Immunohistochemical study of pleomorphic adenoma of lacrimal gland.

Immunohistochemical examinations of lacrimal gland specimens were carried out with monoclonal antibodies to S-100 protein and glial fibrillary acidic protein (GFAP) in 3 cases of normal tissue, 2 cases of hypertrophy and 3 cases of pleomorphic adenoma of the lacrimal gland. In specimens of normal lacrimal gland tissue, S-100 protein was identified in myoepithelial cells and ductal epithelia, but GFAP was not identified in any part of the gland. In specimens of lacrimal gland hypertrophy, the findings were identical. In pleomorphic adenoma of the lacrimal gland, asteroid cells in the myxoid and/or chondroid areas were strongly stained by antibodies to both S-100 protein and GFAP. In the solid areas of pleomorphic adenoma specimens, S-100 protein-positive fusiform or round cells and GFAP-positive round cells were observed. It was thought that S-100 protein-positive cells could have originated from myoepithelial cells and that GFAP could be a tumor-associated antigen. These findings agreed with recent immunohistochemical findings in pleomorphic adenoma of the salivary gland. It was speculated that pleomorphic adenoma of the lacrimal gland could cause mesenchymal metaplasia of the myoepithelial cells, as happens in pleomorphic adenoma of the salivary gland.     

Immunohistochemical study of pleomorphic adenoma of the lacrimal gland

We performed immunohistochemical examinations in 1 hypertrophy and 3 pleomorphic adenomas of the lacrimal glands with monoclonal antibodies to S-100 protein and GFAP (glial fibrillary acidic protein). It was thought that hypertrophy of the lacrimal gland would be cytological equivalent to normal lacrimal gland tissue because of the lack of cytological atypia except when accompanied by lymphoid infiltration. In hypertrophy of lacrimal gland, S-100 protein was identified in myoepithelial cells and parts of the ductal epithelia, but GFAP was not identified in any part. In pleomorphic adenomas of lacrimal glands, asteroid cells of myxoid and/or chondroid areas were strongly stained with both antibodies to S-100 protein and GFAP. In solid areas of pleomorphic adenomas, S-100 protein-positive fusiform or round cells and GFAP-positive round cells were observed. It was thought that S-100 protein-positive cells could have originated from myoepithelial cells and GFAP could be a tumor-associated antigen. The results coincided with recent immunohistochemical findings of pleomorphic adenoma of the salivary gland. It was suspected that pleomorphic adenoma of lacrimal gland could develop from mesenchymal metaplasia of myoepithelial cells as in the case of pleomorphic adenoma of salivary gland.     

Clinical analysis of 109 cases of lacrimal gland pleomorphic adenoma

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 received 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.     

Mucoepidermoid carcinoma ex pleomorphic adenoma of the lacrimal gland: A rare presentation

Carcinoma ex pleomorphic adenoma in lacrimal gland is a rare entity unlike its salivary gland counterpart. This rare tumor poses a diagnostic challenge to clinicians as pre-operative diagnosis is difficult and diagnosis is only by careful pathological assessment. We report this uncommon lesion in a 62-year-old lady, wherein the malignant component was mucoepidermoid carcinoma. The elderly patient remained clinically and radiologically free of the tumor for two years after complete excision of the tumor but computed tomography at the end of two and a half years showed a recurrent lesion in the region of the lacrimal gland. This makes long term follow up of patients with these rare lacrimal tumors imperative with a minimum period of at least five years.     

In situ adenocarcinoma ex pleomorphic adenoma of the lacrimal gland

A case of an in situ adenocarcinoma of the lacrimal gland that developed in a recurring pleomorphic adenoma is presented. Six years after incomplete resection of a pleomorphic adenoma, the patient complained of a progressive swelling of her right upper eyelid without pain or diplopia. An orbital computerized tomography scan showed an inhomogeneous mass in the right lacrimal gland region without bone involvement. After local excision of the in situ adenocarcinoma ex pleomorphic adenoma, the tumour has been in control for over 6 years.     

Carcinosarcoma of the lacrimal gland arising from a pleomorphic adenoma

PURPOSE: To report a case of the lacrimal gland tumor diagnosed as carcinosarcoma (true mixed tumor) arising from a pleomorphic adenoma. DESIGN: Interventional case report. METHODS: An 80-year-old Japanese woman was referred with a well-circumscribed tumor in the lacrimal fossa. RESULTS: The tumor was excised, but the deepest portion remained because of heavy bleeding. Histopathologically, the tumor consisted of osteosarcomatous cells. No epithelial or myoepithelial component was identified by immunohistochemistry. Two months later, the tumor progressed further and was excised completely with lateral orbitotomy. The tumor consisted primarily of osteosarcomatous component but also had epithelial components including carcinoma and pleomorphic adenoma. Examinations did not detect local recurrence and metastasis at 10 post-operative months. CONCLUSION: The tumor was diagnosed as true carcinosarcoma of the lacrimal gland arising from a pleomorphic adenoma. When lacrimal gland tumors containing sarcomatous components are encountered, carcinosarcoma should be included in the differential diagnosis.     

Pleomorphic adenoma of the lacrimal gland in a nine-year-old child

A case of a 9-year-old child with pleomorphic adenoma of the lacrimal gland is described. This tumour is usually found in adults; cases younger than 10 years of age have proved to be extremely rare. Because of its age distribution, pleomorphic adenoma of the lacrimal gland is usually overlooked as a possible cause of unilateral proptosis in children. Also, the history of disease provided by the family may be incorrect, possibly leading to misdiagnosis by the ophthalmologist.     

Unusual presentations of pleomorphic adenoma and adenoid cystic carcinoma of the lacrimal gland

Purpose: To report two atypical cases of pleomorphic adenoma and adenoid cystic tumours of the lacrimal gland. Methods: Two case reports are presented. The first is of a 65-year-old female with a long history of right hypoglobus with sudden recent worsening. Computed tomography (CT) showed a round, well-defined lesion in the fossa for the lacrimal gland with an anterior hypodense exten- sion suggestive of possible malignancy in a pleomorphic adenoma. The tumour in the second case, a 35-year-old male, was diagnosed after presentation following a relatively minor periorbital injury. The smooth rounded mass on CT scan was suggestive of a benign lacrimal gland tumour. Results: The lesion in case 1 was excised with a diagnosis of haemorrhage within a pleomorphic adenoma. The lesion in case 2 was excised with a diagnosis of adenoid cystic carcinoma of the lacrimal gland with pseudocapsule. Conclusions: Haemorrhagic cyst developing beneath the pseudocapsule of a pleomorphic adenoma should be considered in the differential diagnosis of secondary development of malignancy in a pleomorphic adenoma. Adenoid cystic tumours of the lacrimal gland can present with a pseudocapsule.     

Slow-growing large pleomorphic adenoma of ectopic lacrimal gland tissue in the upper eyelid

A few cases of pleomorphic adenoma of an ectopic lacrimal gland involving the deep orbit and lateral canthus have been previously reported. A 75-year-old female with a slow-growing, large pleomorphic adenoma arising from ectopic lacrimal gland tissue in the left upper eyelid is described in the present case report.     

Pleomorphic adenoma of a subconjunctival ectopic lacrimal gland

This report describes a case of pleomorphic adenoma of an ectopic lacrimal gland arising subconjunctivally in the lateral fornix in a 13-year-old girl. The tumor was removed surgically in toto with the capsule. This is probably the first reported case.     

Giant pleomorphic adenoma of the lacrimal gland: A surgical challenge

Pleomorphic adenoma is the most common epithelial neoplasm of the lacrimal gland. The tumor typically presents with a superotemporal mass with inferonasal displacement of the globe. They generally measure less than 3 cm in size and can be removed comfortably via a lateral orbitotomy approach. Pleomorphic adenoma left unattended for a long period grows up to humongous proportions and poses a surgical challenge both for its complete removal and globe salvage. We report a rare case of pleomoprphic adenoma of the lacrimal gland in an adult male, who did not seek any medical advice for 20 years allowing the tumor to expand enormously in all dimensions, with complete obscuration of the globe. The tumor was excised completely and globe could be salvaged.     

泪腺多形性腺瘤组织病理学研究

背景泪腺多形性腺瘤是眼眶部最常见的泪腺上皮性肿瘤,组织学上虽表现为良性,但具有易复发和恶变的特点。其组织学表现呈现多样性,常具有与其他泪腺上皮性肿瘤相同的组织学结构。目的探讨泪腺多形性腺瘤不同病理亚型的组织学特点及其与肿瘤复发的关系,并提高对其组织病理学类型的认识。方法收集1966--2010年在中山大学中山眼科中心病理室存档的181例泪腺多形性腺瘤的基本临床资料和组织石蜡标本,并重新切片行苏木精一伊红染色,观察肿瘤内各组分形态、结构特点及其与肿瘤复发的关系。结果181例泪腺多形性腺瘤可分为上皮细胞为主型90例（49．7％）、问质丰富型38例（21％）以及中间型（上皮和间质成分比例相当）53例（29．3％）。86．5％的复发泪腺多形性腺瘤为间质丰富型,显著高于其他两种类型（χ2=120．896,P=0．000）。在159例有包膜的肿瘤中,138例（86．7％）可见包膜较薄（局部厚度〈20Ixm）,尤其在间质丰富型肿瘤中往往有大片的薄包膜区域,间质丰富型包膜内肿瘤卫星灶或芽状突起比例显著高于其他两种类型（χ2=80．715,P=0．000）。肿瘤上皮成分包含立方形、鳞状上皮、浆细胞样、梭形、透明样和基底样细胞,其中立方形细胞最为常见,鳞状细胞次之。上皮细胞排列方式多样,有导管状、囊状、片状实体及小梁网状结构,其中以导管状结构最为常见。肿瘤内的间质成分包括黏液基质、玻璃样变组织、脂肪细胞以及软骨或钙化组织,其中黏液成分最为常见。结论复发性泪腺多形性腺瘤以间质丰富型常见,其包膜受侵犯可能是肿瘤复发的原因之一。掌握泪腺多形性腺瘤上皮及间质成分的多种形态及排列结构,有助于病理医师的正确诊断。     

Cystic carcinoma ex pleomorphic adenoma of the lacrimal gland

In a 48-year-old male patient, a well-circumscribed, round tumor in the lacrimal fossa was detected by CT. During total excision of the tumor, a wall of the tumor ruptured, resulting in leakage of the fluid contents. The wall of the tumor formed a cyst that contained residual yellowish fluid. Histologically, the cyst wall included an adenocarcinomatous component and a focus of pleomorphic adenoma. The tumor was diagnosed as a carcinoma ex pleomorphic adenoma of the lacrimal gland. The carcinoma cells showed positive immunoreactivity for androgen receptor and BRST-2, indicating that the tumor was equivalent to a salivary duct carcinoma. Postoperatively, the patient underwent involved field irradiation. This case demonstrates that a carcinoma ex pleomorphic adenoma of the lacrimal gland may develop in a cyst.     

Pleomorphic adenoma of a deep orbital ectopic lacrimal gland

Ectopic lacrimal gland, being one of the choristomas, is comprised of lacrimal gland tissue outside the lacrimal gland fossa in the fronto-lateral part of the orbital roof. Ectopic lacrimal gland is a rare condition where the gland may be found in the orbit, eyelids, ocular adnexa or within the globe. Neoplastic transformation of such tissue may occur. A sixty-two-year old male patient presented with right eye proptosis and slight nasal displacement of the globe. Computerized tomography scan revealed a well-defined hypodense lesion of size 19 x 18 x 20 mm supero-lateral to lateral rectus muscle, with mild proptosis and thinning of the right lateral orbital wall. Excisional biopsy was performed through a lateral orbitotomy approach. A well circumscribed globular mass was removed from the right orbit, well behind the fossa for the lacrimal gland in the retrobulbar space. Histopathology was suggestive of pleomorphic adenoma of lacrimal gland. Pleomorphic adenoma is an epithelial tumor of the lacrimal gland which is extremely rare from an ectopic lacrimal gland and only few cases have been reported in literature till date.     

Pleomorphic adenoma (formerly chondroid syringoma) of the eyelid margin with a pseudocystic appearance

Among the adenomas and adenocarcinomas spawned by the adnexal glands of the eyelids, pleomorphic adenoma (also referred to as benign mixed tumor or chondroid syringoma in dermatopathology) is among the rarest. Pleomorphic adenomas (PAs) can arise from sweat glands in the dermis of the eyelid skin and must be distinguished from those of the accessory lacrimal glands of Krause and Wolfring. We describe an eyelid margin skin PA that appeared clinically to be a cyst and was not associated with an accessory lacrimal gland. Histopathologically, the lesion was circumscribed but nonencapsulated and composed of branching ductular structures with a double layer of epithelial cells set in a myxoid and sclerotic stroma that did not contain cartilage. The outer ductular (myoepithelial) cells delaminated to populate the stroma. Histochemistry disclosed abundant extracellular mucopolysaccharides that conferred the “cystic” character clinically. The inner ductular cells were uniformly positive for cytokeratin 7 and focally for gross cystic fluid disease protein-15, an apocrine marker. The inner and outer ductular cells were negative for alpha-smooth muscle actin. These pathologic findings support an apocrine (gland of Moll) origin for this tumor, which is consistent with the fact that there are no eccrine glands at the eyelid margin.     

原发性泪腺上皮性肿瘤患者97例诊治分析

目的　探讨泪腺上皮性肿瘤的临床表现、影像学特征、诊断方法及治疗原则。方法　对２００３年１月至２０１４年１０月在河南省眼科研究所就诊的９７例泪腺上皮性肿瘤的临床表现、影像学检查、治疗方法、术后视力、及随访情况等临床资料进行回顾性分析。采用前路开眶１１例,常规外侧开眶１９例,改良“Ｓ”形皮肤切口并外侧开眶６７例。结果　９７例泪腺上皮性肿瘤中,泪腺多行性腺瘤６５例（６７．０１％）,腺样囊性癌１６例（１６．４９％）,多形性腺癌１２例（１２．３７％）,腺癌２例（２．０６％）,黏液表皮样癌１例（１．０３％）,肌上皮瘤１例（１．０３％）。术后视力提高３７例,无变化５４例,下降６例。１３例出现眼球内陷,早期上睑下垂１１例,４例出现外展运动受限、水平复视。术后６个月随访:上睑下垂４例;１例出现永久性外展不足、水平复视。结论　泪腺上皮性肿瘤具有典型的临床表现和特征性的影像学改变,术前通过准确的定位来选择适宜的手术进路,可顺利并完整地切除肿瘤,避免肿瘤复发和出现严重并发症。     

Posttraumatic benign pleomorphic adenoma of the lacrimal gland

A 55-year-old man presented with a painless and slowly developing mass in the right superior lateral eyelid region. He had sustained periorbital blunt injury about 4 years previously. The mass was observed several months later in the persisting traumatic tumefaction region. Computed tomography showed a cystic soft-tissue mass with central low density and peripheral enhancement over the upper anterior quadrant of the right orbit. Lateral orbitotomy was performed to remove the lesion. Histopathologic examination showed proliferation of epithelial and myoepithelial cells arranged in ductules and nests in the myxoid stroma. Benign pleomorphic adenoma of the lacrimal gland associated with traumatic tumefaction, as demonstrated in our patient, may be rare.     

Atypical presentations of pleomorphic adenoma of the lacrimal gland

PURPOSE: To report 3 cases of pleomorphic adenoma of the lacrimal gland with atypical features. METHODS: The medical records, radiographic imaging, operative reports, and tumor histopathology of 3 patients with unusual presentations of pleomorphic adenoma of the lacrimal gland were reviewed. RESULTS: All 3 patients were females ranging in age from 18 to 64 years. The first patient presented with abrupt orbital inflammation mimicking orbital cellulitis. The second patient presented with a painful subcutaneous nodule. The last patient demonstrated calcification with bony erosion on orbital imaging. Orbitotomy was performed in all cases. Histopathologic evaluation of all specimens with light microscopy was consistent with pleomorphic adenoma of the lacrimal gland. CONCLUSIONS: Pleomorphic adenoma is the most common epithelial tumor of the lacrimal gland. A higher degree of suspicion must be present to make the correct diagnosis in cases with atypical features. Pleomorphic adenoma may present abruptly with orbital inflammation mimicking orbital cellulitis, as a painful subcutaneous nodule, or demonstrate calcification with bony erosion on orbital imaging. Complete surgical excision of the lesion within its pseudocapsule is recommended to prevent local recurrences and, although less likely, the possibility of malignant transformation.     

泪腺多形性腺瘤ras癌基因产物表达与DNA含量的关系

目的:泪腺多形性腺瘤是常见的泪腺肿瘤,约占上皮性肿瘤的50％。手术后常有复发,恶性变也不少见。以往的研究表明,肿瘤的发生发展与癌基因产物的异常表达有关。本研究的目的是观察泪腺多形性腺瘤是否存在有P~(21ras)的异常表达。方法:研究采用免疫组化ABC法测定5例正常泪腺组织及32例多形性腺瘤P~(21ras)的表达,抗体选用单克隆抗体F—132—62。肿瘤细胞核DNA含量测定采用计算机图像分析技术。结果:正常泪腺组织均染色阴性,12例肿瘤染色阳性。14例肿瘤细胞核DNA含量增加,其倍体分布类型呈双峰及多峰型。P~(21ras)表达与肿瘤细胞DNA含量倍体分布类型明显相关,在P~(21ras)染色阳性的肿瘤,DNA含量倍体分布类型主要为双峰及多峰型,经精确概率x~2检验,差异有显著性,P<0.05。结论:本研究的结果提示,泪腺多形性腺瘤有P~(21ras)的增强表达,P~(21ras)的异常表达与肿瘤的发展有关。眼科学报 1996;12;54—57。     

泪腺多形性腺瘤或癌DNA含量和P^53基因产物表达的定量分析

应用流式细胞计量术和免疫荧光染色技术，对１８例多形性腺瘤和６例多形性腺癌细胞的ＤＮＡ含量及Ｐ５３基因产物表达进行定量测定，探讨其在该肿瘤病理学诊断中的意义。结果表明，多形性腺瘤具有正常泪腺细胞的ＤＮＡ二倍体含量；多形性腺癌以ＤＮＡ异倍体为特征；Ｐ５３...