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Congenital transmandibular dermoid: case report
British Journal of Oral and Maxillofacial Surgery (2000) 38, 107–109 © 2000 The British Association of Oral and Maxillofacial Surgeons DOI: 10.1054/bjom.1999.0028
BRITISH JOURNAL OF ORAL
& M A X I L L O FA C I A L S U R G E RY
Congenital transmandibular dermoid: case report O. Koshy,* L. Ion,† N. Waterhouse‡ *Registrar in Plastic Surgery, Whiston General Hospital, Prescot, Merseyside; †Registrar in Plastic Surgery, Chelsea and Westminster Hospital, London; ‡Consultant Craniofacial and Plastic Surgeon, Chelsea and Westminister Craniofacial Unit, Chelsea and Westminster Hospital, London, UK Congenital dermoids arise as result of epidermal sequestration during development and are quite common along the lines of fusion. While submandibular dermoids are common, we could find only a few references to dermoid cysts embedded in the mandibular bone itself and we describe one such case.
CASE REPORT A 21-year-old North African man presented with a painful swelling of the right lower cheek and neck. He said that the swelling had been present for several years, but had become painful during the previous two weeks, preventing him from eating normally. On examination, he was pyrexial, with trismus, and had two discharging sinuses located at the right angle of the jaw and in the submandibular region, respectively. There was no associated cervical lymphadenopathy and no obvious intraoral disease. An orthopantomogram showed a grossly enlarged right mandibular canal (Fig. 1) with missing right premolars and first molar but no dental abscess. Exploration under general anaesthesia confirmed submandibular and mandibular angle abscesses, which were drained through two incisions over each abscess. Granular pus was evacuated and sent for microbiological examination. Actinomycosis was reported and the patient was given long-term treatment with penicillin. Although the cellulitis and trismus subsided and the patient’s condition improved, the two sinuses persisted (Fig. 2). A well-defined lump became palpable under each sinus. As the swellings became more prominent, we did a fine-needle aspiration biopsy and microscopy again suggested ‘actinomyces-like organisms’, so we increased the dose of penicillin. A computed tomogram (CT) scan of the head and neck done shortly afterwards showed two welldefined submandibular masses connected to the inferior border of the ramus and the angle of the mandible (Fig. 3). The enlarged mandibular canal noted on the orthopanthomogram was confirmed by the CT. In view of the persisting masses, we decided to explore the neck with a view to debulking the chronically infected tissues. During surgery, we found two encapsulated cysts on either side of the right submandibular gland which contained hair and sebaceous material. As the diagnosis was now obviously dermoid cysts, we decided to excise the lesions in toto. On further dissection, we found the cysts were connected by a 6 cm transmandibular segment, which was gently and quite easily detached from the bony canal with the aid of sinus probe (Fig. 4). The posterior end of the tract was divided from the cyst, and the transmandibular segment was easily pulled out with the anterior cyst. There was no evidence of the inferior alveolar nerve either on the specimen or
Fig. 1 – Orthopantomogram showing a large right mandibular ‘canal’ with one aperture at the anterior body of the mandible and another at the right mandibular angle, which is attenuated.
Fig. 2 – The anterior sinus with underlying cystic lesion.
within this ‘canal’. The wounds were closed without drainage and the patient made an uneventful recovery. He did, however, retain normal sensation in the inferior alveolar nerve territory and, by digitally enhancing the orthopantomogram, we saw that the inferior alveolar canal was lying immediately above the dermoid canal. Histological examination confirmed the diagnosis of dermoid cyst (Fig. 5) but further microbiological investigation revealed no evidence of actinomycosis. Penicillin was discontinued, the wounds have healed and the patient remains well. 107
108
British Journal of Oral and Maxillofacial Surgery
Fig. 3 – CT showing two masses situated in close proximity to the mandible with breach of the mandibular cortex.
Fig. 4 – Low-power view of a section of the specimen showing epidermis with appendages, in keeping with the appearances of a dermoid cyst. Part of the sinus wall contains hair shaft material and patchy chronic inflammatory cells with fibrosis. Haematoxylin and eosin, original magnification × 25.
There have been numerous reports of dermoids of the neck region, since first documented by Jourdain in 1778.1 A retrospective review by Katz showed the relative rarity of lateral submandibular and intraosseous dermoids.2 There is debate on the embryological origin of these dermoids, the main theories being that they are congenital or acquired.3 The most popular theory about is that they result from entrapment of midline ectodermal tissue during fusion of the mandibular and hyoid branchial arches.4 Dermoid cysts should be differentiated from teratomas, thyroglossal duct cysts and odontogenic cysts. Teratomas are very rare and histologically have
elements of all three primary embryonic germ layers.5 Thyroglossal duct cysts are more common and have some features in common with dermoids. In a review by deMello et al.6 of 75 midline cervical masses preoperatively diagnosed as thyroglossal duct cysts, 11 were reclassified as dermoid cysts because of their squamous epithelial lining and the epidermal appendages within the cysts. A further six cases were classified as ‘mixed’ because of epidermal appendages as well as epithelial sinus tracts or thyroid follicles. They therefore concluded a common origin for both lesions and suggested Sistrunk procedure for all such lesions. In our case, the medial cyst was strictly paramedian and
Congenital transmandibular dermoid
109
appearance of actinomycosis, we conclude that our patient had indeed initially developed actinomycosis and subsequent secondary infection by oral commensals within these congenital transmandibular dermoid cysts. Acknowledgements We thank Dr S. Shousha, Consultant Pathologist at the Charing Cross Hospital, for help in preparing this case report.
References Fig. 5 – Peroperative view showing probe passing along the tumour and through the mandible from submandibular to the lateral mandible angle.
1. 2. 3.
lying above the hyoid. Although a CT scan adequately identified the extent of the lesion in our case, ultrasound scan5 has been suggested as the investigation of first choice.7 Odontogenic cysts only rarely contain sebaceous elements and also do not easily enucleate like dermoids which have a thick fibrous sheath, as in our case. It is, however, difficult to establish whether our dermoid is of odontogenic or soft-tissue origin, as both the cysts were extra-osseous but the intermediate tract was entirely within the body and horizontal ramus of the right side of the mandible. Patten proposed that an abnormal mesodermal growth could cause ectoderm to be trapped and it would seem possible that, in our case, the dermoid developed intraosseously and eroded its way out at the parasymphysis and angle of the mandible.8 Our patient was initially treated as an acute case of actinomycosis. The swelling had gradually increased over a year but the patient could not recollect its presence in adolescence or in childhood. The granular nature of the scanty amount of pus aspirated at the first operation raised the clinical suspicion of actinomycosis and the microbiologist duly reported ‘actinomycosis-like organisms’ on microscopy. Histopathological examination also showed ‘heavy chronic inflammatory cell infilterate in the dermis’. Gupta et al.9 have commented on the difficulties of establishing a diagnosis of actinomycosis and emphasized the need for repeated smear examinations, anaerobic cultures, gas chromatography of metabolic products and sensitivity tests. Although the contents of a dermoid cyst may appear granular and create an apparent
4. 5. 6. 7. 8. 9.
Jourdain A. Traité des maladies chirurgicales de la bouche. Paris: Valleyre, 1778: 457–458. Katz AD. Midline dermoid tumours of the neck. Arch Surg 1974; 109: 822–823. Torres JS. Epidermoid cysts in the oral cavity. Oral Surg Oral Med Oral Pathol 1970; 30: 592–600. Shafer WG, Hine MK, Levy BM (eds) A Textbook of Oral Pathology, 4th edn. Philadelphia: WB Saunders, 1974: 78–79. Vuckovic N, Vuckovic D. Cystic teratoma of the neck. Medicinski Pregled 1991; 44: 39–40. deMello DE, Lima JA, Liapis H. Midline cervical cysts in children: thyroglossal anomalies. Arch Otolaryngo Head Neck Surg 1987; 113: 418–420. Tuffin JR, Theaker E. True lateral dermoid cyst of the neck. Int J Oral Maxillofac Surg 1991; 20: 275–276. Patten BM. The normal development of the facial region. In: Pruzanski S (ed) Congenital Anomalies of the Face and Associated Structures. Springfield 1961: 41. Gupta DS, Gupta MK, Naidu NG. Mandibular osteomyelitis caused by Actinomyces Israelii: report of a case. J Maxillofac Surg 1986; 14: 291–293.
The Authors Ommen Koshy FRCS MCh (Plast) Registrar in Plastic Surgery Whiston General Hospital Prescot, UK Lucian Ion FRCS (Plast) Registrar in Plastic Surgery Chelsea and Westminster Hospital London, UK N. Waterhouse FRCS (Plast) Consultant Craniofacial and Plastic Surgeon Chelsea and Westminster Craniofacial Unit Chelsea and Westminster Hospital Fulham Road London, UK Correspondence and requests for offprints to: Mr L. Ion FRCS (Plast), Registrar in Plastic Surgery, Chelsea and Westminster Hospital, 369 Fulham Road, London SW10 9NH, UK Paper received 11 February 1998 Accepted 2 February 1999
BRITISH JOURNAL OF ORAL
& M A X I L L O FA C I A L S U R G E RY
Congenital transmandibular dermoid: case report O. Koshy,* L. Ion,† N. Waterhouse‡ *Registrar in Plastic Surgery, Whiston General Hospital, Prescot, Merseyside; †Registrar in Plastic Surgery, Chelsea and Westminster Hospital, London; ‡Consultant Craniofacial and Plastic Surgeon, Chelsea and Westminister Craniofacial Unit, Chelsea and Westminster Hospital, London, UK Congenital dermoids arise as result of epidermal sequestration during development and are quite common along the lines of fusion. While submandibular dermoids are common, we could find only a few references to dermoid cysts embedded in the mandibular bone itself and we describe one such case.
CASE REPORT A 21-year-old North African man presented with a painful swelling of the right lower cheek and neck. He said that the swelling had been present for several years, but had become painful during the previous two weeks, preventing him from eating normally. On examination, he was pyrexial, with trismus, and had two discharging sinuses located at the right angle of the jaw and in the submandibular region, respectively. There was no associated cervical lymphadenopathy and no obvious intraoral disease. An orthopantomogram showed a grossly enlarged right mandibular canal (Fig. 1) with missing right premolars and first molar but no dental abscess. Exploration under general anaesthesia confirmed submandibular and mandibular angle abscesses, which were drained through two incisions over each abscess. Granular pus was evacuated and sent for microbiological examination. Actinomycosis was reported and the patient was given long-term treatment with penicillin. Although the cellulitis and trismus subsided and the patient’s condition improved, the two sinuses persisted (Fig. 2). A well-defined lump became palpable under each sinus. As the swellings became more prominent, we did a fine-needle aspiration biopsy and microscopy again suggested ‘actinomyces-like organisms’, so we increased the dose of penicillin. A computed tomogram (CT) scan of the head and neck done shortly afterwards showed two welldefined submandibular masses connected to the inferior border of the ramus and the angle of the mandible (Fig. 3). The enlarged mandibular canal noted on the orthopanthomogram was confirmed by the CT. In view of the persisting masses, we decided to explore the neck with a view to debulking the chronically infected tissues. During surgery, we found two encapsulated cysts on either side of the right submandibular gland which contained hair and sebaceous material. As the diagnosis was now obviously dermoid cysts, we decided to excise the lesions in toto. On further dissection, we found the cysts were connected by a 6 cm transmandibular segment, which was gently and quite easily detached from the bony canal with the aid of sinus probe (Fig. 4). The posterior end of the tract was divided from the cyst, and the transmandibular segment was easily pulled out with the anterior cyst. There was no evidence of the inferior alveolar nerve either on the specimen or
Fig. 1 – Orthopantomogram showing a large right mandibular ‘canal’ with one aperture at the anterior body of the mandible and another at the right mandibular angle, which is attenuated.
Fig. 2 – The anterior sinus with underlying cystic lesion.
within this ‘canal’. The wounds were closed without drainage and the patient made an uneventful recovery. He did, however, retain normal sensation in the inferior alveolar nerve territory and, by digitally enhancing the orthopantomogram, we saw that the inferior alveolar canal was lying immediately above the dermoid canal. Histological examination confirmed the diagnosis of dermoid cyst (Fig. 5) but further microbiological investigation revealed no evidence of actinomycosis. Penicillin was discontinued, the wounds have healed and the patient remains well. 107
108
British Journal of Oral and Maxillofacial Surgery
Fig. 3 – CT showing two masses situated in close proximity to the mandible with breach of the mandibular cortex.
Fig. 4 – Low-power view of a section of the specimen showing epidermis with appendages, in keeping with the appearances of a dermoid cyst. Part of the sinus wall contains hair shaft material and patchy chronic inflammatory cells with fibrosis. Haematoxylin and eosin, original magnification × 25.
There have been numerous reports of dermoids of the neck region, since first documented by Jourdain in 1778.1 A retrospective review by Katz showed the relative rarity of lateral submandibular and intraosseous dermoids.2 There is debate on the embryological origin of these dermoids, the main theories being that they are congenital or acquired.3 The most popular theory about is that they result from entrapment of midline ectodermal tissue during fusion of the mandibular and hyoid branchial arches.4 Dermoid cysts should be differentiated from teratomas, thyroglossal duct cysts and odontogenic cysts. Teratomas are very rare and histologically have
elements of all three primary embryonic germ layers.5 Thyroglossal duct cysts are more common and have some features in common with dermoids. In a review by deMello et al.6 of 75 midline cervical masses preoperatively diagnosed as thyroglossal duct cysts, 11 were reclassified as dermoid cysts because of their squamous epithelial lining and the epidermal appendages within the cysts. A further six cases were classified as ‘mixed’ because of epidermal appendages as well as epithelial sinus tracts or thyroid follicles. They therefore concluded a common origin for both lesions and suggested Sistrunk procedure for all such lesions. In our case, the medial cyst was strictly paramedian and
Congenital transmandibular dermoid
109
appearance of actinomycosis, we conclude that our patient had indeed initially developed actinomycosis and subsequent secondary infection by oral commensals within these congenital transmandibular dermoid cysts. Acknowledgements We thank Dr S. Shousha, Consultant Pathologist at the Charing Cross Hospital, for help in preparing this case report.
References Fig. 5 – Peroperative view showing probe passing along the tumour and through the mandible from submandibular to the lateral mandible angle.
1. 2. 3.
lying above the hyoid. Although a CT scan adequately identified the extent of the lesion in our case, ultrasound scan5 has been suggested as the investigation of first choice.7 Odontogenic cysts only rarely contain sebaceous elements and also do not easily enucleate like dermoids which have a thick fibrous sheath, as in our case. It is, however, difficult to establish whether our dermoid is of odontogenic or soft-tissue origin, as both the cysts were extra-osseous but the intermediate tract was entirely within the body and horizontal ramus of the right side of the mandible. Patten proposed that an abnormal mesodermal growth could cause ectoderm to be trapped and it would seem possible that, in our case, the dermoid developed intraosseously and eroded its way out at the parasymphysis and angle of the mandible.8 Our patient was initially treated as an acute case of actinomycosis. The swelling had gradually increased over a year but the patient could not recollect its presence in adolescence or in childhood. The granular nature of the scanty amount of pus aspirated at the first operation raised the clinical suspicion of actinomycosis and the microbiologist duly reported ‘actinomycosis-like organisms’ on microscopy. Histopathological examination also showed ‘heavy chronic inflammatory cell infilterate in the dermis’. Gupta et al.9 have commented on the difficulties of establishing a diagnosis of actinomycosis and emphasized the need for repeated smear examinations, anaerobic cultures, gas chromatography of metabolic products and sensitivity tests. Although the contents of a dermoid cyst may appear granular and create an apparent
4. 5. 6. 7. 8. 9.
Jourdain A. Traité des maladies chirurgicales de la bouche. Paris: Valleyre, 1778: 457–458. Katz AD. Midline dermoid tumours of the neck. Arch Surg 1974; 109: 822–823. Torres JS. Epidermoid cysts in the oral cavity. Oral Surg Oral Med Oral Pathol 1970; 30: 592–600. Shafer WG, Hine MK, Levy BM (eds) A Textbook of Oral Pathology, 4th edn. Philadelphia: WB Saunders, 1974: 78–79. Vuckovic N, Vuckovic D. Cystic teratoma of the neck. Medicinski Pregled 1991; 44: 39–40. deMello DE, Lima JA, Liapis H. Midline cervical cysts in children: thyroglossal anomalies. Arch Otolaryngo Head Neck Surg 1987; 113: 418–420. Tuffin JR, Theaker E. True lateral dermoid cyst of the neck. Int J Oral Maxillofac Surg 1991; 20: 275–276. Patten BM. The normal development of the facial region. In: Pruzanski S (ed) Congenital Anomalies of the Face and Associated Structures. Springfield 1961: 41. Gupta DS, Gupta MK, Naidu NG. Mandibular osteomyelitis caused by Actinomyces Israelii: report of a case. J Maxillofac Surg 1986; 14: 291–293.
The Authors Ommen Koshy FRCS MCh (Plast) Registrar in Plastic Surgery Whiston General Hospital Prescot, UK Lucian Ion FRCS (Plast) Registrar in Plastic Surgery Chelsea and Westminster Hospital London, UK N. Waterhouse FRCS (Plast) Consultant Craniofacial and Plastic Surgeon Chelsea and Westminster Craniofacial Unit Chelsea and Westminster Hospital Fulham Road London, UK Correspondence and requests for offprints to: Mr L. Ion FRCS (Plast), Registrar in Plastic Surgery, Chelsea and Westminster Hospital, 369 Fulham Road, London SW10 9NH, UK Paper received 11 February 1998 Accepted 2 February 1999