Unilateral gingival lymphangioma: A first case report.

Lymphangiomas are classified as vascular malformations. A 17-year-old male patient was referred with asymptomatic gingival enlargement with a vesicular feature on the buccal aspect of the central and lateral teeth of the right mandible; the lesion showed easy bleeding. After local anesthesia, subgingival, and sulcular incisions was performed on the buccal and lingual gingiva of the right central and lateral incisors of the mandible. Microscopic evaluation showed lymphangioma. A 3-year follow-up showed promising results.

INTRODUCTION

Vascular anomalies cause identifiable birthmarks of the skin and mucosa and a variable degree of underlying soft tissue abnormalities; they are congenital aberrancies in vascular development.[1] Lymphangiomas are relatively rare lesions characterized by the proliferation of lymphatic vessels. Currently, the lymphangiomas are classified as malformations and not neoplasms, with a marked predilection for the head and the neck region. The oral localization of lymphangiomas is less usual, and the most common site is the tongue, especially the anterior two-thirds.[2] Usually, it is superficial in location and shows a pebbly surface that resembles a cluster of translucent vesicles. The surface has been likened to the appearance of frog eggs or tapioca pudding.[3] Although, they are usually present at or around the time of birth, they usually manifest within the first two decades of life.[4] These benign, painless, soft malformations affect both sexes equally.[3] Only three unusual cases of bilateral symmetrical lymphangiomas of the gingiva have been reported by Josephson and van Wyk,[5] McDaniel and Adcock[6] and Motahhary et al.[7] This article reports a first time case of unilateral lymphangioma of the gingiva, its treatment and 3-year follow-up results.

CASE REPORT

A 17-year-old male patient was referred to a periodontist with a complaint of gingival enlargement on the buccal aspect of the central and lateral teeth of the right mandible [Figure 1], with the lingual side intact. Complete blood count, bleeding time, clotting time, prothrombin time, and partial thromboplastin time of the patient were normal. In the clinical examination, vesicular asymptomatic lesions with easy bleeding were found on the buccal aspect of attached and marginal gingiva in the right central and lateral incisors of the mandible. The surface of the lesion was intact without any ulcer present. Before being referred, the patient had scaling and root planning (SRP) done, except for the two involved teeth. He said that the lesion appeared gradually 1 year ago and he experienced easy bleed during biting of solid foods. In the radiographs, alteration could not be detected. Initial treatment included oral hygiene instruction and deep SRP with an ultrasonic device (Woodpecker®, Guilin Woodpecker Medical Instrument Co., China) was done. A plaque control record ≥ 90% was achieved before surgery. Two weeks after SRP [Figure 2], a biopsy was taken. The surgical procedures consisted of local anesthesia (Persocaine-E® Lidocaine Hcl 2%, epinephrine 1:80,000, Daroupakhsh Co., Iran), subgingival and sulcular incisions on the buccal and lingual gingiva on the distal of the right canine to the distal of the left first incisor of the mandible, reflection of the flap, granulation tissue debridement, thorough SRP with an ultrasonic device (Woodpecker®, Guilin Woodpecker Medical Instrument Co., China), inner thinning of the flap, normal saline irrigation, and suture with silk 0-4. There was abundant bleeding during the surgery. The specimen was sent to the Oral Pathology Department of the Babol University of Medical Sciences for microscopic examination by differential diagnosis of pyogenic granuloma and hemangioma. This specimen was diagnosed as a lymphangioma by the following histopathological features: The lesion composed of lymphatic vessels located just beneath the epithelium surface; they contained proteinaceous fluid and few lymphocytes. Furthermore, lymphoid aggregation in the walls of the vessels was observed, endothelium of the vessels was thin, and some channels contained red blood cells. Extension of these vessels into the deeper connective tissue and skeletal muscles were seen [Figure 3]. The patient was recalled 3 weeks after the surgery [Figure 4] and the gingiva showed mild inflammation. However, he did not attend another follow-up appointment until 11 months after surgery. Since his oral hygiene was low, copious calculus and some gingival hyperplasia on the buccal aspect of the central and lateral teeth of the right mandible were seen in this session [Figure 5]. SRP was done and the gingiva was examined 2 weeks later. Enlargement subsided, but gingiva showed mild inflammation. The patient was instructed in correct oral hygiene and then he was visited 1 and 3 months later; the inflammation disappeared. Hence after, he attended his follow-up appointments regularly every 3 months with appropriate oral hygiene. After that, there was no sign of inflammation in the involved area in his follow-up sessions and his 3-year follow-up showed no sign of recurrence.

Figure 1

Gingival enlargement on the buccal aspect of the right central and lateral incisors of the mandible

Figure 2

Two weeks after scaling and root planing

Figure 3

Histopathological features of the lesion (H and E staining); (a) At ×10 (b) At ×40

Figure 4

Three weeks after the surgery

Figure 5

Eleven months after surgery

DISCUSSION

The following issues concerning lymphangiomas of the gingiva are controversial and hence we will discuss them in detail: The manifestation site, treatment methods, histological features, origins, and rate and possible causes of recurrence.

The manifestation site

Josephson and van Wyk[5] have reported a symmetrical bilateral lymphangioma of the mandibular gingiva in a 16-year-old female patient. McDaniel and Adcock[6] have reported a case of bilateral superficial lymphangiomas between the maxillary right central and lateral incisors, the maxillary left central and lateral incisors, and the mandibular right canine and lateral incisor in an 11-year-old female patient. Motahhary et al.[7] had reported lymphangiomas present in the lateral and canine teeth of the maxilla in a 32-year-old male on both sides. However, we have reported the first case of unilateral lymphangioma of the gingival on the buccal aspect of central and lateral incisors of the right mandible in a 17-year-old male patient.

Treatment approaches

Josephson and van Wyk[5] removed bilateral symmetrical lymphangiomas of the mandibular gingiva by conventional gingivectomy. However, McDaniel and Adcock[6] and Motahhary et al.[7] performed excisional biopsies to remove the lymphangiomas.

In the present case, subgingival and sulcular incisions were done on the buccal and lingual gingiva of the right mandibular central and lateral incisors. The lesion involved the attached gingiva; therefore, removal of the whole gingiva could not be applied. Furthermore, thinning of the flap was performed to prevent gingival loss of the attachment.

Histopathological features

Traditionally, lymphangiomas were divided into three groups: Simplex (capillary), cavernous, and cystic.[8] Although, Bill and Sumner[9] suggested that histological differences in various lymphangiomas are attributed to the differences in anatomic location and therefore, histological classification is of little benefit. According to this concept, some authorities classify lymphangiomas into superficial and deep types. Josephson and van Wyk[5] have reported that both of the lesions were a cavernous type and apparently confined to the gingiva. McDaniel and Adcock[6] reported the diagnosis as marked lymphangiectasia, consistent with superficial lymphangioma and Motahhary et al.[7] considered their case as superficial lymphangioma. Lymphangiomas were reported in our case report as superficial lymphangiomas.

Origin

Josephson and van Wyk,[5] and McDaniel and Adcock[6] stated that the distribution and location of the lesions indicate that they are developmental anomalies, probably arising from residua of neonatal lymphangiomas. Furthermore, confined growth potential and bilateral distribution suggests a hamartomatous nature and probably a developmental origin.

Motahhary et al.[7] suggested that bilateral and symmetrical distribution of lesions, in addition to lack of infection or surgery (that may cause localized lymphatic obstruction) rule out the possibility of acquired lymphangiectasia. Conversely, confined growth potential and bilateral distribution suggests a hamartomatous nature and probably a developmental origin.

The probability of neoplasm of lymphangioma in our patient because of lack of continuous growth was eliminated and the probability of development of the lesion considering that it did not exist at the time of birth is low; therefore, it is highly probable that the lesion is hamartomatous.

Rate and possible causes of recurrence

Josephson and van Wyk[5] reported no recurrence after 2 years. They reported that both lesions were of a cavernous type and apparently confined to the gingiva. For this reason, it seems that local excision is adequate even if the mass is incompletely removed, because anomalies do not recur in the same fashion as a tumor.

McDaniel and Adcock[6] reported that 10 months after the initial biopsies, residual vesicles were observed between the maxillary left central and lateral incisors and between the lateral incisor and canine. At that time, a decision was made to observe the lesions periodically for regressive or progressive changes. Shortly thereafter the patient was lost to follow-up.

Motahhary et al.[7] have reported no sign of recurrence after 10 months.

In the 3-year follow-up of our patient, lesion recurrence was not observed. Low inflammation 11 months after the operation was observed at the site. However, by observing proper oral hygiene the inflammation disappeared.

CONCLUSION

Considering that there have been only a few studies done on this lesion and the difference in reported site manifestation, it is presently impossible to specify the lesion's most prevalent site. In our study, we reported the first case of unilateral gingival lymphangioma, although three other studies report bilateral involvement. Hence, lymphangioma should be considered in the differential diagnosis of unilateral lesions of the gingiva.

According to our findings, surgery was an appropriate treatment option to remove existing lesions of the gingiva. Moreover, poor oral hygiene can cause calculus formation leading to erythema and so clinicians should be careful in not associating these problems with the removed lesion and include instructing the patient in good oral hygiene as part of the follow-up care.