- Email: [email protected]
Cervical endometriosis, a lesion of increasing importance
Cervical endometriosis, a lesion of increasing importance HERMAN L. GARDNER, M.D. Houston, Texas
P R r M A R v superficial cervical endometriosis is one of the significant causes of minimal metrorrhagia and is becoming substantially more frequent in occurrence. This increase appears to be etiologically related to the increased use of cervical biopsies, cauterizations, and conizations. That Williams and Richardson t were able to find only 42 reported cases in the world literature before 1955 shows why the lesion had previously been considered extremely rare. Then, their report of an additional 35 cases proved that it was not of such rarity. When this current study was proposed, the group of 31 cases seemed to be reasonably large; however, when, in 1960, Williams 2 reported an additional 76 cases, the present series became less impressive. Material and findings
All patients who were included in this study were white and were taken from a private office practice, during the 5 year period from August, 1955, to August, 1960. All patients with histories of intermenstrual bleeding of any type were carefully screened, and all cervices with lesions the least bit suggestive of endometriosis were studied histologically. All tissues were obtained by punch biopsy techniques in the office. Criteria for diagnosis included the finding of From the Department of Obstetrics and Gynecology, Baylor University College of Medicine. Presented at a meeting of the Texas Association of Obstetricians and Gynecologists, February, 1961.
170
both endometrial glands and endometrial type stroma. Symptoms. Pelvic pain is not a symptom of superficial cervical endometriosis. With these cases, there was only one associated symptom and that was minimal metrorrhagia. Of the 31 patients, 28 had abnormal bleeding of one type or another, either postcoital or spontaneous. Twenty had, on occasion, spotting 3 to 14 days before menstruation; 7 had postmenstrual spotting for periods from 3 to 10 days; and others had erratic spotting. Several patients had all three types of bleeding and, occasionally, a patient reported almost continuous spotting. Williams, in his expanded series of 111 cases published in 1960, found that in 69 patients there was a history of bloody discharge, and Rannei reported that 8 of 16 patients had had intermenstrual bleeding. Gross features. Findings common to aU the lesions observed in this study were superficial submucosal position. failure to form distinct masses (typical of endometriomas), and ectocervical location. The lesions varied considerably in shape and size. Some were macular and some elevated, depending upon cellular and blood capillary activity from hormonal stimulation. For example, macular lesions of a light red color and with poorly defined edges were hardly discernible during the proliferative phase. but they frequently became dark blue, elevated, and enlarged around menstruation. The majority of the lesions consisted mainly of one or more round, slightly elevated, bright red to dark blue areas (Fig. 1) which measured
Volume 84 Number 2
2 to 4 mm. in diameter. Other lesions (Fig. 2) , however, covered larger areas and frequently exhibited bizarre configurations. In 4 patients the entire external os was involved, with irregular peripheral extensions up to 1 em. from the os. This type of lesion occurred only in those patients who had had conizations. Other lesions resembled hemorrhagic blebs or "blood blisters," and in some superficial ulceration was evident. Usually, the overlying squamous epithelium was easily disrupted by a speculum, a wooden spatula, or even a cotton-tipped applicator to cause bleeding. After trauma, some lesions increased in diameter, apparently as the result of submucosal bleeding. No cervices were observed to have "puckered areas" from scarring, and no evidence was found to suggest that the lesion has a predilection for any particular area of the cervix. Microscopic appearance. Although the basic microscopic picture (Fig. 3) of each tissue specimen was identical, the number of glands and the amount of endometrial stroma were highly variable. All lesions were located immediately beneath the stratified squamous epithelium, a finding possibly explained by the method of obtaining specimens. The tissues usually corresponded, at
Fig. 1. Gross, showing several small, slightly elevated lesions of different shades.
Cervical endometriosis 171
Fig. 2. Gross, showing lesion about external os. This type more often seen after conization. Also small papular lesion .
least to a degree, with the phase of the menstrual cycle, thus demonstrating a responsiveness to estrogen-progesterone stimulation. Some lesions appeared to have been mechanically ruptured, while in others maceration of the squamous epithelium from necrosis was evident. Submucosal and intrastromal infiltration with red blood cells was noted more frequently around the menstrual phase, as was an increase in the number of capillaries. Leukocytic infiltration peripheral to and within stroma also was not uncommon. Incidence. While no significant statistics have been compiled as to the numerical incidence of cervical endometriosis, these findings and those of Williams prove its relative frequency. It was of interest that of 52 lesions with gross appearance strongly suggestive of superficial endometriosis 31 were confirmed by histologic study. Diagnosis was established in these cases despite the fact that all tissue specimens were removed by punch biopsy, a technique, which, according to Williams, might result in a negative diagnosis because of extrusion of endometriotic tissue. Williams has also repeatedly emphasized the advantage of examining patients near menstruation. Only a few of the
July 15, 1962 Am. ]. Obst. & Gynec.
172 Gardner
Fig. 3. Endometriosis, demonstrating late secretory stage. Focal hemorrhage beneath squamous epithelium and within gland lumen.
patients were examined at the more propitious time, so undoubtedly some cases escaped detection. Etiology
There is convincing evdence that the occurrence of this lesion is closely related to cervical trauma. On analysis of the 31 case histories, it was learned that 29 of the patients had been previously subjected to a total of 74 traumatizing procedures upon the cervices, including 31 cauterizations, 16 biopsies, 13 conizations, and 11 curettages. Two patients who cited no history of surgical trauma had borne one or more children. In Ranney's report, 12 of 16 patients had had cervical operative procedures with direct trauma, and Williams stated that 106 of 111 patients had borne children and that 74 gave a history of other trauma. I am unable to relate any of these reported cases to the direct extension of endometrial tissue from the endometrial cavity to extension of endometriosis from the cul-desac or to cervical adenomyosis. To attribute any of these 31 cases to metaplasia of cervical epithelium would seem nebulous reasoning. Wolfe and associates/ in a recent report of 17 cases, attributed 2 cases to metaplasia, 2 to "developmental ectopia," 1 to
"heterotopic fault," and 5 to hematogenous spread. Their sampling of cases is hardly representative, however, because several were found in the endocervices of removed uteri. Because of the infrequency of this condition in the previously untraumatized cervix, the only reasonably valid explanation for development in the majority of cases is the transplantation of viable endometrial cells upon traumatized areas of the cervix. Treatment
Patients can be treated by simple excision of the lesions, by means of the punch biopsy instrument, the cold knife, or the loop electrode. Destruction by cauterization or coagulation is also useful, particularly when employed as an adjunct to excision. Several of the patients developed recurrences after the initial treatment, but, admittedly, the majority were not treated vigorously. Comment
These observations and those of others afford additional support for Sampson's theory of implantation, already confirmed by the experimental work of TeLinde and Scott,5 Scott and associates,6 and others. 7 • 8 That these endometrial implants can exhibit the usual response to estrogen-proges-
Volume 84 Number 2
terone stimulation is evidenced by the gross changes in individual lesions during different phases of the menstrual cycle; by their unusual susceptibility to rupture and trauma near menstruation; by the close correlation of their histologic appearance with the phase of the menstrual cycle; and by the decidual reaction noted during pregnancy. Nine of these 31 patients were subsequently observed during a pregnancy, 8 of whom showed typical deciduomas involving identical areas of the cervix as were involved by endometriosis. There appears to be more than a casual etiological relationship between cervical endometriosis and cervical trauma. The common practice of performing cauterizations immediately after menstruation is possibly conducive to the disease, because, when menstruation occurs 2 or 3 weeks later, the slough has disappeared and granulation tissue has usually formed to provide a healthy nidus for desquamated endometrial cells. Perhaps some thought should be given to the "optimal time" when diagnostic and therapeutic procedures can be performed with a minimal risk of implantation. Cervical endometriosis is not a serious or painful disorder, and in the majority of cases the lesions are small. The failure of these superficial lesions to form large and typical endometriomas is easily understood with the realization that they are superficial and spill their contents readily through the easily ruptured squamous epithelium. The published consensus is that proof of this lesion depends upon the demonstration
REFERENCES
1. Williams, G. A., and Richardson, A. C.: Obst. & Gynec. 6: 309, 1955. 2. Williams, G. A.: AM. J. 0BST. & GYNEC. 80:
734, 1960.
3. Ranney, B., and Chung, J. T.: AM. J. 0BsT. & GYNEC. 64: 1333, 1952. 4. Wolfe, S. A., Mackles, A., and Greene, H. J.: AM. J. 0BST. & GYNEC. 81: 111, 1961.
Cervical endometriosis 173
of both endometrial glands and endometrial stroma; however, it might be argued that a grossly characteristic lesion which yields an abundance of typical endometrial stroma should also be considered indicative of the disease. Summary and conclusions 1. Cervical endometriosis is a frequent cause of persistent minimal metrorrhagia and should be suspected when this symptom is present. 2. The etiological relationship between superficial cervical endometriosis and cervical trauma is further elucidated by the finding of 29 of 31 patients with histories of surgical trauma. 3. This and similar reports offer further support of Sampson's theory of implantation of desquamated endometrial cells as a cause of endometriosis. 4. It is postulated that this lesion will be observed frequently in the practice of every gynecologist who is aware of its existence and whose consultant in pathology maintains a comparable degree of suspicion. 5. The findings of this study indicate that, during pregnancy, cervical deciduomas are likely to develop at the site of cervical endometriosis.
Addendum. Since the period covered by the manuscript (up to August, 1960), I have seen 9 additional cases of cervical endometriosis in private office practice. All of these patients had had minor surgical procedures such as cervical biopsy, cauterization, and conization.
5. TeLinde, R. W., and Scott, R. B.: AM. J. 0BST. & GYNEC. 60: 1147, 1950. 6. Seott, R. B., TeLinde, R. W., and Wharton, L. R.: AM. J. OssT. & GYNEC. 66: 1082, 1953. 7. Ridley, J. H., and Edwards, I. K.: AM. ]. 0BST. & GYNEC. 76: 783, 1958. 8. Fuchs, H.: Zentralbl. Gynlik. 59: 914, 1935. 6410 Fannin St. Houston 25, Texas
P R r M A R v superficial cervical endometriosis is one of the significant causes of minimal metrorrhagia and is becoming substantially more frequent in occurrence. This increase appears to be etiologically related to the increased use of cervical biopsies, cauterizations, and conizations. That Williams and Richardson t were able to find only 42 reported cases in the world literature before 1955 shows why the lesion had previously been considered extremely rare. Then, their report of an additional 35 cases proved that it was not of such rarity. When this current study was proposed, the group of 31 cases seemed to be reasonably large; however, when, in 1960, Williams 2 reported an additional 76 cases, the present series became less impressive. Material and findings
All patients who were included in this study were white and were taken from a private office practice, during the 5 year period from August, 1955, to August, 1960. All patients with histories of intermenstrual bleeding of any type were carefully screened, and all cervices with lesions the least bit suggestive of endometriosis were studied histologically. All tissues were obtained by punch biopsy techniques in the office. Criteria for diagnosis included the finding of From the Department of Obstetrics and Gynecology, Baylor University College of Medicine. Presented at a meeting of the Texas Association of Obstetricians and Gynecologists, February, 1961.
170
both endometrial glands and endometrial type stroma. Symptoms. Pelvic pain is not a symptom of superficial cervical endometriosis. With these cases, there was only one associated symptom and that was minimal metrorrhagia. Of the 31 patients, 28 had abnormal bleeding of one type or another, either postcoital or spontaneous. Twenty had, on occasion, spotting 3 to 14 days before menstruation; 7 had postmenstrual spotting for periods from 3 to 10 days; and others had erratic spotting. Several patients had all three types of bleeding and, occasionally, a patient reported almost continuous spotting. Williams, in his expanded series of 111 cases published in 1960, found that in 69 patients there was a history of bloody discharge, and Rannei reported that 8 of 16 patients had had intermenstrual bleeding. Gross features. Findings common to aU the lesions observed in this study were superficial submucosal position. failure to form distinct masses (typical of endometriomas), and ectocervical location. The lesions varied considerably in shape and size. Some were macular and some elevated, depending upon cellular and blood capillary activity from hormonal stimulation. For example, macular lesions of a light red color and with poorly defined edges were hardly discernible during the proliferative phase. but they frequently became dark blue, elevated, and enlarged around menstruation. The majority of the lesions consisted mainly of one or more round, slightly elevated, bright red to dark blue areas (Fig. 1) which measured
Volume 84 Number 2
2 to 4 mm. in diameter. Other lesions (Fig. 2) , however, covered larger areas and frequently exhibited bizarre configurations. In 4 patients the entire external os was involved, with irregular peripheral extensions up to 1 em. from the os. This type of lesion occurred only in those patients who had had conizations. Other lesions resembled hemorrhagic blebs or "blood blisters," and in some superficial ulceration was evident. Usually, the overlying squamous epithelium was easily disrupted by a speculum, a wooden spatula, or even a cotton-tipped applicator to cause bleeding. After trauma, some lesions increased in diameter, apparently as the result of submucosal bleeding. No cervices were observed to have "puckered areas" from scarring, and no evidence was found to suggest that the lesion has a predilection for any particular area of the cervix. Microscopic appearance. Although the basic microscopic picture (Fig. 3) of each tissue specimen was identical, the number of glands and the amount of endometrial stroma were highly variable. All lesions were located immediately beneath the stratified squamous epithelium, a finding possibly explained by the method of obtaining specimens. The tissues usually corresponded, at
Fig. 1. Gross, showing several small, slightly elevated lesions of different shades.
Cervical endometriosis 171
Fig. 2. Gross, showing lesion about external os. This type more often seen after conization. Also small papular lesion .
least to a degree, with the phase of the menstrual cycle, thus demonstrating a responsiveness to estrogen-progesterone stimulation. Some lesions appeared to have been mechanically ruptured, while in others maceration of the squamous epithelium from necrosis was evident. Submucosal and intrastromal infiltration with red blood cells was noted more frequently around the menstrual phase, as was an increase in the number of capillaries. Leukocytic infiltration peripheral to and within stroma also was not uncommon. Incidence. While no significant statistics have been compiled as to the numerical incidence of cervical endometriosis, these findings and those of Williams prove its relative frequency. It was of interest that of 52 lesions with gross appearance strongly suggestive of superficial endometriosis 31 were confirmed by histologic study. Diagnosis was established in these cases despite the fact that all tissue specimens were removed by punch biopsy, a technique, which, according to Williams, might result in a negative diagnosis because of extrusion of endometriotic tissue. Williams has also repeatedly emphasized the advantage of examining patients near menstruation. Only a few of the
July 15, 1962 Am. ]. Obst. & Gynec.
172 Gardner
Fig. 3. Endometriosis, demonstrating late secretory stage. Focal hemorrhage beneath squamous epithelium and within gland lumen.
patients were examined at the more propitious time, so undoubtedly some cases escaped detection. Etiology
There is convincing evdence that the occurrence of this lesion is closely related to cervical trauma. On analysis of the 31 case histories, it was learned that 29 of the patients had been previously subjected to a total of 74 traumatizing procedures upon the cervices, including 31 cauterizations, 16 biopsies, 13 conizations, and 11 curettages. Two patients who cited no history of surgical trauma had borne one or more children. In Ranney's report, 12 of 16 patients had had cervical operative procedures with direct trauma, and Williams stated that 106 of 111 patients had borne children and that 74 gave a history of other trauma. I am unable to relate any of these reported cases to the direct extension of endometrial tissue from the endometrial cavity to extension of endometriosis from the cul-desac or to cervical adenomyosis. To attribute any of these 31 cases to metaplasia of cervical epithelium would seem nebulous reasoning. Wolfe and associates/ in a recent report of 17 cases, attributed 2 cases to metaplasia, 2 to "developmental ectopia," 1 to
"heterotopic fault," and 5 to hematogenous spread. Their sampling of cases is hardly representative, however, because several were found in the endocervices of removed uteri. Because of the infrequency of this condition in the previously untraumatized cervix, the only reasonably valid explanation for development in the majority of cases is the transplantation of viable endometrial cells upon traumatized areas of the cervix. Treatment
Patients can be treated by simple excision of the lesions, by means of the punch biopsy instrument, the cold knife, or the loop electrode. Destruction by cauterization or coagulation is also useful, particularly when employed as an adjunct to excision. Several of the patients developed recurrences after the initial treatment, but, admittedly, the majority were not treated vigorously. Comment
These observations and those of others afford additional support for Sampson's theory of implantation, already confirmed by the experimental work of TeLinde and Scott,5 Scott and associates,6 and others. 7 • 8 That these endometrial implants can exhibit the usual response to estrogen-proges-
Volume 84 Number 2
terone stimulation is evidenced by the gross changes in individual lesions during different phases of the menstrual cycle; by their unusual susceptibility to rupture and trauma near menstruation; by the close correlation of their histologic appearance with the phase of the menstrual cycle; and by the decidual reaction noted during pregnancy. Nine of these 31 patients were subsequently observed during a pregnancy, 8 of whom showed typical deciduomas involving identical areas of the cervix as were involved by endometriosis. There appears to be more than a casual etiological relationship between cervical endometriosis and cervical trauma. The common practice of performing cauterizations immediately after menstruation is possibly conducive to the disease, because, when menstruation occurs 2 or 3 weeks later, the slough has disappeared and granulation tissue has usually formed to provide a healthy nidus for desquamated endometrial cells. Perhaps some thought should be given to the "optimal time" when diagnostic and therapeutic procedures can be performed with a minimal risk of implantation. Cervical endometriosis is not a serious or painful disorder, and in the majority of cases the lesions are small. The failure of these superficial lesions to form large and typical endometriomas is easily understood with the realization that they are superficial and spill their contents readily through the easily ruptured squamous epithelium. The published consensus is that proof of this lesion depends upon the demonstration
REFERENCES
1. Williams, G. A., and Richardson, A. C.: Obst. & Gynec. 6: 309, 1955. 2. Williams, G. A.: AM. J. 0BST. & GYNEC. 80:
734, 1960.
3. Ranney, B., and Chung, J. T.: AM. J. 0BsT. & GYNEC. 64: 1333, 1952. 4. Wolfe, S. A., Mackles, A., and Greene, H. J.: AM. J. 0BST. & GYNEC. 81: 111, 1961.
Cervical endometriosis 173
of both endometrial glands and endometrial stroma; however, it might be argued that a grossly characteristic lesion which yields an abundance of typical endometrial stroma should also be considered indicative of the disease. Summary and conclusions 1. Cervical endometriosis is a frequent cause of persistent minimal metrorrhagia and should be suspected when this symptom is present. 2. The etiological relationship between superficial cervical endometriosis and cervical trauma is further elucidated by the finding of 29 of 31 patients with histories of surgical trauma. 3. This and similar reports offer further support of Sampson's theory of implantation of desquamated endometrial cells as a cause of endometriosis. 4. It is postulated that this lesion will be observed frequently in the practice of every gynecologist who is aware of its existence and whose consultant in pathology maintains a comparable degree of suspicion. 5. The findings of this study indicate that, during pregnancy, cervical deciduomas are likely to develop at the site of cervical endometriosis.
Addendum. Since the period covered by the manuscript (up to August, 1960), I have seen 9 additional cases of cervical endometriosis in private office practice. All of these patients had had minor surgical procedures such as cervical biopsy, cauterization, and conization.
5. TeLinde, R. W., and Scott, R. B.: AM. J. 0BST. & GYNEC. 60: 1147, 1950. 6. Seott, R. B., TeLinde, R. W., and Wharton, L. R.: AM. J. OssT. & GYNEC. 66: 1082, 1953. 7. Ridley, J. H., and Edwards, I. K.: AM. ]. 0BST. & GYNEC. 76: 783, 1958. 8. Fuchs, H.: Zentralbl. Gynlik. 59: 914, 1935. 6410 Fannin St. Houston 25, Texas