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Heliyon
Heliyon
Heliyon
2405-8440
Elsevier

S2405-8440(24)13645-6
10.1016/j.heliyon.2024.e37614
e37614
Case Report
Hepatic glomus tumor, a case report and literature review
Lin Feizhuan
Yang Jianhui
Zhu Xin
Wu Kangze wukangze@zju.edu.cn
⁎
Department of hepatopancreatobiliary surgery, Shaoxing People's Hospital, Shaoxing city, Zhejiang province, China
⁎ Corresponding author. wukangze@zju.edu.cn
07 9 2024
30 9 2024
07 9 2024
10 18 e376142 2 2024
26 8 2024
6 9 2024
© 2024 The Authors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction

This case report documents an exceptionally rare hepatic glomus tumor, contributing valuable insights to the diagnosis and differential diagnosis of such tumors.

Main symptoms and important clinical findings

A 58-year-old male was admitted in 2023 due to unexplained elevation of bilirubin discovered during routine laboratory tests. The patient denied any symptoms, and physical examination revealed no positive findings. Laboratory tests indicated elevated bilirubin, while serum tumor markers remained within normal ranges. Contrast-enhanced computed tomography revealed an indistinctly bordered hepatic mass, displaying uneven and marked enhancement in the arterial phase and sustained enhancement in the venous phase.

The main diagnoses, therapeutic interventions, and outcomes

The patient underwent laparoscopic partial hepatectomy, and pathological examination suggested a spindle cell tumor of mesenchymal origin. Immunohistochemistry confirmed positive staining for smooth muscle actin, leading to the final diagnosis of a hepatic glomus tumor with undetermined malignant potential. Follow-up at 6 months postoperatively showed no signs of metastasis or recurrence.

Conclusion

This case underscores the subtle radiological distinctions of hepatic glomus tumors, resembling hemangiomas but manifesting unique features. Additionally, the crucial role of immunohistochemistry in achieving a definitive diagnosis is emphasized in this report.

Keywords

Hepatic nodules
Glomus tumor
Diagnosis
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pmc1 Introduction

Glomus tumor is a rare mesenchymal tumor composed of modified smooth muscle cells resembling normal vascular globules. It represents a special form of arteriovenous anastomosis, making it prone to misdiagnosis as vascular malformation or arteriovenous malformation in clinical settings. Typically found in dermal regions rich in vascular globules, such as the subungual areas of fingers (toes) or the deep dermis of hands, palms, wrists, forearms, and feet, hepatic glomus tumor are extremely uncommon [1]. To date, there have been fewer than 10 reported cases of hepatic glomus tumors in existing literature [[2], [3], [4], [5], [6], [7], [8]]. Here, we present a case of hepatic glomus tumor. The aim is to provide relevant information for the diagnosis and treatment of glomus tumors in uncommon locations, ultimately improving patient prognosis.

2 Case presentation

2.1 Patient's admission

A 58-year-old male was admitted in 2023 due to unexplained elevation of bilirubin found during laboratory tests. He denied symptoms such as right upper abdominal pain, bloating, loss of appetite, nausea, vomiting, or diarrhea. Physical examination revealed no jaundice or abdominal mass. Previous gastrointestinal endoscopy had identified multiple colonic polyps, which were removed, and pathology suggested tubular adenomas.

2.2 Laboratory tests and imaging findings

Laboratory tests showed a white blood cell count of 6·84 x 10^9/L, hemoglobin of 147 g/L, aspartate aminotransferase of 23·8 IU/L, alanine aminotransferase of 20·9 IU/L, alkaline phosphatase of 98·6 IU/L, total bilirubin elevated to 50·2 μmol/L, direct bilirubin elevated to 7·1 μmol/L, and indirect bilirubin elevated to 43·1 μmol/L. Serum markers including alpha-fetoprotein, carbohydrate antigen 199, and carcinoembryonic antigen were within normal ranges. Contrast-enhanced computed tomography (CT) revealed a slightly hypodense mass in liver segment S6 during the non-contrast phase, approximately 40 × 28 mm in size, with indistinct borders. Late arterial phase enhancement showed uneven and marked enhancement, while the venous phase exhibited sustained enhancement. (Fig. 1).Fig. 1 Imaging Characteristics of Hepatic Glomus Tumor. (A) The non-contrast phase of the CT scan revealed a slightly hypodense mass in liver segment S6, approximately 40 × 28 mm in size, with indistinct borders. (B) Contrast-enhanced imaging during the arterial phase revealing heterogeneous enhancement of the tumor mass. (C) Sustained enhancement of the mass during the venous phase.

Fig. 1

2.3 Surgical intervention

The patient underwent laparoscopic partial hepatectomy, revealing a 3·5x3x5 cm tumor on pathology. The tumor was identified as a spindle cell neoplasm of mesenchymal origin (Fig. 2). Immunohistochemistry (IHC) demonstrated positive staining for smooth muscle actin (SMA) (Fig. 2), vimentin, CD34, CD99, Bcl-2, CD31, and Ki-67 (1 %), while negative for Desmin, CD117, AFP, Hepatocyte, HMB-45, Melan-A, S-100 and TFE3. The final diagnosis was a hepatic glomus tumor with undetermined malignant potential. Follow-up at 6 months postoperatively showed no signs of metastasis, recurrence or adverse events.Fig. 2 Microscopic Examination and SMA Staining of Hepatic Glomus Tumor. (A) Microscopic view of hepatic glomus tumor. (B) SMA staining showing positive immunoreactivity.

Fig. 2

3 Discussion

Hepatic glomus tumor is a rare condition, and due to its uncommon occurrence, there is currently no consensus regarding its nature and prognosis. Owing to its distinctive feature of uneven contrast agent uptake during the CT arterial phase, we promptly performed laparoscopic partial hepatectomy shortly after the CT examination. Pathological confirmation post-surgery indicated malignant potential; however, based on the ongoing follow-up, the patient's prognosis appears promising.

Based on our case and literature reports (Table 1) [[2], [3], [4], [5], [6], [7], [8]], the onset age of hepatic glomus tumors primarily ranges from 50 to 63 years. However, there were two patients aged 18 and 39, respectively. The male-to-female ratio is 6:2, suggesting that hepatic glomus tumors are more common in elderly men. Nonetheless, given that there are only 8 cases, this conclusion may have a significant bias.Table 1 Clinical Presentation, Laboratory Findings, Imaging Characteristics, Treatment, and Immunohistochemistry of Retrieved Hepatic Glomus Tumors

This table enumerates the clinical manifestations, laboratory investigations, radiological features, treatment modalities, and immunohistochemical characteristics associated with retrieved hepatic glomus tumors.

Table 1Authors	Gender	Age	clinical findings	Immunohistochemistry	Treatment	CT findings	
Amoueian et al. [2]	Female	50	Vague epigastric pain and nausea; an enlarged left liver lobe in physical examination; a mild increase in alkaline phosphatase and bilirubin;	Vimentin (+), SMA (+); CD34 (+); HMB45 (−); CEA (−); synaptophysin (−), chromogranin (−), CK (−)	Partial liver resection	a huge hepatic mass that replaced the entire left lobe	
Hirose et al. [3]	Male	39	Fullness in the epigastrium; elevated biliary tract enzymes	Vimentin (+), SMA (+); focal positivity for calponin and synaptophysin; Ki-67 proliferative index was less than 3 % in cystic lesions and 15 % in solid lesions	Partial liver resection	a 21-cm solid and cystic mass in the left liver lobe	
Gassel et al. [4]	Male	61	Lack of appetite and weight loss	Desmin (+); actin (+); vimentin (+)	Partial liver resection	a well-deŽ ned mas s 4 cm in diameterwith nodular uptake of contrast medium	
Jaiswal et al. [5]	Male	57	Right flank pain; microhematuria	Vimentin (+), SMA (+); CD31 (+); CD34 (+)	Partial liver resection	a 3.5-cm hypervascular mass within the posterior segment of the right lobe of the liver	
Geramizadeh et al. [6]	Female	50	Right upper quadrant bulging	SMA (+); CD34 (+); CD31 (+; endothelial cells)	Partial liver resection	None	
Kihara et al. [8]	Male	63	Epigastric pain and hiccups	Vimentin (+); SMA (+); type IV collagen (+)	Partial liver resection	a hepatic monolocular cystic mass	
Li et al. [7]	Male	18	Hypertension and arrhythmia	SMA (+); vimentin (+); syn (+); cyclinD1 (+); CD34 (±); desmin (±); calponin (±); b-catenin (±)	Partial liver resection	an oval lesion with abnormal enhancement in the left lobe of the liver, which was sized 6.0 cm4.6 cm and showed heterogeneous enhancement at the edge of the lesion in early phase of enhancement	
Our case	Male	58	elevation of bilirubin	SMA (++), Vim (++), CD34 (+), CD99 (partially weak +), Bcl-2 (partially weak +), CD31 (vascular +), Ki-67 (+1 %), CKpan (−), Calponin (+), CD117 (−), AFP (−), Hepatocyte (−), HMB-45 (−), Melan-A (−), Desmin (−), S-100 (−), TFE3 (−), Catenin (membrane +)	Partial liver resection	a slightly hypodense mass in liver segment S6, approximately 40 × 28 mm, with indistinct borders. Arterial phase enhancement showed uneven and marked enhancement, while the venous phase exhibited sustained enhancement	

Upon reviewing literature on hepatic glomus tumor, clinical presentations vary (Table 1) [[2], [3], [4], [5], [6], [7], [8]]. Some patients may exhibit nonspecific symptoms such as upper abdominal pain [2,5,8] and nausea [2], while others remain asymptomatic, with only elevated bilirubin levels in laboratory tests [2,3]. The connection between elevated bilirubin and hepatic glomus tumor remains uncertain.

Hepatic glomus tumors should be differentiated from various liver conditions, including malignant liver tumors, hepatic adenomas, hemangiomas, and focal nodular hyperplasia. While the imaging characteristics of hepatic glomus tumors may not be highly specific, they still offer some diagnostic clues. The uneven enhancement during the CT arterial phase and sustained enhancement during the venous phase can help distinguish hepatic glomus tumors from hepatocellular carcinoma, which typically shows rapid arterial phase enhancement and quick washout during the venous phase. However, these features do not significantly differ from those of hepatic hemangiomas, necessitating further investigation for accurate differentiation. Focal nodular hyperplasia typically presents with marked and uniform enhancement during the arterial phase and gradually decreasing enhancement in the portal phase, which differs from the imaging features of hepatic glomus tumors and can aid in differentiation. Nevertheless, both contrast-enhanced CT and magnetic resonance imaging (MRI) are non-invasive methods and cannot provide definitive evidence; ultimately, a conclusive diagnosis requires pathological examination.

Surgical resection is currently the recommended treatment (Table 1) [[2], [3], [4], [5], [6], [7], [8]], and postoperative IHC is crucial for confirming hepatic glomus tumor. Typically, positive staining for SMA and vimentin is observed[2,3,[5], [6], [7], [8]], with SMA being the primary marker. However, there is controversy surrounding other markers like Desmin, CD34, and CD31, with some studies reporting positive results and others negative [[2], [3], [4], [5], [6], [7], [8]].

The prognosis for hepatic glomus tumor is generally favorable, with no reported cases of postoperative recurrence in our case and others (Table 1) [[2], [3], [4], [5], [6], [7], [8]]. However, due to the short follow-up period and the uncertainty regarding its malignant potential, definitive conclusions cannot be drawn, necessitating continued close monitoring.

4 Conclusion

In summary, hepatic glomus tumor is a rare liver tumor with an unclear tissue origin. Clinical manifestations lack specificity, and while imaging aids in differential diagnosis, it provides limited assistance in confirming the disease. Immunohistochemistry is crucial for a definite diagnosis, typically showing positive staining for SMA and vimentin. The nature of hepatic mesenchymal hamartoma remains uncertain, although current reports suggest a favorable prognosis. However, limitations exist due to short follow-up periods in available data, recommending surgical resection followed by long-term monitoring.

Data availability

Data will be made available on request.

Ethics statement

Informed consent has been obtained from all individuals included in this case report/case series. The individuals providing consent understand that the publication of clinical conditions, research findings, de-identified images, clinical reports, and any other information related to each patient discussed in the journal will be widely and freely accessible to the public, without restrictions. The signed informed consent acknowledges that the publication may be promoted on the journal's website, possibly featured in news and/or social media, and may be utilized for future research advancements.

The privacy and confidentiality of the patients involved have been carefully considered. Any personal information that could potentially identify the patients has been appropriately de-identified to protect their privacy. The patients have been made aware that their information may be used for academic research, medical publications, and may be disseminated in various public domains.

This submission adheres to the ethical standards and guidelines set forth by Heliyon regarding the publication of case reports and case series. The authors affirm that they have complied with all relevant ethical considerations, and this submission does not violate any ethical or legal standards.

Funding

This study was supported by the Zhejiang Provincial Health and Medical Commission of China under the Zhejiang Provincial Health Program (No.2024KY473 ).

CRediT authorship contribution statement

Feizhuan Lin: Writing – original draft, Conceptualization. Jianhui Yang: Data curation. Xin Zhu: Data curation. Kangze Wu: Writing – review & editing, Data curation, Conceptualization.

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.

Acknowledgments

None.
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