Oral Carcinoma Cuniculatum Research Articles

Aggressive Oral Carcinoma Cuniculatum Presenting with Recurrence and Metastasis: A Case Report

Aggressive Oral Carcinoma Cuniculatum Presenting with Recurrence and Metastasis: A Case Report

Gingival Carcinoma Cuniculatum: A Case Series of Seven Cases with Emphasis on Immunohistochemical and Histochemical Features.

Oral carcinoma cuniculatum (CC) is a rare distinct variant of oral squamous cell carcinoma (OSCC) displaying unique clinical and histopathological features. CC is a locally aggressive malignancy with lower metastatic potential and has a tendency to invade the underlying bone yielding nonspecific clinical presentation resembling fungal/bacterial infections or osteomyelitis. Seven rare cases of gingival CC are reported with special emphasis on immunohistochemistry and special stains. Clinical and demographic profile were analysed and all cases were subjected to Ki-67 and p53 immunostaining and Ayoub-Shklar histochemical staining. For comparison, age and gender matched seven cases of conventional OSCC with bony involvement were recruited. IBM SPSS statistics software version 26 (IBM Analytics, Armonk, New York, U.S.) was used to analyse the data. All cases of CC lacked p53 expression in contrast to OSCC and demonstrated intermediate to high proliferation index in stroma also. The keratin staining and cytokeratin profile in OSCC and CC were also found to be statistically different. CC is a rare well differentiated variant of OSCC with different pathways involved in the pathogenesis. Local aggressive and invasive nature could be the result of unique interaction between epithelium and stromal elements. Oral health care provider must be familiar with this uncommon variant of OSCC.

Oral carcinoma cuniculatum, a rare variant of squamous cell carcinoma

IntroductionCarcinoma cuniculatum (CC) is a rare variant of squamous cell carcinoma (SCC). Lack of awareness of this tumor and its difficult diagnosis delay management and complicate treatment. The aim of this study is to describe the clinical and histological features, predisposing factors, treatment and survival rate of oral carcinoma cuniculatum. Material & methodAll patients with histologically confirmed CC of the oral cavity treated between January 2013 and August 2022 in a specialized center were retrospectively included. Patients were identified using the database ADICAP (“Association pour le Développement de l'Informatique en Cytologie et Anatomie Pathologique”). Once identified, the charts were reviewed. ResultsTen patients were included. CC lesions were mainly located in the mandibular or maxillary gingiva, while the 2 remaining lesions were located in the mobile tongue. The tumor showed local aggressiveness: mandibular or maxillary osteolysis was systematically found for gingival CC, while tongue lesions were classified cT3. Fifteen biopsies were performed in these 10 patients, histological diagnosis is difficult on a limited tissue sample. All patients underwent curative management with oncological excision surgery combined with neck lymph node dissection, and adjuvant radiotherapy (except one patient). DiscussionThe main problem is the difficulty of diagnosis when biopsies are performed, which leads to a significant delay in diagnosis compared with SCC. In the event of clinico-histological discordance after biopsy, CC should be considered.

Oral Carcinoma Cuniculatum: A Variant of Squamous cell Carcinoma with Unique Histological Characteristics and a Diagnostic Challenge.

Oral carcinoma cuniculatum (OCC) is a rare squamous cell carcinoma (SCC) variant with low incidence often due to misdiagnosis. This report aims to highlight this unique variant of SCC which is suggested to have a better prognosis with a focus on its histopathological features in comparison with similar clinical entities.

ORAL SQUAMOUS CELL CARCINOMA, VARIANT CUNICULATUM: A CASE REPORT

Oral carcinoma cuniculatum is a rare low-grade variant of squamous cells carcinoma, with distinct clinicopathologic outcomes. In the present case, a 63-year-old Black male patient presented with an exophytic mass in the posterior mandible gingiva for 6 months. The lesion appeared after removal of a third molar. Oral examination revealed firm exophytic verruciform growth. Radiographic exam showed irregular radiolucent lesion in the mandible, evidencing bone invasion. A biopsy was carried out with the diagnostic hypothesis of verrucous carcinoma. The initial histopathologic report was inconclusive of a mucosal fragment with papillomatosis and moderate dysplasia. A second and large incisional biopsy was conducted. Microscopically the lesion revealed atypical proliferation of keratinocytes forming multiple, complex, branching keratin-filled crypts, with a burrowing pattern. Mild cytological atypia, few mitotic figures, hyperchromatism, and low cell cohesiveness were also noted. With a final diagnosis of squamous cells carcinoma, subtype cuniculatum, the patient was referred for treatment.

Oral carcinoma cuniculatum, the great mimicker: Report of a challenging case and literature review

AbstractOral carcinoma cuniculatum (OCC) is a rare and locally invasive well‐differentiated variant of squamous cell carcinoma. To date, 74 cases affecting the oral cavity were reported in English‐language literature. We report a case of maxillary OCC in a 71‐year‐old woman and review the demographic, etiological, clinicopathological, therapeutic and prognostic features of the OCC cases published in the English literature. From the 75 cases evaluated, there was no predilection for sex, and the highest incidence was between the seventh and tenth decades of life. Use of alcohol (17.3%), tobacco (29.3%), both (6.7%), and betel chewing (5.3%) were related in some cases. The clinical presentation was variable, and the gingiva and mandibular alveolar ridge were the most affected sites (50.7%). Inconclusive result or misdiagnosis in the initial histopathological examination was reported in more than 40% of cases, which required deeper or more representative biopsies for the definitive diagnosis. Radical surgery was the treatment of choice for 88.0% of cases. Overall, 10.7% of cases had cervical lymph node metastasis and 17.3% had recurrence after initial treatment, with only one death (1.3% of the cases). In conclusion, the wide variety of clinical presentations, lack of cytological atypia in most cases and need for deep incisional biopsies make the diagnosis of OCC challenging. Radical surgery is the main therapeutic modality and local recurrence after initial treatment occurs in less than 20% of cases. Although locally aggressive, regional and distant metastases are rare and the prognosis is good.

Oral carcinoma cuniculatum: an unacquainted entity with diagnostic challenges\u2014a case report

BackgroundOral carcinoma cuniculatum (OCC) is an unacquainted well-differentiated subtype of oral squamous cell carcinoma, which displays unique clinic-pathological features. Up to date, OCC remains rare with about 75 reported cases, and is frequently missed or even misdiagnosed.Case presentationThe aim of the present work was: to report a case of OCC in mandibular gingiva and to highlight its main clinic-pathological diagnostic features: with an exophytic cobble-stone surface and a characteristic endophytic burrowing architecture, as well as to differentiate between it and other closely similar lesions including verrucous carcinoma, papillary squamous cell carcinoma, and well-differentiated conventional oral squamous cell carcinoma.ConclusionsAn accurate diagnosis of OCC entails awareness of the clinicians and pathologists about its entity, proper knowledge of the diagnostic clinical and histopathological evidence, and the ability to differentiate it from closely similar lesions.

Carcinoma Cuniculatum of the Oral Cavity: A Series of 6 Cases and Review of Literature.

Carcinoma cuniculatum (CC) is a rare variant of squamous cell carcinoma (SCC) that is characterized by minimal cytologic atypia and a unique deeply infiltrative growth pattern resembling rabbit burrows (cuniculi). With less than 75 cases reported in the head and neck, the clinical and pathologic spectrum of this entity remains poorly understood. A retrospective review of the clinical and pathologic features of archival cases of oral CC was performed. A total of six cases of oral CC were identified. Age ranged from 25-77 years; the male-to-female ratio was 5:1. All patients had a long-standing history of tobacco and betel-quid consumption. The tumors were distributed in the gingivobuccal sulcus (n = 2), the tongue (n = 2), buccal mucosa (n = 1), and the palate (n = 1). Histology in all cases typically revealed a tumor composed of well-differentiated squamous epithelium, devoid of atypia, lining deeply infiltrative, large-sized, branching, keratin-filled cavities, resembling rabbit-burrows. Dense lymphocytic infiltrates and discharging micro-abscesses were regular features. Underlying bone invasion and lymph node metastasis were observed in 1 patient. One patient with a tongue tumor developed locoregional recurrence at 10months while none developed distant metastasis. Oral CC is a rare and under-recognized variant of SCC with locally aggressive behavior. Lack of familiarity with this variant exacerbated by the absence of cytologic anaplasia makes CC susceptible to multiple negative biopsies and erroneous diagnoses. Awareness of this clinicopathologic entity is essential to allow its accurate diagnosis and optimal management.

Oral Carcinoma Cuniculatum Arising from Odontogenic Keratocyst: A Rare Case Report

Oral Carcinoma Cuniculatum Arising from Odontogenic Keratocyst: A Rare Case Report

GINGIVAL CARCINOMA CUNICULATUM: RARE VARIANT OF SQUAMOUS CELL CARCINOMA OFTEN MISDIAGNOSED AS AN INFLAMMATORY PROCESS

Carcinoma cuniculatum (CC) is a rare variant of oral squamous cell carcinoma (OSCC). To date, 50 cases of oral CC have been reported. A 67-year-old white female patient presented complaining of a painless tumor mass in the left mandibular body with several months of evolution. Previous dental history indicated extraction of teeth #37 and #38 and chronic osteomyelitis. Intraoral examination revealed an irregular exophytic mass in the region of the extracted teeth, which exhibited a yellow purulent discharge during palpation. Radiograph evidenced an osteolytic lesion with irregular and ill-defined borders. An incisional biopsy was performed, which showed an intense squamous epithelial proliferation, with frequent invaginations that delimited pseudocystic cavities filled with parakeratin and peeled cells. In the deepest part, foci of osseous resorption permeated by squamous epithelium were visualized. In conclusion, oral CC should be considered in the differential diagnosis, especially when assessing persistent "reactive inflammatory process." Carcinoma cuniculatum (CC) is a rare variant of oral squamous cell carcinoma (OSCC). To date, 50 cases of oral CC have been reported. A 67-year-old white female patient presented complaining of a painless tumor mass in the left mandibular body with several months of evolution. Previous dental history indicated extraction of teeth #37 and #38 and chronic osteomyelitis. Intraoral examination revealed an irregular exophytic mass in the region of the extracted teeth, which exhibited a yellow purulent discharge during palpation. Radiograph evidenced an osteolytic lesion with irregular and ill-defined borders. An incisional biopsy was performed, which showed an intense squamous epithelial proliferation, with frequent invaginations that delimited pseudocystic cavities filled with parakeratin and peeled cells. In the deepest part, foci of osseous resorption permeated by squamous epithelium were visualized. In conclusion, oral CC should be considered in the differential diagnosis, especially when assessing persistent "reactive inflammatory process."

CARCINOMA CUNICULATUM OF THE ALVEOLAR RIDGE

Carcinoma cuniculatum (CC) is a rare, well-differentiated variant of oral squamous cell carcinoma that usually affects the mucoperiosteum of older patients. Oral CC appears mainly as locally destructive tumors with superficial drainage of keratin, with low risk of metastasis. A 56-year-old white woman, a nonsmoker and nondrinker, presented a nonhealing gingival ulcer with duration of 2 months. Intraoral examination showed a well-defined irregular ulcer in the posterior upper alveolar ridge with drainage of a whitish material suggestive of keratin during palpation. Periapical radiograph showed an ill-defined osteolytic lesion. Microscopic evaluation showed well-differentiated proliferation of squamous epithelium with mild cytologic atypia exhibiting multiple infiltrative and branching keratin-filled epithelial crypts. Oral CC may appear as an irregular ulcer in the alveolar ridge with drainage of whitish material, which may be clinically similar to benign inflammatory diseases of the jaws. Carcinoma cuniculatum (CC) is a rare, well-differentiated variant of oral squamous cell carcinoma that usually affects the mucoperiosteum of older patients. Oral CC appears mainly as locally destructive tumors with superficial drainage of keratin, with low risk of metastasis. A 56-year-old white woman, a nonsmoker and nondrinker, presented a nonhealing gingival ulcer with duration of 2 months. Intraoral examination showed a well-defined irregular ulcer in the posterior upper alveolar ridge with drainage of a whitish material suggestive of keratin during palpation. Periapical radiograph showed an ill-defined osteolytic lesion. Microscopic evaluation showed well-differentiated proliferation of squamous epithelium with mild cytologic atypia exhibiting multiple infiltrative and branching keratin-filled epithelial crypts. Oral CC may appear as an irregular ulcer in the alveolar ridge with drainage of whitish material, which may be clinically similar to benign inflammatory diseases of the jaws.

Oral Carcinoma Cuniculatum: A Case Report

We report a case a of oral carcinoma cuniculatum, an exophytic variant of oral squamous cell carcinoma that has bland cytomorphologic features, and a peculiar and characteristic growth pattern. Despite the lack of cytologic atypia, the tumor exhibited locally aggressive and infiltrative behavior with bone and cutaneous involvement. Pertinent benign and malignant mimics, and helpful differentiating features are also discussed.

Oral carcinoma cuniculatum, an unacquainted variant of oral squamous cell carcinoma: A systematic review.

PurposeOral carcinoma cuniculatum is a rare well-differentiated variant of oral squamous cell carcinoma. The purpose was to systematically review its unique features to differentiate it from other variants as verrucous carcinoma, papillary squamous cell carcinoma and well-differentiated squamous cell carcinoma.Materials and MethodsA systematic review was performed using MEDLINE, Dentistry and Oral Sciences Source and PubMed databases and any existing articles related to the research subject missed in the search strategy to screen ones reporting cases occurring exclusively in the oral cavity in English literature. Variables analyzed included clinical, etiologic, imaging, histopatholgical features, treatment, follow-up and survival rates.ResultsFrom 229 hits, 17 articles with 43 cases were included in the systematic review. Clinically it showed a female predilection with pain and/or ulceration of a relatively long duration and exudation being the most common symptoms. Histologically, it showed more endophytic features comprising well-differentiated squamous epithelium with absent or minimal cytological atypia and multiple keratin filled crypts or cuniculus. Inflammatory stromal reaction and discharging abscesses were reported in most of the cases. Bone destruction was predominant in most imaging features. Complete surgical resection with a safety margin was the treatment of choice in most of the cases with few recorded recurrence cases.ConclusionApprehensive knowledge of oral carcinoma cuniculatum unique features is essential to avoid its misdiagnosis and provide proper treatment especially for recurrent cases.

Long-term survival and management of childhood oral carcinoma cuniculatum

Long-term survival and management of childhood oral carcinoma cuniculatum

Oral carcinoma cuniculatum: two cases illustrative of a diagnostic challenge

Carcinoma cuniculatum is a rare variant of squamous cell carcinoma originally described on the sole of the foot and for a long time believed to be restricted to the skin. However, since its first description in 1954, several cases have been reported at other sites. Carcinoma cuniculatum characteristically invades the underlying subcutaneous, submucosal, or bone tissues, forming so-called "rabbit burrows," with crypts filled with keratin that are of utmost importance for distinguishing this neoplasm from other variants of squamous cell carcinoma. Oral carcinoma cuniculatum is an even rarer entity that is frequently misdiagnosed initially, a phenomenon described in about one-third of previously reported cases. The aim of this report is: (1) to describe the clinicopathologic features of 2 new cases of oral carcinoma cuniculatum, initially misdiagnosed as infected orthokeratinized cyst and abscess, respectively; and (2) to review the related English language literature.

A clinicopathological and immunohistochemical study of oral carcinoma cuniculatum (CC)

A clinicopathological and immunohistochemical study of oral carcinoma cuniculatum (CC)

Clinicopathological evaluation of carcinoma cuniculatum: a variant of oral squamous cell carcinoma

Carcinoma cuniculatum (CC) is a rare neoplasm of oral squamous cell carcinoma (OSCC) and remains a histopathological challenge to pathologists because of its low incidence and unique architecture. Therefore, the purpose of this study was to illustrate the clinicopathological findings and biological behavior about this distinctive lesion. Clinical and follow-up information were retrieved from medical charts, and histopathological analyses were performed. Our series included 15 cases of CC, and the study included seven men and eight women, age 44-92. The tongue and mandible were the most frequently affected sites. There were three lymph node metastasis cases, and three local recurrences; one of the patients died of lung metastasis from oral CC. Histopathologically, the point of differential diagnosis with other subtypes of OSCC included unique, cuniculatum architecture and branching crypts. Because of its well-differentiated status, the prognosis of CC is usually optimistic. However, recurrent cases in our study might have possessed invasive biological characters that were not well controlled by conventional treatment; in one case, a portion of the CC transformed into conventional squamous cell carcinoma (SCC). This might suggest that transformed CC rather than primary CC should be further emphasized.

The sequential changes of carcinoma cuniculatum: a case report with 11-year follow-up

Carcinoma cuniculatum (CC) has been regarded as a well differentiated subtype of oral squamous cell carcinoma (SCC) that is characterized by keratin cores and keratin-filled crypts, but lacking obvious cytological malignancy. There have been relatively few case reports of oral CC, and descriptions of the long-term prognosis are scarce. In addition, the process of change from CC to conventional SCC is still poorly understood. In 1983, an 80-year-old woman admitted for a white spot underwent an excision biopsy, and CC was diagnosed. This lesion then recurred in 1993, with the same diagnosis, but it changed into conventional SCC when it recurred in 1994. Tissue materials from the three surgical treatments were examined with hematoxylin and eosin stain as well as with immunohistochemical stains for Ki-67 and for p53 and p63 gene products (P53, P63). While expressions of P53 protein and Ki-67 became stronger with every recurrence, the opposite tendency was obtained for P63 expression. This is the first report to describe a case of long-term follow-up of CC in which the expression modes of oncogene products were analyzed.

Oral Carcinoma Cuniculatum in a Young Child.

From the Department of Dental Specialties, Birmingham Children's Hospital. This case study describes a rare case of oral carcinoma cuniculatum in a 7-year-old female. She presented with an enlarged mass of the anterior maxilla arising from the gingiva. An anterior maxillectomy with immediate prosthetic replacement and obturation of the residual defect were carried out. The management of this case was challenging given the rare nature of the disease, unclear etiology, the patient's young age and the mutilating effects of surgery. The treatment involved a large multidisciplinary team. The provision of obturators was particularly difficult due to poor patient compliance and the extent of surgery carried out in a growing child. Oral cancer in children under 15 years old is extremely rare and this is the youngest case of oral carcinoma cuniculatum reported in the literature.