British Journal of Oral Surgery
14 (1976) 156-162
ATROPHY OF THE MANDIBLE: RECONSTRUCTION FOLLOWING FRACTURE DAVID R. JAMES,M.B.,
B.Ch., B.D.S., F.D.S.R.C.S.(Eng.)l
Queen Mary’s Hospital, Roehampton, London Summary. A rational approach to the management of spontaneous fracture of the severely atrophic mandible is illustrated by means of a case report, A study of the literature reveals that some confusion exists concerning the terms ‘atrophy’ and ‘osteolysis’, and an attempt is made to distinguish between them.
Introduction Spontaneous fracture of the mandible occurs when the bone is so weakened that it is no longer able to withstand the normal stresses to which it is subjected. This weakening may be due to a specific disease entity within the bone, or to atrophy. There is some confusion in the literature concerning the terms ‘atrophy’ and “osteolysis’, and it is the aim of this paper to distinguish between these two terms, and by means of a case report to propose a rational approach to the management of spontaneous fracture of the atrophic mandible.
Case report A 60-year-old schoolmaster was admitted to hospital in December 1971 with a five-week history of increasing pain and swelling of the right side of his face. There was a past history of cervical spondylosis, and the patient had worn a cervical collar intermittently for eight years. A dental clearance had been performed because of caries when the patient was 24, since when he had worn a number of full dentures. Examination revealed a thin man with a tender swelling over the right body of the mandible which was fractured (Fig. la). Investigations Radiographic examination of the mandible (Fig. 3a) showed an extremely thin body but normal rami, and the inferior dental bundle appeared to be exposed for several centimetres on either side. There was a slightly displaced fracture just anterior to the right angle, and the maxillary alveolus showed advanced resorption. A skeletal survey revealed some mild cervical spondylosis which was within normal limits for the patient’s age, as was the rest of the skeleton. Haematology and serum chemistry results were within normal limits and a biopsy taken from the fracture site at operation was reported as consistent with a resolving inflammatory process; there was no evidence of angiomatosis. (Received
1 Present address: Department London, WCIE 6JD.
19 November
1975; accepted
of Oral Surgery, University
13 January 1976) College Hospital,
Mortimer
Market,
ATROPHY
OF
THE
MANDIBLE
FIGS 1 and 2. 1. Anterior views; and 2. Profile views: (a) Pre-operative. (b) Three months completion of surgery. (c) Three years after completion of surgery.
157
after
hfanagemen t Treatment was directed towards the promotion of rapid healing of the fracture, improving facial contour and minimising the danger of further fracture of the very thin mandible. Under general anaesthesia, upper and lower Gunning splints were wired into position to stabilise the mandible. The right body of the mandible was exposed via an extended submandibular incision; a bone graft taken from the left iliac crest was placed adjacent to the mandible from angle to chin, and wired into position lateral to the fracture. Bone chips were packed around the graft, and the wound was closed in layers. Post-operative recovery was uneventful, and fixation was removed at six weeks. The patient was persuaded to dispense with the cervical collar. The left side of the mandible was similarly augmented with a bone graft nine months later, this time without the use of fixation. Dentures have been worn satisfactorily ever since.
58
BRITISH
JOURNAL
OF
ORAL
SURGERY
ATROPHY
159
OF THE MANDIBLE
~--_~---~Preoperative
I
I
I
I
Mar:
Sept.
Jan.
72
72
FIG. 4. Graph demonstrating
period of remodelling
73
thickness
of Mandible
I
(average)
I
Feb.
Sept.
74
75
the fate of the bone grafts over a period of three years. After an initial there has been little resorption of the graft on either side.
Progress
A comparison of photographs of the patient taken before and after treatment demonstrates that the facial contour has been improved (Figs 1 and 2). Serial orthopantomographs demonstrate the condition of the mandible over a period of three years (Fig. 3). The thickness of the mandible on these radiographs was measured using the anterior transosseous wire on either side as a marker, and the measurements have been expressed graphically (Fig. 4). Allowing that the orthopantomograph is not precisely reproducible, at least a crude indication of the fate of the bone grafts is obtained. The graph shows that after an initial period of remodelling there has been little resorption of the graft on either side.
Discussion It would seem from a study of the literature that there is some confusion concerning the terms ‘atrophy’ and ‘osteolysis’ when used in relation to the mandible. Thoma (1933, 1951, 1954) used the term ‘complete atrophy’ to describe in three separate publications what was in fact the same case of massive osteolysis. Thoma was quoted by Alty (1963) in his paper ‘Atrophy of the Mandible and Spontaneous Fracture’, and the second case described by Alty could well have been one of osteolysis. Rowe and Killey (1968) in their turn quote both Thoma and Alty, and use the term ‘disappearing bone disease’. El-Mofty (1971) entitled his review article ‘Atrophy of the Mandible (Massive Osteolysis)‘. Massive osteolysis is a rare, destructive disease of bone of unknown aetiology, and with a poor prognosis for treatment. It has also been called phantom bone disease, disappearing bone disease, acute spontaneous resorption of bone, progressive atrophy FIG. 3 (opposite). Serial orthopantomographs showing the condition of the mandible over a period of three years. (a) Pre-operatively. (b) On completion of surgery. (c) Seventeen months after surgery. (d) Three years after surgery.
160
BRITISH
JOURNAL
OF
ORAL
SURGERY
of bone, progressive osteolysis, angiomatosis, haemangiomatosis, lymphangiectasis and Gorham’s disease. In this disease there is resorption of bone which usually commences as a localised lesion, but which may progress to complete destruction, and this appears to be the main distinguishing feature of osteolysis. It may occur in any bone, and may be monostotic or polyostotic. Thirteen cases have been reported with mandibular involvement (Romer, 1924; Thoma, 1933; Steenhuis & Nauta, 1936; Pasnikowski & Grec, 1960; Hampton & Arthur, 1966; Ellis & Adams, 1971; ElMofty, 1971; Cherrick et al., 1972; Malter, 1972; Phillips et al., 1972; Kriens, 1973; Black et al., 1974; Booth & Burke, 1974). Young adults of either sex are most frequently affected, although it may occur in older individuals. Laboratory investigations are usually normal, and the most consistent histopathological finding is the presence of an angiomatous tissue proliferation that replaces bone. Reconstruction of the affected bone using bone grafts is universally unsuccessful, although there is one report of the successful use of a metal implant replacing a femur (Aston, 1958). In mandibular atrophy, numerous aetiological factors have been postulated, including disuse, pressure, periodontal disease, senility and endocrine influences, but the relative importance of these is unknown. Severe atrophy of the mandible presents four main problems: (1) Difficulty in wearing dentures. (2) Pain and altered sensation due to pressure from the lower denture on exposed mental and inferior dental nerves. (3) The appearance of senility that results from loss of vertical dimension. (4) The threat of fracture, either spontaneously or as a result of minor trauma. This patient illustrates an extreme example of mandibular atrophy. The fact that he had been edentulous and had worn dentures for 36 years is probably significant, as alveolar atrophy has been shown to be more rapid when dentures are worn (Campbell, 1960; Jozefowicz, 1970). The rigid cervical collar which was worn for eight years may have produced an element of disuse (Fig. 5). A review of the literature has revealed reports of only six patients in whom a total of eight spontaneous fractures have occurred due to mandibular atrophy (Thoma & Holland, 1951; Holland, 1953; Alty, 1963; Berkenbaum, 1965; Van de Mark et al., 1969). In view of the frequency with which mandibular atrophy occurs, this scarcity of such reports is surprising, and it is likely that such cases have been under-reported. Obwegeser and Sailer (1973) reported five patients, each of whom sustained fracture of a severely atrophic mandible, although none occurred spontaneously. These authors acknowledged the difficulty of treating such fractures, and advocated the use of onlay rib grafts with subsequent vestibuloplasty where indicated. Analysis of the six case reports of spontaneous fracture leads to three main conclusions : (1) Treatment has a high failure rate, with bony union being achieved in only four of the eight fractures (Thoma & Holland, 1951; Alty, 1963; Berkenbaum, 1965; Van de Mark et al., 1969). Of these four successes, two fractures were treated with Gunning splints, one by means of open reduction and transosseous wiring, and the third by open reduction and insertion of a split rib graft. (2) There is a risk of further fracture of an atrophic mandible. In two of the six patients, after treatment of a spontaneous fracture on one side of the mandible, a fracture occurred on the other side (Alty, 1963; Van de Mark et al., 1969). (3) In no case was the appearance of the patient seriously considered, although the average age of the six patients was only 58 years, and three were under 50 years. In conclusion, the use of bilateral iliac bone grafts is a rational approach to the treatment of spontaneous fracture of the severely atrophic mandible in the younger
ATROPHY
OF THE MANDIBLE
161
FIG. 5. Patient wearing the rigid cervical collar which he had used for eight years. This may have produced an element of disuse in the mandible.
patient. This results in more rapid and reliable healing of the fracture, and increases the bulk of the mandible, thereby improving the facial contour and reducing the risk of further fracture. Acknowledgements I have pleasure in expressing my thanks to Mr J. E. Bowerman for permission to publish this paper, and for his help in its preparation. I am also gratefu! to Mr E. Ferrill of the Department of Medical Illustration at Queen Mary’s Hospital for production of the illustrations. References 114, 188. Alty, H. M. (1963). British DentalJournal, Aston, J. N. (1958). Journal of BoneanrlJoint surgery, 40, 514. Berkenbaum, D. (1965). Aciu Stomatologica, 62,403. Black, M. J., Cassisi, N. J. & Biller, H. F. (1974). Archives of Otolaryngology, 100, 314. Booth, D. F. & Burke, C. H. (1974). Journal of Oral Surgery, 32, 787. Campbell, R. L. (1960). Journal of the American Dental Association, 60, 143. Cherrick, H. M., King, 0. H. & Dorsey, J. N. (1972). Journal of Oral Medicine, 27, 67. Ellis, D. J. & Adams, T. 0. (1971). Journal of Oral Surgery, 29, 659. El-Mofty, S. (1971). Oval Surgery, Oral Medicitie and Oval Pathology, 31, 690. Hampton, J. & Arthur, J. F. (1966). British Dental Journal, 120, 538. Holland, D. J. (1953). Dental Survey, 29, 894. Jozefowicz, W. (1970). Journal of Prosthetic Dentistry, 24, 137. Kriens, 0. (1973). Dentomaxillofacial Radiology, 2, 73.
162
BRITISH
JOURNAL
OF ORAL
SURGERY
Malter, I. J. (1972). Journal of the American Dental Association, 85, 148. Obwegeser, H. L. & Sailer, H. F. (1973). Journal of Maxillofacial Surgery, 1,213. Pasnikowski, T. & Grec, S. (1960). Polski Tygodnik Lek, 15, 1277. Phillips, R. M., Bush, 0. B. & Hall, H. D. (1972). Oral Surgery, Oral Medicine and Oral Pathology, 34, 886. Romer, 0. (1924). Die Pathologic der Zahne, p. 135. Berlin: Springer. Rowe, N. L. & Killey, H. C. (1968). Fractures of the Facial Skeleton, 2nd Ed., p. 75: Edinburgh and London: Livingstone. Steenhuis, D. J. & Nauta, J. H. (1936). Roentgenpraxis, 8, 607. Thoma, K. H. (1933). JournaZ of Bone and Joint Surgery, 15, 494. Thoma, K. H. & Holland, D. J. (1951). Oral Surgery, 4, 1477. Thoma, K. H. (1954). Oral Pathology, 4th Ed., p. 704. London: H. Kimpton. Van de Mark, T. B., Weinberg, S., Zosky, J. G. & Cryfe, J. H. (1969). Journal of fhe Canadian Dental Association, 35, 98.
14 (1976) 156-162
ATROPHY OF THE MANDIBLE: RECONSTRUCTION FOLLOWING FRACTURE DAVID R. JAMES,M.B.,
B.Ch., B.D.S., F.D.S.R.C.S.(Eng.)l
Queen Mary’s Hospital, Roehampton, London Summary. A rational approach to the management of spontaneous fracture of the severely atrophic mandible is illustrated by means of a case report, A study of the literature reveals that some confusion exists concerning the terms ‘atrophy’ and ‘osteolysis’, and an attempt is made to distinguish between them.
Introduction Spontaneous fracture of the mandible occurs when the bone is so weakened that it is no longer able to withstand the normal stresses to which it is subjected. This weakening may be due to a specific disease entity within the bone, or to atrophy. There is some confusion in the literature concerning the terms ‘atrophy’ and “osteolysis’, and it is the aim of this paper to distinguish between these two terms, and by means of a case report to propose a rational approach to the management of spontaneous fracture of the atrophic mandible.
Case report A 60-year-old schoolmaster was admitted to hospital in December 1971 with a five-week history of increasing pain and swelling of the right side of his face. There was a past history of cervical spondylosis, and the patient had worn a cervical collar intermittently for eight years. A dental clearance had been performed because of caries when the patient was 24, since when he had worn a number of full dentures. Examination revealed a thin man with a tender swelling over the right body of the mandible which was fractured (Fig. la). Investigations Radiographic examination of the mandible (Fig. 3a) showed an extremely thin body but normal rami, and the inferior dental bundle appeared to be exposed for several centimetres on either side. There was a slightly displaced fracture just anterior to the right angle, and the maxillary alveolus showed advanced resorption. A skeletal survey revealed some mild cervical spondylosis which was within normal limits for the patient’s age, as was the rest of the skeleton. Haematology and serum chemistry results were within normal limits and a biopsy taken from the fracture site at operation was reported as consistent with a resolving inflammatory process; there was no evidence of angiomatosis. (Received
1 Present address: Department London, WCIE 6JD.
19 November
1975; accepted
of Oral Surgery, University
13 January 1976) College Hospital,
Mortimer
Market,
ATROPHY
OF
THE
MANDIBLE
FIGS 1 and 2. 1. Anterior views; and 2. Profile views: (a) Pre-operative. (b) Three months completion of surgery. (c) Three years after completion of surgery.
157
after
hfanagemen t Treatment was directed towards the promotion of rapid healing of the fracture, improving facial contour and minimising the danger of further fracture of the very thin mandible. Under general anaesthesia, upper and lower Gunning splints were wired into position to stabilise the mandible. The right body of the mandible was exposed via an extended submandibular incision; a bone graft taken from the left iliac crest was placed adjacent to the mandible from angle to chin, and wired into position lateral to the fracture. Bone chips were packed around the graft, and the wound was closed in layers. Post-operative recovery was uneventful, and fixation was removed at six weeks. The patient was persuaded to dispense with the cervical collar. The left side of the mandible was similarly augmented with a bone graft nine months later, this time without the use of fixation. Dentures have been worn satisfactorily ever since.
58
BRITISH
JOURNAL
OF
ORAL
SURGERY
ATROPHY
159
OF THE MANDIBLE
~--_~---~Preoperative
I
I
I
I
Mar:
Sept.
Jan.
72
72
FIG. 4. Graph demonstrating
period of remodelling
73
thickness
of Mandible
I
(average)
I
Feb.
Sept.
74
75
the fate of the bone grafts over a period of three years. After an initial there has been little resorption of the graft on either side.
Progress
A comparison of photographs of the patient taken before and after treatment demonstrates that the facial contour has been improved (Figs 1 and 2). Serial orthopantomographs demonstrate the condition of the mandible over a period of three years (Fig. 3). The thickness of the mandible on these radiographs was measured using the anterior transosseous wire on either side as a marker, and the measurements have been expressed graphically (Fig. 4). Allowing that the orthopantomograph is not precisely reproducible, at least a crude indication of the fate of the bone grafts is obtained. The graph shows that after an initial period of remodelling there has been little resorption of the graft on either side.
Discussion It would seem from a study of the literature that there is some confusion concerning the terms ‘atrophy’ and ‘osteolysis’ when used in relation to the mandible. Thoma (1933, 1951, 1954) used the term ‘complete atrophy’ to describe in three separate publications what was in fact the same case of massive osteolysis. Thoma was quoted by Alty (1963) in his paper ‘Atrophy of the Mandible and Spontaneous Fracture’, and the second case described by Alty could well have been one of osteolysis. Rowe and Killey (1968) in their turn quote both Thoma and Alty, and use the term ‘disappearing bone disease’. El-Mofty (1971) entitled his review article ‘Atrophy of the Mandible (Massive Osteolysis)‘. Massive osteolysis is a rare, destructive disease of bone of unknown aetiology, and with a poor prognosis for treatment. It has also been called phantom bone disease, disappearing bone disease, acute spontaneous resorption of bone, progressive atrophy FIG. 3 (opposite). Serial orthopantomographs showing the condition of the mandible over a period of three years. (a) Pre-operatively. (b) On completion of surgery. (c) Seventeen months after surgery. (d) Three years after surgery.
160
BRITISH
JOURNAL
OF
ORAL
SURGERY
of bone, progressive osteolysis, angiomatosis, haemangiomatosis, lymphangiectasis and Gorham’s disease. In this disease there is resorption of bone which usually commences as a localised lesion, but which may progress to complete destruction, and this appears to be the main distinguishing feature of osteolysis. It may occur in any bone, and may be monostotic or polyostotic. Thirteen cases have been reported with mandibular involvement (Romer, 1924; Thoma, 1933; Steenhuis & Nauta, 1936; Pasnikowski & Grec, 1960; Hampton & Arthur, 1966; Ellis & Adams, 1971; ElMofty, 1971; Cherrick et al., 1972; Malter, 1972; Phillips et al., 1972; Kriens, 1973; Black et al., 1974; Booth & Burke, 1974). Young adults of either sex are most frequently affected, although it may occur in older individuals. Laboratory investigations are usually normal, and the most consistent histopathological finding is the presence of an angiomatous tissue proliferation that replaces bone. Reconstruction of the affected bone using bone grafts is universally unsuccessful, although there is one report of the successful use of a metal implant replacing a femur (Aston, 1958). In mandibular atrophy, numerous aetiological factors have been postulated, including disuse, pressure, periodontal disease, senility and endocrine influences, but the relative importance of these is unknown. Severe atrophy of the mandible presents four main problems: (1) Difficulty in wearing dentures. (2) Pain and altered sensation due to pressure from the lower denture on exposed mental and inferior dental nerves. (3) The appearance of senility that results from loss of vertical dimension. (4) The threat of fracture, either spontaneously or as a result of minor trauma. This patient illustrates an extreme example of mandibular atrophy. The fact that he had been edentulous and had worn dentures for 36 years is probably significant, as alveolar atrophy has been shown to be more rapid when dentures are worn (Campbell, 1960; Jozefowicz, 1970). The rigid cervical collar which was worn for eight years may have produced an element of disuse (Fig. 5). A review of the literature has revealed reports of only six patients in whom a total of eight spontaneous fractures have occurred due to mandibular atrophy (Thoma & Holland, 1951; Holland, 1953; Alty, 1963; Berkenbaum, 1965; Van de Mark et al., 1969). In view of the frequency with which mandibular atrophy occurs, this scarcity of such reports is surprising, and it is likely that such cases have been under-reported. Obwegeser and Sailer (1973) reported five patients, each of whom sustained fracture of a severely atrophic mandible, although none occurred spontaneously. These authors acknowledged the difficulty of treating such fractures, and advocated the use of onlay rib grafts with subsequent vestibuloplasty where indicated. Analysis of the six case reports of spontaneous fracture leads to three main conclusions : (1) Treatment has a high failure rate, with bony union being achieved in only four of the eight fractures (Thoma & Holland, 1951; Alty, 1963; Berkenbaum, 1965; Van de Mark et al., 1969). Of these four successes, two fractures were treated with Gunning splints, one by means of open reduction and transosseous wiring, and the third by open reduction and insertion of a split rib graft. (2) There is a risk of further fracture of an atrophic mandible. In two of the six patients, after treatment of a spontaneous fracture on one side of the mandible, a fracture occurred on the other side (Alty, 1963; Van de Mark et al., 1969). (3) In no case was the appearance of the patient seriously considered, although the average age of the six patients was only 58 years, and three were under 50 years. In conclusion, the use of bilateral iliac bone grafts is a rational approach to the treatment of spontaneous fracture of the severely atrophic mandible in the younger
ATROPHY
OF THE MANDIBLE
161
FIG. 5. Patient wearing the rigid cervical collar which he had used for eight years. This may have produced an element of disuse in the mandible.
patient. This results in more rapid and reliable healing of the fracture, and increases the bulk of the mandible, thereby improving the facial contour and reducing the risk of further fracture. Acknowledgements I have pleasure in expressing my thanks to Mr J. E. Bowerman for permission to publish this paper, and for his help in its preparation. I am also gratefu! to Mr E. Ferrill of the Department of Medical Illustration at Queen Mary’s Hospital for production of the illustrations. References 114, 188. Alty, H. M. (1963). British DentalJournal, Aston, J. N. (1958). Journal of BoneanrlJoint surgery, 40, 514. Berkenbaum, D. (1965). Aciu Stomatologica, 62,403. Black, M. J., Cassisi, N. J. & Biller, H. F. (1974). Archives of Otolaryngology, 100, 314. Booth, D. F. & Burke, C. H. (1974). Journal of Oral Surgery, 32, 787. Campbell, R. L. (1960). Journal of the American Dental Association, 60, 143. Cherrick, H. M., King, 0. H. & Dorsey, J. N. (1972). Journal of Oral Medicine, 27, 67. Ellis, D. J. & Adams, T. 0. (1971). Journal of Oral Surgery, 29, 659. El-Mofty, S. (1971). Oval Surgery, Oral Medicitie and Oval Pathology, 31, 690. Hampton, J. & Arthur, J. F. (1966). British Dental Journal, 120, 538. Holland, D. J. (1953). Dental Survey, 29, 894. Jozefowicz, W. (1970). Journal of Prosthetic Dentistry, 24, 137. Kriens, 0. (1973). Dentomaxillofacial Radiology, 2, 73.
162
BRITISH
JOURNAL
OF ORAL
SURGERY
Malter, I. J. (1972). Journal of the American Dental Association, 85, 148. Obwegeser, H. L. & Sailer, H. F. (1973). Journal of Maxillofacial Surgery, 1,213. Pasnikowski, T. & Grec, S. (1960). Polski Tygodnik Lek, 15, 1277. Phillips, R. M., Bush, 0. B. & Hall, H. D. (1972). Oral Surgery, Oral Medicine and Oral Pathology, 34, 886. Romer, 0. (1924). Die Pathologic der Zahne, p. 135. Berlin: Springer. Rowe, N. L. & Killey, H. C. (1968). Fractures of the Facial Skeleton, 2nd Ed., p. 75: Edinburgh and London: Livingstone. Steenhuis, D. J. & Nauta, J. H. (1936). Roentgenpraxis, 8, 607. Thoma, K. H. (1933). JournaZ of Bone and Joint Surgery, 15, 494. Thoma, K. H. & Holland, D. J. (1951). Oral Surgery, 4, 1477. Thoma, K. H. (1954). Oral Pathology, 4th Ed., p. 704. London: H. Kimpton. Van de Mark, T. B., Weinberg, S., Zosky, J. G. & Cryfe, J. H. (1969). Journal of fhe Canadian Dental Association, 35, 98.
