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Hickson CJ, Ahmed O, Laycock J, Hone R. Recurrent transient loss of airway secondary to regurgitated pedunculated hypopharyngeal liposarcoma. BMJ Case Rep 2021; 14:14/5/e240503. [PMID: 34011665 DOI: 10.1136/bcr-2020-240503] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
We describe a rare case of hypopharyngeal liposarcoma with an atypical presentation. The patient presented with a 3-month history of intermittent, transient acute airway obstruction. In between episodes, he was asymptomatic. A pedunculated tumour originating in the postcricoid region was seen to be suspended into the oesophagus and intermittently regurgitated into the larynx to cause airway obstruction. The lesion was endoscopically removed and examined histologically to confirm the diagnosis. On-going management of rare lesions such as this should be through multidisciplinary team meetings at a tertiary sarcoma centre.
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Affiliation(s)
- Craig John Hickson
- Otorhinolaryngology, Head & Neck Surgery, East Kent Hospitals University NHS Foundation Trust, Canterbury, Kent, UK
| | - Omar Ahmed
- Otorhinolaryngology, Head & Neck Surgery, East Kent Hospitals University NHS Foundation Trust, Canterbury, Kent, UK
| | - Juliet Laycock
- Otorhinolaryngology, Head & Neck Surgery, East Kent Hospitals University NHS Foundation Trust, Canterbury, Kent, UK
| | - Robert Hone
- Otorhinolaryngology, Head & Neck Surgery, East Kent Hospitals University NHS Foundation Trust, Canterbury, Kent, UK
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2
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Ye YW, Liao MY, Mou ZM, Shi XX, Xie YC. Thoracoscopic resection of a huge esophageal dedifferentiated liposarcoma: A case report. World J Clin Cases 2020; 8:1698-1704. [PMID: 32420304 PMCID: PMC7211538 DOI: 10.12998/wjcc.v8.i9.1698] [Citation(s) in RCA: 11] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/26/2020] [Revised: 04/09/2020] [Accepted: 04/16/2020] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Esophageal liposarcoma is a rare malignant tumor and an esophageal dedifferentiated liposarcoma (DDL) is extremely rare. There are no reports on the treatment of DDL by thoracoscopic surgery.
CASE SUMMARY A 38-year-old woman presented with dysphagia and dyspnea. Imaging examination showed a large mass in the posterior mediastinum. The patient also developed respiratory failure and it was unclear whether this was caused by a mass from inside or outside the esophagus. We decided to perform thoracoscopic exploration to relieve the obstruction caused by tracheal compression. The upper segment of the esophagus was split longitudinally, and most of the mass could be removed from the esophageal lumen to the thoracic cavity. The pedicle was excised by linear cutting closers under mirrors. Little residual mass was visualized by gastroscopy. The mucous and muscular layers were closed by interrupted sutures. Pathological examination showed that the mass was a DDL. The patient did not have any dysphagia or dyspnea 2 wk postoperatively and refused any further treatment. Computed tomography and esophagoscopy did not find any recurrence at up to 20 mo postoperatively.
CONCLUSION Thoracoscopy can be used to treat large esophageal masses.
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Affiliation(s)
- Yi-Wang Ye
- Department of Thoracic Surgery, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Meng-Ying Liao
- Department of Pathology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Zhi-Min Mou
- Department of Thoracic Surgery, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Xiao-Xin Shi
- Department of Pathology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
| | - Yuan-Cai Xie
- Department of Thoracic Surgery, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong Province, China
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3
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Giant Esophageal Liposarcoma in Asymptomatic Young Patient. Cir Esp 2017; 96:381-383. [PMID: 29233581 DOI: 10.1016/j.ciresp.2017.10.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/06/2017] [Revised: 09/25/2017] [Accepted: 10/22/2017] [Indexed: 11/23/2022]
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4
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Jo DH, Chon HK, Woo SH, Kim TH. Endoscopic Resection of a Giant Esophageal Lipoma Causing Sudden Choking. THE KOREAN JOURNAL OF GASTROENTEROLOGY 2017; 68:210-213. [PMID: 27780945 DOI: 10.4166/kjg.2016.68.4.210] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/03/2022]
Abstract
Most esophageal lipomas are discovered incidentally and are small and asymptomatic. However, large (>4 cm) lipomas may cause various symptoms, including dysphagia, regurgitation, or epigastric discomfort. We present a 45-year-old woman with intermittent sudden choking and globus pharyngeus. Upper gastrointestinal endoscopy and endoscopic ultrasound revealed an approximately 10.0×1.5 cm pedunculated subepithelial tumor in the upper esophagus, identified as the cause of her symptoms. A thoracic computed tomography scan revealed a fat attenuated longitudinal mass along the upper esophagus, suggestive of a lipoma. Endoscopic resection of the lesion was performed with a detachable snare to relieve her symptoms, and the pathologic findings were consistent with a lipoma.
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Affiliation(s)
- Dong Ho Jo
- Department of Internal Medicine, Wonkwang University College of Medicine, Iksan, Korea
| | - Hyung Ku Chon
- Department of Internal Medicine, Wonkwang University College of Medicine, Iksan, Korea
| | - Sun Ho Woo
- Department of Internal Medicine, Wonkwang University College of Medicine, Iksan, Korea
| | - Tae Hyeon Kim
- Department of Internal Medicine, Wonkwang University College of Medicine, Iksan, Korea
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Rare Giant Pedunculated Liposarcoma of the Hypopharynx: Case Report and Review of Literature. J Gastrointest Cancer 2017; 47:449-453. [PMID: 26449226 DOI: 10.1007/s12029-015-9767-3] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/23/2022]
Abstract
INTRODUCTION Liposarcoma is considered the most common soft tissue sarcoma in adults. It represents approximately 20 % of all mesenchymal malignancies, and most frequently it involves the retroperitoneum, trunk, and extremities. Hypopharyngeal localization of liposarcoma is extremely rare. METHODS We report a new rare case of giant pedunculated liposarcoma arising from the left antero-lateral wall of the hypopharyngeal-esophageal junction, and we present a complete review of the literature. RESULTS An 81-year-old man presented with a 5-month history of dysphagia and 30-kg weight loss, due to a giant pedunculated liposarcoma of the hypopharynx, which is resected with cervical approach. CT scan imaging revealed a 25-cm-long pedunculated heterogeneous mass with fat-like density originating from the anterior wall of hypopharyngeal-esophageal junction protruding into the lumen. The polypoid mass was identified as well as the stalk, and it was completely delivered through the cervical incision. Histopatological examination showed a dedifferentiated liposarcoma, without positive or close margins. No further chemoradiation therapy was performed due to the patient's age, comorbidities, negative margins, and absence of distant metastases. Dysphagia solved 1 month after surgery. No evidence of tumor recurrence was seen in the 12 months following surgery. CONCLUSIONS When base of the tumor is located in hypopharynx or cervical portion of the esophagus, and tumor is not aggressive, cervical approach is better, irrespective of the tumor size, except for cases in which an endoscopic approach is feasible.
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6
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Riva G, Sensini M, Corvino A, Garzaro M, Pecorari G. Liposarcoma of Hypopharynx and Esophagus: a Unique Entity? J Gastrointest Cancer 2017; 47:135-42. [PMID: 26875081 DOI: 10.1007/s12029-016-9808-6] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
Abstract
PURPOSE Liposarcoma is the most common soft tissue sarcoma in adults. It represents approximately 20 % of all mesenchymal malignancies. It most frequently involves retroperitoneum, trunk, and extremities. Hypopharyngeal and esophageal localization of liposarcoma is extremely rare. METHODS We performed a systematic review of literature and reported 26 and 33 cases of hypopharyngeal and esophageal liposarcoma. We analyzed natural history, imaging features, histology, treatment, and prognosis, with a specific focus to similarities and differences between tumors of hypopharynx and esophagus. RESULTS Hypopharyngeal and esophageal liposarcomas have more similarities than differences. Incidence has a peak at 6th and 7th decades. The diagnostic procedures are barium swallow, endoscopic examination, and CT/MR imaging. Well-differentiated liposarcoma represents the most frequent histological subtype. Surgical excision is the main treatment. Endoscopic resection can be useful for pedunculated tumors of hypopharynx and cervical esophagus. Differences between hypopharyngeal and esophageal liposarcoma are represented by local recurrence rate (greater for hypopharyngeal tumors), number of giant tumors, and time to recurrence (greater for esophageal tumors). Finally, liposarcomas of distal esophagus need more extended approaches. CONCLUSIONS Liposarcomas of hypopharynx and cervical esophagus could be considered a unique pathological entity, with similar features and treatment options. Survival rate is dependent on histological type and location. Local recurrence is common, especially for hypopharyngeal liposarcoma, while the risk of lymph node or distant metastasis is very low. Patients should undergo regular examinations to rule out local recurrence, also for a long time, especially for esophageal tumors.
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Affiliation(s)
- Giuseppe Riva
- 1st ENT Division, Surgical Sciences Department, University of Turin, Via Genova 3, 10126, Turin, Italy.
| | - Matteo Sensini
- 1st ENT Division, Surgical Sciences Department, University of Turin, Via Genova 3, 10126, Turin, Italy
| | - Andrea Corvino
- 1st ENT Division, Surgical Sciences Department, University of Turin, Via Genova 3, 10126, Turin, Italy
| | - Massimiliano Garzaro
- 1st ENT Division, Surgical Sciences Department, University of Turin, Via Genova 3, 10126, Turin, Italy
| | - Giancarlo Pecorari
- 1st ENT Division, Surgical Sciences Department, University of Turin, Via Genova 3, 10126, Turin, Italy
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Dowli A, Mattar A, Mashimo H, Huang Q, Cohen D, Fisichella PM, Lebenthal A. A pedunculated giant esophageal liposarcoma: a case report and literature review. J Gastrointest Surg 2014; 18:2208-13. [PMID: 25190025 DOI: 10.1007/s11605-014-2628-8] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/28/2014] [Accepted: 08/04/2014] [Indexed: 01/31/2023]
Abstract
INTRODUCTION The majority of esophageal tumors arise from the mucosal layer; only 5 % are of mesenchymal origins. Of the latter, barely 0.5 % are liposarcomas. We present a case of an esophageal liposarcoma with a review of the literature. CASE REPORT A 64-year-old male was referred with 5 years of progressive dysphagia. Preoperative evaluation initially suggested a leiomyoma. The polypoid lesion was then resected through a cervical esophagotomy, once endoscopic resection proved to be not feasible. The definitive pathologic diagnosis confirmed a well-differentiated liposarcoma. LITERATURE REVIEW Esophageal liposarcomas are very rare and only 40 such cases have been reported in the literature. Most patients were male (80 %), the median age was 62 years (range 38-83 years), and the most common symptom was dysphagia (85 %). Only in two cases was a liposarcoma detected on preoperative biopsy. The most common histological subtype was well-differentiated liposarcoma. Overall, 77.5 % of the patients were successfully treated with surgery, 20 % endoscopically, and 2.5 % were ablated with CO2 laser. CONCLUSION Esophageal liposarcoma is an extremely rare tumor. The majority of patients are males; dysphagia is the most common initial symptom, and preoperative biopsy is unreliable. Because these tumors are pedunculated, well-circumscribed, and well-differentiated, they can be safely resected locally. All patients need long-term follow-up as this disease can recur many decades after treatment.
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Affiliation(s)
- A Dowli
- Division of Thoracic Surgery, Brigham and Women's Hospital, Boston VA Health Care System, Harvard Medical School, Boston, MA, USA
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Teixeira FJR, Reis AV, Medeiros RSS, Akaishi EH, Moutinho VC, Couto Netto SD, Andrade Lima TM, Perina ALF, Ferreira FO, Mendes GO, Lallé MP, Utiyama EM. Malignant dysphagia treated by esophageal sparing surgery with good prognosis. J Gastrointest Cancer 2014; 45 Suppl 1:241-4. [PMID: 25129043 DOI: 10.1007/s12029-014-9645-4] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/13/2023]
Affiliation(s)
- F J R Teixeira
- Sarcoma and Melanoma Group-General Surgery Department, Cancer Institute-University of Sao Paulo School of Medicine, Sao Paulo, Brazil,
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9
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Abstract
The so-called "giant fibrovascular polyps" of the esophagus and hypopharynx typically present as sausage-like pedunculated structures that protrude into the lumen and cause obstructive symptoms. Most are cured by local resection but they may recur. Microscopically, they display an admixture of fibrovascular and adipose tissue that is coated by unremarkable squamous mucosa. Here, we report a case that had scattered hyperchromatic cells and lipoblasts within the adipose tissue component. In other anatomic sites similar appearing lesions have been interpreted as pedunculated liposarcomas/atypical lipomatous tumors that are more prone to local recurrences than classic giant fibrovascular polyps. Reports of dedifferentiation and metastases are lacking thus raising the possibility that the cytologic findings in such lesions are degenerative. To confirm our suspicion of liposarcomatous differentiation, we performed immunohistochemistry for MDM2 and p53, 2 markers that are known to be negative in benign lipomatous lesions and positive in well-differentiated liposarcomas/atypical lipomatous tumors. The scattered atypical hyperchromatic cells and the lipoblasts both exhibited strong nuclear staining for both markers and supported the diagnosis of pedunculated giant hypopharyngeal atypical lipomatous tumor.
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10
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Page R, Narlawar R, Holemans J, Gosney J, Warwick R, Elsayed H. Giant oesophageal liposarcoma: case report. Radiol Bras 2008. [DOI: 10.1590/s0100-39842008000600013] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/22/2022] Open
Abstract
We describe imaging findings of a oesophageal liposarcoma in a 66 year old man. The computed tomography scan was performed after a chest radiograph showed a large posterior mediastinal mass. Oesophageal liposarcomas are rare tumours. They can achieve large size before they become symptomatic. Our patient was successfully managed with complete surgical removal.
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Affiliation(s)
- Richard Page
- Liverpool Heart and Chest Hospital NHS Trust, United Kingdom
| | | | - John Holemans
- Liverpool Heart and Chest Hospital NHS Trust, United Kingdom
| | - John Gosney
- Liverpool Heart and Chest Hospital NHS Trust, United Kingdom
| | - Richard Warwick
- Liverpool Heart and Chest Hospital NHS Trust, United Kingdom
| | - Hany Elsayed
- Liverpool Heart and Chest Hospital NHS Trust, United Kingdom
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11
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Xu S, Xu Z, Hou Y, Tan Y. Primary pedunculated giant esophageal liposarcoma: case report. Dysphagia 2007; 23:327-30. [PMID: 18058173 DOI: 10.1007/s00455-007-9130-3] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/26/2007] [Accepted: 09/14/2007] [Indexed: 01/01/2023]
Abstract
Liposarcoma is one of the most common soft tissue sarcomas in adults, but the incidence of esophageal liposarcoma is extremely low. To our knowledge there are only 18 cases of esophageal liposarcoma reported in the English-language literature. Here we report a new case of primary esophageal liposarcoma in a 50-year-old male patient who complained of aggravating dysphagia with intermittent vomiting for 6 months. The tumor was located in the upper esophagus. Polypectomy was performed and histologic examination showed a well-differentiated liposarcoma. Diagnostic and therapeutic tools are discussed and the results of a literature review are discussed.
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Affiliation(s)
- Songtao Xu
- Department of Thoracic Surgery, Zhongshan Hospital, Fudan University, Shanghai, China.
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12
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Mica L, Gianom D, Bode B, Jaklin P, Hollinger A. Rare cause of dysphagy: giant polypoid esophageal well-differentiated liposarcoma. Case Rep Gastroenterol 2007; 1:7-14. [PMID: 21487465 PMCID: PMC3073781 DOI: 10.1159/000104225] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/19/2022] Open
Abstract
Liposarcoma represents one of the most frequent (10–20%) malignant mesenchymal tumors in the adult, affecting mostly the soft tissue of extremities, the trunk or the retroperitoneum. This tumor type occurs exceptionally rarely in the gastrointestinal tract with only few cases described in the literature. In this case we present a 73-year-old male patient who was admitted due to loss of weight, anorexia and postprandial emesis with dysphagy. Gastrographin esophagography failed to make precise diagnostics. CT scan of the upper gastrointestinal tract revealed a large esophageal tumor filling out the whole length of the esophagus. The tumor was removed by parasternocleidomastoidal approach with a stapler. Histopathological examination revealed a well-differentiated liposarcoma (grade I). Well-differentiated liposarcomas are characterised by amplified material of the 12q13-15 chromosomal region, present in the form of giant or ring chromosomes and leading to the overexpression of MDM2 and CDK4 genes. MDM2 and CDK4 proteins can be detected immunhistochemically, which was the case in the reported tumor. Overexpression of these proteins leads to suppression of tumor suppressor genes, leading to increased cell survival.
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Affiliation(s)
- Ladislav Mica
- Department of Surgery, University Hospital of Zürich, Zürich, Switzerland
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Maruyama K, Motoyama S, Okuyama M, Sasaki K, Sato Y, Hayashi K, Nanjo H, Ogawa JI. Cervical approach for resection of a pedunculated giant atypical lipomatous tumor of the esophagus. Surg Today 2007; 37:173-5. [PMID: 17243042 DOI: 10.1007/s00595-006-3357-8] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/15/2006] [Accepted: 07/13/2006] [Indexed: 11/26/2022]
Abstract
We describe how we removed a giant pedunculated atypical lipomatous tumor, arising in the cervical to upper thoracic esophagus and occupying a region extending from the cervical to the middle thoracic esophagus, through a cervical esophagotomy without thoracotomy or laparotomy. We suggest that if the base of the tumor is located in the cervical portion of the esophagus, and if the tumor is not aggressive, the cervical approach is best, irrespective of the size of the tumor.
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Affiliation(s)
- Kiyotomi Maruyama
- Department of Surgery, Akita University School of Medicine, 1-1-1 Hondo, Akita, 010-8543, Japan
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Liakakos TD, Troupis TG, Tzathas C, Spirou K, Nikolaou I, Ladas S, Karatzas GM. Primary liposarcoma of esophagus: A case report. World J Gastroenterol 2006; 12:1149-52. [PMID: 16534863 PMCID: PMC4087914 DOI: 10.3748/wjg.v12.i7.1149] [Citation(s) in RCA: 20] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/06/2023] Open
Abstract
Liposarcoma is the most common soft tissue sarcoma in adult life while esophageal liposarcoma is an extremely rare tumor. In the world literature, only 14 cases of esophageal liposarcomas have been described. We report a 72-year old male patient who was urgently admitted to our hospital for acute epigastric pain with a burning retrosternal sensation, persistent nausea, vomiting and dysphagia. Barium swallow, upper gastrointestinal (GI) endoscopy, esophageal manometry and CT scan, failed to accurately diagnose the lesion. After surgical resection of an esophageal polypoid tumor, the histological examination revealed a well-differentiated grade I liposarcoma. Diagnostic and therapeutic tools were discussed and the results of literature were reviewed.
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Affiliation(s)
- Theodore D Liakakos
- 3rd Department of Surgery, University of Athens, ATTIKON University General Hospital, Haidari, Athens, Greece
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Garcia M, Buitrago E, Bejarano PA, Casillas J. Large esophageal liposarcoma: a case report and review of the literature. Arch Pathol Lab Med 2004; 128:922-5. [PMID: 15270604 DOI: 10.5858/2004-128-922-lelacr] [Citation(s) in RCA: 29] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
Liposarcoma is one of the most common neoplasms of adulthood. However, it is exceedingly rare in the gastrointestinal tract. To our knowledge, only 12 cases occurring in the esophagus have been reported in the world literature to date. We report the case of a 42-year-old man with a pleomorphic liposarcoma arising in the esophageal wall. The morphologic, immunophenotypic, and ultrastructural characteristics are presented, as well as the results of literature review.
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Affiliation(s)
- Monica Garcia
- Department of Pathology, University of Miami School of Medicine, Miami, Fla 33136, USA
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