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Cavernous hemangioma of the tongue
Cavernous hemangioma of the tongue Report
of
Q case
Richard G. Lyon, D.M.D.,* Robert A. Amon, D.D.X.,“” and Reubin Seldin, D.D.S.,““” Brooklyn, N. Y. KINGS
COUNTY
HOSPITAL
H
emangioma is a common lesion of the oral cavity. There are, however, many unsettled questions concerning its management and therapy. Hemangiomas are now generally considered to be developmental vascular malformations, rather than true neoplasms. They can occur in any area of the oral cavity, at any age, and there is no sexual or racial predisposition. Their clinical appearance varies considerably in size, in depth of involvement, and in degree of deformity. Shklar and Meye+ describe five types of hemangioma on the basis of their histopathology : the capilla,ry type, the cavernous type, the mixed type, the cellular type, and the sclerosing type. They point out, however, that histologic classification has little value in relation to clinical behavior or to prognosis. Lampe and LaTourette5 point out that lesions that enlarge in the first few months of life and cease growing before the age of one year usually regress spontaneously very early in life, (the involuting type), whereas those lesions that do not grow actively early in life will usually not regress (the noninvoluting type). They further state that for lesions of the involuting type a program of “watch-and-wait” expectant therapy is preferable unless clinical symptoms, such as infection or active bleeding, make definitive treatment mandatory. The literature suggests numerous methods of therapy for hemangiomas; these include observation, surgical removal, electrodesiccation, electroeoagulation, injection of sclerosing agents, cryotherapy, and irradiation. Thus, the decision as to the management of hemangiomas is a difficult and often confusing one. Numerous sclerosing agents are documented in the literatures and have been *Intern, Oral Surgery Service. **Resident, Oral Surgery Service. ***Director of Dentistry. 540
usccl successfully in the management of intraoral hemangiomas : Sotradecol, boiling water, alcohol, dilute hydrochloric acid, quinine compounds, soaps (sodium morrhuate), and others. The action of sclerosing agents is based on the fact that they are both tissue irritants and thrombogenic a.gents. 1 Thry provoke a moderately severe inflammatory rrspo~rsc with subsequent fibrosis anti obliteration of vascular channels. Ba.urmash and Mandell suggest that sclerosing agents are indicated (I) for large, deeply pen&rating lesions; (2) for poorly outlined vascular tumors; (3) for lesions in juxtaposition to vital struct,ures; (1) for patients reluctant to undergo surgical treatment; and (5) for those patients with medical contraindi~iltiOllS to surgical
intervention.
(‘ontl.aintlicaticrrlv to the use of sclcrosing agents include srcontlaril>~ infrctetl lesions, uncontrolled diabetes, and a history of allergy to the agents. The relative limitation of inforrnation in the literature dealing with t,he use of radiation therapy for the treatment of intraoral hemangiomas is significant. Damage to bone, growth centers, and developing tooth buds is a well-documented complication as,wciated with intraoral radiation therapy. This is a particularly significant hazard when radiation therapy is administered to patients in the younger age groups, and the resultant damage may not be evident until years later. Consequently, radiation should not be used to treat intraoral hemangiomas unless adequate protection can be given adjacent structures. This is often a tlifficult problem for the radiologist, and the risk of permanent damage frcquently yields to altcrnatc forms of therapy. Shklar and Meyer’s state that the use of radiation in treating oral mucosal lesions is to be “deplored.” They state further that, because of the potential hazard of radiation, it should never be considered for use in the treatment of asymptomatic lesions. Medina7 reports that he never uses radiation therapy in patients over 3 >-cars of age because of the tcndenc; of these lesions to become more fibrous in nat,urc, and he says that, he generally ~)~t’c~t*silltC’U)t~tC inMliod8 in treating intraoral hcniangiomas. I~iltliwtioll has been
Pig. I. I’veop”atire
view of lesion at time of patient’s admission to Ilospital.
O.S., O.M. & O.P. April, 1968
nsrtl snccwsfally in the in trcatin, 01intraoral as well as extraoral hemangiomas,” ant1 it is still used in carefully selected cases. Methocls employtl in the management of hemangiomas shonld cleprrd 011 the character ilIlt pathologic type of the lesion, as well as its size, location, ant1 growth pattern, and the age and sex of the patient. l)ilSt
CASE REPORT A S-year-old, wzll-developed, well-nourished Negro girl was admitted to the hospital with the chief complaints of tlysphagia, slight pain, and slight bleeding on eating or touch, associated with a raised lesion on the right postrrolateral aspwt of the tlorsum of the tongue (Fig. 1). The history of the lesion was obtained from the child’s stepmother, who stated that to the best of her knowledge the lesion had been present from birth, was slowly enlarging, and only recently had become symptomatic. She stated that 3 months prior to admission the child had been seen at a local clinic and had been told that the lesion was “enlarged muscle” and should be watched closely. The child was placed on iron therapy for anemia 3 weeks prior to admission. One day prior to admission she returned to the same clinic for followup treatment and was immrdiatclg referred to the hospital for diagnosis and treatment of the tongue lesion. The past medical history, review of systems, and physical examination findings were within normal limits, except for the previously diagnosed anemia and the tongue lesion. Oral examination revealed the normal mixed dentition of a g-year-old child, with moderate caries present. The mucous membranes were slightly pale in color but otherwise normal on examination. There was a 4.5 by 2 cm. raised, firm, multinodular lesion on the right posterolateral aspect of the dorsum of the tongue. The surface was nodular and varied in color from bluish to dark red. There were occasional areas of ulceration and slight bleeding on provocation. So pulsation was palpable, and only slight blanching was observed on compression. The child was an obvious introvert, and only minimal speech impediment was elicited by observation and history. The impression at this time was that the lesion was a hemangioma. Other possible included lymphangioma, granular-cell myoblastoma, diagnoses entertained leiomyoma, rhabdomyoma, neurofibroma, granuloma pyogenicum, and their malignant counterparts. and an opthalmology consultation Radiologic examination of the skull was negative, disclosed no pathosis. Thus, the possibility of Sturgc-Weber syndrome was eliminated. Chest x-ray findings were also negative. The results of routine blood tests were within normal limits, and the previously diagnosed anemia was now under control. Blood specimens were also drawn for typing and crossmatching. Eight days after admission, following appropriate consultation with the Head and Neck Service, the patient was taken to the operating room for an excisional biopsy. Suitable blood Under Pcntothal sodium and nitrous-oxide-oxygen-halothane replacement was available. anesthesia with nasotracheal intubation, the patient was prepared and draped in the usual manner. The pharynx was packed with 1 inch wet vaginal gauze. A 1-O black silk suture was placed through the anterior part of the tongue for traction. Three deep 3-O black silk sutures were passed from left t,o right at intervals along the lesion for control of massive hemorrhage. One superficial anteroposterior silk suture was passed lateral to the lesion on both sides for exposure and access to deeper portions of the defect after excision. Such access was an aid in the clamping and tying of the bleeders encountered, both deep and superficial. The lesion was aspirated with a 20.gauge needle, with no resulting blood or fluid. An elliptical incision was made to include the entire lesion with a 0.5 cm. margin of normal tissue. Suture ligatures of 3-O chromic gut were placed for hemostasis after clamping of all bleeders. Three areas of doubtful margins were again excised, and all tissue specimens were The lesion appeared rather superficial, well confined, submitted for microscopic diagnosis. and without invasion of deeper muscle layers.
Volume 25 Number 4
Cavemzous hema~lgiorna
Pig. P. Postoperative view 10 days after surgical procedure.
fs’ig. J.
(iross specimen at time of operation.
of
twague
543
544
I,!yo~, Amon, und, Xeldin
Fig. 4. Photomicrograph containing blood elements.
OS., 0.x 8; 0.1’. April,1968
of lesion demonstrating cavernous spaces lined with cudotheliun+ Note fibrous nature of relatively dense stroma.
patient’s age and the consensus impression that the lesion could be better managed surgically. Second, members of the Tumor Board to whom the cast was presented felt that the lesion should be managed surgically with the possibility of a frozen se&ion to be kept in mind. Third, sclerosing of the lesion was considered thoroughly. It was thought, however, that the youthful age of the patient would limit cooperation and that, because of the large size of the lesion, the possibility of secondary edema was great and might result in airway problems necessitating tracheostomy. Edema was also consider4 to be a possible sequel to surgical management, but it was believed that satisfactory hrmostasis could be obtained, limiting such a complication to a minimum. It was also thought that surgical treatment might have to be considered, even after sclerosing of t,he lesion, if satisfactory resolution was not obtained. In retrospect, the postoperative results seemed to justify our decision to treat this particular hemangioma surgically. We would like to thank Dr. Rosen of the Pathology Department of Kings County IIospital for his contribut,ion of the photomicrographs and Dr. Gilbert Eiseman of the Plastic Surgery Department for his assistance at surgery and for his eontribution of the clinical photographs. REFERENCES
The Nonsurgical Treatment of Hemangioma with 1. Baurmash, H., and Mandel, L.: Sotradecol, ORAL SURG., ORN, MED. & ORAL PATH. 16: 777-782, 1963. Report of Four Cases, ORAL SITRG., 2. Christensen, R,. W.: Treatment of Oral Hemangiomas; ORAL MED. & ORAL PATH. 12: 912.921, 1959. 3. Erich, J. B.: Benign and Malignant Tumors of .JRWS, Surg. Clin. North America 32: 1161, 1952. 4. Geshickter, C. F., and Keasby, L. E.: Tumors of Blood Vessels, Am. J. Cancer 23: 568-591, 1935. 5. Lampe, I., and LaTourettc, H. B.: Managment of Hemagiomas in Infants, Pediat. Clin. North America 6: 511-528, 1959. 6. Medina, A., and Alexander, L.: Radium Management of Cutaneous Hemdngioma, J. Nat. M. A. 58: 6-11, 1966. 7. Medina, A.: Personal interview, June, 1966.
8. Olech, IL: Hemangioma of the Cheek Involving the Opening of Rtcnsen’s Duet Treated by Tnjection of a Hclcrosiny Solution, ORAr, HURG., ORAL MEI). $ ORAL PATH. 16: 641-644, 1963. 3. Peahler, G. E.: The Treatment of Hrwangioma Chiefly 1)~ Tnadiation, AwlI Dermat. & Ryph. 72: 425437, 1955. 10. Rhklar, G., and Meyer, I.: Vascular Tumors cf tile AI\Ioutlt and Jaws, ORAL SCRG., ORAL MED. & ORAL PATIt. 19: 335.358, 1965. Il. Stockdale, CL R.: Peripheral Angioma a,nd Their Treatment \Vith Sclerosing Solution, ORAL SURG., ORAL IMED. $ ORAL PATH. 12: 1157-1162, 1959.
of
Q case
Richard G. Lyon, D.M.D.,* Robert A. Amon, D.D.X.,“” and Reubin Seldin, D.D.S.,““” Brooklyn, N. Y. KINGS
COUNTY
HOSPITAL
H
emangioma is a common lesion of the oral cavity. There are, however, many unsettled questions concerning its management and therapy. Hemangiomas are now generally considered to be developmental vascular malformations, rather than true neoplasms. They can occur in any area of the oral cavity, at any age, and there is no sexual or racial predisposition. Their clinical appearance varies considerably in size, in depth of involvement, and in degree of deformity. Shklar and Meye+ describe five types of hemangioma on the basis of their histopathology : the capilla,ry type, the cavernous type, the mixed type, the cellular type, and the sclerosing type. They point out, however, that histologic classification has little value in relation to clinical behavior or to prognosis. Lampe and LaTourette5 point out that lesions that enlarge in the first few months of life and cease growing before the age of one year usually regress spontaneously very early in life, (the involuting type), whereas those lesions that do not grow actively early in life will usually not regress (the noninvoluting type). They further state that for lesions of the involuting type a program of “watch-and-wait” expectant therapy is preferable unless clinical symptoms, such as infection or active bleeding, make definitive treatment mandatory. The literature suggests numerous methods of therapy for hemangiomas; these include observation, surgical removal, electrodesiccation, electroeoagulation, injection of sclerosing agents, cryotherapy, and irradiation. Thus, the decision as to the management of hemangiomas is a difficult and often confusing one. Numerous sclerosing agents are documented in the literatures and have been *Intern, Oral Surgery Service. **Resident, Oral Surgery Service. ***Director of Dentistry. 540
usccl successfully in the management of intraoral hemangiomas : Sotradecol, boiling water, alcohol, dilute hydrochloric acid, quinine compounds, soaps (sodium morrhuate), and others. The action of sclerosing agents is based on the fact that they are both tissue irritants and thrombogenic a.gents. 1 Thry provoke a moderately severe inflammatory rrspo~rsc with subsequent fibrosis anti obliteration of vascular channels. Ba.urmash and Mandell suggest that sclerosing agents are indicated (I) for large, deeply pen&rating lesions; (2) for poorly outlined vascular tumors; (3) for lesions in juxtaposition to vital struct,ures; (1) for patients reluctant to undergo surgical treatment; and (5) for those patients with medical contraindi~iltiOllS to surgical
intervention.
(‘ontl.aintlicaticrrlv to the use of sclcrosing agents include srcontlaril>~ infrctetl lesions, uncontrolled diabetes, and a history of allergy to the agents. The relative limitation of inforrnation in the literature dealing with t,he use of radiation therapy for the treatment of intraoral hemangiomas is significant. Damage to bone, growth centers, and developing tooth buds is a well-documented complication as,wciated with intraoral radiation therapy. This is a particularly significant hazard when radiation therapy is administered to patients in the younger age groups, and the resultant damage may not be evident until years later. Consequently, radiation should not be used to treat intraoral hemangiomas unless adequate protection can be given adjacent structures. This is often a tlifficult problem for the radiologist, and the risk of permanent damage frcquently yields to altcrnatc forms of therapy. Shklar and Meyer’s state that the use of radiation in treating oral mucosal lesions is to be “deplored.” They state further that, because of the potential hazard of radiation, it should never be considered for use in the treatment of asymptomatic lesions. Medina7 reports that he never uses radiation therapy in patients over 3 >-cars of age because of the tcndenc; of these lesions to become more fibrous in nat,urc, and he says that, he generally ~)~t’c~t*silltC’U)t~tC inMliod8 in treating intraoral hcniangiomas. I~iltliwtioll has been
Pig. I. I’veop”atire
view of lesion at time of patient’s admission to Ilospital.
O.S., O.M. & O.P. April, 1968
nsrtl snccwsfally in the in trcatin, 01intraoral as well as extraoral hemangiomas,” ant1 it is still used in carefully selected cases. Methocls employtl in the management of hemangiomas shonld cleprrd 011 the character ilIlt pathologic type of the lesion, as well as its size, location, ant1 growth pattern, and the age and sex of the patient. l)ilSt
CASE REPORT A S-year-old, wzll-developed, well-nourished Negro girl was admitted to the hospital with the chief complaints of tlysphagia, slight pain, and slight bleeding on eating or touch, associated with a raised lesion on the right postrrolateral aspwt of the tlorsum of the tongue (Fig. 1). The history of the lesion was obtained from the child’s stepmother, who stated that to the best of her knowledge the lesion had been present from birth, was slowly enlarging, and only recently had become symptomatic. She stated that 3 months prior to admission the child had been seen at a local clinic and had been told that the lesion was “enlarged muscle” and should be watched closely. The child was placed on iron therapy for anemia 3 weeks prior to admission. One day prior to admission she returned to the same clinic for followup treatment and was immrdiatclg referred to the hospital for diagnosis and treatment of the tongue lesion. The past medical history, review of systems, and physical examination findings were within normal limits, except for the previously diagnosed anemia and the tongue lesion. Oral examination revealed the normal mixed dentition of a g-year-old child, with moderate caries present. The mucous membranes were slightly pale in color but otherwise normal on examination. There was a 4.5 by 2 cm. raised, firm, multinodular lesion on the right posterolateral aspect of the dorsum of the tongue. The surface was nodular and varied in color from bluish to dark red. There were occasional areas of ulceration and slight bleeding on provocation. So pulsation was palpable, and only slight blanching was observed on compression. The child was an obvious introvert, and only minimal speech impediment was elicited by observation and history. The impression at this time was that the lesion was a hemangioma. Other possible included lymphangioma, granular-cell myoblastoma, diagnoses entertained leiomyoma, rhabdomyoma, neurofibroma, granuloma pyogenicum, and their malignant counterparts. and an opthalmology consultation Radiologic examination of the skull was negative, disclosed no pathosis. Thus, the possibility of Sturgc-Weber syndrome was eliminated. Chest x-ray findings were also negative. The results of routine blood tests were within normal limits, and the previously diagnosed anemia was now under control. Blood specimens were also drawn for typing and crossmatching. Eight days after admission, following appropriate consultation with the Head and Neck Service, the patient was taken to the operating room for an excisional biopsy. Suitable blood Under Pcntothal sodium and nitrous-oxide-oxygen-halothane replacement was available. anesthesia with nasotracheal intubation, the patient was prepared and draped in the usual manner. The pharynx was packed with 1 inch wet vaginal gauze. A 1-O black silk suture was placed through the anterior part of the tongue for traction. Three deep 3-O black silk sutures were passed from left t,o right at intervals along the lesion for control of massive hemorrhage. One superficial anteroposterior silk suture was passed lateral to the lesion on both sides for exposure and access to deeper portions of the defect after excision. Such access was an aid in the clamping and tying of the bleeders encountered, both deep and superficial. The lesion was aspirated with a 20.gauge needle, with no resulting blood or fluid. An elliptical incision was made to include the entire lesion with a 0.5 cm. margin of normal tissue. Suture ligatures of 3-O chromic gut were placed for hemostasis after clamping of all bleeders. Three areas of doubtful margins were again excised, and all tissue specimens were The lesion appeared rather superficial, well confined, submitted for microscopic diagnosis. and without invasion of deeper muscle layers.
Volume 25 Number 4
Cavemzous hema~lgiorna
Pig. P. Postoperative view 10 days after surgical procedure.
fs’ig. J.
(iross specimen at time of operation.
of
twague
543
544
I,!yo~, Amon, und, Xeldin
Fig. 4. Photomicrograph containing blood elements.
OS., 0.x 8; 0.1’. April,1968
of lesion demonstrating cavernous spaces lined with cudotheliun+ Note fibrous nature of relatively dense stroma.
patient’s age and the consensus impression that the lesion could be better managed surgically. Second, members of the Tumor Board to whom the cast was presented felt that the lesion should be managed surgically with the possibility of a frozen se&ion to be kept in mind. Third, sclerosing of the lesion was considered thoroughly. It was thought, however, that the youthful age of the patient would limit cooperation and that, because of the large size of the lesion, the possibility of secondary edema was great and might result in airway problems necessitating tracheostomy. Edema was also consider4 to be a possible sequel to surgical management, but it was believed that satisfactory hrmostasis could be obtained, limiting such a complication to a minimum. It was also thought that surgical treatment might have to be considered, even after sclerosing of t,he lesion, if satisfactory resolution was not obtained. In retrospect, the postoperative results seemed to justify our decision to treat this particular hemangioma surgically. We would like to thank Dr. Rosen of the Pathology Department of Kings County IIospital for his contribut,ion of the photomicrographs and Dr. Gilbert Eiseman of the Plastic Surgery Department for his assistance at surgery and for his eontribution of the clinical photographs. REFERENCES
The Nonsurgical Treatment of Hemangioma with 1. Baurmash, H., and Mandel, L.: Sotradecol, ORAL SURG., ORN, MED. & ORAL PATH. 16: 777-782, 1963. Report of Four Cases, ORAL SITRG., 2. Christensen, R,. W.: Treatment of Oral Hemangiomas; ORAL MED. & ORAL PATH. 12: 912.921, 1959. 3. Erich, J. B.: Benign and Malignant Tumors of .JRWS, Surg. Clin. North America 32: 1161, 1952. 4. Geshickter, C. F., and Keasby, L. E.: Tumors of Blood Vessels, Am. J. Cancer 23: 568-591, 1935. 5. Lampe, I., and LaTourettc, H. B.: Managment of Hemagiomas in Infants, Pediat. Clin. North America 6: 511-528, 1959. 6. Medina, A., and Alexander, L.: Radium Management of Cutaneous Hemdngioma, J. Nat. M. A. 58: 6-11, 1966. 7. Medina, A.: Personal interview, June, 1966.
8. Olech, IL: Hemangioma of the Cheek Involving the Opening of Rtcnsen’s Duet Treated by Tnjection of a Hclcrosiny Solution, ORAr, HURG., ORAL MEI). $ ORAL PATH. 16: 641-644, 1963. 3. Peahler, G. E.: The Treatment of Hrwangioma Chiefly 1)~ Tnadiation, AwlI Dermat. & Ryph. 72: 425437, 1955. 10. Rhklar, G., and Meyer, I.: Vascular Tumors cf tile AI\Ioutlt and Jaws, ORAL SCRG., ORAL MED. & ORAL PATIt. 19: 335.358, 1965. Il. Stockdale, CL R.: Peripheral Angioma a,nd Their Treatment \Vith Sclerosing Solution, ORAL SURG., ORAL IMED. $ ORAL PATH. 12: 1157-1162, 1959.