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Feline cystadenomatosis affecting the ears and skin of 57 cats (2011–2019)
57只猫耳部和皮肤的猫囊腺瘤病(2011-2019年)
作者：Klaus E Loft, Julie Soohoo, Brooke Simon and Christian E Lange
翻译：王思权； 校对：王帆
Abstract
Objectives：This study aimed to understand epidemiological factors associated with feline cystadenomatosis, including signalment and papillomavirus PCR status. Cystadenomatosis is an uncommon condition primarily involving the ceruminous and apocrine skin and ear glands.
Methods：This was a retrospective case series. Clinical records from 2011 to 2019 from a tertiary referral hospital in Boston, MA, USA were screened for cases, and case data were re-evaluated and analyzed. The total patient pool contained 65,385 individual cats, of which 797 were referred to the dermatology service. Medical records and biopsy specimens were reviewed; the information collected included signalment, clinical signs, physical examination and diagnostic tests, comorbidities and histopathologic findings. PCR was performed on biopsy specimens to test for papillomavirus DNA.
Results：The cystadenomatosis population consisted of 57 cases (7.1% of total cases referred to the dermatology service) with 105 affected ears. Twenty-seven cases (48 ears) were confirmed via histopathology; four cats (7%) exhibited clinically cystic lesions on the periocular, periorbital and perianal regions; only one cat did not have pinnal lesions. Domestic shorthair cats were most often affected. Relative risk for cystadenomatosis was 2.24 times higher in male cats. In 48 cats (84.2%), ears were bilaterally affected. Seven cats (12.3%) had malignant neoplasia, which included: inflamed adenocarcinoma (n = 5); mast cell tumor (n = 1); or squamous cell carcinoma (n = 1). PCR testing on biopsy specimens from 24 cats revealed feline papillomavirus type 2 DNA in only four cats.
Conclusions and relevance：Cystadenomatosis was more prevalent in senior non-purebred cats, over-represented in male cats and did not appear to be associated with papillomavirus, feline infectious peritonitis, feline immunodeficiency virus/feline leukemia virus status or other identifiable illnesses. Further studies are needed to investigate the causes of cystadenomatosis.
摘要：
目的：本研究旨在了解与猫囊腺瘤病相关的流行病学因素，包括特征和乳头瘤病毒PCR（聚合酶链式反应）情况。囊腺瘤病是一种不常见的疾病，主要涉及皮肤顶浆汗腺及耳道耵聍腺。
方法：回顾性研究。筛选了美国马萨诸塞州波士顿一家三级转诊医院2011年至2019年的临床记录进行了病例筛选，并对病例数据进行了重新评估和分析。医院病历总共有65385只猫，其中797只被分诊到皮肤科就诊。检查病例记录和活检标本；收集病例信息包括特征、临床症状、体格检查和诊断测试、共病和组织病理学结果。对活检标本进行PCR检测乳头瘤病毒DNA。
结果：囊腺瘤病患猫57例(占皮肤科治疗病例总数的7.1%)，共105耳。27例(48耳)经组织病理学证实，4只猫(7%)在眼周、眶周、肛周表现为囊性病变，仅1只猫无耳廓病变。家养短毛猫最容易患病。公猫患囊腺瘤的相对风险是母猫的2.24倍。其中48只猫(84.2%)双耳患病。7只猫(12.3%)有恶性肿瘤，其中炎性腺癌5只，肥大细胞瘤1只，鳞状细胞癌1只。对24只猫的活检标本进行了PCR检测，发现只有4只猫存在猫乳头状瘤病毒2型DNA
结论和相关性：囊腺瘤病在老年非纯种猫中更为普遍，在公猫中比例过高，似乎与乳头瘤病毒、猫传染性腹膜炎、猫免疫缺陷病毒/猫白血病病毒状态或其他已知的疾病无关。囊腺瘤病的病因有待进一步研究。
Keywords: Otitis externa; periocular cysts; cysts; cystadenomatosis; apocrine glands; ceruminous glands
关键词：外耳炎；眼周囊肿；囊肿；囊腺瘤病；大汗腺；耵聍腺
Introduction
介绍
Cystadenomatosis involves the formation of multiple cysts originating from the secretory portion of modified apocrine sweat glands, leading to the development of characteristic focal to coalescing bluish-black cystic skin and ear nodules that range from 1 mm to several centimeters in diameter (Figure 1). The cysts are often located in areas of the body where modified apocrine sweat glands are known to be located.They have also been reported to occur in dogs and humans.Cystadenomatosis has been found to primarily affect the preauricular region, concave pinna, concha, tragus and antitragus, and the external ear canal of cats, while less commonly affected areas include the periocular, perioral and perianal regions (Figure 2).
囊腺瘤病是由起源于改良顶浆汗腺分泌部分多个囊肿形成的，导致发展成特征性局灶性聚集性的蓝黑色囊性皮肤和耳部结节，直径从1毫米到几厘米不等（图1）。囊肿通常位于已知改良顶浆汗腺的机体部位。据报道，也会发生在犬和人身上。囊腺瘤病已被发现主要影响猫的耳前区、凹耳廓、耳甲（concha）、耳屏和对耳屏，以及外耳道，而眼周、口周和肛周区域(图2)较少发生。
Figure 1 Extensive classic bluish-black cystic lesions on the entire concave pinna and into tragus folds
图1.整个耳廓凹面及耳屏皱褶处大面积典型的蓝黑色囊性病变
Figure 2 Open arrows indicate the apocrine cystic lesions along the eyelids. The solid arrows indicate pre-aural cysts in an Abyssinian cat. Courtesy of Dr M Coster
图2.空箭头指示沿眼睑的顶浆腺囊性病变。实箭头表示阿比西尼亚猫耳前囊肿。由Dr.M Coster提供
The nomenclature for this condition can be confusing, and terms such as sebaceous or ceruminous cysts,apocrine cystadenoma, ceruminous cystadenoma, apocrine cysts, apocrine hidrocystomas, sweat gland cysts of the skin, melanotic ceruminous cysts, ceruminous gland adenomas, ceruminous gland cysts, ceruminous gland hyperplasia, ceruminous cystomatosis and epitrichial cystadenomas have all been used. We have previously used the term ‘cystomatosis’ in abstracts.
这种病变的命名可能很难，术语像是皮脂腺或耵聍腺囊肿、顶浆腺囊腺瘤、耵聍腺囊腺瘤、顶浆腺囊肿、顶浆腺汗腺囊肿、皮肤汗腺囊肿、黑色素性耵聍腺囊肿、耵聍腺腺瘤、耵聍腺囊肿、耵聍腺增生、耵聍囊瘤病和毛上汗腺囊腺瘤都使用过。所以我们使用摘要中用过的词“囊腺瘤病”。
An adenoma is defined as a benign epithelial neoplasm of glandular origin; however, it is non-specific and hence the microanatomical glandular tissue of origin is often indicated (ie, apocrine gland) to further specify the pathology . ‘Cystadenoma’ describes an adenoma in which the glandular space is cystic.According to the Merriam-Webster medical dictionary , the term ‘adenomatosis’ is defined as ‘a condition marked by growths consisting of glandular tissue’. In our view, the term ‘cystadenomatosis’ is a more comprehensive term for the description of this particular dermatosis. We therefore propose that the term ‘feline cystadenomatosis’ be used to describe this clinical syndrome.
“腺瘤”被定义为一种腺体来源的良性上皮性肿瘤；然而，它是非特异性的，因此，为了进一步明确病理学，通常会指出其来源于的微解剖学的腺体组织（如顶浆腺）。“囊腺瘤”是指腺体间隙为囊状的腺瘤。根据《韦氏医学词典》，“腺瘤病”一词被定义为“一种以腺体组织生长为特征的疾病”。在本文作者看来，术语“囊腺瘤病”是一个更全面的术语来描述这种特殊的皮肤病。因此，我们建议用“猫囊腺瘤病”一词来描述这种临床综合征。
Cystadenomatosis has been reported in Europe, North America, Asia and South America.The clinical sign associated with this benign cutaneous neoplasia is minimal inflammation in the surrounding skin.However, it can cause obstructive conformational changes in ear canals and enhance the likelihood and severity of secondary infectious otitis externa.
囊腺瘤病在欧洲、北美、亚洲和南美均有报道。这种良性皮肤瘤样病变的临床表现为周围皮肤轻度炎症。但它可引起耳道结构的阻塞性改变，增加继发感染性外耳炎的可能性和严重程度。
The glands involved in cystadenomatosis are the modified sweat apocrine glands, which can be found in the skin surrounding the eyelid margins (Moll’s glands), lip margins and perianal regions, but are most prevalent in and around the ears.The external acoustic meatus of cats contains ceruminous and sebaceous glands, with mixed epitrichial and atrichial units, which produce the waxy cerumen emulsion that coats the ear canal surface.Cerumen is a mixture of desquamated flattened keratinized squamous epithelial cells and the fatty secretions from sebaceous and ceruminous glands, and helps to maintain barrier function within the ear canal.In cats, the ceruminous glands of the external acoustic meatus are folded tubular glands with a wide tubular glandular architecture and are surrounded by a single or double layer of myoepithelial cells (Figure 3). The secretory material from these modified sweat glands in and around the ears, eyes and lip margins can be both atrichial (away from the follicular infundibulum) and epitrichial (terminate in the follicular infundibulum).
囊腺瘤病涉及的腺体是改良的顶浆汗腺，这种腺体存在于眼睑边缘周围的皮肤(Moll腺)、唇缘和肛周区域，但其在耳道内和耳周围最常见。猫的外耳道包含耵聍腺和皮脂腺，以及混合的毛上汗腺和无毛汗腺单位，以产生覆盖于耳道表面被毛的蜡样耵聍。耵聍是脱落的扁平角质化鳞状上皮细胞与皮脂腺和耵聍腺分泌的脂肪的混合物，其有助于维持耳道内的屏障功能。在猫中，外耳道的耵聍腺是折叠的管状腺，具有宽管状的腺结构，并被单层或双层肌上皮细胞包围(图3)。耳、眼睛和唇缘的改良汗腺分泌的物质既可以是无毛汗腺(远离毛囊漏斗)，也可以是有毛汗腺(终止于毛囊漏斗)。
Figure 3 Histopathology from a cystadenomatosis-affected ear.
图3.一只囊腺瘤病患耳的组织病理学。
Human and canine ceruminous glands are located in the deeper layers of the tissue, just beneath the deeper aspects of the apocrine glands of the ear canal.To our knowledge, this has not been shown in the feline literature and, according the authors of Skin Diseases of the Dog and Cat, ‘Interestingly , cysts derived from eccrine sweat glands have not been observed in dogs or cats’. The proportion of the ceruminous to sebaceous glands within the length of the ear canal appears to change as one moves away from the tympanic membrane. This ratio of mainly ceruminous glands is higher in the horizontal meatus compared with the vertical meatus, where there are more sebaceous and apocrine glands.The thin, smooth, epidermally stratified squamous epithelial lining of the external ear canal is histologically similar to that of the pinna and epidermis; however, in most dog breeds, there are less simple single hair follicles within the canal, and the hairs do not extend throughout the length of the canal except in the Cocker Spaniel, which has more compound hair follicles within the acoustic meatus.Little has been published regarding this in cats. According to Fernando’s 1965 paper, secretions from the cells of the ceruminous glands do not stain histologically on Mallory’s triple staining, which differs from what is reported in dogs. The modified apocrine glands in cat’s external ears contain acidic mucous substances, which are found in the apical parts of the glandular cells and the lumen of these glands, and have properties similar to mucous glands. The diastase-resistant periodic acid–Schiff-positive granules of the apocrine glands found in the external ear canal are located near the apical borders of the glandular cells in cats.
人类和犬的耵聍腺位于组织的深层，就在耳道顶浆腺的深处。据我们所知，这一点在猫科动物文献中尚未得到证实，《犬和猫的皮肤病》的作者表示，“有趣的是，在犬或猫身上没有发现来自于小汗腺的囊肿（666页）。耳道内耵聍腺与皮脂腺的比例似乎随着远离鼓膜的距离而变化。水平耳道中耵聍腺的比率更高，而垂直耳道则有更多的皮脂腺和顶浆腺。外耳道的薄而光滑的表皮复层鳞状上皮在组织上类似于耳廓和表皮的上皮；然而，除了可卡犬外，别的大多数犬种的耳道内较少有简单的单个毛囊，并且毛发不会延伸至整个耳道。可卡犬在耳道内则有较多的复合毛囊。关于猫的这方面的研究很少。根据Fernando 1965年的论文，在组织学上，来自耵聍腺细胞的分泌物不会在Mallory氏三色染色法上染色，这与在犬中报道的情况不同。猫外耳中改良的顶浆汗腺含有酸性粘液物质，这些酸性粘液物质存在于腺细胞的顶部和腺腔中，类似于粘膜腺体的特性。外耳道中存在于顶浆腺的抗淀粉酶高碘酸-希夫阳性颗粒位于猫腺细胞的顶端边缘。
The bluish-black clinical appearance of the cystadenomatosis lesions are partially a reflection of the distended skin and partially attributed to the pigmentary contributions from hemosiderin and ceroid of the thick acellular or cell-poor fluid filling the cystic lumen. Histopathologically, these cysts have been theorized to be caused by obstructed glandular ducts, or retention cysts, based on the flattened appearance of epithelial cells with dermal fibrosis or due to compression exerted from benign adenomas process with papillary digital projections into the cystic cavities. No known etiology has been identified as a potential cause for the cyst formation, but some authors have theorized that a senile change may play a role in cyst formation.
囊腺瘤病变的临床表现为蓝黑色，有些是肿胀的表现，有些是由于囊腔内充满富含铁血黄素和蜡样色素的无细胞或缺乏细胞的液体。在组织病理学上，理论认为这些囊肿是因为腺管阻塞或囊肿滞留导致的，理由基于上皮细胞扁平外观，伴随真皮纤维化或由于良性腺瘤形成过程时压迫细微的乳头状指突进入囊腔。目前还没有确定囊肿形成的潜在原因，但一些作者从理论上推测，年龄增加可能导致囊肿形成。
Feline cystadenomatosis has been reported in the literature to be a rare benign neoplasia, and has been observed to occur most commonly in middle-aged to older Abyssinian, Himalayan and Persian cats.Therapeutic approaches ranging from benign neglect to various topical and systemic anti-inflammatory treatments to draining of the cysts to chemical ablation, cauterization, cryosurgery, surgical excision or laser surgery have been suggested; however, continued development of new lesions after removal is common.
据文献报道，猫囊腺瘤病是一种罕见的良性肿瘤，并发现最常见于中年至老年阿比西尼亚猫、喜马拉雅猫和波斯猫。对囊腺瘤的治疗方法多样，从不做处理到建议进行各种局外部和全身抗炎治疗，再到囊肿引流、化学消融、烧灼、冷冻手术、手术切除或激光手术。但是，移除囊腺瘤后，也很常见出现新的病变。
In order to better understand this clinical syndrome, a retrospective study was conducted to investigate any potential association between cystadenomatosis and factors such as age, sex, breed, feline immunodeficiency virus (FIV)/feline leukemia virus (FeLV) status and comorbidities. We also decided to screen for the presence of papillomavirus (PV) DNA in tissue collected from 24/44 cystadenomatosis cases, as PV is known to be associated with the development of several types of cutaneous neoplasms. Only cases that were examined and sampled in the period 2011–2015 were tested for PV.
为了更好地了解这种临床特征，我们进行了一项回顾性研究，以调查囊腺瘤病与发病年龄、性别、品种、猫免疫缺陷病毒(FIV)/猫白血病病毒(FeLV)状态和共病因素之间的潜在联系。我们还决定在24/44例囊腺瘤病例的组织中筛查乳头瘤病毒(PV) DNA的存在，因为PV已知与几种类型的皮肤肿瘤的发展有关。本篇研究仅对2011-2015年期间检查和采样的病例进行了PV检测。
Materials and methods
材料与方法
The medical records of 797 individual feline patients, seen by the dermatology service at Angell Animal Medical Center in Boston, MA, USA from January 2011 to December 2019 exclusively , were reviewed.
研究人员回顾了2011年1月至2019年12月美国马萨诸塞州波士顿安吉尔动物医疗中心皮肤科诊疗室收治的797名猫的病例记录。
Signalment information and other data were included in the study and reviewed. To determine prevalence, the number of cases consistent with a clinical diagnosis of cystadenomatosis was compared with the total referred feline population seen by the dermatology service during the same 84 months. The medical records of all patients were reviewed. We used the first mention of otitis externa or lesions consistent with a likely diagnosis of cystadenomatosis. The identification of cysts in medical records was used as the best estimate for age of onset of cystadenomatosis in a given patient. Where such information was missing from the records, we used the date of first presentation to the dermatology service for referral as the age of onset. Data regarding sex, breed (when provided by owner) and FIV/FeLV status (when available) were collected. The histopathologic findings from 27 cats (if applicable; see Figure 3) and concurrent medical conditions were also collected.
研究中包括临床表现信息和其他数据。为了确定发病率，将符合囊腺瘤病临床诊断的病例数与同样84个月内皮肤科诊疗所见的总转诊猫群数量进行了比较。查阅了所有病历信息。我们使用了首诊就提到是外耳炎或与囊腺瘤病变相似的病例。病历中将发现囊性病变的日期作为对患猫囊腺瘤病最佳的发病估算年龄。如果记录中没有这样的信息，我们使用首次向皮肤科诊所提交转诊的日期作为发病年龄。收集了有关性别、品种(如果主人提供)和FIV/FeLV情况(如果有)的数据。还收集了27只猫的组织病理学结果(如果适用；见图3)和同时发生的医疗情况。
Biopsy specimens from 24 of these animals were submitted for PCR testing to evaluate for the presence of PV.Either fresh frozen or paraffin-embedded tissue was used, and DNA was extracted with a DNeasy extraction kit (Qiagen) according to the manufacturer’s recommendations. In order to detect PV DNA in the extracts, six different PCR assays were employed. Two of the PCRs used degenerate primers for conserved regions to detect a broad spectrum of PV types, with the FAP59/FAP64 assay targeting the L1 gene and the CP4/CP5 assay targeting the E1 gene.3Additionally , four PCRs were used to test specifically for feline PV2 (FcaPV) types 1–4. For FcaPV2, the JMPF/JMPR primers were selected, while new primers were designed for FcaPV1, FcaPV3 and FcaPV4: FcaPV1_L1 f (5’-TTG GGA GAG GAC AAC CTT TG-3’)/FcaPV1_L1 r (5’-GGC TTA CAG CCC ACC ATA AA-3’), FcaPV3_L1 f (5’-GTT TGC TTT GGC AGA CCC TA-3’)/FcaPV3_L1 r (5’-TGG ACA CAT TTT GCC TTT CA-3’) and FcaPV4_L1 f (5’-TGG TGA TGA AAG GCA AAA CA-3’)/FcaPV4_L1 r (5’-CAA AGC CCA CAT CAC ACA TC-3’), respectively .4The FAP59/FAP64, CP4/CP5 and JMPF/JMPR PCRs were performed as previously published,39,41,42,43 while for the other three PCR assays an initial denaturation of 94°C for 3 mins was used, followed by 40 cycles of 94°C for 30 s, 55°C for 30 s and 72°C for 30 s. PCR products were loaded and visualized on an agarose gel, amplicons were cut out and DNA was purified using a QIAquick DNA extraction kit (Qiagen).Sequencing was performed in an ABI 3730 sequencer (Applied Biosystems), and the results were compared with the National Center for Biotechnology Information (NCBI) database using the BLAST N algorithm. The PCR protocols were performed as published and, in the case of the novel PCR assays, an initial denaturation of 94°C for 3 mins was used, followed by 40 cycles of 94°C for 30 s, 55°C for 30 s and 72°C for 30 s. PCR products were loaded and visualized on an agarose gel, amplicons were cut out and DNA was purified using a QIAquick DNA extraction kit (Qiagen). Sequencing was performed in an ABI 3730 sequencer (Applied Biosystems), and the results were compared with the NCBI database using the BLAST N algorithm.
其中24只动物的活检标本被提交进行PCR检测以评估PV的存在。文章使用新鲜冷冻组织或石蜡包埋组织，并根据制造商的说明书，用DNeasy提取试剂盒(Qiagen)提取DNA。为了检测提取液中的PV DNA，采用了6种不同的PCR方法。其中两个PCR使用简并引物对保守区域进行检测，以检测广泛的PV类型，FAP59/FAP64法靶向L1基因，CP4/CP5法靶向E1基因。此外，四种PCR用于猫PV2 (FcaPV) 1-4型特异性检测。对于FcaPV2，选择JMPF/JMPR引物，而对FcaPV1、FcaPV3和FcaPV4分别设计了新的引物: FcaPV1_L1 f (5’-TTG GGA GAG GAC AAC CTT TG-3’)/FcaPV1_L1 r (5’-GGC TTA CAG CCC ACC ATA AA-3’), FcaPV3_L1 f (5’-GTT TGC TTT GGC AGA CCC TA-3’)/FcaPV3_L1 r (5’-TGG ACA CAT TTT GCC TTT CA-3’) and FcaPV4_L1 f (5’-TGG TGA TGA AAG GCA AAA CA-3’)/FcaPV4_L1 r (5’-CAA AGC CCA CAT CAC ACA TC-3’)。FAP59/FAP64、CP4/CP5和JMPF/JMPR 的PCR按之前发表过的方法进行，而对于其他三种PCR分析，初始变性94°C 3 min，随后进行40个循环，94°C 30 s, 55°C 30 s和72°C 30 s。PCR产物利用琼脂糖凝胶跑胶，扩增子被切割后用QIAquick DNA提取试剂盒(Qiagen)纯化。测序在ABI 3730测序仪(Applied Biosystems)中进行，并使用BLAST N算法将结果与国家生物技术信息中心(NCBI)数据库进行比较。按照已发表的PCR方案进行，在新型PCR分析的情况下，使用94°C初始变性3分钟，然后进行40个循环，94°C为30 s, 55°C为30 s, 72°C为30 s。PCR产物装载在琼脂糖凝胶上，扩增子被切割，DNA用QIAquick DNA提取试剂盒(Qiagen)纯化。测序在ABI 3730测序仪(Applied Biosystems)中进行，并使用BLAST N算法将结果与NCBI数据库进行比较。
Statistical analysis
统计学分析
Descriptive data were generated using www.medcalc.org and Microsoft Excel; comparison of means and a χ2 test of independence were performed.
使用www.medcalc.org和Microsoft Excel生成描述性数据;进行均数比较和χ2独立检验。
Results
结果
Cystadenomatosis was found in 7.1% (n = 57/797) of referrals and in 0.1% of the total hospital feline population seen (n = 57/65,385) during the study period. Of the 57 cats that had lesions consistent with cystadenomatosis, 84.2% (n = 48/57) had both ears affected, and in 4/57 cats (7.0%), clinically similar cystic lesions were also noted in other locations on the body (one in the periocular region, two in the periorbital region and one in the perianal region). In 27 cats (48 ears) (47.4%, n = 27/57), the diagnosis of cystadenomatosis was confirmed histopathologically and PCR testing for the presence of PV was conducted on biopsy material from 24 of these cats.FcaPV2 DNA was detected in 4/24 (16.7%) cats.
在研究期间，7.1%的转诊猫(n = 57/797)和0.1%的医院就诊猫(n = 57/65,385)发现了囊腺瘤病。在57只出现囊腺瘤样病变的猫中，84.2% (n = 48/57)双耳均有病变，4/57只猫(7.0%)在身体其他部位也发现了临床相似的囊性病变(1只在眼周区，2只在眶周区，1只在肛周区)。27只猫(48只耳)(47.4%，n = 27/57)中，组织病理学证实了囊腺瘤病的诊断，并对其中24只猫的活检材料进行了PV的PCR检测。4/24只猫(16.7%)检测到FcaPV2 DNA。
The mean ± SD age of onset among affected cats was 11 ± 3.0 years (range 3.5–17). The mean ± SD age of unaffected cats seen by the dermatology service (n = 740) at the time of referral was 7.31 ± 4.37 years (range 0.5–19.2) (P <0.0001). Domestic shorthair cats were most commonly affected (57.9%, n = 33/57).Other breeds included domestic mediumhair (n = 13 [22.8%]), domestic longhair (n = 7 [14%]), Persian (n = 2 [3.5%]), Himalayan (n = 1 [1.8%]) and Abyssinian (n = 1 [1.8%]) (Table 1).There was no significant breed predisposition in the overall population of cats referred to the dermatology service (χ2 [, n = 797] = 0.786275, P = 0.992439, P <0.05).
患病猫发病的平均±SD年龄为11±3.0岁(范围3.5-17岁)。皮肤科检查未患猫的平均±SD年龄为7.31±4.37岁(0.5~19.2岁)(P<0.0001)。家养短毛猫最常见(57.9%，n = 33/57)。结果还包括其余品种如家养中毛猫(n = 13[22.8%])、家养长毛猫(n = 7[14%])、波斯毛(n = 2[3.5%])、喜马拉雅毛(n = 1[1.8%])和阿比西尼亚毛(n = 1[1.8%])(表1)。皮肤科就诊猫病例中未发现明显的品种易感性(χ2[自由度,n = 797] = 0.786275, P = 0.992439, P <0.05)。
Cystadenomatosis was found in one intact female (1.8%), 14 spayed females (24.6%), two intact males (3.5 %) and 40 castrated males (70.2%). Most of the cats in both the affected and the total referred populations were spayed or castrated (n = 54/57 [94.7%] and n = 776/797 [97.4%], respectively) (Table 2). Of the 57 affected cats, there was a male:female ratio of 2.8. The male (intact plus castrated, n = 387) to female (intact and spayed, n = 410) ratio in the referral population was 0.94. The male cats had a 2.24 higher relative risk of developing cystadenomatosis than female cats (99% confidence interval 1.27–1.809).
这些数据显示，在1只未绝育雌性（1.8%）、14只已绝育雌性（24.6%）、2只未去势雄性（3.5%）和40只已去势雄性（70.2%）猫中发现了囊腺瘤病。囊腺瘤病例种群和总参考种群中的大多数猫都已绝育或已去势（分别为n=54/57[94.7%]和n=776/797[97.4%]）（表2）。在57只囊腺瘤病例的猫中，雄性与雌性的比例为2.8。转诊病例中雄性（已去势和未去势，n=387）与雌性（已绝育和未绝育，n=410）的比例为0.94。雄猫患囊腺瘤病的相对风险比雌猫高2.24倍（99%置信区间1.27–1.809）。
Seven of the 57 cases (12.3%) were diagnosed with malignant neoplasia in and around the ears and included the following diagnoses: inflamed adenocarcinoma (n = 5); mast cell tumor on pinna (n = 1); and squamous cell carcinoma (n = 1). A range of concurrent clinical conditions was reported in the 57 affected cats, with a range of clinical signs and/or a medical history of dermatologic problems and treatments suggestive of some form of allergic dermatosis (n = 3); chronic renal failure (n = 2); pancreatitis (n = 1); chylothorax (n = 1); diabetes mellitus (n = 1); hyperthyroidism (n = 2); atopic dermatitis (n = 1); and lymphoma (n = 1). However, the majority of cases (n = 48) had no known clinical disease requiring medical attention besides secondary otitis externa (bacterial, yeast or mixed) owing to cystadenomatosis. No cats in the study had required treatment for an ear infection prior to developing cysts, other than a single cat that had a history of ear mites (Otodectes species). A review of the medical records showed that in 42 of the cases of cystadenomatosis, there had been clinical signs consistent with secondary otitis externa unilaterally or bilaterally upon or within the 6 months prior to referral.
57例病例中有7例（12.3%）被诊断为耳内及耳周恶性肿瘤，包括以下诊断：炎性腺癌（n=5）；耳廓肥大细胞瘤（n=1）；和鳞状细胞癌（n＝1）。据报道，这57只猫中，一些猫患囊腺瘤病的同时，伴发了一系列其他临床症状，这些临床表现和/或过去的皮肤病史和治疗提示，其中一些猫出现某种过敏性皮肤病（n = 3）；慢性肾衰竭（n = 2）；胰腺炎（n = 1）；乳糜胸（n = 1）；糖尿病（n = 1）；甲状腺功能亢进（n = 2）；特应性皮炎（n = 1）和淋巴瘤（n = 1）。然而，大多数病例（n = 48）除了由于囊腺瘤病引起的继发性外耳炎（细菌、酵母或混合型）之外，没有已知的需要医疗关注的临床疾病。除了一只有耳螨病史的猫（耳螨属）外，研究中没有一只猫在出现囊肿之前需要治疗耳感染。对就诊记录的审查显示，在转诊前6个月内或在转诊时，42例囊腺瘤病例在单侧或双侧耳道出现与继发性外耳炎一致的临床症状。
Medical management of clinical signs in these 42 cats ranged from maintenance cleaning with various cleaning solutions to prescription otic topical medications for inflammatory and/or infectious otitis signs. No obvious pattern was seen, and comparison of effect was not performed on the different treatments, since patients were presented at various times during their treatment and disease progression.
这42只猫的临床症状的用药管理包括使用各种洗耳液进行维持性清洁，以及使用治疗炎症和/或感染性耳炎症状的处方外用耳药。由于患猫会在治疗和疾病进展的不同时间就诊，因此没有观察到明显的用药模式和不同治疗效果的比较。
Four cats (seven ears) needed to undergo total ear canal ablation and bulla osteotomy (TECABO) owing to progression of the cystadenomatosis and/or complications from chronic otitis externa and/or neoplasia in or around the ears. Two of the cats that underwent TECABO had initially had their diagnosis confirmed via histopathology , and had failed to resolve with CO2 laser therapy prior to needing more extensive surgery . A single case had a unilateral TECABO and two had pinnectomy performed prior to referral owing to a diagnosis of suspected cystadenomatosis.
由于囊腺瘤病的进展和/或慢性外耳炎和/或耳内或耳周围肿瘤的并发症，四只猫（七只耳）需要接受全耳道消融术和鼓泡截骨术（TECABO）。其中接受TECABO的两只猫最初通过组织病理学确认了诊断，在TECABO这种广大面积手术之前，CO2激光治疗未能解决问题。因疑似囊腺瘤病，一例患猫在转诊前进行了单侧TECABO，还有两例患猫进行了耳廓切除术。
The FIV/FeLV status of 5/57 cats was unknown.Fifty-one cats were either listed as negative for FIV and FeLV by owners, or as current or up to date with vaccine protection for FeLV . One cat was FIV-positive and was euthanized due to a brain tumor. One cat was considered positive for feline infectious peritonitis (FIP) but remained non-clinical for FIP . None of the other cats with unknown FIV/FeLV status had malignant neoplasia in the ears. Only three (5.3%) of the 57 cats with cystadenomatosis had signs and/or medical history of dermatologic problems and treatments suggestive of some form of allergic dermatosis, prior to developing clinical cystadenomatosis.
5/57只猫的FIV/FeLV状态未知。其中的51只猫要么被主人列为FIV和FeLV阴性，要么是受目前最新的FeLV疫苗保护。剩余的1只猫FIV呈阳性，因脑瘤被安乐死。这57只猫中有1只猫曾经被诊断是猫传染性腹膜炎(FIP)阳性，但目前FIP没有临床症状。其他FIV/FeLV状态不明的猫耳内均无恶性肿瘤。在57只患有囊腺瘤的猫中，只有3只(5.3%)在发展为临床囊腺瘤病之前，通过临床表现或用药史提示可能存在某种形式的过敏性皮肤病。
Discussion
讨论
In this retrospective study from a referral tertiary animal hospital population, we found that feline cystadenomatosis was not associated with certain breeds such as Persians or Himalayans, as previously suggested, although in a 2005 textbook one author noted that DSH cats were the most affected breed.In this study , cystadenomatosis was over-represented in non-purebred cats, though the association was not statistically significant. In the present study , male cats were 1.52 times more likely to develop ceruminous cysts than female cats, and affected male cats were more likely to be castrated than intact, consistent with previous studies and publications.This raises the question of whether male sex hormones could potentially affect glandular tissue and play some role in its progression into a cystic pathology , even with the majority of patients being spayed or castrated. Similar to previous studies, the condition appears to occur in cats as young as 3.5 years of age but more commonly affects older cats, as in our study population (mean of 11 years).
这项针对转诊的动物医院病例的回顾性研究发现，猫囊腺瘤病与某些品种(如波斯猫或喜马拉雅猫)无关，但在2005年的教科书中，一位作者指出DSH猫是最易患病品种。在本研究中，虽然其相关性在统计学上不显著，但发现囊腺瘤病在非纯种猫中比例过高。在现阶段研究中，雄猫患耵聍腺囊肿的可能性是母猫的1.52倍，耵聍腺囊肿的雄猫比没有发病的雄猫更容易被要求去势，这与之前的研究和发表的文献一致。这就带来了一个问题，即使大多数患猫已绝育或已去势，雄激素是否可能潜在地影响腺体组织，并在其发展为囊性病理过程中发挥一定作用。与之前的研究相似，这种情况似乎出现在3.5岁的猫身上，并且常见于年龄更大的猫，就如同我们的研究样本(平均11岁)一样。
Most cats had bilateral disease, with or without lesions associated with the ears (n = 4/57). The periocular zone appears to be the second most common area to develop cystic lesions (n = 2/57), which is consistent with the ophthalmology literature.The condition has been recognized in almost all regions of the world short of Oceania and Africa. In our view, this is more likely to be due to a lack of publications on the topic, rather a true reflection of the prevalence of disease. The most common clinical presentation beside cysts was the owner experiencing increasing difficulty with the management of secondary bacterial and/or Malassezia otitis externa. This was likely due to growth of the cystic lesions in the tragus and meatic entrance, making topical treatments more difficult for owners.
不论是否出现与耳相关的病变，大多数猫都有是出现双侧病变的 (n=4/57)。眼周似乎是发生囊性病变的第二常见区域(n=2/57)，这与眼科文献一致。除大洋洲和非洲外，世界上几乎所有地区都已知这种疾病。作者认为，这更有可能是因为大洋洲和非洲缺乏关于这一主题的文献，而并不能真实地反映了疾病流行率。除囊肿外，最常见的临床现象是宠主在处理继发性细菌性和/或马拉色菌外耳炎方面越来越多的困难。这可能是由于囊性病变在耳屏和耳道入口处的生长，使得外部治疗对于主人来说更加困难。
CO2 laser surgery using a model LX-20SP (www.aesculight.com) was carried out by two of the authors (KEL and BS) on a total of 34 ears and in three non-ear locations (chin, lip, perianal area) in 23 patients (three cats had TECABO or pinnectomy prior to referral). In this study , we found that CO2 laser surgery was, subjectively , a good treatment option for patients where cysts had reached a size or distribution that caused increased risk of otitis externa owing to the obstructive nature of the lesions, patient discomfort or increased difficulty in resolving secondary infections. Post-laser complications were short term and only three cats required 14 days of treatment. Regarding secondary infections, the most common clinical post-procedural observation by owners and clinicians was, surprisingly , minimal local discomfort for a few days after the procedure (Figure 4). However, a limitation of CO2 laser therapy is that it is unable to treat cysts that extend deep into the horizontal canal without causing damage to unaffected tissues. Diode laser and holmium type lasers will likely be advantageous in these cases, but the cost might be a limiting factor. We have carried out ‘touch-up’ treatment on four severely affected cases over the span of the study , mainly when the cats where under general anesthesia for other reasons such as for dental cleaning.
In two cats with malignant ear tumors, we performed a second round of CO2 laser treatment in an attempt to give those cats additional quality of life and allowing for topical palliative care, without resolving the malignancy
两位作者(KEL和BS)使用LX-20SP模型对23例患猫(其中3只猫在转诊前接受了TECABO或34耳廓切除术)的34只耳及3处非耳部位(下巴、嘴唇、肛周区域)进行了CO2激光手术。在本研究中，我们主观地认为， CO2激光手术在囊肿由于大小或分布导致患猫易患外耳炎、患猫不适或继发感染难以解决的情况下，是一种有效的治疗选择。手术后的并发症是短期的，只有3只猫需要进行为期14天的治疗。并且令人惊讶的是，至于继发感染，宠主或医生最常见的现象是动物在几天内仅出现轻微的局部不适（图4）。然而，CO2激光治疗的局限性在于，它无法在不损害未患病组织的情况下深入水平耳道治疗囊肿。二极管激光器和钬激光器在这些情况下是有优势的，但成本可能是一个限制因素。在整个研究过程中，我们对四个严重患病病例进行了“补充”治疗（touch up treatment），主要是因为猫正巧需要其他原因(如牙齿清洁)而进行全身麻醉。在两只患有恶性耳肿瘤的猫身上，我们进行了第二轮CO2激光治疗，尝试提高这些猫的生活质量，并进行局部姑息治疗，但没有解决恶性肿瘤。
Figure 4 Post CO2 laser of same cat as in Figure 1. Courtesy of Dr P Mouser
图4.与图1相同猫的CO2激光后。由Dr. P Mouser提供
The nine cats with concurrent non-cystadenoma medical conditions were considered to be within the normal occurrence in a population of senior cats, and we could not establish any pattern between their conditions and cystadenomatosis. No association with PV infection was found with the PCR testing carried out in 24 cases.Although FcaPV2, in particular, has been found to be causally involved in the etiology of certain human and feline cutaneous tumors,the virus may also be found in or on the skin of cats without any associated clinical signs.4The detection of FcaPV2 DNA in 4/24 tested cats indicates no connection between cystomatosis and PV infection. We also did not establish any association between FIV/FeLV or FIP and cystadenomatosis in our population. In a previous study , 1/4 cats with benign ear tumors tested positive for FeLV and two and one of 56 cats with malignant ear tumors were FeLV positive and FIV positive, respectively.
9例猫同时患有其他疾病（非囊腺瘤），这些疾病被认为是在老年猫中出现的正常疾病，作者无法将它们的病情与囊腺瘤病之间建立任何模式。24例猫进行PCR检测，未发现与PV感染相关。特别是FcaPV2，但它被发现与人类和猫皮肤肿瘤的病因相关，但该病毒也可能在猫的皮肤中或皮肤上被发现但没有任何相关的临床症状。在4/24只被测试的猫中检测到FcaPV2 DNA，表明囊腺瘤和PV感染之间没有联系。作者也没有在此研究的病例样本中建立FIV/FeLV或FIP与囊腺瘤病之间的任何关联。在之前的一项研究中，1/4患有良性耳部肿瘤的猫检测出FeLV阳性，56只患有恶性耳部肿瘤的猫中，FeLV阳性的猫有2只，FIV阳性的猫有1只。
Based on the documented communication with the client or owner, the cats in our study did not have a history of aural clinical signs prior to the cysts being noted by the owners and/or veterinarians. Only one of the affected cats was confirmed to have had ear mites (Otodectes cynotis) prior to the development of cystadenomatosis. However, the lack of previous history of otitis externa suggests that cystadenomatosis does not appear to be a reactive hyperplastic change for the majority of cases or appear to be over-represented among allergic or hypersensitive cats since only 3/57 had signs of allergic skin disease.Furthermore, the prevalence of ear mite infestations is highest in young individuals owing to transmission from the queen to the kitten.However, feline cystadenomatosis is more commonly found in cats that are middleaged and older and could support a possible senile or degenerative change in the tissue or its associated structures, and be less supportive of a congenital etiology .2The finding of neoplastic conditions in 17% of the cases is in line with what has previously been reported in the literature, where the mean age of onset for malignant ear tumors (such as ceruminous adenocarcinoma and squamous cell carcinoma) is 11 years.7In the present study , there was no significant association between cystadenomatosis and malignant ear neoplasia.
根据与客户或主人的交流记录，作者研究中的猫在被主人和/或兽医注意到囊肿之前没有耳部临床症状相关的病史。只有一只患猫在发生囊腺瘤病之前被证实患有耳螨。然而，外耳炎病史的缺乏表明囊腺瘤病对大多数病例来说似乎不是一种反应性增生性的改变，也似乎不过度地出现在过敏症或过敏反应的猫中，因为只有3/57表现出过敏性皮肤病的症状。此外，耳螨的流行率在年轻个体中最高，这是因为耳螨由母猫传播给小猫。然而，猫囊腺瘤病更常见于中老年猫，这个现象比较支持囊腺瘤由于组织或其相关结构的老年或退行性产生的病变，而不太支持先天性的病因。在本研究中，17％的病例中发现恶性肿瘤，这与先前文献中报告的情况一致，其中恶性耳肿瘤（如腺癌和鳞状细胞癌）的发病年龄平均为11岁。本研究发现，囊腺瘤与恶性耳肿瘤之间没有显著的相关性。
Conclusion
结论
Further investigation into the pathology of adenoma formation, and particularly into the feline apocrine and ceruminous glands, will be needed to identify specific etiologic factors and subsequent potential effective strategies for reducing the complications associated with cystadenomatosis.
减少与囊腺瘤病相关的并发症需要进一步研究腺瘤形成的病理学，特别是猫顶浆汗腺和耵聍腺，以确定特定的病因因素和后续潜在的有效治疗策略。
In our experience, CO2 laser therapy appears to be a useful approach for ablating cysts in more mild cases and minimizing the risk of obstruction in more severe cases.
根据作者经验，CO2激光治疗似乎是一种有效的方法，可消融较轻病例的囊肿，并最大限度地减少处理严重病例时，由耳道阻塞带来的风险。
It is possible that a similar effect could be achieved with cryotherapy and/or electrocautery , but we have had less success with this approach. In cases with moderate obstructive changes of the upper sections of the acoustic meatus, the reconstructive surgery proposed by Pavletic31 could be a viable consideration.
冷冻疗法和/或电灼疗法也有可能达到类似的效果，但我们使用这种方法的成功率较低。在耳道上部有中度堵塞的情况下，Pavletic建议可以考虑手术重建耳道。
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发表于 2023-6-24 16:54:49
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