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Lymphangioma: Anlage proliferation induced by chronic irritation
Oral pathology GENERAL SECTION
Lymphangioma: Anlage proliferation induced by chronic irritation Jerome Lichtenstein, D.D.S.,* Elmhurst, N. Y., Williana D.D.S.,“” Elmhurst, N. Y., and Arthur Post, D.D.X.,*“* Washington Heights, N. Y.
K. Kopp,
A
ngiomas are tumors composed of blood vessels or lymph channels. The lymphangioma is the analogue in the lymphatic system of the hemangioma of the vascular system. The most widely accepted classification of lymphangiomas is as follows 1. Capillary lymphangioma 2. Cavernous lymphangioma 3. Cystic hygroma (Fig. 1). The lymphangioma is a benign tumor. In the majority of casesit is present at birth or arises soon after. According to Watson and M:cCarthy,l 95 per cent of these tumors were seen to arise before the age of 10. In a similar study by Nix,l 71 per cent of t.he tumors were noted before the age of 15. ETIOLOGY
AND
CLINICAL
FINDINGS
The cause of the lymphangioma is controversial, but it is generally accepted that these tumors are hamartomas. A hamartoma is a localized error in the composition of the tissue elements of an organ. This error may be in abnormal quantity, abnormal structure, or degree of maturation of the component tissue.? During the development of a structure, the vascular component often may be exaggerated or overemphasized. As the infant develops, these lesions become macroscopically visible. Both sexes are equally affected by this tumor. In the oral cavity, lymphangiomas are seen to occur principally on the tongue. Less frequently involved sites are the buccal mucosa and the lips; the palate and gingivne arc rarely involved. Lymphangiomas may occur as papillary *Chief Resident, Oral Surgery, Elmhurst~ Hospital. **Intern, Oral Surgery, Elmhurst Hospital ; at prawns t Assistxnt Hospital (New York), Elmhurst Herviccs Unit. ‘*“Attending Oral Surgeon, Elmhurst Hospital.
Kt~sitlrnt,
Mount
Hiuni
363
lesions Or as diffuse cnlarg:~mcnts. 1~ 1IIV la1 tcr ilM;illc+o. il’ the: tongue is inT-olved, S~CCC~ and mast,ica t ion a I’(~ illlI):lil*(4 l~ca~~sc~ 0 II t hc resnltant macroglossia. Lymphangiomas form lesions with ~a11 \-csicles of :I ycIlowish or purplish hue covering the surface of the tlmlor. If allowed to progress to sufficient size, the tumor may drain spontaneously and become ulcerated and painful. At, this time the tumor will be seen to undergo intermittent alterations in size. Scarring 01’ the surface mncosa may be the end result of this p~ocess.:~ HISTOLOGY
The lymphangioma is composed of n&works of’ endothelium-lined lymph channels that can be differentiated from vascular channels by the absence of blood elements. A scant pink debris from the protein of the lymph fluid and sometimes lymphocytes and macrophaqs may bc seen in these spaces4 The sept,a of these spaces are cithcr reduced to thin strands of acellular connective tissue or are thicker and participate in its proliferation.” The endothelial cells lining these spaces sometimes hypertrophy and assume the appearance of glandular cpithclinm.’ TREATMENT
AND
PROGNOSIS
The treatment ol’ choice is cscision, since the lymphangioma is extremely radioresistant and insensitive to sclcrosing agents, such ilS sodium ps.vlliat,c 01 sodillm morrhuatc.‘, ‘$ The prognosis following complctc IWWIYI~ is csc~~llcnt-.l+‘ollowing incomplete removal, liowevc~r, rcciirrcnw is often obscrvc4.” CASE 011
Elmhurst the laid The
REPORT 17, 1963, :L i-yc~l~ld N,~~Iw Iwy was ~.~~ft~rrwl 10 111,. Owl (iener;tl IIospitnl for cxitrnitdion ant1 tro:ltrllc:Iit of il “papillary mucosa of the lower lip. patient ~-as a healthy, alert, at1c1 responsive child. He hat1 lweu
Ocl-.
Surgc~ry C’liniv projwtioll” rwt~iving
rouliue
at on
Volume Number
Lynzphangionza
20 3
Fig.
2’. Note
Fig.
3. Close-up
lesion
in direct
of papillary
relationship
mass with
to fractured
sessile
maxillary
central
365
incisor.
base.
dental treat,ment at the public school dental clinic which referred him. The past medical history was noncontributory. Intraoral examination revealed a normally developed dentition and supporting structures. The maxillary left central incisor had sustained a Class II Ellis fracture when the patient \vas 7 years 1 month of age s-ml had been present and untreated for 5 months since. The lip lesion was seen to be in direct relation with the fractured central incisor (Fig. 2). As the history was being elicited, the child could be seen to maneuver the lower lip in such a manner as to bring the mass into constant interplay with the fractured tooth. Roth the child and his mother maintained that this mass was not present before the maxillary central incisor was fractured and that the lesion began after the tooth was first fractured. The lesion was a papillary, sessile mass, approximately 1 cm. in diameter and raised 0.5 cm. The overlying mucosa appeared bluish gray, with several gray-white stippled areas (Fig. 3). A provisional diagnosis of fibroepithelial polyp (irritation fibroma) was made. Total surgical removal of the lesion was indicated. Operation
Under was made underlying
local anesthesia (bilateral about the lesion. Sharp tissue. No tumor tissue
mental foramen nerve blocks), an elliptical incision dissection was used to free the specimen from the was seen to invade the subcutaneous part of the lip.
The
pathologist
returned
a diagnosis
of 1!.111p)1an~io1rra.
DISCUSSION
This cast was of interest not brcansc a lymphangioma occurred on the lip, but rather because of the significance of the fact that certain irritational factors hare been positirtly associated with the stimulation and subsequent growth of an anlagc of a lymphangioma. Tn the documentation of this case, a direct cause-and-effect relationship was established in the neoplasia of this tumor entity. Whether this malformation would have proliferated at some future time in the absenceof the fractured tooth is subject to conjecture’. REFERENCES
1. Shafer W. G., Hine, X. K., and Levy, 1s. M.: Texthook of Oral Pathology, Philadelphia, 195X 6. B. Saunders Company, pp. 110-111. 2. Beriier, J. L.: Maaagement of Oral Ijisease, ed. 2, St. T,ouis, 959, The (1. X’. Mosb) Company, pp. 74-75, 758-761. 3. Thoma, K. H.: Oral Surgery, ed. 4. St. Louis, 1963, The (‘. Y. Nosby Company, pp. 943-944. 4. Robbins, S. L.: Textbook of Pathology, ed. 2, Philatlelphia, 1962, TV. 1% Saunders Company, p. 540. 5. Ewing, J.: Neoplastic Ijiseases, ed. 4, Philadelphia, 1940~ \2’. R. Saunders Company, p. 263. 6. Ward, 0. E., and Hendrick, J. IV.: l)iagnosis and Treatment of Tumors of the Head and Neck, Baltimore, 1950, Williams & Wilkins (‘ompany, pp. 97-98.
Lymphangioma: Anlage proliferation induced by chronic irritation Jerome Lichtenstein, D.D.S.,* Elmhurst, N. Y., Williana D.D.S.,“” Elmhurst, N. Y., and Arthur Post, D.D.X.,*“* Washington Heights, N. Y.
K. Kopp,
A
ngiomas are tumors composed of blood vessels or lymph channels. The lymphangioma is the analogue in the lymphatic system of the hemangioma of the vascular system. The most widely accepted classification of lymphangiomas is as follows 1. Capillary lymphangioma 2. Cavernous lymphangioma 3. Cystic hygroma (Fig. 1). The lymphangioma is a benign tumor. In the majority of casesit is present at birth or arises soon after. According to Watson and M:cCarthy,l 95 per cent of these tumors were seen to arise before the age of 10. In a similar study by Nix,l 71 per cent of t.he tumors were noted before the age of 15. ETIOLOGY
AND
CLINICAL
FINDINGS
The cause of the lymphangioma is controversial, but it is generally accepted that these tumors are hamartomas. A hamartoma is a localized error in the composition of the tissue elements of an organ. This error may be in abnormal quantity, abnormal structure, or degree of maturation of the component tissue.? During the development of a structure, the vascular component often may be exaggerated or overemphasized. As the infant develops, these lesions become macroscopically visible. Both sexes are equally affected by this tumor. In the oral cavity, lymphangiomas are seen to occur principally on the tongue. Less frequently involved sites are the buccal mucosa and the lips; the palate and gingivne arc rarely involved. Lymphangiomas may occur as papillary *Chief Resident, Oral Surgery, Elmhurst~ Hospital. **Intern, Oral Surgery, Elmhurst Hospital ; at prawns t Assistxnt Hospital (New York), Elmhurst Herviccs Unit. ‘*“Attending Oral Surgeon, Elmhurst Hospital.
Kt~sitlrnt,
Mount
Hiuni
363
lesions Or as diffuse cnlarg:~mcnts. 1~ 1IIV la1 tcr ilM;illc+o. il’ the: tongue is inT-olved, S~CCC~ and mast,ica t ion a I’(~ illlI):lil*(4 l~ca~~sc~ 0 II t hc resnltant macroglossia. Lymphangiomas form lesions with ~a11 \-csicles of :I ycIlowish or purplish hue covering the surface of the tlmlor. If allowed to progress to sufficient size, the tumor may drain spontaneously and become ulcerated and painful. At, this time the tumor will be seen to undergo intermittent alterations in size. Scarring 01’ the surface mncosa may be the end result of this p~ocess.:~ HISTOLOGY
The lymphangioma is composed of n&works of’ endothelium-lined lymph channels that can be differentiated from vascular channels by the absence of blood elements. A scant pink debris from the protein of the lymph fluid and sometimes lymphocytes and macrophaqs may bc seen in these spaces4 The sept,a of these spaces are cithcr reduced to thin strands of acellular connective tissue or are thicker and participate in its proliferation.” The endothelial cells lining these spaces sometimes hypertrophy and assume the appearance of glandular cpithclinm.’ TREATMENT
AND
PROGNOSIS
The treatment ol’ choice is cscision, since the lymphangioma is extremely radioresistant and insensitive to sclcrosing agents, such ilS sodium ps.vlliat,c 01 sodillm morrhuatc.‘, ‘$ The prognosis following complctc IWWIYI~ is csc~~llcnt-.l+‘ollowing incomplete removal, liowevc~r, rcciirrcnw is often obscrvc4.” CASE 011
Elmhurst the laid The
REPORT 17, 1963, :L i-yc~l~ld N,~~Iw Iwy was ~.~~ft~rrwl 10 111,. Owl (iener;tl IIospitnl for cxitrnitdion ant1 tro:ltrllc:Iit of il “papillary mucosa of the lower lip. patient ~-as a healthy, alert, at1c1 responsive child. He hat1 lweu
Ocl-.
Surgc~ry C’liniv projwtioll” rwt~iving
rouliue
at on
Volume Number
Lynzphangionza
20 3
Fig.
2’. Note
Fig.
3. Close-up
lesion
in direct
of papillary
relationship
mass with
to fractured
sessile
maxillary
central
365
incisor.
base.
dental treat,ment at the public school dental clinic which referred him. The past medical history was noncontributory. Intraoral examination revealed a normally developed dentition and supporting structures. The maxillary left central incisor had sustained a Class II Ellis fracture when the patient \vas 7 years 1 month of age s-ml had been present and untreated for 5 months since. The lip lesion was seen to be in direct relation with the fractured central incisor (Fig. 2). As the history was being elicited, the child could be seen to maneuver the lower lip in such a manner as to bring the mass into constant interplay with the fractured tooth. Roth the child and his mother maintained that this mass was not present before the maxillary central incisor was fractured and that the lesion began after the tooth was first fractured. The lesion was a papillary, sessile mass, approximately 1 cm. in diameter and raised 0.5 cm. The overlying mucosa appeared bluish gray, with several gray-white stippled areas (Fig. 3). A provisional diagnosis of fibroepithelial polyp (irritation fibroma) was made. Total surgical removal of the lesion was indicated. Operation
Under was made underlying
local anesthesia (bilateral about the lesion. Sharp tissue. No tumor tissue
mental foramen nerve blocks), an elliptical incision dissection was used to free the specimen from the was seen to invade the subcutaneous part of the lip.
The
pathologist
returned
a diagnosis
of 1!.111p)1an~io1rra.
DISCUSSION
This cast was of interest not brcansc a lymphangioma occurred on the lip, but rather because of the significance of the fact that certain irritational factors hare been positirtly associated with the stimulation and subsequent growth of an anlagc of a lymphangioma. Tn the documentation of this case, a direct cause-and-effect relationship was established in the neoplasia of this tumor entity. Whether this malformation would have proliferated at some future time in the absenceof the fractured tooth is subject to conjecture’. REFERENCES
1. Shafer W. G., Hine, X. K., and Levy, 1s. M.: Texthook of Oral Pathology, Philadelphia, 195X 6. B. Saunders Company, pp. 110-111. 2. Beriier, J. L.: Maaagement of Oral Ijisease, ed. 2, St. T,ouis, 959, The (1. X’. Mosb) Company, pp. 74-75, 758-761. 3. Thoma, K. H.: Oral Surgery, ed. 4. St. Louis, 1963, The (‘. Y. Nosby Company, pp. 943-944. 4. Robbins, S. L.: Textbook of Pathology, ed. 2, Philatlelphia, 1962, TV. 1% Saunders Company, p. 540. 5. Ewing, J.: Neoplastic Ijiseases, ed. 4, Philadelphia, 1940~ \2’. R. Saunders Company, p. 263. 6. Ward, 0. E., and Hendrick, J. IV.: l)iagnosis and Treatment of Tumors of the Head and Neck, Baltimore, 1950, Williams & Wilkins (‘ompany, pp. 97-98.