Volume Number

Intraosseous

41 5

10. Shklar, G.: Observations, Reprint

reqwests

Oral Arch.

Lesions Dermatol.

of Erythema 92: 495-500,

angiolipoma

Multiforme: 1965.

of mandible

Histologic

and

637

Histochemical

to :

Dr. John Giunta Department of Oral Pathology Tufts University School of Dental 1 Kneeland St. Boston, Mass. 02111

Medicine

Intraosseous angiolipoma of the mandible Hans W. Polte, Lieutenant Colonel, USAF (DC),* Stanley C. Kolodny, Colonel, USAF (DC),** and Southern P. Hooker, Colonel, USAF (DC)+++ WILFORD

HALL

USAF

MEDICAL

CENTER,

LACKLAND

AIR

FORCE

BASE,

TEXAS

A case of intraosseous angiolipoma, one of the rarest benign tumors of bone, is reported. This tumor represents an example of an intraosseous neoplasm consisting of both blood vessels and fat. To our knowledge, such a tumor of the mandible has not been reported previously.

A

n intraosseous lipoma is a relatively uncommon entity. Dahlin reported the incidence of bony lipomas to be 1 per 1,000 cases.3s l5 Several types of lipomas involving bone have been recognized. One type is known as lipomatosis, a condition arising in the soft tissue and invading the bone secondarily.‘, I3 A second type is the periosteal (parosteal) lipoma, which is uncommon but not rare and has its primary origin associated with the periosteum.ly 2* I3 The true intramedullary or intraosseous lipoma is one of the rarest benign tumors of bone. Less than a dozen cases have been documented in the literature. Probably the first intraosseous lipoma was described by Brault, in 1868, involving the diaphysis of the femur. Several have since been rep0rted.l” Since then, intraosseous lipomas have been reported in the fibula,14* l6 the tibia,4, I) the ulna and frontal bone,l the calcaneus,2 the humerusI and the rib.6 In a series of 3,829 *USAF **Director ***Staff

Hospital Chanute. of Dental Services, Wilford oral pathologist, Wilford Hall

Hall USAF Medical Center. USAF Medical Center.

638

Polte, Kolodny,

and Hooker

Oral May,

surg. 1976

examinations of the vertebrae, .Junghannsl” found intraosseous lipomatous areas Present in 0.6 per cent of all the cases. He never encountered these in patients under 50 years of age and suggested that these findings represented fatty degeneration of the bone marrow. In some isolated cast>s, areas IvitIl concentrated vascularity within the lipoma were noted which were thought to be variants of angioma5.12 In a series of autopsies, Makrycostas found four cases of vertebral lipomas, three of which were associated with angiomas of vertebrae.” All patients were over 70 years of age. Again these lesions were thought to represent variants of angiomas or possibly intermediate stages of forming hemangiomas.2, 12 Another intraosseous lesion consisting of blood vessels and fat has been described by Krepp.’ He reported a hemangiolipoma in the tibia of a 30-year-old woman. However, it is not clear whether this lesion represented a true neoplasm or a reactive lesion associated with preexisting trauma.15 In the dental literature, Oringer,ll in 1948, described a lipoma in the body of the mandible associated with the mesial and distal roots of a mandibular second molar. In 1969, Johnson5 reported an intraosseous lipoma in the ramus of the mandible, which was stated to have some increased vascularity. A third lesion of the mandible, diagnosed as a fibrolipoma, was reported by Newn1an.l” The present case of an angiolipoma is an esamplc of an intraosseous neoplasm consisting of both blood vessels and fat. To our knowledge, an example of a true intramedullary angiolipoma of the mandible has not been reported previously. CASE REPORT A 39-year-old Latin American man was first seen at Randolph Air Force Base, Texas, with the chief complaint of hypesthesia of the left lower lip and chin of one week’s duration. There was apparently no previous history of trauma to the mandible. Radiographs demonstrated bilateral radiolucencies of the body of the mandible. The patient was referred to the Oral Surgery Service at Wilford Hall USAF Medical Center for evaluation and treatment. The patient, an aircraft mechanic, had a 22-year history of active duty with world-wide assignments in the USAF. He had had the usual childhood diseases, without sequelae. To his knowledge, he had no history of drug sensitivity or allergies. The patient’s father had carcinoma of tho lungs and tuberculosis. Review

of systems

Except for chronic sinusitis and degenerative disc disease of the cervical spine, the patient claimed to be in good health. On Aug. 28, 1972, he had undergone a discectomy of C5-6 and C6-7, with anterior cervical fusion at both levels. A bone graft from the right iliac crest was utilized. Physical examination revealed a slightly obese male in no acute distress, alert, cooperative, and oriented in all spheres. Except for minimal limitation of motion of the neck, the examination of the head, eyes, ears, nose, and throat yielded findings that mere within normal limits. There was hypesthesia of the lower left lip; however, the patient was able to perceive painful stimuli. No inflammation, ulceration, or tenderness of the intraoral soft tissues was noted. The dentition was in good repair. There was no expansion of the cortical bone of the mandible. All mandibular teeth responded normally to the vitalometer. There was no mobility or extrusion of the mandibular premolars or molars. auscultation over the radiolucent lesions bilaterally revealed absence of bruits. Attempts at bilateral aspiration of the mandibular first molar areas were unsuccessful. Neurologic examination revealed that, grossly, the cranial nerves were intact. Deep tendon reflexes were +2 throughout, with the exception of the left biceps and left triceps, retlexes of

Volume Number

Idmosseous

41 5

Fig.

1. Bilateral

radiolucencies

in body

of mandible

angiolipoma

noted

on panoramic

of nmdible

639

radiograph.

which were diminished. Numbness of the left thumb and first two fingers was noted. The findings of the remainder of the general physical examination were within normal limits. Laboratory data, including complete blood count, urinalysis, glucose, blood urea nitrogen, electrolytes, alkaline phosphatase, SGOT, and LDH, were within normal limits. The VDRL was nonreactive, and Bence-Jones protein was negative. Radiographs showed bilateral radiolucent lesions in the body of the mandible (Fig. 1). The left mandibular lesion had a well-circumscribed appearance, with scalloping of the upper border between the molars and the premolars. It extended from the cuspid to the mesial root of the third molar and inferiorly slightly below the inferior alveolar canal (Fig. 2). The right mandibular lesion extended from the root apex of the second premolar to the second molar, having a less defined demarcation at its posterior extent (Fig. 3). Posteroanterior and lateral chest radiographs, as well as a survey of the long bones, were within normal limits. Treatment

The patient was admitted to Wilford Hall USAF Medical Center on July 30, 1973. On the following day, under general anesthesia, he underwent exploration of the bilateral radiolucent lesions of the mandible via an intraoral approach. A mucoperiosteal flap was reflected to expose the lateral aspect of the body of the angle of the left mandibIe. The overlying cortical bone was found to be intact. There was no evidence of periosteal reaction, with neither expansion nor erosion of the cortical bone. A 1.5 by 2.0 cm. window was cut in the cortical plate just below the apices of the premolars and molars. Immediately upon removal of the cortical plate, a pink, myxedematous, and extremely hemorrhagic soft mass was encountered within the medullary cavity. Under continuous aspiration, the friable, jellylike mass was removed iu segments with curettes. The neurovascular bundle was encountered and carefully dissected free of the mass. No epithelial lining or fibrous capsule was visualized. An iodoform gauze pack was placed into the cavity in order to assure hemostasis. The mucoperiosteal flap was repositioned and sutured with 4-O black silk. In a similar fashion, the right mandibular lesion was explored. It was found to be an osteoporotic bone marrow defect. Specimens from the right and left sides of the mandihle were submitted for histopathologic evaluation. The estimated loss of blood was 600 c.c., none of which was replaced. The procedure was tolerated well, and the patient’s postoperative course was uneventful. On the basis of the operative findings, the left mandibular lesion was considered to be either a myxoma or a lipoma. The right mandibular lesion was thought to be an osteoporotic hone marrow defect. The specimen obtained from the right mandible consisted of three fragments of firm, irregular tissue, reddish brown in color, which consisted of trabecular bone that contained normal marrow elements. This was consistent with the diagnosis of an osteoporotic bone marrow defect. The specimen from the left mandible consisted, grossly, of multiple fragments of yellowish tan, fatty-appearing tissue, with some small fragments of bone. Microscopic examination

640

Polte,

Kolodny,

Fig. 2. Lateral lesion with scalloped

Fig.

Oral May,

and Hooker

oblique view appearance

3. Close-up

of left mandible of upper border.

view

of right

demonstrating

mandibular

well-circumscribed

radiolucent

Surg. 1976

radiolucent

lesion.

of the left mandibular lesion revealed a mass of mature fat cells, with numerous arterioles terspersed throughout the tissue. Several of the larger vessels showed thrombus formation, varying amounts of chronic inflammation present within the vessels. The final diagnosis intraosseous angiolipoma (Figs. 4 and 5).

inwith was

DISCUSSION

Despite the abundance of adipose connective tissue in bone marrow, lipomas of bone, particularly intramedullary ones, are extremely rare.3l I1 The intraosseous lipoma is a benign, slow-growing tumor consisting of a massof mature fat cells. When the vascular component within these tumors is a prominent feature, they are considered to be angiolipomas.lz, I7

Volume Number

41 5

Iwtraosseous

angiolipoma

of mandible

Fig. 4. Numerous arterioles interspersed throughout mass of mature fat cells. Some thrombus formation. (Magnification, x25.) Fig. 5. Section of left mandibular tumor demonstrating mature fat cells surrounding sels. (Magnification, x100.)

641

vessels

show

ves-

The cause of these neoplasms is uncertain. Clinically, the intraosseous lipoma may be asymptomatic, may cause swelling, or may show neurologic signsI such as hypesthesia or paresthesia, as was manifested in this case. Radiographically, the lesion usually appears as a well-circumscribed radiolucency within the bone.13 Grossly, this tumor presented as an extremely hemorrhagic, friable, jellylike, yellow, lobulated mass with a distinctive greasy appearance. Microscopic studies reveal numerous blood vessels interspersed throughout a mass of mature fat cells. No specific data are available as to definitive treatment of intraosseous

642

Polte, Kolodny,

Fig. filling

6. Panoramic with bone.

and Hooker

radiograph

of mandible

eight

months

postoperatively

showing

defects

lipomas. The presumption is that conservative curettage with the complete removal of the lipomatous mass is indicated. 3, 5, I5 Follow-up of the present case at eight months showed radiographic evidence of bony regeneration bilaterally (Fig. 6). SUMMARY

Described is a case of intraosseous angiolipoma of the left mandible with an osteoporotic bone marrow defect of the right mandible in a 39-year-old man. Treatment of the angiolipoma consisted of conservative removal of the lipomatous mass. A search of the literature has revealed no previously reported case of an intramedullary angiolipoma. REFERENCES 1. Caruolo,

f :

4. 5. 6. 7. 8. 9. 10. 11. 12. 13. 14. 15. 16.

J. E., and Dahlin, D. C. : Lipoma Involving Bone and Simulating Malignant Bone Tumor: Report of Case, Proc. Staff Meet. Mayo Clin. 28: 361, 1953. of Case, Am. J. Clin. Pathol. 25: 1050, 1955. Child, P. E. : Lipoma of the 0s Calcis : Report Dahlin, D. C.: Lipoma and Liposarcoma. In Bone Tumors, ed. 2, Springfield, Ill., 1967, Charles C Thomas Publisher, pp. 110-113. Dickson, A. B., Ayres, TV. W., Mason, M. W., and Miller, W. R.: Lipoma of Boue of Intraosseous Origin, J. Bone Joint Surg. [Am.] 33: 257, 1951. Johnson, E. C. : Intraosseous Lipoma: Report of Case, J. Oral Surg. 27: 8681969. Kawakami, A. : Intraosseous Lipoma of Rib (in Japanese), Jap. J. Thoracic Surg. 19: 561, 1966. Krepp, 8.: iiher ein Knochenhiimangio-Lipom, Zentralbl. Chir. 90: 1674, 1965. Lichtenstein, L. : Bone Tumors, ed. 4, St. Louis, 1972, The C. V. Moshy Company, pp. 330. 334. Mueller, M. C., and Robbins, J. L.: Intramedullary Lipoma of Bone: Report of Case, J. Bone Joint Surg. [Am.] 42: 517, 1960. Newman, C. W.: Fibrolipoma of the Mandible: Report of Case, J. Oral Surg. 15: 257, 1957. Oringer, M. J.: Lipoma of the Mandible, ORAL SURG. 1: 1134, 194% Riissle, R.: Spezielle Pathologic des Skeletts und seiner Teile. In Henke, F., and Lubarsch, 0.: Handbuch der speziellen pathologischen Anatomie und Histologie, Bd. 9, Abd. 5, S 50. 55, Berlin, 1944, Julius Springer. Skinner, G. G., and Fraser, R. G.: Medullarv - Lipoma of Bone, J. Can. Assoc. Radiol. 8: 19, 1957. Lipoma of Bone, Cancer 10: 1151, 1957. Smith, W. E., and Fienberg, R.: Intraosseous Spjut, H. J., Dorfman, H., Fechner, R., and Ackerman, L. V.: Tumors or Tumor-like Lesions of Marrow Origin. In Tumors of Bone and Cartilage, 2nd series, No. 5, Washington D. C., 1971, Armed Forces Institute of Pathology, pp. 197-198. Wehwig, G.: Lipom des Knochenmarks, Centralbl. f. allg. Path. u. path. Anat. 21: 243, 1910.

Volume Number

Sweat

41 5

gland

carcinoma

17. Greer, R. O., and Richardson, J. F.: The Nature of Lipomas and Their Oral Cavity: Review and Report of Cases, J. Oral Surg. 36: 551, 1973.

of lips

Significance

643 in the

Reprint requests to : Colonel Stanley C. Kolodny, USAF (DC) Wilford Hall USAF Medical Center Box 369446, CMR 8 Lackland Air Force Base, Texas 78236

Sweat gland carcinoma of the lips Suleyman Gulme-rt, D.D.S., M.S.D.,” and Peter A. Pullo~, D.D.S., M.S., M.Ed.,“* St. Louis, MO. PATHOLOGY

DEPARTMENT,

WASHIKGTON

UXIVERSITY

SCHOOL

OF

DENTAL

MEDICINE

Sweat gland carcinomas are extremely rare tumors with a fully malignant potential. Two cases of primary sweat gland carcinoma of the lips are presented. Because of the unique nature of this lesion, the histopathology and biologic behavior are discussed, and the pertinent literature is reviewed.

S

weat gland carcinomas are uncommon tumors that arise from the eccrine and apocrine sweat glands of the skin. Because they are uncommon lesions, sweat gland carcinomas may not be recognized early in the course of their development. Histologic diagnosis of sweat gland carcinomas is difficult becauseof the rarity of the lesions and also because of the variation in their microscopic patterns. The histologic features of malignancy depend upon violation of the tumor capsule with subsequent invasion of the adjacent tissues, Direct extension and lymphatic metastasis are the usual course of clinical behavior. Two casesof sweat gland carcinoma of the lip are presented in order to alert the clinician and the pathologist to the nature of this malignant neoplasm. CASE CASE

REPORTS 1

A 35-year-old University School

*Assistant **Professor

Caucasian woman was seen by a physician at Barnes Hospital, of Medicine, because of a slightly indurated and raised lesion,

Professor. and Chairman.

Washington 2 by 1 cm.

Intraosseous angiolipoma of the mandible.

A case of intraosseous angiolipoma, one of the rarest benign tumors of bone, is reported. This tumor represents an example of an intraosseous neoplasm...
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