Multiple keratocystic odontogenic tumors in a non-syndromic minor patient: Report of an unusual case

Rai, Shalu; Rana, A S; Kalra, Puneet; Gupta, Deepak; Goel, Sumit

doi:10.4103/0975-8844.113708

Cited by 3 publications

(4 citation statements)

References 2 publications

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“…showed non-syndromic multiple keratocysts and multiple radiolucencies with ill-defined borders in the maxilla [11] . Rai et al reported multiple radioloucencies with well-defined borders in both the mandible and maxilla [6] .…”

Section: Discussionmentioning

confidence: 99%

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Non-syndromic multiple keratocystic odontogenic tumor: A case report

Sohanian

Seifi

Dezfouli

et al. 2017

JOA

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A Keratocystic Odontogenic Tumor (KCOT) is a benign odontogenic lesion that is well-known for its histopathological features and invasive clinical behaviors. KCOTs may be non-syndromic or syndromic, and the syndromic type presents a higher risk of recurrence and invasive behaviors. Non-syndromic KCOTs are uncommon and account for only 5% of the cases. The purpose of the present case report is to conduct clinical, radiographic and pathologic evaluations and treat a 37-year-old man with non-syndromic nevoid basal cell carcinoma. Syndromic and non-syndromic keratocysts present more invasive behaviors than the solitary type.The initial symptoms of syndromic keratocysts appear as multiple keratocysts of the mandible and maxilla that are diagnosed by a dentist; as a result, the diagnosis and treatment of the disease is very crucial.

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Section: Discussionmentioning

confidence: 99%

“…Odontogenic keratocysts may turn into invasive neoplasms such as ameloblastoma and squamous cell carcinoma. The syndromic and non-syndromic types of keratocysts are more invasive than the solitary types [6] .…”

Section: Introductionmentioning

confidence: 96%

Non-syndromic multiple keratocystic odontogenic tumor: A case report

Sohanian

Seifi

Dezfouli

et al. 2017

JOA

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“…OKCs when associated with NBCCS occur earlier in life and are associated with higher tendency to recur and are more aggressive than nonsyndromic cases and occasionally may get transformed into aggressive neoplasms such as ameloblastoma and squamous cell carcinoma. 4 OKCs are the most common form of cystic lesions affecting the maxillofacial region with an incidence rate of 12-14% and two peaks around the ages of 30 and 60 years, and seem to be more frequent in males (M/F 2:1). Around 60-80% of the reported cases occur in the mandible, mainly in the molar, angle, and ramus areas.…”

Section: Introductionmentioning

confidence: 99%

“…These multiple lesions may be the first manifestation of the NBCCS or otherwise, it may be attributed to the multifocal nature of OKCs. 4 This syndrome is known to express a high degree of penetrance, variable expressivity, and is Journal of Contemporary Dentistry, Volume 9 Issue 1 (January-April 2019) 47 characterized by several developmental defects. Among patients with NBCCS, 65-75% have OKCs.…”

Section: Introductionmentioning

confidence: 99%

Multiple Odontogenic Keratocysts in Nonsyndromic Patients—A Case Report and Review of Literature

Jamwal¹,

K²,

Ramakanth³

et al. 2019

Journal of Contemporary Dentistry

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Aim:The aim of this study is to highlight a behavioral pattern of odontogenic keratocysts (OKCs) in syndromic and nonsyndromic patients in terms of clinical course, genetic makeup, local destruction, management modalities, and recurrences such that detection in initial stages and intervention when done prevents extensive morbidity and a better quality of life. Background: OKC is a common developmental odontogenic cyst affecting the oral and the maxillofacial region, which arises from the dental lamina or its remnants. Multiple OKCs are usually seen in association with the nevoid basal cell carcinoma syndrome (NBCCS) but approximately 5% of the cases have multiple cysts without concomitant syndromic presentation. As it spreads by way of the bone marrow, it results in more destruction before any clinical manifestation appears. Although lesions are benign but are aggressive, locally destructive, and highly recurrent. Case description: A 15-year-old male patient, a rare case of nonsyndromic multiple OKCs, is presented here along with a review of literature with emphasis on the importance of thorough evaluation, diagnosis, treatment strategy, and strict follow-up. Conclusion: Although our case does not fulfill the major and minor criteria of syndrome, but considering the reported literature and clinical presentation, early age, multiple OKCs, involvement of both the jaws, cleft lip and cleft palate, and histological correlation, we suggest our case to be a partial expression of NBCCS. Clinical significance: Any case presenting with single or multiple OKCs should be thoroughly investigated locally and systemically for any other signs of NBCCS or other syndrome. A definite treatment protocol should be formulated depending on patient's age, number and size of lesion, remaining subsequent growth, and proximity to adjacent vital structures, to provide a better quality of life with a minimal amount of morbidity and kept on strict regular follow-up.

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