梭形细胞嗜酸细胞瘤
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摘要
目的总结梭形细胞嗜酸细胞瘤的临床表现、影像学和病理学特点、诊断与鉴别诊断要点。方法与结果男性患者,59岁,主诉双眼视物模糊1年余伴反应迟钝、易疲劳;头部MRI显示鞍区占位性病变。组织学形态,肿瘤组织由梭形细胞构成,呈束状或不规则排列,肿瘤细胞胞质丰富、呈嗜酸性颗粒样,胞核呈卵圆形或短梭形、无明显核分裂象,间质内可见散在淋巴细胞浸润。免疫组织化学染色,部分肿瘤细胞胞核甲状腺转录因子-1阳性,胞质抗线粒体抗体、S-100蛋白、半乳糖凝集素-3和波形蛋白阳性,Ki-67抗原标记指数约3%。最终病理诊断为(鞍区)梭形细胞嗜酸细胞瘤。患者术后2个月出现视力急剧下降,复查MRI显示鞍上占位性病变,遂予放射治疗。随访10个月,视力无好转,肿瘤无明显改变。结论梭形细胞嗜酸细胞瘤是临床罕见的发生于鞍区的肿瘤,属WHOⅠ级,应注意与发生于鞍区的其他肿瘤相鉴别,治疗以手术全切除为主,部分切除患者术后辅以放射治疗。
Objective To study the clinical manifestations, imaging and pathological features,diagnosis and differential diagnosis of spindle cell oncocytoma. Methods and Results A 59-year-old man presented with one year history of blurred vision, lags in response and fatiguability. Head MRI showed a mass at the sellar region. Histological examination showed the tumor was mainly composed of spindle cells arranged in fascicularis or irregularity with abundant eosinophilic granular cytoplasm and oval or short spindle nuclei without typical mitotic activity. Infiltration of scattered lymphocytes was observed in the stroma. Immunohistochemical staining showed partial tumor cells were immunoreactive to thyroid transcription factor-1(TTF-1) in nuclei, anti-mitochondria antibody(AMA), S-100 protein(S-100), Galectin-3 and vimentin(Vim) in cytoplasm. Ki-67 labeling index was 3%. The final diagnosis was spindle cell oncocytoma in sellar region. The patient presented rapid loss of vision 2 months after surgery. Reexamined MRI showed a mass at the sellar region, and the patient was treated by radiotherapy. During the 10-month follow-up, the vision was not recovered, and the tumor was still existed. Conclusions Spindle cell oncocytoma is a rare benign tumor in the sellar region, which is considered as WHO grade Ⅰ tumor. It should be considered in the differential diagnosis of various sellar region lesions. Complete removal of tumor is important to prevent tumor recurrence. For patients who achieved partial resection, postoperative adjuvant radiotherapy is necessary.
Objective To study the clinical manifestations, imaging and pathological features,diagnosis and differential diagnosis of spindle cell oncocytoma. Methods and Results A 59-year-old man presented with one year history of blurred vision, lags in response and fatiguability. Head MRI showed a mass at the sellar region. Histological examination showed the tumor was mainly composed of spindle cells arranged in fascicularis or irregularity with abundant eosinophilic granular cytoplasm and oval or short spindle nuclei without typical mitotic activity. Infiltration of scattered lymphocytes was observed in the stroma. Immunohistochemical staining showed partial tumor cells were immunoreactive to thyroid transcription factor-1(TTF-1) in nuclei, anti-mitochondria antibody(AMA), S-100 protein(S-100), Galectin-3 and vimentin(Vim) in cytoplasm. Ki-67 labeling index was 3%. The final diagnosis was spindle cell oncocytoma in sellar region. The patient presented rapid loss of vision 2 months after surgery. Reexamined MRI showed a mass at the sellar region, and the patient was treated by radiotherapy. During the 10-month follow-up, the vision was not recovered, and the tumor was still existed. Conclusions Spindle cell oncocytoma is a rare benign tumor in the sellar region, which is considered as WHO grade Ⅰ tumor. It should be considered in the differential diagnosis of various sellar region lesions. Complete removal of tumor is important to prevent tumor recurrence. For patients who achieved partial resection, postoperative adjuvant radiotherapy is necessary.
引文
[1]Matyja E, Maksymowicz M, Grajkowska W, Olszewski W,Zieliński G, Bonicki W. Spindle cell oncocytoma of the adenohypophysis:a clinicopathological and ultrastructural studyof two cases[J]. Folia Neuropathol, 2010, 48:175-184.
[2]Roncaroli F, Scheithauer BW, Cenacchi G, Horvath E, KovacsK, Lloyd RV, Abell-Aleff P, Santi M, Yates AJ.'Spindle celloncocytoma'of the adenohypophysis:a tumor of folliculostellatecells[J]? Am J Surg Pathol, 2002, 26:1048-1055.
[3]Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC,Jouvet A, Scheithauer BW, Kleihues P. The 2007 WHOclassification of tumor of the central nervous system[J]. ActaNeuropathol, 2007, 114:97-109.
[4]Louis DN, Perry A, Reifenberger G, von Deimling A, Figarella-Branger D, Cavenee WK, Ohgaki H, Wiestler OD, Kleihues P,Ellison DW. The 2016 World Health Organization Classificationof Tumors of the Central Nervous System:a summary[J]. Acta[Neuropathol, 2016, 131:803-820.
[5]Vuong HG, Kondon T, Tran TM, Oishi N, Nakazawa T,Mochizuki K, Inoue T, Kasai K, Tahara I, Jieying W, Katoh R.Spindle cell oncocytoma of adenohypophysis:report of a caseand immunohistochemical review of literature[J]. Pathol ResPract, 2016, 212:222-225.
[6]Yoshimoto T, Takahashi-Fujigasaki J, Inoshita N, Fukuhara N,Nishioka H, Yamada S. TTF-1-positive oncocytic sellar tumorwith follicle formation/ependymal differentiation:non-adenomatous tumor capable of two different interpretations as apituicytoma or spindle cell oncocytoma[J]. Brain Tumor Pathol,2015, 32:221-227.
[7]Mete O, Lopes MB, Asa SL. Spindle cell oncocytomas andgranular cell tumor of the potuitary are variants of pituicytoma[J]. Am J Surg Pathol, 2013, 37:1694-1699.
[8]Kloub O, Perry A, Tu PH, Lipper M, Lopes MB. Spindle celloncocytoma of edenohypophysis:report of two recurrent case[J]. Am J Surg Pathol, 2005, 29:247-253.
[9]Dahiya S, Sarkar C, Hedley-Whyte ET, Sharma MC, Zervas NT,Sridhar E, Louis DN. Spindle cell oncocytoma of theadenohypophysis:report of two cases[J]. Acta Neuropathol,2005, 110:97-99.
[10]Vajtai I, Sahli R, Kappeler A. Spindle cell oncocytoma of the adenohypophysis:report of a case with a 16-year follow-up[J].Pathol Res Pract, 2006, 202:745-750.
[11]Farooq MU, Bhatt A, Chang HT. Teaching neuroimage:spindlecell oncocytoma of the pituitary gland[J]. Neurology, 2008, 71:E3.
[12]Borota OC, Scheithauer BW, Fouger SL, Hald JK, Ramm-Pettersen J, Bollerslev J. Spindle cell oncocytoma of theadenohypophysis:report of a case with marked cellular atypiaand recurrence despite adjuvant treatment[J]. ClinNeuropathol, 2009, 28:91-95.
[13]CoiréCI, Horvath E, Smyth HS, Kovas K. Rapidly recurringfolliculostellate cell tumor of the adenohypophysis with themorphology of a spindle cell oncocytoma:case report with electronmicroscopic studies[J]. Clin Neuropathol, 2009, 28:303-308.
[14]Demssie YN, Joseph J, Dawson T, Roberts G, de Carpentier J,Howell S. Recurrent spindle cell oncocytoma of the pituitary:acase report and review of literature[J]. Pituitary, 2011, 14:367-370.
[15]Borges MT, Lillehei KO, Kleinschmidt-DeMasters BK. Spindlecell oncocytoma with late recurrence and unique neuroimagingcharacteristics due to recurrent subclinical intratumoral bleeding[J]. J Neurooncol, 2011, 101:145-154.
[16]Millka M, Azouz H, Chelly I, Said IB, Jemel H, Haouet S,Zitouna M, Kchir N. Spindle cell oncocytoma of theadenohypophysis in a woman:a case report and review of the literature[J]. J Med Case Rep, 2011, 5:64.
[17]Romero-Rojas AE, Melo-Uribe MA, Barajas-Solano PA,Chinchilla-Olaya SI, Escobar LI, Hernandez-Walteros DM.Spindle cell oncocytoma of the adenohypophysis[J]. BrainTumor Pathol, 2011, 28:359-364.
[18]Ogiwara H, Dubner S, Shafizadeh S, Raizer J, Chandler JP.Spindle cell oncocytoma of the pituitary and pituicytoma:twotumors mimicking pituitary adenoma[J]. Surg Neurol Int, 2011,2:116.
[19]Vajtai I, Beck J, Kappeler A, Hewer E. Spindle celloncocytoma of the pituitary gland with follicle-like component:organotypic differentiation to support its origin from folliculo-stellate cells[J]. Acta Neuropathol, 2011, 122:253-258.
[20]Fujisawa H, Tohma Y, Muramatsu N, Kida S, Kaizaki Y,Tamamura H. Spindle cell oncocytoma of the adenohypophysiswith marked hypervascularity:case report[J]. Neurol Med Chir(Toyko), 2012, 52:594-598.
[21]Alexandrescu S, Brown RE, Tandon N, Bhattacharjee MB.Neuron precursor features of spindle cell oncocytoma ofadenohypophysis[J]. Ann Clin Lab Sci, 2012, 42:123-129.
[22]Singh G, Agarwal S, Sharma MC, Suri V, Sarkar C, Garg A,Kale SS. Spindle cell oncocytoma of the adenohypophysis:report of a case and review of literature[J]. Clin NeurolNeurosurg, 2012, 114:267-271.
[23]Rotman JA, Kucharczyk W, Zadeh G, Kiehl TR, Al-Ahmadi H.Spindle cell oncocytoma of the adenohypophysis:a case reportillustrating its natural history with 8-year observation and areview of the literature[J]. Clin Imaging, 2014, 38:499-504.
[24]Mu Q, Yu J, Qu L, Hu X, Gao H, Liu P, Zheng X, Sun Y,Huang H. Spindle cell oncocytoma of the adenohypophysis:twocase reports and a review of the literature[J]. Mol Med Rep,2015, 12:871-876.
[25]Bie L, Peng T, Yu HQ, Xu HY, Zhao G, Zhang XF. Spindlecell oncocytoma of the adenohypophysis:two case reports and areview of the literature[J]. Zhonghua Shen Jing Wai Ke Za Zhi,2013, 29:783-785[.别黎,彭涛,于洪泉,许海洋,赵刚,张显峰.垂体前叶梭形细胞嗜酸性细胞瘤二例并文献复习[J].中华神经外科杂志, 2013, 29:783-785.]
[26]Zygourakis CC, Rolston JD, Lee HS, Partow C, Kunwar S, AghiMK. Pituicytomas and spindle cell oncocytomas:modern caseseries from the University of California, San Francisco[J].Pituitary, 2015, 18:150-158.
[27]Mansour M, Juanita G, Tyler S, Edison V, Shams H, Teresa A.Spindle cell oncocytoma of the pituitary gland:case report andliterature review[J]. Neuro Oncol, 2015, 17(Suppl 5):202.
[28]Ryu WH, Hamilton L, Starreveld Y, Chan J. An unusualoncocytic sellar neoplasm in a patient with Birt-Hogg-Dubésyndrome[J]. Int J Neuropathol, 2015, 3:43-54.
[29]Custodio PJ, Jho DH, Pu C, Gordon MB, Donangelo I. Spindlecell oncocytoma of the pituitary presenting with severehyponatremia[J]. AACE Clin Case Rep, 2016, 2:E237-243.
[30]Billeci D, Marton E, Giordan E, Carraro V, Ronzon M, Rossi S.Spindle cell oncocytoma:report of two cases with massivebleeding and review of the literature[J]. J Clin Neurosci, 2017,39:39-44.
[31]Osman M, Wild A. Spindle cell oncocytoma of the anteriorpituitary presenting with an acute clinical course due tointraventricular hemorrhage:a case report and review ofliterature[J]. Am J Case Rep, 2017, 18:894-901.
[32]Kong X, Li D, Kong Y, Zhong D. Malignant adenohypophysisspindle cell oncocytoma with repeating recurrences and a highKi-67 index[J]. Medicine(Baltimore), 2017, 96:E5657.
[33]Sali A, Epari S, Tampi C, Goel A. Spindle cell oncocytoma ofadenohypophysis:review of literature and report of anotherrecurrent case[J]. Neuropathology, 2017, 37:535-543.
[34]Zhao LH, Wu XH, Xu MH, Wang Y, Xu LS, Xiao HL. Spindlecell oncocytoma of adenohypophysis:a clinicopathologicalobservation[J]. Zhen Duan Bing Li Xue Za Zhi, 2009, 16:440-443[.赵连花,吴晓华,许民辉,王毅,徐伦山,肖华亮.腺垂体梭形细胞嗜酸细胞瘤临床病例观察[J].诊断病理学杂志,2009, 16:440-443.]
[35]Hasiloglu ZI, Ure E, Comunoglu N, Tanriover N, Oz B,Gazioglu N, Mihmanli I. New radiological clues in diagnosis ofspindle cell oncocytoma of the adenohypophysis[J]. ClinRadiol, 2016, 71:E5-11.
[36]Guadagno E, Cervasio M, Di Somma A, Califano M, Solari D,Del Basso De Caro M. Essential role of ultrastructuralexamination for spindle cell oncocytoma:case report of a rareneoplasm and review of the literature[J]. Ultrastruct Pathol,2016, 40:121-124.
[2]Roncaroli F, Scheithauer BW, Cenacchi G, Horvath E, KovacsK, Lloyd RV, Abell-Aleff P, Santi M, Yates AJ.'Spindle celloncocytoma'of the adenohypophysis:a tumor of folliculostellatecells[J]? Am J Surg Pathol, 2002, 26:1048-1055.
[3]Louis DN, Ohgaki H, Wiestler OD, Cavenee WK, Burger PC,Jouvet A, Scheithauer BW, Kleihues P. The 2007 WHOclassification of tumor of the central nervous system[J]. ActaNeuropathol, 2007, 114:97-109.
[4]Louis DN, Perry A, Reifenberger G, von Deimling A, Figarella-Branger D, Cavenee WK, Ohgaki H, Wiestler OD, Kleihues P,Ellison DW. The 2016 World Health Organization Classificationof Tumors of the Central Nervous System:a summary[J]. Acta[Neuropathol, 2016, 131:803-820.
[5]Vuong HG, Kondon T, Tran TM, Oishi N, Nakazawa T,Mochizuki K, Inoue T, Kasai K, Tahara I, Jieying W, Katoh R.Spindle cell oncocytoma of adenohypophysis:report of a caseand immunohistochemical review of literature[J]. Pathol ResPract, 2016, 212:222-225.
[6]Yoshimoto T, Takahashi-Fujigasaki J, Inoshita N, Fukuhara N,Nishioka H, Yamada S. TTF-1-positive oncocytic sellar tumorwith follicle formation/ependymal differentiation:non-adenomatous tumor capable of two different interpretations as apituicytoma or spindle cell oncocytoma[J]. Brain Tumor Pathol,2015, 32:221-227.
[7]Mete O, Lopes MB, Asa SL. Spindle cell oncocytomas andgranular cell tumor of the potuitary are variants of pituicytoma[J]. Am J Surg Pathol, 2013, 37:1694-1699.
[8]Kloub O, Perry A, Tu PH, Lipper M, Lopes MB. Spindle celloncocytoma of edenohypophysis:report of two recurrent case[J]. Am J Surg Pathol, 2005, 29:247-253.
[9]Dahiya S, Sarkar C, Hedley-Whyte ET, Sharma MC, Zervas NT,Sridhar E, Louis DN. Spindle cell oncocytoma of theadenohypophysis:report of two cases[J]. Acta Neuropathol,2005, 110:97-99.
[10]Vajtai I, Sahli R, Kappeler A. Spindle cell oncocytoma of the adenohypophysis:report of a case with a 16-year follow-up[J].Pathol Res Pract, 2006, 202:745-750.
[11]Farooq MU, Bhatt A, Chang HT. Teaching neuroimage:spindlecell oncocytoma of the pituitary gland[J]. Neurology, 2008, 71:E3.
[12]Borota OC, Scheithauer BW, Fouger SL, Hald JK, Ramm-Pettersen J, Bollerslev J. Spindle cell oncocytoma of theadenohypophysis:report of a case with marked cellular atypiaand recurrence despite adjuvant treatment[J]. ClinNeuropathol, 2009, 28:91-95.
[13]CoiréCI, Horvath E, Smyth HS, Kovas K. Rapidly recurringfolliculostellate cell tumor of the adenohypophysis with themorphology of a spindle cell oncocytoma:case report with electronmicroscopic studies[J]. Clin Neuropathol, 2009, 28:303-308.
[14]Demssie YN, Joseph J, Dawson T, Roberts G, de Carpentier J,Howell S. Recurrent spindle cell oncocytoma of the pituitary:acase report and review of literature[J]. Pituitary, 2011, 14:367-370.
[15]Borges MT, Lillehei KO, Kleinschmidt-DeMasters BK. Spindlecell oncocytoma with late recurrence and unique neuroimagingcharacteristics due to recurrent subclinical intratumoral bleeding[J]. J Neurooncol, 2011, 101:145-154.
[16]Millka M, Azouz H, Chelly I, Said IB, Jemel H, Haouet S,Zitouna M, Kchir N. Spindle cell oncocytoma of theadenohypophysis in a woman:a case report and review of the literature[J]. J Med Case Rep, 2011, 5:64.
[17]Romero-Rojas AE, Melo-Uribe MA, Barajas-Solano PA,Chinchilla-Olaya SI, Escobar LI, Hernandez-Walteros DM.Spindle cell oncocytoma of the adenohypophysis[J]. BrainTumor Pathol, 2011, 28:359-364.
[18]Ogiwara H, Dubner S, Shafizadeh S, Raizer J, Chandler JP.Spindle cell oncocytoma of the pituitary and pituicytoma:twotumors mimicking pituitary adenoma[J]. Surg Neurol Int, 2011,2:116.
[19]Vajtai I, Beck J, Kappeler A, Hewer E. Spindle celloncocytoma of the pituitary gland with follicle-like component:organotypic differentiation to support its origin from folliculo-stellate cells[J]. Acta Neuropathol, 2011, 122:253-258.
[20]Fujisawa H, Tohma Y, Muramatsu N, Kida S, Kaizaki Y,Tamamura H. Spindle cell oncocytoma of the adenohypophysiswith marked hypervascularity:case report[J]. Neurol Med Chir(Toyko), 2012, 52:594-598.
[21]Alexandrescu S, Brown RE, Tandon N, Bhattacharjee MB.Neuron precursor features of spindle cell oncocytoma ofadenohypophysis[J]. Ann Clin Lab Sci, 2012, 42:123-129.
[22]Singh G, Agarwal S, Sharma MC, Suri V, Sarkar C, Garg A,Kale SS. Spindle cell oncocytoma of the adenohypophysis:report of a case and review of literature[J]. Clin NeurolNeurosurg, 2012, 114:267-271.
[23]Rotman JA, Kucharczyk W, Zadeh G, Kiehl TR, Al-Ahmadi H.Spindle cell oncocytoma of the adenohypophysis:a case reportillustrating its natural history with 8-year observation and areview of the literature[J]. Clin Imaging, 2014, 38:499-504.
[24]Mu Q, Yu J, Qu L, Hu X, Gao H, Liu P, Zheng X, Sun Y,Huang H. Spindle cell oncocytoma of the adenohypophysis:twocase reports and a review of the literature[J]. Mol Med Rep,2015, 12:871-876.
[25]Bie L, Peng T, Yu HQ, Xu HY, Zhao G, Zhang XF. Spindlecell oncocytoma of the adenohypophysis:two case reports and areview of the literature[J]. Zhonghua Shen Jing Wai Ke Za Zhi,2013, 29:783-785[.别黎,彭涛,于洪泉,许海洋,赵刚,张显峰.垂体前叶梭形细胞嗜酸性细胞瘤二例并文献复习[J].中华神经外科杂志, 2013, 29:783-785.]
[26]Zygourakis CC, Rolston JD, Lee HS, Partow C, Kunwar S, AghiMK. Pituicytomas and spindle cell oncocytomas:modern caseseries from the University of California, San Francisco[J].Pituitary, 2015, 18:150-158.
[27]Mansour M, Juanita G, Tyler S, Edison V, Shams H, Teresa A.Spindle cell oncocytoma of the pituitary gland:case report andliterature review[J]. Neuro Oncol, 2015, 17(Suppl 5):202.
[28]Ryu WH, Hamilton L, Starreveld Y, Chan J. An unusualoncocytic sellar neoplasm in a patient with Birt-Hogg-Dubésyndrome[J]. Int J Neuropathol, 2015, 3:43-54.
[29]Custodio PJ, Jho DH, Pu C, Gordon MB, Donangelo I. Spindlecell oncocytoma of the pituitary presenting with severehyponatremia[J]. AACE Clin Case Rep, 2016, 2:E237-243.
[30]Billeci D, Marton E, Giordan E, Carraro V, Ronzon M, Rossi S.Spindle cell oncocytoma:report of two cases with massivebleeding and review of the literature[J]. J Clin Neurosci, 2017,39:39-44.
[31]Osman M, Wild A. Spindle cell oncocytoma of the anteriorpituitary presenting with an acute clinical course due tointraventricular hemorrhage:a case report and review ofliterature[J]. Am J Case Rep, 2017, 18:894-901.
[32]Kong X, Li D, Kong Y, Zhong D. Malignant adenohypophysisspindle cell oncocytoma with repeating recurrences and a highKi-67 index[J]. Medicine(Baltimore), 2017, 96:E5657.
[33]Sali A, Epari S, Tampi C, Goel A. Spindle cell oncocytoma ofadenohypophysis:review of literature and report of anotherrecurrent case[J]. Neuropathology, 2017, 37:535-543.
[34]Zhao LH, Wu XH, Xu MH, Wang Y, Xu LS, Xiao HL. Spindlecell oncocytoma of adenohypophysis:a clinicopathologicalobservation[J]. Zhen Duan Bing Li Xue Za Zhi, 2009, 16:440-443[.赵连花,吴晓华,许民辉,王毅,徐伦山,肖华亮.腺垂体梭形细胞嗜酸细胞瘤临床病例观察[J].诊断病理学杂志,2009, 16:440-443.]
[35]Hasiloglu ZI, Ure E, Comunoglu N, Tanriover N, Oz B,Gazioglu N, Mihmanli I. New radiological clues in diagnosis ofspindle cell oncocytoma of the adenohypophysis[J]. ClinRadiol, 2016, 71:E5-11.
[36]Guadagno E, Cervasio M, Di Somma A, Califano M, Solari D,Del Basso De Caro M. Essential role of ultrastructuralexamination for spindle cell oncocytoma:case report of a rareneoplasm and review of the literature[J]. Ultrastruct Pathol,2016, 40:121-124.