- Email: [email protected]
Cavernous hemangioma of striated muscle
Cavernous REVIEW
Hemangioma OF
THE
LITERATURE
of Striated AND
REPORT
ANTONIO F. LA SORTE, M.D., Binghamton,
From Our Lady New York.
of
Lourdes
Hospital,
Bingbamton,
of striated muscle are fascinating tumors. Since only some 386 cases of this entity have been reported in the world Iiterature, it is doubtfu1 that many surgeons have seen a case or wouId chance to diagnose it correctIy. Jones [5] states that Liston (I 843) recorded the first case of hemangioma of muscIe. The Iatter was an erectile tumor of the popliteal space. ExcelIent and comprehensive reviews of this subject have been made by Davis and KitIowski in 1930 [I], Jenkins and DeIaney in 1932 [z], ShaIIow, Eger and Wagner in 1944 [3], BonvaIIet in 1940 [4], and Jones in 1953 [5]. Bendeck and Lichtenberg [6] (1957) recentIy reviewed the literature and added two cases bringing the composite tota to 386. The object of this paper is to record another case with review of the literature since the paper of Bendeck and Lichtenberg [6]. Since the Iatter paper, tweIve new cases have been reported and are summarized in TabIe I.
C
OF
Muscle
ONE
CASE
York
New
On physica examination, the patient was very well developed and we11 nourished with a blood
AVERNOUS hemangiomas
TABLE RECENTLY
REPORTED
HEMANGIOMA
OF
I
CASES
OF
SKELETAL
COMPOSITE
Author
CAVERNOUS
MUSCLE
AND
TOTAL
P40. of (2ases
Location of Tumor
_ Bendeck and Lichtenberg [6] (previousIy reported cases) Prignacchi [7] (not included by Bendeck and Lichtenberg) Chironi [8] Scott [9]
386
jemimembranosus left
Mercadente Zampi [rz]
J. B., a twenty-six year old white, married, construction worker, was admitted to Our Lady of Lourdes Hospital in Binghamton, New York, on December 2, 1957. Eight months previousIy he had noted a pain in his back. The pain was intermittent and aching in character. He had recentIy had the impression that a sweIIing was present. The patient consuIted with his physician who at first gIance thought that the tumor represented a Iipoma. On a more thorough and carefuI examination, the swelIing seemed to pulsate and auscuItation reveaIed a systolic bruit.
Giugiano [I31
I
BrachiaIis ScaIenus posticus Mylohyoid Rectus femoris Soleus and gastrocnemius Muscles under tendon AchiIIes Quadriceps Vastus medialis Pectorahs major MuscIes of mid-thigh aIong the course of the saphenous nerve
Parrini [ro]
CASE REPORT
_
[I I]
I 2
5
I I
and Bertola
Present case
I
Abductor haIIucis FIexor digitorum brevis Levator scapulae with contiguous four muscles
Total................................
I
I
399
Note: Tumors involving mainIy one muscIe with spread to another or more were counted as one case.
593
American
Journal
of Surgery,
Volume
roe,
October
1960
La Sorte was compressible. AuscuItation reveaIed a grade 2 systoIic bruit. The skin over the area was norma in consistency and coIor and not unusuaIIy warm. One had the impression that it Iay certainly beIow the subcutaneous Iayer and perhaps deeper. A posteroanterior roentgenogram of the chest showed an obvious soft tissue sweIIing in the Ieft supracIavicuIar area. Angiocardiogram reveaIed at Ieast two prominent arterial feeders entering the tumor from the Ieft axiIIary artery. The vascuIar network within the mass was tortuous. DetaiIed studies of the Ieft scapuIa reveaIed no bony erosion or destruction. Tentative diagnosis prior to surgery was hemangio-endotheIioma or angiosarcoma. On December 5, 1957, the patient was operated upon under endotracheal anesthesia. He was pIaced in the right IateraI decubitus position. A generous incision was outIined foIlowing the contour of the scapula. Bleeding in the subcutaneous and the muscIe Iayers was rather severe and necessitated meticuIous hemostasis with fine cotton Iigatures. The trapezius muscIe was transected for the entire length of the incision. This provided a good exposure of the area of disease. The tumor invoIved mainIy the Ievator scapulae, but, in addition, portions of the rhomboideus minor and major were invoIved as they inserted into the media1 border of the scapuIa. A portion of the supraspinatus and some of the subscapuIaris as we11as the attachment of the serratus magnus were invoIved. Since the nature of the tumor was not known, a wide margin of normal muscle was obtained in addition to transecting a greater portion of the superior angIe of the scapuIa. Entering the tumor from above were at Ieast two arteries the size of an average radial
FIG. I. The circuIar marking describes the maxima1 area of sweIIing. pressure of I 10/70 mm. Hg, and a puIse of 84. The positive physical findings were Iimited to his Ieft upper back. Media1 to the superior angIe of the Ieft scapuIa, a sweIIing was present. (Fig. I .) The sweIIing, aIthough rather noticeabIe, was not discreet nor were its boundaries sharpIy defined. Pressure upon it resuIted in a visibIe and tactiIe puIsation. It
FIG. 2. The cavernous spaces supported by a fibrobIastic stroma in which are interspersed isIands of striated mu&e are weIl demonstrated (high power).
594
Cavernous
Hemangioma
artery. The tumor was rubbery and firm and seemed grossly infiltrative. Its coIor was a yelIowish, reddish gray. Closure was effected by uniting the sectioned supraspinatus muscle to the underlying subscapuIaris muscIe and thus seaIing the transected edge of scapuIa. The wound was irrigated with saIine and penicillin soIution. Cotton sutures were utilized to close and approximate the muscle Iayers and the subscapuIar space was drained by means of a Penrose drain. The postoperative course was uncompIicated. There was a moderate amount of serosanguinous fluid drainage for the first two or three postoperative days as the drain was progressiveIy shortened. ShouIder movement was exceIIent and has continued to be so. There has been no recurrence of the tumor. Pathological Report. The specimen consisted of
a partiaIIy encapsulated and fairIy we11 circumscribed paIe gray spongy mass with Iarge pieces of skeIeta1 muscle and a narrow, Iong piece of Aat bone attached to its surface. The mass measured 18 cm. by 12 cm. by IO cm. The growth was incorporated with the periosteum aIong the entire Iength of the bony piece attached to its surface. Microscopic Report. Sections reveaIed a vascuIar tumor consisting of Iarge and smaI1 bIood sinuses and scattered foci of we11formed capiIIaries. Some of the sinusoids were thrombosed. Most of the stroma was hyaIinized. FibrobIastic proIiferation between the sinusoids and isIands of striated muscIe was noted. (Fig. 2.) COMMENT
Hemangiomas
of striated
muscle,
according
to most authors
[r-3,5,6], are probabIy congenitaI in origin. Trauma may pIay a role as a contributing factor in their growth. A simple cIassification into capiIIary, cavernous, venous and arteria1 is a practica1 working scheme reaIizing that a mixing of this pattern is present in about 38 per cent of those reported. A strictIy cavernous arrangement was present in 54 per cent whiIe a pureIy capiIIary pattern was present in 3 per cent of the cases. PureIy arteria1 and venous types accounted for 2 per cent each of the tota cases reported. GrossIy the tumors may have a bIuish hue or present a reddish, yeIIowish gray appearance. The tumors may be spongy, compressibIe, hard, or firm. On section, the vesseIs may stand out Iike stems. Thirteen per cent of these tumors were we11 circumscribed with a distinct capsuIe within a muscIe. In 5 per cent of the reported cases, they were partiaIIy circumscribed having in part a distinct capsuIe whiIe the remainder
595
of Striated
MuscIe
i&Itrated the muscle. The Iargest number were of the diffuse type and these hemangiomas infiItrated in an irreguIar fashion into one or adjacent muscles and invoIved veins, arteries or nerves. The diffuse type accounted for 3g per cent of the cases. These tumors differ from the usua1 benign tumor in that growth is by infiItration and not by expansion. MicroscopicaIIy one sees cavernous, capiIIary, venous and arterial vascular eIements in a connective tissue stroma. RareIy are there ossified areas. CaIcuIi or phIeboIiths are present where there is stasis. Remnants of striated muscIe are found in varying stages of degeneration. Eighty-five per cent of the patients with hemangiomas of striated muscIe are in the first three decades of Iife. These tumors are sIightIy more common in women. Pain is a cardinal symptom occurring in 60 per cent of the cases. A mass is aImost aIways present and is found in g8 per cent of the tumors reported. Only 3 per cent of these tumors pulsate and a bruit is heard in I .8 per cent. The overrying skin was norma in 39 per cent but can be bIuish, warm, teIangiectatic, uIcerated or contain nevi. Any striated muscle can be invoIved but the quadriceps is the most frequentIy invoIved muscIe. One muscIe was invoIved in 66 per cent of the cases reported. Two muscIes were invoIved in 12.5 per cent, three muscIes in 3.6 per cent, four muscIes in 1.2 per cent and five muscIes in 0.3 per cent of the tota number reported. In the differentia1 diagnosis, these possibilities have been entertained: Iipoma, sarcoma, fibroma, hematoma, primary carcinoma, dermoid cysts, syphiIoma, chronic myositis, tubercuIar myositis, myositis ossificans, hydatid cyst, and hernia of muscle or of Iung. Treatment of course shouId be compIete excision when this is feasibIe. PartiaI excision, amputation and the use of scIerosing agents have been utiIized. Recoverv and cure is the ruIe if complete excision ;s carried out. This patient’s first compIaint was pain and the discovery of a mass. InitiaI diagnosis was a Iipoma aIthough further examination reveaIed puIsation and the presence of a bruit. Angiocardiogram was very successfu1 in outIining the area of disease and revearing the vascuIar nature of the tumor. This tumor showed the more “rare characteristics of hemangiomas of muscIe. It puIsated, had a bruit and invoIved a tota of five muscIes by virtue of its infiItrative
La Sorte 4. BONVALLET, J. M. Angiomas des muscIes de squelette. Presse mkd., 58: 535, 1950. 5. JONES, K. G. Cavernous hemangioma of striated muscIe; a review of the Iiterature and a report of four cases. J. Bone @ Joint Surg., 35: 717, 1953. 6. BENDECK, T. E. and LICHTENBERG, F. Cavernous hemangioma of striated muscle; review of the Iiterature and report of two cases. Ann, Surg., 146: 1011, 19ri7. 7. PRI&ACCHI; q: ‘A proposito della Semeiotic degli Emangiomi MuscoIari. Cbir. d. org. di movimento, 43: 470. ‘956. 8. CHIRONI, P. In Tema di Emangiomi MuscuIari degIi Arti, descrizione di un case di mio angiome de1 brachiaIe anteriore. Minerua ortop., 8: 389,
quaIities. A partial scapuIectomy was necessary for its compIete abIation. SUMMARY
A case of hemangioma of striated muscIe is reported bringing the tota reported to 399. This tumor exhibited the more rare characteristics such as pukation, bruit and muItipIe muscIe invoIvement. Cure was effected by wide excision and partiaI scapuIectomy. A brief review of the pertinent data concerning these tumors is presented.
1957. g. SCOTT, J. E. S. Hemangiomata in skeIeta1 muscIes. Brit. J. Surg., 44: 496, 1957. IO. PARRINI, L. GIi Emangiomi deh’apparato muscoIare. Arch. ortop., 70: 708, 1957. II. MERCADENTE, T. GIi Angiomi primitivi dei muscoIi striati. Tumori, 43: 235, 1957. 12. ZAMPI, G. Rara ossificazione de110 stroma in un angioma cavernoso de1 muscoIo. Arch. “de Veccbi” anat. pat., 27: 181, 1957. 13. GIUGIANO, A. and BERTOLA, L. Emangioma dei muscoIi de1 piede. Arch. SC. med., 106: I 13, 1958.
REFERENCES
I. DAVIS, J. S. and KITLOWSKI, E. A. Primary intrahemangiomas of striated muscIe. muscuIar Arch. Surg., 20: 39. 1930. 2. JENKINS. H. P. and DELANEY. P. A. Benien angiomatous tumors of skeIeta1 muscIes. Surg., Cynec. Ed Obst., 55: 464, 1932. 3. SHALLOW,T. A., EGER, S. A. and WAGNER, F. B., JR. Primary hemangiomatous tumors of skeIeta1 muscIe. Ann. Surg., I Ig: 700, 1944.
596
Hemangioma OF
THE
LITERATURE
of Striated AND
REPORT
ANTONIO F. LA SORTE, M.D., Binghamton,
From Our Lady New York.
of
Lourdes
Hospital,
Bingbamton,
of striated muscle are fascinating tumors. Since only some 386 cases of this entity have been reported in the world Iiterature, it is doubtfu1 that many surgeons have seen a case or wouId chance to diagnose it correctIy. Jones [5] states that Liston (I 843) recorded the first case of hemangioma of muscIe. The Iatter was an erectile tumor of the popliteal space. ExcelIent and comprehensive reviews of this subject have been made by Davis and KitIowski in 1930 [I], Jenkins and DeIaney in 1932 [z], ShaIIow, Eger and Wagner in 1944 [3], BonvaIIet in 1940 [4], and Jones in 1953 [5]. Bendeck and Lichtenberg [6] (1957) recentIy reviewed the literature and added two cases bringing the composite tota to 386. The object of this paper is to record another case with review of the literature since the paper of Bendeck and Lichtenberg [6]. Since the Iatter paper, tweIve new cases have been reported and are summarized in TabIe I.
C
OF
Muscle
ONE
CASE
York
New
On physica examination, the patient was very well developed and we11 nourished with a blood
AVERNOUS hemangiomas
TABLE RECENTLY
REPORTED
HEMANGIOMA
OF
I
CASES
OF
SKELETAL
COMPOSITE
Author
CAVERNOUS
MUSCLE
AND
TOTAL
P40. of (2ases
Location of Tumor
_ Bendeck and Lichtenberg [6] (previousIy reported cases) Prignacchi [7] (not included by Bendeck and Lichtenberg) Chironi [8] Scott [9]
386
jemimembranosus left
Mercadente Zampi [rz]
J. B., a twenty-six year old white, married, construction worker, was admitted to Our Lady of Lourdes Hospital in Binghamton, New York, on December 2, 1957. Eight months previousIy he had noted a pain in his back. The pain was intermittent and aching in character. He had recentIy had the impression that a sweIIing was present. The patient consuIted with his physician who at first gIance thought that the tumor represented a Iipoma. On a more thorough and carefuI examination, the swelIing seemed to pulsate and auscuItation reveaIed a systolic bruit.
Giugiano [I31
I
BrachiaIis ScaIenus posticus Mylohyoid Rectus femoris Soleus and gastrocnemius Muscles under tendon AchiIIes Quadriceps Vastus medialis Pectorahs major MuscIes of mid-thigh aIong the course of the saphenous nerve
Parrini [ro]
CASE REPORT
_
[I I]
I 2
5
I I
and Bertola
Present case
I
Abductor haIIucis FIexor digitorum brevis Levator scapulae with contiguous four muscles
Total................................
I
I
399
Note: Tumors involving mainIy one muscIe with spread to another or more were counted as one case.
593
American
Journal
of Surgery,
Volume
roe,
October
1960
La Sorte was compressible. AuscuItation reveaIed a grade 2 systoIic bruit. The skin over the area was norma in consistency and coIor and not unusuaIIy warm. One had the impression that it Iay certainly beIow the subcutaneous Iayer and perhaps deeper. A posteroanterior roentgenogram of the chest showed an obvious soft tissue sweIIing in the Ieft supracIavicuIar area. Angiocardiogram reveaIed at Ieast two prominent arterial feeders entering the tumor from the Ieft axiIIary artery. The vascuIar network within the mass was tortuous. DetaiIed studies of the Ieft scapuIa reveaIed no bony erosion or destruction. Tentative diagnosis prior to surgery was hemangio-endotheIioma or angiosarcoma. On December 5, 1957, the patient was operated upon under endotracheal anesthesia. He was pIaced in the right IateraI decubitus position. A generous incision was outIined foIlowing the contour of the scapula. Bleeding in the subcutaneous and the muscIe Iayers was rather severe and necessitated meticuIous hemostasis with fine cotton Iigatures. The trapezius muscIe was transected for the entire length of the incision. This provided a good exposure of the area of disease. The tumor invoIved mainIy the Ievator scapulae, but, in addition, portions of the rhomboideus minor and major were invoIved as they inserted into the media1 border of the scapuIa. A portion of the supraspinatus and some of the subscapuIaris as we11as the attachment of the serratus magnus were invoIved. Since the nature of the tumor was not known, a wide margin of normal muscle was obtained in addition to transecting a greater portion of the superior angIe of the scapuIa. Entering the tumor from above were at Ieast two arteries the size of an average radial
FIG. I. The circuIar marking describes the maxima1 area of sweIIing. pressure of I 10/70 mm. Hg, and a puIse of 84. The positive physical findings were Iimited to his Ieft upper back. Media1 to the superior angIe of the Ieft scapuIa, a sweIIing was present. (Fig. I .) The sweIIing, aIthough rather noticeabIe, was not discreet nor were its boundaries sharpIy defined. Pressure upon it resuIted in a visibIe and tactiIe puIsation. It
FIG. 2. The cavernous spaces supported by a fibrobIastic stroma in which are interspersed isIands of striated mu&e are weIl demonstrated (high power).
594
Cavernous
Hemangioma
artery. The tumor was rubbery and firm and seemed grossly infiltrative. Its coIor was a yelIowish, reddish gray. Closure was effected by uniting the sectioned supraspinatus muscle to the underlying subscapuIaris muscIe and thus seaIing the transected edge of scapuIa. The wound was irrigated with saIine and penicillin soIution. Cotton sutures were utilized to close and approximate the muscle Iayers and the subscapuIar space was drained by means of a Penrose drain. The postoperative course was uncompIicated. There was a moderate amount of serosanguinous fluid drainage for the first two or three postoperative days as the drain was progressiveIy shortened. ShouIder movement was exceIIent and has continued to be so. There has been no recurrence of the tumor. Pathological Report. The specimen consisted of
a partiaIIy encapsulated and fairIy we11 circumscribed paIe gray spongy mass with Iarge pieces of skeIeta1 muscle and a narrow, Iong piece of Aat bone attached to its surface. The mass measured 18 cm. by 12 cm. by IO cm. The growth was incorporated with the periosteum aIong the entire Iength of the bony piece attached to its surface. Microscopic Report. Sections reveaIed a vascuIar tumor consisting of Iarge and smaI1 bIood sinuses and scattered foci of we11formed capiIIaries. Some of the sinusoids were thrombosed. Most of the stroma was hyaIinized. FibrobIastic proIiferation between the sinusoids and isIands of striated muscIe was noted. (Fig. 2.) COMMENT
Hemangiomas
of striated
muscle,
according
to most authors
[r-3,5,6], are probabIy congenitaI in origin. Trauma may pIay a role as a contributing factor in their growth. A simple cIassification into capiIIary, cavernous, venous and arteria1 is a practica1 working scheme reaIizing that a mixing of this pattern is present in about 38 per cent of those reported. A strictIy cavernous arrangement was present in 54 per cent whiIe a pureIy capiIIary pattern was present in 3 per cent of the cases. PureIy arteria1 and venous types accounted for 2 per cent each of the tota cases reported. GrossIy the tumors may have a bIuish hue or present a reddish, yeIIowish gray appearance. The tumors may be spongy, compressibIe, hard, or firm. On section, the vesseIs may stand out Iike stems. Thirteen per cent of these tumors were we11 circumscribed with a distinct capsuIe within a muscIe. In 5 per cent of the reported cases, they were partiaIIy circumscribed having in part a distinct capsuIe whiIe the remainder
595
of Striated
MuscIe
i&Itrated the muscle. The Iargest number were of the diffuse type and these hemangiomas infiItrated in an irreguIar fashion into one or adjacent muscles and invoIved veins, arteries or nerves. The diffuse type accounted for 3g per cent of the cases. These tumors differ from the usua1 benign tumor in that growth is by infiItration and not by expansion. MicroscopicaIIy one sees cavernous, capiIIary, venous and arterial vascular eIements in a connective tissue stroma. RareIy are there ossified areas. CaIcuIi or phIeboIiths are present where there is stasis. Remnants of striated muscIe are found in varying stages of degeneration. Eighty-five per cent of the patients with hemangiomas of striated muscIe are in the first three decades of Iife. These tumors are sIightIy more common in women. Pain is a cardinal symptom occurring in 60 per cent of the cases. A mass is aImost aIways present and is found in g8 per cent of the tumors reported. Only 3 per cent of these tumors pulsate and a bruit is heard in I .8 per cent. The overrying skin was norma in 39 per cent but can be bIuish, warm, teIangiectatic, uIcerated or contain nevi. Any striated muscle can be invoIved but the quadriceps is the most frequentIy invoIved muscIe. One muscIe was invoIved in 66 per cent of the cases reported. Two muscIes were invoIved in 12.5 per cent, three muscIes in 3.6 per cent, four muscIes in 1.2 per cent and five muscIes in 0.3 per cent of the tota number reported. In the differentia1 diagnosis, these possibilities have been entertained: Iipoma, sarcoma, fibroma, hematoma, primary carcinoma, dermoid cysts, syphiIoma, chronic myositis, tubercuIar myositis, myositis ossificans, hydatid cyst, and hernia of muscle or of Iung. Treatment of course shouId be compIete excision when this is feasibIe. PartiaI excision, amputation and the use of scIerosing agents have been utiIized. Recoverv and cure is the ruIe if complete excision ;s carried out. This patient’s first compIaint was pain and the discovery of a mass. InitiaI diagnosis was a Iipoma aIthough further examination reveaIed puIsation and the presence of a bruit. Angiocardiogram was very successfu1 in outIining the area of disease and revearing the vascuIar nature of the tumor. This tumor showed the more “rare characteristics of hemangiomas of muscIe. It puIsated, had a bruit and invoIved a tota of five muscIes by virtue of its infiItrative
La Sorte 4. BONVALLET, J. M. Angiomas des muscIes de squelette. Presse mkd., 58: 535, 1950. 5. JONES, K. G. Cavernous hemangioma of striated muscIe; a review of the Iiterature and a report of four cases. J. Bone @ Joint Surg., 35: 717, 1953. 6. BENDECK, T. E. and LICHTENBERG, F. Cavernous hemangioma of striated muscle; review of the Iiterature and report of two cases. Ann, Surg., 146: 1011, 19ri7. 7. PRI&ACCHI; q: ‘A proposito della Semeiotic degli Emangiomi MuscoIari. Cbir. d. org. di movimento, 43: 470. ‘956. 8. CHIRONI, P. In Tema di Emangiomi MuscuIari degIi Arti, descrizione di un case di mio angiome de1 brachiaIe anteriore. Minerua ortop., 8: 389,
quaIities. A partial scapuIectomy was necessary for its compIete abIation. SUMMARY
A case of hemangioma of striated muscIe is reported bringing the tota reported to 399. This tumor exhibited the more rare characteristics such as pukation, bruit and muItipIe muscIe invoIvement. Cure was effected by wide excision and partiaI scapuIectomy. A brief review of the pertinent data concerning these tumors is presented.
1957. g. SCOTT, J. E. S. Hemangiomata in skeIeta1 muscIes. Brit. J. Surg., 44: 496, 1957. IO. PARRINI, L. GIi Emangiomi deh’apparato muscoIare. Arch. ortop., 70: 708, 1957. II. MERCADENTE, T. GIi Angiomi primitivi dei muscoIi striati. Tumori, 43: 235, 1957. 12. ZAMPI, G. Rara ossificazione de110 stroma in un angioma cavernoso de1 muscoIo. Arch. “de Veccbi” anat. pat., 27: 181, 1957. 13. GIUGIANO, A. and BERTOLA, L. Emangioma dei muscoIi de1 piede. Arch. SC. med., 106: I 13, 1958.
REFERENCES
I. DAVIS, J. S. and KITLOWSKI, E. A. Primary intrahemangiomas of striated muscIe. muscuIar Arch. Surg., 20: 39. 1930. 2. JENKINS. H. P. and DELANEY. P. A. Benien angiomatous tumors of skeIeta1 muscIes. Surg., Cynec. Ed Obst., 55: 464, 1932. 3. SHALLOW,T. A., EGER, S. A. and WAGNER, F. B., JR. Primary hemangiomatous tumors of skeIeta1 muscIe. Ann. Surg., I Ig: 700, 1944.
596