Mandibular Carcinoma Cuniculatum around the Dental Implant in a Patient with Concurrent Management for Pemphigus Vulgaris: A Case Report

Background: Carcinoma cuniculatum (CC) is a rare subtype of squamous cell carcinoma that is difficult to diagnose owing to the lack of cellar atypia and/or associated oral epithelial dysplasia. The prognosis is good given proper resection, but it often has a poor prognosis with recurrence. We presen...

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Main Authors: Chika Murai (Author), Ken-ichiro Sakata (Author), Chisato Ouchi (Author), Masanao Sonobe (Author), Kazuhito Yoshikawa (Author), Jun Sato (Author), Akira Satoh (Author), Aya Matsuda (Author), Yoshimasa Kitagawa (Author)
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Published: MDPI AG, 2022-02-01T00:00:00Z.
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LEADER 00000 am a22000003u 4500
001 doaj_7842d5b65ae3419e8e6c1d166b42a1b1
042 |a dc 
100 1 0 |a Chika Murai  |e author 
700 1 0 |a Ken-ichiro Sakata  |e author 
700 1 0 |a Chisato Ouchi  |e author 
700 1 0 |a Masanao Sonobe  |e author 
700 1 0 |a Kazuhito Yoshikawa  |e author 
700 1 0 |a Jun Sato  |e author 
700 1 0 |a Akira Satoh  |e author 
700 1 0 |a Aya Matsuda  |e author 
700 1 0 |a Yoshimasa Kitagawa  |e author 
245 0 0 |a Mandibular Carcinoma Cuniculatum around the Dental Implant in a Patient with Concurrent Management for Pemphigus Vulgaris: A Case Report 
260 |b MDPI AG,   |c 2022-02-01T00:00:00Z. 
500 |a 10.3390/oral2010007 
500 |a 2673-6373 
520 |a Background: Carcinoma cuniculatum (CC) is a rare subtype of squamous cell carcinoma that is difficult to diagnose owing to the lack of cellar atypia and/or associated oral epithelial dysplasia. The prognosis is good given proper resection, but it often has a poor prognosis with recurrence. We present the case of a 78-year-old man who visited our department with an ulcer around the implant in tooth 35. With Nikolsky's phenomenon in the gingiva, a detailed examination revealed pemphigus vulgaris. Steroid administration remarkably improved the oral symptoms but caused osteomyelitis and rapid bone destruction, leading to pathological fracture. After multiple biopsies, mandibular segment resection was performed with a diagnosis of mandibular osteomyelitis, and no malignant findings were found. Four months later, the wound reopened, a white keratinized lesion appeared, and a biopsy revealed CC. Unresectable tumor infiltration was already observed, but the patient died of aspiration pneumonia 15 months after diagnosis. Conclusions: It took 20 months to make a definitive diagnosis of CC in this case. Pemphigus vulgaris may have made the diagnosis particularly difficult. Since other CCs are often diagnosed with osteomyelitis or odontogenic keratocyst preoperatively, we recommend keeping in mind the possibility of CC in refractory cases. 
546 |a EN 
690 |a carcinoma cuniculatum (CC) 
690 |a pemphigus vulgaris (PV) 
690 |a refractory mandibular osteomyelitis 
690 |a multiple biopsies 
690 |a Dentistry 
690 |a RK1-715 
655 7 |a article  |2 local 
786 0 |n Oral, Vol 2, Iss 1, Pp 49-61 (2022) 
787 0 |n https://www.mdpi.com/2673-6373/2/1/7 
787 0 |n https://doaj.org/toc/2673-6373 
856 4 1 |u https://doaj.org/article/7842d5b65ae3419e8e6c1d166b42a1b1  |z Connect to this object online.