The Journal of Craniofacial Surgery
Volume 26, Number 6, September 2015
and there was no abnormality in intraoral examination. The patient had no history of dentomaxillofacial trauma, craniofacial anomalies, syndromes, or infection. A computerized tomography (CT) scan was performed, which revealed a supernumerary intranasal tooth in the floor of left nasal cavity, 2 cm posterior to the anterior end of the inferior turbinate, with the same attenuation as that of the oral teeth (Fig. 1). The intranasal tooth was removed by forceps with endoscopic guidance under general anesthesia. The postoperative course was uneventful, except for a few days of serious discharge, which diminished. Ectopic teeth is a rare disease, which may be supernumerary or may result from abnormal dentition.1 The supernumerary intranasal teeth may be observed at various ages, ranging from 6 to 61 years. It is reported to be more common in male patients (62%) and in the left side.2 The etiology of intranasal tooth eruption has not been clarified. Abnormal tissue interactions during development may potentially lead to ectopic tooth development and eruption.3 A total of 84.6% of patients are symptomathic.3 Nasal teeth can cause nasal discharge, foul smell, nasal obstruction, nasal discomfort, headache, facial pain, epistaxis, epiphora, sinusitis, rhinitis, nasal septal deviations, nasal septal abscess, and oronasal fistula.1,2 Sometimes an intranasal tooth may be asymptomatic as in the current case. The diagnosis of an intranasal tooth can be confirmed clinically and radiologically. An intranasal tooth is often a hard white mass and can sometimes be covered completely by nasal mucosa and surrounded by granulation tissue and necrotic debris.2 Radiologic examinations, especially CT, are useful to identify the exact position and helps to decide the surgical approach.1 The differential diagnosis of intranasal white mass includes nasal foreign body, rhinolith, exocytosis, odontomas, osteomas, malignant tumors.1,2 The general opinion on the treatment of nasal teeth is the extraction when diagnosed because of potential morbidity, even if, asymptomatic.1 It may be extracted with either transnasal or transpalatal approach. Endoscopic removal of intranasal teeth can provide good illumination, better visualization, and has less morbidity compared with conventional approach.2 As the diagnosis of the disease is easy with a simple endoscopic examination, all of the patients referred to an otorhinolaryngology clinic should be completely examined. The supernumerary intranasal teeth are rare cases. Early extraction is advocated because of potential morbidity.
Correspondence
REFERENCES 1. Krishnan B, Parida PK, Gopalakrishnan S, et al. An unusual cause of epistaxis in a young patient: the supernumerary nasal tooth. Oral Maxillofac Surg 2013;17:315–317 2. Iwai T, Aoki N, Yamashita Y, et al. Endoscopic removal of bilateral supernumerary intranasal teeth. J Oral Maxillofac Surg 2012;70: 1030–1034 3. Mohan S, Kankariya H, Harjani H. Ectopic third molar in the maxillary sinus. Natl J Maxillofac Surg 2011;2:222–224
Clinicopathologic Assessment of Myositis Ossificans Circumscripta of the Masseter Muscles To the Editor: Myositis ossificans (MO) is a heterotopic bone formation within the muscle, and can be classified into 3 subtypes: fibrodysplasia ossificans progressiva, neurogenic MO, and traumatic MO circumscripta, which is a benign and reactive bone formation within the muscle after trauma. Traumatic MO circumscripta mostly occurs in the thigh and brachium,1 and the occurrence in the masticatory muscles, though rare, usually results in temporomandibular joint dysfunction. Although the exact cause of MO circumscripta remains unknown, it has been presumed that infection, hematoma, tearing of the periosteum and bony metaplasia of the muscle and fibrous connective tissues cause heterotopic ossification.2 A 36-year-old man experiencing severe trismus was referred to our hospital. He was, at the age of 21, under confinement for 1 year and frequently abused about the face. On physical examination, maximal interincisal distance (MID) was 10 mm and hard masses were palpated in the bilateral cheeks (Fig. 1). Computed tomography scan showed calcified masses in the bilateral masseter muscles extending from the zygomatic arch to the ascending ramus of the mandible (Fig. 1). He was diagnosed as MO circumscripta, and as the disease state had not been progressive for the past several years, surgical intervention was selected to alleviate the trismus. We performed bilateral osteotomies of the bony fragments, and bilateral coronoidectomies to eliminate any possibility of further restriction of the jaw function. Following this, gentle manipulation of the jaw resulted in a full range of motion, achieving an MID of 40 mm,
Muhammed Sedat Sakat, MD Korhan Kilic, MD Otorhinolaryngology Clinics Palandoken State Hospital Erzurum, Turkey Mustafa Sitki Gozeler, MD Harun Ucuncu, MD Department of Otorhinolaryngology Atatu¨rk University Erzurum, Turkey [email protected] Ozan Kuduban, MD Department of Otorhinolaryngology Erzurum Region Education and Research Hospital Erzurum, Turkey
#
2015 Mutaz B. Habal, MD
FIGURE 1. The maximal interincisal distance improved from 10 mm (before treatment) to 36 mm (2 months postoperatively). Preoperative 3D-CT images revealed large ossified masses in the masseter region of the left and right sides (50 and 21ı ¨mm in maximum diameter, respectively). Pathologic findings of the left ossified mass (hematoxylin and eosin stain) showed that rigid bony structure constituted a large portion of the specimen, and striate muscles (black arrow) existed between bone trabeculae where bone marrow is normally present. 3D-CT, three-dimensional computed tomography.
2025
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Correspondence
The Journal of Craniofacial Surgery
and the wound was closed in layers. The day after the operation, the patient’s trismus was temporarily aggravated probably because of postoperative edema and pain in the masseter region. After 2 months of physical therapy, the patient, however, presented an MID of 36 mm (Fig. 1). And, as of 1 year after the operation, there was no recurrence of trismus or heterotopic ossification, providing a high level of patient satisfaction and a good functionality. The most common treatment of MO circumscripta is excision of the lesion or osteotomy, though exacerbation or repeated relapses may occur in some patients.3 Two successive phases of the disease are known: an early phase with abundant primary vascular cells and angiogenesis, and an ossification phase with premature ectopic bone formation.1 To prevent recurrence, surgical intervention is recommended after the end of ossification phase. In addition to the removal of a mass, coronoidectomy, or at least coronoidotomy, can be the choice of treatment to eliminate possibility of disturbances in jaw movements.
PATHOLOGIC ASSESSMENT The pathologic examination of the decalcified, hematoxylin–eosin stained specimens revealed that the ossified mass had a dense and rigid bony structure of mature lamellar bone, including Haversian systems and bone trabeculae (Fig. 1). This indicates osteogenesis was once dynamic though later subsided. Moreover, most interestingly, there were striate muscles between the trabeculae, where bone marrow is normally present. This suggests that osteogenesis was induced along existing masseter muscles, presumably by muscle-derived stem cells differentiating into osteoblasts.4 There, however, remains a slight possibility of migration of marrowderived stem cells into the muscle region. Takanobu Mashiko, MD Department of Plastic Surgery Tokyo Metropolitan Police Hospital Nakano, Nakano-Ku Tokyo, Japan [email protected] Tanetaka Akizuki, MD Yorikatsu Watanabe, MD Department of Plastic Surgery Tokyo Metropolitan Police Hospital Nakano-ku, Tokyo, Japan Ryo Sasaki, DDS Department of Plastic Surgery Tokyo Women’s Medical University School of Medicine Shinjuku-ku, Tokyo, Japan
Volume 26, Number 6, September 2015
REFERENCES 1. Godhi SS, Singh A, Kukreja P, et al. Myositis ossificans circumscripta involving bilateral masticatory muscles. J Craniofac Surg 2011;22:e11–e13 2. Molloy JC, McGuirk RA. Treatment of traumatic myositis ossificans circumscripta: use of aspiration and steroids. J Trauma 1976;16:851–857 3. Aoki T, Naito H, Ota Y, et al. Myositis ossificans traumatica of the masticatory muscles: review of the literature and report of a case. J Oral Maxillofac Surg 2002;60:1083–1088 4. Shi X, Garry DJ. Muscle stem cells in development, regeneration, and disease. Genes Dev 2006;20:1692–1708
Basosquamous or Basal Cell Carcinoma? The Importance of the Differential Diagnosis To the Editor: Basosquamous carcinoma (BSC) is a rare and aggressive type of basal cell carcinoma (BCC) more frequently involving head and neck region1 characterized by high tendency to recur locally or at distant sites. Metastasis have been observed with a latency period that can vary from 0 to 30 years.2 In some cases, first diagnosis of the skin lesion is consistent with BCC, and only after metastasis detection diagnosis of BSC has been done.3,4 Immunohistochemistry is useful to diagnose BSC: areas of squamous cell carcinoma are cytokeratin AE1, cytokeratin AE3, and cell adhesion molecule 52 positive and show variable staining with epithelial membrane antigen. Characteristically, the transition zone shows a decline of staining with graded reactivity for Ber-EP4 as the morphology of the tumor moves from BCC to squamous cell carcinoma.1,5 Nevertheless, approximately 80% of the lesions initially diagnosed as BCC were found to have basosquamous histology upon metastasis, as in our case.2 It can support the hypothesis that BSC derives from differentiation of a BCC, or can be explained as an histopathologic misdiagnosis or sample bias because of tumor heterogenity.3,4 A 60-years-old woman presented at our department referring a progressive unpainful swelling of the left submandibular region. Medical history was unremarkable, except for a BCC of the left nose that was treated with surgical resection in free margins approximately 10 years before. Clinical examination revealed no signs of local recurrence, and no suspect lesions were detected after fibrolaringoscopy. Computed tomography scan with contrast revealed a 2 cm colliquative lymphadenopathy at level Ib in the left neck, with the presence of some enlarged nodes at the same side (Fig. 1A). Fine-needle aspiration cytology was consistent with carcinoma. Whole body positron emission tomography with 2-deoxy-2-[fluorine-18]fluoro-D-glucose integrated with computed
Munehiro Yokoyama, MD Department of Pathology Tokyo Metropolitan Police Hospital Nakano, Nakano-Ku, Tokyo, Japan Kotaro Yoshimura, MD Kazuhide Mineda, MD Department of Plastic Surgery University of Tokyo School of Medicine Hongo, Bunkyo-Ku, Tokyo, Japan
2026
FIGURE 1. A, Preoperative axial CT-scan with contrast. B, Preoperative PET-CT (sagittal view). Arrow: pathologic submandibular node.
#
2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Volume 26, Number 6, September 2015
and there was no abnormality in intraoral examination. The patient had no history of dentomaxillofacial trauma, craniofacial anomalies, syndromes, or infection. A computerized tomography (CT) scan was performed, which revealed a supernumerary intranasal tooth in the floor of left nasal cavity, 2 cm posterior to the anterior end of the inferior turbinate, with the same attenuation as that of the oral teeth (Fig. 1). The intranasal tooth was removed by forceps with endoscopic guidance under general anesthesia. The postoperative course was uneventful, except for a few days of serious discharge, which diminished. Ectopic teeth is a rare disease, which may be supernumerary or may result from abnormal dentition.1 The supernumerary intranasal teeth may be observed at various ages, ranging from 6 to 61 years. It is reported to be more common in male patients (62%) and in the left side.2 The etiology of intranasal tooth eruption has not been clarified. Abnormal tissue interactions during development may potentially lead to ectopic tooth development and eruption.3 A total of 84.6% of patients are symptomathic.3 Nasal teeth can cause nasal discharge, foul smell, nasal obstruction, nasal discomfort, headache, facial pain, epistaxis, epiphora, sinusitis, rhinitis, nasal septal deviations, nasal septal abscess, and oronasal fistula.1,2 Sometimes an intranasal tooth may be asymptomatic as in the current case. The diagnosis of an intranasal tooth can be confirmed clinically and radiologically. An intranasal tooth is often a hard white mass and can sometimes be covered completely by nasal mucosa and surrounded by granulation tissue and necrotic debris.2 Radiologic examinations, especially CT, are useful to identify the exact position and helps to decide the surgical approach.1 The differential diagnosis of intranasal white mass includes nasal foreign body, rhinolith, exocytosis, odontomas, osteomas, malignant tumors.1,2 The general opinion on the treatment of nasal teeth is the extraction when diagnosed because of potential morbidity, even if, asymptomatic.1 It may be extracted with either transnasal or transpalatal approach. Endoscopic removal of intranasal teeth can provide good illumination, better visualization, and has less morbidity compared with conventional approach.2 As the diagnosis of the disease is easy with a simple endoscopic examination, all of the patients referred to an otorhinolaryngology clinic should be completely examined. The supernumerary intranasal teeth are rare cases. Early extraction is advocated because of potential morbidity.
Correspondence
REFERENCES 1. Krishnan B, Parida PK, Gopalakrishnan S, et al. An unusual cause of epistaxis in a young patient: the supernumerary nasal tooth. Oral Maxillofac Surg 2013;17:315–317 2. Iwai T, Aoki N, Yamashita Y, et al. Endoscopic removal of bilateral supernumerary intranasal teeth. J Oral Maxillofac Surg 2012;70: 1030–1034 3. Mohan S, Kankariya H, Harjani H. Ectopic third molar in the maxillary sinus. Natl J Maxillofac Surg 2011;2:222–224
Clinicopathologic Assessment of Myositis Ossificans Circumscripta of the Masseter Muscles To the Editor: Myositis ossificans (MO) is a heterotopic bone formation within the muscle, and can be classified into 3 subtypes: fibrodysplasia ossificans progressiva, neurogenic MO, and traumatic MO circumscripta, which is a benign and reactive bone formation within the muscle after trauma. Traumatic MO circumscripta mostly occurs in the thigh and brachium,1 and the occurrence in the masticatory muscles, though rare, usually results in temporomandibular joint dysfunction. Although the exact cause of MO circumscripta remains unknown, it has been presumed that infection, hematoma, tearing of the periosteum and bony metaplasia of the muscle and fibrous connective tissues cause heterotopic ossification.2 A 36-year-old man experiencing severe trismus was referred to our hospital. He was, at the age of 21, under confinement for 1 year and frequently abused about the face. On physical examination, maximal interincisal distance (MID) was 10 mm and hard masses were palpated in the bilateral cheeks (Fig. 1). Computed tomography scan showed calcified masses in the bilateral masseter muscles extending from the zygomatic arch to the ascending ramus of the mandible (Fig. 1). He was diagnosed as MO circumscripta, and as the disease state had not been progressive for the past several years, surgical intervention was selected to alleviate the trismus. We performed bilateral osteotomies of the bony fragments, and bilateral coronoidectomies to eliminate any possibility of further restriction of the jaw function. Following this, gentle manipulation of the jaw resulted in a full range of motion, achieving an MID of 40 mm,
Muhammed Sedat Sakat, MD Korhan Kilic, MD Otorhinolaryngology Clinics Palandoken State Hospital Erzurum, Turkey Mustafa Sitki Gozeler, MD Harun Ucuncu, MD Department of Otorhinolaryngology Atatu¨rk University Erzurum, Turkey [email protected] Ozan Kuduban, MD Department of Otorhinolaryngology Erzurum Region Education and Research Hospital Erzurum, Turkey
#
2015 Mutaz B. Habal, MD
FIGURE 1. The maximal interincisal distance improved from 10 mm (before treatment) to 36 mm (2 months postoperatively). Preoperative 3D-CT images revealed large ossified masses in the masseter region of the left and right sides (50 and 21ı ¨mm in maximum diameter, respectively). Pathologic findings of the left ossified mass (hematoxylin and eosin stain) showed that rigid bony structure constituted a large portion of the specimen, and striate muscles (black arrow) existed between bone trabeculae where bone marrow is normally present. 3D-CT, three-dimensional computed tomography.
2025
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Correspondence
The Journal of Craniofacial Surgery
and the wound was closed in layers. The day after the operation, the patient’s trismus was temporarily aggravated probably because of postoperative edema and pain in the masseter region. After 2 months of physical therapy, the patient, however, presented an MID of 36 mm (Fig. 1). And, as of 1 year after the operation, there was no recurrence of trismus or heterotopic ossification, providing a high level of patient satisfaction and a good functionality. The most common treatment of MO circumscripta is excision of the lesion or osteotomy, though exacerbation or repeated relapses may occur in some patients.3 Two successive phases of the disease are known: an early phase with abundant primary vascular cells and angiogenesis, and an ossification phase with premature ectopic bone formation.1 To prevent recurrence, surgical intervention is recommended after the end of ossification phase. In addition to the removal of a mass, coronoidectomy, or at least coronoidotomy, can be the choice of treatment to eliminate possibility of disturbances in jaw movements.
PATHOLOGIC ASSESSMENT The pathologic examination of the decalcified, hematoxylin–eosin stained specimens revealed that the ossified mass had a dense and rigid bony structure of mature lamellar bone, including Haversian systems and bone trabeculae (Fig. 1). This indicates osteogenesis was once dynamic though later subsided. Moreover, most interestingly, there were striate muscles between the trabeculae, where bone marrow is normally present. This suggests that osteogenesis was induced along existing masseter muscles, presumably by muscle-derived stem cells differentiating into osteoblasts.4 There, however, remains a slight possibility of migration of marrowderived stem cells into the muscle region. Takanobu Mashiko, MD Department of Plastic Surgery Tokyo Metropolitan Police Hospital Nakano, Nakano-Ku Tokyo, Japan [email protected] Tanetaka Akizuki, MD Yorikatsu Watanabe, MD Department of Plastic Surgery Tokyo Metropolitan Police Hospital Nakano-ku, Tokyo, Japan Ryo Sasaki, DDS Department of Plastic Surgery Tokyo Women’s Medical University School of Medicine Shinjuku-ku, Tokyo, Japan
Volume 26, Number 6, September 2015
REFERENCES 1. Godhi SS, Singh A, Kukreja P, et al. Myositis ossificans circumscripta involving bilateral masticatory muscles. J Craniofac Surg 2011;22:e11–e13 2. Molloy JC, McGuirk RA. Treatment of traumatic myositis ossificans circumscripta: use of aspiration and steroids. J Trauma 1976;16:851–857 3. Aoki T, Naito H, Ota Y, et al. Myositis ossificans traumatica of the masticatory muscles: review of the literature and report of a case. J Oral Maxillofac Surg 2002;60:1083–1088 4. Shi X, Garry DJ. Muscle stem cells in development, regeneration, and disease. Genes Dev 2006;20:1692–1708
Basosquamous or Basal Cell Carcinoma? The Importance of the Differential Diagnosis To the Editor: Basosquamous carcinoma (BSC) is a rare and aggressive type of basal cell carcinoma (BCC) more frequently involving head and neck region1 characterized by high tendency to recur locally or at distant sites. Metastasis have been observed with a latency period that can vary from 0 to 30 years.2 In some cases, first diagnosis of the skin lesion is consistent with BCC, and only after metastasis detection diagnosis of BSC has been done.3,4 Immunohistochemistry is useful to diagnose BSC: areas of squamous cell carcinoma are cytokeratin AE1, cytokeratin AE3, and cell adhesion molecule 52 positive and show variable staining with epithelial membrane antigen. Characteristically, the transition zone shows a decline of staining with graded reactivity for Ber-EP4 as the morphology of the tumor moves from BCC to squamous cell carcinoma.1,5 Nevertheless, approximately 80% of the lesions initially diagnosed as BCC were found to have basosquamous histology upon metastasis, as in our case.2 It can support the hypothesis that BSC derives from differentiation of a BCC, or can be explained as an histopathologic misdiagnosis or sample bias because of tumor heterogenity.3,4 A 60-years-old woman presented at our department referring a progressive unpainful swelling of the left submandibular region. Medical history was unremarkable, except for a BCC of the left nose that was treated with surgical resection in free margins approximately 10 years before. Clinical examination revealed no signs of local recurrence, and no suspect lesions were detected after fibrolaringoscopy. Computed tomography scan with contrast revealed a 2 cm colliquative lymphadenopathy at level Ib in the left neck, with the presence of some enlarged nodes at the same side (Fig. 1A). Fine-needle aspiration cytology was consistent with carcinoma. Whole body positron emission tomography with 2-deoxy-2-[fluorine-18]fluoro-D-glucose integrated with computed
Munehiro Yokoyama, MD Department of Pathology Tokyo Metropolitan Police Hospital Nakano, Nakano-Ku, Tokyo, Japan Kotaro Yoshimura, MD Kazuhide Mineda, MD Department of Plastic Surgery University of Tokyo School of Medicine Hongo, Bunkyo-Ku, Tokyo, Japan
2026
FIGURE 1. A, Preoperative axial CT-scan with contrast. B, Preoperative PET-CT (sagittal view). Arrow: pathologic submandibular node.
#
2015 Mutaz B. Habal, MD
Copyright © 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
