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Congenital epulis of the newborn
British Journal of Oral Surgery (1980) 18,238-243 @ The British Association
0007-117X/80/00350238$02.00
of Oral Surgeons
CONGENITAL EPULIS OF THE NEWBORN R. R. WELBURY, B.D.S. Dental & Oral Surgery Department, Newcastle General Hospital, Westgate Road, Newcastle Upon Tyne, NE4 6BE Summary. Two cases of Congenital Epulis of the Newborn, one treated by surgical removal, the other conservatively, are presented. In both patients the dentition erupted normally and although after five years a residual swelling remains in the patient treated conservatively, it is asymptomatic. It is suggested that the natural history of the congenital epulis is one of regression, and no treatment is required unless the lesion causes feeding or respiratory problems.
Introduction This tumour was first described by Neumann (1871), as a soft tissue growth which may be found on the gingiva of a newborn infant. Since then many cases have been reported but as yet the tissue of origin is not proven. The theories reported by Fuhr and Krogh (1972) in their centennial review of the congenital epulis include fibroblastic, histiocytic, myogenic and neurogenic origins. The tumours are present at birth and are located on either the maxillary or mandibular gingiva in the ratio of 3 : 1. They are usually pink, non-ulcerated and pedunculated, arising on the crest of the alveolar ridge but may be attached by a wide base. They are commonly found in the incisor regions and rarely posteriorly and their size may vary from over a few millimetres to several centimetres in diameter. The lesion is more common in females. Custer and Fust (1952) reviewed the literature and although the congenital epulis resembles the granular cell myoblastoma, they regarded the following histological and clinical features as evidence that they were most likely separate lesions, (1) Uniformity of structure in the congenital epulis is not found in the granular cell myoblastoma. (2) Congenital epulis contains no demonstrable neural components, nor is there hyperplasia of the adjacent squamous epithelium, both of which are present in the granular cell myoblastoma. (3) The prominent vascular component in the congenital epulis is not found in the granular cell myoblastoma. (4) The congenital epulis occurs in newborn infants. Their structure is not precisely reduplicated by granular cell neurofibromas of the gingiva occurring later in life. (5) The typical location of the congenital epulis in the incisor region of the maxilla or mandible is not a matter of chance. (6) There is a higher incidence of epulides in female infants. There is no preferential sex incidence in granular cell myoblastoma. Campbell (1955) although stating that histologically the congenital epulis and granular cell myoblastoma are practically identical, acknowledged that the congenital epulis with its predilection for the gums, especially the incisor region, and its preponderance in female babies, set it apart to some extent. (Received 25 January 1980; accepted 25 February 238
1980)
CONGENITAL
EPULIS
OF THE
NEWBORN
239
O’Brien (1972) felt it necessary to emphasise the points in which the two lesions appeared to differ in his series of experiments. Namely that the pseudoepitheliomatous hyperplasia is characteristic of the myoblastoma, and that the high degree of vascularity in the stroma is characteristic of the congenital epulis. Histologically the congenital epulis is overlaid by gingival mucosa which occasionally may be ulcerated. The tumour mass comprises sheets of large closely packed cells, showing fine abundant eosinophilic cytoplasm and sometimes they may encompass small islands of epithelium. These well defined sheets of cells are separated from the surface epithelium by a zone of connective tissue which, however, does not form a definite capsule. The cells are mostly round, but oval, irregular or elongated strap forms may be present. The nuclei may be centrally or eccentrically placed, and in the large cells they have a clearly defined nucleolus. Neither mitoses nor crossstriations are visible (Shafer et al., 1974) and in between individual cells is a fine collagenous stroma. Many capillaries are seen to traverse the tumour either through all layers, or aggregated in the deeper parts. Although the lesion is benign, removal either under local or general anaesthesia may be necessary because it causes feeding or respiratory problems. Case reports Case 1
A six-day-old female baby was referred because she had a smooth, round, red pedunculated tumour about 1 cm in diameter arising from a pedicle situated on the crest of the mandibular ridge in the cbl region (Fig. 1). There was no sign of ulceration and the tumour did not interfere with the baby’s feeding.
FIG. 1, Case 1. Clinical appearance
six days after birth.
240
BRITISH
JOURNAL
OF ORAL
SURGERY
FIG. 3, Case 1. Photomicrograph showing granularity of tumour cells, and endotheliumlined vascular channels (H&E, x 90). FIG. 2, Case 1. Lesion after removal under general
anaesthetic.
The tumour had been present at birth and had been observed regularly in the interim period without any noticeable enlargement. The baby’s condition was satisfactory after 40 weeks gestation and she had no other abnormalities apart from a small capillary haemangioma over the sacral area. Under general anaesthesia on the eighth day of life a Dexon horizontal mattress suture was placed below the pedicle, the base of the pedicle excised and the suture tied (Fig. 2). Microscopic examination revealed a congenjtal epulis with superficial ulceration present (Fig. 3). The baby was discharged from hospital the following day and was reviewed at regular intervals up to the age of eighteen months. Healing was good without residual scarring and the deciduous teeth erupted normally. There was no sign of recurrence and the child was discharged. Case 2
A one-day-old female baby was found to have a bilobed, smooth, round, red tumour arising from a broad base on the crest of the mandibular ridge in the cbaj region (Fig. 4). The anterior lobe was twice as big as the posterior, and more vascular. The total length of the tumour was about 1 cm, it was not ulcerated and did not interfere with feeding.
CONGENITAL
EPULIS
OF
THE
FIG. 4, Case 2. Clinical appearance
NEWBORN
241
one day after birth.
The tumour had been present since birth. The baby’s condition was satisfactory after an uneventful full-term gestation and normal delivery. The diagnosis of congenital epulis of the newborn was made. The baby was seen regularly and after three weeks the tumour appeared less vascular with no change in size. After a further three weeks its size was reduced, and at five months old, the tumour was firmer and smaller. Similarly at eight months it was smaller and firmer, the posterior lobe having disappeared and a/a erupting normally (Fig. 5). The other teeth erupted normally and at 18 months the tumour was localised to the interdental septa in the ba/ region (Fig. 6). Radiographs taken revealed no bony or dental abnormality. Regular reviews showed the tumour continued to regress. At the most recent examination at age five years, the size of the residual lesion was 3 mm diameter, firm and asymptomatic. Discussion
Histological examination has shown that many specimens have not been entirely excised and yet no recurrence has been reported after excision. Surgery, when necessary, need not be radical, thus minimising the danger of damaging underlying alveolar bone and developing tooth buds (Freedman et a/., 1968).
242
BRITISH
JOURNAL
OF ORAL
FIG. 5, Case 2. Clinical appearance
FIG. 6, Case 2. Clinical appearance
SURGERY
eight months after birth.
18 months after birth.
CONGENITAL
EPULIS
OF THE
243
NEWBORN
However a residual lesion may require local removal at a later date due to its possible deleterious effect on the health of the periodontium in that area. Its presence could lead to an area of localised plaque increase, whereby initiating and perpetuating a periodontal pocket, or it may be subject to toothbrush abrasion which could lead to an increase in its size. Surgery, when and if necessary for these reasons, would be a relatively simple and quick procedure. Campbell (1955) first noted that no reports gave any information on the natural history of the lesion. O’Brien and Pielou (1971) described two patients, each of which had two lesions. The larger anterior ones in each patient were removed and the smaller posterior ones regressed spontaneously, one within five weeks, the other within 13 months. He suggested that the natural history of the congenital epulis is to undergo resolution. Bowe (1974) in presenting one surgically treated case, invited speculation on the natural history of untreated lesions. The congenital epulis does not lend itself easily to a study of its natural history primarily because it frequently causes nursing difficulties necessitating its removal, and also because of its rarity. Campbell (1955) suggested that with an increased awareness of the lesion it may not be as rare as was believed. Some 20 years later this has been borne out. Perhaps with an even greater awareness its natural history will become more evident. Acknowledgements The author would like to express his gratitude to Mr G. H. Steel, for permitting the publication of Case 2 and to Mr P. Bradnum for permitting publication of Case 1 and for his advice in preparing this paper. References Bowe, J. J. (1974). Congenital
Epulis Tumour:
Case Report. Plastic and Reconstrucfive
Surgery,
53, 227.
Campbell, J. A. H. (1955). Congenital Epulis. Journal ofPathology and Bacteriology, 70, 223. Custer, R. F. & Fust, J. A. (1952). Congenital Epulis. American Journal of Clinical Pathology, 22, 1044.
Freedman,
G. L., Hooley, J. R. & Gordon, R. C. (1968). Congenital Epulis in the Newborn. Journal
of Oral Surgery,
26, 61.
Fuhr, A. H. & Krogh, P. H. J. (1972). Congenital
Epulis of the Newborn. Journal
of Oral Surgery,
30, 30.
Neumann, E. (1871). Ein fall von Congenitaler Epulis. Archiv fiir Ohrenheilkunde, 12, 189. O’Brien, F. V. & Pielou, W. D. (1971). Congenital Epulis: Its Natural History. Archives of Disease in Childhood, 46, 559.
O’Brien, F. V. (1972). Granular Cell Lesions of the Mouth. British Journal of Oral Surgery, 10, 89. Shafer, W. G., Hine, M. Ii. & Levy, B. M. (1974). A Text Book of Oral Pathology, 3rd Ed., pp. 182-185. Philadelphia & London: Saunders.
0007-117X/80/00350238$02.00
of Oral Surgeons
CONGENITAL EPULIS OF THE NEWBORN R. R. WELBURY, B.D.S. Dental & Oral Surgery Department, Newcastle General Hospital, Westgate Road, Newcastle Upon Tyne, NE4 6BE Summary. Two cases of Congenital Epulis of the Newborn, one treated by surgical removal, the other conservatively, are presented. In both patients the dentition erupted normally and although after five years a residual swelling remains in the patient treated conservatively, it is asymptomatic. It is suggested that the natural history of the congenital epulis is one of regression, and no treatment is required unless the lesion causes feeding or respiratory problems.
Introduction This tumour was first described by Neumann (1871), as a soft tissue growth which may be found on the gingiva of a newborn infant. Since then many cases have been reported but as yet the tissue of origin is not proven. The theories reported by Fuhr and Krogh (1972) in their centennial review of the congenital epulis include fibroblastic, histiocytic, myogenic and neurogenic origins. The tumours are present at birth and are located on either the maxillary or mandibular gingiva in the ratio of 3 : 1. They are usually pink, non-ulcerated and pedunculated, arising on the crest of the alveolar ridge but may be attached by a wide base. They are commonly found in the incisor regions and rarely posteriorly and their size may vary from over a few millimetres to several centimetres in diameter. The lesion is more common in females. Custer and Fust (1952) reviewed the literature and although the congenital epulis resembles the granular cell myoblastoma, they regarded the following histological and clinical features as evidence that they were most likely separate lesions, (1) Uniformity of structure in the congenital epulis is not found in the granular cell myoblastoma. (2) Congenital epulis contains no demonstrable neural components, nor is there hyperplasia of the adjacent squamous epithelium, both of which are present in the granular cell myoblastoma. (3) The prominent vascular component in the congenital epulis is not found in the granular cell myoblastoma. (4) The congenital epulis occurs in newborn infants. Their structure is not precisely reduplicated by granular cell neurofibromas of the gingiva occurring later in life. (5) The typical location of the congenital epulis in the incisor region of the maxilla or mandible is not a matter of chance. (6) There is a higher incidence of epulides in female infants. There is no preferential sex incidence in granular cell myoblastoma. Campbell (1955) although stating that histologically the congenital epulis and granular cell myoblastoma are practically identical, acknowledged that the congenital epulis with its predilection for the gums, especially the incisor region, and its preponderance in female babies, set it apart to some extent. (Received 25 January 1980; accepted 25 February 238
1980)
CONGENITAL
EPULIS
OF THE
NEWBORN
239
O’Brien (1972) felt it necessary to emphasise the points in which the two lesions appeared to differ in his series of experiments. Namely that the pseudoepitheliomatous hyperplasia is characteristic of the myoblastoma, and that the high degree of vascularity in the stroma is characteristic of the congenital epulis. Histologically the congenital epulis is overlaid by gingival mucosa which occasionally may be ulcerated. The tumour mass comprises sheets of large closely packed cells, showing fine abundant eosinophilic cytoplasm and sometimes they may encompass small islands of epithelium. These well defined sheets of cells are separated from the surface epithelium by a zone of connective tissue which, however, does not form a definite capsule. The cells are mostly round, but oval, irregular or elongated strap forms may be present. The nuclei may be centrally or eccentrically placed, and in the large cells they have a clearly defined nucleolus. Neither mitoses nor crossstriations are visible (Shafer et al., 1974) and in between individual cells is a fine collagenous stroma. Many capillaries are seen to traverse the tumour either through all layers, or aggregated in the deeper parts. Although the lesion is benign, removal either under local or general anaesthesia may be necessary because it causes feeding or respiratory problems. Case reports Case 1
A six-day-old female baby was referred because she had a smooth, round, red pedunculated tumour about 1 cm in diameter arising from a pedicle situated on the crest of the mandibular ridge in the cbl region (Fig. 1). There was no sign of ulceration and the tumour did not interfere with the baby’s feeding.
FIG. 1, Case 1. Clinical appearance
six days after birth.
240
BRITISH
JOURNAL
OF ORAL
SURGERY
FIG. 3, Case 1. Photomicrograph showing granularity of tumour cells, and endotheliumlined vascular channels (H&E, x 90). FIG. 2, Case 1. Lesion after removal under general
anaesthetic.
The tumour had been present at birth and had been observed regularly in the interim period without any noticeable enlargement. The baby’s condition was satisfactory after 40 weeks gestation and she had no other abnormalities apart from a small capillary haemangioma over the sacral area. Under general anaesthesia on the eighth day of life a Dexon horizontal mattress suture was placed below the pedicle, the base of the pedicle excised and the suture tied (Fig. 2). Microscopic examination revealed a congenjtal epulis with superficial ulceration present (Fig. 3). The baby was discharged from hospital the following day and was reviewed at regular intervals up to the age of eighteen months. Healing was good without residual scarring and the deciduous teeth erupted normally. There was no sign of recurrence and the child was discharged. Case 2
A one-day-old female baby was found to have a bilobed, smooth, round, red tumour arising from a broad base on the crest of the mandibular ridge in the cbaj region (Fig. 4). The anterior lobe was twice as big as the posterior, and more vascular. The total length of the tumour was about 1 cm, it was not ulcerated and did not interfere with feeding.
CONGENITAL
EPULIS
OF
THE
FIG. 4, Case 2. Clinical appearance
NEWBORN
241
one day after birth.
The tumour had been present since birth. The baby’s condition was satisfactory after an uneventful full-term gestation and normal delivery. The diagnosis of congenital epulis of the newborn was made. The baby was seen regularly and after three weeks the tumour appeared less vascular with no change in size. After a further three weeks its size was reduced, and at five months old, the tumour was firmer and smaller. Similarly at eight months it was smaller and firmer, the posterior lobe having disappeared and a/a erupting normally (Fig. 5). The other teeth erupted normally and at 18 months the tumour was localised to the interdental septa in the ba/ region (Fig. 6). Radiographs taken revealed no bony or dental abnormality. Regular reviews showed the tumour continued to regress. At the most recent examination at age five years, the size of the residual lesion was 3 mm diameter, firm and asymptomatic. Discussion
Histological examination has shown that many specimens have not been entirely excised and yet no recurrence has been reported after excision. Surgery, when necessary, need not be radical, thus minimising the danger of damaging underlying alveolar bone and developing tooth buds (Freedman et a/., 1968).
242
BRITISH
JOURNAL
OF ORAL
FIG. 5, Case 2. Clinical appearance
FIG. 6, Case 2. Clinical appearance
SURGERY
eight months after birth.
18 months after birth.
CONGENITAL
EPULIS
OF THE
243
NEWBORN
However a residual lesion may require local removal at a later date due to its possible deleterious effect on the health of the periodontium in that area. Its presence could lead to an area of localised plaque increase, whereby initiating and perpetuating a periodontal pocket, or it may be subject to toothbrush abrasion which could lead to an increase in its size. Surgery, when and if necessary for these reasons, would be a relatively simple and quick procedure. Campbell (1955) first noted that no reports gave any information on the natural history of the lesion. O’Brien and Pielou (1971) described two patients, each of which had two lesions. The larger anterior ones in each patient were removed and the smaller posterior ones regressed spontaneously, one within five weeks, the other within 13 months. He suggested that the natural history of the congenital epulis is to undergo resolution. Bowe (1974) in presenting one surgically treated case, invited speculation on the natural history of untreated lesions. The congenital epulis does not lend itself easily to a study of its natural history primarily because it frequently causes nursing difficulties necessitating its removal, and also because of its rarity. Campbell (1955) suggested that with an increased awareness of the lesion it may not be as rare as was believed. Some 20 years later this has been borne out. Perhaps with an even greater awareness its natural history will become more evident. Acknowledgements The author would like to express his gratitude to Mr G. H. Steel, for permitting the publication of Case 2 and to Mr P. Bradnum for permitting publication of Case 1 and for his advice in preparing this paper. References Bowe, J. J. (1974). Congenital
Epulis Tumour:
Case Report. Plastic and Reconstrucfive
Surgery,
53, 227.
Campbell, J. A. H. (1955). Congenital Epulis. Journal ofPathology and Bacteriology, 70, 223. Custer, R. F. & Fust, J. A. (1952). Congenital Epulis. American Journal of Clinical Pathology, 22, 1044.
Freedman,
G. L., Hooley, J. R. & Gordon, R. C. (1968). Congenital Epulis in the Newborn. Journal
of Oral Surgery,
26, 61.
Fuhr, A. H. & Krogh, P. H. J. (1972). Congenital
Epulis of the Newborn. Journal
of Oral Surgery,
30, 30.
Neumann, E. (1871). Ein fall von Congenitaler Epulis. Archiv fiir Ohrenheilkunde, 12, 189. O’Brien, F. V. & Pielou, W. D. (1971). Congenital Epulis: Its Natural History. Archives of Disease in Childhood, 46, 559.
O’Brien, F. V. (1972). Granular Cell Lesions of the Mouth. British Journal of Oral Surgery, 10, 89. Shafer, W. G., Hine, M. Ii. & Levy, B. M. (1974). A Text Book of Oral Pathology, 3rd Ed., pp. 182-185. Philadelphia & London: Saunders.