Post-orthognathic surgical ciliated cyst of the anterior maxilla: a case report and narrative review

Abstract

Background

Post-orthognathic surgical ciliated cyst (POSCC) is a benign, acquired cystic lesion associated with a history of orthognathic surgery, maxillofacial trauma, and similar events. This study presents a case of a maxillary POSCC that developed five years after a Le Fort I advancement osteotomy.

Case presentation

A 25-year-old male patient developed a painless, gradually fluctuant swelling in the anterior maxillary area following previous surgical treatment. Radiographic investigations revealed a unilocular radiolucent lesion from the left to right premolars, encompassing the apices of the teeth with a scalloped border. With clinicopathological correlation and consideration of the patient’s history, the diagnosis of surgical ciliated cyst was confirmed. Decompression followed by lesion excision was performed for the patient. A one year follow-up showed no recurrence or complications.

Conclusion

Although POSCC is a rare complication following surgery involving the maxillary bone and sinus, it can lead to large lesions with extensive growth. Long-term radiographic follow up is highly recommended after Le Fort osteotomy.

Introduction

The surgical ciliated cyst, also known as postoperative maxillary cyst or post-orthognathic surgical ciliated cyst (POSCC), was first described by Kubo in 1927. This uncommon benign acquired cystic lesion is associated with a history of radical sinus surgery, orthognathic surgery, maxillofacial trauma, upper molar retrograde root treatment, and sinus augmentation, with onset reported between 6 and 49 years post-procedure [1,2,3]. The lesion typically presents in adults in their 4th or 5th decade as a slow-growing maxillary swelling. It may be asymptomatic or accompanied with pain, discomfort, or swelling either in the intraoral or extraoral region. Additionally, mandibular POSCC have also been reported [4].

Though the pathogenesis of POSCC remains speculative, the prevailing theory is that it results from the sequestration of respiratory epithelium within the wound during surgical procedures or trauma. This is followed by an inflammatory process, cystic changes, and mucin secretion [5].

Radiologically, POSCC typically presents as a well-defined, unilocular or multilocular radiolucency, with or without bony perforation [2]. Histologically, the cyst is lined by pseudostratified ciliated columnar epithelium, or in some cases, cuboidal epithelium interspersed with goblet cells [6].

This study presents a patient with a massive POSCC that appeared in the maxilla five years post Le Fort I advancement osteotomy. The long-term follow-up and management of such a lesion are particularly important, as they provide valuable insights into the evolution and clinical handling of POSCC several years after orthognathic surgery. This case emphasizes the need for vigilant post-surgical monitoring and underscores the potential for delayed complications, helping to fill a gap in the current understanding of POSCC in the context of orthognathic surgery.

Case report

Due to a dentofacial deformity, a 25-year-old male patient underwent Le Fort I surgery in 2017. The patient remained symptom-free until March of 2022 when he developed a painless, gradually enlarging mass in the anterior maxillary area, several years after his previous surgical treatment. The patient’s past medical history was unremarkable. Intraoral examination revealed a large well-defined, fluctuant swelling located beneath the upper lip and in the anterior palatal area (Fig. 1).

Fig. 1

Intraoral view of the lesion. The white arrows indicate the bone perforation associated with the lesion, which has resulted in swelling in the palatal mucosa

An endodontic evaluation confirmed that all the adjacent teeth were vital, but their response was slightly reduced compared to the adjacent teeth. Radiographic investigations, including Panoramic imaging and cone beam computed tomography, revealed a unilocular radiolucent lesion extending from the left premolars to right premolars, from the alveolar bone to the nasal floor and involving left and right maxillary sinuses. The lesion encompassed the apices of the mentioned teeth’s with a scalloped border. Root resorption and blunting of the incisors, canines and right first premolar was observed. The cyst measured 38.8 × 25.8 × 24.8 mm, leading to thinning, expansion and significant perforation of the buccal and palatal cortical plates (Figs. 2, 3 and 4).

Fig. 2

Extraoral swelling in nasal base area

Fig. 3

Initial panoramic view of the patient. Note the scalloped shape of lesion between the roots, accompanied by minimal periapical destruction

Fig. 4

The initial cone beam computed tomography (CBCT) view shows perforation of the buccal, palatal, and nasal floor walls

Following aspiration of Thick bloody mucous fluid, an incisional biopsy was performed under local anesthesia through an intraoral approach via an incision in the labial sulcus. Histopathological examination revealed a cystic lesion lined by a single layer of cuboidal to columnar cells, with areas of stratified columnar ciliated cells resembling respiratory mucosa containing numerous goblet cells. Occasional mucus goblet cells were also seen in the epithelium, along with scattered chronic inflammatory cells in the surrounding connective tissue (Fig. 5A, magnification 100x). The presence of ciliated epithelium and goblet cells confirmed the diagnosis of a surgical ciliated cyst. A higher magnification (Fig. 5B, magnification 400x) illustrates the chronic inflammatory infiltration in the connective tissue and occasional mucus goblet cells.

Fig. 5

A: Histopathological image showing pseudostratified ciliated columnar epithelium (magnification 100x). B: Higher magnification showing occasional mucus goblet cells and chronic inflammatory cells in the connective tissue (magnification 400x)

With clinicopathological correlation and the patient’s history, the diagnosis of a surgical ciliated cyst was confirmed. Given the significant size and scalloped characteristics of the lesion, a decompression treatment plan was considered for the patient. During the incisional biopsy surgery, the thick secretions of the lesion were carefully washed and drained. Subsequently, over the following three months, the secretions were aspirated and drained using a high-gauge needle during monthly visits. As the thickening of secretions persisted, a second-stage surgery for marsupialization was required, during which a sterile Nelaton catheter connector was used to keep the created window open. The patient was advised to continue daily washing until the discharge was completely clear.

Upon the 9-month post-operative assessment, follow-up imaging revealed a significant reduction in the size of the lesion, indicating favorable conditions for lesion excision (Figs. 6 and 7). Under local anesthesia (Lidocaine 2% with 1/80000 epinephrine), access to the lesion was obtained via sulcular incision. The lesion was then carefully separated from the nasal floor and the palatal mucous membrane. The resulting defect was filled with mineralized bone allograft with 150–1000 μm particle size (Regen allograft, Iranian Tissue Products, Iran) and lyophilized cellular dermis membrane (Iranian Tissue Products, Iran) was used to cover the buccal wall and nasal floor (Fig. 8). Excision of the entire lesion via curettage and enucleation confirmed the initial diagnosis.

Fig. 6

A panoramic view of the lesion after nine months of decompression and marsupialization shows bone formation around the roots and a reduction in the size of the lesion

Fig. 7

The cone beam computed tomography (CBCT) view of the lesion after 9 months of decompression and marsupialization shows bone formation in the palatal wall and nasal floor. The buccal window created for marsupialization, as well as the device used to keep the window open, are also visible

Fig. 8

A: access to the lesion through the created window. B: placement of membrane to separate the lesion from the nasal mucosa and allograft particles to fill the cavity. C: the view of the reconstruction before suturing

One year after the removal of the lesion, the teeth remain asymptomatic. Given the unreliability of vitality tests post-surgery, the endodontist recommends periodic radiographic evaluations to assess the condition of the teeth and determine the need for root canal treatment if necessary (Figs. 9 and 10).

Fig. 9

Two months after the final surgery and bone reconstruction. There is no evidence of infection or recurrence

Fig. 10

One year after the final surgery and bone reconstruction. Note bone formation in periapical area of affected teeth

Discussion

This case report presents a massive POSCC that developed in the maxilla five years after a Le Fort I advancement osteotomy. The lesion was managed with decompression, excision, and bone grafting, with a successful outcome. The long-term follow-up highlighted the delayed onset of POSCC, emphasizing the importance of vigilant post-surgical monitoring for potential complications even years after orthognathic surgery. This study is important as it highlights a delayed occurrence of a POSCC in the maxilla, emerging five years after Le Fort I advancement osteotomy, a scenario not commonly encountered in the literature. The rarity of POSCC itself, combined with the long-term follow-up provided in this case, offers significant clinical value by shedding light on the evolution and progression of this lesion over an extended period. Although POSCC is generally recognized in the immediate post-surgical phase, this report emphasizes the potential for its delayed onset, underscoring the need for ongoing post-surgical monitoring even several years after orthognathic procedures. The case further reinforces the necessity for clinicians to remain vigilant for late complications, contributing to a more comprehensive understanding of the long-term management of patients who undergo major facial surgeries. In addition to the clinical relevance, this case adds to the literature on POSCC by providing a detailed account of its management in the context of orthognathic surgery, highlighting the standard treatment approach (decompression, excision, and bone grafting) with a favorable outcome. It provides valuable insights into the clinical decision-making process in handling such rare lesions and serves as a reminder for surgeons to consider this possibility in their differential diagnosis when managing post-surgical patients. This report also helps to bridge the gap in understanding between early and delayed occurrences of POSCC, which emphasizes the need for future research to explore long-term outcomes and better define the risk factors for its development.

The surgical ciliated cyst is a non-odontogenic lesion that can occur as a complication following sinus surgery, traumatic dental extraction, and orthognathic surgery [1, 2]. POSCC mostly affects maxilla, especially in the posterior region. It may present as an asymptomatic lesion or as an expansile swelling in the vestibule, accompanied by pain, purulent discharge in cases of secondary infection [7]. Li et al. reported a case involving complaints of numbness in lower lip and chin [8]. Cases of mandibular POSCC have also been documented, predominantly occurring in the anterior mandibular area and, in some cases, in ramus, often with a history of prior genioplasty with nasal cartilage autograft and rhinoplasty, genioplasty concomitant with maxillary osteotomies, chin augmentation, and Le Fort surgery [9, 10]. The time interval between surgery and diagnosis of the lesion ranges from six to 49 years. This report differentiates itself from previously documented cases by providing a detailed narrative of a POSCC that occurred five years post-surgery, which is at the higher end of the reported latency period. Most prior cases described in the literature report shorter latency periods, ranging from 2 to 22 years after orthognathic surgery (Table 1). The current case underscores the potential for POSCC development even after mid-range follow-up periods, a finding not commonly emphasized in prior reports (Table 1).

Table 1 Main clinicopathological features of post-orthognathic surgical ciliated cyst

Radiographic evaluation of POSSC typically reveals a well-demarcated unilocular or multilocular radiolucent lesion that may be surrounded by sclerotic borders [2]. In our case, a well-defined unilocular radiolucent lesion was observed. This radiographic presentation is consistent with other cases in the literature, such as those reported by Hayhurst et al. [12] and Kim et al. [13], who also observed unilocular radiolucent lesions in the maxilla. Clinically and radiographically, POSCC can be included in the differential diagnosis of pulpoperiapical lesions, which can be distinguished through a pulp vitality test. In addition, traumatic bone cyst, glandular odontogenic cyst, keratocyst, and central giant cell granuloma must be considered. Positive aspiration and fluid drainage can aid in diagnosis [31]. Furthermore, a mucocele is another lesion that must be rule out. The growth pattern and epicenter of these two lesions differ: a mucocele grows within the maxillary sinus and expands the sinus walls, while POSCC’s epicenter is in trabecular bone, with expansion originating from there [9, 32].

In evaluating POSCC histopathologically, there are several epithelial linings that can be observed, each providing valuable insight into the lesion’s nature and behavior. These linings include ciliated pseudostratified columnar epithelium (respiratory epithelium), squamous epithelium with mucous cells, and metaplastic squamous epithelium [10]. The variations in epithelial presentation are crucial in understanding the pathogenesis and biological behavior of the cyst, as well as in guiding treatment strategies. In our case, the histopathological examination revealed a squamous epithelium with mucous cells lining the lesion, which aligns with findings in the study by Gates et al. [6], who also reported squamous epithelium with mucous cells in their cases of POSCC. This type of epithelium suggests a metaplastic transformation from normal respiratory epithelium, possibly due to chronic irritation or mechanical injury, which is consistent with the origin of the lesion in the sinonasal region. The mucous cells found within the squamous epithelium may have contributed to the cystic nature of the lesion, promoting its expansion and subsequent bone erosion.

In terms of treatment, various techniques have been proposed, including transnasal endoscopic surgery and Caldwell-Luc approach for simple cystectomy, cystectomy with antrostomy, and marsupialization for thin-walled, unilocular cysts with extensive bony perforations [9, 32, 35]. However, each technique comes with its own set of advantages and limitations. Transnasal endoscopic surgery, for example, offers minimally invasive access but may not be suitable for larger, more complex lesions or those near vital structures. Similarly, while the Caldwell-Luc approach provides excellent access to the maxillary sinus, it can be associated with a higher rate of postoperative complications such as infection or damage to the nasal structures. In comparison, our case utilized a combined approach of decompression, excision, and bone grafting, a strategy that was also seen in other cases, such as those by Sugar et al. [11] and Bourgeois et al. [16], who employed excision and curettage or enucleation, but with a greater emphasis on simpler excision methods. This combined approach, while more invasive, was necessary due to the lesion’s extensive size and involvement with the surrounding bony structures. All investigations agree that due to the expansile and destructive behavior of POSCC, surgical removal is necessary, unlike sinus mucocele, which arises from ostial obstruction and can often be treated with marsupialization without the need for extensive surgical treatment. In our case, a combined approach of decompression, excision, and bone grafting was employed, considering the lesion’s extent and proximity to important anatomical structures such as the maxillary sinus and nasal floor. Interestingly, while most of the cases in the literature (e.g., Amin et al. [15] and Golaszewski et al. [30]) involved enucleation or curettage as the primary treatment, our patient’s case required a more complex intervention due to the lesion’s size and location, emphasizing the need for tailored management (Table 1).

Recurrence is rare, although it may occur due to possible remnant cyst lining in the area. A review of the literature on POSCCs reports two cases of recurrence after treatment- one after 5 years and the other one after 2 years [8, 9]. While recurrence is uncommon, it underscores the need for ongoing monitoring post-treatment. Long-term follow-up strategies vary in the literature, from frequent imaging during the first 2–3 years to annual imaging. Our case, with a 2-year follow-up period, demonstrated a successful outcome with no recurrence, which aligns with the findings of Lee et al. [22], who also observed no recurrence at 21 months. This suggests the efficacy of the combined approach used, which included decompression and excision. However, recurrence may still be a consideration in future cases, and long-term follow-up is important.

The choice of treatment plan depends on the size of the lesion and its proximity to nearby structures. A study by Sfondrini et al. [3] recommends marsupialization for large lesions that carry a risk of jaw fracture. This study highlights additional benefits of marsupialization. Enucleation becomes more challenging for lesions that have infiltrated between the roots, making bone curettage impossible. The process of marsupialization encourages thickening of the cyst wall and shrinking of the lesion, which can eliminate scalloping and facilitate enucleation. Additionally, for large lesions in the maxilla that compromise the sinus wall and nasal floor, marsupialization can promote the formation of thin bone between these cavities. This helps surgeons remove the lesion without damaging the delicate membranes of the sinus and nasal floor. Despite these benefits, the effectiveness of marsupialization in large, aggressive lesions like POSCC remains uncertain, particularly in terms of long-term recurrence and functional outcomes. In contrast to this approach, our case opted for a direct excision and bone grafting method due to the lesion’s defined nature, similar to the approach used by Shakib et al. [20] and Moe et al. [19], who focused on excision without marsupialization. Overall, this study demonstrates that marsupialization positively impacts the reduction of intra-cystic pressure in SCC cases. For our patient, the decision to perform decompression and excision with bone grafting was supported by the lesion’s significant size and involvement of the surrounding bony structures, making a more conservative approach less suitable.

Another consideration is the use of local versus general anesthesia during treatment. In this case, all procedures were conducted under local anesthesia. The initial surgery for the incisional biopsy presented some challenges in achieving adequate anesthesia, as sensitivity in the sinus mucosa persisted despite administering an anterior superior alveolar nerve block. However, as the lesion shrank, the excision was performed under local anesthesia with minimal issues. Other studies have indicated that excisions can also be successfully completed in one step using general anesthesia with favorable outcomes [3, 36].

Conclusions

Although POSCC remains a rare but significant complication following surgeries involving the maxillary bone and sinus, it has the potential to evolve into large and aggressive lesions, leading to substantial growth and potential bone involvement. Given this, it is crucial to emphasize the need for long-term radiographic monitoring following procedures such as Le Fort osteotomy to detect any emerging or recurrent lesions at an early stage. Early identification allows for more effective intervention, minimizing the risk of further complications. Additionally, the inclusion of POSCC in the differential diagnosis for any unexplained sinonasal mass or cystic lesion is essential for avoiding both misdiagnosis and inappropriate management. Considering the varied clinical presentations and histopathological diversity of POSCC, maintaining a high index of suspicion can significantly improve treatment outcomes and reduce the chances of delayed or inadequate therapeutic measures. Therefore, a multidisciplinary approach with careful follow-up is recommended to ensure optimal patient care and outcomes.