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Hemangioma of the maxilla
Hemangioma of the maxilla
Vincent S. Pailadino,* M D, East Meadow, N Y , and Arthur E. Danziger,f DDS, New York In patients with hemangioma, severe hemorrhage, both fatal and nonfatal, is an attendant hazard in surgical interven tion of the lesion itself or in exodontia. Successful results, however, were achieved by surgical removal of a cavernous type hemangioma of the maxilla in a 50-yearold woman. No exceptional bleeding oc curred during surgery; there were no complications or sequelae postoperatively. In addition to the case report, a review of the literature, a discussion of the diag noses and nomenclature of vascular lesions found in the literature and a working classification of vascular lesions are pre sented. Hemangiomas of the jawbones rarely are reported. The possibility of severe hemor rhage after surgical intervention or tooth extraction makes these lesions potentially dangerous. Experience in successful treat ment of a patient with a hemangioma, cavernous type, prompts the report of a case and a review of the literature. REVIEW OF LITERATURE
In surveying the literature, we sought chiefly those reports of cases in which the
lesion arose in or was limited to jawbones and was labeled “hemangioma” or “an gioma,” mostly interpreted as cavernous or capillary varieties. Thus, for example, recent reports of aneurysmal bone cysts,1 so-called hemangioameloblastoma2,3 and varieties of aneurysms4,5 were excluded. Also eliminated is a case reported by Stafne,® who, in a discussion of “roent genograms in diagnosis of tumors of the jaws,” shows a roentgenogram (his Fig. 6) of an “angioma of the mandible;” no details are given. Smith,7 in his review of the literature and report of a new case, brought the total of reported cases, by 1959, to 29, 10 in which the lesion was in the max illa. Many of the cases in his review were not well documented, since, as Smith states, “Many of these cases were only referred to. . . .” His chart indicates three instances of fatal hemorrhage associated with tooth extraction (Siegmund and Weber,8 Broderick and Round9 and Stoy10) and two instances of severe, nonfatal hemorrhage associated with ex traction (Broderick and Round® and Brodsky11) . Severe hemorrhage associated with surgical intervention is described by Kamphues12 and by Smith in his own report of a case. Of the cases listed by Smith, we call attention to severe, non-
Palladino and Danziger: H E M A N G IO M A O F M A N D IB L E • 637
fatal hemorrhage associated with therapy in Smith’s case no. 25 (Villa and Limchayseng13) and his case no. 26 (Battersby14). The latter case was not docu mented histologically but was assumed by Battersby to represent a hemangioma. Also, we add the case recently reported by Dibble and Whelan,15 who diagnosed a patient’s mandibular lesion as a “circoid aneurysm,” which they considered a variant of cavernous hemangioma. Severe hemorrhage occurred after extraction and necessitated removal of much of the man dible. Dibble and Whelan also refer to a case reported by Kroh16 in which autopsy showed a cavernous hemangioma of the mandible. Thus, of 31 cases reported in the literature, four involved fatal hemor rhage after extraction, three involved severe, nonfatal hemorrhage associated with extraction and four reports recorded severe, nonfatal hemorrhage associated with surgical treatment of the lesion. Al though it is possible that hemangiomas with fatal or near fatal hemorrhage tend to find their way to the literature, the frequency and severity of hemorrhage is nonetheless impressive. Interesting examples of vascular lesions that are not restricted to jawbones but which involve adjacent structures as well are presented in the discussion that fol lows. Jorgensen17 (1936) reported a cavern ous hemangioma of the maxilla that rep resented maxillary involvement by a large “nevus angiomatosus” of much of the skin of the left side of the face, gin giva and hard and soft palate. Fairly copious bleeding continued for 24 hours after extraction. Cadenat18 (1941) reports a lesion he considered to consist of vascular adipose tissue (questionably “angiolipoma” ) in volving much of the left temporal apo neurosis and part of the mandible. Bower and others19 (1942), in an ar ticle titled “Arteriovenous Angioma of the Mandible and Retina with Pro nounced Hematemesis and Epistaxis,” describe what may represent separate le
sions of mandible and retina. A loose tooth was associated with bleeding. Al though no photomicrographs are in cluded, their diagnosis should likely be accepted. The determination of the precise num ber of hemangiomas reported in the lit erature and firm evaluation of many of the diagnoses established on the basis of pathologic findings are impossible for the following reasons: 1. Case reports, at times, are incom plete and contain little discussion of pathologic findings and inadequate to no photomicrographs. Thus, examples of non-neoplastic vascular lesions may be reported as “hemangioma.” A variety of nonvascular lesions may be similarly misdiagnosed. 2. The relative scarcity of reports of diagnoses determined by pathologic find ings of other than “capillary hemangi oma” or “cavernous hemangioma” sug gests that more unusual hemangiomas are overlooked. 3. Variable interpretations of nomen clature of vascular lesions and common references to less than authoritative sources make evaluation of some pub lished cases difficult, particularly when descriptions of pathologic findings and photomicrographs are scanty, inadequate or absent. An interesting report of a case, “Primary Hemangiosarcoma of the Jaw,”20 illustrates the problem encoun tered in evaluating more unusual lesions. This paper presents but two photomicro graphs of only fair quality. These photo micrographs suggest a diagnosis of malig nant hemangioendothelioma to us. The term “hemangiosarcoma” is considered too inclusive by the present authors for all but infrequent and generally debat able lesions; more precise terminology usually is possible. The case reported by Williams and Gilbert21 (hemangioma of the mandible) may well represent he mangioendothelioma, benign juvenile type. Smith’s7 interpretation of the con dition reported by Stoy10 as “capillary hemangioma” is not supported by Stoy’s
438 • J. A M E R . DENT. A SSN .: Vol. 70, March 1965
Fig. I • a: Preoperative roentgenogram showing radiolucent area separating right central and lateral incisors, b-d: Roentgenograms taken 3, 7 and 49 months after surgery, respectively
description or by the single photomicro graph of the lesion, which showed large, thick-walled vascular spaces. WORKING CLASSIFICATION OF VASCULAR LESIONS
Mesenchymoma (only partially vascu lar)36 Angiofibroma (generally “juvenile na sal angiofibroma” ) Granuloma pyogenicum (questionably neoplastic)
II. Neoplasia of debatable, possibly vascu lar, origin The foregoing reasons for questioning the Ewing’s tumor (“endothelioma” )37'39 accuracy of the diagnoses of some of the “Adamantinoma” of long bones40 (ma lesions reported in the literature as “he lignant angioblastoma41)
mangioma” prom pt inclusion here of one author’s (V. S. P.) working classification III. “Vascular” lesions, probably non of vascular lesions as used in this labora neoplastic Aneurysmal bone cyst42,43 tory. References are cited for the more Cystic hygroma (may mimic lymphanunusual or debatable lesions, and they are giectasis and lymphangioma) generally authoritative. Aneurysm (some are considered con
genital) Congenital anomalies (these include I. Neoplastic or very probably neoplastic others in Group III) vascular lesions Hamartoma (may mimic mesenchy Hemangioma, capillary type moma)— often in combination with Hemangioma, cavernous type other components, such as adipose Hemangioma, mixed capillary and tissue cavernous type Pregnancy “tumor” Fibrosing (hyalinizing) hemangioma Lymphangiectasis (example: Milroy’s (not identical to so-called “sclerosing disease) (may mimic cystic hygro hemangioma” of Gross and Wolma) bach22) Telangiectasis (includes “spider” forms) Hemangioendothelioma, benign juven Sclerosing hemangioma (of question ile type (benign hemangioendotheli able vascular origin)22 (“dermato oma23) fibroma,” “xanthoma” )44 Glomus tumor24,25 “Hemangiomatosis” (example: LindauAngiomyoma von Hippel disease) Lymphangioma Angiokeratoma (includes Fabry’s dis Hemangiopericytoma (both benign and ease) malignant clinical behavior are de scribed with identical histology J*-28 Lymphangiopericytoma20' 80 REPORT OF CASE Lymphangiosarcoma (postradical mas tectomy)31 Hemangioendothelioma (malignant )32 Roentgenographic examination of a 50Kaposi’s sarcoma33“35 year-old patient revealed a radiolucent
Palladino and Danziger: H E M A N G I O M A O F M A N D IB L E • 639
Fig. 2 • Left: Low-power photomicrograph of al most entire specimen showing discrete periphery and uniformly distributed vascular channels. Larg est dimension is 1 cm. Top right: Congested•vas cular spaces lined by unexceptional endothelium and separated by small to moderate amounts of smooth muscle. Bottom right: Flat to mildly plump endothelium lining congested vascular spaces
area in the anterior portion of the max illa lying between roots of the maxillary right central and lateral incisors. • Several months before surgery, the patient had noticed that the maxillary right central incisor was being displaced inferiorly and labially. She consulted her dentist who, on a roentgenogram, noticed a pea-sized radio lucent area between the roots of the aforementioned teeth (Fig. la ). The pa tient was referred to one of the authors (A. E. D.) for surgery. Present Illness
• Infiltration anesthesia was in duced with a 2 per cent solution of butethamine hydrochloride, and the patient was prepared and draped in the usual manner in the office.
Surgery
A “half-moon” incision was made on the labial gingiva over the right maxil lary incisors and cuspids. The flap was reflected, exposing the labial plate of the maxilla. By use of bone drills and rongeur
640 • J. A M E R . DENT. A SSN .: Vol. 70, March 1965
forceps, a window was created in the bone, exposing what appeared to be a rubbery, discrete, encapsulated mass. The mass was easily removed from its bony crypt by use of curets; a smooth-walled cavity in the bone remained. There was no exceptional bleeding at the time of surgery. The flap then was returned to position with no. 000 interrupted silk su tures, and the patient was discharged. The patient’s postoperative course progressed without complications and without sequelae. Postoperative roent genograms taken 3, 7 and 49 months after surgery show normal bone healing (Fig. lb -d ). The patient had no com plaints in reference to the lesion when last seen 49 months postoperatively. Pathologist’s Report • Grossly, the speci men consisted of a lobular to granular, rubbery, oval, tan-brown, discrete mass measuring 1.1 by 1.0 by 0.7 cm. On cross section, the lesion was spongy and graypurple. (The entire specimen was sub mitted in four sections.) Microscopically, the lesion had nu merous oval to mildly undulating con gested vascular channels, most of which measured about 1,000 ¡x. in greatest di mension (Fig. 2, left). The vessel walls varied from a slender ribbon composed of one to two smooth muscle cells to oc casional broad bands of smooth muscle (Fig. 2, top and bottom right). The en dothelial cells were flat to slightly plump (Fig. 2, bottom right). The intervascular tissue consisted almost entirely of smooth muscle, much of which was sepa rated by basophilic vacuolated stroma. The periphery was sharply delimited and consisted of a narrow rim of fibrous tis sue and smooth muscle that blended with the perivascular and intervascular stroma. Diagnosis • The diagnosis of the lesion was established as hemangioma, cavern ous type.
ADDENDUM
During the time this paper was being processed for publication, an additional four instances of central hemangioma were reported by Lund and Dahlin.45 Two of these lesions were in the maxilla, and two were in the mandible. PO Box 175
From th e d e p a rtm e n ts o f p a th o lo g y and c lin ic a l la b o ra to rie s and d iv is io n o f o ra l surgery, M e a d o w b ro o k H o s p ita l, East M eadow , N e w Y ork 11554. * D ire c to r o f p a th o lo g y a n d c lin ic a l la b o ra to rie s , M e a d o w b ro o k H o s p ita l; le c tu re r on o ra l surgery (p o s t g ra d u a te ), N ew Y ork U n iversity, New York. fA s s is ta n t a tte n d in g ora ! surgeon, M e a d o w b ro o k H o s p ita l. 1. Vianna, M . R. A n eurysm al bone cyst in th e m a x illa : re p o r t o f case. J . O ra l S u rg., Anesth. & H o sp. D. Serv. 20:432 S e pt. 1962. 2. V illa , V. G . H e m a n g io m a o f the cavernous ty p e associated w ith a m e lo b la s to m a {a m e lo b la s tic h e m a n g i o m a ). J . O ra l Surg., A n esth. & H osp. D. Serv. 18:429 Sept. I960. 3. O liv e r, R. T., M cKenna, W . F., and Shafer, W . G . H e m a n g io -a m e lo b la s to m a : re p o rt o f case. J . O ra l Surg., Anesth. & H osp. D. Serv. 19:245 M ay 1961. 4. C o ok, T. J ., and Z b a r, M . J . A rte rio v e n o u s aneu rysm o f th e m a n d ib le . O ra l Surg., O ra l M e d . & O ra l Path. 15:442 A p r il 1962. 5. Thom a, K. H . Case r e p o r t o f a so-ca lle d la te n t bone cyst. O ra l Surg., O ra l M e d . & O ra l Path. Sept. 1955. 6. Stafne, E. C . Value o f roen tg e n o g ra m s in diagnosis o f tum o rs o f th e ¡aws. O ra l Surg., O ra ! M e d . & O ra l Path. 6:82 Jan. 1953. 7. Sm ith, H . W . H e m a n g io m a o f the jaws: re v ie w o f th e lite ra tu re a n d r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v . 1959. 8. S iegm und, H ., and W e b e r, R. Pathologische H isto lo g ie d e r M undho hle, L e ip z ig , S. H irz e l, 1926. C ite d in Sm ith, H . W ., H e m a n g io m a o f the jaws: review o f th e lite ra tu re and re p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v . 1959. 9. B rode rick, R. A ., and Round, H . Cavernous an g io m a o f th e m a x illa : fa ta l hae m orrhage a fte r te e th e x tra c tio n , w ith notes o f s im ila r non -fata l case. Lancet 2:13 J u ly I, 1933. C ite d in S m ith, H . W ., H e m an giom a o f the ¡aws: re v ie w o f th e lite ra tu re and r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 Nov. 1959. 10. Stoy, P. J . A n unusual tu m o r. B rit. D. J . 82:100 M arch 7, 1947. 11. Brodsky, R. H . M a n d ib u la r cavernous he m a n g i om a . D. D ig e s t 40:60 Feb. 1934. C ite d in Sm ith, H . W ., H e m a n g io m a o f th e ¡aws: re v ie w o f the lite ra tu re and r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v. 1959. 12. Kamphues, T. K ie rfe ra n g io m a . In augural d is s e rta tio n , M unster, G erm any, H . Buschmann, 1936. C ite d in S m ith, H . W ., H e m a n g io m a o f the ¡aws: review o f the lite ra tu re and r e p o r t o f a case. A M A A rc h . O to la ry n g . 70:579 N ov. 1959. 13. V illa , V. G ., and Lim chayseng, Felix. C e n tra l hem ang iom a o f th e m a n d ib le (c e n tra l a n g io m a )..R e p o rt o f a case. O ra l Surg., O ra l M e d . & O ra l Path. 8:1254 Dec. 1955. 14. Battersby, T. G . C avernous a ng iom a o f the m an d ib le . R e port o f a case. B rit. D. J . 103:347 Nov. 19, 1957.
Pailadino and Danziger: H E M A N G I O M A O F M A N D IB L E • 641
15. D ib b le , P. A . pand W helan, T. J . Cavernous hem ang iom a o f th e m a n d ib le . A rc h . O to la ry n g . (C hi* c a g o ) 75:114 Feb. 1962. 16. Kroh, F. Fatal hem orrh age a fte r e x tra c tio n o f to o th . (A b s t.) J A M A 86:236 Jan. 16, 1926. 17. Jorgensen, A . H a e m a n g lo -c a v e rn o m a ossis m a x il lae. Hals-, Nasen- u. O hre narzt. (Teil I) 27:132, 1936. 18. C a d e n a t, F. M . Un curieux ” l ;p o m e " de la ¡oue. Presse m ed. 49:175 Feb. 12-15, 1941. 19. Bower, L. E., and others. A rte rio v e n o u s a ng iom a o f m a n d ib le and re tin a w ith pronou nced hem atem esis and epista xis. A m . J . Dis. C h ild . 64:1023 Dec. 1942. 20. T oto, P. D., and L a vie ri, John . P rim ary h e m a n g io sarcom a o f th e ¡aw. O ra l Surg., O ra l M e d . & O ra l Path. 12:1459 Dec. 1959. 21. W illia m s , W . J., and G ilb e rt, R. K. H a em a n g io m a o f th e m a n d ib le . B rit. D. J . 98:445 June 21, 1955. 22. Gross, R. E., and W o lb a c h , S. B. Sclerosing he m angiom a s: th e ir re la tio n s h ip to d e rm a to fib ro m a , h is tio cytom a , xanthom a and to ce rta in p ig m e n te d lesions o f the skin. A m . J. Path. 19:533 J u ly 1943. 23. Kauffm an, S. L., and S tout, A . P. Tum ors o f th e m a jo r sa liva ry g lands in c h ild re n . C a ncer 16:1317 O c t. 1963. 24. M urray, M . R., and Stout, A . P. G lom us tu m o r. In ve stig a tio n o f its d is trib u tio n and b e h a v io r, and th e id e n tity o f its " e p ith e lio id " cells. A m . J . Path. 18:183 M arch 1942. 25. Shugart, R. R., Soule, E. H ., and Johnson, E. W ., J r. G lom us tu m o r. Surg., G ynec. & O b s t. 117:334 Sept. 1963. 26. Stout, A . P. H e m a n g io p e ric y to m a : s tudy new cases. C a ncer 2:1027 N o v. 1949.
o f 25
27. M arcial-R ojas, R. A . P rim ary h e m a n g io p e ric y to m a o f b on e: review o f th e lite ra tu re and r e p o r t o f the firs t case w ith metastases. C a ncer 13:308 M a rc h -A p ril 1960. 28. Kauffm an, S. L., and Stout, A . P. H e m a n g io p e ri cytom a in c h ild re n . C a n ce r 13:695 J u ly -A u g . I960. 29. Enterline, H . T., and Roberts, Brooke. Lym phang io p e rlc y to m a : case r e p o rt o f p revio usly undescribed tu m o r ty p e . C a ncer 8:582 M ay-June 1955. 30.
Pachter, M . R., and Lattes, R. M esenchym al tum o rs
o f th e m e d ia s tin u m . I I I . Tumors o f lym ph vascular o rig in . C a ncer 16:108 Jan. 1963. 31. S tew art, F. W ., and Treves, N. Lym phangiosarcom a in postm astectom y ly m p h e d e m a : r e p o rt o f 6 cases in elephantia sis c h iru rg ic a . C a n ce r 1:64 M ay 1948. 32. S tout, A . P. H e m a n g io -e n d o th e lio m a : tu m o r o f b lo o d vessels fe a tu rin g vascular e n d o th e lia l c ells. A nn. Surg. 118:445 Sept. 1943. 33. Tedeschi, C . G ., Folsom , H . F., and C a rn ic e lli, T. J . V isceral K aposi's disease. A rc h . Path. 43:335 A p r il 1947. 34. Tedeschi, C . G . Some consid e ra tio n s concerning th e nature o f so-called sarcom a o f K aposi. A M A A rc h . Path. 66:656 N o v . 1958. 35. Dutz, W ., and Stout, A . P. K aposi's sarcom a in in fan ts and c h ild re n . C a n c e r 13:684 J u ly -A u g . I960. 36. Stout, A . P. M esenchym om a, m ixed tu m o r o f mes enchym al d e riv a tiv e s . A n n. Surg. 127:278 Feb. 1948. 37. Foote, F. W ., J r., and A n derson, H . R. H is to genesis o f Ew ing's tu m o r. A m . J . Path. 17:497 J u ly 1941. 38. J a ffe , H e nry L. Tumors and tum o rou s c o n d itio n s of the bones and jo in ts . P h ila d e lp h ia , Lea & Feb iger, 1958, p. 350. 39. Stout, A . P. Discussion o f p a th o lo g y and h is to genesis o f Ewing's tu m o r o f bone m arrow . A m . J . Roent g e n o l. 50:334 Sept. 1943. 40. J a ffe , H e nry L. Tum ors and tum o rou s con d itio n s o f th e bones and ¡oints. P h ila d e lp h ia , Lea & Febiger, 1958, p. 213. 4 !. Changus, G . W ., Speed, J . S., and Stew art, F. W . M a lig n a n t a n g io b la s to m a o f b on e: re a p p ra is a l o f a d a m antinom a o f long bone. C a n c e r 10:540 M ay-June 1957. 42. J a ffe , H enry L. Aneurysm al bone cyst. Bui. H osp. J o in t Dis. 11:3 A p r il 1950. 43. Lichte nstein, L. Aneurysm al bone cyst: p a th o lo g i cal e n tity co m m o n ly m istaken fo r g ia n t-c e ll tu m o r and occa s io n a lly fo r hem ang iom a and oste o g e n ic sarcom a. C a ncer 3:279 M arch 1950. 44. Lund, H . Z . Tumors o f th e skin. In A rm e d Forces In s titu te o f P athology, A tla s o f tu m o r p a th o lo g y , Sec. I, Fase. 2. W as h in g to n , DC, 1957. p . 274. 45. Lund, B. A ., and D a hlin, D. C . H e m an giom as o f the m a n d ib le and m a x illa . J . O ra l Surg., Anesth. & Hosp. D. Serv. 22:234 M ay 1964.
General Impressions * General impressions are never to be trusted. Unfortunately, when they are of long-standing they become fixed rules of life and assume a prescriptive right not to be questioned. Consequently, those who are not accustomed to original inquiry entertain a hatred and horror of statistics. They cannot endure the idea of submitting their sacred impressions to cold-blooded verification but it is a triumph of scientific men to rise superior to such supersti tions, to desire tests by which the value of beliefs may be ascertained, and to feel sufficiently masters of themselves to discard contemptuously whatever may be found untrue. Sir Francis Galton.
Vincent S. Pailadino,* M D, East Meadow, N Y , and Arthur E. Danziger,f DDS, New York In patients with hemangioma, severe hemorrhage, both fatal and nonfatal, is an attendant hazard in surgical interven tion of the lesion itself or in exodontia. Successful results, however, were achieved by surgical removal of a cavernous type hemangioma of the maxilla in a 50-yearold woman. No exceptional bleeding oc curred during surgery; there were no complications or sequelae postoperatively. In addition to the case report, a review of the literature, a discussion of the diag noses and nomenclature of vascular lesions found in the literature and a working classification of vascular lesions are pre sented. Hemangiomas of the jawbones rarely are reported. The possibility of severe hemor rhage after surgical intervention or tooth extraction makes these lesions potentially dangerous. Experience in successful treat ment of a patient with a hemangioma, cavernous type, prompts the report of a case and a review of the literature. REVIEW OF LITERATURE
In surveying the literature, we sought chiefly those reports of cases in which the
lesion arose in or was limited to jawbones and was labeled “hemangioma” or “an gioma,” mostly interpreted as cavernous or capillary varieties. Thus, for example, recent reports of aneurysmal bone cysts,1 so-called hemangioameloblastoma2,3 and varieties of aneurysms4,5 were excluded. Also eliminated is a case reported by Stafne,® who, in a discussion of “roent genograms in diagnosis of tumors of the jaws,” shows a roentgenogram (his Fig. 6) of an “angioma of the mandible;” no details are given. Smith,7 in his review of the literature and report of a new case, brought the total of reported cases, by 1959, to 29, 10 in which the lesion was in the max illa. Many of the cases in his review were not well documented, since, as Smith states, “Many of these cases were only referred to. . . .” His chart indicates three instances of fatal hemorrhage associated with tooth extraction (Siegmund and Weber,8 Broderick and Round9 and Stoy10) and two instances of severe, nonfatal hemorrhage associated with ex traction (Broderick and Round® and Brodsky11) . Severe hemorrhage associated with surgical intervention is described by Kamphues12 and by Smith in his own report of a case. Of the cases listed by Smith, we call attention to severe, non-
Palladino and Danziger: H E M A N G IO M A O F M A N D IB L E • 637
fatal hemorrhage associated with therapy in Smith’s case no. 25 (Villa and Limchayseng13) and his case no. 26 (Battersby14). The latter case was not docu mented histologically but was assumed by Battersby to represent a hemangioma. Also, we add the case recently reported by Dibble and Whelan,15 who diagnosed a patient’s mandibular lesion as a “circoid aneurysm,” which they considered a variant of cavernous hemangioma. Severe hemorrhage occurred after extraction and necessitated removal of much of the man dible. Dibble and Whelan also refer to a case reported by Kroh16 in which autopsy showed a cavernous hemangioma of the mandible. Thus, of 31 cases reported in the literature, four involved fatal hemor rhage after extraction, three involved severe, nonfatal hemorrhage associated with extraction and four reports recorded severe, nonfatal hemorrhage associated with surgical treatment of the lesion. Al though it is possible that hemangiomas with fatal or near fatal hemorrhage tend to find their way to the literature, the frequency and severity of hemorrhage is nonetheless impressive. Interesting examples of vascular lesions that are not restricted to jawbones but which involve adjacent structures as well are presented in the discussion that fol lows. Jorgensen17 (1936) reported a cavern ous hemangioma of the maxilla that rep resented maxillary involvement by a large “nevus angiomatosus” of much of the skin of the left side of the face, gin giva and hard and soft palate. Fairly copious bleeding continued for 24 hours after extraction. Cadenat18 (1941) reports a lesion he considered to consist of vascular adipose tissue (questionably “angiolipoma” ) in volving much of the left temporal apo neurosis and part of the mandible. Bower and others19 (1942), in an ar ticle titled “Arteriovenous Angioma of the Mandible and Retina with Pro nounced Hematemesis and Epistaxis,” describe what may represent separate le
sions of mandible and retina. A loose tooth was associated with bleeding. Al though no photomicrographs are in cluded, their diagnosis should likely be accepted. The determination of the precise num ber of hemangiomas reported in the lit erature and firm evaluation of many of the diagnoses established on the basis of pathologic findings are impossible for the following reasons: 1. Case reports, at times, are incom plete and contain little discussion of pathologic findings and inadequate to no photomicrographs. Thus, examples of non-neoplastic vascular lesions may be reported as “hemangioma.” A variety of nonvascular lesions may be similarly misdiagnosed. 2. The relative scarcity of reports of diagnoses determined by pathologic find ings of other than “capillary hemangi oma” or “cavernous hemangioma” sug gests that more unusual hemangiomas are overlooked. 3. Variable interpretations of nomen clature of vascular lesions and common references to less than authoritative sources make evaluation of some pub lished cases difficult, particularly when descriptions of pathologic findings and photomicrographs are scanty, inadequate or absent. An interesting report of a case, “Primary Hemangiosarcoma of the Jaw,”20 illustrates the problem encoun tered in evaluating more unusual lesions. This paper presents but two photomicro graphs of only fair quality. These photo micrographs suggest a diagnosis of malig nant hemangioendothelioma to us. The term “hemangiosarcoma” is considered too inclusive by the present authors for all but infrequent and generally debat able lesions; more precise terminology usually is possible. The case reported by Williams and Gilbert21 (hemangioma of the mandible) may well represent he mangioendothelioma, benign juvenile type. Smith’s7 interpretation of the con dition reported by Stoy10 as “capillary hemangioma” is not supported by Stoy’s
438 • J. A M E R . DENT. A SSN .: Vol. 70, March 1965
Fig. I • a: Preoperative roentgenogram showing radiolucent area separating right central and lateral incisors, b-d: Roentgenograms taken 3, 7 and 49 months after surgery, respectively
description or by the single photomicro graph of the lesion, which showed large, thick-walled vascular spaces. WORKING CLASSIFICATION OF VASCULAR LESIONS
Mesenchymoma (only partially vascu lar)36 Angiofibroma (generally “juvenile na sal angiofibroma” ) Granuloma pyogenicum (questionably neoplastic)
II. Neoplasia of debatable, possibly vascu lar, origin The foregoing reasons for questioning the Ewing’s tumor (“endothelioma” )37'39 accuracy of the diagnoses of some of the “Adamantinoma” of long bones40 (ma lesions reported in the literature as “he lignant angioblastoma41)
mangioma” prom pt inclusion here of one author’s (V. S. P.) working classification III. “Vascular” lesions, probably non of vascular lesions as used in this labora neoplastic Aneurysmal bone cyst42,43 tory. References are cited for the more Cystic hygroma (may mimic lymphanunusual or debatable lesions, and they are giectasis and lymphangioma) generally authoritative. Aneurysm (some are considered con
genital) Congenital anomalies (these include I. Neoplastic or very probably neoplastic others in Group III) vascular lesions Hamartoma (may mimic mesenchy Hemangioma, capillary type moma)— often in combination with Hemangioma, cavernous type other components, such as adipose Hemangioma, mixed capillary and tissue cavernous type Pregnancy “tumor” Fibrosing (hyalinizing) hemangioma Lymphangiectasis (example: Milroy’s (not identical to so-called “sclerosing disease) (may mimic cystic hygro hemangioma” of Gross and Wolma) bach22) Telangiectasis (includes “spider” forms) Hemangioendothelioma, benign juven Sclerosing hemangioma (of question ile type (benign hemangioendotheli able vascular origin)22 (“dermato oma23) fibroma,” “xanthoma” )44 Glomus tumor24,25 “Hemangiomatosis” (example: LindauAngiomyoma von Hippel disease) Lymphangioma Angiokeratoma (includes Fabry’s dis Hemangiopericytoma (both benign and ease) malignant clinical behavior are de scribed with identical histology J*-28 Lymphangiopericytoma20' 80 REPORT OF CASE Lymphangiosarcoma (postradical mas tectomy)31 Hemangioendothelioma (malignant )32 Roentgenographic examination of a 50Kaposi’s sarcoma33“35 year-old patient revealed a radiolucent
Palladino and Danziger: H E M A N G I O M A O F M A N D IB L E • 639
Fig. 2 • Left: Low-power photomicrograph of al most entire specimen showing discrete periphery and uniformly distributed vascular channels. Larg est dimension is 1 cm. Top right: Congested•vas cular spaces lined by unexceptional endothelium and separated by small to moderate amounts of smooth muscle. Bottom right: Flat to mildly plump endothelium lining congested vascular spaces
area in the anterior portion of the max illa lying between roots of the maxillary right central and lateral incisors. • Several months before surgery, the patient had noticed that the maxillary right central incisor was being displaced inferiorly and labially. She consulted her dentist who, on a roentgenogram, noticed a pea-sized radio lucent area between the roots of the aforementioned teeth (Fig. la ). The pa tient was referred to one of the authors (A. E. D.) for surgery. Present Illness
• Infiltration anesthesia was in duced with a 2 per cent solution of butethamine hydrochloride, and the patient was prepared and draped in the usual manner in the office.
Surgery
A “half-moon” incision was made on the labial gingiva over the right maxil lary incisors and cuspids. The flap was reflected, exposing the labial plate of the maxilla. By use of bone drills and rongeur
640 • J. A M E R . DENT. A SSN .: Vol. 70, March 1965
forceps, a window was created in the bone, exposing what appeared to be a rubbery, discrete, encapsulated mass. The mass was easily removed from its bony crypt by use of curets; a smooth-walled cavity in the bone remained. There was no exceptional bleeding at the time of surgery. The flap then was returned to position with no. 000 interrupted silk su tures, and the patient was discharged. The patient’s postoperative course progressed without complications and without sequelae. Postoperative roent genograms taken 3, 7 and 49 months after surgery show normal bone healing (Fig. lb -d ). The patient had no com plaints in reference to the lesion when last seen 49 months postoperatively. Pathologist’s Report • Grossly, the speci men consisted of a lobular to granular, rubbery, oval, tan-brown, discrete mass measuring 1.1 by 1.0 by 0.7 cm. On cross section, the lesion was spongy and graypurple. (The entire specimen was sub mitted in four sections.) Microscopically, the lesion had nu merous oval to mildly undulating con gested vascular channels, most of which measured about 1,000 ¡x. in greatest di mension (Fig. 2, left). The vessel walls varied from a slender ribbon composed of one to two smooth muscle cells to oc casional broad bands of smooth muscle (Fig. 2, top and bottom right). The en dothelial cells were flat to slightly plump (Fig. 2, bottom right). The intervascular tissue consisted almost entirely of smooth muscle, much of which was sepa rated by basophilic vacuolated stroma. The periphery was sharply delimited and consisted of a narrow rim of fibrous tis sue and smooth muscle that blended with the perivascular and intervascular stroma. Diagnosis • The diagnosis of the lesion was established as hemangioma, cavern ous type.
ADDENDUM
During the time this paper was being processed for publication, an additional four instances of central hemangioma were reported by Lund and Dahlin.45 Two of these lesions were in the maxilla, and two were in the mandible. PO Box 175
From th e d e p a rtm e n ts o f p a th o lo g y and c lin ic a l la b o ra to rie s and d iv is io n o f o ra l surgery, M e a d o w b ro o k H o s p ita l, East M eadow , N e w Y ork 11554. * D ire c to r o f p a th o lo g y a n d c lin ic a l la b o ra to rie s , M e a d o w b ro o k H o s p ita l; le c tu re r on o ra l surgery (p o s t g ra d u a te ), N ew Y ork U n iversity, New York. fA s s is ta n t a tte n d in g ora ! surgeon, M e a d o w b ro o k H o s p ita l. 1. Vianna, M . R. A n eurysm al bone cyst in th e m a x illa : re p o r t o f case. J . O ra l S u rg., Anesth. & H o sp. D. Serv. 20:432 S e pt. 1962. 2. V illa , V. G . H e m a n g io m a o f the cavernous ty p e associated w ith a m e lo b la s to m a {a m e lo b la s tic h e m a n g i o m a ). J . O ra l Surg., A n esth. & H osp. D. Serv. 18:429 Sept. I960. 3. O liv e r, R. T., M cKenna, W . F., and Shafer, W . G . H e m a n g io -a m e lo b la s to m a : re p o rt o f case. J . O ra l Surg., Anesth. & H osp. D. Serv. 19:245 M ay 1961. 4. C o ok, T. J ., and Z b a r, M . J . A rte rio v e n o u s aneu rysm o f th e m a n d ib le . O ra l Surg., O ra l M e d . & O ra l Path. 15:442 A p r il 1962. 5. Thom a, K. H . Case r e p o r t o f a so-ca lle d la te n t bone cyst. O ra l Surg., O ra l M e d . & O ra l Path. Sept. 1955. 6. Stafne, E. C . Value o f roen tg e n o g ra m s in diagnosis o f tum o rs o f th e ¡aws. O ra l Surg., O ra ! M e d . & O ra l Path. 6:82 Jan. 1953. 7. Sm ith, H . W . H e m a n g io m a o f the jaws: re v ie w o f th e lite ra tu re a n d r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v . 1959. 8. S iegm und, H ., and W e b e r, R. Pathologische H isto lo g ie d e r M undho hle, L e ip z ig , S. H irz e l, 1926. C ite d in Sm ith, H . W ., H e m a n g io m a o f the jaws: review o f th e lite ra tu re and re p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v . 1959. 9. B rode rick, R. A ., and Round, H . Cavernous an g io m a o f th e m a x illa : fa ta l hae m orrhage a fte r te e th e x tra c tio n , w ith notes o f s im ila r non -fata l case. Lancet 2:13 J u ly I, 1933. C ite d in S m ith, H . W ., H e m an giom a o f the ¡aws: re v ie w o f th e lite ra tu re and r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 Nov. 1959. 10. Stoy, P. J . A n unusual tu m o r. B rit. D. J . 82:100 M arch 7, 1947. 11. Brodsky, R. H . M a n d ib u la r cavernous he m a n g i om a . D. D ig e s t 40:60 Feb. 1934. C ite d in Sm ith, H . W ., H e m a n g io m a o f th e ¡aws: re v ie w o f the lite ra tu re and r e p o rt o f a case. A M A A rc h . O to la ry n g . 70:579 N o v. 1959. 12. Kamphues, T. K ie rfe ra n g io m a . In augural d is s e rta tio n , M unster, G erm any, H . Buschmann, 1936. C ite d in S m ith, H . W ., H e m a n g io m a o f the ¡aws: review o f the lite ra tu re and r e p o r t o f a case. A M A A rc h . O to la ry n g . 70:579 N ov. 1959. 13. V illa , V. G ., and Lim chayseng, Felix. C e n tra l hem ang iom a o f th e m a n d ib le (c e n tra l a n g io m a )..R e p o rt o f a case. O ra l Surg., O ra l M e d . & O ra l Path. 8:1254 Dec. 1955. 14. Battersby, T. G . C avernous a ng iom a o f the m an d ib le . R e port o f a case. B rit. D. J . 103:347 Nov. 19, 1957.
Pailadino and Danziger: H E M A N G I O M A O F M A N D IB L E • 641
15. D ib b le , P. A . pand W helan, T. J . Cavernous hem ang iom a o f th e m a n d ib le . A rc h . O to la ry n g . (C hi* c a g o ) 75:114 Feb. 1962. 16. Kroh, F. Fatal hem orrh age a fte r e x tra c tio n o f to o th . (A b s t.) J A M A 86:236 Jan. 16, 1926. 17. Jorgensen, A . H a e m a n g lo -c a v e rn o m a ossis m a x il lae. Hals-, Nasen- u. O hre narzt. (Teil I) 27:132, 1936. 18. C a d e n a t, F. M . Un curieux ” l ;p o m e " de la ¡oue. Presse m ed. 49:175 Feb. 12-15, 1941. 19. Bower, L. E., and others. A rte rio v e n o u s a ng iom a o f m a n d ib le and re tin a w ith pronou nced hem atem esis and epista xis. A m . J . Dis. C h ild . 64:1023 Dec. 1942. 20. T oto, P. D., and L a vie ri, John . P rim ary h e m a n g io sarcom a o f th e ¡aw. O ra l Surg., O ra l M e d . & O ra l Path. 12:1459 Dec. 1959. 21. W illia m s , W . J., and G ilb e rt, R. K. H a em a n g io m a o f th e m a n d ib le . B rit. D. J . 98:445 June 21, 1955. 22. Gross, R. E., and W o lb a c h , S. B. Sclerosing he m angiom a s: th e ir re la tio n s h ip to d e rm a to fib ro m a , h is tio cytom a , xanthom a and to ce rta in p ig m e n te d lesions o f the skin. A m . J. Path. 19:533 J u ly 1943. 23. Kauffm an, S. L., and S tout, A . P. Tum ors o f th e m a jo r sa liva ry g lands in c h ild re n . C a ncer 16:1317 O c t. 1963. 24. M urray, M . R., and Stout, A . P. G lom us tu m o r. In ve stig a tio n o f its d is trib u tio n and b e h a v io r, and th e id e n tity o f its " e p ith e lio id " cells. A m . J . Path. 18:183 M arch 1942. 25. Shugart, R. R., Soule, E. H ., and Johnson, E. W ., J r. G lom us tu m o r. Surg., G ynec. & O b s t. 117:334 Sept. 1963. 26. Stout, A . P. H e m a n g io p e ric y to m a : s tudy new cases. C a ncer 2:1027 N o v. 1949.
o f 25
27. M arcial-R ojas, R. A . P rim ary h e m a n g io p e ric y to m a o f b on e: review o f th e lite ra tu re and r e p o r t o f the firs t case w ith metastases. C a ncer 13:308 M a rc h -A p ril 1960. 28. Kauffm an, S. L., and Stout, A . P. H e m a n g io p e ri cytom a in c h ild re n . C a n ce r 13:695 J u ly -A u g . I960. 29. Enterline, H . T., and Roberts, Brooke. Lym phang io p e rlc y to m a : case r e p o rt o f p revio usly undescribed tu m o r ty p e . C a ncer 8:582 M ay-June 1955. 30.
Pachter, M . R., and Lattes, R. M esenchym al tum o rs
o f th e m e d ia s tin u m . I I I . Tumors o f lym ph vascular o rig in . C a ncer 16:108 Jan. 1963. 31. S tew art, F. W ., and Treves, N. Lym phangiosarcom a in postm astectom y ly m p h e d e m a : r e p o rt o f 6 cases in elephantia sis c h iru rg ic a . C a n ce r 1:64 M ay 1948. 32. S tout, A . P. H e m a n g io -e n d o th e lio m a : tu m o r o f b lo o d vessels fe a tu rin g vascular e n d o th e lia l c ells. A nn. Surg. 118:445 Sept. 1943. 33. Tedeschi, C . G ., Folsom , H . F., and C a rn ic e lli, T. J . V isceral K aposi's disease. A rc h . Path. 43:335 A p r il 1947. 34. Tedeschi, C . G . Some consid e ra tio n s concerning th e nature o f so-called sarcom a o f K aposi. A M A A rc h . Path. 66:656 N o v . 1958. 35. Dutz, W ., and Stout, A . P. K aposi's sarcom a in in fan ts and c h ild re n . C a n c e r 13:684 J u ly -A u g . I960. 36. Stout, A . P. M esenchym om a, m ixed tu m o r o f mes enchym al d e riv a tiv e s . A n n. Surg. 127:278 Feb. 1948. 37. Foote, F. W ., J r., and A n derson, H . R. H is to genesis o f Ew ing's tu m o r. A m . J . Path. 17:497 J u ly 1941. 38. J a ffe , H e nry L. Tumors and tum o rou s c o n d itio n s of the bones and jo in ts . P h ila d e lp h ia , Lea & Feb iger, 1958, p. 350. 39. Stout, A . P. Discussion o f p a th o lo g y and h is to genesis o f Ewing's tu m o r o f bone m arrow . A m . J . Roent g e n o l. 50:334 Sept. 1943. 40. J a ffe , H e nry L. Tum ors and tum o rou s con d itio n s o f th e bones and ¡oints. P h ila d e lp h ia , Lea & Febiger, 1958, p. 213. 4 !. Changus, G . W ., Speed, J . S., and Stew art, F. W . M a lig n a n t a n g io b la s to m a o f b on e: re a p p ra is a l o f a d a m antinom a o f long bone. C a n c e r 10:540 M ay-June 1957. 42. J a ffe , H enry L. Aneurysm al bone cyst. Bui. H osp. J o in t Dis. 11:3 A p r il 1950. 43. Lichte nstein, L. Aneurysm al bone cyst: p a th o lo g i cal e n tity co m m o n ly m istaken fo r g ia n t-c e ll tu m o r and occa s io n a lly fo r hem ang iom a and oste o g e n ic sarcom a. C a ncer 3:279 M arch 1950. 44. Lund, H . Z . Tumors o f th e skin. In A rm e d Forces In s titu te o f P athology, A tla s o f tu m o r p a th o lo g y , Sec. I, Fase. 2. W as h in g to n , DC, 1957. p . 274. 45. Lund, B. A ., and D a hlin, D. C . H e m an giom as o f the m a n d ib le and m a x illa . J . O ra l Surg., Anesth. & Hosp. D. Serv. 22:234 M ay 1964.
General Impressions * General impressions are never to be trusted. Unfortunately, when they are of long-standing they become fixed rules of life and assume a prescriptive right not to be questioned. Consequently, those who are not accustomed to original inquiry entertain a hatred and horror of statistics. They cannot endure the idea of submitting their sacred impressions to cold-blooded verification but it is a triumph of scientific men to rise superior to such supersti tions, to desire tests by which the value of beliefs may be ascertained, and to feel sufficiently masters of themselves to discard contemptuously whatever may be found untrue. Sir Francis Galton.