Non-puerperal inversion of the uterus

NON~PUERPERAL

INVERSION

HOWARD W.

JONES, JR.,

OF THE UTERUS* M.D.

Baltimore, Maryland

I

NVERSION of the uterus is not common in obstetric practice and is indeed a rarity for the gynecoIogist. The purpose of this paper is to emphasize the gynecoIogic as distinct from the obstetric knowIedge of this condition by summarizing the Iiterature and reporting a most extraordinary example of this condition. Inversion of the uterus may be classihed in two groups: (I) puerpera1 or obstetric, (a) acute, (b) chronic; (2) non-puerperal or gynecoIogic, (a) tumor-produced, (b) idiopathic. PuerperaI inversion is associated with abortion, miscarriage or term debvery, non-puerpera inversion occurs in a non-pregnant uterus. In the puerpera1 variety acute inversion is considered to occur within thirty days of the termination of pregnancy, chronic after that. In the non-puerperal variety Buseyl suggests the term, sudden and gradua1 for acute and chronic, whereas Winke12 considers a11 nonpuerpera1 cases as chronic. Because the thirtyday Iimit serves no usefu1 purpose in the nonpuerperal variety of inversion, it may be better to emphasize etioIogy by cIassifying these cases into tumor-produced and idiopathic while realizing that either may sometimes present themseIves as an acute emergency. Non-puerperal, tumor-produced inversion is associated with a neopIasm of the body ofthe uterus whiIe the idiopathic variety has no recognizabIe cause. This cIassification may be considered a modification from Mante13 who used obstetric and surgica1 types of inversion rather than puerpera1 and non-puerpera1. However, he thought that surgica1 was aIways associated with tumors. Provision must be made in the cIassification for the idiopathic non-puerpera1 variety aIthough there seems to be but a singIe case reported in the Iast thirty-eight years. The German Iiterature has referred to onkogenetic inversion, but the term tumor-produced has been used instead. An exact transIation of onkogenetic inversion wouId indicate a tumor produced by inversion whereas the reverse is intended. * From the Department

of Gynecology

The non-puerpera1 variety of inversion is Ieast common and for the most part has received attention onIy incidenta to the more common variety. Das4 in 1940 was abIe to coIIect 391 cases of inversion of which about fifty-eight or 13 per cent were non-puerpera1. CASE

REPORT

This patient, No. 426410, gave a history of having a norma menopause in 1913 at the age of fifty-three. FoIlowing this there was no further vagina1 bleeding unti1 December, 1946, after which there was continuous vagina1 spotting. On June 23, 1947, the patient was admitted for diIation and curettage. She was then eighty-seven years old. Examination at that time showed a cervica1 OS diIated by the protrusion of a necrotic poIypoid mass. Examination of the uterus was unsatisfactory but it was thought to be at Ieast two times its normal size but movabIe. Because of the patient’s advanced age no anesthetic was used for the endometria1 curettage. A pathologic examination reveaIed a Iow grade sarcoma probabIy of endometrial origin. AIthough a genera1 medica evaIuation showed the patient to be somewhat feebIe, no serious abnormality was demonstrated. Nevertheless, a decision was made to use intrauterine radium as the onIy therapy. AccordingIy on JuIy 24, 1947, she received 3,375 mg. hours intrauterine radium by three 29 mg. tandem tubes. FoIIowing this she was observed from time to time in the outpatient department. There were no symptoms but on severa occasions it was noted that the uterus was about four times its normaI size. On August 16, 1948, it was noted that the Iower abdomina1 mass was much smaIIer and that there was a Iarge papiIIary mass in the vagina. This was thought to be a papiIIary hbrosarcoma projecting through the cervix and she was admitted to the hospita1 for a vaginal remova of this tumor. A genera1 medica survey at that time showed arterioscIerotic heart disease with minimal cardiac faiIure. She was digitaIized prior to the

of the Johns Hopkins Hospital and University,

492

American

BaItimore,

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Md.

of Surgery

Jones-Inversion operation which took pIace on August 23, 1948. IJnder pentothal anesthesia@ it was noted that :I papiIIary necrotic tumor arose from the region of the cervix and filIed the vagina. The cervix could not be identified. The fundus was noted to be about normaI in size or somewhat below normaI. As the pedicIe of the tumor was large, it was grasped with a kidney pedicle clamp and severed. Examination of the specimen revealed that the amputation had removed the body of the uterus which was inverted. Tumor was stiI1 present in the stump of the uterus remaining high in the vagina. (Fig. I.) As the patient was in good condition, an immediate tota abdomina1 hysterectomy and biIatera1 saIpingo-oophorectomy was undertaken. The technic of this operation varied in important detaiIs from a tota hysterectomy in a non-inverted uterus and is iIIustrated and described. (Fig. 2.) AI1 visibIe neopIastic tissue was removed. Abdominal rather than vagina1 hysterectomy was selected because a Iarge amount of tumor remained in the vagina1 vauIt and it was judged that the operative procedure in its presence wouId be technicaIIy diffrcuIt. Furthermore the Iarge diameter of the cervix made it at once apparent that the ureters were in cIose proximity. The patient was out of bed on the day of operation and had a remarkably smooth postoperative course. She was discharged on the eIeventh postoperative day. PathoIogic examination revealed sarcoma of the uterus with extension to the cervix. The tubes and ovaries showed no growth. When Iast heard from on May I, 1950, she was enjoying her former fair state of heaIth and had passed her ninetieth birthday. Inversion has been described as compIete or incompIete. According to Thorn5 if any part of the fundus passes through the cervical OS the inversion is called complete. He states that 75 per cent of tumor-produced inversions are compIete. In Das’ coIIection 92 per cent were compIete whiIe a11 the more recent cases reported have been complete. The diagnosis of incomplete non-puerperal inversion is attended with obvious difhculties. For practica1 purposes a11 Important cases of non-puerperal inversion may be considered as complete. Inversion of the vagina and proIapse of the uterus have been reported associated with ,VlCl~‘, I951

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uterine inversion. The patient whose case was reported herein is an example of complctc, tumor-produced, inversion of the uterus. EtioZo,q-. Idiopathic non-pucrperal invcrsion is, as the name implies, not associated with an4 demonstrabIe abnormality. Thorn5 MEDIAN

SECTION

INVERSION OF UTERUS

SHOWING

FIG. I. Semi-diagrammatic median section showing the inversion of the uterus associated with endomctrial sarcoma. was abIe to collect thirteen cases and there has been apparently onIy one case6 reported in the Iast thirty-eight years. Tumor-produced, nonpuerperal inversion is most often associated with submucous uterine fibroids but has been reported with carcinoma of the body of the uterus and sarcoma of the uterus. Das in 1940 coIIected forty-seven cases due to fibroids, four due to sarcoma and three due to carcinoma, of which one was an adeno-acanthoma. Since his report an additiona case has appeared due to fibroids7 and one due to sarcoma! The present case is therefore the sixth patient to be recorded with a sarcoma. SJ’mptoms and Diagnosis. In non-puerperaI inversion the accident of inversion is usually diffIcuIt to recognize even in retrospect. The idiopathic variety in which the accident is often abrupt may be an exception and accompanied with pain and shock as in the case reported by Aronson and Karen.6 For the most part the tumor-produced variety is associated with abnorma1 vagina1 bleeding and a vagina1 mass. As wouId be expected the non-puerperal variety occurs most often in oIder women. This is in contrast to the puerpera1 variety in which according to Das over haIf the cases occur in

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Jones-Inversion

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FIG. 2. Total hysterectomy and biIatera1 salpingo-oophorectomy for tumor produced non-puerperal (gynecologic) inversion of the uterus. Upper: the findings at operation; the round ligaments have been severed and the right infundibuIopelvic ligament is being severed. Lower Ieft: Because the bladder may be invoIved in the inversion, care must be exercised in denuding the bladder peritoneum; it shouId be severed as far within the inverted uterus as possibIe. Lower right: the inverted uterus presents a mass of much greater diameter than a normal cervix; for this reason the ureters are in close proximity. The T-shaped inversion in the pubocervica1 fascia, as recommended by Richardson, is particuIarIy desirabIe as this aIIows the ureters to drop away from the immediate operative area; after the specimen has been removed the operation is compIeted as in usua1 tota hysterectomy.

patients under the age of thirty. Of the women in Das’ non-puerperai series 21 per cent were between thirty-one and forty, zg per cent between forty-one and fifty and 25 per cent between fifty-one and sixty. The youngest patient was sixteen years oId and the oIdest seventy-nine years old. Our patient, therefore, exceeds by ten years the oIdest previously reported case. If the pelvic examination is carefuIIy made and the findings properly interpreted, the diagnosis of inversion shouId be easily made. However, in most instances incIuding our own the possibility is not entertained, and the diagnosis is made onIy at operation. In severa

reported instances and in the case reported the diagnosis was not recognized until the inverted body of the uterus was amputated and the peritoneal cavity thereby opened in an attempt to remove what was thought to be a submucous myoma or large endometria1 polyp. The history or record of an abdomina1 tumor which has diminished in size, the presence of a vagina1 tumor, the absence of a cervica1 ring, inability to sound the uterine cavity and the cup-shaped or apparent absence of the uterine body shouId make the diagnosis very cIear if the possibiIity is kept in mind. A differentia1 diagnosis wiI1 usuaIly Iie between a submucous vagina1 myoma without American

Journal

of Surgery

Jones-Inversion inversion and the same tumor with inversion. The diagnosis should he considered in casesof vagina1 submucous myoma, especiaIIy in the older age group. Treatment. Non-puerperal inversion of the uterus is too rare and experience with this condition has accumuIated over too many years to render statistical tabuIations of value in determining the best method of therapy. Suffke it to say that vagina1 hysterectomy, abdominal hysterectomy, vagina1 excision of the tumor and reposition of the uterus both spontaneousIy and manuaIIy and combinations of these procedures have been used. The mortaIity has been surprisingIy Iow. Das coIIected twenty-two cases of vagina1 hysterectomy with two deaths, abdomina1 hysterectomy in two cases without mortaIity and vagina1 excision of the tumor foIIowed with spontaneous reduction in three cases without mortality. It is cIear that with non-puerpera1 inversion associated with submucous myomas hysterectomy may not be necessary if the peritonea1 cavity is not opened. If the cavity is opened, either vagina1 or abdomina1 hysterectomy becomes necessary. In inversion associated with maIignant tumors a radical operation may be indicated. The technic of tota abdomina1 hysterectomy in uterine inversion varies from the usua1 hysterectomy in important aspects as indicated in the accompanying figure. Most patients with idiopathic non-puerpera1 inversion have been treated with hyster-

May,

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ectomy; but if such a diagnosis couId be made with assurance, reposition might be possible. SUMMARY

Non-puerpera1 inversion of the uterus occurs most frequentIy in the oIder age group in contrast to puerpera1 inversions. The tumorproduced variety has been reported associated with submucous fibroids, endometrial carcinoma or sarcbma of the uterus. The diagnosis shouId be cIear aIthough it is seIdom made preoperativeIy. An extraordinary exampIe of this condition associated with sarcoma of the uterus is presented with the technic of tota abdomina1 hysterectomy used in the presence of inversion. REFERENCES I. BUSEY, S. C.

2. 3.

4.

5. 6.

7. 8.

Chronic inversion of the uterus. In: A

System of Gynecology and Obstetrics. Edited by Mann and Hurst. Edinburg, I 889. WINKEL, F. Handbuch der GeburtshuIfe. Wiesbaden, I 904. MANTEL, P. Un cinquieme cas d’inversion uterin, traite avec succes par I’application du baloon de M. Champetier de Ribes go&e avec de I’air. Ann. de gyntc. et d’obst., 3: 257, 1906. DAS, P. Inversion of the uterus. J. Obst. e’~ Gynec. Brit. Emp., 47: 525, 1940. THORN, W. Zur inversio uteri. Samml. Klin. Vort., 1gr I. SELKIN-ARONSON, E. and KAREN, B. Nonpuerperal inversion of the uterus. Am. J. Obst. C* Gynec., 42: 54% 1941. CANIZARES VERSON, R. Inversion due to hbroma of fundus. Arch. cubanos cancerol., I : 264, 1942. REICH, W. J. and NECHTON, M. J. Chronic inversion with fibrosarcoma of corpus. Am. J. h-g., 71: 710, 1946.