Gastric hibernoma: a novel location and presentation of a rare tumor

Gastric hibernoma: a novel location and presentation of a rare tumor We present a case of gastric hibernoma, an unusual tumor with a location novel to the literature. A 39-year-old female pre- sented with one year of upper gastrointestinal bleeding and dysphagia. Gastroenterology performed an esophagogastroduo- denoscopy with ultrasound and identified a 6 cm mass within the muscularis propria of the antrum. Computed tomography demonstrated a 9.7 × 7.8 × 4.8 cm heterogeneous antral mass with internal septa. A distal gastrectomy with Bilroth I gastro- duodenostomy was performed with 4 cm proximal and 2 cm distal margins. Excision was appropriate to make the diagnosis, exclude malignancy, and remove a symptomatic mass. Hibernoma was confirmed by histopathology. These are rare tumors of brown fat named for their resemblance to the thermogenic tissue found in hibernating animals. They typically present as a slowly enlarging mass of the thigh or shoulder. To our knowledge, this is the first presentation of a hibernoma as a sub- mucosal gastric mass. endoscopy with mucosal biopsy of a large antral mass. She declined INTRODUCTION endoscopic ultrasound (EUS) and was briefly lost to follow up over Upper gastrointestinal (GI) bleeding is a common presentation the course of a pregnancy. She returned to seek care for ongoing requiring hospitalization; with ~100 per 100 000 hospitalizations anemia and new epigastric pain. Esophagogastroduodenoscopy annually in the USA [1]. The most common etiology of upper GI (EGD) with EUS was performed identifying a 6 cm mass within the bleeds in the USA is ulcerative disease. GI neoplasia rarely presents muscularis propria of the antrum. Based on these findings, she as upper GI bleeding and even more uncommon are fat-derived was referred to a surgeon and underwent pre-operative computed gastric tumors [2]. Here, we present a rare cause of an upper GI tomography (CT) of the abdomen and pelvis which demonstrated bleedand araretumor with alocation novel to the literature. a9.7 × 7.8 × 4.8 cm , heterogeneous mass with internal septa adherent to the posterior wall of the gastric antrum (Figs 1 and 2). She was lost to follow up a second time before presenting to CASE REPORT our emergency department with complaints of melena. Repeat A 39-year-old female presented to her primary care physician EGD was notable for mucosal ulceration overlying the mass. with ~1 year of upper GI bleeding. She was a recent Ukrainian Biopsy results were consistent with adipose tissue. With lipo- immigrant and had not undergone previous evaluation. She sarcoma in the differential and the symptomatic nature of the was referred to a gastroenterologist and underwent upper mass, surgery was offered. Received: February 15, 2018. Accepted: May 14, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 S.P. Smith et al. Figure 2: Gross pathology: partial gastrectomy specimen composed of distal portion of stomach and duodenum. Adipocytic neoplasm encapsulated and well circumscribed within the submucosa of the antrum. No gross or micro- scopic evidence of tumor necrosis. The tumor does not extend past the muscu- laris propria. Figure 1: Computer tomography demonstrating a large (9.7 × 7.8 × 4.8 cm ) mur- al based mass extending along the posterior wall of the lesser curve of the stomach, effacing the lumen. (A) Coronal. (B) Axial. The mass contains predom- inantly fat signal with areas of hazy internal enhancement. No pathological lymphadenopathy was noted. She ultimately underwent a laparoscopic exploration with plans for resection of this mass. This was converted to an open procedure due to the size of the mass and significant adhesion of the posterior gastric wall to the lesser sac. A distal gastrec- tomy was performed and 4 cm proximal and 2 cm distal mar- gins were achieved. She was reconstructed with a Bilroth I gastroduodenostomy. Her post-operative course was uneventful. Nutrition was provided enterally via a jejunostomy tube placed at the time of surgery and then advanced after a contrast study was completed without evidence of a leak. Following this she tol- erated a general diet. She was discharged home on post- Figure 3: Histopathology of gastrectomy specimen (×200): Brown fat cells (circle) and scattered immature adipocytes (square) intermixed with Lipoma-like adi- operative day eight. pocytes (arrow), the later including. No mitotic figures or nuclear hyperchroma- Her final pathology was notable for a well circumscribed 10 3 sia identified. × 7 × 5cm yellow-orange mass within the wall of the antrum (Fig. 3). There was evidence of ulceration of the overlying histology and location, a second opinion was obtained and the mucosa, but no hemorrhage or necrosis within the mass itself. Histologically, the mass was composed of lipoma-like adipo- unusual diagnosis of a hibernoma was confirmed. cytes (large, univacuolated cells with eccentric nuclei) inter- mixed with immature adipocytes (mutivacuolated cells with DISCUSSION bland central nucleoli) and granular eosinophilic brown fat cells (Fig. 3). FISH MDM2 amplification testing was negative, ruling Hibernomas are rare tumors of brown fat initially described by out well differentiated liposarcoma. Given this unusual Merkel in 1906 for their resemblance to the thermogenic tissue Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Gastric hibernoma: a novel location and presentation 3 found in hibernating animals [3]. In humans, brown fat is pre- margins were obtained as a precautionary measure. Hibernomas sent in the fetus and newborn with the amount decreasing do not recur when completely excised and can usually be enu- with age. It is widely distributed, with adult rests seen in the cleated from the surrounding tissue [4, 5]. Excision was appropri- neck, axilla, mediastinum and inter-scapular regions [3–5]. ate to confirm the diagnosis, exclude malignancy and remove a Hibernomas are generally seen between the ages of 20–40 years symptomatic rare tumor in an unusual location. [3–6]. They are believed to have a female predominance, although this has been called into question by the Armed Forces Pathology Institute study of hibernomas which showed CONFLICT OF INTEREST STATEMENT a higher male occurrence (58%) [5]. Hibernomas are found None declared. throughout the body, most commonly in the thigh, shoulder, back and neck [5]. Hibernomas typically present as a slow growing palpable REFERENCES mass or with pain secondary to compression of surrounding structures. They are mobile and are found intramuscularly or 1. Longstreth GF. Epidemiology of hospitalization for acute subcutaneously [3–6]. CT imaging of these tumors demonstrates gastrointestinal hemorrhage: a population based study. Am J heterogenous, hypodense lesions. On T1 weighted MRI, hiberno- Gastroenteol 1995;90:206–10. mas are hyperintense and similarly heterogenous. These find- 2. Savides TJ, Jensen DM, Cohen J, Randall GM, Kovacs TO, ings, paired with the occasional intramuscular position of Pelayo E, et al. Severe upper gastrointestinal tumor bleeding: hibernomas requires histologic evaluation to rule out liposarco- endoscopic findings, treatment, and outcome. Endoscopy ma [3, 4]. Hibernomas are typically Flurodeoxyglucose (FDG) avid; 1996;28:244–8. consistent with their metabolic role in heat production, but PET 3. DeRosa DC, Lim RB, Lin-hurtubise K, Johnson EA. Symptomatic scan cannot safely distinguish these tumors from malignant hibernoma: a rare soft tissue tumor. Hawaii J Med Public Health liposarcomas [6, 7]. On gross pathology, hibernomas are encap- 2012;71:342–5. sulated and appear dark yellow to brown secondary to the higher 4. Lath N, Familua O, Adu A, Oluwole S. Massive abdominal number of mitochondria in these tumors [3, 4, 8]. Histologically, wall hibernoma: case report and literature review of a rare hibernomas are characterized by the presence of multi-vacuolated soft-tissue tumor. J Natl Med Assoc 2017;103:372–4. brown fat cells with eosinophilic cytoplasm and a small, central 5. Furlong MA, Smith JCF, Miettinen M. The morphologic spec- nucleus in the setting of normal adipose tissue (characterized by trum of hibernoma a clinicopathologic study of 170 cases. univaculated cells with an eccentric nucleus) [3, 5]. There are four Am J Surg Pathol 2001;25:809–14. histologic variants of hibernomas including typical (the most com- 6. Kamata Y, Kikuta K, Susa M, Nishimoto K, Sasaki A, mon), myxoid, lipoma-like and spindle cell, these are described Kameyama K, et al. Hibernoma showing high uniform accu- separately but behave similarly [5]. mulation on an F-fluorodeoxyglucose positron emission To our knowledge, this is the first presentation of a hibernoma tomography scan: a case report. Case Rep Oncol 2016;9:427–31. as a submucosal gastric mass. The differential for gastric mass is 7. Park JH, Ogura K, Fujiwara T, Nagano A, Numoto K, Terauchi broad ranging from adenocarcinoma to GI stromal tumors to T, et al. Case report the values and limitations of FDG-PET/ purely benign tumors like lipomas and leiomyomas. In this CT for diagnosis of hibernoma. Case Rep Orthop 2015;2015: patient’s case, the presentation of GI bleeding was non-specific, 958690. but biopsy identifying lipomatous tissue narrowed the differential 8. Worsey J, Mcguirt W, Carrau RL, Peitzman AB. Hibernoma of but did include liposarcoma. In this case, the diagnosis of hiberno- the neck: a rare cause of neck mass. Am J Otolaryngol 1994;15: ma was not apparent by histology and therefore grossly negative 152–4. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Surgical Case Reports Oxford University Press

Gastric hibernoma: a novel location and presentation of a rare tumor

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Abstract

We present a case of gastric hibernoma, an unusual tumor with a location novel to the literature. A 39-year-old female pre- sented with one year of upper gastrointestinal bleeding and dysphagia. Gastroenterology performed an esophagogastroduo- denoscopy with ultrasound and identified a 6 cm mass within the muscularis propria of the antrum. Computed tomography demonstrated a 9.7 × 7.8 × 4.8 cm heterogeneous antral mass with internal septa. A distal gastrectomy with Bilroth I gastro- duodenostomy was performed with 4 cm proximal and 2 cm distal margins. Excision was appropriate to make the diagnosis, exclude malignancy, and remove a symptomatic mass. Hibernoma was confirmed by histopathology. These are rare tumors of brown fat named for their resemblance to the thermogenic tissue found in hibernating animals. They typically present as a slowly enlarging mass of the thigh or shoulder. To our knowledge, this is the first presentation of a hibernoma as a sub- mucosal gastric mass. endoscopy with mucosal biopsy of a large antral mass. She declined INTRODUCTION endoscopic ultrasound (EUS) and was briefly lost to follow up over Upper gastrointestinal (GI) bleeding is a common presentation the course of a pregnancy. She returned to seek care for ongoing requiring hospitalization; with ~100 per 100 000 hospitalizations anemia and new epigastric pain. Esophagogastroduodenoscopy annually in the USA [1]. The most common etiology of upper GI (EGD) with EUS was performed identifying a 6 cm mass within the bleeds in the USA is ulcerative disease. GI neoplasia rarely presents muscularis propria of the antrum. Based on these findings, she as upper GI bleeding and even more uncommon are fat-derived was referred to a surgeon and underwent pre-operative computed gastric tumors [2]. Here, we present a rare cause of an upper GI tomography (CT) of the abdomen and pelvis which demonstrated bleedand araretumor with alocation novel to the literature. a9.7 × 7.8 × 4.8 cm , heterogeneous mass with internal septa adherent to the posterior wall of the gastric antrum (Figs 1 and 2). She was lost to follow up a second time before presenting to CASE REPORT our emergency department with complaints of melena. Repeat A 39-year-old female presented to her primary care physician EGD was notable for mucosal ulceration overlying the mass. with ~1 year of upper GI bleeding. She was a recent Ukrainian Biopsy results were consistent with adipose tissue. With lipo- immigrant and had not undergone previous evaluation. She sarcoma in the differential and the symptomatic nature of the was referred to a gastroenterologist and underwent upper mass, surgery was offered. Received: February 15, 2018. Accepted: May 14, 2018 Published by Oxford University Press and JSCR Publishing Ltd. All rights reserved. © The Author(s) 2018. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/ licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018 2 S.P. Smith et al. Figure 2: Gross pathology: partial gastrectomy specimen composed of distal portion of stomach and duodenum. Adipocytic neoplasm encapsulated and well circumscribed within the submucosa of the antrum. No gross or micro- scopic evidence of tumor necrosis. The tumor does not extend past the muscu- laris propria. Figure 1: Computer tomography demonstrating a large (9.7 × 7.8 × 4.8 cm ) mur- al based mass extending along the posterior wall of the lesser curve of the stomach, effacing the lumen. (A) Coronal. (B) Axial. The mass contains predom- inantly fat signal with areas of hazy internal enhancement. No pathological lymphadenopathy was noted. She ultimately underwent a laparoscopic exploration with plans for resection of this mass. This was converted to an open procedure due to the size of the mass and significant adhesion of the posterior gastric wall to the lesser sac. A distal gastrec- tomy was performed and 4 cm proximal and 2 cm distal mar- gins were achieved. She was reconstructed with a Bilroth I gastroduodenostomy. Her post-operative course was uneventful. Nutrition was provided enterally via a jejunostomy tube placed at the time of surgery and then advanced after a contrast study was completed without evidence of a leak. Following this she tol- erated a general diet. She was discharged home on post- Figure 3: Histopathology of gastrectomy specimen (×200): Brown fat cells (circle) and scattered immature adipocytes (square) intermixed with Lipoma-like adi- operative day eight. pocytes (arrow), the later including. No mitotic figures or nuclear hyperchroma- Her final pathology was notable for a well circumscribed 10 3 sia identified. × 7 × 5cm yellow-orange mass within the wall of the antrum (Fig. 3). There was evidence of ulceration of the overlying histology and location, a second opinion was obtained and the mucosa, but no hemorrhage or necrosis within the mass itself. Histologically, the mass was composed of lipoma-like adipo- unusual diagnosis of a hibernoma was confirmed. cytes (large, univacuolated cells with eccentric nuclei) inter- mixed with immature adipocytes (mutivacuolated cells with DISCUSSION bland central nucleoli) and granular eosinophilic brown fat cells (Fig. 3). FISH MDM2 amplification testing was negative, ruling Hibernomas are rare tumors of brown fat initially described by out well differentiated liposarcoma. Given this unusual Merkel in 1906 for their resemblance to the thermogenic tissue Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018 Gastric hibernoma: a novel location and presentation 3 found in hibernating animals [3]. In humans, brown fat is pre- margins were obtained as a precautionary measure. Hibernomas sent in the fetus and newborn with the amount decreasing do not recur when completely excised and can usually be enu- with age. It is widely distributed, with adult rests seen in the cleated from the surrounding tissue [4, 5]. Excision was appropri- neck, axilla, mediastinum and inter-scapular regions [3–5]. ate to confirm the diagnosis, exclude malignancy and remove a Hibernomas are generally seen between the ages of 20–40 years symptomatic rare tumor in an unusual location. [3–6]. They are believed to have a female predominance, although this has been called into question by the Armed Forces Pathology Institute study of hibernomas which showed CONFLICT OF INTEREST STATEMENT a higher male occurrence (58%) [5]. Hibernomas are found None declared. throughout the body, most commonly in the thigh, shoulder, back and neck [5]. Hibernomas typically present as a slow growing palpable REFERENCES mass or with pain secondary to compression of surrounding structures. They are mobile and are found intramuscularly or 1. Longstreth GF. Epidemiology of hospitalization for acute subcutaneously [3–6]. CT imaging of these tumors demonstrates gastrointestinal hemorrhage: a population based study. Am J heterogenous, hypodense lesions. On T1 weighted MRI, hiberno- Gastroenteol 1995;90:206–10. mas are hyperintense and similarly heterogenous. These find- 2. Savides TJ, Jensen DM, Cohen J, Randall GM, Kovacs TO, ings, paired with the occasional intramuscular position of Pelayo E, et al. Severe upper gastrointestinal tumor bleeding: hibernomas requires histologic evaluation to rule out liposarco- endoscopic findings, treatment, and outcome. Endoscopy ma [3, 4]. Hibernomas are typically Flurodeoxyglucose (FDG) avid; 1996;28:244–8. consistent with their metabolic role in heat production, but PET 3. DeRosa DC, Lim RB, Lin-hurtubise K, Johnson EA. Symptomatic scan cannot safely distinguish these tumors from malignant hibernoma: a rare soft tissue tumor. Hawaii J Med Public Health liposarcomas [6, 7]. On gross pathology, hibernomas are encap- 2012;71:342–5. sulated and appear dark yellow to brown secondary to the higher 4. Lath N, Familua O, Adu A, Oluwole S. Massive abdominal number of mitochondria in these tumors [3, 4, 8]. Histologically, wall hibernoma: case report and literature review of a rare hibernomas are characterized by the presence of multi-vacuolated soft-tissue tumor. J Natl Med Assoc 2017;103:372–4. brown fat cells with eosinophilic cytoplasm and a small, central 5. Furlong MA, Smith JCF, Miettinen M. The morphologic spec- nucleus in the setting of normal adipose tissue (characterized by trum of hibernoma a clinicopathologic study of 170 cases. univaculated cells with an eccentric nucleus) [3, 5]. There are four Am J Surg Pathol 2001;25:809–14. histologic variants of hibernomas including typical (the most com- 6. Kamata Y, Kikuta K, Susa M, Nishimoto K, Sasaki A, mon), myxoid, lipoma-like and spindle cell, these are described Kameyama K, et al. Hibernoma showing high uniform accu- separately but behave similarly [5]. mulation on an F-fluorodeoxyglucose positron emission To our knowledge, this is the first presentation of a hibernoma tomography scan: a case report. Case Rep Oncol 2016;9:427–31. as a submucosal gastric mass. The differential for gastric mass is 7. Park JH, Ogura K, Fujiwara T, Nagano A, Numoto K, Terauchi broad ranging from adenocarcinoma to GI stromal tumors to T, et al. Case report the values and limitations of FDG-PET/ purely benign tumors like lipomas and leiomyomas. In this CT for diagnosis of hibernoma. Case Rep Orthop 2015;2015: patient’s case, the presentation of GI bleeding was non-specific, 958690. but biopsy identifying lipomatous tissue narrowed the differential 8. Worsey J, Mcguirt W, Carrau RL, Peitzman AB. Hibernoma of but did include liposarcoma. In this case, the diagnosis of hiberno- the neck: a rare cause of neck mass. Am J Otolaryngol 1994;15: ma was not apparent by histology and therefore grossly negative 152–4. Downloaded from https://academic.oup.com/jscr/article-abstract/2018/5/rjy105/5005848 by Ed 'DeepDyve' Gillespie user on 21 June 2018

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Journal of Surgical Case ReportsOxford University Press

Published: May 22, 2018

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