WGDCs have received little attention in published literature, and data on surgical outcomes are scarce. Since Weatherhead reported his case series of Wolfring dacryops in 1991 [1], only few case reports have addressed this topic [2, 12, 15, 16]. Other case series included several types of dacryops without a specific characterization of WGDCs. To the best of our knowledge, this is the largest case series describing the characteristics and surgical outcomes of WGDCs.

WGDC has been reported to be associated with conjunctival cicatrization, particularly trachoma, but the exact etiology has not been determined [1, 3]. In the present study, palpebral conjunctival scarring was present in 87.5% of the patients (79.2% due to trachoma and 8.3% due to VKC). Galindo-Ferreiro et al. reported that up to 85% of dacryops have been linked to earlier conjunctival scarring based on a review of the literature [3]. Trachomatous scarring was present in all the patients in Weatherhead’s series [1].

This association could explain the relative rarity of dacryops in Western countries; Salam et al. reported only 10 patients in an 18-year period in the UK [13]. Von Holstein et al.17 reported 24 patients in a 33-year period in Denmark [17], and Lam et al. identified 5 patients with dacryops over a 3-year period in the USA [18]. Most large series of accessory lacrimal gland cysts were reported in Saudi Arabia, previously endemic for trachoma [1, 3, 11].

Cicatrization seems to play a key role in the pathogenesis of dacryops [5, 19]. Our findings of the presence of fibrotic bands along the proximal tarsal border in all patients, palpebral conjunctival scarring in most patients and chronic inflammatory cells observed in most specimens may support this mechanism. Previous reports of accessory lacrimal ductal cysts in the context of conjunctival scarring also support this theory [1, 4, 15].

However, the role of cicatricial ductular blockage as the sole factor in dacryops formation has been challenged experimentally [9]. Some authors propose concurrent IgA hypersecretion by activated plasma cells with a resultant osmotic effect [1, 18]. Others have suggested dysfunction of the periductular neural plexus [5].

The presence of lacrimal glandular tissue next to the cyst is considered the main histologic clue to lacrimal gland (LG) dacryops [5]. However, this diagnostic feature was not universally present in most patients in the present study (for only 1/4 of the patients). Lacrimal gland acini were absent in all the patients in Weatherhead’s series [1]. The associated lacrimal tissue is smaller in accessory lacrimal gland lesions than in main LG lesions. Moreover, glandular acinar tissue undergoes progressive atrophy during dacryops formation [1, 5]. This can be missed without meticulous tissue sampling and sectioning. Additionally, meticulous surgical separation of the cyst from any surrounding tissues, including lacrimal glandular tissue, may explain the lack of associated lacrimal tissue.

A microscopic serpiginous configuration of cyst cavities was present in most patients (43/48) in our series. This finding was highlighted by Jakobiec et al.5 and was present in 12/14 of their patients.

In the present study, the mean age at presentation was 28.5 years (range 3–66). In previously reported cases of accessory lacrimal gland ductal cysts, the mean age at disease onset was 30.5 years (range 2–81). Reports including main LG dacryops preponderance showed a higher mean age ranging from 48 to 52 years, based on a review of the literature by Fung et al. [16].

A female predilection was reported by Shield et al. [20] (75%), Alsarhani et al. [11] (72.7%) and Betharia et al. [8] (100%). Similarly, there was a female prevalence (64.6%) in our study. This can be attributed to the fact that trachomatous scarring, the most frequent etiological association of WGDCs in this study, occurs at higher rates in women [21].

WGDCs can be diagnostically challenging with a possibility of misdiagnosis [5, 11].

Clinical clues aiding in the diagnosis include the cyst’s subconjunctival location, adherence to the nonmarginal tarsal border, immobility, lack of tenderness, and positive transillumination. These lesions more frequently appear in the upper eyelid medially or centromedially, likely because more WGs exist in this location [1, 3, 11, 19]. WGDCs tend to grow insidiously; however, a rapid increase in size may occur and this should raise the suspicion of cyst complications such as hemorrhage or infection [19]. A transverse fibrotic band along the nonmarginal tarsal border in the area of the cyst was noted in all patients, independent of the eyelid affected. These fibrotic changes were different from those observed in palpebral conjunctival scarring or in Arlt’s line, which is pathognomonic for trachoma. This finding was observed in the photographs in previous reports and may have been overlooked [2, 3, 16]. It is unclear whether this finding represents a sequela of previous conjunctival inflammation that leads to obstruction of several adjacent duct orifices contributing to dacryops formation or whether it is simply a reactive response to fluid leakage from the cysts.

Distinguishing WGDCs from other cysts in the eyelid or anterior orbit can pose diagnostic complexities, both clinically and histologically. Krause gland ductal cysts exhibit similar characteristics; however, they are anatomically located within the fornices and lack fixation to the tarsal plate [22]. Another condition that is frequently confused with WGDC is conjunctival inclusion cyst, which results from the entrapment of conjunctival epithelial tissue within the substantia propria. These cysts are typically lined by either a double layer of low cuboidal cells or, more commonly, multilaminar nonkeratinizing squamous cells [5, 11, 23]. Absence of history of trauma or surgery, fixation to the tarsal plate, serpiginous microscopic cyst configuration, and presence of bilaminar cuboidal cells, rather than multilaminar nonkeratinizing squamous cells, are indicative features of WGDCs rather than conjunctival inclusion cysts [5, 11].

Various treatment modalities for dacryops have been described. Generally, complete excision through a conjunctival cul-de-sac route has been accepted as the treatment of choice for accessory lacrimal gland ductal cysts [1, 3]. However, this method is associated with more adverse events than the transcutaneous approach in our series. Intraoperative cyst rupture was greater with the transconjunctival route. The scarred conjunctiva restricts lid eversion of the eyelid, particularly in the upper eyelid, with poor cyst exposure. Additionally, everted lids force thin-walled cysts under tension and increase the risk of rupture. In contrast, the transcutaneous approach allows wider exposure of the cyst. Dissection can be performed easily away from the area of fibrotic adhesions and tarsal attachment where cyst rupture usually occurs.

Intraoperative cyst rupture did not significantly increase the risk of cyst recurrence in the study groups. Nevertheless, early cyst rupture with wall collapse during surgery poses challenges in identifying the cyst capsule through the conjunctiva, increasing the likelihood of incomplete excision of the cyst wall and the potential for recurrence [24].

An incision through the scarred conjunctiva leads to a large gap with an increased risk of tissue loss, making primary closure difficult. Therefore, conjunctival grafting is more frequently needed with this approach.

Postoperative ptosis was significantly greater in patients treated via the transconjunctival route. A direct conjunctival incision may trigger conjunctival inflammation and further scarring and shortening of the fornix, increasing the risk of mechanical ptosis. In contrast, the skin approach allows exploration of the levator aponeurosis and repair of dehiscence, if any [1].

While the higher detection of orbital signs in the transconjunctival group might suggest more advanced forms of disease, intraoperative inspection of the cysts showed no orbital extension. This indicates that the detected orbital signs were not necessarily due to advanced disease. This is further supported by the lack of significant difference between the two groups in the intraoperative measurements of the cyst.

Less invasive surgical methods, including simple aspiration, needling and marsupialization, have been used [12, 13]. Cysts readily refill after simple aspiration. Similarly, we feel that needling is an inappropriate procedure, and only one reported case was treated with needling [12]. Six of our patients had recurrent cysts following aspiration or needling performed before being referred to our institution.

Marsupialization entails deroofing of the cyst wall and is a less invasive treatment. The procedure does not require meticulous dissection of the cyst wall, which is technically trickier and more time consuming [13]. However, this approach may lead to recurrence with additional scarring in the future.

The retrospective nature of the study, nonrandomization, and small sample size are notable limitations to the present study. In addition, there may have been some bias with regards to transcutaneous versus transconjunctival approach selection by the surgeon. The authors historically utilized the transconjunctival approach for removing the WGDCs but have progressively switched to the transcutaneous approach in recent years. This shift was prompted by observing a notable occurrence of intraoperative cyst rupture, postoperative ptosis, and chronic eye irritation associated with conjunctival grafting associated with the transconjunctival route. Due to insufficient data on patients treated with the transconjunctival approach in earlier times, they were excluded from the study. This explains the relatively smaller number of patients and longer duration of follow-up among those treated with this approach.