LITHOPEDION
(Review with a Case Report)
by
PusHPA GuPTA, M.B., M.S., D.G.O. and ANGULYABHARANAM SAROJINI, M.B., M.D., D.G.O.
History curious epigram alluding to the classi cal myth that after the flood the world The term lithopedion is applied to a was repopulated by the two survivors, foetus which has been retained within Deucalion and Pyrrha, who walked the maternal abdomen, and the tissues over the earth and cast stones behind of which have become more or less them which on striking ground be completely infiltrated with calcium came people". The epigram read as salts. follows: "Deucalion cast stones be According to Dorland and Hubeney hind him and thus fashioned our ten (1926), lithopedion formation had der race from hard marble. How been recorded as early as the 16th comes it that now-a-days by a re century by Veneteiss (1595), Albo versal of things, the tender body of a ' sius (1597) and Densingeus (1661). little babe has limbs nearer akin to In the early days the discovery with stone"? in a woman's abdomen of such a hard stony mass having th2 form of a child Classification excited wonder and speculation, and In 1881 Kuchenmeister collected was even viewed at times with con 45 cases of various forms of foetal cal siderable awe. Bainbridge (1912) cification recorded during the 200 quoted an interesting account of the year period, antedating 1880. He first case (from Gould and Pyle, pointed out that calcification in the 1897). He said "Israel Spach in a;u products of conception may not be extensive gynaecological work, pubh entirely limited to the foetus, but may shed in 1557, figures a lithopedion also involve the membranes and the drawn in s·du in the case of a woman placenta or may be entirely limited to I with her belly laid open. He !lecided the latter structures. On the basis of this to be a calcified foetus, which he these variations 'he proposed their regarded as a reversion, following the division into three groups. f 1. Lithokelyphos (Stone-sheath or Department of Obstetrics & Gynaeco egg shell), in which the membranes logy, Sardar Patel Medical College and alone are calcified and f9rm a hard P. B. M. Group of Hospitals, Bikaner. shell surrounding the foetus. The Received for publication on 29-1-65. foetus may undergo only slight - LITHOPEDION 317
_. change or it may completely be skele lithopedion, or sterile calcification of tonized, but it is not involved in the the foetus. The above classification process of calcification. Kuchenmeis is based e>n modern pathological eon ter believed this to be a most common cept and is not limited to the retained type and the result of the membranes calcified foetus in the abdominal remaining intact around the foetus at cavity but indicates the various the termination of pregnancy. changes which can take place in the 2. Lithokelphopaedion (Stone foetus in an intra-abdominal preg sheath child), in which both the nancy. membranes and the foetus are cal cified. The amniotic fluid has escap Etiology ed or has been absorbed. The factors leading to the calcifica 3. True lithopedion (Stone child), tion of the foetus are vague. Leopold in which the foetus is infiltrated with (1881) did some experimental work calcium salts and in which the calci to show the fate of rabbit foetuses, fication of the foetal membranes is removed from the maternal uterus negligible. This type, he believed, and left in the abdomen. He placed, resulted when the foetus escaped un embryos 2.5, 5 and 8 ems. in length attached into the abdominal cavity, in the abdominal cavities of adult the membranes being either left be rabbits. In some instances he used hind or closely wrapped about the the foetus alone and in others the en foetus. tire embryo with its membranes in While this classification has no tact. There was no reaction on the clinical value, it provides a con part of the host except in those cases venient method of grouping the speci in which peritonitis developed by the mens. The confusion resulting from second day; the 2.5 ems. embryos had the double use of the word litho almost completely disappeared and pedion in referring to the general by the 9th or lOth week the 8 ems. group as well as to the third group foetus had been reduced to few rem could be avoided by substituting the nants of the paws, the integument, word lithotecnon when referring to and the skeletal system. In the pre the third group, the true lithopedion sence of infection the process of de of Kuchenmeister, in which the foetus struction and absorption progressed alone is calcified. much more rapidly. If the umbilical D'Aunov and King (1922) propos cord was ligated, absorption was ed another classification of retained slower. The miniature foetus meets abdominal pregnancies into four the same fate as the one extruded possible categories: (1) skeletoniza into the abdomen of a woman on the tion or retention of a collection of terminatio'n of extra-uterine preg foetal bones with absorption of foetal nancy. soft parts; (2) adipocere, or replace An analysis of the pregnancies re ment of the soft parts by soap and sponsible for the production of the fats; (3) suppuration, or abscess for foetus of lithopedion shows evidence mation due to infection with destruc that in 73 to 84% of cases there is tion of the foetal tissues; and ( 4) true clinical evidence of development to 13 318 JOURNAL OF OBSTETRICS AND GYNAECOLOGY OF INDIA or beyond the seventh month and in The exact chemical process by which ... 60 % to term. · The average term of calcium is deposited in the tissues or development was about 7 months, elsewhere in the body is not well which shows that only the more understood. mature foetuses undergo this trans formation. It is impossible to postu Wells (1918) found that pieces of late the types of extra-uterine preg sterilized cartilage placed in the nancy that are most likely to result in peritoneal cavity of a rabbit soon be the production of lithopedion. In came calcified, having taken up lime general it is the type in which the from the fluid in which they bathed. foetus is most likely to develop to He concluded that the process is a near term, and while this may occur physical rather than a chemical one, ' in practically every type, it is more the calcium and phosphorus being ab common in some than in others. sorbed by the degenerated tissue. Mac Callum (1916) stated that there The retention of the foetus in the seems to be something peculiar about abdomen, its development to the third tissues, living or dead, which gives month or more, are essential for the them power to catch up the calcium It production of lithopedion. is ex from the circulating fluid and hold it tremely doubtful if a foetus retained firmly in solid form. Later on, Mac within a normal uterus ever under Callum ( 1940) suggested that some goes this transformation because local chemical process must be re there the occurrence of infection sponsible for the precipitation of cal leads rather to destruction of the cium in the dead materials exposed tissues of the foetus and skeletoniza to the circulating fluid, as well as in tion. Oden and Lee (1940) listed the bone. He stated that iron is practical- following conditions necessary for ly always demonstrable in areas of lithopedion formation: calcification, but it is possible that it 1. There must be an extra-uterine is merely absorbed by the calcium pregnancy. salt or precipitated by phosphoric 2. The foetus must survive in the acid liberated in that position. l\~ s abdomen over 3 months. bitt (1955) suggests that calcium is 3. The pregnancy must escape deposited in dying or dead tissue medical notice. without reference to the blood cal 4. The foetus must remain sterile. cium. Fatty degeneration occurs 5. Conditions necessary for the de initially, then hydrolisation takes position of calcium must be present, place and a fatty acid is liberated . .._ - namely sluggish circulation. The fatty acid is replaced by carbonic acid Mummification, adipocere and and phosphoric acid in the blood to form calcium carbonate and cal- · various types of fatty change have cium phosphate. Schumann (1921) been described in these retained categorized the final results of ectopic mature foetuses, but it is not known gestation as follows: if these changes are entirely different processes or merely successive steps Resorption, mole formation. in the production of a lithopedion. haematocele, suppuration, skeleto- LITHOPEDION 319
nization, adipocere and lithopedion early months except possibly pain or formation. unusually active foetal movements,. Owens (1962) stated that the foetal 2. The termination of pregnancy. tissue undergoes aseptic autolysis and The later months of the pregnancy circulating calcium from maternal are usually uneventful and at or near blood is precipitated locally in the term, labour is initiated in an ap acidic necrotic tissue, as a passive parently normal manner. The pains physico-chemical process, to produce continue to be mild; however, after a lithopedion. 24 to 48 hours, they cease entirely without the delivery of the child. This Diagnosis is commonly referred to as "false" or The diagnosis of lithopedion can be "missed" labour. The patient is no made from the history but its pre longer aware of foetal movements. sence is frequently not recognised be She gradually recovers, menstruation cause the condition is not borne in returns and the abdomen decreases mind and the various events in the in size. history are not correlated until the 3. Latent period. The foetus now diagnosis has been made at operation. ceases to exist as such. It becomes The symptoms may be divided chro a parasite in the maternal abdomen nologically into four groups cor and while it may continue to derive responding to the successive stages in some blood supply from the mother, the development (Masson and Simon, it leads only a passive existence. Its 1928). bulk decreases with the absorption of 1. The onset of pregnancy. The the amniotic fluid. Dehydration patient gives a history of a pregnancy occurs and tissues begin to be infil which had been atypical in some re trated by calcium salts. During this spects. The symptoms may be those period the patient is free from symp of ruptured tubal gestation which toms except that she may still be continues as a secondary abdominal aware of the presence of a mass in the pregnancy. In such cases the symp lower abdomen. From two to many toms of rupture subside but the abdo more years may lapse in this manner I men continues to enlarge and the while the foetus is being gradually other evidences of pregnancy persist. transformed into a lithopedion. In other cases the symptoms may be 4. Late symptoms. In about 65 o/t mistaken for those of miscarriage but of cases, if the patient lives, late the inquiry discloses the fact that a symptoms eventually develop. They ~"' foetus was not passed and that instead consist of mild abdominal pains or a of usual sequence of events following consciousness of fullness in the lower a miscarriage, the abdomen continued abdomen frequently associated with to enlarge and the other evidences of a persistent foul vaginal discharge. pregnancy persisted. Symptoms of bladder irritation may In the event of primary abdominal result from pressure of the mass on or ovarian gestation, the patient con the bladder or from secondary siders herself pregnant and may cystitis. If the patient is untreated notice nothing abnormal during the the lithopedion may rupture into the
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adjacent viscera or through the abdo cidence of 2.0 per cent, Schumann minal wall. (1921) 1.5 to 1.8 per cent and Ander The general health of the patient son et al., (1951) 0.81 per cent in 370 may be unimpaired or thzre may b·e cases of extra-uterine pregnancy. loss of weight. A hard mass usually Clark and Ellison (1959) reported an associated with slight tenderness is incidence of 0.29 pzr cent based on present in the lower part of the abdo 351 ectopic pregnancies operated men. On vaginal examination this during the period 1947-1957. Bland mass may be palpated on one or the et al., (1933) brought the total ag other side of the pelvis and is fre gregate of reported cases to 197. quently attached to the uterus. Later Anderson et al. , (1951) X-ray evidence is characteristic. reported a total of 252 cases of_,., • Kirklin and Simon (1928) have authentic lithopedion formation in pointzd out that the foetus appears the world's literature. Subsequent much more distinctly than in a nor ly, various authors have report mal pregnancy because of its increas ed cases: McClure and Eppers~n ed density. The foetus may also oc (1952); Schwarz (1952); Roberts cupy an abnormal position. Other (1952) ; Sordo Noreiga (1952); tissues besides the skeleton, which Herrera and Casimiro ( 1954) ; Menon have become infiltrated with calcium (1954); Nesbitt (1955); Gupta ~alts, will produce an extra-skeletal (1955); Steinberger and Pogue shadow of varying density and out (1956); Parikh (1956); Clark and line, depending on the extent of cal Ellison ( 1959); Temple and Hester cification of the products of concep (1959); Woodbury and Jarrett , tion. (1960); Benerjee (1961) ; Owens The conditi01fs with which litho (1962) and O'Lzary and Bepko pedion are most frequently confused (1963): These cases with the one are calcified fibromyomata and herein described make an aggregate ovarian dermoids. The fibromyo of 269. mata, when sufficiently calcified, There are innumerable instances rarely produce symptoms. Ovarian where women carrying lithopedion or dermoids may be a source of con· retained foetal parts have had COj l fusion except when they are bilateral. comitant normal pregnancies and de Previous miscarriage or atypical preg liveries. Williams (1946) reported a nancy is not an essential part of the case of an intra-uterine lithopedion history of ovarian dermoid. being delivered wrapped around the, Incidence neck of a living twin. .... - The discovery of a lithopedion in The true incidence of abdominal the second half of the twentieth cen pregnancy is difficult and varies wide tury implies that either the patient ly in different series. The occurrence involved has had no medical attention of lithopedion formation has been re whatsoever, or rather some serious ported as from one to two per cent of mistakes in medical judgement have all extra-ut~rine n:regnancies. Masson been made. Obviously, a retained and Simon (1928) reported an in- abdominal pregnancy should be re- LITHOPEDION. 321
cognised eventually in any modern nancy or calcified fibroid in the broad liga obstetrical practice. However, with ment was made. Skiagram showed a small foetus with close medical supervision and im over-riding of bones of skull. Spalding's proved diagnostic methods ectopic sign, was present. The spine and extremities gestation escapes surgical treatment were crowded in a small mass-dead foetus less frequently, so that lithopedion (Fig. 1). The diagnosis of a lithopedion development is now a truly rare oc currence. This case is therefore re ported because of the rarity of its typ~.
Case Report Mrs. L., aged 30 years, married for 12 years, came to the hospital on lOth April, 1963, for sterility, scanty menstruation and a lump in the abdomen of 5 years' duration. Previous medical and surg'cal histories were not significant. History of present ill ness - 5 years ago she had a period of amenorrhoea for 7 months and she also felt foetal movements followed by a bout of vaginal bleeding along with acute abdo-· m"nal pain, after which the foetal move ments stopped. She had no complaint and the size of abdomen also reduced, her ~ periods had been regular but scanty. She had her last period 15 days ago. One F.T.N.D. -7 years ago but child died at the age of 5 months. General examination revealed nothing abnormal in her heart and lungs. Her blood pressure was 110/70 mm. Hg., her haemoglob1n 13 gm. and urine Fig. 1 normal. Skiagram showing the shadow of lithopedion. Clinical Examination was made before the operation as skiagram Abdominal examination revealed ·a hard was absolutely conclusive. lump arising ·from the pelvis and filling Operation: Under spinal anaesthes'a the up the lower abdomen almost up to the abdomen was opened by a median subum umbil"cus. The margins were well defined bilical incision. A hard irregular encap all round except where the mass dipped sulated mass was seen. It was adherent deep into pelvis; the surface was smooth. anteriorly. to the omentum, to the parietal There was no tenderness. Liver and spleen peritoneum on the right s"de, and posterior were not palp References 1. Andrson, J. R., Counseller, U. S. and Woolner, N. B.: Am. J. Obst. & Gynec. 62: 439, 1951. 2. Bainbridge, W. S.: Am. J. Obst. 65: 31, 1912. 3. Banerjee, D.: J. Obst. & Gynec. Fig. 2 India. 12: 303, 1961. Showing details of lithopedion. 4. Bland, P. B., Goldstein, L. and Bol- ~ ton, W. W.: Surg. Gynec. & Obst. adherent to the body of the foetus; fea tures of foetus were well marked and defin 56: 939, 1933. ed. All body parts were well developed. 5. Clark, J. F. J. and Ellison, H. R.: Obst. & Gynec. 14: 537, 1959. Summary 6. D'Aunoy, R. ahd King,- . L.: Am. 1. The present case of lithopedion J. Obst. & Gynec. 3: 377, 1922. formation brings the total of reported 7. Dorland, W. A. N. and Hubeney, cases in the world's literature to 269, M. J.: The X-Ray in Embrology including the cases reported in this and Obstetrics, St. Paul, Minn., country. 1926, Bruce Publ. Co. 2. The lithopedion developed over 8. Gould, G. M. and Pyle, W. L.: a period of 5 years. The initial attack Anomalies and Curiosities of Medi - of acute abdominal pain was un cine, 1897, Quoted by Bainbridge, ... doubtedly due to rupture of an extra W. S.: Am. J. Obst. 65: 31, 1912. uterine pregnancy with extrusion of 9. Gupta, U.: J. Obst. & Gynec. India. the unattached foetus into the broad 5: 81, 1955. ligament where it subsequently be came calcified. The patient came to 10. Herrera, M. and Casimiro, C. M.: the hospital for treatment of sterility. Philippine J. Surg. 9: 131, 1954. 3. A brief review of the literature 11. Kirklim, B. R. and Simon, H. E.: of lithopedion is given. Ill. Med. J. 1928, Quoted by Mas- LITHOPEDION son, J. C. and Simon, H. E.: Surg. 22. Owens, W. L.: Obst. & Gynec. 19: Gynec. & Obst. 46: 500, 1928. 401, 1962. 12. Kuchenmeister, V.: Arch. f. Gynak. 23. Parikh, R. M.: J. Ind. Med. Assoc. 17: 153, 1881. 26: 104, 1956. 13. Leopold, G.: Arch. f. Gynak. 18: 53, 24. Roberts, J. K.: Brit. Med. J. 2: 130, 1881. 1952. 14. MacCallum, W. G.: Text Book of 25. Schumann, E. A.: Extra-Uterine Pathology, Philadelphia, 1916, W. B. Pregnancy, New York, 1921, D. Saunders Co. Appleton & Co., p. 33. 15. MacCallum, W. G.: Text Book of 26. Schwarz, F.: B. M. J. 2: 131, 1952. Pathology, ed. 7, Philadelphia, 1940, 27. Sordo Noriega, A.: Gac. Med. W. B. Saunders Co. p. 117. Mexico. 82: 183, 1952. 16. Masson, J. C. and Simon, H. E.: 28. Steinberger, W. and Pogue, E. A.: Surg. Gynec. & Obst. 46: 500, 1928. Obst. & Gynec. 7: 54, 1956. 17. McClure, J. H. and Epperson, J. W. 29. Temple, H. R. and Hester, L. L.: W.: Ohio Med. J. 48: 225, 1952. Obst. & Gynec. 14: 537, 1959. 18. Menon, M. K. K.: J. Obst. & Gynec. 30. Wells, H. G.: Chemical Pathology, India. 5: 89, 1954. ed. 3, Philadelphia, 1918, W. B. 19. Nesbitt, R. E. L., Jr.: U.S. Armed Saunders Co., p. 439. Forces Med. J. 6: 903, 1955. 20. Oden, P. W. and Lee, H. C.: Virgi 31. Williams, M.: Am. J. Surg. 72: 750, nia Med. Month. 67: 304, 1940. 1946. 21. O'Leary, J. A. and Bepko, F. J.: 32. Woodbury, J. W. and Jarrett, J. C.: Obst. & Gynec. 21: 250, 1963. Am. J. Obst. & Gynec. 80: 590, 1960.