Le Fort I osteotomy combined with horseshoe osteotomy has been established as a technique for total maxillary setback. In this study, the stability of the maxillary and mandibular positions was examined in five cases in which was performed total maxillary setback by Le Fort I osteotomy combined with horseshoe osteotomy.
Subjects and methods
The stability of the maxilla was examined in five cases that had undergone total maxillary setback by horseshoe Le Fort I (H- LF I) osteotomy and bilateral sagittal split osteotomy (BSSO) including mandibular anterior subapical osteotomy (MASO) in one case. The changes in the position of the maxillary and mandibular positions were analyzed with lateral cephalograms taken immediately before (T0), a few days after (T1), and one year after (T2) the surgery.
Results
Postoperative maxillary changes both horizontally and vertically were all less than 2 mm. Postoperative horizontal mandibular changes showed little skeletal relapse, but all postoperative vertical mandibular changes were within 2 mm.
Conclusion
Our results suggest that H-LF I osteotomy is a reliable, safe, and effective surgical procedure for postoperative skeletal stability of total maxillary setback in skeletal class II cases.
目的 Le Fort I截骨术联合马蹄形截骨术已被确立为上颌骨全退位的一种技术。研究对象和方法研究了五例通过马蹄形 Le Fort I（H- LF I）截骨术和双侧矢状劈开截骨术（BSSO）（包括一例下颌骨前方根尖下截骨术（MASO））进行全上颌骨后移的病例的上颌骨和下颌骨位置的稳定性。术前（T0）、术后几天（T1）和术后一年（T2）拍摄的侧位头影分析了上颌骨和下颌骨位置的变化。结论我们的研究结果表明，H-LF I 截骨术是一种可靠、安全、有效的手术方法，可用于骨骼Ⅱ级病例上颌骨全后移的术后骨骼稳定。
{"title":"Stability after maxillary setback by horseshoe Le Fort I osteotomy in skeletal class II cases","authors":"Myo Maung Maung, Daichi Hasebe, Masaki Kasahara, Zhuoyang Zheng, Ryoko Takeuchi, Hidenobu Sakuma, Daisuke Saito, Tadaharu Kobayashi","doi":"10.1016/j.ajoms.2024.05.002","DOIUrl":"10.1016/j.ajoms.2024.05.002","url":null,"abstract":"<div><h3>Purpose</h3><div>Le Fort I osteotomy combined with horseshoe osteotomy has been established as a technique for total maxillary setback. In this study, the stability of the maxillary and mandibular positions was examined in five cases in which was performed total maxillary setback by Le Fort I osteotomy combined with horseshoe osteotomy.</div></div><div><h3>Subjects and methods</h3><div>The stability of the maxilla was examined in five cases that had undergone total maxillary setback by horseshoe Le Fort I (H- LF I) osteotomy and bilateral sagittal split osteotomy (BSSO) including mandibular anterior subapical osteotomy (MASO) in one case. The changes in the position of the maxillary and mandibular positions were analyzed with lateral cephalograms taken immediately before (T0), a few days after (T1), and one year after (T2) the surgery.</div></div><div><h3>Results</h3><div>Postoperative maxillary changes both horizontally and vertically were all less than 2 mm. Postoperative horizontal mandibular changes showed little skeletal relapse, but all postoperative vertical mandibular changes were within 2 mm.</div></div><div><h3>Conclusion</h3><div>Our results suggest that H-LF I osteotomy is a reliable, safe, and effective surgical procedure for postoperative skeletal stability of total maxillary setback in skeletal class II cases.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 36-41"},"PeriodicalIF":0.4,"publicationDate":"2024-05-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141045740","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Central Giant Cell Granuloma (CGCG) in jaw bones have characteristic diagnostic features. But rarely they may present with slightly different features or features related to other lesions. It can be challenging when histopathological features of two or more different entities are seen within a single lesion.
Case report
We report a jaw lesion in a 47-year-old male patient who presented unusually as a painful diffuse left mandibular swelling. Although the 2-dimensional imaging study suggested a cystic lesion, the 3-dimensional imaging study pointed to a more well-demarcated pathology. A complete excision of the lesion when examined in detail confirmed a histopathologic diagnosis of a CGCG. The challenge however was the large reactive bone areas that raised a suspicion of an Ossifying Fibroma lesion.
Conclusion
Diagnosing a jaw lesion consisting of areas showing two or more different features is always challenging. This report highlights the key histopathologic findings that can help distinguish between a hybrid lesion and a case of CGCG with large reactive bone areas.
{"title":"An unusual case of Central Giant Cell Granuloma","authors":"Yap Boon How , Ajay Telang , Lahari Telang , Wanninayake Mudiyanselage Tilakaratne","doi":"10.1016/j.ajoms.2024.04.018","DOIUrl":"10.1016/j.ajoms.2024.04.018","url":null,"abstract":"<div><h3>Introduction</h3><div>Central Giant Cell Granuloma (CGCG) in jaw bones have characteristic diagnostic features. But rarely they may present with slightly different features or features related to other lesions. It can be challenging when histopathological features of two or more different entities are seen within a single lesion.</div></div><div><h3>Case report</h3><div>We report a jaw lesion in a 47-year-old male patient who presented unusually as a painful diffuse left mandibular swelling. Although the 2-dimensional imaging study suggested a cystic lesion, the 3-dimensional imaging study pointed to a more well-demarcated pathology. A complete excision of the lesion when examined in detail confirmed a histopathologic diagnosis of a CGCG. The challenge however was the large reactive bone areas that raised a suspicion of an Ossifying Fibroma lesion.</div></div><div><h3>Conclusion</h3><div>Diagnosing a jaw lesion consisting of areas showing two or more different features is always challenging. This report highlights the key histopathologic findings that can help distinguish between a hybrid lesion and a case of CGCG with large reactive bone areas.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 239-242"},"PeriodicalIF":0.4,"publicationDate":"2024-05-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141053381","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-05-04DOI: 10.1016/j.ajoms.2024.05.001
Shinichi Sato, Masato Takahashi
Supernumerary teeth are commonly observed in the anterior region of the maxilla; however, reports of their occurrence in the molar region, particularly near the third molars, are rare. A 21-year-old man with no genetic disease visited our department for a third-molar extraction. Panoramic radiography and computed tomography revealed one and four supernumerary teeth in the right and left maxillary third-molar regions, respectively. The bilateral third molars and supernumerary teeth were extracted under general anesthesia. The supernumerary teeth were incompletely developed, and one tooth morphologically resembled a premolar. The patient’s postoperative course was uneventful.
{"title":"Five impacted supernumerary teeth in the bilateral maxillary third-molar regions: A case report","authors":"Shinichi Sato, Masato Takahashi","doi":"10.1016/j.ajoms.2024.05.001","DOIUrl":"10.1016/j.ajoms.2024.05.001","url":null,"abstract":"<div><div>Supernumerary teeth are commonly observed in the anterior region of the maxilla; however, reports of their occurrence in the molar region, particularly near the third molars, are rare. A 21-year-old man with no genetic disease visited our department for a third-molar extraction. Panoramic radiography and computed tomography revealed one and four supernumerary teeth in the right and left maxillary third-molar regions, respectively. The bilateral third molars and supernumerary teeth were extracted under general anesthesia. The supernumerary teeth were incompletely developed, and one tooth morphologically resembled a premolar. The patient’s postoperative course was uneventful.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 112-115"},"PeriodicalIF":0.4,"publicationDate":"2024-05-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141034839","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ameloblastic carcinoma (AC) is an aggressive odontogenic tumor. Spindle cell variant of AC (SCAC) is a rare entity and only sixteen cases of SCACs have been reported in the literature till date. We reported a case of 42-years-old male presented with swelling in right lower posterior region for 3 years. Radiology revealed multilocular osteolytic with cortical thinning and rarefaction showing internal non-enhancing areas. Histologically, the tumor revealed areas characteristic of ameloblastoma along with hypercellular areas of monomorphic spindle shaped cells merging into the stroma displaying cytological atypia, suggestive of AC with spindle cell variant. The purpose of this case report is to document an unusual case of spindle cell variant of AC, which can mimic other oral spindle cell lesions. Additionally, proliferative marker along with histological parameters like cytological atypia, mitoses and cell morphology is emphasized.
绒毛细胞癌（AC）是一种侵袭性牙源性肿瘤。纺锤形细胞变异型牙髓母细胞癌（SCAC）是一种罕见的肿瘤，迄今为止，文献中仅报道了16例SCAC。我们报告了一例 42 岁男性病例，患者右下后部肿胀 3 年。放射学检查发现肿瘤为多发性溶骨性，皮质变薄、稀疏，内部为非增强区。组织学上，肿瘤显示出釉母细胞瘤的特征区域，以及单形纺锤形细胞与基质融合的高细胞区，显示出细胞学不典型性，提示为具有纺锤细胞变异的 AC。本病例报告的目的是记录一例不寻常的 AC 纺锤形细胞变异病例，该病例可模拟其他口腔纺锤形细胞病变。此外，报告还强调了增殖标志物以及细胞学不典型性、有丝分裂和细胞形态等组织学参数。
{"title":"Spindle cell variant of ameloblastic carcinoma of mandible","authors":"Kiran Jot , Rahul Yadav , Aanchal Kakkar , Deepika Mishra","doi":"10.1016/j.ajoms.2024.04.017","DOIUrl":"10.1016/j.ajoms.2024.04.017","url":null,"abstract":"<div><div>Ameloblastic carcinoma (AC) is an aggressive odontogenic tumor. Spindle cell variant of AC (SCAC) is a rare entity and only sixteen cases of SCACs have been reported in the literature till date. We reported a case of 42-years-old male presented with swelling in right lower posterior region for 3 years. Radiology revealed multilocular osteolytic with cortical thinning and rarefaction showing internal non-enhancing areas. Histologically, the tumor revealed areas characteristic of ameloblastoma along with hypercellular areas of monomorphic spindle shaped cells merging into the stroma displaying cytological atypia, suggestive of AC with spindle cell variant. The purpose of this case report is to document an unusual case of spindle cell variant of AC, which can mimic other oral spindle cell lesions. Additionally, proliferative marker along with histological parameters like cytological atypia, mitoses and cell morphology is emphasized.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 243-247"},"PeriodicalIF":0.4,"publicationDate":"2024-05-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141043782","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Carcinoma ex pleomorphic adenoma (CXPA) is a tumor arising from a pleomorphic adenoma and classified as an independent type in the 2005 WHO classification. These tumors mainly occur in the parotid gland or palate, though rarely in the buccal region. The carcinoma component is most often a salivary duct carcinoma, while findings of a squamous cell carcinoma (SCC) are rare. Here, we report a case of carcinoma of pleomorphic adenoma origin with SCC as the main component occurring in the buccal region. A 77-year-old woman was referred to our department because of swelling in the right buccal mucosa. An elastic hard mass was found in the right buccal subcutaneous region and induration in the right buccal mucosa due to the mass was noted by palpation. Biopsy results from an obtained specimen led to a diagnosis of SCC. The patient underwent excision of a right-sided buccal malignant tumor in combination with skin resection and partial right-sided maxillary resection. Histopathological examination findings of the excised tumor indicated CXPA primarily composed of SCC with lymph node metastasis. Thereafter, multiple sites of distant metastasis were found. The patient died at two years six months after surgery.
{"title":"Carcinoma ex pleomorphic adenoma primarily composed of squamous cell carcinoma in buccal region – Case report","authors":"Takako Naruse , Kouji Ohta , Hideo Shigeishi , Toshinori Ando , Takayuki Nakagawa , Nao Yamakado , Yoshiaki Ninomiya , Shinichi Sakamoto , Yoko Ishida , Tomonao Aikawa","doi":"10.1016/j.ajoms.2024.04.014","DOIUrl":"10.1016/j.ajoms.2024.04.014","url":null,"abstract":"<div><div>Carcinoma ex pleomorphic adenoma (CXPA) is a tumor arising from a pleomorphic adenoma and classified as an independent type in the 2005 WHO classification. These tumors mainly occur in the parotid gland or palate, though rarely in the buccal region. The carcinoma component is most often a salivary duct carcinoma, while findings of a squamous cell carcinoma (SCC) are rare. Here, we report a case of carcinoma of pleomorphic adenoma origin with SCC as the main component occurring in the buccal region. A 77-year-old woman was referred to our department because of swelling in the right buccal mucosa. An elastic hard mass was found in the right buccal subcutaneous region and induration in the right buccal mucosa due to the mass was noted by palpation. Biopsy results from an obtained specimen led to a diagnosis of SCC. The patient underwent excision of a right-sided buccal malignant tumor in combination with skin resection and partial right-sided maxillary resection. Histopathological examination findings of the excised tumor indicated CXPA primarily composed of SCC with lymph node metastasis. Thereafter, multiple sites of distant metastasis were found. The patient died at two years six months after surgery.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 99-103"},"PeriodicalIF":0.4,"publicationDate":"2024-05-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141046580","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-05-02DOI: 10.1016/j.ajoms.2024.04.016
Hisanobu Kamio , Kana Oue , Yasuyuki Asada , Nanako Ito , Serika Imamura , Mitsuru Doi , Yoshitaka Shimizu , Mitsuhiro Yoshida , Souichi Yanamoto , Hiroshi Hanamoto
Noonan syndrome (NS) is characterized by various abnormalities and is complicated with cardiac diseases, such as hypertrophic cardiomyopathy, in > 80% of cases. Minimum hemodynamic changes are a crucial factor during general anesthesia in such patients. We report the case of a patient with NS and hypertrophic obstructive cardiomyopathy (HOCM) who underwent general anesthesia using remimazolam, a new ultrashort-acting benzodiazepine anesthetic, which is expected to cause less circulatory depression. A 19-year-old woman with NS complicated with HOCM was scheduled to undergo extraction of the wisdom teeth and second molar under general anesthesia. Preoperative evaluation revealed HOCM with left ventricular outflow tract obstruction and the patient had chronic heart failure. After the placement of an arterial line under moderate sedation with remimazolam, general anesthesia was induced with remimazolam, fentanyl, and rocuronium, followed by anesthesia maintenance with remimazolam and remifentanil. Tracheal intubation was performed using videolaryngoscopy. Local anesthesia and inferior alveolar nerve block were performed using adrenaline-free local anesthetics. Intraoperatively, low-dose phenylephrine was administered continuously to maintain peripheral vascular resistance. At the end of surgery, the endotracheal tube was replaced with an i-gel® supraglottic airway device before emergence from general anesthesia. After full recovery from anesthesia, the i-gel® was removed, and the patient was transferred to the hospital ward. The perioperative blood pressure and heart rate were maintained within normal ranges, and no cardiovascular events occurred during anesthesia. Anesthesia management using remimazolam and low-dose phenylephrine with reduction in perioperative stress may provide an appropriate circulatory condition for noncardiac surgery in patients with HOCM.
{"title":"General anesthesia with remimazolam for tooth extraction in a patient with Noonan syndrome and hypertrophic obstructive cardiomyopathy: A case report","authors":"Hisanobu Kamio , Kana Oue , Yasuyuki Asada , Nanako Ito , Serika Imamura , Mitsuru Doi , Yoshitaka Shimizu , Mitsuhiro Yoshida , Souichi Yanamoto , Hiroshi Hanamoto","doi":"10.1016/j.ajoms.2024.04.016","DOIUrl":"10.1016/j.ajoms.2024.04.016","url":null,"abstract":"<div><div>Noonan syndrome (NS) is characterized by various abnormalities and is complicated with cardiac diseases, such as hypertrophic cardiomyopathy, in > 80% of cases. Minimum hemodynamic changes are a crucial factor during general anesthesia in such patients. We report the case of a patient with NS and hypertrophic obstructive cardiomyopathy (HOCM) who underwent general anesthesia using remimazolam, a new ultrashort-acting benzodiazepine anesthetic, which is expected to cause less circulatory depression. A 19-year-old woman with NS complicated with HOCM was scheduled to undergo extraction of the wisdom teeth and second molar under general anesthesia. Preoperative evaluation revealed HOCM with left ventricular outflow tract obstruction and the patient had chronic heart failure. After the placement of an arterial line under moderate sedation with remimazolam, general anesthesia was induced with remimazolam, fentanyl, and rocuronium, followed by anesthesia maintenance with remimazolam and remifentanil. Tracheal intubation was performed using videolaryngoscopy. Local anesthesia and inferior alveolar nerve block were performed using adrenaline-free local anesthetics. Intraoperatively, low-dose phenylephrine was administered continuously to maintain peripheral vascular resistance. At the end of surgery, the endotracheal tube was replaced with an i-gel® supraglottic airway device before emergence from general anesthesia. After full recovery from anesthesia, the i-gel® was removed, and the patient was transferred to the hospital ward. The perioperative blood pressure and heart rate were maintained within normal ranges, and no cardiovascular events occurred during anesthesia. Anesthesia management using remimazolam and low-dose phenylephrine with reduction in perioperative stress may provide an appropriate circulatory condition for noncardiac surgery in patients with HOCM.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 108-111"},"PeriodicalIF":0.4,"publicationDate":"2024-05-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141032474","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Infants tend to place anything in their mouths; however, foreign bodies embedded in the hard palate of infants are extremely rare in clinical practice. Infants are often difficult to examine, and foreign bodies in the hard palate are misdiagnosed as neoplastic or inflammatory lesions in some cases. We present a rare case of a foreign body that was not diagnosed until its removal from the hard palate of an infant under general anesthesia. A healthy 12-month-old girl with a protruding foreign body on the hard palate was referred for close investigation. A white-yellow protruding foreign body measuring approximately 10 mm was found on the palate. Computed tomography revealed a radiopaque foreign body at the border. It was decided to examine and performed biopsy under general anesthesia, and the foreign body removed easily was an obviously plastic object with a semicircular shape and a brim around it. The histopathological examination of the abnormal gingiva under the foreign body revealed inflammatory granulation tissue. This report highlights the difficulty in diagnosing an infant with a foreign body in the hard palate and the safety of its removal under general anesthesia. Clinicians must consider the possibility of foreign bodies causing lesions on an infant’s hard palate. Therefore, careful examination and safe removal are necessary to prevent serious aspiration complications.
{"title":"Foreign body in infant hard palate: A report of a rare case and literature review","authors":"Shohei Takaoka , Kenji Yamagata , Satoshi Fukuzawa , Fumihiko Uchida , Naomi Ishibashi-Kanno , Hiroki Bukawa","doi":"10.1016/j.ajoms.2024.04.015","DOIUrl":"10.1016/j.ajoms.2024.04.015","url":null,"abstract":"<div><div>Infants tend to place anything in their mouths; however, foreign bodies embedded in the hard palate of infants are extremely rare in clinical practice. Infants are often difficult to examine, and foreign bodies in the hard palate are misdiagnosed as neoplastic or inflammatory lesions in some cases. We present a rare case of a foreign body that was not diagnosed until its removal from the hard palate of an infant under general anesthesia. A healthy 12-month-old girl with a protruding foreign body on the hard palate was referred for close investigation. A white-yellow protruding foreign body measuring approximately 10 mm was found on the palate. Computed tomography revealed a radiopaque foreign body at the border. It was decided to examine and performed biopsy under general anesthesia, and the foreign body removed easily was an obviously plastic object with a semicircular shape and a brim around it. The histopathological examination of the abnormal gingiva under the foreign body revealed inflammatory granulation tissue. This report highlights the difficulty in diagnosing an infant with a foreign body in the hard palate and the safety of its removal under general anesthesia. Clinicians must consider the possibility of foreign bodies causing lesions on an infant’s hard palate. Therefore, careful examination and safe removal are necessary to prevent serious aspiration complications.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 104-107"},"PeriodicalIF":0.4,"publicationDate":"2024-05-02","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"141051016","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-04-27DOI: 10.1016/j.ajoms.2024.04.013
Seiichiro Oda , Hirofumi Okada , Atsushi Kihara , Norito Ishii , Hiroshi Koga , Yoshiyuki Mori , Tadahide Noguchi
Mucous membrane pemphigoid (MMP) is an autoimmune subepithelial/subepidermal blistering disease characterized by linear deposition of immunoglobulin G (IgG), IgA, and/or C3 in the basement membrane zone (BMZ) and predominant mucosal lesions involving the oral cavity and conjunctivae. Here, we describe a case of a 58-year-old Japanese man with MMP. Intraoral examination revealed desquamative gingivitis in the upper and lower gingivae. No conjunctival or cutaneous lesions were observed. Histopathological examination revealed a subepithelial split with inflammatory cell infiltration of the epithelial and connective tissues. Direct immunofluorescence revealed a linear deposition of C3 in the BMZ. IgG autoantibodies against BP230 were detected using an enzyme-linked immunosorbent assay. Indirect immunofluorescence using 1 mol/L NaCl-split skin sections revealed no reactivity for serum IgG or IgA. Immunoblotting using normal human epidermal extract as a substrate did not detect IgG reactivity against the 230-kDa antigen. Based on the above results, the definitive diagnosis of MMP with anti-BP230 antibody alone was established. Our case was categorized as low-risk MMP. The patient was treated with 0.1% triamcinolone acetonide ointment, oral minocycline, and oral nicotinamide, which improved the oral erosions. However, the mechanism underlying IgG autoantibody production against intracellular BP230 remains unclear. Further research and more such case studies are necessary to elucidate the mechanism of anti-BP230 antibody production and the clinical feature of anti-BP230 type MMP.
{"title":"Anti-BP230 type mucous membrane pemphigoid with desquamative gingivitis: A case report","authors":"Seiichiro Oda , Hirofumi Okada , Atsushi Kihara , Norito Ishii , Hiroshi Koga , Yoshiyuki Mori , Tadahide Noguchi","doi":"10.1016/j.ajoms.2024.04.013","DOIUrl":"10.1016/j.ajoms.2024.04.013","url":null,"abstract":"<div><div>Mucous membrane pemphigoid (MMP) is an autoimmune subepithelial/subepidermal blistering disease characterized by linear deposition of immunoglobulin G (IgG), IgA, and/or C3 in the basement membrane zone (BMZ) and predominant mucosal lesions involving the oral cavity and conjunctivae. Here, we describe a case of a 58-year-old Japanese man with MMP. Intraoral examination revealed desquamative gingivitis in the upper and lower gingivae. No conjunctival or cutaneous lesions were observed. Histopathological examination revealed a subepithelial split with inflammatory cell infiltration of the epithelial and connective tissues. Direct immunofluorescence revealed a linear deposition of C3 in the BMZ. IgG autoantibodies against BP230 were detected using an enzyme-linked immunosorbent assay. Indirect immunofluorescence using 1 mol/L NaCl-split skin sections revealed no reactivity for serum IgG or IgA. Immunoblotting using normal human epidermal extract as a substrate did not detect IgG reactivity against the 230-kDa antigen. Based on the above results, the definitive diagnosis of MMP with anti-BP230 antibody alone was established. Our case was categorized as low-risk MMP. The patient was treated with 0.1% triamcinolone acetonide ointment, oral minocycline, and oral nicotinamide, which improved the oral erosions. However, the mechanism underlying IgG autoantibody production against intracellular BP230 remains unclear. Further research and more such case studies are necessary to elucidate the mechanism of anti-BP230 antibody production and the clinical feature of anti-BP230 type MMP.</div></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"37 1","pages":"Pages 210-214"},"PeriodicalIF":0.4,"publicationDate":"2024-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142660547","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2024-04-24DOI: 10.1016/j.ajoms.2024.04.011
Farzad Esmaeili , Narges Bayat , Amir Ebrahimpour Tolouei , Mohammad Azimzadeh , Mehrdad Nateghi , Kasra Rahimipour
This study aimed to investigate the impact of COVID-19 on the trabecular bone thickness in the mandible using fractal analysis. The researchers selected 82 healthy patients who had previously been infected with COVID-19 and had dental panoramic radiographs (DPRs) prior to contracting the virus. DPRs were obtained again after infection, and the trabecular thickness was analyzed before and after COVID-19 using statistical tests. The results revealed that the average trabecular thickness of the mandible at the angle and condyle regions decreased significantly after contracting COVID-19. The mean thickness before infection was 1.479 ± 0.128 at the angle point and 1.469 ± 0.124 at the condylar region, while after infection, it decreased to 1.200 ± 0.123 and 1.159 ± 0.114, respectively. The reduction in trabecular thickness was found to be statistically significant. This study underscores the importance of monitoring bone density in COVID-19 patients. The findings suggest that COVID-19 can have extrapulmonary effects on skeletal health, specifically the mandibular bone. The results highlight the need for preventive interventions to mitigate pathological bone loss in individuals affected by COVID-19. Overall, this research emphasizes the significance of considering skeletal manifestations and implementing appropriate measures for COVID-19 patients to safeguard their bone health.
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Pub Date : 2024-04-23DOI: 10.1016/j.ajoms.2024.04.012
Maroteaux-Lamy syndrome (mucopolysaccharidoses VI) is a rare genetic disease results in functional deficiency of arylsulfatase B and accumulation of dermatan sulfate within lysosomes in various tissues. Here we present an 8-year-old male who was referred for treatment of limited mouth opening. The patient had been diagnosed with mucopolysaccharidoses VI at age of 6 months. He had received bone marrow transplantation at age of 3.5 years, developed graft-versus-host disease and subsequent vitiligo-like changes. The patient presented with short stature, and some skeletal anomalies. In radiographic images bilateral hyperplasia of the coronoid process, underdeveloped condyles, and dentigerous cyst-like follicles in the jaws were noted. Bilateral coronoidectomy was performed to improve the maximum opening of the mouth. Post-op follow-up examinations in 5 years showed moderate improvement. Abnormal coronoid and condylar processes are unusual in MPS VI. Proper management is needed to improve patients’ oral and general health for the remainder of their lives.
Maroteaux-Lamy 综合征（粘多糖病 VI）是一种罕见的遗传性疾病，会导致芳基硫酸酯酶 B 功能性缺乏，并在各种组织的溶酶体中积累硫酸皮质酯。我们在此介绍一名因张口受限而转诊治疗的 8 岁男性患者。患者在 6 个月大时被诊断出患有粘多糖病 VI。他在 3.5 岁时接受了骨髓移植，后来患上了移植物抗宿主病，并出现了白癜风样改变。患者表现为身材矮小和一些骨骼异常。放射影像显示，患者双侧冠状突增生，髁状突发育不全，下颌有齿状囊肿样滤泡。为了改善口腔的最大张开度，医生对他进行了双侧冠状突切除术。术后5年的随访检查显示病情有适度改善。冠状突和髁突异常在 MPS VI 中并不常见。需要进行适当的治疗，以改善患者余生的口腔和全身健康状况。
{"title":"Maroteaux-lamy syndrome (mucopolysaccharidosis VI) with abnormal coronoid and condylar processes of the mandible: Report of a case with surgical intervention","authors":"","doi":"10.1016/j.ajoms.2024.04.012","DOIUrl":"10.1016/j.ajoms.2024.04.012","url":null,"abstract":"<div><p>Maroteaux-Lamy syndrome (mucopolysaccharidoses VI) is a rare genetic disease results in functional deficiency of arylsulfatase B<span><span> and accumulation of dermatan sulfate<span><span> within lysosomes in various tissues. Here we present an 8-year-old male who was referred for treatment of limited mouth opening. The patient had been diagnosed with </span>mucopolysaccharidoses<span> VI at age of 6 months. He had received bone marrow transplantation at age of 3.5 years, developed graft-versus-host disease and subsequent vitiligo-like changes. The patient presented with short stature, and some </span></span></span>skeletal anomalies<span><span>. In radiographic images bilateral hyperplasia of the coronoid process, underdeveloped condyles, and dentigerous cyst-like follicles in the jaws were noted. Bilateral coronoidectomy was performed to improve the maximum opening of the mouth. Post-op follow-up examinations in 5 years showed moderate improvement. Abnormal coronoid and </span>condylar processes are unusual in MPS VI. Proper management is needed to improve patients’ oral and general health for the remainder of their lives.</span></span></p></div>","PeriodicalId":45034,"journal":{"name":"Journal of Oral and Maxillofacial Surgery Medicine and Pathology","volume":"36 6","pages":"Pages 872-877"},"PeriodicalIF":0.4,"publicationDate":"2024-04-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"140767252","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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