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Chen S, Zhao Y, Zhao Y. Thoracoscopic resection of a giant esophageal schwannoma: A case report and review of literature. Medicine (Baltimore) 2024; 103:e39507. [PMID: 39213227 PMCID: PMC11365663 DOI: 10.1097/md.0000000000039507] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/12/2024] [Accepted: 08/09/2024] [Indexed: 09/04/2024] Open
Abstract
INTRODUCTION Benign esophageal tumors are uncommon, accounting for approximately 2% of esophageal tumors. Esophageal schwannoma is a much rarer solid tumor with few cases reported in the literature. Open surgery is the surgical approach of choice for the treatment of esophageal tumors. With the advent of thoracoscopy, more and more countries are adopting a thoracoscopic approach to treat esophageal tumors, but there is still no clear surgical standard or modality for the treatment of esophageal tumors. PATIENT CONCERNS A 50-year-old woman was admitted to our hospital. Over the past 2 months, her clinical presentation has included progressively worse swallowing disorder and weight loss. Gastroscopy showed an elevated lesion with a smooth surface visible 18 cm out from the incisors. An electron circumferential ultrasound endoscopy showed a hemispherical bulge with a smooth surface 18 to 23 cm from the incisor; the bulge originated from the intrinsic muscular layer and showed a heterogeneous mixed moderate ultrasound with a little blood flow signal and blue-green elastography in 1 of the sections measuring approximately 4 cm × 3 cm. Chest computed tomography (CT) showed a mass-like soft tissue shadow in the upper esophagus measuring approximately 39 mm × 34 mm, with a CT The lumen was compressed and narrowed, and the lumen of the upper part of the lesion was dilated, and the adjacent trachea was compressed and displaced to the right. INTERVENTIONS After completion of the examination, assisted by artificial pneumothorax and thoracoscopic resection of esophageal masses were performed. DIAGNOSIS AND OUTCOMES Postoperative pathology report: Mesenchymal-derived tumor (esophagus), combined with immunohistochemical staining results and morphologic features supported schwannoma. The patient's postoperative course was calm. The patient's postoperative dysphagia subsided. CONCLUSION We describe a case of successful treatment of a schwannoma of the upper esophagus using artificial pneumothorax-assisted VATS. The combined use of Sox10 and S100 helps to improve the sensitivity and specificity of schwannoma diagnosis. Damage to the esophageal lining was avoided by mixed thoracoscopic and endoscopic exploration. This approach can also be applied to benign esophageal tumors in the thoracic and subthoracic segments, leading to better minimally invasive results.
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Affiliation(s)
- Shu Chen
- Department of Thoracic Surgery, the Second Hospital of Jilin University, Jilin, China
| | - Yixuan Zhao
- Department of Ultrasound Medicine, the Second Hospital of Jilin University, Jilin, China
| | - Yinghao Zhao
- Department of Thoracic Surgery, the Second Hospital of Jilin University, Jilin, China
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Fritchie KJ, Dermawan JK, Astbury C, Sharma A, Bakhshwin A, Fuller L, Agrawal S, Wieland CN, Greipp PT, Azzato EM, Folpe AL, Billings SD. Novel NONO::TFE3 fusion and ALK co-expression identified in a subset of cutaneous microcystic/reticular schwannoma. Virchows Arch 2023; 483:237-243. [PMID: 37468653 DOI: 10.1007/s00428-023-03605-7] [Citation(s) in RCA: 6] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/25/2023] [Revised: 07/09/2023] [Accepted: 07/12/2023] [Indexed: 07/21/2023]
Abstract
Microcystic/reticular schwannoma (MRS) is a benign variant of schwannoma with a predilection for the gastrointestinal tract and skin. To date, genetic characterization of this tumor is limited. Prompted by the identification of TFE3::NONO fusion and ALK overexpression in an index case of MRS, a cohort of tumors was collected from institutional and consultation archives of two institutions. Next-generation sequencing (NGS), TFE3 fluorescence in situ hybridization (FISH), and TFE3 and ALK immunohistochemistry were performed, while clinicopathologic variables were documented. Eighteen MRS cases were identified (35 to 85 years) arising in the skin (n=8), gastrointestinal tract (n=5), adrenal gland (n=3), abdominal wall (n=1), and unknown site (n=1). Tumors showed a circumscribed to multinodular to plexiform low-power architecture with variable amounts of microcystic/reticular and solid schwannian components. Mitotic figures were scarce (0-1/10 HPFs), and atypia was absent. S100 protein and/or SOX10 immunoreactivity was noted in the microcystic/reticular and schwannian areas of all cases. NGS performed on two cutaneous tumors yielded NONO exon 12 fusion with TFE3 exon 4, and these lesions also showed HMB45 and ALK expression. Two additional cases showed ALK expression (1 weak), while a third was positive for TFE3, but these cases failed to show ALK or TFE3 rearrangement by FISH/NGS. There were no morphologic variables that correlated with the presence of NONO::TFE3. We identified a subset of microcystic/reticular schwannomas with NONO::TFE3 fusions and ALK co-expression, adding to the cohort of mesenchymal neoplasms that show ALK overexpression without rearrangement of the ALK gene.
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Affiliation(s)
- Karen J Fritchie
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA.
| | - Josephine K Dermawan
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Caroline Astbury
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Anurag Sharma
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Ahmed Bakhshwin
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Lanisha Fuller
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Shruti Agrawal
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, 55905, USA
- Department of Dermatology, Mayo Clinic, Rochester, MN, 55905, USA
| | - Carilyn N Wieland
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, 55905, USA
- Department of Dermatology, Mayo Clinic, Rochester, MN, 55905, USA
| | - Patricia T Greipp
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, 55905, USA
| | - Elizabeth M Azzato
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
| | - Andrew L Folpe
- Department of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, MN, 55905, USA
| | - Steven D Billings
- Department of Pathology, Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, 2119 E 93rd Street, L15, Cleveland, OH, 44195, USA
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Mu YZ, Zhang Q, Zhao J, Liu Y, Kong LW, Ding ZX. Total removal of a large esophageal schwannoma by submucosal tunneling endoscopic resection: A case report and review of literature. World J Clin Cases 2023; 11:2510-2520. [PMID: 37123315 PMCID: PMC10130983 DOI: 10.12998/wjcc.v11.i11.2510] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/23/2022] [Revised: 01/14/2023] [Accepted: 03/21/2023] [Indexed: 04/06/2023] Open
Abstract
BACKGROUND Primary schwannoma is a rare submucosal tumor of the esophagus, which is most often benign, and surgery is the only effective treatment. So far, only a few cases have been reported. Herein, we reported a single case diagnosed with primary esophageal schwannoma that was totally removed by submucosal tunneling endoscopic resection (STER).
CASE SUMMARY A 62-year-old man presented to the hospital with a history of resection of a malignant gastric tumor and mild dysphagia. Endoscopic examination revealed a large submucosal elevated lesion in the esophagus 25-30 cm from the incisors. Endoscopic ultrasonography detected a 45 mm × 35 mm × 31 mm hypoechoic lesion; chest computed tomography showed a mass of approximately 55 mm × 35 mm × 29 mm. A preliminary examination showed features suggestive of a stromal tumor. Pathological findings indicated esophageal schwannoma. Next, STER alone was performed to completely resect the mass, and the patient recovered well post-surgery. Afterward, the patient was discharged and showed no tumor recurrence at 33 mo of follow-up.
CONCLUSION Endoscopic resection is still an effective treatment for large esophageal schwannomas (> 30 mm) under meticulous morphological evaluation.
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Affiliation(s)
- Yu-Zhu Mu
- Department of Radiology, The Fourth School of Clinical Medicine, Zhejiang Chinese Medical University, Hangzhou 310006, Zhejiang Province, China
- Department of Radiology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Qi Zhang
- Department of Radiology, The First Affiliated Hospital of Zhejiang Chinese Medical University, Hangzhou 310006, Zhejiang Province, China
| | - Jing Zhao
- Department of Gastroenterology, The First Affiliated Hospital of Zhejiang Chinese Medical University, Hangzhou 310006, Zhejiang Province, China
| | - Yan Liu
- Department of Pathology, The First Affiliated Hospital of Zhejiang Chinese Medical University, Hangzhou 310006, Zhejiang Province, China
| | - Ling-Wei Kong
- Department of Radiology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
| | - Zhong-Xiang Ding
- Department of Radiology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
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Bianchi R, Fraternali Orcioni G, Spina B, Vellone VG, Ravetti JL, Gaggero G. A microcystic/reticular schwannoma in an unusual site: description of a retroperitoneal location and review of the literature. Pathologica 2022; 114:159-163. [PMID: 35481567 PMCID: PMC9248245 DOI: 10.32074/1591-951x-266] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/11/2021] [Accepted: 07/06/2021] [Indexed: 11/30/2022] Open
Abstract
Microcystic/reticular (MRV) schwannoma has been described since 2008, but remains a rarely encountered entity. MRV has a predilection for visceral locations and has variable histologic appareances. Given its rarity and anatomic variability, this entity could raise differential diagnostic issues with other tumours and malignancies. We describe the case of a 69-year-old male followed at IRCCS Ospedale Policlinico San Martino of Genoa for his previous history of non-Hodgkin lymphoma. A para-aortic mass was discovered during follow-up, which -due to its stability, also after chemotherapy- had been hypothesized to be a non-lymphomatous lesion; given the dimensions and the site, the mass was removed. Histological evaluation showed a nodule limited by a slight fibrous capsule and characterized by a proliferation of medium-sized fusiform cells, with elongated nuclei and scarce eosinophilic cytoplasm. Given the lack of malignant signs and the strong expression of protein S-100, a diagnosis of mesenchymal neoplasia with expression of neural markers compatible with reticular schwannoma was made. The neoplasm has not recurred since its removal. The case we present is, at our best knowledge, the first described in the retroperitoneum, a site where the exclusion of other mesenchymal malignancies is mandatory. The rarity and variability of presentations could create problems of differential diagnosis both with mucinous-producing carcinomas or with other soft tissue tumours, with myxoid or reticular structure. The description of this case could help raise information on this rare neoplasm and help distinguish it from other malignancies, especially in unusual sites.
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Affiliation(s)
- Rita Bianchi
- Unit of Pathology, Ospedale di Sestri Levante, ASL4, Genoa, Italy
| | | | - Bruno Spina
- Unit of Pathology, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | | | - Jean Luis Ravetti
- Unit of Pathology, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | - Gabriele Gaggero
- Unit of Pathology, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
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Hashizume R, Matsuda S, Nagai M, Hirata K, Imai H, Kushima R. An unusual presentation of chordoma as a pyloric ring submucosal tumor: The first case report of a primary gastrointestinal lesion in humans. Int J Surg Case Rep 2022; 94:107032. [PMID: 35398781 PMCID: PMC9006247 DOI: 10.1016/j.ijscr.2022.107032] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/18/2022] [Revised: 04/01/2022] [Accepted: 04/02/2022] [Indexed: 11/23/2022] Open
Abstract
Introduction and importance Chordomas are rare malignant bone neoplasms that are presumed to arise from chordal remnants in the fetal stage and typically occur along the axial skeleton. The extra-skeletal chordomas reported to date include soft tissue of the extremities and nasopharynx. Chordoma arising from the gastrointestinal wall has not been previously described. Case presentation We report on a 42-year-old man with primary chordoma presenting as a gastroduodenal submucosal tumor centered on the pyloric ring. The patient was consistently asymptomatic, and the tumor was an incidental finding. However, during a follow-up at approximately 1.6 years, an increase in tumor size was identified on computed tomography (CT), and surgical resection was performed without a definite pathologic diagnosis. The patient was successfully treated with distal gastrectomy, and the histological diagnosis was a conventional chordoma. The diagnosis was confirmed via immunohistochemical staining for brachyury, pan-cytokeratin, S-100, and SOX9. Postoperative CT and magnetic resonance imaging revealed no recurrence or metastasis during the 1.5-year follow-up period. Clinical discussion Primary chordomas of the digestive tract are rare. Embryologic development of the notochord does not explain the existence of remnants in the gastrointestinal wall. Moreover, notochordal remnants, as precursors of chordoma, were not identified in the current case. The gastroduodenal chordoma may not have originated from embryonic notochordal remnants but through aberrant brachyury activation without a notochordal precursor. Conclusion We report the first case of primary gastrointestinal chordoma in humans. The tumor was completely removed surgically, without postoperative recurrence.
This is a case of primary gastrointestinal submucosal chordoma in a 42-year-old man Chordoma is a rare bony tumor that most frequently affects the axial skeleton Extra-skeletal chordomas are extremely rare in any species This is the first report of primary chordoma of the digestive tract in humans
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Affiliation(s)
- Ryotaro Hashizume
- Department of Pathology and Matrix Biology, Mie University Graduate School of Medicine, Tsu, Mie 514-8507, Japan; Department of Genomic Medicine, Mie University Hospital, Tsu, Mie 514-8507, Japan; Department of Surgery, Nagai Hospital, Tsu, Mie 514-8508, Japan.
| | | | - Moritaka Nagai
- Department of Surgery, Nagai Hospital, Tsu, Mie 514-8508, Japan
| | - Kazuki Hirata
- Department of Pathology Laboratory, Nagai Hospital, Tsu, Mie 514-8508, Japan
| | - Hiroshi Imai
- Department of Genomic Medicine, Mie University Hospital, Tsu, Mie 514-8507, Japan; Pathology Division, Mie University Hospital, Tsu, Mie 514-8508, Japan
| | - Ryoji Kushima
- Department of Pathology, Undergraduate School of Medicine, Shiga University of Medical Science, Otsu, Shiga 520-2192, Japan; Pathology Division, National Cancer Center Hospital, Tokyo 104-0045, Japan
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Zhao X, Zhou X, Chen X, Pan J, Li B. Primary microcystic/reticular schwannoma of the frontal bone: illustrative case. JOURNAL OF NEUROSURGERY: CASE LESSONS 2021; 1:CASE21175. [PMID: 35855082 PMCID: PMC9245779 DOI: 10.3171/case21175] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 03/23/2021] [Accepted: 04/02/2021] [Indexed: 12/03/2022]
Abstract
BACKGROUND To the best of the authors’ knowledge, this is the first case of microcystic/reticular schwannoma arising in the frontal bone. OBSERVATIONS An 18-year-old man presented to the authors’ orthopedic clinic with a complaint of a progressively enlarging, painless mass in the frontal bone. It showed significant hyperintensity on T2-weighted imaging with progressive enhancement. Computed tomography combined with three-dimensional reconstruction showed expansive bone destruction with a soft tissue mass in the left side of the frontal bone, without calcification inside or a sclerotic margin around it. The mass was a microcystic/reticular schwannoma as confirmed by surgical pathology. LESSONS The authors report a rare case of a microcystic/reticular schwannoma arising in the frontal bone, with relatively comprehensive imaging data that enabled them to learn more about this tumor.
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Affiliation(s)
| | - Xinmu Zhou
- Pathology, The Fifth Affiliated Hospital of Wenzhou Medical University, Lishui City, Zhejiang Province, China
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Clinicopathological features of esophageal schwannomas in mainland China: systematic review of the literature. Int J Clin Oncol 2020; 26:284-295. [PMID: 33216242 DOI: 10.1007/s10147-020-01809-4] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/26/2019] [Accepted: 10/09/2020] [Indexed: 12/20/2022]
Abstract
OBJECTIVE Esophageal schwannoma (ES) are rare and mostly benign neurogenic tumors. The clinical misdiagnosis rate of it is high. In this study, the clinicopathologic features of ES in mainland China were studied to better understand the disease and improve the diagnosis and treatment rate. METHODS A systematic review was conducted in accordance with PRISMA guidelines. The keywords "esophageal schwannoma", "esophageal neurinoma" and "esophageal neurilemoma" were searched for databases such as Pubmed, EMbase, Wanfang Database and Chinese National Knowledge Infrastructure. The search time frame for database was until July 2019. Combined with our patient, the clinicopathological data and the diagnosis and treatment of ES were summarized. RESULTS ES occurs in the upper part of the mediastinum and in the thoracic esophagus in most patients in the neck, upper and middle segments. CT and PET/CT examinations can be used for diagnosis, but the differentiation value of both benign and malignant ES is similar. The histopathological findings of forceps biopsy specimens are often difficult to diagnose, and deep tissue biopsies may increase pathological accuracy. EUS-FNA is also recommended for ES diagnosis, but it may also be misdiagnosed. Pathological features include a fusiform arrangement in a palisade-like structure or a tumor cell arranged in a network to form a loose structure. ES characteristic immunohistochemistry results showed that S-100 protein has strong immunological activity. CONCLUSION The definitive diagnosis requires immunohistochemistry, especially immunological reaction with S-100 protein. The appropriate treatment plan should be selected according to the diameter of the lesion. The overall prognosis of ES is good, but attention should be paid to follow-up.
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Souza LCDA, Pinto TDA, Cavalcanti HODF, Rezende AR, Nicoletti ALA, Leão CM, Cunha VC. Esophageal schwannoma: Case report and epidemiological, clinical, surgical and immunopathological analysis. Int J Surg Case Rep 2019; 55:69-75. [PMID: 30710876 PMCID: PMC6357786 DOI: 10.1016/j.ijscr.2018.10.084] [Citation(s) in RCA: 14] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/22/2018] [Accepted: 10/31/2018] [Indexed: 12/11/2022] Open
Abstract
INTRODUCTION Schwannoma is a tumor of the peripheral nervous system originated in the Schwann cells of the neural sheath. PRESENTATION OF CASE A 43-years-old male complained of odynophagia, dysphagia and hemoptysis. The upper gastrointestinal endoscopy showed a smooth elevated lesion, 20 cm from the incisor teeth, occupying the entire lumen of the esophagus. The chest computed tomography (CT) scan showed a lesion of 7 cm and superior mediastinal, lower paraesophageal and cardiac enlarged lymph nodes. A posterolateral thoracotomy was performed with total esophagectomy without intraoperative complications. The anatomopathological analysis revealed fusocellular mesenchymal neoplasia of low malignancy potential. The immunohistochemical study showed positivity for S-100 protein and KI67 antibodies and absence of staining for CD117, CD34, ALK protein, SMA and Desmin. Thus, the morphological and immunohistochemical findings pointed to the diagnosis of esophageal Schwannoma. DISCUSSION Although rare and indolent, Schwannoma occurs in the peripheral nervous system, being uncommon in the esophagus. CONCLUSION The immunohistochemical study is essential for the diagnosis, which is based on the positivity for S-100 protein and absence of staining for CD34 and CD117.
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Affiliation(s)
- Luiz Carlos de Araújo Souza
- Undergraduates of Medicine in the University Center of Brasilia (UniCEUB) and Researchers in the Department of Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil.
| | - Thiago David Alves Pinto
- Physician Anatomopathologist of Diagnose laboratory and Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil
| | | | - Alexandre Rezende Rezende
- Physician Anatomopathologist of Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil
| | - Ana Luiza Alves Nicoletti
- Undergraduates of Medicine in the University Center of Brasilia (UniCEUB) and Researchers in the Department of Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil
| | - Cinthia Mares Leão
- Undergraduates of Medicine in the University Center of Brasilia (UniCEUB) and Researchers in the Department of Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil
| | - Vinícius Carvalhêdo Cunha
- Undergraduates of Medicine in the University Center of Brasilia (UniCEUB) and Researchers in the Department of Cytopathology and Pathological Anatomy of the Base Institute of the Federal District (NUCAP-IHBDF), Brasilia, Brazil
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Xie J, Wang C, Wang H. Microcystic/reticular schwannoma of the adrenal gland: a case report and literature review. HUMAN PATHOLOGY: CASE REPORTS 2018. [DOI: 10.1016/j.ehpc.2018.03.001] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022] Open
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Liu C, Yan L, Liu Q, Li J, Jin H, Wang J, Deng Y. Lumbar intraspinal microcystic/reticular schwannoma: Case report and literature review. Medicine (Baltimore) 2018; 97:e12474. [PMID: 30278533 PMCID: PMC6181603 DOI: 10.1097/md.0000000000012474] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/15/2022] Open
Abstract
RATIONALE Microcystic/reticular schwannoma (MRS) is a rare histological variant of schwannoma which was initially described in 2008 with a predilection for the visceral organs. This distinct tumor had been reported to mainly affect gastrointestinal tract, subcutaneous and soft tissue, various glands and head and neck region. However, MRS involving spine is extremely rare. PATIENT CONCERNS The authors report the first case of MRS occurring in the lumbar (L) spinal canal of a 40-year-old male who presented with continuous pain and numbness in both feet for 2.5 years. Physical examination revealed weakness of lower extremities and hyperalgesia of both feet. DIAGNOSES AND INTERVENTIONS The findings of pre-operative investigation were suspicious for either a schwannoma or a spinal meningioma. Accordingly, total laminectomy, complete tumor resection, instrumentation and spinal fusion were performed. Post-operative histopathologic examination revealed a well-encapsulated neoplasm with reticular and microcystic growth pattern. Antoni A and Antoni B regions, Verocay bodies and hyalinized blood vessels were observed. And cytologic atypia, necrosis or mitosis was absent. Immunohistochemically, the tumor cells showed strong and diffuse positivity for S-100 as well as SOX 10. Therefore, a histopathological diagnosis of MRS was finally made. OUTCOMES The patient remains well with no evidence of recurrence at a 22-month follow-up. LESSONS This is the first case of MRS which is located in the L spinal canal. Awareness of this distinctive entity is helpful in preventing diagnostic pitfalls and making correct treatment strategies.
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Affiliation(s)
- Congcong Liu
- Department of Orthopedics, The Second Xiangya Hospital, Central South University, Changsha, Hunan
| | - Lianqi Yan
- Department of Orthopedics, The Second Xiangya Hospital, Central South University, Changsha, Hunan
- Department of Orthopedics, Clinical medical college of Yangzhou University
- Orthopedics Institute, Subei People's Hospital of Jiangsu Province, Yangzhou, Jiangsu
| | - Qing Liu
- Department of Spine Surgery, The Second Xiangya Hospital, Central South University, Changsha, Hunan
| | - Jing Li
- Department of Spine Surgery, The Second Xiangya Hospital, Central South University, Changsha, Hunan
| | - Hongtao Jin
- Department of Pathology, Shen Zhen People's Hospital, Second Clinical Medical College of Jinan University, Shenzhen, Guangdong
| | - Jingcheng Wang
- Department of Orthopedics, The Second Xiangya Hospital, Central South University, Changsha, Hunan
- Department of Orthopedics, Clinical medical college of Yangzhou University
- Orthopedics Institute, Subei People's Hospital of Jiangsu Province, Yangzhou, Jiangsu
| | - Youwen Deng
- Department of Spine Surgery, The Second Xiangya Hospital, Central South University, Changsha, Hunan
- Department of Emergency Medicine, The Second Xiangya Hospital, Central South University, Changsha, Hunan, China
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Microcystic/Reticular Schwannoma of the Frontal Lobe: An Unusual Occurrence. Case Rep Pathol 2017; 2017:4728585. [PMID: 28428901 PMCID: PMC5385903 DOI: 10.1155/2017/4728585] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/02/2016] [Accepted: 02/26/2017] [Indexed: 12/14/2022] Open
Abstract
Schwannoma is a benign peripheral nerve sheath tumor that typically involves cranial nerves of the head and neck region. Intraparenchymal occurrence of this tumor is uncommon. Even rarer in this site is the microcystic/reticular pattern of schwannoma. This histologic variant, first described in 2008, has a predilection for visceral organs. Herein, we report the first case of microcystic/reticular schwannoma of the frontal lobe.
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12
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Ieni A, Barresi V, Reggiani Bonetti L, Branca G, Caruso RA, Tuccari G. Cytohistological and immunohistochemical characteristics of spindle-shaped mesenchymal neoplasms occurring in the gastrointestinal tract. Scand J Gastroenterol 2017; 52:291-299. [PMID: 27817254 DOI: 10.1080/00365521.2016.1251607] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/08/2023]
Abstract
The purpose of the present review is to analyze the cytohistological and immunohistochemical characteristics of spindle-shaped mesenchymal gastrointestinal neoplams (MGNs), a group of unusual neoplastic conditions with different biological behavior. These tumors exhibit clinical pictures strictly related to the site of origin and dimensions, even if they appear generally with an intramural localization. This latter point may suggest an useful application of endoscopic ultrasound-guided fine-needle aspiration (EUS-FNA), mainly followed by the cell-block procedure (CBP) in the differential diagnostic approach. First of all, we discuss the most common entity of MGNs represented by gastrointestinal stromal tumors (GISTs), analyzing the morphologic characteristics and stressing the strength of immunohistochemical algorithm for diagnostic purposes. Successively, we have reported the less common group of spindle-shaped MGNs comprehensive of those arising elsewhere the soft tissues, such as leiomyomas, leiomyosarcomas, schwannomas, inflammatory myofibroblastic tumor and intra-abdominal desmoid fibromatosis. Finally, very uncommon spindle-shaped MGNs, like clear cell, follicular dendritic cell, undifferentiated pleomorphic and radiation-induced sarcomas as well as spindle cell dedifferentiated liposarcomas, have been briefly mentioned.
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Affiliation(s)
- Antonio Ieni
- a Department of Human Pathology of adult and evolutive age "Gaetano Barresi" , University of Messina, A.O.U. "Policlinico G.Martino" , Messina , Italy
| | - Valeria Barresi
- a Department of Human Pathology of adult and evolutive age "Gaetano Barresi" , University of Messina, A.O.U. "Policlinico G.Martino" , Messina , Italy
| | - Luca Reggiani Bonetti
- b Department of Laboratory Integrated Activities, Anatomic Pathology and Legal Medicine , University of Modena and Reggio Emilia , Modena , Italy
| | - Giovanni Branca
- a Department of Human Pathology of adult and evolutive age "Gaetano Barresi" , University of Messina, A.O.U. "Policlinico G.Martino" , Messina , Italy
| | - Rosario Alberto Caruso
- a Department of Human Pathology of adult and evolutive age "Gaetano Barresi" , University of Messina, A.O.U. "Policlinico G.Martino" , Messina , Italy
| | - Giovanni Tuccari
- a Department of Human Pathology of adult and evolutive age "Gaetano Barresi" , University of Messina, A.O.U. "Policlinico G.Martino" , Messina , Italy
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Lau RP, Melamed J, Yee-Chang M, Marcus S, Givi B, Zamuco R. Microcystic/Reticular Schwannoma Arising in the Submandibular Gland: A Rare Benign Entity that Mimics More Common Salivary Gland Carcinomas. Head Neck Pathol 2015; 10:374-8. [PMID: 26621673 PMCID: PMC4972748 DOI: 10.1007/s12105-015-0674-5] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/01/2015] [Accepted: 11/24/2015] [Indexed: 12/12/2022]
Abstract
Microcystic/reticular schwannoma is a recently described variant of schwannoma with a predilection for the gastrointestinal tract, rarely involving the head/neck region. This is the first reported case involving the submandibular gland. We present a case in a 34 year old man with 4.5 cm submandibular mass. Fine needle aspiration suggested a spindle cell lesion. Frozen section evaluation raised the possibility of mucoepidermoid carcinoma. Resection showed a well circumscribed mass with a mucoid appearance. Histologic findings include a lobular architecture with fibrous septa, a lympho-plasmacytic infiltrate, and scattered lymphoid aggregates at the periphery. There are two distinct histologic patterns with solid areas of spindle cells and areas of spindle/ovoid cells with a microcystic pattern in a myxoid background. The tumor has a pushing border, with extension into adipose and adjacent parenchyma, without cytologic atypia or necrosis. Immunohistochemical stains are positive for S-100 and CD34, and negative for calponin, mammoglobin, ALK1, p63, ER, GFAP, SMA, desmin, cytokeratin 7, cytokeratin AE1/AE3, and C-Kit. Mucicarmine stain is negative. Recognition of this benign unusual variant of schwannoma is paramount for appropriate conservative treatment due to the morphologic and immunohistochemical overlap with primary salivary gland carcinomas.
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Affiliation(s)
- Ryan P. Lau
- Department of Pathology, New York University School of Medicine, 462 1st Avenue, Office 4W35, New York, NY 10016 USA
| | - Jonathan Melamed
- Department of Pathology, New York University School of Medicine, 462 1st Avenue, Office 4W35, New York, NY 10016 USA
| | - Melissa Yee-Chang
- Department of Pathology, New York University School of Medicine, 462 1st Avenue, Office 4W35, New York, NY 10016 USA
| | - Sonya Marcus
- Department of Otolaryngology – Head and Neck Surgery, New York University School of Medicine, 462 First Avenue, Suite 5SE5, New York, NY 10016 USA
| | - Babak Givi
- Department of Otolaryngology – Head and Neck Surgery, New York University School of Medicine, 462 First Avenue, Suite 5SE5, New York, NY 10016 USA
| | - Ronaldo Zamuco
- Department of Pathology, New York University School of Medicine, 462 1st Avenue, Office 4W35, New York, NY 10016 USA
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Yin Y, Wang T, Cai YP, Huang XJ, Li YJ, Chen SH, Qin R, Wang CF, Wu Q. Microcystic/Reticular Schwannoma of the Mandible First Case Report and Review of the Literature. Medicine (Baltimore) 2015; 94:e1974. [PMID: 26559274 PMCID: PMC4912268 DOI: 10.1097/md.0000000000001974] [Citation(s) in RCA: 10] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 02/07/2023] Open
Abstract
Schwannoma comprises a group of nerve sheath tumors. Morphologic variants of schwannoma have no distinct relationship to clinical behavior, but unawareness of rare variants may lead to diagnostic pitfall and risk of mistreatment. Microcystic/reticular schwannoma is a recently described rare variant of schwannoma. We report a case of a 61-year-old female with a 5.0 cm × 3.5 cm × 3.0 cm mass in the right mandible, which has never been reported to date. Light microscopic evaluation showed that the mass was circumscribed with focal infiltration. Arranged in a prominent microcystic and reticular growth pattern, tumor cells were spindle-shaped with eosinophilic cytoplasm. No evidence of cytologic atypia, mitosis, or necrosis was observed. The stroma of the tumor mainly contained myxoid material with local infiltration of hyalinized collagen. Tumor cells showed diffuse and strong nuclear and cytoplasmic immunoreactivity for S100 protein. Tumor cells were also positive for CD34, CD99, and NSE, but negative for CK, EMA, CK5/6, P63, Calponin, CD10, SMA, Desmin, GFAP, NF, Syn, and CgA. The proliferation marker MIB-1 showed <1% nuclear reaction. Furthermore, we reviewed the clinical and pathological features of 24 previously reported cases of microcystic/reticular schwannoma. Unlike classic schwannoma, the reticular variant showed striking microcystic and reticular architecture microscopically. Recognition of these distinct entities is essential in avoiding misdiagnosis. Unlike classic schwannoma with a complete capsule, some masses were reported to lack encapsulation or contain focal infiltration. Further follow-up of tentative or identified cases is necessary to better understand this schwannoma.
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Affiliation(s)
- Yu Yin
- From the Department of Pathology, Anhui Medical University, Hefei, Anhui Province (YY, Y-PC, X-JH, Y-JL, S-HC, RQ, QW) and Department of Pathology, Shanghai Cancer Center, Fudan University, Shanghai, China (TW, C-FW)
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