,.. s. No. Year Author Incidence. ADENOMYOSIS (A Review of 73 Cases)

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1 (A Review of 73 Cases) by VINAYA PENDSE M.S. Adenomyosis is a pathological condition in which the endometrial glands and stroma are found in the mtyometrium separate from the basal layer of the endometrium. This condition was first recognised by Rokitansky in 1860> while the first monograph of its clinical and pathological features was published by Von Recklinghausen in Major contribution to this subject was that of Cullen in He studied the pathological process and recognised the localised and the generalised variety and named these as ade- nomyoma and adenomyosis respectively. In the beginning of this century adeno - TABLE I myosis was considered to be a rarity since only a few clinicians recognised it as an entity. Adenomyosis is not as uncommon in our country as indicated by the paucity of the published reports (Hilda 19>50; Bhatt 1960; and Rosario 1968). The present paper is based on a clinical study of 73 cases of adenomyosis encountered at Zenana Hospital Jaipur from to Incidence The final diagnosis of adenomyosis is always histopathological after hysterectomy. Out of 668 hysterectomies done for Showing Incidence of Adenomyosis as Reported by Various Authors s. No. Year Author Incidence Cullen 5.00% McCarty and Blackm<Jn 5.43% Westman 8.00% Brines ei ell. ]0.7% Dreyfuss 8.10o/o Crossen and C! ossen 20.3% Hunter et al. 37.8% Benson and Sneedet\ 21.4% Israel and W ontersz 10.0% Bhatt 6.00% Emge 15.0% Weed 6.00% Rosario 8.50% Reader in Gyn. & Obst. S.M.S. Medical College Jaipur. *Present address: Reader in Gyn. & Obst. R. N. T. Medical College Udaipur. Received for pnblicat'ion un various condition's adenomyosis was found in 73 specimen an incidence of 10.9 per cent. The incic1ence of adenomyosis as reported in the literature shows wide variation as can be made out from Table I.....

2 401.. The lowest incidence is that given by Cullen and highest is that given by Hunter et al (1947). Incidence in the present study is comparable with that given by Israel and Wontersz (1959) and Brines et al (1943). The difference in the incidence of ade nomyosis as reported by various workers may be accounted for by the fact that slight 'dipping down' of the endometrium into the myometrium is labelled as ade-.nomyosis by some but ignored by others. In the present series all the cases had marked downgrowth of endometrium in the muscular layer. Age The highest incidence of adenomyosis was found between years age group. In the present series per cent were of years age group 2'4.63 per cent were below the age of 40 years and 8.25 per cent were above 55 years age group. The oldest case in the present series was a 68 years old while the.youngest was 28 years old. The same age incidence has been reported by almost all the authors. For example Green (1966) has found the highest incidence between years of age Israel and Wontersz gave the average age incidence of 45 years while that given. by Benson and Sneeden (1958) was 4'0.9 years. Cullen (1908) Jeffcoate and Potter (1934) Dreyfuss (1940) Spatt (1946) Rosario (\.1968) and Bhatt (1960) all agree that by far the greate st number of cases occur in the fifth decade. Cullen in 1908 reported this condition in a girl of 19 years. Youngest patient reported in Benson and Sneeden's series was 18 years old while Holden reported it in a 14 year old girl wh()se symptoms began 6 months after the onsef of meris.. truation and consisted of severe dysmenorrhoea. Pai"'ity Unlike endometriosis externa adenomyosis is not associated with sterility and low parity. In the present series 8.3% were sterile 4.1% had one child 10.1% had 2 to 4 children and 68.5% had more than 4 children. Hence the highest incidence of adenomyosis wa:s found in grandmultiparae. In the series presented hy Rosario 4.4% were sterile 36.7% had 2 to 4 children while 57.3% were grandmultiparae. In Benson and Sneeden's series multiparity was 3 to 4 times more _cominon in the age groups studied. In the series published by Israel and Wontersz (1968) 8.0% had borne children and only 16% were multiparous. Hunter et al (1956) suggested that the majority of cases were multiparous while nullipuae were few. In Bhatt's series majority were multiparous while 25% were sterile. Controver sial figures of incidence were given by Green (1966) who stated that only 4% were grandmultiparae and by Hilda (1950) who stated that 46.4% were multiparous. Since the majority of the cases were between years nearing menopause a correlation was sought between the last childbirth and the date of diagnosis. Only 20%had last delivery 1-5 years back and 30% had it 6-10 years back while 41.6% had it years back and 8.3% ha~ it over 20 years back. In the series present~ ed by Bhatt 42% had last delivery.10 years back while in the series presented by Rosario 70.5% had last delivery 10 \Years back and 17.6% had it over 20 years back. This shows a relatively long periog o both voluntary and involuntary infer: tility in the majotity-'2f - <!'ris_~~:. ' ---~ _

3 402 JOURNAL OF OBSTETRICS AND GYNAECOLOGY OF INDIA Symptomatology The clinical diagnosis is always provisional and is one of the many diagnostic possibilities. The symptoms of adenomyositi are very difficult to ascertain as the lesion is often associated with other pelvic pathology which ma.y cause similru symptoms. The symptoms produced by adenomyo sis are menorrhagia menometrorrhagia dysmenorrhoea a feeling of heaviness in the lower abdomen and backache; the most prominent of these being menorrhagia. In the present series menorrhagia was present in 62 % of cases (i.e. 45 cases) while 20 % had menometron hagia. The duration of menorrhagia was 1-5 years in 40% of cases 5~10 years in 40% of cases more than 10 years duration in 5% cases and in 15% of cases it was less than 1 year. Menorrhagia and menometrorrhagia were the outstanding symptoms and 32 patients (70.1%) of menorrhagic type had undergone dilatation and curettage once twice or thrice without any relief. ' Menorrhagia was the main cause of anaemia so prevalent in the majority of the cases of adenomyosis. 4.3% of cases had continuous vaginal bleeding of one to three months' duration and as curettage had failed to control the bleeding hysterectomy was done. 19.2% had dysmenorrhoea of moderate to severe degree and in the majority of the cases it was congestive in type. Ten cases (13.7 %) were postmenopausal and were mainly operated upon for genital prolapse. In 5 patients (6.8%) the main complaint was excessive vaginal discharge. In 3 cases ( 4.1%) there was a history of severe low backache not cured by any remedy. Four patients (5.4%) had suf- fered from pain and heaviness in the lower abdomen. Incidence of menorrhagia given by various authors is as follows: -Hunter et al 85% Rosario 83.8% Bhatt 82.0 % Emge 70.0% Payne 57.0% Israel and Wontersz 55.4% Spatt 53.0% Bayly and Yates 51.8% and Novak 36.0%. Incidence of dysmenorrhoea given by various authors is as follows-hunter et al 61.0% Green 45.6% Emge 25.4% Bhatt 39.0% and Rosario 26.0 % (for endometriosis in general). The duration of dysmenorrhoea in almost all the series was 1-5 years and it was progressive in nature. The condition may remain asymptoma tic. In the present series cases who were operated upon for prolapse had no symptoms suggestive of adenomyosis and 10 cases (13.7%) were postmenopausal. Israel and Wontersz reported that more than one third of the patients whose uterus showed adenomyosis had neither of its classical symptoms of menorrhagia and dysmenorrhoea. Associated Conditions (i) Fibromyoma uterus: As adenomyosis and fibromyoma uterus are diseases of the same age group and have ~ more or less simliar symptoms the two lesions are often associated with each other. In the present sedes only 5 (6.8%) cases had association of adenomyosis with fibroid uterus. This is the lowest incidence reported so far. The incidence of the same reported by various authors is as follows: Cullen 100% Israel and Wontersz 62.4% Novak et al. 62.5% Crossen and Crossen 60.7%. Bayly and Yates 62.0% Hilda 60.0% Hunter et al 58.8% Benson and Sneeden 56.6% Green 53.2% Spatt..

4 % Jeffcoate 28.0% Rosario 25.0% and Bhatt 8.0%. (ii) Endometriosis externa: In the present series only 2 (2.7%) cases had associated endometriosis externa. Comparatively higher incidences were reported by almost all the authors: Benson and Sneeden 56.6% Hilda 39.0% Green 15.5% Dreyfuss 12.4% Novak 10.8% and Rosario 4.4%. (iii) Ovarian pathology: "in the present series 10 (13.7%) cases had follicular cysts of the ovary along with adenomyosis. Four cases had serous cystadenoma of the ovary. No case was associated with ovarian malignancy. The incidence of follicular cyst associated with adenomyosis reported by Spatt was 21.9% and by Rosario was 19.2%. (iv) Cervical pathology: One case was associated with cancer of the cervix in the present series. Twenty-seven cases of adenomyosis showed squamous metaplasis of the cervix. Pre-Operative Diagnosis In the present series pre-operative diag. nosis was made in 10 (7.3%) cases. In 25 (34.2%) patients the uterus was larger than normal. Emge (1962) reported correct pre-operative diagnosis in 64.8% of cases Bhatt in 21.0% Rosario in 19.1% Benson and Sneeden in less than 10.0% Green in 4.7% and Israel and Wontersz in 2.6% of the cases. Pre-operative diagnosis is always provisional. It is based upon the clinical picture it produces menorrhagia and dysmenorrhoea of progressive type after the age of 40 years always goes in favour of adenomyosis as suggested by Emge. Curettage is unreliable as a diagnostic rocedure. Green has described characteristic appearances in hysterosalpingo- grams in the form of spicules extending perpendicularly from the ~terine cavity. In the present series only clinical features have been taken into consideration in coming to a pre-operative diagnosis. Thirty-two (43.8%) cases had undergone endometrial curettage pre-operatively. Twenty-eight (38.3%) had repeated curettage without any r elief. In the majority of the cases the histopathological report of endometrium was_ proliferative phase. Only 2 patients had hormone therapy of oral progestogens before operation without any relief. Treatment In all the cases hysterectomy was done by the abdominal or by the vaginal route with or without ovarian conservation. Discussion Adenomyosis is a frequently encountered pathological lesion found in 10.9% o.f hysterectomy specimens. The incidence of adenomyosis as reported by the majority of western clinicians is higher as compared to our cases. Endometriosis externa is associated with sterility or low parity in the majority of cases. But adenomyosis is more frequently seen in grandmultiparae. In the present series 8.3% patients were sterile while 68.5% had more than 4 children. Comparable incidence was given by Rosario Bhatt Israel and W ontersz and by Benson and Sneeden. This shows that repeated childbirth may play a role in the causation or in the acceleration of the growth process in adenomyosis. The process of benign invasion of myometrium by the endometrium under the influence of ovarian dysfunction seems to be easy and extensive in a parous uterus which ' il.

5 404 JOURNAL OF OBSTETRICS AND GYNAECOLOGY OF INDIA has undergone the process of childbirth several times. Ovarian dysfunction particularly anovulation or hyperoestrogenism is frequent a few years before the onset of menopause. At this age both adenomyosis and leiomyoma are more frequently encountered. Novak (1948) states that "adenomyosis represents merely an increased growth activity of otherwise normal tissues that it does not assume the circumscribed form so characteristic of g-enuine neoplasm and that the immediate normal cause of endometrial and muscle growth is the oestrogenic hormone of the ovany". Even in grandmultiparae there is a long interval of several years between the last childbirth and the occurrence or the diagnosis of adenomyosis. This is the period of voluntary or involuntary infertilit.y in the majority of cases. This indicates that the hormonal dysfunction comes into action much earlier in the process and that in some of the cases it may be the initiating factor. The role of hyperoestrogenism was championed by Jeffcoate and Potter. Why some in the presence of hormonal dysfunction develop adenomyosis while others develop leiomyoma and still others remain merely case of dysfunctional uterine bleeding is difficult to answer. Some hereditary factor as postulated by Emge may be significant. He has given 7 instances where the mother of the patients having adenomyosis was operated for the same condition. Similar observations were made for endometriosis externa by several workers. The existence of possible hereditary factor in the production of adenomyosis is also supported by the discovery of this lesion in a foetus at term by Robert Meyer in Similar discoveries in children of 4-14 years of age were observed by Holden (1956).J ;wert and Philip (Quoted by Emge). The other possible aetiological factors are previous infections and inflammation in the uterine cavity and trauma on account of deep and vigorous curettage. In the present series 22 cases had undergone endometrial curettage pre-operatively and two had loop insertion. In all the cases curettage was done well after the onset of symptoms. In 2 cases loop was inserted 2 and 5 years back and was removed on account of metrorrhagia and pelvic pain within 1-2 years of insertion... Trauma of repeated childbirth has also been incriminated by majority of workers. Multiparity (more than 4 children) was present in 68.5% of cases. Comparable incidence of multiparity in adenomyosis has been reported hy Benson and Sneeden Hunter Israel and Wontersz Rosario and Bhatt. The significance of multiparity however becomes disputed in view of the occurrence of this condition in a foetus at term as reported by Robert Meyer in 1897 and in a young girl by Cullen in Adenomyosis is commonly found in premenopausal age group but this may possibly be due to the fact that hysterectomy is performed most commonly in this age group. No age is however exempt as the condition has been reported in a full term foetus as well as in a 87 year old woman. Menorrhagia and menometrorrhagia are the outstanding symptoms. The cause of menorrhagia may be the assl""iated endometrial hyperplasia and may also be because of myometrial congestion and weak uterine haemostatic action on account of the presence of islands of endometrium. Usually there is histor.y of long duration of these symptoms which are not controlled by currettage or hormones and ultimate resort is hysterectomy. Congestive type of dysmenorrhoea starting-...

6 405 after the age of 35 years when associated with other symptoms favours the diagnosis of adenomyosis. Heaviness of the lower abdomen and backache are supposed to be due to heavy enlarged uterus and pelvic congestion. Classical symptoms of menorrhagia and dysmenorrhoea ma1y be absent in about 10-20% of cases. Associated lesions along with adenomyosis are fibromyoma endometriosis externa ovarian pathology pelvic inflammations and lastl.y malignancy of the cervix and the uterus. In the present series 5 cases were associated with leiomyoma 2 with endometriosis externa 10 with follicular cyst of the ovary 4 with serous cystadenoma of the ovar.y 4 with chronic pelvic inflammation 1 with cancer cervix and 27 cases were associated with squamous metaplasia of the cervix. The last mentioned condition is due to trauma and infection. Its association with adenomyosis may be because of a common aetiological factor i.e. repeated childbirth trauma. The presence of follicl.llar oyst of the ovary in a good number of cases may be because of the ovarian activity of hyperoestrogenism associated with adenomyosis. Endometriosis externa is infrequently encountered in our rural and undernouri~hed women hence we have cmne across only 2 cases associated with adenomyosis. Pre-operative diagnosis was made in only 10 (7.3 % ) cases. In the majority of cases the provisional ' diagnosis was dysfunctional uterine bieeding and fibroid uterus. Pre-operative diagnosis is usually difficult on account of its association with other lesions and its inability to produce specific symptoms. When there is no obvious pathology in the pelvis the commonest provisional diagnosis is dysfunctional uterine bleeding. The diagnosis stands even after first endometrial curettage. When it fails to cure even after repetition then adenomyosi~ usually strikes the clinicians mind and after hysterectomy it is confirmed by the pathologist. History of previous endometrial curettage was present in 32 (43.9%) cases out of which 28 had repeated curettage. Only 2 cases had received hormones pre-operatively in the form of oral progestogens. The operative treatment done in all the cases was hysterectomy with or without salpingo-oophorectomy. In 53 cases it was by the abdominal route and in 19 cases it was done vaginally. In 1 case it was Wertheim's hysterectomy for cancer cervix. Summary Adenomyosis is a pathological entity of uncertain aetiology and possibly several factors are involved in the genes1s o.f this condition. It may be present singly or in association with other conditions involving the uterus and adnexa. In the absence of pathognomonic symptoms and signs correct pre-operative diagnosis is possible in small percentage of cases but an awareness and high index of suscpicion would make the diagnosis more frequent. The clinical aspects and aetiopathogenesis of the condition has been reviewed and 73 cases of adenomyosis have been analysed. Acknowledgements I am grateful to the Superintendent Zenana Hospital J aipur and Head of the Department of Gynec. & Obst. S. M. S. Med'ical College J aipur for permitting me to make use of the hospital records....

7 406 JOURNAL OF OBSTETRICS AND GYNAECOLOGY OF INDIA References 1. Bayly M. A. and Yates C. J.: Obst. & Gynec. 10: Benson R. C. and Sneeden V. D.: Am. J. Obst. & Gynec. 76: Bhatt R. V.: J. Obst. & Gynec. India 11: Brines 0. A. and Blaines J. P.: Surg. Gynec. & Obst. 76: Counseller V. S.: Am. J. Obst. & Gynec. 36: Crossen H. S. and Crossen P. J.: 'Diseases of Women' p. 176 St. Louis The C. V. Mosby Co. 7. Cullen T. S.: J. Amer. Med. Ass. 50: Dreyfuss M. L.: Am. J. Obst. & Gynec. 39: Emge L. A.: ~ J. Obst. & Gynec. 83: Giammalvo J. T. and Kaplan K.: Am. J. Obst. & Gynec. 75: Green H. T.: Clin. Obst. & Gynec. 9: Hilda L.: J. Obst. & Gynec. India 1: Holden F. C.: Am. J. Obst. & Gynec. 22: Hunter W. E. Smith L. L. and Reiner W. C.: Am. J. Obst. & Gynec. 53': Israel S. L. and Wontersz T. B.: Obst. & Gynec. 14: Jeffcoate T. N. A. and Potter A. L.: J. Obst. & Gynec. Brit. Emp. 41: Kanter A. E. Klawans A. H. and Bauer C. P.: Am. J. Obst. & Gynec. 32: McCarty W. M. and Blackman R. H.: Ann. Surg. 69: Novak E. and DeLima 0. A.: Am. J. Obst. & Gynec. 56: Payne F. L.: Am. J. Obst. & Gynec. 39: Rosario; P. Y.: J. Obst. & Gynec. India 18: Spatt S. D.: Am. J. Obst. & Gynec. 52: Taylor H. C. Jr.: Am. J. Obst. & Gynec. 23: Weed J. Geavy W. and Hollard J.: Clin. Obst. & Gynec. 9: Westman S.: Arch. F. Gynak. 116: (Quoted by Spatt 22)... \

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