Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts

<h3>Objective</h3><p dir="ltr">This case series reports the clinical presentation and management of multiple <u>odontogenic keratocysts</u> (OKCs) in patients with <u>Gorlin Goltz</u> syndrome and in non-syndromic patients. </p><h3>Mate...

وصف كامل

محفوظ في:
التفاصيل البيبلوغرافية
المؤلف الرئيسي: Omer Sefvan Janjua (21347903) (author)
مؤلفون آخرون: Ramish Tariq (21347906) (author), Muhammad Usman Khalid (19457347) (author), Sana Mehmood Qureshi (21347909) (author), Kamran Ali (8861576) (author)
منشور في: 2022
الموضوعات:
الوسوم: إضافة وسم
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author Omer Sefvan Janjua (21347903)
author2 Ramish Tariq (21347906)
Muhammad Usman Khalid (19457347)
Sana Mehmood Qureshi (21347909)
Kamran Ali (8861576)
author2_role author
author
author
author
author_facet Omer Sefvan Janjua (21347903)
Ramish Tariq (21347906)
Muhammad Usman Khalid (19457347)
Sana Mehmood Qureshi (21347909)
Kamran Ali (8861576)
author_role author
dc.creator.none.fl_str_mv Omer Sefvan Janjua (21347903)
Ramish Tariq (21347906)
Muhammad Usman Khalid (19457347)
Sana Mehmood Qureshi (21347909)
Kamran Ali (8861576)
dc.date.none.fl_str_mv 2022-03-31T09:00:00Z
dc.identifier.none.fl_str_mv 10.1016/j.omsc.2022.100253
dc.relation.none.fl_str_mv https://figshare.com/articles/journal_contribution/Presentation_and_management_of_syndromic_and_non-syndromic_patients_with_multiple_odontogenic_keratocysts/29045423
dc.rights.none.fl_str_mv CC BY 4.0
info:eu-repo/semantics/openAccess
dc.subject.none.fl_str_mv Biomedical and clinical sciences
Clinical sciences
Dentistry
Paediatrics
Gorlin Goltz syndrome
Odontogenic
Keratocyst
Nevoid basal cell carcinoma syndrome
dc.title.none.fl_str_mv Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
dc.type.none.fl_str_mv Text
Journal contribution
info:eu-repo/semantics/publishedVersion
text
contribution to journal
description <h3>Objective</h3><p dir="ltr">This case series reports the clinical presentation and management of multiple <u>odontogenic keratocysts</u> (OKCs) in patients with <u>Gorlin Goltz</u> syndrome and in non-syndromic patients. </p><h3>Material and methods</h3><p dir="ltr">Eight patients presented with features of multiple odontogenic keratocyst at department of Oral and Maxillofacial Surgery. The diagnosis was confirmed following clinical, radiographic and histopathological examination. Initially the diagnosis of OKC was confirmed on an incisional biopsy. Major and minor criteria were followed for the diagnosis of Gorlin Goltz syndrome. Smaller cysts in all patients were enucleated and for larger cysts <u>marsupialization </u>was planned either alone or followed by <u>enucleation</u>. Patients were followed at 1 month, 3 months, 6 months and yearly interval to check for bone healing and recurrences. </p><h3>Results</h3><p dir="ltr">Association of Gorlin Goltz syndrome was identified in four patients all of whom were males with age range of 12–37 years. Among four patients that had non-syndromic OKCs, three were female and one was male with age range of 09–50 years; two patients had familial non-syndromic multiple OKCs. In syndromic patients, multiple OKCs, recognized manifestations of Gorlin Goltz syndrome were identified with variable frequency: calcification of falx cerebri and <u>chest deformity</u> (100%); <u>macrocephaly</u> with <u>frontal bossing </u>(100%);<u> hypertelorism</u> (75%); b<u>asal cell carcinoma </u>(25%); pectus deformity with flame shaped hands and feet (25%) and <u>syndactyly</u> (50%). Palmar or plantar pits, <u>cleft lip</u> or palate, ovarian<u> fibroma</u> and <u>medulloblastoma</u> was not identified in any patient. In patients with non-syndromic multiple OKCs <u>mandible</u> was more commonly involved than <u>maxilla</u>. </p><h3>Conclusion</h3><p dir="ltr">Patients with multiple OKCs, should be evaluated thoroughly and <u>basal cell</u> carcinomatous lesions should be ruled out. Meticulous follow up is vital as Gorlin Goltz syndrome is associated with<u> malignancies</u> and OKCs may be the first manifestation of this syndrome. Given the fact that OKCs associated with this syndrome have higher rate of recurrence than the isolated OKCs, long term follow up is warranted.</p><h2>Other Information</h2><p dir="ltr">Published in: Oral and Maxillofacial Surgery Cases<br>License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br>See article on publisher's website: <a href="https://dx.doi.org/10.1016/j.omsc.2022.100253" target="_blank">https://dx.doi.org/10.1016/j.omsc.2022.100253</a></p>
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network_acronym_str Manara2
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oai_identifier_str oai:figshare.com:article/29045423
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spelling Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocystsOmer Sefvan Janjua (21347903)Ramish Tariq (21347906)Muhammad Usman Khalid (19457347)Sana Mehmood Qureshi (21347909)Kamran Ali (8861576)Biomedical and clinical sciencesClinical sciencesDentistryPaediatricsGorlin Goltz syndromeOdontogenicKeratocystNevoid basal cell carcinoma syndrome<h3>Objective</h3><p dir="ltr">This case series reports the clinical presentation and management of multiple <u>odontogenic keratocysts</u> (OKCs) in patients with <u>Gorlin Goltz</u> syndrome and in non-syndromic patients. </p><h3>Material and methods</h3><p dir="ltr">Eight patients presented with features of multiple odontogenic keratocyst at department of Oral and Maxillofacial Surgery. The diagnosis was confirmed following clinical, radiographic and histopathological examination. Initially the diagnosis of OKC was confirmed on an incisional biopsy. Major and minor criteria were followed for the diagnosis of Gorlin Goltz syndrome. Smaller cysts in all patients were enucleated and for larger cysts <u>marsupialization </u>was planned either alone or followed by <u>enucleation</u>. Patients were followed at 1 month, 3 months, 6 months and yearly interval to check for bone healing and recurrences. </p><h3>Results</h3><p dir="ltr">Association of Gorlin Goltz syndrome was identified in four patients all of whom were males with age range of 12–37 years. Among four patients that had non-syndromic OKCs, three were female and one was male with age range of 09–50 years; two patients had familial non-syndromic multiple OKCs. In syndromic patients, multiple OKCs, recognized manifestations of Gorlin Goltz syndrome were identified with variable frequency: calcification of falx cerebri and <u>chest deformity</u> (100%); <u>macrocephaly</u> with <u>frontal bossing </u>(100%);<u> hypertelorism</u> (75%); b<u>asal cell carcinoma </u>(25%); pectus deformity with flame shaped hands and feet (25%) and <u>syndactyly</u> (50%). Palmar or plantar pits, <u>cleft lip</u> or palate, ovarian<u> fibroma</u> and <u>medulloblastoma</u> was not identified in any patient. In patients with non-syndromic multiple OKCs <u>mandible</u> was more commonly involved than <u>maxilla</u>. </p><h3>Conclusion</h3><p dir="ltr">Patients with multiple OKCs, should be evaluated thoroughly and <u>basal cell</u> carcinomatous lesions should be ruled out. Meticulous follow up is vital as Gorlin Goltz syndrome is associated with<u> malignancies</u> and OKCs may be the first manifestation of this syndrome. Given the fact that OKCs associated with this syndrome have higher rate of recurrence than the isolated OKCs, long term follow up is warranted.</p><h2>Other Information</h2><p dir="ltr">Published in: Oral and Maxillofacial Surgery Cases<br>License: <a href="http://creativecommons.org/licenses/by/4.0/" target="_blank">http://creativecommons.org/licenses/by/4.0/</a><br>See article on publisher's website: <a href="https://dx.doi.org/10.1016/j.omsc.2022.100253" target="_blank">https://dx.doi.org/10.1016/j.omsc.2022.100253</a></p>2022-03-31T09:00:00ZTextJournal contributioninfo:eu-repo/semantics/publishedVersiontextcontribution to journal10.1016/j.omsc.2022.100253https://figshare.com/articles/journal_contribution/Presentation_and_management_of_syndromic_and_non-syndromic_patients_with_multiple_odontogenic_keratocysts/29045423CC BY 4.0info:eu-repo/semantics/openAccessoai:figshare.com:article/290454232022-03-31T09:00:00Z
spellingShingle Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
Omer Sefvan Janjua (21347903)
Biomedical and clinical sciences
Clinical sciences
Dentistry
Paediatrics
Gorlin Goltz syndrome
Odontogenic
Keratocyst
Nevoid basal cell carcinoma syndrome
status_str publishedVersion
title Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
title_full Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
title_fullStr Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
title_full_unstemmed Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
title_short Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
title_sort Presentation and management of syndromic and non-syndromic patients with multiple odontogenic keratocysts
topic Biomedical and clinical sciences
Clinical sciences
Dentistry
Paediatrics
Gorlin Goltz syndrome
Odontogenic
Keratocyst
Nevoid basal cell carcinoma syndrome