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Malignant Glomus Tumour with A Rare Localization.

INTRODUCTION

Glomus tumor is a mesenchymal tumor comprising smooth muscle cells known as glomus cells; it is responsible for thermoregulation (1). It was first described by Masson in 1924 (2). Glomus tumors constitute <2% of soft tissue tumors (3). They are seen more commonly in the fourth and fifth decades of life in all age groups. Clinically, they are usually red and bluish-purple lesions and are <1 cm in size. Cold sensitivity, point sensibility, and paroxysmal pain are the most important features of these tumors (4). Glomus tumors, which usually show a subungual location, are rarely found outside the finger; however, cases have been reported in the foot, ankle, knee, hip, thigh, thorax, sacrum, and coccyxtic (5). A malignant glomus tumor is seen very rarely. It constitutes 1% of all glomus tumors and usually progresses with local infiltration (6, 7). Post-surgery local recurrence has generally been reported as 10% and is usually attributed to poor excision (8). In this article, a case of patellar malignant glomus tumor was presented due to the fact that it had a rare localization and recurred after treatment.

CASE REPORT

A 31-year-old male consulted the clinic due to pain in his right knee. Open biopsy performed for the same complaints two years ago reported "fusiform cell mesenchymal tumor," and resection and patellar reconstruction operation performed two months later revealed "lymphovascular proliferation, inflammatory granulation tissue. No residual tumor was seen, no tumor was seen at the surgical margins." At his subsequent application to the clinic, physical examination revealed pain after 110[degrees] flexion and palpable tenderness in the right knee, and a mass was seen at the same location. Excisional biopsy revealed a macroscopic mass sized 2x3 cm. Microscopic examination of the mass revealed tumor cell islands that were surrounded by vascular structures in various sizes, had eosinophilic cytoplasm, oval-round nucleus, wide and single nucleoli, and polygonal and oval appearance. These cells contain cytologic atypia and mitotic activity (Figure 1). Positive reaction was observed in tumor cells with immunohistochemically applied vimentin and smooth muscle actin; however, no reaction was observed in tumor cells with pancytokeratin, carcinoembryonic antigen, and desmin (Figure 2). The case was evaluated in favor of malignant glomus tumor in the presence of clinical, morphological, and immunohistochemical findings. Informed consent was obtained from the patient for the publication of this case report.

DISCUSSION

Glomus tumors constitute a small proportion of soft tissue tumors; they can be solitary or multicentric, and are quite rare. Glomus tumors, which usually show a subungual location, are rarely found outside the finger; however, cases have been reported in the foot, ankle, knee, hip, thigh, thorax, sacrum, and coccxy (5, 9). A glomus tumor located in the anterior region of the patella has been reported with two cases in the literature (8). The case we present here also has a patellar localization; it is rarely seen and shows local recurrence. Local recurrence after surgery was generally reported as 10%; it is usually attributed to inadequate excision. Complete surgical resection was performed in our case. After resection, granulation tissue was observed at the same location. However, the patient had recur-rent lesions in the same location in the examinations performed two years later. Putti and Tato (8) reported that local recurrence developed after local excision in two cases with glomus tumors located in the anterior region of the patella, and they attributed this situation to the insufficient excision of the tumor. Contrarily, in our case, recurrence did not develop after local excision; however, it developed despite the fact that wide excision was performed and no tumor was seen at the surgical margin. Folpe et al. (7) defined the criteria for malignant glomus tumors as follows: deeply-located tumor, diameter >2 cm, presence of marked atypia along with atypical mitotic figures, and mitotic activity. The presence of these characteristics has been found to be associated with the risk of metastasis. The presence of atypical mitosis revealed via the microscopic examination of our case overlaps with the malignancy criteria, i.e., atypia in tumor-forming cells and macroscopic diameter >2 cm.

CONCLUSION

Glomus tumors may rarely develop in regions outside the finger; it may be difficult to diagnose its involvements in these regions, and in cases of unexplained pain, glomus tumors should be considered in the differential diagnosis. Follow-up is required in terms of recurrence. In tumors sized >2 cm, malignant glomus tumors should be considered for differential diagnosis in the presence of nuclear atypia and atypical mitosis and should be followed up more closely in terms of local recurrence. It should also be considered that these tumors may have metastatic potential.

Informed Consent: Verbal informed consent was obtained from patient who participated in this case.

Peer-review: Externally peer-reviewed.

Author Contributions: Concept--E.K., A.A.I.; Design--E.K., F.A.A.; Supervision - E.K.; Resources--O.G.; Data Collection and/or Processing--E.K.; Analysis and/or Interpretation--N.E.; Literature Search--O.G.; Writing Manuscript - E.K.; Critical Review--E.K.

Conflict of Interest: No conflict of interest was declared by the authors.

Financial Disclosure: The authors declared that this study has received no financial support.

REFERENCES

(1.) Miettinen M, Paal E, Lasota J, Sobin H. Gastrointestinal glomus tumors: a clinicopathologic,immunohistochemical, and molecular genetic study of 32 cases. Am J Surg Pathol 2002; 26: 301-11. [CrossRef]

(2.) Masson P. Le glomus neuro-myo-arteriel des regions tactiles et ses tumeurs. Lyon Chi 1924; 21: 257-80.

(3.) Folpe AL. Pericytic (perivascular) tumours. In: Fletcher CDM, Unni KK, Mertens E, editors. Pathology and Genetics of Tumours of Soft Tissue and Bone. Lyon, France: IARC Press; 2002. p.136-7.

(4.) Tabanlioglu D, Leblebicioglu G, Boztepe G, Uzumcugil A, Erkin G, Ayhan M, et al. Glomus Tumor of Skin and Nail Bed: 3 Cases. Turkiye Klinikleri J Dermatol 2005; 15: 200-4.

(5.) GenCosmanoglu R, Inceoglu R, Kurtkaya-Yapicier O. Glomangioma of the hip. Dermatol Surg 2003; 29: 1244-7. [CrossRef]

(6.) Gould EW, Maniveland JC, Albores-Saavedra J. Locally infiltrative glomus tumors and glomangiosarcomas: a clinical, ultrastructural and immunohistochemical study. Cancer 1990; 65: 310-8. [CrossRef]

(7.) Folpe AL, Fanburg-Smith JC, Miettinen M, Weiss SW. Atypical and malignant glomus tumors:analysis of 52 cases, with a proposal for there classification of the glomus tumors. Am J Surg Pathol 2001; 25: 1-12. [CrossRef]

(8.) Putti E, Tato FB. Two cases of glomus tumor localized in the right knee: removal and recurrence. Chir Organi Mov 1991; 76: 375-8.

(9.) Baleato-Gonzalez S, Garcia-Figueiras R, Trujillo-Ariza MW, Carrera-Alvarez JJ. Malignant Glomus Tumor of the Peritoneum: Case Report. Korean J Radiol 2014; 15: 61-5. [CrossRef]

Elife Kimiloglu (1) [iD], Fatih Alper Ahlatci (2) [iD], Aysenur Akyildiz Igdem (1) [iD], Nusret Erdogan (1) [iD], Ozgecan Gundogar (1) [iD]

(1) Clinic of Medical Pathology, University of Health Sciences Gaziosmanpasa Taksim Training and Research Hospital, Istanbul, Turkey

(2) Clinic of Medical Pathology, Corlu State Hospital, Tekirdag, Turkey

Address for Correspondence: Elife Kimiloglu, E-mail: elife.sahan@gmail.com

Received Date: 09.11.2016 Accepted Date: 26.05.2017

DOI: 10.5152/jarem.2017.1305

Cite this article as: Kimiloglu E, Ahlatci FA, Akyildiz Igdem A, Erdogan N, Gundogar O. Malignant Glomus Tumour with A Rare Localization. JAREM 2018; 8: 50-1.
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Article Details
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Title Annotation:Olgu Sunumu / Case Report
Author:Kimiloglu, Elife; Ahlatci, Fatih Alper; Igdem, Aysenur Akyildiz; Erdogan, Nusret; Gundogar, Ozgecan
Publication:Journal of Academic Research in Medicine
Date:Apr 1, 2018
Words:1182
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