Rare presentation of hibernoma as a cystic swelling


 Table of Contents   CASE REPORT Year : 2022  |  Volume : 12  |  Issue : 1  |  Page : 51-53  

Rare presentation of hibernoma as a cystic swelling

Prashant Sawarkar, Bhupendra Mehra, Siddharth Pramod Dubhashi, Fatema Kamal
Department of Surgery, All India Institute of Medical Sciences, Nagpur, Maharashtra State, India

Date of Submission03-Jun-2021Date of Decision10-Oct-2021Date of Acceptance02-Dec-2021Date of Web Publication31-Jan-2022

Correspondence Address:
Prashant Sawarkar
Department of Surgery, All India Institute of Medical Sciences, Nagpur - 441 108, Maharashtra
India
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Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/ijabmr.ijabmr_365_21

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   Abstract 


Hibernoma is a rare benign tumor of brown fat origin. It presents as a painless, slowly growing soft tissue tumor mimicking lipoma or liposarcoma, usually affecting adults, with a slight male predominance and a peak of incidence between the third and fourth decades of life. This is a case report of 35-year male, who presented with a mobile, fluctuant and transilluminant, swelling of 7 cm × 5 cm, on medial aspect of the right upper leg. Ultrasonography showed well-defined cystic lesion of varied echotexture with dense internal echoes and septation. Lesion was excised in-toto. On gross examination, it was a subcutaneous, multiseptate cyst containing clear serous fluid with cholesterol crystals with a solid area of 3 cm × 2 cm. Histopathological examination revealed large tumor cells with abundant granular (multivacuolated) cytoplasm. Postoperative recovery period was uneventful. We present this case as cystic degeneration in case of hibernoma.

Keywords: Cystic swelling, dermoid cyst, hibernoma, multivacuolated cytoplasm


How to cite this article:
Sawarkar P, Mehra B, Dubhashi SP, Kamal F. Rare presentation of hibernoma as a cystic swelling. Int J App Basic Med Res 2022;12:51-3
How to cite this URL:
Sawarkar P, Mehra B, Dubhashi SP, Kamal F. Rare presentation of hibernoma as a cystic swelling. Int J App Basic Med Res [serial online] 2022 [cited 2022 Jan 31];12:51-3. Available from: 
https://www.ijabmr.org/text.asp?2022/12/1/51/336959    Introduction Top

Lipoma is considered as universal tumor and its prevalence increases with age. About 2% of people are affected with lipoma.[1] Hibernomas are nonmalignant lipomatous tumors, a slow growing and painless with prevalence of 1.6% of all lipomatous tumors.[2] Brown fat is abundant in newborns. Brown fat cells come from mesoderm, primary function being thermoregulation by nonshivering thermogenesis. It is also present and metabolically active in adult humans[3],[4] but its prevalence decreases as humans age.[5] Several names have been used to describe the lesion, including lipoma of immature adipose tissue, lipoma of embryonic fat, and fetal lipoma. In adults, inter-scapular region, mediastinum, retroperitoneum, back, and thigh are the most common areas of residual brown fat and, sometimes, it is located in head-and-neck area.[6],[7] It usually affects adult males in their thirties and forties. It is clinically and radiologically indistinguishable from benign lesions like lipoma or liposarcoma.

   Case Report Top

A 35-year-old gentleman presented with a single painless, progressive 7 cm × 5 cm size swelling on medial aspect of right upper leg. There was no history of any inciting factors. The lesion was nontender, fluctuant, transilluminant and was free from skin and underlying tissues. Ultrasonography (USG) [Figure 1] showed well-defined cystic lesion with collection which showed varied echotexture with dense internal echoes and septation. Peripheral small calcific foci of size 7.4 mm were noted along the inferior wall. Underlying muscles were normal. Imaging features favored the possibility of epidermoid/dermoid cyst. All hematological and biochemical parameters were within normal limits. Excision of swelling was done under local anesthesia [Figure 2]. On gross examination, it was a multiseptate cyst containing clear serous fluid with cholesterol crystals and solid area of 3 cm × 2 cm identified which was tan yellow color, firm to hard. Histopathological examination [Figure 3]a and [Figure 3]b revealed unremarkable epidermis and dermis with subcutaneous located encapsulated tumor mass. Large tumor cells were arranged in organoid pattern, with granular (multivacuolated) cytoplasm and small central nuclei. Mature adipocytes were seen at the periphery. Sections from cystic areas showed similar cells and no capsular lining was identified. Areas of hyalinization, myxoid degeneration, hemorrhage, and granulation tissue along with few cholesterol clefts and scattered similar tumor cells were noted. The patient has been asymptomatic in the follow-up period of 1 year till date.

Figure 3: (a) High power ×40 view of hibernoma showing cells with granular/multivacuolated eosinophilic cytoplasm with central round to oval nucleus, (b) Low power image ×10 of hibernoma, showing organoid pattern of adipocytes with granular to multivacuolated cytoplasm with central round nucleus with eosinophilic to pale cytoplasm

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   Discussion Top

The first person who described this tumor was Merkel in 1906, who labeled it as pseudo-lipoma.[8] The term Hibernoma coined by Gery in 1914, when he observed the similarity in hibernating animals fat cells and cells present in this tumor.[9] Furlong et al.[10] have studied 170 cases of hibernoma and showed most common anatomic locations as lower extremity and thigh 32%, trunk 23%, upper extremity 21%, head and neck 14%, and abdominal cavity/retroperitoneum 10%. Out of 170 cases, 58% were males and 42% were females. The tumor occurs usually in middle- aged patients with mean age of 38 years. Four morphologic variants of hibernoma were identified: (1) typical hibernoma constituted 82%, (2) myxoid hibernoma constituted 8.2%, (3) lipoma-like variant constituted 7%, and (4) spindle cell hibernoma constituted 2.8%.

Hibernomas are classically slow-growing, but sometimes it can grow fast and cause pain when the adjoining structures get compressed. Their average size is 5–10 cm, but may be found as much as up to 20 cm.[11] Majority of the Hibernomas are subcutaneous as in our case with only 10% being intramuscular.[2] There is no reported malignant transformation in Hibernoma. USG is initial investigation of choice followed by fine-needle aspiration cytology for tissue diagnosis. Later computed tomography (CT) or magnetic resonance imaging can be done if required to know the relation of the surrounding structures for excision. 18F-fluorodeoxyglucose position emission tomography may be needed to differentiate from other tumours. Angiography may be needed, as some Hibernomas are hypervascular.[12] Uniform hyperechogenicity is characteristic of Hibernoma on USG, but in our case, it was described as a well-defined thick-walled exophytic cystic lesion in subcutaneous location, suggestive of dermoid or epidermoid cyst. CT scan shows the lesions as isointense or slightly hypointense compared to the subcutaneous fat, with enhancement after contrast enhancement.

Macroscopically, hibernoma is a well-circumscribed, encapsulated, lobulated soft greasy mass of usually 5–10 cm size. Cut surface reveals yellow to red brown appearance with rare areas of hemorrhage. It was cystic with a very small solid component in our reported case. Microscopic sections reveal encapsulated tumor comprising large multivacuolated cells and univacuolated cells. Multivacuolated cells have eccentric vesicular nucleus and characteristically fine granular, vacuolated cytoplasm. Abundant capillaries with a lack of mitosis or atypia are a usual feature. The presence of atypia and lipoblasts helps to differentiate liposarcoma from lipoma and hibernoma. These adipocytic tumors can be reliably diagnosed with the help of molecular markers such as MDM-2, CDK-4, and p-16.[13] Chromosomal aberration is present in hibernomas with reciprocal translocation in chromosomes 9 and 11.[14]

Incomplete excision may result in local recurrence of the tumor, thereby warranting a complete resection for this type of neoplasm.

   Conclusion Top

Hibernoma is a benign soft tissue tumor clinically and radiologically mimicking lipoma or liposarcoma. Cystic variant of hibernoma is a rare entity. Treatment includes excision of the lesion in-toto. Histological examination will clinch the diagnosis.

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Conflicts of interest

There are no conflicts of interest.

 

   References Top
1.Cherian SS, George EV, Chellappa PM. Parosteal lipomas of the right femur – A case report. J Evid Based Med Healthc 2017;4:905-7.  Back to cited text no. 1
    2.Mavrogenis AF, Coll-Mesa L. Soft tissue: Hibernomas. Atlas Genet Cytogenet Oncol Haematol 2013;17:60-74.  Back to cited text no. 2
    3.Nedergaard J, Bengtsson T, Cannon B. Unexpected evidence for active brown adipose tissue in adult humans. Am J Physiol Endocrinol Metab 2007;293:E444-52.  Back to cited text no. 3
    4.Saito M, Okamatsu-Ogura Y, Matsushita M, Watanabe K, Yoneshiro T, Nio-Kobayashi J, et al. High incidence of metabolically active brown adipose tissue in healthy adult humans: Effects of cold exposure and adiposity. Diabetes 2009;58:1526-31.  Back to cited text no. 4
    5.Graja A, Schulz TJ. Mechanisms of aging-related impairment of brown adipocyte development and function. Gerontology 2015;61:211-7.  Back to cited text no. 5
    6.Chen CL, Chen WC, Chiang JH, Ho CF. Intercapsular hibernoma – Case report and literature review. Kaohsiung J Med Sci 2011;27:348-52.  Back to cited text no. 6
    7.Hertoghs M, Van Schil P, Rutsaert R, Van Marck E, Vallaeys J. Intrathoracic hibernoma: Report of two cases. Lung Cancer 2009;64:367-70.  Back to cited text no. 7
    8.Merkel H. On a pseudolipoma of the breast. Beitr Pathol Anat 1906;39:152-7.  Back to cited text no. 8
    9.Kosem M, Karakok M. Hibernoma: A case report and discussion of a rare tumor. Turk J Med Sci 2001;25:175-2.  Back to cited text no. 9
    10.Furlong MA, Fanburg-Smith JC, Miettinen M. The morphologic spectrum of hibernoma: A clinicopathologic study of 170 cases. Am J Surg Pathol 2001;25:809-14.  Back to cited text no. 10
    11.Murphey MD, Carroll JF, Flemming DJ, Pope TL, Gannon FH, Kransdorf MJ. Benign musculoskeletal lipomatous lesions. Radiographics 2004;24:1433-66.  Back to cited text no. 11
    12.Daubner D, Spieth S, Pablik J, Zöphel K, Paulus T, Laniado M. Hibernoma – Two patients with a rare lipoid soft-tissue tumour. BMC Med Imaging 2015;15:4.  Back to cited text no. 12
    13.Patil SD, Sheik AR, Tewari V, Mutreja D. Hibernoma: A missed diagnosis!! Indian J Pathol Microbiol 2019;62:461-3.  Back to cited text no. 13
    14.Turaga KK, Silva-Lopez E, Sanger WG, Nelson M, Hunter WJ, Miettinen M, et al. A (9;11)(q34;q13) translocation in a hibernoma. Cancer Genet Cytogenet 2006;170:163-6.  Back to cited text no. 14
    
  [Figure 1], [Figure 2], [Figure 3]

 

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