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Primary Nonpigmented Sarcoma of the Vulva, with Report of a Case Complicating Pregnancy
PRIMARY NONPIGMENTED SARCOMA OF THE VULVA, WITH REPORT OF A CASE COMPLICATING PREGNANCY RAYMOND 1 From
P. NoLAN, M.D., NEW YoRK, N.Y.
the Obstetrical and Gynecological Service [Third Surgical Division], Bellevue Hospital, and the Department of Obstetrics and Gynecology, New York Unive1·sity College of Medicine)
nRIMARY nonpigmented sarcoma of the vulva is an extremely rare type of tumor, as sho~n by the sparsity of cases in the literature, and the brief discussion allotted to it in textbooks. Certainly, it is an exceedingly rare complication of pregnancy. Any type of maligant growth of the vulva is unusual during the childbearing age, perhaps the most common such tumor being the melanoma. Because of the location of the lesion in the case presented here, an anatomical definition of the term, vulva, wili be mentioned. Taussig,' 4 in his monograph on the diseases of the vulva, quotes Poirer and Charpy as follows: ''The vulva can, therefore, be considered to include the following structures: the labia majora and minora, the mons veneris, the perineum, the clitoris and its folds, the Bartholin's glands, thr vestibule of the vagina, the meatus of the urethra, and the hymen.'' A review of the literature on primary nonpigmented sarcoma of the vulva shows no recent cases complicating pregnancy. One of the E-arliest collections of cases of vulvar sarcoma was by Blair BelP in 1907, who found only 6 in patients under 30 years of age out of a total of 18 cases. In only one instance was the tumor in the region of the vestibule. Kehrer, 6 in 1929, rrferred to 7i cases of primary sarcoma of the vulva, which was apparently all he was able to collect from the literature. In 1937, Taussig 15 reported a case of liposarcoma of the labium majus occurring in a 29-year-old primipara. There was no relation to pregnancy. The tumor, although present as a small lump for about four years, had grown suddenly larger in a six-month period. IJocal excision was followed six weeks later by a unilateral vulvectomy and inguinal lymph node dissection. The patient died with widespread metastases about six months after the original enucleation of the tumor. Taussig 15 suggested that, with modern staining methods, a few of thr eases reported as other types of sarcoma in the older liter·ature may have shown evidence of arising from the fatty tissue of the vulva. He quoted several characteristics of liposareoma in general as describe(! by Shaw in 19;{6. Included are the facts that they may grow slowly for several years, with a sudden increase in size; that the encapsulation does not suggest malignancy; and that they have a varied hisologic structure.
r
134
\"olumc73
Number I
PRIMARY NONPIGMENTED SARCOMA OF VULVA
135
In 1928, a case of unpigmented sarcoma of the labium majus in a girl of sixteen years was reported. 8 It was of the alveolar round-cell type. There were widespread metastases less than four months after the tumor was originally noted. In 1937, congenital myxosarcoma of the left labium majus was described in an infant. 1 Death occurred within a few months. There were mr•tastases to the lungs and the mediastinal lymph nodes. In 1939, a case of primary sarcoma of the vulva was reported by Bernstein 2 in a 45-year-ohl woman. It had grown slowly over a sixteen-year period to the size of a gr·apefruit. It was treated by local excision follow<'d hy radiation. A fouryear follow-up showed no evidence of recurr<'llt<'. In 1940, a case of fibr·osarcoma of the vulva in a 7-month-old infant was d<>scrilwd by Hauptman and Taussig. 5 Local excision was carried out, followed later by radiation to local r·ef•.urrences and inguinal metastases. but the infant died with prlvic tumor masses about a year later. In the recent literature only a few cases ar·e reported. Servir·e and Derbyshirc11 described in 1946 a pedunculated fibromyxosarcoma of the left labium minor in a 3-year-old girl. A local wide excision was performed. Five and one-half months later, metastases were found in the right inguinal lymph nodes. Keller, 7 in 1951, reported a large fibrosarcoma of the left labia in a G8-ycar-old patient. Sacrez and associates/ 0 also in 1951, reported a casf' of rhabdomyoblastic sarcoma of the vulva in a 4-month-old infant. Hr· believNI this to h<' congenital in origin. Recently, in 19!15, Buckingham and McClure' r·cported a reticulum-cell sarcoma of the vulva in a 33-year-old patient, which had grovv·n to a considerable size in a period of a fevv W\l(lks. She was treated hy loral exeision of the tumor, followed by radiation tht>rapy. Th<' patient was asymptomatic up to six months later, althoug-h rE'current masses w<>rE' felt ill the pf'lvis at this time. EvE'n from this small group of cases, it is t'vident that the age incidt>rw• nf this type of vulvar tumor is very wide, from birth to old age, and that the prognosis is poor at any age. A 23-year·old married white primigravida, a nur~e, was first seen on July 30, 1954, when she was admitterl to the Obstetrical Service with a diagnosis of acute pyelonephritis in the seventh month of pregnancy. She improved rapidly on antibiotic therapy anrl was discharged, asymptomatic, on Aug. 10, 1954. At the time of that arlmission, the patient offerer! no complaint of any growth of the ,-agina or the vulva, and none was noted on the physical examination. She was subsequently followed in the prenatal clinic, where a routine pelvic examination on Aug. 17, 1954, did not reveal any vaginal or vulvar growth. The patient later stated that she had noted a small mass, about the size of a pea, on the left side of the introitus a few tlays afterward. She first complained about it on Sept. 7, 1954, during a visit to the prenatal clinic. It was slightly larger at this time, and on examination it was considered to be merely a small cyRt arising from the left side of the vestibule. Since the estimated date of confinement was Sept. 17, 1954, only ten da~·s away, it was thought advisable to "?fait and remove it at a later date if necessary. The 1na:..;s giew rapidly over the next few~ weeks, but apparently caused no symptoms. No further note was made of it. On Oct. ·L 1954, twenty-seven •lays after the patient had first complained of the nodule, and approximately six weeks after she had first noted it, she was admitted to the ho8pital in acti \·e labor.
136
NOLAN
i\m . .J. Obst. & Gynec. January, 1917
Arising from near the lower portion of the inner margin of the left labium minor, was a nontender, pedunculated, moderately firm tumor (Fig. 1). It was oval in shape, and measured about 2.5 by 2.5 by 3.0 em. It was hanging on an elastic, thin pedicle of mucosa which measured about 0.5 em. in diameter and 2.5 em . in length. The color resembled that of the labia minora except for a 1.5 em. area of ecchymosis and ulceration on the anterior surface. The patient was seen shortly after admission by two members of the attending staff, who suggested ren1oval of the n1ass at the time o:f delivery by ligation and transection of the pedicle at its base. The growth was considered to be benign, either a lipoma or a fibroma. After an uneventful labor of eleven hours, a normal spontaneous delivery of a 6 pound, 10 ounce, normal infant occurred over a right mediolateral episiotomy. At delivery, the tumor wa~ removed by double ligation of the baRe of the pedicle
Fig. 1.-Sketch showing tumor hanging from pedicle on left side of the vulva.
with No. 00 chromic catgut sutures, followeu by transection at the base. The specimen was sent to the pathology laboratory for routine examination, the report of which follows: ''Accession No. 5314-54. The specimen received fixed in Bouin 's solution consists of a firm spherical mass averaging 2.5 em, in diameter. The cut surface has a pale fibrous homogeneous appearance. [Fig. 2.] On microscopic examination, the tumor is made up of two types of cells. The more frequent type is a spindle shaped cell ·with a small round ovoid nucleus and indistinct cell borders. These are arranged in interlacing cords and whorls. [Fig. 3.] The second type of cell is an irregular and bizarre giant cell with abunaant sharply aenned, somewhat basophlllC cytoplasm. These latter cells are multinucleate, with nuclei which vary considerably in shape, size, and staining properties. They lie within the whorls of spindle cells, and vary from round, to polygonal, to rectangular in shape. [Figs.·4 and 5.] Elongated forms lie in the axis of the containing cords of spindle cells. An occasional bizarre mitotic figure is seen in these larger cells. In some areas the giant cells are quite sparse, but in other areas they are so numerous as to dominate the
Fig. 2.-The photograph of the fixed specimen shows, on th e left, the external surface <•f the two ha lv es after the tumor had been split through th e cente r. Th e lightPr ti ssm• represents chiefly the shrunken pedicle of mucosa. On the right are sef•n the cut s urfn('<·., of the two halves.
Fig. 3.-An area of somewhat loose fibrillar appearance. 1\-lany multinucleated la rge bizarre cells are present In a loose stroma. In the upper left-hand corner is seen a thin walled blood vessel.
138
NOLAN
·\ m.
J.
Obst. & Gyne-c Jan ua ry, 1 9 ~7
Fi g. 4.--An a rea somewha t more fibrou~ in a ppea rance. Many o"f the same biza rre multi nucl eat ed cells are seen interspersed amo ng- spindle cell whorls.
Fig. 5.-High-power view of a portion of the field .
Volume 73 !\umber l
PRIMARY NONPIGMENTED SARCOMA OF VULVA
13!1
microscopic field. The surface of much of the tumor is ulcerated, covered by fibrinopuru· lent exudate, and strikingly vascularized by capillary channels. At the edges, however, the tumor extends to the overlying squamous epithelium. This epithelium, though somewhat hyperplastic, is sharply demarcated from the underlying tumor by its intact basement membrane. With the trichrome stain, all of the fibrillar spi ndle cells or their intracellular ~uhstance stain green, and the cy toplasm of the giant <•ells is somewhat pinkish. No r.r
l''ig. 6.-Sketch showing base of tumor pedicle as represented by the irregular. small, solid are" on the left of the vestibule. The extent of excision is outlined by the dashes.
'' Diagno;;is: Sarcoma. Note: The exact character of this sarcoma cannot Le de· 1e rmincd. It may represent an atypical fibrosarcoma arising in the
140
NOLAN
Am.
J.
Obst. & Gyner. January, 1957
'l'his was dissected out to a uepth of about 1.5 em. in the center (Fig. 6). Examination of this specimen showe1l no evidence of tumor. Follow-up examinations of the patient at frequent intervals have shown no recurrence. The inguinal lymph nodes have be1m negative. Repeated chest x-rays, and two skeletal surveys have shown no evirlence of meta~ tases during the period of sixteen months from the first observation of the tumor. It is of interest to note that her husband gives a history of the removal of a teratoma of the testicle over ten years previously, with no evidence of recurrence. The tissue sections have been examined by Dr. Arthur Hertig and Dr. Emil Novak who considered it to be most likely a fibrosarcoma. Dr. Arthur Purdy Stoutl3 considers the tumor to be a liposarcoma. His comment on the histology may be quoted: "The giant cells with bizarre pyknotic nuclei, the area of myxoid aspect, with a marked degree of vascularity, ancl the presence of occasional partly differentiated lipoblasts in the form of signet ring cells, are sufficient to establish the diagnosis.'' He feels that the tumor belongs to the variety of liposarcomas capable of metastasizing.
Comment It is evident that if this tumor were present before the onset of the pregnancy, it must have been very small. Its rapid growth during the last trimester is probably coincidental, and unrelated to gestation. That sarcomatous change can occur in an ordinary lipoma or fi.broma has been shown in well-documented cases. 9 • 12 However, the likelihood that this neoplasm was probably malignant in type from its very beginning is indicated by the rapid growth from an unnoticeable size.
Summary A case is presented of sarcoma of the vulva, originating in the vestibule at the base of the hymenal ring, in a 23-year-old white woman, during the last trimester of pregnancy. A fourteen-month follow-up has shown her to be symptom free. The pathology is described. The literature on primary nonpigmented sarcoma of the vulva is reviewed. I wish to express my thanks to Dr. Albert M. Deal of Statesboro, Georgia, and to Dr. Henry C. Frech of Savannah, Georgia, for their most helpful cooperation in following this patient.
References 1. 2w 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13. 14. 15.
Amolsch, A. L.: Arch. Path. 24: 777, 1937. Bernstein, P.: Am. J. Surg. 45: 591, 1939. Bell, W. B.: J. Obst. & Gynaec. Brit. Emp. 12: 275, 1907. Buckingham, J. C., and McClure, J. H.: Obst. & Gynec. 6: 138, 1955. Hauptman, H., and Taussig, F . .T.: .T. Pediat. 16: 350, 1940. Kehrer, E.: Veit-Stoekel Handbuch der Gynakologie, ed. 5, Munich, J. F. Bergmann, part 1, pp. 496-503. Keller, J.: Canad. M.A. J. 64: 534, 1951. Morgan, H. S.: AM. J. 0BST. & GYNEC. 15: 861, 1928. Nelson, H. M.: AM. J. OBST. & GYNEC. 25: 594, 1933. Sacrez, R., Kern, P., and Deltombe, J.: Arch. fran<;. de pediat. 8: 202, 1951. Service, A. C., and Derbyshire, R. C.: J. Pediat. 29: 228, 1946. Sternberg, S. S.: Cancer 5: 975, 1952. Stout, A. P.: Ann. Surg. 119: 86, 1944. Taussig, F. J.: Diseases of the Vulva, New York, 1923, D. Appleton Company, p. 3. Taussig, F . .J.: AM . .J. 0BST. & GYNEC. 33: 1017, 1937.
P. NoLAN, M.D., NEW YoRK, N.Y.
the Obstetrical and Gynecological Service [Third Surgical Division], Bellevue Hospital, and the Department of Obstetrics and Gynecology, New York Unive1·sity College of Medicine)
nRIMARY nonpigmented sarcoma of the vulva is an extremely rare type of tumor, as sho~n by the sparsity of cases in the literature, and the brief discussion allotted to it in textbooks. Certainly, it is an exceedingly rare complication of pregnancy. Any type of maligant growth of the vulva is unusual during the childbearing age, perhaps the most common such tumor being the melanoma. Because of the location of the lesion in the case presented here, an anatomical definition of the term, vulva, wili be mentioned. Taussig,' 4 in his monograph on the diseases of the vulva, quotes Poirer and Charpy as follows: ''The vulva can, therefore, be considered to include the following structures: the labia majora and minora, the mons veneris, the perineum, the clitoris and its folds, the Bartholin's glands, thr vestibule of the vagina, the meatus of the urethra, and the hymen.'' A review of the literature on primary nonpigmented sarcoma of the vulva shows no recent cases complicating pregnancy. One of the E-arliest collections of cases of vulvar sarcoma was by Blair BelP in 1907, who found only 6 in patients under 30 years of age out of a total of 18 cases. In only one instance was the tumor in the region of the vestibule. Kehrer, 6 in 1929, rrferred to 7i cases of primary sarcoma of the vulva, which was apparently all he was able to collect from the literature. In 1937, Taussig 15 reported a case of liposarcoma of the labium majus occurring in a 29-year-old primipara. There was no relation to pregnancy. The tumor, although present as a small lump for about four years, had grown suddenly larger in a six-month period. IJocal excision was followed six weeks later by a unilateral vulvectomy and inguinal lymph node dissection. The patient died with widespread metastases about six months after the original enucleation of the tumor. Taussig 15 suggested that, with modern staining methods, a few of thr eases reported as other types of sarcoma in the older liter·ature may have shown evidence of arising from the fatty tissue of the vulva. He quoted several characteristics of liposareoma in general as describe(! by Shaw in 19;{6. Included are the facts that they may grow slowly for several years, with a sudden increase in size; that the encapsulation does not suggest malignancy; and that they have a varied hisologic structure.
r
134
\"olumc73
Number I
PRIMARY NONPIGMENTED SARCOMA OF VULVA
135
In 1928, a case of unpigmented sarcoma of the labium majus in a girl of sixteen years was reported. 8 It was of the alveolar round-cell type. There were widespread metastases less than four months after the tumor was originally noted. In 1937, congenital myxosarcoma of the left labium majus was described in an infant. 1 Death occurred within a few months. There were mr•tastases to the lungs and the mediastinal lymph nodes. In 1939, a case of primary sarcoma of the vulva was reported by Bernstein 2 in a 45-year-ohl woman. It had grown slowly over a sixteen-year period to the size of a gr·apefruit. It was treated by local excision follow<'d hy radiation. A fouryear follow-up showed no evidence of recurr<'llt<'. In 1940, a case of fibr·osarcoma of the vulva in a 7-month-old infant was d<>scrilwd by Hauptman and Taussig. 5 Local excision was carried out, followed later by radiation to local r·ef•.urrences and inguinal metastases. but the infant died with prlvic tumor masses about a year later. In the recent literature only a few cases ar·e reported. Servir·e and Derbyshirc11 described in 1946 a pedunculated fibromyxosarcoma of the left labium minor in a 3-year-old girl. A local wide excision was performed. Five and one-half months later, metastases were found in the right inguinal lymph nodes. Keller, 7 in 1951, reported a large fibrosarcoma of the left labia in a G8-ycar-old patient. Sacrez and associates/ 0 also in 1951, reported a casf' of rhabdomyoblastic sarcoma of the vulva in a 4-month-old infant. Hr· believNI this to h<' congenital in origin. Recently, in 19!15, Buckingham and McClure' r·cported a reticulum-cell sarcoma of the vulva in a 33-year-old patient, which had grovv·n to a considerable size in a period of a fevv W\l(lks. She was treated hy loral exeision of the tumor, followed by radiation tht>rapy. Th<' patient was asymptomatic up to six months later, althoug-h rE'current masses w<>rE' felt ill the pf'lvis at this time. EvE'n from this small group of cases, it is t'vident that the age incidt>rw• nf this type of vulvar tumor is very wide, from birth to old age, and that the prognosis is poor at any age. A 23-year·old married white primigravida, a nur~e, was first seen on July 30, 1954, when she was admitterl to the Obstetrical Service with a diagnosis of acute pyelonephritis in the seventh month of pregnancy. She improved rapidly on antibiotic therapy anrl was discharged, asymptomatic, on Aug. 10, 1954. At the time of that arlmission, the patient offerer! no complaint of any growth of the ,-agina or the vulva, and none was noted on the physical examination. She was subsequently followed in the prenatal clinic, where a routine pelvic examination on Aug. 17, 1954, did not reveal any vaginal or vulvar growth. The patient later stated that she had noted a small mass, about the size of a pea, on the left side of the introitus a few tlays afterward. She first complained about it on Sept. 7, 1954, during a visit to the prenatal clinic. It was slightly larger at this time, and on examination it was considered to be merely a small cyRt arising from the left side of the vestibule. Since the estimated date of confinement was Sept. 17, 1954, only ten da~·s away, it was thought advisable to "?fait and remove it at a later date if necessary. The 1na:..;s giew rapidly over the next few~ weeks, but apparently caused no symptoms. No further note was made of it. On Oct. ·L 1954, twenty-seven •lays after the patient had first complained of the nodule, and approximately six weeks after she had first noted it, she was admitted to the ho8pital in acti \·e labor.
136
NOLAN
i\m . .J. Obst. & Gynec. January, 1917
Arising from near the lower portion of the inner margin of the left labium minor, was a nontender, pedunculated, moderately firm tumor (Fig. 1). It was oval in shape, and measured about 2.5 by 2.5 by 3.0 em. It was hanging on an elastic, thin pedicle of mucosa which measured about 0.5 em. in diameter and 2.5 em . in length. The color resembled that of the labia minora except for a 1.5 em. area of ecchymosis and ulceration on the anterior surface. The patient was seen shortly after admission by two members of the attending staff, who suggested ren1oval of the n1ass at the time o:f delivery by ligation and transection of the pedicle at its base. The growth was considered to be benign, either a lipoma or a fibroma. After an uneventful labor of eleven hours, a normal spontaneous delivery of a 6 pound, 10 ounce, normal infant occurred over a right mediolateral episiotomy. At delivery, the tumor wa~ removed by double ligation of the baRe of the pedicle
Fig. 1.-Sketch showing tumor hanging from pedicle on left side of the vulva.
with No. 00 chromic catgut sutures, followeu by transection at the base. The specimen was sent to the pathology laboratory for routine examination, the report of which follows: ''Accession No. 5314-54. The specimen received fixed in Bouin 's solution consists of a firm spherical mass averaging 2.5 em, in diameter. The cut surface has a pale fibrous homogeneous appearance. [Fig. 2.] On microscopic examination, the tumor is made up of two types of cells. The more frequent type is a spindle shaped cell ·with a small round ovoid nucleus and indistinct cell borders. These are arranged in interlacing cords and whorls. [Fig. 3.] The second type of cell is an irregular and bizarre giant cell with abunaant sharply aenned, somewhat basophlllC cytoplasm. These latter cells are multinucleate, with nuclei which vary considerably in shape, size, and staining properties. They lie within the whorls of spindle cells, and vary from round, to polygonal, to rectangular in shape. [Figs.·4 and 5.] Elongated forms lie in the axis of the containing cords of spindle cells. An occasional bizarre mitotic figure is seen in these larger cells. In some areas the giant cells are quite sparse, but in other areas they are so numerous as to dominate the
Fig. 2.-The photograph of the fixed specimen shows, on th e left, the external surface <•f the two ha lv es after the tumor had been split through th e cente r. Th e lightPr ti ssm• represents chiefly the shrunken pedicle of mucosa. On the right are sef•n the cut s urfn('<·., of the two halves.
Fig. 3.-An area of somewhat loose fibrillar appearance. 1\-lany multinucleated la rge bizarre cells are present In a loose stroma. In the upper left-hand corner is seen a thin walled blood vessel.
138
NOLAN
·\ m.
J.
Obst. & Gyne-c Jan ua ry, 1 9 ~7
Fi g. 4.--An a rea somewha t more fibrou~ in a ppea rance. Many o"f the same biza rre multi nucl eat ed cells are seen interspersed amo ng- spindle cell whorls.
Fig. 5.-High-power view of a portion of the field .
Volume 73 !\umber l
PRIMARY NONPIGMENTED SARCOMA OF VULVA
13!1
microscopic field. The surface of much of the tumor is ulcerated, covered by fibrinopuru· lent exudate, and strikingly vascularized by capillary channels. At the edges, however, the tumor extends to the overlying squamous epithelium. This epithelium, though somewhat hyperplastic, is sharply demarcated from the underlying tumor by its intact basement membrane. With the trichrome stain, all of the fibrillar spi ndle cells or their intracellular ~uhstance stain green, and the cy toplasm of the giant <•ells is somewhat pinkish. No r.r
l''ig. 6.-Sketch showing base of tumor pedicle as represented by the irregular. small, solid are" on the left of the vestibule. The extent of excision is outlined by the dashes.
'' Diagno;;is: Sarcoma. Note: The exact character of this sarcoma cannot Le de· 1e rmincd. It may represent an atypical fibrosarcoma arising in the
140
NOLAN
Am.
J.
Obst. & Gyner. January, 1957
'l'his was dissected out to a uepth of about 1.5 em. in the center (Fig. 6). Examination of this specimen showe1l no evidence of tumor. Follow-up examinations of the patient at frequent intervals have shown no recurrence. The inguinal lymph nodes have be1m negative. Repeated chest x-rays, and two skeletal surveys have shown no evirlence of meta~ tases during the period of sixteen months from the first observation of the tumor. It is of interest to note that her husband gives a history of the removal of a teratoma of the testicle over ten years previously, with no evidence of recurrence. The tissue sections have been examined by Dr. Arthur Hertig and Dr. Emil Novak who considered it to be most likely a fibrosarcoma. Dr. Arthur Purdy Stoutl3 considers the tumor to be a liposarcoma. His comment on the histology may be quoted: "The giant cells with bizarre pyknotic nuclei, the area of myxoid aspect, with a marked degree of vascularity, ancl the presence of occasional partly differentiated lipoblasts in the form of signet ring cells, are sufficient to establish the diagnosis.'' He feels that the tumor belongs to the variety of liposarcomas capable of metastasizing.
Comment It is evident that if this tumor were present before the onset of the pregnancy, it must have been very small. Its rapid growth during the last trimester is probably coincidental, and unrelated to gestation. That sarcomatous change can occur in an ordinary lipoma or fi.broma has been shown in well-documented cases. 9 • 12 However, the likelihood that this neoplasm was probably malignant in type from its very beginning is indicated by the rapid growth from an unnoticeable size.
Summary A case is presented of sarcoma of the vulva, originating in the vestibule at the base of the hymenal ring, in a 23-year-old white woman, during the last trimester of pregnancy. A fourteen-month follow-up has shown her to be symptom free. The pathology is described. The literature on primary nonpigmented sarcoma of the vulva is reviewed. I wish to express my thanks to Dr. Albert M. Deal of Statesboro, Georgia, and to Dr. Henry C. Frech of Savannah, Georgia, for their most helpful cooperation in following this patient.
References 1. 2w 3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13. 14. 15.
Amolsch, A. L.: Arch. Path. 24: 777, 1937. Bernstein, P.: Am. J. Surg. 45: 591, 1939. Bell, W. B.: J. Obst. & Gynaec. Brit. Emp. 12: 275, 1907. Buckingham, J. C., and McClure, J. H.: Obst. & Gynec. 6: 138, 1955. Hauptman, H., and Taussig, F . .T.: .T. Pediat. 16: 350, 1940. Kehrer, E.: Veit-Stoekel Handbuch der Gynakologie, ed. 5, Munich, J. F. Bergmann, part 1, pp. 496-503. Keller, J.: Canad. M.A. J. 64: 534, 1951. Morgan, H. S.: AM. J. 0BST. & GYNEC. 15: 861, 1928. Nelson, H. M.: AM. J. OBST. & GYNEC. 25: 594, 1933. Sacrez, R., Kern, P., and Deltombe, J.: Arch. fran<;. de pediat. 8: 202, 1951. Service, A. C., and Derbyshire, R. C.: J. Pediat. 29: 228, 1946. Sternberg, S. S.: Cancer 5: 975, 1952. Stout, A. P.: Ann. Surg. 119: 86, 1944. Taussig, F. J.: Diseases of the Vulva, New York, 1923, D. Appleton Company, p. 3. Taussig, F . .J.: AM . .J. 0BST. & GYNEC. 33: 1017, 1937.