Correspondence to:Satoru Kase.Department of Ophthalmology,Faculty of Medicine and Graduate School of Medicine,Hokkaido University,N-15,W-7,Kita-ku,Sapporo 060-8638,Japan.kaseron@med.hokudai.ac.jp AIM:To evaluate the c...Correspondence to:Satoru Kase.Department of Ophthalmology,Faculty of Medicine and Graduate School of Medicine,Hokkaido University,N-15,W-7,Kita-ku,Sapporo 060-8638,Japan.kaseron@med.hokudai.ac.jp AIM:To evaluate the clinical features of primary lacrimal gland pleomorphic adenoma(LGPA).METHODS:This was a 2-center,retrospective,observational study of primary LGPA patients who underwent tumor resection.Ophthalmic examinations and orbital computed tomography(CT)and/or magnetic resonance imaging(MRI)were performed.RESULTS:Totally 18 patients(10 males and 8 females,mean age 56.3±13.8y)were enrolled.Initial symptoms were unilateral proptosis in 8 patients,diplopia in 3 patients,and pressure sensation and no chief complaint in 2 patients each.The best-corrected visual acuity of the affected eye was 0.26±0.44 logMAR,and the intraocular pressure(IOP)of the affected and healthy eyes was 20.1±9.9 and 15.8±4.3 mm Hg,respectively.The difference in degree of proptosis between the affected and healthy eyes was 4.1±2.2 mm based on the Hertel ocular protrusion meter.One case had a history of breast cancer.Seventeen of 18 patients with an orbital lacrimal gland origin underwent total tumor excision by anterior/lateral orbitotomy.CONCLUSION:Orbital LGPA can complicate IOP elevation,and require total tumor excision by orbitotomy.In rare cases,systemic malignancy may complicate LGPA,and in such cases total removal of the lacrimal gland tumor should be considered at the initial surgery.展开更多
AIM:To examine carcinoma ex pleomorphic adenoma(CXPA)and its development to provide information for its clinical assessment and prognosis.METHODS:The clinical data of 26 patients with pathologically diagnosed CXPA wer...AIM:To examine carcinoma ex pleomorphic adenoma(CXPA)and its development to provide information for its clinical assessment and prognosis.METHODS:The clinical data of 26 patients with pathologically diagnosed CXPA were included for analysis.The patients’medical histories and data(e.g.,gender,age,eye laterality,clinical manifestations,pathologic and immunohistochemical indices,treatments,and prognosis)were recorded.RESULTS:The average age of the 26 patients was 59.6±15.7y.There was no significant difference in the gender distribution.The incidence of CXPA bone destruction was approximately 57.7%,and the incidence of optic nerve involvement and extraocular muscle involvement was approximately 15.4%and 19.2%,respectively.The most common pathological type was adenocarcinoma(34.6%),followed by ductal carcinoma(26.9%).Five patients had recurrence or metastasis(19.2%).The 5-year recurrencefree survival rate was 59.0%.There were no significant differences in survival rates among patients with different pathologic types and stages(P>0.05).Bone destruction,nerve invasion,and peripheral tissue invasion did not significantly affect survival rate(P>0.05).Surgery combined with 125I radiation therapy leads to a better survival prognosis(P<0.05).CONCLUSION:CXPA has a variety of pathologic classifications,with characteristics of bone destruction and peripheral tissue invasion.Surgery combined with ^(125)I endoradiotherapy is a preferable treatment option.However,long-term follow-up and close observation for recurrence or metastasis should be performed.展开更多
Background: Mucoepidermoid carcinoma ex-pleomorphic adenoma (MECxPA) is an extremely rare salivary gland malignancy. With only nine prior reported cases, this entity represents a challenging histopathological diagnosi...Background: Mucoepidermoid carcinoma ex-pleomorphic adenoma (MECxPA) is an extremely rare salivary gland malignancy. With only nine prior reported cases, this entity represents a challenging histopathological diagnosis. Methods: We present a case of a 71-year-old male with an enlarging left neck mass over several months. CT showed both a parapharyngeal space mass and a separate level II neck mass. Results: The patient underwent resection of the left parapharyngeal mass and ipsilateral selective level II-IV lymphadenectomy. The final pathologic diagnosis was metastatic high-grade mucoepidermoid carcinoma ex-pleomorphic adenoma. Conclusions: We describe a novel presentation of high-grade mucoepidermoid carcinoma ex-pleomorphic adenoma as a metastatic parapharyngeal mass.展开更多
Giant cell tumors of the pancreas come in three varieties-osteoclastic,pleomorphic,and mixed histology.These tumors have distinctive endoscopic,clinical,and cytological features.Giant cell tumors have a controversial ...Giant cell tumors of the pancreas come in three varieties-osteoclastic,pleomorphic,and mixed histology.These tumors have distinctive endoscopic,clinical,and cytological features.Giant cell tumors have a controversial histogenesis,with some authors favoring an epithelial origin and others favoring a mesenchymal origin.The true origin of these lesions remains unclear at this time.These are also very rare tumors but proper identification and differentiation from more common pancreatic adenocarcinoma is important.The risk factors of these tumors and the prognosis may be different from those associated with standard pancreatic adenocarcinoma.Recognition of these differences can significantly affect patient care.These lesions have a unique appearance when imaged with endoscopic ultrasound(EUS),and these lesions can be diagnosed via EUS guided Fine Needle Aspiration(FNA).This manuscript will review the endoscopic,clinical,and pathologic features of these tumors.展开更多
BACKGROUND:A malignant fibrous histiocytoma,recently referred to as an undifferentiated pleomorphic sarcoma (UPS),is very rare in the liver,and only 34 cases of primary hepatic malignant fibrous histiocytoma have been...BACKGROUND:A malignant fibrous histiocytoma,recently referred to as an undifferentiated pleomorphic sarcoma (UPS),is very rare in the liver,and only 34 cases of primary hepatic malignant fibrous histiocytoma have been reported in the English literature. METHODS:We report a rare case of a hepatic UPS presenting as a unilocular cystic lesion with respect to histopathologic features,the newly revised diagnostic criteria,and the differential diagnosis. RESULTS:A 60-year-old man presented for evaluation of epigastric pain of 7 months duration.Abdominal computed tomography revealed a hypodense,unilocular cystic mass in the right lobe of the liver,measuring 14.0×11.1 cm.A right lobectomy was performed.Grossly,the cut surface showed a single,well-circumscribed unilocular cystic tumor mass containing dark red-brown necrotic debris and blood clots, which occupied most of the mass.Microscopically,the tumor consisted of haphazardly arranged mononuclear pleomorphic tumor cells,admixed with abundant osteoclast- like multinucleated giant cells.Immunohistochemically,the tumor cells expressed vimentin only.The histopathologic and immunohistochemical findings were compatible with a UPS.The patient is alive and well 41 months after surgery without recurrence. CONCLUSIONS:Clinically,most of the hepatic UPSs are solid masses.Only two cases have presented as multilocular cystic masses.A primary hepatic UPS presenting as a unilocular cyst has never been reported.A UPS should be included in the differential diagnosis of unilocular cystic lesions in the liver.展开更多
AIM: To investigate C-myc, Ki-67, pan-cytokeratin, and vimentin immunohistochemical features of carcinoma ex pleomorphic adenoma(Ca-ex-PA) and pleomorphic adenoma(PA) in the lacrimal gland in order to find some clues ...AIM: To investigate C-myc, Ki-67, pan-cytokeratin, and vimentin immunohistochemical features of carcinoma ex pleomorphic adenoma(Ca-ex-PA) and pleomorphic adenoma(PA) in the lacrimal gland in order to find some clues in the differential diagnosis between them.METHODS: We reviewed microscopic slides and clinical records of 64 cases of PA and 15 cases of Ca-ex-PA in the lacrimal gland. Immunohistochemical antibodies for C-myc, Ki-67, pan-cytokeratin, and vimentin were employed.RESULTS: Median age of PA was 43.2 y(from 21 to 75). The 35 patients(54.7%) were male and 29 patients(45.3%) were female. For the PAs, the average positivity of C-myc was 4.6%;the average proliferation index of Ki-67 was 3.2%;pan-cytokeratin was positive in ductal cells, and vimentin was positive in myoepithelial cells. Median age of Ca-ex-PA was 54.3 y(from 26 to 76). There were 7 male patients(46.7%) and 8 female patients(53.3%). Among 15 Ca-ex-PAs, there were 6 myoepithelial carcinomas, 4 adenocarcinomas, 3 epithelial-myoepithelial carcinomas, and 2 squamous cell carcinomas. For the Ca-ex-PAs, the average positivity of C-myc was 36.4%;the average proliferation index of Ki-67 was 29.2%;pan-cytokeratin was positive in all cases, and vimentin was positive in myoepithelial carcinomas.CONCLUSION: PA has a lower positivity of C-myc and Ki-67, while Ca-ex-PA had a higher positivity of these two biomarkers. These four biomarkers as a set could provide valuable clues in the differential diagnosis between Ca-exPA and PA. Our results indicate that the activation of C-myc could play an important role in the pathogenesis of Ca-exPA and PA.展开更多
BACKGROUND Pleomorphic adenoma(PA)is the most common benign tumor that occurs in the salivary glands;however,tracheobronchial PA is rarely observed.To the best of our knowledge,fewer than 50 cases have been reported i...BACKGROUND Pleomorphic adenoma(PA)is the most common benign tumor that occurs in the salivary glands;however,tracheobronchial PA is rarely observed.To the best of our knowledge,fewer than 50 cases have been reported in the literature.We report a 49-year-old woman who had been treated for asthma for 2 years before being diagnosed with PA of the trachea.CASE SUMMARY A 49-year-old woman was referred to our hospital due to dyspnea upon exertion and chronic cough with wheezing for 2 years.Laboratory tests showed an elevated white blood cell count,absolute neutrophil count,and percentage of neutrophils.A chest computerized tomography scan showed a well-defined,softtissue density lesion measuring 2.4 cm×2.1 cm in the lower trachea.Flexible bronchoscopy revealed that nearly 90%of the tracheal lumen was obstructed.The histopathological and immunohistochemistry features suggested PA of the trachea.Furthermore,we review the characteristics of 29 patients with tracheobronchial PA over the last 30 years.CONCLUSION Tracheobronchial PA occurs without gender predominance,mostly in the lower or upper trachea,and has a low recurrence rate.The median age at diagnosis is 48 years.The most common symptoms are cough,stridor,dyspnea,and wheezing.展开更多
BACKGROUND Carcinoma ex pleomorphic adenoma (CXPA) is defined as a malignant salivary gland tumor arising from a primary or recurrent pleomorphic adenoma.Only three cases of CXPA of the trachea have been reported in t...BACKGROUND Carcinoma ex pleomorphic adenoma (CXPA) is defined as a malignant salivary gland tumor arising from a primary or recurrent pleomorphic adenoma.Only three cases of CXPA of the trachea have been reported in the literature.CASE SUMMARY We report a case of tracheal CXPA in a 55-year-old woman,who presented with a more than 3-mo history of progressive dyspnea.Computed tomography of the neck and thorax revealed an inhomogeneous,broad-based lesion arising from the tracheal wall on the right side.Endoscopy revealed a subglottic neoplasm causing up to 90% luminal stenosis.The tumor was resected using a highfrequency electrosurgical snare combined with argon plasma coagulation.Histopathology and immunohistochemistry revealed that the tumor was a CXPA of the trachea.CONCLUSION We report the fourth case of tracheal CXPA,and present the first instance of resection of CXPA using high-frequency electrosurgical snare and laser ablation.We also discuss the pathogenesis,diagnosis,histopathology,and systemic therapy of this rare disease.展开更多
BACKGROUND Pleomorphic lipoma (PL) is a rare benign mesenchymal tumor occurring predominantly in middle-aged and elderly men. It is typically found in the subcutaneous tissue of the posterior neck, back, and shoulders...BACKGROUND Pleomorphic lipoma (PL) is a rare benign mesenchymal tumor occurring predominantly in middle-aged and elderly men. It is typically found in the subcutaneous tissue of the posterior neck, back, and shoulders. There have also been a few reported cases in the face, scalp, and upper extremities. Currently, there is no report on its occurrence in the anterior mediastinum. CASE SUMMARY Herein, we report the case of a 67-year-old woman diagnosed with PL in the anterior mediastinum. The tumor was removed by thoracoscopic surgery. There was no recurrence during the 24-mo follow-up period, and the prognosis was good. Most PL are located on the skin surface. However, they may also occur within the body, even in the mediastinum. CONCLUSION PL occurring in the anterior mediastinum is rare, and it may be differentiated from other tumors.展开更多
AIM:To monitor the prognosis of patients with lacrimal gland pleomorphic adenoma(LGPA)following surgical resection within the past 10y.The factors affecting patient prognosis are analyzed and the experience of surgica...AIM:To monitor the prognosis of patients with lacrimal gland pleomorphic adenoma(LGPA)following surgical resection within the past 10y.The factors affecting patient prognosis are analyzed and the experience of surgical treatment is summarized.METHODS:In this retrospective,comparative case series,clinical records from 109 cases of LGPA treated chiefly at the same institution between November 2009 and May 2019 were reviewed.All 109 patients underwent surgery,histopathological examination,and imaging examination.For patients who underwent surgery for the first time,LGPA could be resected completely,including tumor and capsule tissues,using a surgical approach via the eyebrow arch or double eyelid crease.RESULTS:The ratio of males to females was 1:1.60,the ages ranged from 19 to 74 years old with a mean age of 43.64±13.07 years old,and the ratio of left to right eyes was 1:1.37.A total of 109 patients underwent surgical excision and five of these收稿日期radiotherapy after malignant transformation.Of these patients,15 were lost to follow up within the April 1,2020 deadline and 1 was diagnosed as a recurring pathology.The 5-year recurrence rate for 86 patients who underwent initial surgery was 7.27%.Single factor analysis revealed that the course of disease,bone destruction,invasion of surrounding tissues,tumor size,capsule integrity,and expression of Ki-67 were statistically significant(P<0.05).Binomial Logistic regression analysis showed that capsule integrity was a risk factor influencing recurrence(P=0.008).CONCLUSION:LGPA has a risk of recurrence and potential for malignant transformation.Complete removal of the tumor and capsule in the initial surgery is a key factor in preventing recurrence.展开更多
BACKGROUND Papillary thyroid cancer(PTC)is the most common malignant tumor of the thyroid.However,the coexistence of PTC and sarcoma in one patient is rare.In this article,we report the case of a patient who presented...BACKGROUND Papillary thyroid cancer(PTC)is the most common malignant tumor of the thyroid.However,the coexistence of PTC and sarcoma in one patient is rare.In this article,we report the case of a patient who presented with both PTC and undifferentiated pleomorphic sarcoma(UPS),which has not been previously reported in the online Medline database(PubMed).CASE SUMMARY A 71-year-old man was admitted to our hospital for a mass on the right side of his neck for one month,which rapidly enlarged within 2 wk with distending pain.The patient was diagnosed with a thyroid malignancy by fine-needle aspiration and underwent total thyroidectomy and bilateral central lymph node dissection.Histology and immunohistochemistry revealed features of both PTC and UPS.The thyroid cancer 8 gene detection kit results showed BRAF and telomerase reverse transcriptase mutations.The disease progressed rapidly,and the patient died four months after surgery from extensive lung metastasis.CONCLUSION Our report highlights the patient’s pathological characteristics and related genetic mutations.Due to the rapid development and poor prognosis of cooccurring PTC and sarcoma,it is important for clinical physicians and pathologists to raise awareness of this type of tumor.展开更多
·AIM: To detect proteomic differences in tears between adenoid cystic carcinoma(ACC) and pleomorphic adenoma(PA).·METHODS: Tear samples were collected from 4 patients with ACC, 5 with PA, and 4 control cases...·AIM: To detect proteomic differences in tears between adenoid cystic carcinoma(ACC) and pleomorphic adenoma(PA).·METHODS: Tear samples were collected from 4 patients with ACC, 5 with PA, and 4 control cases. Label-free analysis and parallel reaction monitoring(PRM) were used to screen and validate the tear proteome. Gene Ontology(GO) annotation and Kyoto Encyclopedia of Genes and Genomes(KEGG) were conducted for bioinformatics analysis.·RESULTS: In total, 1059 proteins in tear samples were identified by label-free analysis. Between ACC and PA, 415 differentially expressed proteins were detected. Based on the GO annotation, enzyme regulator activity and serinetype endopeptidase inhibitor activity in the molecular function category, blood microparticle and extracellular matrix in the cellular component category, and response to nutrient levels in the biological process category were most predominant. By KEGG pathway annotation, the different proteins between ACC and PA mainly par ticipated in complement and coagulation cascades, amoebiasis, African trypanosomiasis and cholesterol metabolism. Eight proteins with mostly significant differences were verified by PRM, and five proteins with more than 10-fold increases in ACC compared with PA, including integrin β, α-2-macroglobulin, epididymal secretory sperm binding protein Li 78p, RAB5C, and complement C5, were identified.·CONCLUSION: The combined tools of label-free analysis and PRM are very effective and efficient, especially for samples such as tears. Some proteomic dif ferences in tears between ACC and PA are identified and these protein candidates may be specific biomarkers for future exploration.展开更多
BACKGROUND Pleomorphic rhabdomyosarcoma(RMS)of the spermatic cord is a group of rare neoplasms,and a secondary hydrocele testis occasionally occurs.The misdiagnosis of paratesticular mass may lead to a therapeutic del...BACKGROUND Pleomorphic rhabdomyosarcoma(RMS)of the spermatic cord is a group of rare neoplasms,and a secondary hydrocele testis occasionally occurs.The misdiagnosis of paratesticular mass may lead to a therapeutic delay.CASE SUMMARY A 79-year-old man presented to our clinic complaining of a 1-mo history of painless scrotal swelling.Physical examination revealed approximately a 15 cm×10 cm×5 cm inguinal mass with limited mobility.Contrast-enhanced magnetic resonance imaging showed a hydrocele testis,several enlarged inguinal lymph nodes,and a heterogeneously enhanced lesion with a relatively well-defined margin in the left inguinal region.Due to the imaging findings,he was diagnosed with pleomorphic RMS and received a wide resection of the mass,an inguinal incision with a high section of the left spermatic cord,and a left radical orchiectomy.He experienced local relapse 1 mo postoperatively and received radiotherapy and anlotinib hydrochloride-based immunotherapy as adjuvant therapy.The patient died 3 mo after the surgery.CONCLUSION The optimal interventions for advanced-stage pleomorphic RMS patients should be investigated by more preclinical studies and clinical trials.Physicians need to be aware of the occurrence of pleomorphic RMS in unusual locations,especially when accompanied by a hydrocele testis.展开更多
We present a case of spontaneous undifferentiated/unclassified sarcoma,of a pleomorphic subtype formerly known as malignant fibrous histiocytoma(UPS/MFH),arising from the pancreas of a laboratory rat.The mass was exci...We present a case of spontaneous undifferentiated/unclassified sarcoma,of a pleomorphic subtype formerly known as malignant fibrous histiocytoma(UPS/MFH),arising from the pancreas of a laboratory rat.The mass was excised after laparotomy from a 6‐month‐old female laboratory Wistar rat.It presented a giant multilobulated mass of irregular shape,which had arisen from the pancreas and occupied almost the entire peritoneal cavity.Histologically the tumor was characterized by a highly variable morphological pattern,with frequent transitions from storiform to pleomorphic areas.An extensive immunohistochemical examination revealed no specific lines of differentiation.Immunohistochemical positivity was observed only to MIB‐1(high Ki‐67 proliferation index),vimentin and CD68 antibodies.The diagnosis was compatible with UPS/MFH.To the best of our knowledge,the present case is the first report of a spontaneous primary UPS/MFH arising from the pancreas of a laboratory rat.展开更多
BACKGROUND Myoepithelial carcinoma(MC)is a clinically rare malignancy,there is controversy regarding its etiology and its biological behavior is not fully elucidated.Extensive surgical resection is the main treatment ...BACKGROUND Myoepithelial carcinoma(MC)is a clinically rare malignancy,there is controversy regarding its etiology and its biological behavior is not fully elucidated.Extensive surgical resection is the main treatment method.We describe a case of pleomorphic adenoma(PA)with multiple postoperative recurrences after malignant transformation,and the history of the disease in this patient was more than 20 years.Complete resection during the first surgery of PA and long-term postoperative follow-up is necessary.CASE SUMMARY A 34-year-old male with PA and a history of 5 postoperative recurrences over 21 years,each surgically removed,presented 15 d ago with headache,nasal congestion,protrusion of the right eyeball and loss of vision in the right eye,with progressively worsening symptoms.The patient underwent surgery,and MC was confirmed by pathology examination.A small PA component was locally visible under light microscope.The patient had a recurrence of the tumor 2 mo after surgery and underwent surgical resection.CONCLUSION During the first operation for PA,care should be taken not to rupture the envelope to prevent tumor cell implantation,and when complete resection is not possible due to the anatomical site,postoperative radiotherapy is necessary to control the lesion and prevent infiltration and malignant transformation of the tumor to MC.Computed tomography and magnetic resonance imaging is important for establishing diagnosis and developing a treatment plan.展开更多
Undifferentiated pleomorphic sarcoma is an extremely rare malignant thyroid tumor.Thyroid sarcoma differs from common malignant thyroid tumors,such as thyroid follicular cell carcinoma.It is usually highly malignant,p...Undifferentiated pleomorphic sarcoma is an extremely rare malignant thyroid tumor.Thyroid sarcoma differs from common malignant thyroid tumors,such as thyroid follicular cell carcinoma.It is usually highly malignant,progresses rapidly,and is prone to remote metastasis.Currently,there is no standard protocol for the treatment of thyroid sarcomas,and most treatment effects are unsatisfactory.Argon-helium cryoablation is an important method of local treatment that is widely used in patients with unresectable advanced tumors.However,owing to the low incidence of thyroid sarcomas,there are no relevant literature reports on the treatment of thyroid sarcomas using cryoablation in China.This study reports the case of a patient with undifferentiated pleomorphic sarcoma of the thyroid gland who was treated with argon-helium cryoablation,and the immediate outcome was good.Based on a review of relevant literature,we discussed the effectiveness and safety of argon-helium cryoablation treatment,to provide clinical guidance and references for the treatment of patients with thyroid sarcoma.展开更多
We report a case of carcinoma ex pleomorphic adenoma of a sublingual gland in a 70-year-old man.Under a clinical diagnosis of benign salivary gland tumor,excision of the mass with the sublingual salivary gland in an e...We report a case of carcinoma ex pleomorphic adenoma of a sublingual gland in a 70-year-old man.Under a clinical diagnosis of benign salivary gland tumor,excision of the mass with the sublingual salivary gland in an en bloc fashion via an intraoral approach was performed.Histopathologically,there was a rupture of the fibrous capsule and diffuse cell-rich sheets composed of myoepithelial cells with round nuclei were also seen.Immunohistochemically,the cells that composed of cell rich sheets were positive to smooth muscle actin.Final diagnosis of myoepithelial carcinoma ex pleomorphic adenoma was made.展开更多
Introduction: Rhabdomyosarcoma (RMS) is the most common childhood soft tissue sarcoma, but it represents only a small portion of soft tissue sarcoma in adult population. There is a treatment protocol based on Intergro...Introduction: Rhabdomyosarcoma (RMS) is the most common childhood soft tissue sarcoma, but it represents only a small portion of soft tissue sarcoma in adult population. There is a treatment protocol based on Intergroup Rhabdomyosarcoma Study (IRS) that provides satisfactory results in treating RMS in children, but there is only limited evidence regarding the outcome and prognosis in extrapolating the IRS protocol to treat RMS in adults. We report a case of adult pleomorphic RMS treated with multidisciplinary approach and the results we have obtained. Case presentation: A 48-year-old woman was admitted in February 2011 due to a painful mass on her left thigh. Diagnosis of pleomorphic rhabdomyosarcoma was made by histopathology and immunohistochemistry. After multimodal treatment that includes Trans-Arterial Chemotherapy Infusion, Cryosurgery, and wide excision surgery, our patient remains disease-free as of the latest annual follow up examination on June 2017. Conclusions: The pleomorphic type of Rhabdomyosarcoma is very rare in adults and is often associated with a poor prognosis. In our case, a multidisciplinary approach with multimodal treatment provides excellent result, even after a routine follow up spanning through six years.展开更多
Pleomorphic adenoma is the most common benign parotid gland tumor. Although its local recurrence rate is known to be high, the recurrence extending to the cervical region is rare. Here we report a case of a young fema...Pleomorphic adenoma is the most common benign parotid gland tumor. Although its local recurrence rate is known to be high, the recurrence extending to the cervical region is rare. Here we report a case of a young female (25 years old) with pleomorphic adenoma of the parotid gland which showed multiple recurrences through facial to cervical regions over a span of eight years. We also discuss how this benign tumor with a high recurrence rate has been treated in other cases, and how it should be treated.展开更多
Salivary gland tumors, the second most common neoplasm of the mouth after squamous cell carcinoma, account for a significant proportion of tumors of the oral and perioral regions. An unusual case of adenoma presented ...Salivary gland tumors, the second most common neoplasm of the mouth after squamous cell carcinoma, account for a significant proportion of tumors of the oral and perioral regions. An unusual case of adenoma presented as a solitary intraoral palatine mass in a 32-year-old woman is reported here. The tumor was interpreted as an unusual pleomorphic adenoma because of the presence of exuberant squamous metaplasia, clinically mimicking squamous cell carcinoma. Moreover, the presence of cystic structures filled with keratinized material was also salient feature. Pleomorphic adenomas may occasionally display focal squamous metaplastic changes;when extensive, it presents the potential for misinterpretation of the histology as indicative of well-differentiated squamous cell carcinoma.展开更多
文摘Correspondence to:Satoru Kase.Department of Ophthalmology,Faculty of Medicine and Graduate School of Medicine,Hokkaido University,N-15,W-7,Kita-ku,Sapporo 060-8638,Japan.kaseron@med.hokudai.ac.jp AIM:To evaluate the clinical features of primary lacrimal gland pleomorphic adenoma(LGPA).METHODS:This was a 2-center,retrospective,observational study of primary LGPA patients who underwent tumor resection.Ophthalmic examinations and orbital computed tomography(CT)and/or magnetic resonance imaging(MRI)were performed.RESULTS:Totally 18 patients(10 males and 8 females,mean age 56.3±13.8y)were enrolled.Initial symptoms were unilateral proptosis in 8 patients,diplopia in 3 patients,and pressure sensation and no chief complaint in 2 patients each.The best-corrected visual acuity of the affected eye was 0.26±0.44 logMAR,and the intraocular pressure(IOP)of the affected and healthy eyes was 20.1±9.9 and 15.8±4.3 mm Hg,respectively.The difference in degree of proptosis between the affected and healthy eyes was 4.1±2.2 mm based on the Hertel ocular protrusion meter.One case had a history of breast cancer.Seventeen of 18 patients with an orbital lacrimal gland origin underwent total tumor excision by anterior/lateral orbitotomy.CONCLUSION:Orbital LGPA can complicate IOP elevation,and require total tumor excision by orbitotomy.In rare cases,systemic malignancy may complicate LGPA,and in such cases total removal of the lacrimal gland tumor should be considered at the initial surgery.
基金Supported by National Key R&D Program of China(No.2023YFC2410203)Beijing Hospitals Authority’Ascent Plan(No.DFL20190201)+1 种基金Beijing Hospitals Authority Clinical Medicine Development of Special Funding Support(No.ZLRK202503)Natural Science Foundation of Beijing(No.7222025).
文摘AIM:To examine carcinoma ex pleomorphic adenoma(CXPA)and its development to provide information for its clinical assessment and prognosis.METHODS:The clinical data of 26 patients with pathologically diagnosed CXPA were included for analysis.The patients’medical histories and data(e.g.,gender,age,eye laterality,clinical manifestations,pathologic and immunohistochemical indices,treatments,and prognosis)were recorded.RESULTS:The average age of the 26 patients was 59.6±15.7y.There was no significant difference in the gender distribution.The incidence of CXPA bone destruction was approximately 57.7%,and the incidence of optic nerve involvement and extraocular muscle involvement was approximately 15.4%and 19.2%,respectively.The most common pathological type was adenocarcinoma(34.6%),followed by ductal carcinoma(26.9%).Five patients had recurrence or metastasis(19.2%).The 5-year recurrencefree survival rate was 59.0%.There were no significant differences in survival rates among patients with different pathologic types and stages(P>0.05).Bone destruction,nerve invasion,and peripheral tissue invasion did not significantly affect survival rate(P>0.05).Surgery combined with 125I radiation therapy leads to a better survival prognosis(P<0.05).CONCLUSION:CXPA has a variety of pathologic classifications,with characteristics of bone destruction and peripheral tissue invasion.Surgery combined with ^(125)I endoradiotherapy is a preferable treatment option.However,long-term follow-up and close observation for recurrence or metastasis should be performed.
文摘Background: Mucoepidermoid carcinoma ex-pleomorphic adenoma (MECxPA) is an extremely rare salivary gland malignancy. With only nine prior reported cases, this entity represents a challenging histopathological diagnosis. Methods: We present a case of a 71-year-old male with an enlarging left neck mass over several months. CT showed both a parapharyngeal space mass and a separate level II neck mass. Results: The patient underwent resection of the left parapharyngeal mass and ipsilateral selective level II-IV lymphadenectomy. The final pathologic diagnosis was metastatic high-grade mucoepidermoid carcinoma ex-pleomorphic adenoma. Conclusions: We describe a novel presentation of high-grade mucoepidermoid carcinoma ex-pleomorphic adenoma as a metastatic parapharyngeal mass.
文摘Giant cell tumors of the pancreas come in three varieties-osteoclastic,pleomorphic,and mixed histology.These tumors have distinctive endoscopic,clinical,and cytological features.Giant cell tumors have a controversial histogenesis,with some authors favoring an epithelial origin and others favoring a mesenchymal origin.The true origin of these lesions remains unclear at this time.These are also very rare tumors but proper identification and differentiation from more common pancreatic adenocarcinoma is important.The risk factors of these tumors and the prognosis may be different from those associated with standard pancreatic adenocarcinoma.Recognition of these differences can significantly affect patient care.These lesions have a unique appearance when imaged with endoscopic ultrasound(EUS),and these lesions can be diagnosed via EUS guided Fine Needle Aspiration(FNA).This manuscript will review the endoscopic,clinical,and pathologic features of these tumors.
文摘BACKGROUND:A malignant fibrous histiocytoma,recently referred to as an undifferentiated pleomorphic sarcoma (UPS),is very rare in the liver,and only 34 cases of primary hepatic malignant fibrous histiocytoma have been reported in the English literature. METHODS:We report a rare case of a hepatic UPS presenting as a unilocular cystic lesion with respect to histopathologic features,the newly revised diagnostic criteria,and the differential diagnosis. RESULTS:A 60-year-old man presented for evaluation of epigastric pain of 7 months duration.Abdominal computed tomography revealed a hypodense,unilocular cystic mass in the right lobe of the liver,measuring 14.0×11.1 cm.A right lobectomy was performed.Grossly,the cut surface showed a single,well-circumscribed unilocular cystic tumor mass containing dark red-brown necrotic debris and blood clots, which occupied most of the mass.Microscopically,the tumor consisted of haphazardly arranged mononuclear pleomorphic tumor cells,admixed with abundant osteoclast- like multinucleated giant cells.Immunohistochemically,the tumor cells expressed vimentin only.The histopathologic and immunohistochemical findings were compatible with a UPS.The patient is alive and well 41 months after surgery without recurrence. CONCLUSIONS:Clinically,most of the hepatic UPSs are solid masses.Only two cases have presented as multilocular cystic masses.A primary hepatic UPS presenting as a unilocular cyst has never been reported.A UPS should be included in the differential diagnosis of unilocular cystic lesions in the liver.
基金Supported by the National Natural Science Foundation of China (No.30371515)
文摘AIM: To investigate C-myc, Ki-67, pan-cytokeratin, and vimentin immunohistochemical features of carcinoma ex pleomorphic adenoma(Ca-ex-PA) and pleomorphic adenoma(PA) in the lacrimal gland in order to find some clues in the differential diagnosis between them.METHODS: We reviewed microscopic slides and clinical records of 64 cases of PA and 15 cases of Ca-ex-PA in the lacrimal gland. Immunohistochemical antibodies for C-myc, Ki-67, pan-cytokeratin, and vimentin were employed.RESULTS: Median age of PA was 43.2 y(from 21 to 75). The 35 patients(54.7%) were male and 29 patients(45.3%) were female. For the PAs, the average positivity of C-myc was 4.6%;the average proliferation index of Ki-67 was 3.2%;pan-cytokeratin was positive in ductal cells, and vimentin was positive in myoepithelial cells. Median age of Ca-ex-PA was 54.3 y(from 26 to 76). There were 7 male patients(46.7%) and 8 female patients(53.3%). Among 15 Ca-ex-PAs, there were 6 myoepithelial carcinomas, 4 adenocarcinomas, 3 epithelial-myoepithelial carcinomas, and 2 squamous cell carcinomas. For the Ca-ex-PAs, the average positivity of C-myc was 36.4%;the average proliferation index of Ki-67 was 29.2%;pan-cytokeratin was positive in all cases, and vimentin was positive in myoepithelial carcinomas.CONCLUSION: PA has a lower positivity of C-myc and Ki-67, while Ca-ex-PA had a higher positivity of these two biomarkers. These four biomarkers as a set could provide valuable clues in the differential diagnosis between Ca-exPA and PA. Our results indicate that the activation of C-myc could play an important role in the pathogenesis of Ca-exPA and PA.
基金Supported by the Natural Science Foundation of Guangdong Province,China,No.2020A1515010119.
文摘BACKGROUND Pleomorphic adenoma(PA)is the most common benign tumor that occurs in the salivary glands;however,tracheobronchial PA is rarely observed.To the best of our knowledge,fewer than 50 cases have been reported in the literature.We report a 49-year-old woman who had been treated for asthma for 2 years before being diagnosed with PA of the trachea.CASE SUMMARY A 49-year-old woman was referred to our hospital due to dyspnea upon exertion and chronic cough with wheezing for 2 years.Laboratory tests showed an elevated white blood cell count,absolute neutrophil count,and percentage of neutrophils.A chest computerized tomography scan showed a well-defined,softtissue density lesion measuring 2.4 cm×2.1 cm in the lower trachea.Flexible bronchoscopy revealed that nearly 90%of the tracheal lumen was obstructed.The histopathological and immunohistochemistry features suggested PA of the trachea.Furthermore,we review the characteristics of 29 patients with tracheobronchial PA over the last 30 years.CONCLUSION Tracheobronchial PA occurs without gender predominance,mostly in the lower or upper trachea,and has a low recurrence rate.The median age at diagnosis is 48 years.The most common symptoms are cough,stridor,dyspnea,and wheezing.
文摘BACKGROUND Carcinoma ex pleomorphic adenoma (CXPA) is defined as a malignant salivary gland tumor arising from a primary or recurrent pleomorphic adenoma.Only three cases of CXPA of the trachea have been reported in the literature.CASE SUMMARY We report a case of tracheal CXPA in a 55-year-old woman,who presented with a more than 3-mo history of progressive dyspnea.Computed tomography of the neck and thorax revealed an inhomogeneous,broad-based lesion arising from the tracheal wall on the right side.Endoscopy revealed a subglottic neoplasm causing up to 90% luminal stenosis.The tumor was resected using a highfrequency electrosurgical snare combined with argon plasma coagulation.Histopathology and immunohistochemistry revealed that the tumor was a CXPA of the trachea.CONCLUSION We report the fourth case of tracheal CXPA,and present the first instance of resection of CXPA using high-frequency electrosurgical snare and laser ablation.We also discuss the pathogenesis,diagnosis,histopathology,and systemic therapy of this rare disease.
文摘BACKGROUND Pleomorphic lipoma (PL) is a rare benign mesenchymal tumor occurring predominantly in middle-aged and elderly men. It is typically found in the subcutaneous tissue of the posterior neck, back, and shoulders. There have also been a few reported cases in the face, scalp, and upper extremities. Currently, there is no report on its occurrence in the anterior mediastinum. CASE SUMMARY Herein, we report the case of a 67-year-old woman diagnosed with PL in the anterior mediastinum. The tumor was removed by thoracoscopic surgery. There was no recurrence during the 24-mo follow-up period, and the prognosis was good. Most PL are located on the skin surface. However, they may also occur within the body, even in the mediastinum. CONCLUSION PL occurring in the anterior mediastinum is rare, and it may be differentiated from other tumors.
基金Beijing Hospitals Authority’Ascent Plan (No.DFL20190201).
文摘AIM:To monitor the prognosis of patients with lacrimal gland pleomorphic adenoma(LGPA)following surgical resection within the past 10y.The factors affecting patient prognosis are analyzed and the experience of surgical treatment is summarized.METHODS:In this retrospective,comparative case series,clinical records from 109 cases of LGPA treated chiefly at the same institution between November 2009 and May 2019 were reviewed.All 109 patients underwent surgery,histopathological examination,and imaging examination.For patients who underwent surgery for the first time,LGPA could be resected completely,including tumor and capsule tissues,using a surgical approach via the eyebrow arch or double eyelid crease.RESULTS:The ratio of males to females was 1:1.60,the ages ranged from 19 to 74 years old with a mean age of 43.64±13.07 years old,and the ratio of left to right eyes was 1:1.37.A total of 109 patients underwent surgical excision and five of these收稿日期radiotherapy after malignant transformation.Of these patients,15 were lost to follow up within the April 1,2020 deadline and 1 was diagnosed as a recurring pathology.The 5-year recurrence rate for 86 patients who underwent initial surgery was 7.27%.Single factor analysis revealed that the course of disease,bone destruction,invasion of surrounding tissues,tumor size,capsule integrity,and expression of Ki-67 were statistically significant(P<0.05).Binomial Logistic regression analysis showed that capsule integrity was a risk factor influencing recurrence(P=0.008).CONCLUSION:LGPA has a risk of recurrence and potential for malignant transformation.Complete removal of the tumor and capsule in the initial surgery is a key factor in preventing recurrence.
文摘BACKGROUND Papillary thyroid cancer(PTC)is the most common malignant tumor of the thyroid.However,the coexistence of PTC and sarcoma in one patient is rare.In this article,we report the case of a patient who presented with both PTC and undifferentiated pleomorphic sarcoma(UPS),which has not been previously reported in the online Medline database(PubMed).CASE SUMMARY A 71-year-old man was admitted to our hospital for a mass on the right side of his neck for one month,which rapidly enlarged within 2 wk with distending pain.The patient was diagnosed with a thyroid malignancy by fine-needle aspiration and underwent total thyroidectomy and bilateral central lymph node dissection.Histology and immunohistochemistry revealed features of both PTC and UPS.The thyroid cancer 8 gene detection kit results showed BRAF and telomerase reverse transcriptase mutations.The disease progressed rapidly,and the patient died four months after surgery from extensive lung metastasis.CONCLUSION Our report highlights the patient’s pathological characteristics and related genetic mutations.Due to the rapid development and poor prognosis of cooccurring PTC and sarcoma,it is important for clinical physicians and pathologists to raise awareness of this type of tumor.
文摘·AIM: To detect proteomic differences in tears between adenoid cystic carcinoma(ACC) and pleomorphic adenoma(PA).·METHODS: Tear samples were collected from 4 patients with ACC, 5 with PA, and 4 control cases. Label-free analysis and parallel reaction monitoring(PRM) were used to screen and validate the tear proteome. Gene Ontology(GO) annotation and Kyoto Encyclopedia of Genes and Genomes(KEGG) were conducted for bioinformatics analysis.·RESULTS: In total, 1059 proteins in tear samples were identified by label-free analysis. Between ACC and PA, 415 differentially expressed proteins were detected. Based on the GO annotation, enzyme regulator activity and serinetype endopeptidase inhibitor activity in the molecular function category, blood microparticle and extracellular matrix in the cellular component category, and response to nutrient levels in the biological process category were most predominant. By KEGG pathway annotation, the different proteins between ACC and PA mainly par ticipated in complement and coagulation cascades, amoebiasis, African trypanosomiasis and cholesterol metabolism. Eight proteins with mostly significant differences were verified by PRM, and five proteins with more than 10-fold increases in ACC compared with PA, including integrin β, α-2-macroglobulin, epididymal secretory sperm binding protein Li 78p, RAB5C, and complement C5, were identified.·CONCLUSION: The combined tools of label-free analysis and PRM are very effective and efficient, especially for samples such as tears. Some proteomic dif ferences in tears between ACC and PA are identified and these protein candidates may be specific biomarkers for future exploration.
文摘BACKGROUND Pleomorphic rhabdomyosarcoma(RMS)of the spermatic cord is a group of rare neoplasms,and a secondary hydrocele testis occasionally occurs.The misdiagnosis of paratesticular mass may lead to a therapeutic delay.CASE SUMMARY A 79-year-old man presented to our clinic complaining of a 1-mo history of painless scrotal swelling.Physical examination revealed approximately a 15 cm×10 cm×5 cm inguinal mass with limited mobility.Contrast-enhanced magnetic resonance imaging showed a hydrocele testis,several enlarged inguinal lymph nodes,and a heterogeneously enhanced lesion with a relatively well-defined margin in the left inguinal region.Due to the imaging findings,he was diagnosed with pleomorphic RMS and received a wide resection of the mass,an inguinal incision with a high section of the left spermatic cord,and a left radical orchiectomy.He experienced local relapse 1 mo postoperatively and received radiotherapy and anlotinib hydrochloride-based immunotherapy as adjuvant therapy.The patient died 3 mo after the surgery.CONCLUSION The optimal interventions for advanced-stage pleomorphic RMS patients should be investigated by more preclinical studies and clinical trials.Physicians need to be aware of the occurrence of pleomorphic RMS in unusual locations,especially when accompanied by a hydrocele testis.
文摘We present a case of spontaneous undifferentiated/unclassified sarcoma,of a pleomorphic subtype formerly known as malignant fibrous histiocytoma(UPS/MFH),arising from the pancreas of a laboratory rat.The mass was excised after laparotomy from a 6‐month‐old female laboratory Wistar rat.It presented a giant multilobulated mass of irregular shape,which had arisen from the pancreas and occupied almost the entire peritoneal cavity.Histologically the tumor was characterized by a highly variable morphological pattern,with frequent transitions from storiform to pleomorphic areas.An extensive immunohistochemical examination revealed no specific lines of differentiation.Immunohistochemical positivity was observed only to MIB‐1(high Ki‐67 proliferation index),vimentin and CD68 antibodies.The diagnosis was compatible with UPS/MFH.To the best of our knowledge,the present case is the first report of a spontaneous primary UPS/MFH arising from the pancreas of a laboratory rat.
文摘BACKGROUND Myoepithelial carcinoma(MC)is a clinically rare malignancy,there is controversy regarding its etiology and its biological behavior is not fully elucidated.Extensive surgical resection is the main treatment method.We describe a case of pleomorphic adenoma(PA)with multiple postoperative recurrences after malignant transformation,and the history of the disease in this patient was more than 20 years.Complete resection during the first surgery of PA and long-term postoperative follow-up is necessary.CASE SUMMARY A 34-year-old male with PA and a history of 5 postoperative recurrences over 21 years,each surgically removed,presented 15 d ago with headache,nasal congestion,protrusion of the right eyeball and loss of vision in the right eye,with progressively worsening symptoms.The patient underwent surgery,and MC was confirmed by pathology examination.A small PA component was locally visible under light microscope.The patient had a recurrence of the tumor 2 mo after surgery and underwent surgical resection.CONCLUSION During the first operation for PA,care should be taken not to rupture the envelope to prevent tumor cell implantation,and when complete resection is not possible due to the anatomical site,postoperative radiotherapy is necessary to control the lesion and prevent infiltration and malignant transformation of the tumor to MC.Computed tomography and magnetic resonance imaging is important for establishing diagnosis and developing a treatment plan.
文摘Undifferentiated pleomorphic sarcoma is an extremely rare malignant thyroid tumor.Thyroid sarcoma differs from common malignant thyroid tumors,such as thyroid follicular cell carcinoma.It is usually highly malignant,progresses rapidly,and is prone to remote metastasis.Currently,there is no standard protocol for the treatment of thyroid sarcomas,and most treatment effects are unsatisfactory.Argon-helium cryoablation is an important method of local treatment that is widely used in patients with unresectable advanced tumors.However,owing to the low incidence of thyroid sarcomas,there are no relevant literature reports on the treatment of thyroid sarcomas using cryoablation in China.This study reports the case of a patient with undifferentiated pleomorphic sarcoma of the thyroid gland who was treated with argon-helium cryoablation,and the immediate outcome was good.Based on a review of relevant literature,we discussed the effectiveness and safety of argon-helium cryoablation treatment,to provide clinical guidance and references for the treatment of patients with thyroid sarcoma.
文摘We report a case of carcinoma ex pleomorphic adenoma of a sublingual gland in a 70-year-old man.Under a clinical diagnosis of benign salivary gland tumor,excision of the mass with the sublingual salivary gland in an en bloc fashion via an intraoral approach was performed.Histopathologically,there was a rupture of the fibrous capsule and diffuse cell-rich sheets composed of myoepithelial cells with round nuclei were also seen.Immunohistochemically,the cells that composed of cell rich sheets were positive to smooth muscle actin.Final diagnosis of myoepithelial carcinoma ex pleomorphic adenoma was made.
文摘Introduction: Rhabdomyosarcoma (RMS) is the most common childhood soft tissue sarcoma, but it represents only a small portion of soft tissue sarcoma in adult population. There is a treatment protocol based on Intergroup Rhabdomyosarcoma Study (IRS) that provides satisfactory results in treating RMS in children, but there is only limited evidence regarding the outcome and prognosis in extrapolating the IRS protocol to treat RMS in adults. We report a case of adult pleomorphic RMS treated with multidisciplinary approach and the results we have obtained. Case presentation: A 48-year-old woman was admitted in February 2011 due to a painful mass on her left thigh. Diagnosis of pleomorphic rhabdomyosarcoma was made by histopathology and immunohistochemistry. After multimodal treatment that includes Trans-Arterial Chemotherapy Infusion, Cryosurgery, and wide excision surgery, our patient remains disease-free as of the latest annual follow up examination on June 2017. Conclusions: The pleomorphic type of Rhabdomyosarcoma is very rare in adults and is often associated with a poor prognosis. In our case, a multidisciplinary approach with multimodal treatment provides excellent result, even after a routine follow up spanning through six years.
文摘Pleomorphic adenoma is the most common benign parotid gland tumor. Although its local recurrence rate is known to be high, the recurrence extending to the cervical region is rare. Here we report a case of a young female (25 years old) with pleomorphic adenoma of the parotid gland which showed multiple recurrences through facial to cervical regions over a span of eight years. We also discuss how this benign tumor with a high recurrence rate has been treated in other cases, and how it should be treated.
文摘Salivary gland tumors, the second most common neoplasm of the mouth after squamous cell carcinoma, account for a significant proportion of tumors of the oral and perioral regions. An unusual case of adenoma presented as a solitary intraoral palatine mass in a 32-year-old woman is reported here. The tumor was interpreted as an unusual pleomorphic adenoma because of the presence of exuberant squamous metaplasia, clinically mimicking squamous cell carcinoma. Moreover, the presence of cystic structures filled with keratinized material was also salient feature. Pleomorphic adenomas may occasionally display focal squamous metaplastic changes;when extensive, it presents the potential for misinterpretation of the histology as indicative of well-differentiated squamous cell carcinoma.
