Papillary oncocytoma of the eyelid. A previously undescribed tumor of apocrine gland origin

Oncocytomas are uncommon tumors arising within the ductular cell lining of glandular structures. The oncocytic cells are characteristically large and rich in eosinophilic cytoplasm. Electron microscopic studies show densely packed mitochondria with irregular, whorled, or fragmented cristae. Ocular adnexal oncocytomas have been reported to arise in the caruncle, lacrimal gland, and lacrimal sac. The authors recently encountered an oncocytic tumor occurring in a most unusual location, the skin of the medial lid margin and commissure. Microscopically, the lesion was papillary and cystic in architecture, and arose from an adjacent apocrine gland of the eyelid margin (gland of Moll). Electron microscopic studies showed that the tumor cells were packed with malformed mitochondria to the exclusion of other organelles. This is the first report documenting an oncocytic lesion arising in the skin from an adnexal gland.     

Papillary hidradenoma of the eyelid margin: clinical and immunohistochemical observations further supporting an apocrine rather than an eccrine origin

A 46-year-old woman was evaluated for a “recurring papilloma” of the left medial upper eyelid margin. Beneath the papillary lesion medial to the punctum was a 5-mm diameter cutaneous mass thought to be cystic. After excisional biopsy, histopathologic analysis documented the presence of an epidermal keratinizing squamous papilloma surmounting a circumscribed dermal papillary hidradenoma composed of deeply eosinophilic columnar cells. Additionally, there was intraductal proliferation of tumor extending toward a subclinical poral opening through the epidermis. Immunohistochemistry proved the apocrine nature of the benign, non-cystic lesion by virtue of its nuclear androgen receptor and cytoplasmic gross-cystic disease fluid protein-15 positivity, along with its smooth muscle actin–positive myoepithelial layer. This and prior cases establish that apocrine tumors, both benign and malignant, are strictly localized at or near the eyelid margin where only apocrine glands are found. These tumors are more often papillary than solid adenomas, and most exceptionally can be malignant. We review the differential diagnosis of simulating eccrine eyelid tumors. We recommend wide local excision for benign lesions, in view of possible intraductal extension that can be eccentric to the main tumor and the miniscule potential for malignant transformation.     

Hidradenoma papilliferum of the upper eyelid arising from the apocrine gland of Moll

Hidradenoma papilliferum is derived from apocrine sweat glands and was originally described as a tumor located in the anogenital area. The authors recently examined a 78-year-old man with a hidradenoma papilliferum at the margin of the upper eyelid. While the tumor was located in the dermis, serial sections showed a connection to the epidermis and neoplastic cells partially replacing a gland of Moll. There were glandular structures with luminal cells that showed apical decapitation-type secretion, indicating the apocrine origin of the tumor. The luminal cells also contained granules that stained with periodic acid-Schiff (PAS) stain and were diastase resistant. Results of examination of the duct-forming cells by transmission electron microscopy showed adluminal villi, focal decapitation secretion, luminal cytoplasmic debris, and electron-dense cytoplasmic secretory granules. Because the apocrine glands of Moll are localized adjacent to the eyelid cilia, hidradenoma papilliferum and other tumors of apocrine origin should be considered in the clinical setting of a mass at the eyelid margin.     

A rare case of atypical chondroid syringoma of the lower eyelid and review of the literature

A 46-year-old man presented with a painless firm mass in the eyelid margin of the left lower eyelid, which had been present for 9 years. Biopsy nine years previously had not established a diagnosis. We performed excisional biopsy of the lesion and lateral advancement skin flap for reconstruction of the skin defect. Histopathologic and immunohistochemical findings were consistent with atypical chondroid syringoma with eccrine differentiation. The systemic metastatic work-up was negative, and no recurrence or metastasis was present at 30-month follow-up. Chondroid syringoma is a rare skin tumor that occurs, very rarely, in the eyelids, especially the lower eyelid. Only 26 cases of chondroid syringoma in the periorbital area, including our case, have been reported, with various characteristics. Chondroid syringomas are classified as two types, the apocrine type characterized by tubular and cystic branching lumina lined by two layers of epithelial cells, and the eccrine type, which has small tubular lumina lined by a single layer of epithelial cells. The tumor can have benign, atypical, and malignant variants. Our patient had the atypical variant, which is characterized by benign cytological features, as well as the presence of infiltrative margins and/or satellite nodules. Even benign chondroid syringomas, if incompletely excised, can recur with malignant transformation; thus, complete excision and regular follow-up is recommended for all chondroid syringomas.     

Relationship between eyelid margin irregularity and meibomian gland dropout

PurposeTo examine the relationship between lid margin abnormalities and meibomian gland loss in infrared meibography.MethodsThis study was a retrospective chart review of 170 patients with dry eye disease. A correlation analysis between eyelid margin abnormalities and meibomian gland dropout in infrared meibography was performed using data from 141 eyes. We graded the following eyelid margin abnormalities: irregular lid margin, vascular engorgement, plugging, anterior placement of the mucocutaneous junction, exposed terminal duct, and presence of tattoos. Multiple regression analyses were performed with meiboscore (meibomian gland dropout grade) as the dependent variable and age, sex, lid margin abnormality grades, and total grading score of lid margin abnormalities as the covariates. In addition, Meibomian glands structure were examined in those with eyelid margin dimpling using meibography in 25 eyes.ResultsIn the multiple regression analysis, an irregular lid margin in the upper eyelid was associated with a higher meiboscore after controlling for age and sex (coefficients B = 1.379, p = 0.025). Other lid margin abnormalities did not significantly affect the meiboscore. In the lower eyelids, irregular lid margin (coefficients B = 0.602, p = 0.009) and total grading score of lid margin abnormality were associated with higher meiboscores (coefficients B = 0.100, p = 0.022). Of the 25 eyes with dimples, 21 (84%) showed focal or complete meibomian gland loss at the site.ConclusionsLid margin abnormalities were found to be associated with meibomian gland dropout.     

Tubular apocrine adenoma of the eyelid – A case report and literature review

Tubular apocrine adenoma is a rare benign adnexal neoplasm most commonly identified in the scalp, composed of a dermal proliferation of apocrine tubules in a background of hyalinized stroma. Tubular apocrine adenoma can be a component of various sweat gland tumors and can also morphologically overlap with other sweat gland neoplasms. Isolated tubular apocrine adenoma arising in the glands of Moll is exceedingly rare, with only 4 previously reported cases. We present a 63-year-old male with tubular apocrine adenoma of the left upper eyelid, which recurred following initial incomplete excision. Although the lesion showed focal morphologic similarity to the apocrine variant of pleomorphic adenoma (chondroid syringoma), the diagnosis of tubular apocrine adenoma was supported by fluorescence in situ hybridization studies, which demonstrated absence of PLAG1 and HMGA2 gene rearrangements seen in pleomorphic adenoma. This case illustrates the clinical, microscopic and immunohistochemical features of tubular apocrine adenoma. The recent advances in our understanding of the molecular genetics of tubular apocrine adenoma and related tumors, and how these advances shape the evolving classification of sweat gland tumors are reviewed.     

Mucoepidermoid carcinoma of an accessory lacrimal gland with orbital invasion

PURPOSE: To report a case of mucoepidermoid carcinoma of an accessory lacrimal gland with orbital invasion. METHODS: The clinical history and pathologic findings of a patient with a left upper eyelid lesion were reviewed. RESULTS: The patient was evaluated and found to have an epithelial tumor arising in an accessory lacrimal gland. Special stains showed mucin production by individual tumor cells. The tumor was classified as mucoepidermoid carcinoma. CONCLUSIONS: Mucoepidermoid carcinoma may arise in accessory lacrimal glands and invade the orbit.     

睑板腺癌术后眼睑缺损的整复

目的探讨睑板腺癌术后眼睑缺损的整复方法。方法睑板腺癌6例。1例上睑肿物切除后上睑缺损小于1/4,创缘直接分层缝合;1例上睑肿物切除后上睑缺损大于1/2,但未侵及睑板上缘,残留睑板结膜瓣向下滑行修复缺损;2例肿物侵及睑板上缘,切除后缺损分别大于1/2和3/4,下睑板结膜滑行修复上睑缘,上睑提肌瓣向下滑行和滑行睑板吻合;1例下睑肿物切除后缺损大于3/4,鼻颊部皮瓣转移修复;1例部分眶内容摘除植皮修复。结果术后随访观察12～19个月,1例眶内容摘除眶内皮片成活良好,其余5例眼睑外形及功能均恢复,眼睑闭合好,活动自如。结论睑板腺癌切除术后根据眼睑缺损的部位和大小选择不同的修复方案,均取得了良好的效果。     

Mucoepidermoid Carcinoma of the Eyelid： A Case Report and Review of the Literature

Purpose: To report the clinical features, therapeutic method, and histopathological findings of a case of mucoepidermoid carcinoma in the lower eyelid and review the literature about the mucoepidermoid carcinoma arising from the eye. Methods : Case report and review of the literature. Results: An 88-year-old man developed a painless, indurated nodule in the left lower eyelid for two years and ulceration of the skin existed for a year. He underwent tumor resection and reconstruction of the eyelid. By histopathology, tumor cells showed an admixture of epidermoid and mucus-secreting cells, which was consistent with mucoepidermoid carcinoma. Mucoepidermoid carcinoma is a common malignant tumor of the salivary glands, but rare in the eye tissues among which conjunctiva and lacrimal gland are most commonly involved. It has a higher degree of malignancy than basal cell carcinoma and squamous cell carcinoma. It displays an unusual capacity of aggressive local invasion and recurs easily after simple excision and tumors may require enucleation or exenteration because of the involvement of the intraocular structures and/or orbit. Frequent follow-up is necessary for the patient after operation. Conclusions: Mucoepidermoid carcinoma arising from the eye is rare and has a high degree of malignancy. It should be differentiated from other neoplasms such as basal cell carcinoma and squamous cell carcinoma. Eye Science 2005;21:152-157.     

The acinar and ductal organisation of the tarsal accessory lacrimal gland of Wolfring in rabbit eyelid

The purpose of this study was to objectively assess the tarsal accessory lacrimal gland of Wolfring and its excretory duct by transmission electron microscopy (TEM). The upper eyelid of 6 female grey rabbits (2 kg) was fixed in the fully extended configuration with buffered glutaraldehyde. At the tarsal/orbital portion of the palpebral conjunctiva, a series of prominent acinar glands were located by both light microscopy and TEM. Cells within the acini were characterised by apical tight junctions and desmosomal connections as well as abundant intra-cytoplasmic osmophillic (secretory) granules. The apical surfaces were profusely decorated with microvilli-like extensions. In the vicinity of the acini, and traversing over several millimetres of adjacent sub-epithelial parenchyma and epithelium, was an extensive system of duct-like spaces lined with secretory cells decorated with microvilli. These ducts run a tortuous course within the palpebral conjunctival epithelium leading to crypt-like termination's along the eyelid inner surface. The tarsal accessory lacrimal gland of Wolfring thus has an acinar structure similar to the main lacrimal glands, and its ducts are lined by secretory cells along the entire tortuous course towards the palpebral conjunctival surface, where the ducts emerge at multiple points at the tarsal/orbital portion of the surface.     

Vasoactive intestinal polypeptide (VIP) innervation of the human eyelid glands.

This study was conducted to obtain morphological proof of innervating nerve fibres in the glands of the human eyelid (accessory lacrimal glands of Wolfring, meibomian glands, goblet cells, glands of Zeis, glands of Moll, sweat glands, glands of lanugo hair follicles) and identification of the secretomotorically active neuropeptide vasoactive intestinal polypeptide (VIP) as a common transmitter. Epoxy-embedded ultrathin sections of tissue samples from human eyelids were studied using electron microscopy. Paraffin sections fixed in Bouin-Hollande solution were immunostained with rabbit antiserum against VIP. With the electron microscope we were able to identify nerves in the glandular stroma of all the glands examined with the exception of goblet cells. Intraepithelial single axons were only seen in the parenchyma of Wolfring glands. The morphological findings corresponded with the immunological finding of VIP-positive, nerve-like structures in the same locations, with the exception of lanugo hair follicle glands, and goblet cells. Our findings indicate that the glands of the eyelids and main lacrimal gland represent a functional unit with VIP as a possible common stimulating factor.     

泪腺上皮性肿瘤彩色多普勒超声特征分析

目的 分析泪腺上皮性肿瘤的彩色多普勒超声声像特征,证实其临床应用价值.方法 对28例经病理证实为泪腺上皮性肿瘤的彩色多普勒超声声像图进行回顾性分析.结果 泪腺多形性腺瘤多表现为边界清楚,形态规则,均匀中等内回声或不均匀内回声,不可压缩,彩色多普勒血流分级为I～Ⅱ级;腺样囊性癌多表现为边界清楚,形态不规则,不均匀内回声,不可压缩,彩色多普勒血流分级为Ⅲ～Ⅵ级,其中原发性泪腺腺样囊性癌收缩期峰值血流速度明显升高,与原发性泪腺多形性腺瘤间差异有统计学意义(P<0.05 ).结论 彩色多普勒超声对泪腺上皮性肿瘤的诊断及区别泪腺肿瘤良、恶性有重要临床应用价值.     

Meibomian gland morphogenesis requires developmental eyelid closure and lid fusion

Background and purpose

Meibomian glands (MGs) play an important role in the maintenance of ocular surface health, but the mechanisms of their development are still poorly understood. The MGs arise from the epithelium at the junction of eyelid fusion, raising the possibility that defective eyelid fusion disturbs the formation of MGs.

Primary infiltrating signet ring carcinoma of the eyelids

A 61-year old man presented with a five-year history of a swelling initially developing in his right lower lid that progressed to involve the lateral canthal skin and eventually the upper lid and anterior orbit. He was discovered to have an infiltrating, poorly differentiated, mucin-producing carcinoma. Systemic work-up failed to disclose a visceral malignancy, and it was concluded that his tumor was primary in the lids, arising from an adnexal sweat gland. Three other reports in the literature also share almost identical clinical and pathologic features, in that all of the earlier reports dealt with middle-aged or elderly men who had diffusely indurated lids. Histopathologically, the tumor cells grow diffusely in a sclerotic stroma, and resemble the "histiocytoid" variant of metastatic breast carcinoma to the lids in women. Ultrastructural studies in our case point toward an apocrine origin, although earlier authors have favored an eccrine origin. Despite its indolent clinical course, the tumor is capable of producing regional and distant metastases on long-term follow-up. Complete local excision, possibly necessitating radical surgery, is probably the preferred method of treatment, but local radiotherapy may have a beneficial effect in retarding spread of the disease.     

Inner eyelid surface temperature as a function of warm compress methodology

PURPOSE: To (1) determine an optimal method of warm compress (WC) application to maximize heating meibomian glands in minimal time, (2) determine the maximum inner eyelid temperature achievable during 30 min of routine WC application, (3) examine the cooling curve for 10 min after WCs have been discontinued. METHODS: Subjects were randomly assigned to one of three WC methodologies. Group A (GrA), (n = 10): 15 min WC application without reheating. GrB, (n = 10): 30 min WC application with reheating every 2 min. GrC, (n = 12): 30 min WC application optimizing contact with the lower lid and reheating every 2 min. WCs were heated to 45 +/- 0.5 degrees C. Outer and inner lower eyelid surface temperatures were measured at baseline and regular intervals. RESULTS: GrA: the maximum outer and inner lower eyelid surface temperatures = 41.2 +/- 0.3 degrees C at 1 min and 38.8 +/- 0.2 degrees C after 4 min, respectively. GrB: the maximum outer upper eyelid temperature, 43.3 +/- 0.5 degrees C, was reached after 6 min while it required 30 min to reach the maximum inner lower eyelid temperature, 40.4 +/- 0.3 degrees C. GrC: it required 4 min to reach the maximum outer lower eyelid temperature, 42.2 +/- 0.4 degrees C, while it required 20 min to reach the maximum inner lower eyelid temperature, 40.8 +/- 0.3 degrees C. CONCLUSIONS: To optimize WC efficiency, patients should (1) heat the WC to approximately 45 degrees C, (2) optimize contact between the WC and outer eyelid surfaces, (3) reheat the WC frequently and have a replacement heated WC on hand for exchange, and (4) perform the activity for at least 4 min in order to achieve an inner lower eyelid temperature > or = 40 degrees C. Longer therapy may be necessary for more severe obstructions. These data suggest that precise, customized, labor-intensive WC procedure is necessary to optimize treating meibomian gland dysfunction and obstruction using WCs.     

睑板腺功能障碍患者睑缘形态与睑板腺形态学的相关性研究

目的 研究睑板腺功能障碍（MGD）患者睑缘形态与睑板腺形态的相关性。设计 横断面研究。研究对象 2019年北京同仁医院行Lipiflow热脉冲治疗前的MGD患者30例（30眼）。方法 通过眼前节照相技术和Oculus眼表分析仪拍摄睑缘和睑板腺,并依据睑缘形态、睑板腺主体部形态、睑板腺开口和分泌物性状进行分类。睑缘形态分为肥厚、充血、角化和变形;睑板腺主体部形态分为缺失、白色节段、导管膨胀、萎缩、扭曲和分叉;睑板腺开口形态分为圆形、脂帽、脂塞和脂栓。依上述形态进行评分,运用Spearman相关分析研究睑缘形态与睑板腺形态的相关性。主要指标 睑缘及睑板腺形态评分,睑缘及睑板腺形态学相关系数（r）。结果 睑板腺开口形态与分泌物性状之间呈正相关（r=0.590,P=0.001）,睑板腺白色节段与睑板腺开口形态呈正相关（r=0.439,P=0.015）;睑板腺萎缩和分叉与睑板腺的分泌物性状呈负相关（r=-0.349,-0.374;P=0.048,0.042）。在睑缘形态中,睑缘角化的评分与睑缘形态总评分呈正相关,且相关关系最强（r=0.842,P＝0.000）;睑缘角化与睑板腺开口形态改变呈正相关（r=0.517,P=0.003）。在睑板腺形态中,睑板腺白色节段评分与睑板腺总评分呈正相关,且相关关系最强（r=0.535,P＝0.002）。结论 MGD患者睑缘形态与睑板腺形态部分存在相关关系。睑板腺白色节段作为睑板腺主体部的形态评估指标更为合理;睑缘角化作为睑缘的形态评估指标更为合理,且睑缘角化与睑板腺开口的形态改变相关。（眼科, 2020, 29： 355-360）     

Endocrine Mucin-Producing Sweat Gland Carcinoma: An Uncommon Presentation

Endocrine mucin-producing sweat gland carcinoma (EMPSGC) is a rare, often underrecognized, low-grade sweat gland carcinoma of the skin of the eyelid. To date, only 20 cases of this carcinoma have been reported, most frequently in Caucasian females with an average age of 70 years. Although the diagnosis is primarily made with immunohistochemical stain, compared to endocrine ductal carcinoma in situ, clinical detection serves as a potentially curative treatment. Further, its benign appearance clinically makes this tumor often misdiagnosed and undertreated. This disease commonly presents in Caucasian women of advanced age, aiding in the diagnosis of this tumor, which presents an even more critical diagnosis in a patient with a rare presentation. In the available literature, we could find no case of EMPSGC in younger African American women. The following case is the first case presented in the literature. Here, we present a case of an atypical presentation of the tumor in a young African American female, as well as a review of literature on the pathophysiology, clinical presentation, and treatment of EMPSGC.     

眼睑恶性肿瘤控制性切除术后疗效观察

 【摘要】 目的 探讨控制性切除术对眼睑恶性肿瘤患者预后的影响。设计 回顾性病例系列。研究对象2011年1月至2015年12月河南省立眼科医院经病理诊断为眼睑恶性肿瘤并行控制性切除术的患者68例（68眼）。方法 收集患者的临床资料,随访其预后及肿瘤复发情况,分析眼睑恶性肿瘤控制性切术的治疗效果。主要指标  肿瘤分期、大小、手术、组织病理类型、复发情况。结果 68例（68眼）中眼睑基底细胞癌者39眼（57.3%）,睑板腺癌28眼（41.2%）,鳞状细胞癌者1眼（1.5%）。随访5~57个月,5例（7.4%）复发。39例基底细胞癌患者中1例（2.6%）术后2年复发;28例睑板腺癌患者中4例（14.3%）复发。所有随访到的无复发的63例患者中20例（31.7%）经组织病理检查,发现切除远端组织内仍残留肿瘤细胞,并行二次手术切除,至切除远端未见肿瘤细胞残留,行眼睑缺损修补术。结论 眼睑恶性肿瘤石蜡组织学控制切除术能有效降低患者的复发率,尽可能保留肿瘤周围正常的眼睑组织,有利于组织缺损修复。     

Immunohistochemical evidence for estrogen receptors in meibomian glands

PURPOSE: To look for sex hormone receptor distribution in three structures contributing to the normal human tear film: the conjunctiva, the accessory lacrimal glands, and the meibomian glands. DESIGN: An immunohistochemical study. TISSUES AND CONTROLS: Forty-one upper eyelid specimens were collected from 15 male and 26 female patients (age range, 1.5-85 years) during blepharoptosis surgery via posterior tarsoconjunctival mullerectomy (Fasanella-Servat or Gavaris). In addition, control sections of histologically normal breast, prostate, and skin tissue were obtained. METHODS: Immunohistochemical staining using mouse monoclonal antibodies against estrogen, progesterone, and androgen receptors was performed on all tissues and controls. Quantitation of the receptors was performed and expressed as percentage nuclear positivity. Specimens were divided into three groups based on the age of the patient: <12 years (n = 9); 18-55 years (n = 1); >55 years (n = 12). RESULTS: Forty-one specimens contained conjunctiva. All were negative for estrogen, progesterone, and androgen receptors. Twenty-four specimens contained accessory lacrimal glands of Wolfring. All were negative for the three receptors. Twenty-two specimens contained meibomian glands. All were positive for estrogen receptors; one was positive for progesterone receptors and one for androgen receptors. Using Minitab statistical software (Minitab Inc. State College, PA), analysis of variation revealed no statistical difference between sexes or between age groups studied. The sebaceous glands of skin were uniformly positive for androgen receptors. Sebaceous glands of the face and scalp (3 of the 15 skin samples) were also positive for estrogen receptors. CONCLUSIONS: Estrogen receptors are present in the meibomian glands of the upper eyelid. Unlike sebaceous glands elsewhere on the skin, the meibomian glands lack androgen receptors. Estrogen receptors may play a role in modulation of the lipid layer of the tear film, and their activity may be linked to meibomian gland dysfunction and dry eye syndrome.     

Cutaneous benign mixed tumor (chondroid syringoma) of the eyelid: clinical presentation and management

PURPOSE: To describe the clinical presentation of cutaneous benign mixed tumor of the eyelid and its management options. METHODS: Periocular cases of cutaneous benign mixed tumor were gathered from members of an oculoplastics specialty Internet discussion group. A total of 9 patients are described in this retrospective, interventional case series. The clinical presentation, histopathology, and management of these lesions is reviewed. RESULTS: Patients were typically asymptomatic, presenting with a slowly enlarging, nontender nodule of 2 to 8 years' duration. The lesions ranged from 4 mm to 17 mm in greatest dimension. Four of the lesions were on the eyelid margin, three in the sub-brow area of the upper eyelid, and two in the central lids. All six cases not involving the brow were fixed to the tarsus; one brow lesion was believed to be adherent to the skin. None of the lesions was associated with significant changes of the overlying epidermis, although one lesion showed overlying pigmentation. All patients underwent excisional biopsy for diagnostic or cosmetic reasons. On histopathologic examination, the tumors were biphasic, with an epithelial component exhibiting apocrine or hair follicle differentiation and a myxoid, adipocytic, chondroid, and/or fibrous stroma. The pathologic diagnoses were all consistent with cutaneous benign mixed tumor (chondroid syringoma, pleomorphic adenoma). Follow-up ranged from 2 weeks to 12 months, although several patients failed to keep scheduled follow-up appointments. No clinical recurrences were identified. CONCLUSIONS: Cutaneous benign mixed tumor may occur in the eyelid, and, although uncommon, should be included in the differential diagnosis of firm, nodular eyelid tumors. The histopathologic features are similar to those seen in this tumor type arising in other areas of the body. Preoperative consideration of this diagnostic possibility may allow the surgeon to plan for complete excision, thereby reducing the possibility of recurrence or malignant transformation.