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Cavernous haemangioma in the mandible
C A V E R N O U S HAEMANGIOMA IN THE MANDIBLE J. A. CRAN, D.D.Sc., Adel.
Reader in Oral Surgery and Pathology, Dental School, University qf Adelaide and Hon. Dental Surgeon at Royal Adelaide Hcspital CASE REPORT A FEMALEchild aged IO years was referred to the writer on 31/8/61 with a circumscribed hard swelling in the region of the lower left first and second deciduous molars. A radiograph showed an erupted lower left second bicuspid with no surrounding radiolucency. The same condition was present on the opposite side. There was some inflammation
FIG. I
Section of gingivae showing large blood vessels × 8o. of the gingivae surrounding the lower left second deciduous molar but this was considered to be due to trauma. Clinical examination indicated considerable buccal displacement of the lower left second bicuspid. The patient was referred to an orthodontist who recommended extraction of the lower second deciduous molars. At the time of extraction a small piece of hyperplastic gingivae was sent for examination but the examining pathologist was not prepared to make a report owing to the damaged state of the tissue (Fig. I). The patient was seen again on 28/9/6I and on 29/I/62 when it was recorded that the lower left second bicuspid had moved lingually. On I8/5/62 a lateral oblique radiograph of the mandible was taken and a radiolucency found below the apices of the lower left first and second molars (Fig. 2). The second molar was mobile and displaced lingually. The patient was referred again to the writer who found at this 32
CAVERNOUS HAEMANGIOMA IN THE MANDIBLE
33
stage enlargement of the lower part of the left side of the face and a history of spontaneous bleeding at night. It was considered that a biopsy was necessary and this was done by Mr. D. N. Robinson, Hon. Plastic Surgeon, Royal Adelaide Hospital. As a result of the extensive bleeding only a small piece of tissue from the bony enlargement was obtained. This was examined but no definite report could be made but the writer made these comments in his report to Mr. Robinson: 'As it is important to you that an opinion be given and as I was present at the operation I feel I must give you some information on the lesion.
FIG. 2
Lateral oblique radiograph showing a radiolucency at the apices of the left first and second
molars and involving the third molar region. Basing my opinion on the radiographic appearance, biopsy material, clinical evidence both prior to and during operation I would suggest that this is possibly a blood vessel malformation or hamartoma.' The piece of gingival tissue removed on 3I/8/6I was re-examined and it was considered that it represented a clue to the condition (Fig. I). It was decided to defer treatment and re-examine the patient at a later date. On i/2/63 it was apparent that further enlargement of the face had occurred and again the mother gave a history of nocturnal haemorrhage. The lower left first and second molars were by this time very mobile and second bicuspid was slightly mobile. The patient complained of pulsation in the area and this was most noticeable in the region of the facial artery. The patient was again examined on 28/2/63 when it was considered that the face had enlarged further. In addition small blood vessels were radiating from the gingivae into the buccal sulcus and floor of the mouth (Fig. 3). At this stage Mr. D. N. Robinson decided that it was necessary to resect the mandible and the patient was admitted to the Royal Adelaide Hospital where the iaw was resected on 9/5/63. Silver cap splints were constructed and immediately after the resection and insertion of a bone graft the iaws
FIG. 3 Photograph showing lingual displacement of the lower left first and second molar and radiating blood vessels in the gingivae.
FIG. 4 Section of gingivae taken from the lower left molar area of the resected jaw showing large blood vessels, one about to rupture x 400.
CAVERNOUS HAEMANGIOMA IN THE MANDIBLE
35
were held together by intermaxillary elastics. The operation was long and accompanied by serious haemorrhage which was difficult to control and thirteen pints of blood were administered. The jaw was resected from below the inferior alveolar foramen to the central incisor region. The turnout was seen almost to have perforated the lingual cortical bone. A biopsy was taken from the lingual area where perforation was occurring and from the gingivae. The biopsy report on the lingual tissue was: 'Tissue consists of thin layer of normal bone on the surface, and the underlying tissue consists of very large vascular spaces separated by cellular connective tissue. The diagnosis is cavernous haemangioma.'
The report for the tissues from the gingivae was: (Fig. 4): 'Small piece of tissue covered with stratified squamous epithelium. T h e underlying connective tissue contains large thin walled blood vessels one of which is covered by a layer consisting of three flattened epithelial cells indicating impending rupture of the blood vessel.'
This explained the spontaneous haemorrhages. A partial denture has been constructed and this is functioning very satisfactorily. The patient has no difficulty in opening the jaws, the only disability being a slight deviation to the affected side. DISCUSSION Recently a case of central haemangioma in an infant has been reported Hankey (I964). In this case radiotherapy was undertaken. In the case just reported it was considered that radiotherapy would complicate surgical treatment undertaken later. As in this case another blood vessel malformation was present. Any operative interferences with this lesion by the dental surgeon could have resulted in a fatal haemorrhage as has happened in other reported cases Broderick et al. (I933). These blood vessel lesions vary considerably in their histology and their clinical behaviour. Some are much more active than others. A case recently reported by the writer Lindsay et al. (I963), is suggestive of a hamartoma which ceased development at a certain stage in jaw development. The present case leads one to speculate whether if surgery had not become an urgent necessity growth would have ceased at the time when jaw development was less active. This case emphasizes the importance of a satisfactory diagnosis and avoidance of treatment until a diagnosis has been made. REFERENCES HANKEY~ G. T. (I964). Brit. ft. oral Surg. x, 3. BRODERICK, R. A. & ROUND, H. (I933). Lancet, 2, 13. BRODSKY, R. H. (I934). Dent. Dig. 4o, 60. ERICH, J. B. (I947). Amer. J. Orthodont. and oral Surg. 33, 6II. LINDSAY, BRUCE & CRAN, J. A. (I963). Aust. dent. J. 8, 2.
Reader in Oral Surgery and Pathology, Dental School, University qf Adelaide and Hon. Dental Surgeon at Royal Adelaide Hcspital CASE REPORT A FEMALEchild aged IO years was referred to the writer on 31/8/61 with a circumscribed hard swelling in the region of the lower left first and second deciduous molars. A radiograph showed an erupted lower left second bicuspid with no surrounding radiolucency. The same condition was present on the opposite side. There was some inflammation
FIG. I
Section of gingivae showing large blood vessels × 8o. of the gingivae surrounding the lower left second deciduous molar but this was considered to be due to trauma. Clinical examination indicated considerable buccal displacement of the lower left second bicuspid. The patient was referred to an orthodontist who recommended extraction of the lower second deciduous molars. At the time of extraction a small piece of hyperplastic gingivae was sent for examination but the examining pathologist was not prepared to make a report owing to the damaged state of the tissue (Fig. I). The patient was seen again on 28/9/6I and on 29/I/62 when it was recorded that the lower left second bicuspid had moved lingually. On I8/5/62 a lateral oblique radiograph of the mandible was taken and a radiolucency found below the apices of the lower left first and second molars (Fig. 2). The second molar was mobile and displaced lingually. The patient was referred again to the writer who found at this 32
CAVERNOUS HAEMANGIOMA IN THE MANDIBLE
33
stage enlargement of the lower part of the left side of the face and a history of spontaneous bleeding at night. It was considered that a biopsy was necessary and this was done by Mr. D. N. Robinson, Hon. Plastic Surgeon, Royal Adelaide Hospital. As a result of the extensive bleeding only a small piece of tissue from the bony enlargement was obtained. This was examined but no definite report could be made but the writer made these comments in his report to Mr. Robinson: 'As it is important to you that an opinion be given and as I was present at the operation I feel I must give you some information on the lesion.
FIG. 2
Lateral oblique radiograph showing a radiolucency at the apices of the left first and second
molars and involving the third molar region. Basing my opinion on the radiographic appearance, biopsy material, clinical evidence both prior to and during operation I would suggest that this is possibly a blood vessel malformation or hamartoma.' The piece of gingival tissue removed on 3I/8/6I was re-examined and it was considered that it represented a clue to the condition (Fig. I). It was decided to defer treatment and re-examine the patient at a later date. On i/2/63 it was apparent that further enlargement of the face had occurred and again the mother gave a history of nocturnal haemorrhage. The lower left first and second molars were by this time very mobile and second bicuspid was slightly mobile. The patient complained of pulsation in the area and this was most noticeable in the region of the facial artery. The patient was again examined on 28/2/63 when it was considered that the face had enlarged further. In addition small blood vessels were radiating from the gingivae into the buccal sulcus and floor of the mouth (Fig. 3). At this stage Mr. D. N. Robinson decided that it was necessary to resect the mandible and the patient was admitted to the Royal Adelaide Hospital where the iaw was resected on 9/5/63. Silver cap splints were constructed and immediately after the resection and insertion of a bone graft the iaws
FIG. 3 Photograph showing lingual displacement of the lower left first and second molar and radiating blood vessels in the gingivae.
FIG. 4 Section of gingivae taken from the lower left molar area of the resected jaw showing large blood vessels, one about to rupture x 400.
CAVERNOUS HAEMANGIOMA IN THE MANDIBLE
35
were held together by intermaxillary elastics. The operation was long and accompanied by serious haemorrhage which was difficult to control and thirteen pints of blood were administered. The jaw was resected from below the inferior alveolar foramen to the central incisor region. The turnout was seen almost to have perforated the lingual cortical bone. A biopsy was taken from the lingual area where perforation was occurring and from the gingivae. The biopsy report on the lingual tissue was: 'Tissue consists of thin layer of normal bone on the surface, and the underlying tissue consists of very large vascular spaces separated by cellular connective tissue. The diagnosis is cavernous haemangioma.'
The report for the tissues from the gingivae was: (Fig. 4): 'Small piece of tissue covered with stratified squamous epithelium. T h e underlying connective tissue contains large thin walled blood vessels one of which is covered by a layer consisting of three flattened epithelial cells indicating impending rupture of the blood vessel.'
This explained the spontaneous haemorrhages. A partial denture has been constructed and this is functioning very satisfactorily. The patient has no difficulty in opening the jaws, the only disability being a slight deviation to the affected side. DISCUSSION Recently a case of central haemangioma in an infant has been reported Hankey (I964). In this case radiotherapy was undertaken. In the case just reported it was considered that radiotherapy would complicate surgical treatment undertaken later. As in this case another blood vessel malformation was present. Any operative interferences with this lesion by the dental surgeon could have resulted in a fatal haemorrhage as has happened in other reported cases Broderick et al. (I933). These blood vessel lesions vary considerably in their histology and their clinical behaviour. Some are much more active than others. A case recently reported by the writer Lindsay et al. (I963), is suggestive of a hamartoma which ceased development at a certain stage in jaw development. The present case leads one to speculate whether if surgery had not become an urgent necessity growth would have ceased at the time when jaw development was less active. This case emphasizes the importance of a satisfactory diagnosis and avoidance of treatment until a diagnosis has been made. REFERENCES HANKEY~ G. T. (I964). Brit. ft. oral Surg. x, 3. BRODERICK, R. A. & ROUND, H. (I933). Lancet, 2, 13. BRODSKY, R. H. (I934). Dent. Dig. 4o, 60. ERICH, J. B. (I947). Amer. J. Orthodont. and oral Surg. 33, 6II. LINDSAY, BRUCE & CRAN, J. A. (I963). Aust. dent. J. 8, 2.