Ocular surface squamous neoplasia is a common neoplasm of ocular surface with significant morbidity [26]. It is a great masquerader and can masquerade as conjunctivitis, inflamed pterygium or an abnormal growth at limbal area, superior limbic conjunctivitis, necrotising scleritis and sclerokeratitis [27, 28, 29, 30]. Hence, it is often misdiagnosed and not treated appropriately resulting in recurrence of the lesion. The traditional treatment in localized OSSN is excision with no- touch technique combined with adjuvant therapies to mitigate recurrence rate. However, the recurrence rate is about 5% and regional metastasis is about 2% even with the modern techniques due to the microscopic extension of disease beyond the edge of the surgical margin at the time of presentation because of late diagnosis of this clinical masquerade.[12,14,15] The other causes of recurrence are the tumor size, positive surgical excision margins, location of tumor, histopathological grade, lack of adjuvant treatment in addition to surgical excision and length of follow-up [17, 20].
In our study the mean interval between initial treatment and recurrence was 07 months. In the current study recurrence was found more common in the sixth decade with a male preponderance. This is in concordance with the study which was undertaken by Li et al, Kirre et al,Cevantes et al, Kim et al, Meel et al and Mckelvie et al as they also reported higher incidence of this disease in sixth decade and in males [15, 26, 29, 31, 32, 34]. However, all these studies reported similar finding in recently diagnosed cases of OSSN. Risk factors which we found in our study like smoking, HIV infection, Xeroderma Pigmentosa and immunosuppression were similar to studies conducted by Li et al, Kim et al and Meel et al.
In our study limbus was involved in all cases and most of the lesion were small (5 mm in size).This is in conformity with Li et al, Meel et al which also showed the similar finding [15, 33]. However, Meel et al and Mirzayev et al reported higher frequency of medium to larger tumors in their study [33, 35]. The common clinical presentation in our study was papilliform mass which was similar to study conducted by Kim et al [32]. But current studies finding were in discordance with Li et al which reported maximum patient had leukoplakic lesion(16/43), whereas Meel et al found majority of tumors had a gelatinous appearance(35/57,61.4%) [15, 33]. The main cause of recurrence in our study was found to be incorrect diagnosis hence not treated appropriately. The other reason of recurrence of the lesion in this study as patients were not followed-up at their initial centre where they were treated. Hence, awareness among general population about the entity is important so that these lesions are diagnosed early and treated on time with appropriate treatment modalities. Galor et al reported tarsal involvement and positive pathologic margins were the strongest predictors of clinical recurrence. [12] Yousef and finger et al reported that tumor size more than 5 mm is associated with a higher risk of recurrence [4]. All patients with recurrent OSSN had unilateral presentation. This is in accordance with Kiire et al,Kim et al and Meel et al which also reported the similar findings [26, 32, 33]. Most of the patients had already undergone excision somewhere else before reporting at our centre. The common pathological grade which was found in current study was invasive squamous cell carcinoma.This is in conformity with Mirzayev et al which also reported invasive SCC in most of the lesion( 46.4%) [35]. However, these findings are in discordance with Li et al and Kao et al as they found more number of squamous cell carcinoma in situ during histological examination of biopsied tissues [15, 36]. The treatment with different treatment modalities achieved good clinical outcomes in all cases of recurrent OSSN in the current study with no recurrence of the lesion over a period of 12 months. This is similar to study conducted by Sturges et al and Palamer et al as they also reported nil recurrence in their study population during a follow -up period of 35 months [20, 37]. But current study finding differed from Li et al and Nanji et al which, as in their studies recurrence rate was 7% and 6% respectively after a mean follow-up period of 24 months [15, 38]. In16 patients complete regression of the tumor was noted whereas in one patient there was reduction in the size of the tumor but lesion did not disappear completely. In patients who underwent wide excision with amniotic membrane graft transplantation, AMG was found displaced after one week.The AMG was placed again over bare area and secured with fibrin glue. Two patients in whom we started interferon, they complained of pain, irritation which subsided after administration of lubricants. The patient who underwent exenteration complaining of oozing from the operated site, which subsided after pressure bandage of few days. Other than this we did not encounter any major complications in our study. In a study conducted by Kirre at al, corneal scarring was found in 09 patients, symplepharon in 01 patient and allergic reaction in 01 patient [26]. Mirzayev et al reported dry eye symptoms in 14 (12.5%) patients after topical chemotherapy and mild stem cell deficiency in 5 (4.5%) cases [35]. Overall, these studies also reported minimal complication rates in their studies like our study.
The present study is unique in many ways. In all published data regarding OSSN, whatever may be the mode of treatment, recurrence was seen in few percentage of cases. But in the current study we did not find any sign of recurrence after 12 months of follow- up period. Our institutional devised treatment protocol yielded good results in the study population,hence this treatment protocol may be considered for managing recurrent cases of OSSN. Second, to the best of our knowledge, our study addressed the outcome in recurrent OSSN among our clientele for the first time as there are very few published studies which have analysed the outcome in recurrent OSSN worldwide and in India till date. Third, the common cause of recurrence in the current study was found to be misdiagnosis of the lesion and not following patients on time whereas in other studies reported larger size of the lesion, immunosuppression, positive pathological margins, multifocal lesion, nodular morphology and incomplete excision as the common cause of recurrence in their study population. Fourth, the current study found majority of the tumors were small when treated initially somewhere else, so size of the lesion plays no role in increased rate of recurrence.Whatever is the size of the lesion,if not treated properly it will definitely escalate the incidence of recurrence in the affected population.
The limitation of study is small sample size and short follow-up. The outcomes of the study may not be the true representative of the entire population. Recurrence of the disease has been reported upto12 years after surgical excision in some published studies.[10] Hence, longer follow-up and study from larger population need to be carried out to validate the outcome result in recurrent OSSN.