left boundary extended to the midline of the abdomen, and the right boundary reached the abdominal wall. In addition, the giant HH also adhered to the small intestine, right colon and omentum. The patient subsequently underwent omentectomy and irregular right hepatectomy (Fig. 1E) based on the preoperative model and auxiliary line during intermittent liver inflow occlusion. Surgical anatomy confirmed the diagnosis of internal hemorrhagic necrosis (Fig. 1F). The liver was dark red without cirrhosis during intraoperative exploration. The patient's vital signs, excluding blood pressure, were stable throughout the procedure, and a vasopressor drug was used to sustain blood pressure. The operative procedure was successful. Conservative drug treatment for protecting liver continued to be given to patient after surgery.

5. Final diagnosis

Combined with the patient's medical history, the final diagnoses were giant hepatic hemangioma with internal hemorrhage necrosis and postoperative changes after TAE.

6. Treatment

Postoperative CT scans and a three-dimensional visualized model revealed complete remission without necrosis (Fig. 1G and H). The patient recovered well and was discharged from the hospital.

7. Outcome and follow-up

The patient had no recurrence or complications in a 4-month follow-up.

8. Discussion

HH is the most common benign liver tumor with nonspecific symptoms, good prognoses and little malignant tendency, and cavernous hemangioma is its major subtype [1,2]. The etiology may be associated with changes in female hormones [11,12]. In most cases, HH is usually asymptomatic and easy to ignore and is discovered as an incidental imaging finding. The sensitivity of ultrasound, CT, or magnetic resonance imaging for diagnosing hepatic hemangioma is over 90 % [13]. However, when HH is quite large (diameter >10 cm), some patients may have symptoms such as abdominal discomfort, rupture, internal hemorrhage, and coagulation disorder [4,14,15]. If patients with overt clinical signs cannot be promptly and appropriately managed, the risk of mortality will rise [6,16]. Therefore, accurate diagnosis and timely treatment are crucial. In nonhemorrhagic HH, the typical features of enhanced CT are swift enhancement and slow expurgation. When complicated with hemorrhage, the images may manifest as medium hyperintensity. Nevertheless, reports of hepatic hemangioma with spontaneous internal hemorrhage are rare worldwide [8,9]. It is difficult to differentiate it from liver abscess, hepatic malignancies, or other diseases. In this case, the patient with distending pain was diagnosed with hemangioma with internal hemorrhage by enhanced CT, which showed high-density hemorrhagic foci. It was confirmed as internal hemorrhagic necrosis through intraoperative surgical anatomy. This study found that the patient had only distending pain. In addition, there were no other clinical symptoms, such as fever or shock.

Given the rarity of giant HH with internal hemorrhagic necrosis, a few studies have investigated its clinical features and prognosis. We reviewed published reports of giant HH with internal hemorrhage and/or necrosis. Their characteristics and those of the current case report are summarized in Table 2[[9], [17], [18], [19], [20], [21], [22]]. In these cases, all of the tumors were giant HHs with sizes larger than 10 cm. Through a literature review, we found only 2 case reports of giant HH with internal hemorrhage necrosis (including our case report). There were 4 case reports of simple internal hemorrhage and 2 case reports of simple necrosis. Most patients were female and over 50 years old. The main complaint of most patients was fever, which responded poorly to antibiotic and antipyretic therapy. Fever associated with HH may be the release of endogenous pyrogens, acute thrombosis and necrosis within the tumor or internal hemorrhage. The complaint of epigastric pain was relatively rare in the literature review. The patients’ abdominal pain was likely due to distension of the liver capsule. In this case report, the capsule of the HH was intact and tense during surgery, and surgical anatomy confirmed hemorrhage and necrosis. Therefore, we believe that the main cause of epigastric pain in this patient was mainly distension of the liver capsule due to internal hemorrhage. We noted that cases with complaint of fever had a longer onset time. Therefore, we speculate that onset time may be the reason for different complaints. Fever requires a long progression process, and abdominal pain can occur in a short period of time. In summary, giant HH with internal hemorrhage and/or necrosis can present with fever of unknown origin and epigastric pain. It should be considered one of the causes of fever or epigastric pain, provided that all other sources have been excluded. The duration of onset also helps with diagnosis. These findings will contribute to a more accurate preliminary diagnosis and suitable treatment plan in similar situations for clinicians (Table 2).

Table 2.

Review of giant HH characteristics with the complication of internal hemorrhage and/or necrosis.

Case No.	Gender	Age (years)	Size of hemangioma (diameter)	Chief Complaint	Management	Cause of Chief Complaint	Outcome	Reference
1	Male	52	15.8 cm	Right epigastric pain for 1 day	Embolization Conservative medication Surgical resection	Internal hemorrhagic necrosis	Cured	Present case
2	Female	59	10.0 cm	Recurrent cough and sputum accompanied by fever for 6 months and symptom aggravation for 3 months	Antibiotic therapy Surgical resection	Internal hemorrhagic necrosis	Cured	17
3	Female	52	16.0 cm	Fever for over 20 days	Antibiotic therapy Blood transfusion Embolization Surgical resection	Internal hemorrhage	Cured	9
4	Female	53	10.0 cm	Fever for about 3 months	Antibiotic therapy Antipyretic therapy Surgical resection	Internal hemorrhage	Cured	18
5	Female	56	10.5 cm	Anemia and low-grade fever for 2 months	Surgical resection	Internal hemorrhage	Cured	19
6	Male	65	11.3 cm	Persistent dull pain in the upper right abdomen lasting 5 days	Surgical resection	Internal hemorrhage	Cured	20
7	Female	49	15.0 cm	Fever for about 3 months	Surgical resection	Internal necrosis	Cured	21
8	Male	33	20.0 cm	Fever for about 2 months	Surgical resection	Internal necrosis	Cured	22

The treatment principle of HH should be to achieve the most satisfactory outcome with minimal trauma. Surgical resection and hepatic artery embolization are the main treatment methods for liver hemangioma. Surgical resection is associated with a high risk for morbidity and mortality and is not recommended for asymptomatic patients [23]. TAE is a treatment with minimal trauma, low cost, and fast postoperative recovery, which can reduce the size of HH through embolizing blood-supply arteries. Expert consensus indicates that the short-term efficacy of TAE is accurate, but the long-term recurrence rate is relatively high [4]. However, the curative effect of two cases treated with embolization was not ideal (Table 2). Surgical resection is still the most definitive and effective therapeutic method for HH [23]. Most patients were treated via surgery through a literature review (Table 2). Currently, three-dimensional visualization technology has become increasingly important in surgical treatment [10]. In this case, due to the diagnosis of giant HH with internal hemorrhage and the limited space of the abdominal cavity, exploratory laparotomy was chosen. 3DVT clearly displayed the spatial relationship between HH and intrahepatic blood vessels and ensured that each liver segment has enough blood supply by virtual surgery. Through preoperative three-dimensional planning, the actual surgery is more purposeful, which will effectively shorten the surgical time to reduce unnecessary exposure and dissection. Irregular right hepatectomy was performed in this case to avoid expanding the scope of hepatectomy, keep functional liver tissue as much as possible, and reduce the occurrence of complications.

Some studies have indicated that TAE can shorten hemangioma, which can reduce the difficulty of secondary operations [24,25]. However, this was not applicable in this case. Under the guidance of the three-dimensional visualized model, the difficulty of operation was high, which is worth noting for surgeons. First, due to the treatment history of TAE, neovascularization led to wound bleeding and oozing during the liver disconnection process, increasing the difficulty of transection. This required frequent hemostasis and close monitoring of vital signs. Second, HH was extensively adhered to surrounding tissues and organs. There was a high risk of massive hemorrhage caused by vascular injury or hemangioma rupture. This also needed careful separation and exploration without omission during the separation process. Finally, the massive volume of giant HH (15.8 × 14.2 × 14.7 cm) with intact encapsulation, poor mobility and high tension restricted the intraoperative operation to a certain degree and increased the risks mentioned above. In summary, although TAE has advantages such as minimal trauma and fast postoperative recovery, clinicians should keep a strict watch over the patient by strengthening postoperative follow-up to evaluate the efficacy of TAE and regard surgical resection as a radical treatment method.

9. CONCLUSION

HH is the most common type of liver tumor, but there are few reports about giant HH with internal hemorrhagic necrosis. Sufficient attention should be paid to atypical symptoms, such as fever or epigastric pain, if all other sources have been excluded, as their onset time can aid in diagnosis. Given the rarity of the condition, specific management guidelines have yet to be established. Surgical resection is still the most effective therapeutic method. The application of three-dimensional visualization technology for preoperative planning and intraoperative guidance can effectively reduce surgical risks and complications. For such rare and high-risk cases, this is an effective method that is worthy of clinical promotion.

Informed consent statement

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

Ethics approval and consent to participate

Ethical review and approval were not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided their written informed consent to participants in this study. Written informed consent was obtained from the individual for the publication of any potentially identifiable images or data included in this article. This report has obtained written informed consent from the patient's legal representative (his family) for publication.

Data availability statement

The authors declare that all data supporting the findings of this study are available within the article.

CRediT authorship contribution statement

Hao Zhong: Writing – review & editing, Writing – original draft, Formal analysis, Data curation. Si-lue Zeng: Visualization, Validation, Supervision. Zhen-ju Huang: Formal analysis, Data curation, Conceptualization. Yi Zhou: Validation, Software, Resources, Project administration. Pei-lin Cai: Software, Resources. Yu-cun Zhu: Resources, Project administration. Ning Zeng: Methodology, Investigation, Formal analysis.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Abbreviation

HH

hepatic hemangioma

CT

computed tomography

3DVT

three-dimensional visualized technology

TAE

transhepatic arterial embolization

WBC

white blood cell

Neut

neutrophil

Lymph

lymphocyte

Mono

monocyte

Eos

eosinophil granulocyte

Baso

basophil leucocyte

RBC

red blood cell

Hb

hemoglobin

Hct

hematocrit

PLT

platelet

ESR

erythrocyte sedimentation rate

eGFR

evaluation of glomerular filtration rate

TP

total protein

ALb

albumin

TBiL

total bilirubin

DBiL

direct bilirubin

AST

aspartate aminotransferase

ALT

alanine aminotransferase

ALP

alkaline phosphatase

GGT

gamma-glutamyl transferase

LDH

lactate dehydrogenase

LAP

leucineaminopeptidase

CK

creatine kinase

BUN/Cr

blood urea nitrogen/creatinine

chol

cholesterol

TG

triglyceride

Na

sodium

K

potassium

Cl

chloride

Ca

calcium

UA

uric acid

HSCRP

high-sensitivity C-reactive protein

Glu

glucose

AMY

amylase

CON

control

TT

thrombin time

PT

prothrombin time

APTT

activated partial thromboplastin time

INR

international normalized ratio

PTA

prothrombin activity

Fg

fibrinogen

DDI

D-Dimer

PLG

plasminogen

NSE

neuron specific enolase

CEA

carcinoembryonic antigen

Fer

ferritin

AFP

α-fetoprotein

PSA

prostate specific antigen

HGH

human growth hormone

HBsAg

hepatitis B surface antigen

HBsAb

hepatitis B surface antibody

HBeAg

hepatitis B e antigen

HBeAb

hepatitis B e antibody

HBcAb

hepatitis B core antibody; HCV: hepatitis C virus

IgG

immunoglobulin G

NIT

nitrite

ERY

erythrocyte