Rare malignant glomus tumor of the esophagus with pulmonary metastasis: a case report

Xiao, Annie; Ahlers, Michael; Dry, Sarah M.; Weber, Andrew T.; Chiu, Victor; Pessegueiro, Antonio

doi:10.21037/acr-21-72

Cited by 9 publications

(8 citation statements)

References 11 publications

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“…To date, 12 cases of esophageal GT are reported in English, including the present case [2,3,[8][9][10][11][12][13][14][15][16]. They are summarized in Table 1.…”

Section: Discussionmentioning

confidence: 99%

Thoracoscopic enucleation of an esophageal glomus tumor in the prone position: a case report and literature review

Matsumoto,

Okumura,

Miwa

et al. 2024

surg case rep

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Background Glomus tumors (GT) generally occur in the skin. However, esophageal GT, an extremely rare condition, has no established standardized treatment guidelines. Herein, we report the case of an esophageal GT successfully removed by thoracoscopic enucleation in the prone position using intra-esophageal balloon compression. Case presentation A 45-year-old man underwent an annual endoscopic examination and was found to have a submucosal tumor in the lower esophagus. Endoscopic ultrasound (EUS) revealed a hyperechoic mass originating from the muscular layer. Contrast-enhanced computed tomography identified a 2 cm mass lesion with high contrast enhancement in the right side of the lower esophagus. Pathologic findings of EUS-guided fine needle aspiration biopsy (EUS–FNA) revealed round to spindle shaped atypical cells without mitotic activity. Immunohistochemically, the tumor was positive for alpha-smooth muscle actin, but negative for CD34, desmin, keratin 18, S-100 protein, melan A, c-kit, and STAT6. He was diagnosed with an esophageal GT and a thoracoscopic approach to tumor resection was planned. Under general anesthesia, a Sengstaken–Blakemore (SB) tube was inserted into the esophagus. The patient was placed in the prone position and a right thoracoscopic approach was achieved. The esophagus around the tumor was mobilized and the SB tube balloon inflated to compress the tumor toward the thoracic cavity. The muscle layer was divided and the tumor was successfully enucleated without mucosal penetration. Oral intake was initiated on postoperative day (POD) 3 and the patient discharged on POD 9. No surgical complications or tumor metastasis were observed during the 1-year postoperative follow-up. Conclusions As malignancy criteria for esophageal GT are not yet established, the least invasive procedure for complete resection should be selected on a case-by-case basis. Thoracoscopic enucleation in the prone position using intra-esophageal balloon compression is useful to treat esophageal GT on the right side of the esophagus.

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“…To date, 12 cases of esophageal GT are reported in English, including the present case [2,3,[8][9][10][11][12][13][14][15][16]. They are summarized in Table 1.…”

Section: Discussionmentioning

confidence: 99%

Thoracoscopic enucleation of an esophageal glomus tumor in the prone position: a case report and literature review

Matsumoto,

Okumura,

Miwa

et al. 2024

surg case rep

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“…To date, 13 cases of hematocytomas of esophageal origin have been reported, but only 3 of these cases exhibited both a malignant histopathological phenotype and regional or distant metastasis [3][4][5]. Due to the rarity of this disease and scarcity of case reports, we have summarized the three cases of malignant hematoma of the esophagus.…”

Section: Discussion Andmentioning

confidence: 99%

Combined treatment for a rare malignant glomus tumor of the esophagus with pulmonary and liver metastases: a case report and review of literature

Liu,

Mao,

Shen

et al. 2023

Preprint

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Background: Glomus tumors are typically benign soft tissue tumors that occur at the extremities; malignant and viscerally occurring cases are extremely rare. Case Presentation: We report a 49-year old male patient with a malignant esophageal glomus tumor that was complicated by lung and liver metastases. Genetic test results guided the patient's individualized treatment. Consequently, treatment with Anlotinib combined with Tislelizumab achieved significant clinical benefits. Conclusion: Our case report demonstrates that immunotherapy combined with anti-angiogenic therapy in patients with malignant esophageal glomus tumors can achieve significant efficacy and suggests the potential value of Next-Generation Sequencing(NGS) detection in guiding personalized treatments in patients with malignant esophageal glomus tumors.

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“…7 9,17 One patient with a malignant oesophageal glomus tumour experienced rapid clinical deterioration with multiple painful nodules and was transferred to hospice care. 9 Based upon our brief literature review and anecdotal observations, we hypothesize that oesophageal glomus tumours have a propensity for malignant behaviour, compared to their more common gastric counterparts. We aim to further explore the clinical, pathologic, and molecular features of oesophageal glomus tumours through a review of cases identified at two large academic institutions.…”

Section: Introductionmentioning

confidence: 96%

“…5 To our knowledge, there are 12 additional case reports of oesophageal glomus tumours in the English literature (Table 1). [6][7][8][9][10][11][12][13][14][15][16][17] Of the eight additional cases with sufficient clinicopathologic parameters reported, four would be classified as malignant according to the criteria proposed by Papke et al 5 Metastases occurred in three cases (two of which would be classified as malignant, and one with insufficient clinicopathologic data), with involvement of lymph nodes (two cases), lung (two cases), liver (one case), bones (one case), scalp (one case), and subcutaneous tissue (one case). 7 9,17 One patient with a malignant oesophageal glomus tumour experienced rapid clinical deterioration with multiple painful nodules and was transferred to hospice care.…”

Section: Introductionmentioning

confidence: 99%

“… 6–17 Of the eight additional cases with sufficient clinicopathologic parameters reported, four would be classified as malignant according to the criteria proposed by Papke et al 5 . Metastases occurred in three cases (two of which would be classified as malignant, and one with insufficient clinicopathologic data), with involvement of lymph nodes (two cases), lung (two cases), liver (one case), bones (one case), scalp (one case), and subcutaneous tissue (one case) 7 9,17 . One patient with a malignant oesophageal glomus tumour experienced rapid clinical deterioration with multiple painful nodules and was transferred to hospice care 9 …”

Section: Introductionmentioning

confidence: 99%

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Oesophageal glomus tumours: rare neoplasms with aggressive clinical behaviour

2023

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Aims: Glomus tumours are neoplasms with perivascular smooth muscle differentiation, which rarely occur in the oesophagus and may behave aggressively in this site based upon prior case reports. This study describes the clinicopathologic features of three oesophageal glomus tumours diagnosed at two large academic institutions between 1984 and 2022. Methods and results: Three cases of oesophageal glomus tumours were identified. Patients included two females and one male, with an age range of 19-65 years. All three tumours behaved in a malignant fashion, with metastases to various sites (lymph nodes, lung, pericardium, pleura, diaphragm, scalp). One patient developed an aorto-oesophageal fistula, resulting in a fatal haemorrhage. Tumours ranged in size from 4.5 to 8.1 cm. Histologically, all tumours had a multinodular, perivascular growth pattern. The neoplasms showed varying degrees of cytologic atypia and spindling, elevated mitotic activity (2-12 mitotic figures per 10 high-power fields), and necrosis was seen in in two cases. All tumours expressed smooth muscle actin by immunohistochemistry, and harboured NOTCH gene alterations (MIR143::NOTCH2 fusion in two cases; NOTCH3 rearrangement and NOTCH1 point mutation in one case). An ATRX splicing mutation in exon 10 was also identified in one case. Conclusion: Oesophageal glomus tumours pose diagnostic challenges, given their rarity at this site, but can be recognised by their characteristic perivascular growth pattern, round central nuclei, and supportive ancillary studies. Given the propensity for aggressive behaviour in this location, we recommend management by a multidisciplinary sarcoma team for optimal outcome.

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Rare malignant glomus tumor of the esophagus with pulmonary metastasis: a case report

Cited by 9 publications

References 11 publications

Thoracoscopic enucleation of an esophageal glomus tumor in the prone position: a case report and literature review

Thoracoscopic enucleation of an esophageal glomus tumor in the prone position: a case report and literature review

Combined treatment for a rare malignant glomus tumor of the esophagus with pulmonary and liver metastases: a case report and review of literature

Oesophageal glomus tumours: rare neoplasms with aggressive clinical behaviour

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