一例犬头部局部广泛性毛囊错构瘤伴毛囊囊肿和皮样囊肿 ...

王帆

Locally extensive follicular hamartomas with concurrent follicular cysts and dermoid cysts on the head of a dog

一例犬头部局部广泛性毛囊错构瘤伴毛囊囊肿和皮样囊肿

 

翻译：美联众合动物医院爱康分院 王羽迪

 

Abstract  

摘要

A 5- month- old, male intact Australian shepherd dog was presented with nodular lesions affecting the dorsal head. A locally diffuse form of follicular hamartomas, with concurrent dermoid cysts and follicular cysts, was diagnosed by histopathological examination. Complete surgical excision of the affected skin and closure with a subdermal plexus advancement flap led to long- term resulotion. 

一只5月龄，未去势雄性澳大利亚牧羊犬出现波及头部背侧的结节性病变。经组织病理学检查为局部广泛性毛囊错构瘤，同时伴有皮样囊肿和毛囊囊肿。手术完全切除患病皮肤，真皮下神经丛推进皮瓣闭合，达到长期消退。

 

INTRODUCTION

简介

Canine follicular hamartomas (FHs) are considered to be developmental skin lesions. The age, breed, site predilections and recurrence rates have not been reported.  Typically single and occasionally multiple, FHs present as skin nodules with tufts of thick, brush-like hairs arising from dilated follicular ostia.Distinctly large anagen hair follicles with normal orientation may be associated with an increase of dermal collagen.Mildly dysplastic and tortuous folliculosebaceous units may be observed. Follicular hamartomas can expand over time, potentially involving a larger area of skin and sometimes requiring multiple surgical procedures.

犬毛囊错构瘤(FHs)被认为是发育性皮肤病变。年龄、品种、易感部位和复发率尚未报道。典型单一病变，偶见多灶性，FHs表现为皮肤结节，伴一簇浓密的、刷子样的毛发从扩张的毛囊口长出。明显可见正常方向的大的生长期毛囊，可能与真皮胶原蛋白的增加有关。可以观察到轻度发育不良和曲折的毛囊皮脂腺的单位。毛囊错构瘤会随着时间的推移而扩大，可能涉及更大的皮肤区域，有时需要多次手术。

 

In contrast to FHs, follicular cysts (FCs) are fairly common in dogs and can present as solitary or multiple lesions.Rupture of the cysts results in a marked endogenous  foreign body reaction. Presumably of congenital aetiology, clusters of FCs have been reported on the dorsal midline of the head of young dogs.

与FHs相反，毛囊囊肿(FCs)在犬中相当常见，可以表现为单发或多发病变。囊肿破裂导致明显的内源性异物反应。据推测是先天性病因，在幼犬头部背中线有FCs簇。

 

Dermoid cysts (DCs) are considered to be inherited, congenital dermal and subcutaneous cysts caused by incomplete separation of the skin and neural tube during  embryonic development.The term dermoid sinus  (DS) has been mostly used for lesions which maintain a connection with the vertebral column.Both DCs and DSs are most commonly reported in the Rhodesian ridgeback dog, and have been described in other dog breeds.Cervical and thoracic dorsal midline are predilection sites, while the head and sacrococcygeal region are less often affected. The DC is lined by squamous epithelium from which small folliculosebaceous units,referred to as “folliculettes”,are seen within the wall or radiate into surrounding dense collagen. If DCs open to the skin surface, tufts of very delicate hair shafts emerge. Complete surgical excision is curative if there are no neurological signs associated with cyst extension to the spinal cord.

皮样囊肿(DCs)被认为是具有遗传性、先天性真皮和皮下囊肿，是由于胚胎发育过程中皮肤和神经管的不完全分离引起的。术语“皮样窦”(DS)主要描述用于与脊柱连接的病变。DCs和DSs最常见于罗得西亚脊背犬，在其他犬种也有描述。颈和胸背中线是好发部位，而头部和骶尾部则较少患病。DC内衬鳞状上皮细胞，从中有小的毛囊皮脂腺单位(称为“毛囊”folliculettes)位于壁内或辐射到周围的致密胶原蛋白中。如果DCs开口于皮肤表面，则会出现一簇非常细小的毛干。如果没有与囊肿延伸到脊髓相关的神经系统症状，则完整手术切除可治愈。

 

The current case presented with a locally extensive area affected by a combination of FCs, FHs and cysts with features of DCs.

本病例表现为局部广泛性区域患病，同时出现FCs、FHs和具有DCs特征的囊肿。

 

CASE REPORT  

病例报告

A 5- month- old, male intact Australian shepherd dog presented with a history of nodular lesions and partial alopecia of the parietal region of the head, present since adoption two months previously. The affected area appeared mildly sensitive on palpation and minimally pruritic.Some nodules were erythematous. Treatment with amoxicillin-clavulanic acid and cefpodoxime (see Table S1 in Supporting information) decreased the erythema, yet nodules remained unchanged. Cytologica examination revealed neutrophils and cocci. Complete blood count and blood chemistry were unremarkable.

一只5月龄的雄性未去势的澳大利亚牧羊犬，2月前被收养，头顶有结节性病变和部分脱毛病史。触诊患处时表现为轻度敏感和轻度瘙痒。一些结节病变发红。使用阿莫西林-克拉维酸和头孢泊肟治疗(见补充信息表S1)发红减轻，但结节未见变化。细胞学检查显示中性粒细胞和球菌。全血细胞计数和血液化学无异常。

 

Upon presentation to the veterinary teaching hospital's dermatology service, the dog was still receiving cefpodoxime, as well as monthly lotilaner (Table S1).  Multifocal, 0.2– 1 cm diameter, firm, raised, dermal nodules affected most of the parietal region of the head. Many nodules had a large, singular, follicular ostium  with numerous emerging hair shafts. Other nodules lacked an obvious skin opening. Mild erythema and crusting were noted within the lesional area. The skin between the nodules was smooth, slightly thin, and alopecic, suggestive of scar tissue (Figure 1a, b).

就诊于兽医教学医院皮肤科时，该犬仍接受头孢泊肟以及每月的洛替拉纳驱虫(表S1)。皮肤病变多灶性，头顶大部分区域可见直径0.2-1厘米，坚硬，凸起的真皮结节。许多结节具大的，单一毛囊开口，并长出许多毛干。其他结节缺乏明显的皮肤开口。病变区域内发现轻度皮肤发红和结痂。结节间皮肤光滑、稍薄、脱毛，提示瘢痕组织 (图1a，b)

 

 

FIGURE 1 Locally extensive follicular hartomas (FHs), follicular cysts and dermoid cysts on the dorsal aspect of the head (a) Irregular region of complete alopecia on dorsal head with partial alopecia along cranial portion of lesion. (b) Same area postclipping, revealing several crusted nodules and multiple large follicular ostia with numerous emerging hair shafts within the alopecic, wrinkled skin. (c) Postoperative view of the subdermal plexus advancement flap. (d) Complete hair regrowth with no FH recurrence nine months postoperatively

图1 头部背侧局部广泛性毛囊错构瘤(FHs)、毛囊囊肿和皮样囊肿(a)头部背侧完全脱毛的不规则区域，沿病变的头侧有部分脱毛。(b)同一区域剃毛后表现，在脱毛、褶皱的皮肤中发现几个结痂性结节和多个大的毛囊开口，并有大量毛干出现(c) 真皮下丛推进皮瓣的术后视图。(d) 术后9个月毛发完全再生，无FH复发。

 

Surface cytological examination revealed a few cocci and occasional inflammatory cells, suggesting superficial pyoderma. Trichoscopic examination of hair shafts  plucked from lesional areas was unremarkable. Under sedation, three 6– 8 mm punch biopsies were collected  for histopathological examination. While obtaining the  skin biopsies, rare, pinpoint draining tracts with a brown exudate were noted, and the same exudate drained  into the open biopsy sites. Cytological examination of  the exudate revealed numerous cocci, and aerobic culture confirmed meticillin-susceptible Staphylococcus pseudintermedius. Cefpodoxime was continued as  prescribed previously.

表面细胞学检查发现一些球菌和偶见炎症细胞，提示浅表性脓皮病。从病变区域拔下毛干进行毛发镜检，无明显变化。在镇静下，取三处6-8毫米皮肤打孔活检进行组织病理学检查。在获得皮肤活检时，注意到罕见的，带有棕色渗出物的针尖大小窦道，并且相同的渗出物开口于活检部位。渗出物的细胞学检查显示出许多球菌，需氧细菌培养证实为甲氧西林敏感的假中间型葡萄球菌。按照以前的处方继续使用头孢泊肟。

 

Histopathological examination revealed two types of lesions. Some nodules were composed of large anagen hair follicles accompanied by increased dermal collagen.  Some follicles had an irregular shape (Figure 2a). Another section revealed deep, large compound hair follicles in anagen phase merging into a more superficial  irregular cyst lined by a thin squamous epithelium, surrounded by dense fibrosis and mixed inflammatory cells (Figure 2b); the cyst communicated with the skin  surface and contained keratin flakes, cocci and numerous hair shafts. Mild perivascular lymphoplasmacellular inflammation and occasional small superficial crusts were present.

组织病理学检查发现两种类型的病变。一些结节由大的生长期毛囊组成，并伴真皮胶原蛋白增加。一些毛囊具有不规则形状(图2a)。另一切片显示，处于生长期的深而大的复合毛囊合并成一个更浅表的不规则囊肿，内衬一层薄的鳞状上皮细胞，周围有致密的纤维化和混合炎性细胞(图2b);囊肿与皮肤表面连通，并包含角蛋白片、球菌和大量毛干。存在轻度的血管周淋巴浆细胞炎症和偶见浅表小结痂。

 

 

FIGURE 2 Multifocal follicular hartomas (FHs), follicular cysts (FCs) and dermoid cysts (DCs) (haematoxylin & eosin, ×20) (a) FHs with mild dysplastic changes, (b) DC type IIIb, (c) FCs and (d) ruptured cysts with endogenous foreign body reaction (furunculosis)

图2多灶毛囊错构瘤(FHs)、毛囊囊肿(FCs)和皮样囊肿(DCs) (苏木精和伊红，20倍) (a) FHs伴有轻度发育不良变化，(b) IIIb型DC，(c) FCs (d) 破裂的囊肿伴内源性异物反应 (疖病)

 

A diagnosis of FHs with mild follicular dysplasia  (Figure 2a), fibrosis and marked atypical cyst formation was given, with the cysts demonstrating features reminiscent  of DCs (Figure 2b).

诊断为FHs伴轻度毛囊发育不良(图2a)，纤维化和明显的非典型囊肿形成，囊肿表现为DCs相似特征(图2b)。

 

One month after presentation, the dog underwent general anaesthesia to remove the lesion en bloc to the level of the deep subcutaneous tissue. The skin defect  produced by surgical excision was roughly 9 cm in diameter and was closed with a subdermal plexus advancement flap (SPAF; Appendix S1).

就诊后一个月，犬接受了全麻，手术将病变整体切除，深度到达皮下组织。手术切除产生的皮肤缺损直径大约9厘米，并使用真皮下丛推进皮瓣技术(SPAF; 附录S1)。

 

The excised skin had dozens of small dermal nodules (<1 cm diameter), some demonstrating numerous  hair shafts protruding from a single follicular ostium. On  cut section, solid nodules and well- demarcated cysts  were noted. The latter contained hair shafts and pasty, tan material. Some cysts communicated with the skin  surface; others were completely enclosed within the  dermis. In addition to histological features seen in the  biopsies submitted previously, intradermal cysts with  no connection to the skin surface were noted. These cysts were lined by thin squamous epithelium with keratohyalin granules and lacked associated hair follicles or adnexa (Figure 2c). Multifocal ruptured cysts resulted in endogenous foreign body reactions centred on free hair  shafts and keratin (Figure  2d). Surgical margins were clean, and narrow.

切除的皮肤有数十个小的真皮结节 (直径＜1厘米)，其中一些显示出许多毛干从单个毛囊口长出。在切面上，发现了实性结节和边界清晰的囊肿。后者包含毛干和膏状棕褐色物质。一些囊肿与皮肤表面相连，其他的则完全封闭在真皮内。除了在先前提交的活检中看到的组织学特征外，还注意到与皮肤表面不相连的皮内囊肿。这些囊肿内衬有薄的鳞状上皮细胞和透明角质颗粒，缺乏相关的毛囊或附件(图2c)。多灶性囊肿破裂导致以游离毛干和角蛋白为中心的内源性异物反应(图2d)。手术边缘干净，狭窄。

 

The final diagnosis included locally extensive FHs  and coexisting cysts with and without associated  follicles and adnexa, the former presenting features of DCs while the latter were considered FCs. Some cysts had ruptured, eliciting a secondary foreign body reaction.

最终诊断包括局部广泛性FHs，并存含或不含相关毛囊和附件的囊肿，前者表现出DCs的特征，而后者被认为是FCs。一些囊肿破裂，引起继发性异物反应。

 

Postoperatively, tension from the SPAF led to  mild dorsal bilateral displacement of the superior eyelids, with a normal blink response still present.Displacement of the eyelids partially improved during four days of postoperative hospitalisation. A drain system was in place for two days postoperatively, and the dog was discharged from the hospital on cefpodoxime, trazodone and carprofen (Table S1). Aerobic culture of serous exudate from the surgical site yielded no growth.

术后，SPAF的张力导致上眼睑的双侧背侧轻度移位，仍然存在正常的眨眼反应。术后住院四天，眼睑的移位得到了部分改善。术后引流系统放置了两天，犬出院后使用头孢泊肟、曲唑酮和卡洛芬(表S1)。手术部位血清渗出液进行需氧细菌培养没有生长出细菌。

 

A postsurgical seroma was addressed with a Penrose drain and, upon recurrence, with warm compresses. Multiple tension- releasing skin incisions  mildly improved the persistent dorsal displacement of the superior eyelids at three months (Figure S1). The SPAF site fully healed with complete hair regrowth; at the time of writing, there has been no recurrence of lesions 23 months postsurgery (Figure 1d).

术后血清肿用Penrose引流管解决，复发后用热敷。多个释放张力的皮肤切口在三个月时轻微改善了上眼睑的持续背侧移位 (图S1)。SPAF部位完全愈合，毛发完全再生; 在撰写本文时，术后23个月无复发病变(图1d)。

 

DISCUSSION  

讨论

To the best of the authors' knowledge, this is the first report of an extensive skin area affected by coexisting FHs and cysts, with the latter presenting features of  either FCs or DCs. Numerous DCs have been reported on the head of a Rottweiler, Saint Bernard and miniature schnauzer.Boxers and ridgebacks have been reported to be predisposed to clusters of FCs on the dorsal head.

据作者所知，这是首例关于皮肤广泛性FHs合并囊肿的报道，后者具有FCs或DC的特征。据报道，在罗威纳犬、圣伯纳犬和迷你雪纳瑞犬的头部高发DCs。据报道，拳师犬和脊背犬容易在头部背部形成簇状FCs。

 

The solid nodules had features typical of FHs, including large anagen hair follicles retaining normal orientation and increased associated fibrous tissue with no  evidence of cyst formation.Some follicles displayed areas with irregular shape, suggestive of mild dysplasia, as reported previously.

实质结节具有FHs的典型特征，包括保持正常方向的大生长期毛囊和相关纤维组织增加，没有囊肿形成的症状。如先前报道，一些毛囊显示出不规则形状的区域，提示轻度发育不良。

 

Cystic lesions with tufts of hair emerging from their ostia affecting the regions of the coronal sutures and squamosal sutures support the diagnosis of DCs in this Australian shepherd dog. Parasagittal DCs on the dorsal head may be associated with defective embryological fusion of the bone synostoses of the cranial vault.The squamous epithelium lining the cyst walls had emerging folliculosebaceous units,  features described with DCs.However, these large, regularly spaced anagen compound hair follicles and associated glands were limited to the deep portion of the cystic space, while follicles described with DCs are typically small, referred to as folliculettes.Typical folliculettes radiate perpendicularly from the cyst wall  and may or may not have adnexa.According to a classification system developed for canine DSs, which includes DCs, these would represent type IIIb DSs, as no fibrous cords extended below the subcutaneous tissue.

囊肿病变的开口长出一簇毛发，影响了冠状线和鳞状线的区域，支持了澳大利亚牧羊犬DCs的诊断。头背侧的旁矢状侧DCs可能与颅穹隆的骨缝融合胚胎学缺陷有关。囊肿壁内衬鳞状上皮细胞出现毛囊皮脂腺单位，描述了DCs特征。然而，这些大的、有规则间隔的生长期复合毛囊和相关腺体仅限于囊腔的深部，而用DCs描述的毛囊通常很小，称为毛囊（folliculettes）。典型的毛囊从囊壁垂直向周围生长，含或不含附件。根据针对犬DSs分类系统，其包括DCs，判断这些为IIIb型DSs，因为皮下组织以下没有纤维索延伸。

 

The second type of cysts observed had features consistent with infundibular FCs; they were completely enclosed within the dermis with no connection to the skin surface. These cysts were lined by infundibular type epithelium and lacked associated hair follicles and glands.In a previous publication, similar lesions seen in a Saint Bernard were classified as type Vb DSs.

观察到的第二种类型的囊肿具有与漏斗状FCs一致的特征，它们完全封闭在真皮内，与皮肤表面没有任何联系。这些囊肿内衬有漏斗状上皮细胞，缺乏相关的毛囊和腺体。在先前的出版物中，在圣伯纳犬中看到的类似病变被归类为Vb型DSs。

 

In conclusion, this is the first report of a locally extensive area affected by FHs and cysts with and without associated folliculosebaceous units. Follicular origin was considered for all three different lesions seen in this young dog, and clinical and histological features suggest a more complex process characterised by the coexistence of FHs, FCs and DCs or, alternatively, FHs and DCs previously classified as DSs types IIIb and Vb. After successful management of  seromas and tension- releasing skin incisions to address  dorsal displacement of superior eyelids,the surgical site healed and no recurrence was reported 23 months postoperatively.

总之，这是首例局部广泛区域FHs合并囊肿含有和不含相关毛囊皮脂腺单位的报道。在这只年轻犬中看到的所有三种不同病变都认为是毛囊起源，临床和组织学特征表明，FHs、FCs和DCs共存，或者FHs与以前归类为IIIb型和Vb型DSs的DCs共存，是更复杂的过程。在成功处理血清肿和皮肤切口张力释放以解决上眼睑的背侧移位后，手术部位愈合，术后23个月未报告复发。

 

 

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