Canad. Med. Ass. J. 226 Case Reports: Lithopedion Aug. 3, 1968, vol. 99 CASEREPORTS Lithopedion LILLIAN A. CHASE, B.A., M.B., C.R.C.P.[C], Toronto

discovery of a lithopedion is as dramatic anything recognizable as tissue. She developed con¬ THEas it is rare. Although the condition was siderable dysuria at this time, and on two occasions, known and in ancient times, because of acute urinary retention, had to be cathe¬ recognized only at a about 270 cases have been recorded in the litera¬ terized hospital in Kingston, . After ture. been in this some months she was no longer able to feel move¬ Most of these have century; up ments, but the bleeding and the pain continued. At until 1900 there had been only 38 recorded no time was she told that she was or had been 12 more cases, and in the next 12 years only pregnant. After being on a waiting list for eight cases were added; the remainder have been re¬ months she was finally admitted to hospital, where ported since then. she underwent an abdominal operation. She did not Today the occurrence of a lithopedion is rarer know what type of procedure had been carried out than ever; it is unknown among women who re¬ and could only say that "a string was giving me ceive care or medical trouble and they loosened it". Postoperatively she adequate prenatal regular some but the so it is found where passed clots, bleeding finally stopped examinations, usually only and she no had any She felt modern health services are unavailable or when longer pain. stronger than before and soon returned to work. Although a gross error has been made. her lower abdomen had decreased in size, she was still able to feel a mass there. She stated that her A 54-year-old Jamaican woman applying for a physician at that time had assured her that this was post on the domestic staff of Sunnybrook Hospital, of no significance. Postoperatively she never again Toronto, was found to have an abdominal mass dur¬ had a menstrual period, and although the mass ing a pre-employment examination in the outpatient appeared to get somewhat smaller, it was still department on February 9, 1966. Although aware of present when she was seen at Sunnybrook Hospital. the mass, she had no complaints. Her history in¬ In 1961 she emigrated to England from cluded the following particulars: Her menarche oc¬ Jamaica and in 1965 she came to Canada; thus on at curred at the age of 15; since then her menses had least two occasions she had been examined by been regular, occurring every 28 days and lasting immigration medical officers. Although these exam¬ for 5 days. The flow was scanty, but was never inations included chest radiographs, at no time was associated with pain. In 1933, at the age of 21, she she informed that anything was wrong. had her first pregnancy and was delivered of a male She had no history of tuberculosis, pelvic in¬ child after a 14-hour labour. The second pregnancy flammatory disease, venereal disease or urethral dis¬ occurred a year later and this delivery apparently charge, and no dysuria, nocturia or stress incon- was painless. During neither of these pregnancies tinence. She had had no recent vaginal bleeding and nor at delivery was she attended by a physician. only a normal amount and type of vaginal dis¬ Her resumed their normal after the charge. periods pattern On examination second pregnancy. Although she never used con¬ physical this 135-pound woman, traceptives, she did not become pregnant 5'5" in height, was found to be edentulous; her again; blood was her was 65 and she had not had any pressure 140/80; pulse miscarriages. A mass was In at the of she noticed that her regular. hard, stone-like, irregular pal¬ 1957, age 45, pable, lying across the lower abdomen. A lower abdomen was increasing in size and she was able abdominal midline scar was The film to fetal movements. she did not think present. plain feel However, of the abdomen showed a lithopedion, and it was she was pregnant at this time, because she had had therefore concluded that it was the head that was vaginal bleeding, which was occasionally regular but palpable in the left lower quadrant and the buttocks more often at irregular intervals. At times the bleed¬ on the right side. The mass appeared to be quite ing lasted for as long as 15 to 21 days. It was accom- superficial and was mobile; it measured 27 cm. in panied by considerable lower abdominal pain and length and 10 cm. in width. was frequently quite profuse, enough to soak the The pelvic examination confirmed that the present newspapers with which she attempted to control it. mass appeared to be of uterine origin, filling the She said that she had not at any time passed pelvis and extending into the abdomen. The cervix was healthy. Laboratory investigation revealed a Consultant in Medicine, Sunnybrook Hospital, and Staff of 12.8 a sedimentation rate of 34 Women's Ontario. hemoglobin g., Member, College Hospital, Toronto, mm. in one hour a white cell count Reprint requests to: Dr. Lillian A. Chase, 228 Medical (Westergren), Arts Building, 170 St. George Street, Toronto 5, Ontario. of 6200 and a blood urea nitrogen of 12 mg. per Canad. Med. Ass. J. Aug. 3, 1968, vol. 99 Case Reports: Lithopedion 227

Fig. 1..Anteroposterior radiograph of the abdomen. Fig. 2..Lateral view of the abdomen seven days later.

100 ml. The prothrombin time and the fibrinogen calcified. Attached to the right cornu in front of level were both within normal limits. Urinalysis the right was a smaller calcified showed a specific gravity of 1.016, a pH of 5.5, a mass, firmly adherent to the adjacent structures. trace of albumen and one or two red blood cells per This mass was also removed after separation and high-power field. The VDRL was negative. A smear division of the many adhesions. Except for a dilated and culture examination of her sputum revealed no esophageal hiatus, no other abnormalities were abnormalities. found in the abdomen. The plain x-ray film of the abdomen was reported Regrettably, no pathological report regarding the as follows: or its membranes was available, and the "A single A.P. view of the abdomen shows the weight was not recorded. However, Figs. 4 and 5 presence of a fetus, the size of which would suggest display the lithopedion immediately after it was that it has developed fairly close to term. There removed from the abdomen. The pathological report are many signs which indicate that the fetus is dead of the smaller mass was recorded as follows: and that it has been dead for a considerable period mass 8 x 4 x 3 cm. with of time. In addition there is evidence of calcifica¬ "A calcified measuring fat 9 x 5 x 1 cm. was received. tion of the soft tissues to the fetus and attached measuring adjacent The calcified mass is oval in One surface is probably of the fetus itself. The fetus occupies a shape. comparatively high position in the pelvis, and this hard and contains calcific material; the other surface would be in with an intra-abdominal is much softer and is composed of a capsule which keeping preg¬ is in colour and is nodular. It contains nancy. The above findings are in agreement with yellowish is ex¬ the radiological diagnosis of a lithopedion" (Figs. 1 yellow debris which quite soft. Microscopic and 2). amination shows stretches of dense cellular collagen The was admitted to Hos¬ in which there are irregular zones of granular and patient Sunnybrook At there pital, and on February 15, 1966, under general partially calcified debris. the margins ap¬ anesthesia, a laparotomy was performed using a pears to be a capsule containing some lymphocytes sub-umbilical right paramedian incision. A partially and near the margins in a loose stroma there are calcified fetus was found lying obliquely in the some collections of vascular channels. There is no abdomen with the head in the left lower quadrant evidence of malignancy. The pathological diagnosis and the buttocks on the right at a slightly higher is calcified omentum and degenerate ." level (Fig. 3). The omentum surrounded and was adherent to the superior and anterior aspects of Discussion the mass. After division and separation of the ad¬ hesions the fetal mass was removed. The uterus, en¬ A lithopedion usually results from an ab¬ larged to about the size of a 12-week pregnancy, dominal pregnancy, either primary or, more contained multiple fibroids, some of which were usually, secondary, i.e., where the gestation Canad. Med. Ass. J. 228 Case Reports: Lithopedion Aug. 3, 1968, vol. 99 Fig. 3

Fig. 5

Fig. 3..Lithopedion as it lies in the wound. The surgeon's gloved hand appears on the pa¬ tient's left side. Fig. 4..Lithopedion lying in the surgeon's hand. The head of the calcified fetus can be seen on the right side. >..Lithopedion after operative removal.

Fig. 4 actually occurs free in the peritoneal cavity, calcified (without calcification of the mem¬ even though it may have originated in a fal¬ branes ) .2 lopian tube or even in the uterus.16 The circum¬ D'Aunoy and King3 have listed four changes stances under which lithopedions are usually which an abdominal fetus may undergo if it is found are listed by Cave11: the pregnancy is not removed: (a) skeletonization, where only extrauterine; the fetus has developed for more the bones of the fetus remain following the dis- than three months; the conditions for the de¬ integration and absorption of the soft parts; (b) position of calcium must be present, i.e., the adipocere, where the soft parts are replaced by local circulation must be sluggish and the fetus fatty acids, soaps and salts of palmitic and must remain sterile. Only in a few patients can stearic acids; (c) suppuration, where the fetus is a placenta be identified. destroyed after an abscess has formed, usually Kuchenmeister,1 to whom we owe so much due to E. coli . Under these circum¬ for his investigation of ancient records of this stances the abscess may rupture into the vagina, condition, classified three types: lithokelyphos, in the rectum or other parts of the bowel, and fetal which only the fetal membranes are calcified bones may be discharged through any of the and the fetus degenerates within them; litho- body's orifices, including the mouth. (d) True kelyphopedion, when both the fetus and the lithopedion formation occurs if the fetus remains surrounding membranes show calcium deposits, sterile and to varying degrees becomes infiltrated and true lithopedion, where only the fetus is with calcium salts. Canad. Med. Ass. J. Case Reports: Lithopedion 229 Aug. 3, 1968, vol. 99

The case reported here has the classical symp¬ the patient died the fetus was found. In 1540, toms and signs of an . Christopher Bain, the "travelling surgeon", suspension of regular menses, development of operated on an Italian woman and extracted the irregular uterine bleeding, lower abdominal pain, skeleton of a male child; the patient survived to dysuria and the appearance of the abdominal have four more children. In 1721 Anna Mullern, mass; even fetal movements were noticed. Oc¬ 46 years before her death, declared herself preg¬ casionally the condition can cause remarkably nant; the swelling remained until she died at few symptoms and these are due mainly to pres¬ the age of 94. She desired that her body be sure, such as dyspareunia and bladder symp¬ opened after death; the surgeon broke open the toms. Once the lithopedion forms the only evi¬ mass with a hatchet.a lithopedion. dence may be an abdominal mass, as in this The first operation in North America for re¬ case. In this era the diagnosis is usually made moval of a fetus from an abdominal pregnancy by a radiologist, but if the clinician fails to order was done by John Baird of in 1759. an x-ray examination of the abdomen, the diag¬ Details of this pregnancy were recorded by nosis will then be made only at laparotomy. It Fothergill.5 seems likely that this patient had her abdominal "Memoir on Extra-uterine Gestation", pub¬ pregnancy in 1957, but it is difficult to explain lished in Edinburgh in 1840,6 is a mine of in¬ why the laparotomy at that time failed to reveal formation. Up to that time the condition was it. regarded more as a medical curiosity than as Cases have been recorded where the normal "one of the most dreadful calamities to which development of an intrauterine pregnancy has a woman can be occurred with the formation of subjected".5 simultaneously In 1960, Woodbury and Jarrett2 reported a pa¬ a lithopedion from a concomitant extrauterine Normal deliveries have been ob¬ tient in whom an intra-abdominal lithopedion pregnancy.2 was retained for 13 years. also refer to a served in women who at the same time have They of an intrauterine delivered carried a report lithopedion lithopedion. while "wrapped around the neck of a living Although at one time the maternal mortality ".5 from abdominal was recorded as pregnancy Masson and Simon7 in a comprehensive article 35% (1919), it is now so rare that it is not re¬ 9 in 44 cases of extra¬ corded reported lithopedions separately. uterine pregnancy at the Mayo Clinic from 1903 One of the earliest recorded cases of litho¬ to 1926, and added 9 more cases that were seen pedion occurred 1000 years ago. It was reported there. by Albucasis, the greatest surgeon of the Arabic a well-illustrated 936- King,8 in very interesting, era, who flourished in Cordova, Spain (A.D. article, wrote of his experiences with 12 cases of 1013 ).* This article, one of the earliest medical advanced extrauterine pregnancy during his 25 writings, was first published in Venice shortly in . One patient passed what she in it was re- years after the advent of printing 1497; was a chicken bone rectum; it was in Oxford in 1779. The record thought by published later a fetal femur. His illustration shows the fetal says that the mother passed fetal bones through bones neatly arranged, apparently all of them the abdominal wall but recovered. having been passed by rectum. In 1557 Israel Spach, quoted by Bainbridge4 has the only case of a litho¬ and showed the of a Auvray9 reported others, figure lithopedion in which a carcinoma developed. drawn in situ with the mother's belly laid open. pedion related this to the that after the This condition has also been a theme for a Spach myth novel. In 1944 Samuel Adams12 flood the world was repopulated by the two Hopkins pub¬ survivors, Deucalion and Pyrrha, who walked lished "Canal Town", which tells of a young in the un¬ over the earth, casting stones behind them doctor who diagnosed pregnancy which, on striking the ground, became people. married daughter of the town's leading citizen; The epigram reads: "Deucalion cast stones be¬ no baby was born, but after her death from hind him and thus fashioned our tender race cholera her body was exhumed, and the doctor, from hard marble. How comes it, that nowadays, previously castigated by the father, showed the by a reversal of things the tender body of a little lithopedion to him. This story is based on a babe has limbs nearer akin to stone?" specimen which is now in the museum of the New York. The Other early cases are those reported by Albany Medical School of Venetiss in 1595 and by Denesingeus in 1661. factual account of this case, based on the report A famous lithopedion occurring in the city of of Dr. Davis Little (1768-1852) to the Albany Sens was reported in Gynarcorium by Cordaeus County Medical Society, appeared in The New in the early part of the sixteenth century; when Yorker of December 10, 1938. Ass. J. 230 CASE REPORTS: LICORICE AND HYPERTENSION Cnad.Au.3, Med.1968, vol. 99

Thanks are due to Dr. W. A. Harvie for the 3. DAUNOY, R. AND KING, E. L.: Ibid., 3: 377, 1922. making 4. BAINBRIDGE, W. S.: Amer. J. Obstet. Dis. Wom., 65: diagnosis by radiographic examination; to Dr. J. K. M. 31, 1912. Morrison, Medical Superintendent of Sunnybrook Hos- 5. FOTHERGILL, W. E.: J. Obstet. Gynaec. Brit. Emp., pital, for prompt arrangement of the admission; 9: 67, 1906 (abstract). patient's 6. CAMPBELL, W.: Memoir on extra-uterine gestation, to Dr. H. F. McConnachie for the pathological report A. & C. Black Ltd., Edinburgh, 1840. and to Dr. A. W. Harrison for the successful surgical 7. MASSON, J. C. AND SIMON, H. E.: Burg. Gynec. Obstet., 46: 500, 1928. operation. Special thanks go to Dr. Laliab Chase of 8. KING, G.: Amer. J. Obstet. Gynec., 67: 712, 1954. Port Williams, Nova Scotia, for making the publication 9. AUVRAY, M.: Gynec. Obstet. (Paris), 9: 346. 1924. of the colour plates possible, and to Mr. William Meth- 10. BENJAMIN, A. E.: Minnesota Med., 17: 412, 1934. 11. CAVE, P.: Brit. Med. J., 1: 383, 1937. ven, medical photographer. 12. ADAMS, S. H.: Canal town, Random House Inc., New York, 1944. REFERENCES 13. OWENS, W. L.: Obstet. Gynec., 19: 401, 1962. 14. BINNS, J. H.: Brit. Med. J., 1: 169, 1966. 1. KUCHENMEISTER, F.: Arch. Gynaek., 17: 153, 1881. 15. GILMAN, R. A. et al.: Amer. J. Obstet. Gynec., 68: 2. WOODBURY, J. W. AND JARRETT, J. C.: Amer. J. Ob8tet. 1187, 1954. Gynec., 80: 590, 1960. 16. ROBERTS, J. K.: Brit. Med. J., 2: 130, 1952.

Licorice and Hypertension ROBERT E. LEFEBVRE, M.D.* and JULIEN MARC-AURELE, M.D.,t Montreal THIS case report is intended to illustrate that pitations, anxiety and episodes of dyspnea. After a complete history is still invaluable in pro- consultation with her doctor, who noted her blood viding clues as to the nature of disease and re- pressure to be 180/120, she was treated with mains the corner stone for subsequent logical phenobarbital. and warranted investigation. Despite this therapy her symptoms did not sub- side and in June 1966 she underwent a complete thyroid investigation the results of which were with- Mrs. P.F., a 34-year-old gravida 3, para 2, aborta in normal limits. The patient was subsequently given 1, white wooman, presented with a history of known other types of sedative without relief of her com- hypertension of about two years' duration. plaints. The patient's symptoms first began about Decem- Convinced that her symptoms and hypertension ber 1965, when she complained of intermittent were emotional in origin, the patient decided to headaches, mostly frontal in location, of variable return to her occupation of nursing. While working duration and described as a sensation of heaviness. in a postoperative recovery room, she would have They occurred most often in the morning on rising her blood pressure taken regularly and noted it to and were not relieved by acetylsalicylic acid. No be consistently elevated between 200/100 and medical attention was sought at this time. 240/140. Disturbed by this persistent high blood In April 1966, the patient was hospitalized for a pressure, she consulted us. dilatation and curettage following a spontaneous The family history disclosed that the father had abortion. On admission, her blood pressure was noted died at 54 years of age from a cerebrovascular ac- to be 170/100. Because the blood pressure remained cident, and that the mother had been slightly hyper- elevated, a full renal and hypertensive investigation tensive since her . was undertaken. All investigated parameters were The patient's own history was non-contributory; found to be normal. Approximately one week after there was only one hospitalization at 22 years of her admission the blood pressure spontaneously re- age for investigation of fever of unknown origin. was verted to normal. She was asymptomatic and During questioning, the patient volunteered the discharged with a diagnosis of essential hypertension information that she had developed a peculiar habit of neurogenic origin. -that it was not unusual for her to eat at least two Approximately two weeks after discharge, the pounds of fresh licorice a week. headaches recurred and were accompanied by pal- Physical examination at this time revealed only an elevated blood pressure of 220/115, and there From the Department of Medicine, Section of Nephrology, HOtel-Dieu de Montreal, Montreal 18, Quebec. were no signs of neuromuscular disturbance. Pre- *Assistant Resident II, Department of Medicine, H6tel- liminary investigation, including a rogitine test, was Dieu de Montreal. tAssistant Professor of Medicine, Department of Medicine, negative. University of Montreal, and Nephrologist, H4tel-Dieu de Montr4al. In the ensuing days, the patient on her own ac- Reprint requests to: Dr. J. Marc-Aur,le, Department of cord decided to stop eating licorice. She continued Medicine, HOtel-Dieu de MontrMal, 3840 St-Urbain Street, Montreal 18, Quebec. to have her blood pressure taken regularly at work,