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Lithopedion with a report of two cases presenting as rectal lesions
Lithopedion WITH A REPORT OF TWO CASES PRESENTING AS RECTAL LESIONS THOMAS
A.
RODENBERG, M.D . AND ANTHONY V . POSTOLOFF, M .D .,
Diplomate, American Board of
Pathology, Buffalo, New York From the Department of Obstetrics and Gynecology and the Department of Pathology, Millard Fillmore Hospital, Buffalo, N. I'.
E
CTOPIC gestation is stated to occur approximately once in 300 pregnancies .' -' Among
the more unusual results of such extrauterine fetation is mummification or calcification of the fetus-the lithopedion or "stone child ." It has been postulated 1,3,4 that a fetus which has been retained in the uterus for a considerable period of time could, by the deposition of lime salts, be transformed into a lithopedion, particularly in instances of missed labor or missed abortion . Others," however, believe that calcification of the fetus or the membranes, or both, occurs only with extrauterine pregnancy . Masson and Simon' state that three conditions are essential to the production of a lithopedion : (i) the development of a fetus to at least the third month, (2) retention of such a fetus within the abdomen and (3) the absence of infection . Thus it would be doubtful if lithopedion formation would take place within the uterus, for the occurrence of resultant infection in this organ would cause destruction of the fetal tissues . At present there are two systems of classifying the condition of calcification, in whole or in part, of the products of conception . Kuchenmeister' proposed a most satisfactory grouping. In his first category is the lithokelyphos, meaning stone sheath, the condition in which the calcification is limited to the fetal membranes . The fetus, which is surrounded by the stone sheath, may be subject to some change, but the calcifying process does not implicate it . His second category is that of the lithokelyphopedion, in which both the fetus and the membranes are petrified and the amniotic fluid is absent, due possibly to leakage or absorption . The third division is the lithopedion or stone child . In this instance the fetus alone has become stony or petrified by the deposition of lime salts .
Another equally adequate and actually very similar classification has been offered by Masson and Simon 6 They use the term lithopedion to describe the entire group . Lithokelyphos and lithokelyphopedion are retained as in Kuchenmeister's grouping, and the word "lithotecnon" is introduced to describe the fetus infiltrated with calcium salts, with no or only negligible involvement of the fetal membranes . The incidence of lithopedion formation in eccyesis has been variously reported', 6,8,9 in the last decade as o .81 per cent, 1 .5 to 1 .8 per cent, 2 .o per cent, and 1 .03 per cent . Undoubtedly a decrease even in this low incidence can be expected since the diagnosis of extrauterine gestation is now made more promptly and accurately, and early surgical intervention is universal . In 1951 Anderson, Counselor and Woolners reported three cases of lithopedion or lithotecnon and stated the total number of reported cases to be 252 at that time . Four additional cases have been reported" -" since 1951 and two cases are presented herein . Lithopedions may affect their hosts in various ways . They may be carried for many years as asymptomatic, benign foreign bodies, sometimes being found only at necropsy,'" 14 or they may initiate distressing, disabling and perplexing symptoms, either early or late . Immediate surgical removal is indicated upon recognition .' In the two cases reported herein the patients' symptoms and the initial examinations indicated rectal lesions, the first as a case of apparent fecal impaction or obstruction due to a foreign body and the second as a draining rectal fistula . Foreign bodies of the intestinal tract are found most frequently in the sigmoidal and rectal areas because of the anatomic configuration of these organs . These foreign bodies can be grouped according to their origin, as follows : 898
Lithopedion (i) those ingested, (2) those formed within the intestinal tract, including the fecal impaction resulting from a fecalith or coprolith, (3) those from a neighboring viscus and (4) those introduced via the rectum ." Symptoms depend on many factors including the size and shape of the material, the duration of its presence and the amount of damage caused . The most frequent symptoms of fecal impaction are those of obstipation, coming on rather suddenly with some intestinal distention, and rectal pain ." There may be a frequent desire to defecate . An anorectal fistula is an abnormal tract or communication connecting the anorectum with some adjacent viscus or skin surface . Anorectal fistulas are almost always preceded by abscess formation in the anal region ." Pelvirectal abscess is not uncommon and its etiology is varied, but among the factors giving rise to this condition is the traumatism arising from the presence of a foreign body . CASE REPORTS CASE i . An eighty-one year old white widow was admitted to the Millard Fillmore Hospital on August 20, 1952, with the chief complaints of lower abdominal pain, slight abdominal distention and obstipation of approximately thirty-six hours' duration . There was no nausea or emesis . Very little flatus had been passed from the onset of her symptoms until the time of admission . The past history was essentially negative . She had never had any similar episodes . She had had no surgery or any serious illnesses . She had been pregnant three times and had three children . Her menses, except for the three instances of pregnancy, had always been regular and had terminated thirty-two years previously . Physical examination revealed no abnormalities other than distention and hyper-resonance of the abdomen . Rectal examination disclosed a firm, hard mass obstructing the rectum approximately 8 cm . from the anus . Pelvic examination showed a marital atrophic introitus . The uterus was small and anteflexed . Results of laboratory studies were within normal limits . The diagnosis of fecal impaction was made, and because of the patient's age it was decided to remove this obstruction under general anesthesia . Operation was performed the same day . The rectal mass was grasped with a sponge forceps,
899
brought into view and carefully dissected from the bowel wall to which it was attached . A small hole observed in the anterior rectal wall was closed . The patient's postoperative course was uneventful and she was discharged on the eighth day . The specimen removed was an irregular, yellowish gray fragment of firm, osseous tissue in which two small spicules of bone could be seen . These fragments of bone measured approximately 12 mm . in length and i mm . in diameter ; their surface was irregular and there were numerous small projections . In one area of the specimen there were several laminated fragments of bony tissue which suggested the thoracic cage in outline . Cut section disclosed some yellowish cheesy material and small particles of bone . Microscopic examination revealed multiple fragments of partially calcified, degenerated hone, hyaline cartilage and necrotic tissue intermingled with old blood . Along one edge portions of tubal wall with preserved epithelium were present . CASE 11 . A twenty-one year old married colored woman was admitted to the Millard Fillmore Hospital on October 29, 1953, with a history of having dull pain in the suprapubic area and in the right lower quadrant of the abdomen of about one month's duration . Following onset of this pain she said she had noticed a whitish mucoid discharge from her rectum ; one week prior to hospitalization she had a rather profuse rectal discharge of a yellow, purulent fluid, after which the pain had disappeared . A whitish discharge, however, continued . Examination by her physician prior to admission revealed a marital introitus, bilateral adnexal tenderness and a fullness in the cul-de-sac . The uterus was not enlarged or remarkable in any way . Proctoscopy showed a fistula draining into the rectum . Her past history was non-contributory and included no surgery or serious illnesses . Her menstrual periods had always been regular with the exception of two pregnancies, one at the age of fifteen and the other at the age of eighteen . Both terminated by spontaneous abortion . Subsequent to the first abortion she had had a vaginal discharge for which she received oral medication . Physical examination showed no abnormalities except for slight protuberance of the abdomen and mild right lower quadrant tender-
Lithopedion
I
2
Lateral view of gross specimen (lithopedion) removed from abscess cavity in Case u . FIG. 2 . Contralateral view of same specimen . The formation of the rib cage, femur, tibia, fibula and other bones can be seen . FIG . I .
4 3 In the roentgenogram of the specimen, same view as Figure 2, the bony parts can be further identified . FIG . 4 . This view also clearly demonstrates calcium deposition in soft tissues . FIG . 3 .
goo
Lithopedion ness . Sigmoidoscopy revealed an anorectal fistula which was surrounded by an area of inflammation and induration . Roentgenographic studies included a scout film of the abdomen, an upper gastrointestinal tract series and a mucosal relief roentgenogram . These were negative . Results of laboratory studies were within normal limits . On November loth a laparotomy was performed . Lysis of adhesions between the right fallopian tube and ovary and the sigmoid colon revealed an abscess cavity containing a hard calcified mass about 41.2 cm . in its longest axis . This mass had eroded into the rectum . The mass was removed and a right salpingooophorectomy and a sigmoidal loop colostomy were performed . Three ,Weeks later an end-toend anastomosis of the sigmoid colon was effected . The patient's postoperative course Was uneventful and she was discharged on December 12, 1953 . The specimen was a partially calcified bone structure approximately 4t°2 e m . i n length and 2 1 9 cm . in its widest diameter . This possessed the anatomic configuration of a fetus (Figs . i and 2) and consisted of multiple bones, including ribs, scapula, femur and spine . Roentgenogranms of the specimen clearly defined the fetal structure. (Figs . 3 and 4 .) SUMMARY 1 . The incidence and the classification of Iithopedion formation are reviewed . 2 . Two cases of lithotecnons or lithopedions are presented, bringing the total number of reported cases to 258 . 3 . In one case the patient apparently had carried her calcified fetus as an asymptomatic foreign body for over thirty years .
4 . In both cases the presence of the lithopedion resulted in rectal symptoms and lesions. REFERENCES
C . B . and KIMBROUGH, R . A . Clinical Obstetrics . Philadelphia, 1953 . J . B . Lippincott Co . 2 . TM S, P . Management of Obstetrical Difficulties, 4th ed . St . Louis, 1940 . C. V . Mosby Co. 3 . EASTMAN, N . J . Williams' Obstetrics, Loth ed . New York, 1950 . Appleton-Century-Crofts, Inc . 4- GREENHILL, J . P . Principles and Practices of Obstetrics, loth ed . Philadelphia, 1951 . AV . B . Saunders Co . i- MASSON, J . C. and SID1oN, FL E . Extra-uterine pregnancy ; lithopedion, Surg ., Gvnec . e'- Obst ., 46 : 500, 1028 . 6 . ANDERSON, J . R ., COUNSELOR, V . S . and WOOLNER, L . B . Lithopedion, report of an unusual case and notes of two other cases . Am . J. Obst . e'-° Cvnec ., 62 :439, 1 951 . KUCHENx1EISTER . Arcb . f. Gvndk ., 17 : 1 53, 1881 . Quoted by Brandman, L ." 8 . SCHt .MANN, E . A . Extra-uterine Pregnancy . New York, 1921 . D . Appleton Co . Quoted by Anderson, Counselor and Woolner." 9 . A1ADERN4, N . A rare case of lithopedion diagnosed radiologically . Arcb . Radiol., 21 : 121, 1947 . Quoted by Anderson, Counselor and \\•'oolner ." 10 .\IIRGON, B . Two cases of extra-uterine pregnancy at term : the first, lithopedion at term, the second, macerated fetus at terns . Afrique franc . chit ., 3 : 65, 1952 . 1 1 . \MCC1 .i RE, J . 11 . and EPPERSON, J . \V . NN' . Lithopedion pregnancy . Ohio State M . J., 48 : 3, 1952 . 12 . DANIS, J . E . Lithopedion . Am . J. Ob.ct . e- Cvnec ., 65 : 673, 19i313 . GLASS, B . A . and ABRAMSON, P . D . Volvulus of cecum due to lithopedion . Ain . J . Surg ., 86 : 348, 1 . LL 1.1,
1953 . 14 . BRANDMAN,
15 .
16 .
L . True lithopedion ; incidental finding at necropsy and review of the literature . Am . J . Obst . c'" Gy-nec ., 54 : 887, 1947 . BACON, II . E . Anus, Rectum, and Sigmoid Colon, vol . 2, 3rd ed . Philadelphia, 1049-J . B . Lippincott
CO . FIIRSCHMAN, L . J .
Synopsis of Ano-rectal Diseases, 2nd ed . St . Louis, 1942 . C . V . Mosby Co .
A.
RODENBERG, M.D . AND ANTHONY V . POSTOLOFF, M .D .,
Diplomate, American Board of
Pathology, Buffalo, New York From the Department of Obstetrics and Gynecology and the Department of Pathology, Millard Fillmore Hospital, Buffalo, N. I'.
E
CTOPIC gestation is stated to occur approximately once in 300 pregnancies .' -' Among
the more unusual results of such extrauterine fetation is mummification or calcification of the fetus-the lithopedion or "stone child ." It has been postulated 1,3,4 that a fetus which has been retained in the uterus for a considerable period of time could, by the deposition of lime salts, be transformed into a lithopedion, particularly in instances of missed labor or missed abortion . Others," however, believe that calcification of the fetus or the membranes, or both, occurs only with extrauterine pregnancy . Masson and Simon' state that three conditions are essential to the production of a lithopedion : (i) the development of a fetus to at least the third month, (2) retention of such a fetus within the abdomen and (3) the absence of infection . Thus it would be doubtful if lithopedion formation would take place within the uterus, for the occurrence of resultant infection in this organ would cause destruction of the fetal tissues . At present there are two systems of classifying the condition of calcification, in whole or in part, of the products of conception . Kuchenmeister' proposed a most satisfactory grouping. In his first category is the lithokelyphos, meaning stone sheath, the condition in which the calcification is limited to the fetal membranes . The fetus, which is surrounded by the stone sheath, may be subject to some change, but the calcifying process does not implicate it . His second category is that of the lithokelyphopedion, in which both the fetus and the membranes are petrified and the amniotic fluid is absent, due possibly to leakage or absorption . The third division is the lithopedion or stone child . In this instance the fetus alone has become stony or petrified by the deposition of lime salts .
Another equally adequate and actually very similar classification has been offered by Masson and Simon 6 They use the term lithopedion to describe the entire group . Lithokelyphos and lithokelyphopedion are retained as in Kuchenmeister's grouping, and the word "lithotecnon" is introduced to describe the fetus infiltrated with calcium salts, with no or only negligible involvement of the fetal membranes . The incidence of lithopedion formation in eccyesis has been variously reported', 6,8,9 in the last decade as o .81 per cent, 1 .5 to 1 .8 per cent, 2 .o per cent, and 1 .03 per cent . Undoubtedly a decrease even in this low incidence can be expected since the diagnosis of extrauterine gestation is now made more promptly and accurately, and early surgical intervention is universal . In 1951 Anderson, Counselor and Woolners reported three cases of lithopedion or lithotecnon and stated the total number of reported cases to be 252 at that time . Four additional cases have been reported" -" since 1951 and two cases are presented herein . Lithopedions may affect their hosts in various ways . They may be carried for many years as asymptomatic, benign foreign bodies, sometimes being found only at necropsy,'" 14 or they may initiate distressing, disabling and perplexing symptoms, either early or late . Immediate surgical removal is indicated upon recognition .' In the two cases reported herein the patients' symptoms and the initial examinations indicated rectal lesions, the first as a case of apparent fecal impaction or obstruction due to a foreign body and the second as a draining rectal fistula . Foreign bodies of the intestinal tract are found most frequently in the sigmoidal and rectal areas because of the anatomic configuration of these organs . These foreign bodies can be grouped according to their origin, as follows : 898
Lithopedion (i) those ingested, (2) those formed within the intestinal tract, including the fecal impaction resulting from a fecalith or coprolith, (3) those from a neighboring viscus and (4) those introduced via the rectum ." Symptoms depend on many factors including the size and shape of the material, the duration of its presence and the amount of damage caused . The most frequent symptoms of fecal impaction are those of obstipation, coming on rather suddenly with some intestinal distention, and rectal pain ." There may be a frequent desire to defecate . An anorectal fistula is an abnormal tract or communication connecting the anorectum with some adjacent viscus or skin surface . Anorectal fistulas are almost always preceded by abscess formation in the anal region ." Pelvirectal abscess is not uncommon and its etiology is varied, but among the factors giving rise to this condition is the traumatism arising from the presence of a foreign body . CASE REPORTS CASE i . An eighty-one year old white widow was admitted to the Millard Fillmore Hospital on August 20, 1952, with the chief complaints of lower abdominal pain, slight abdominal distention and obstipation of approximately thirty-six hours' duration . There was no nausea or emesis . Very little flatus had been passed from the onset of her symptoms until the time of admission . The past history was essentially negative . She had never had any similar episodes . She had had no surgery or any serious illnesses . She had been pregnant three times and had three children . Her menses, except for the three instances of pregnancy, had always been regular and had terminated thirty-two years previously . Physical examination revealed no abnormalities other than distention and hyper-resonance of the abdomen . Rectal examination disclosed a firm, hard mass obstructing the rectum approximately 8 cm . from the anus . Pelvic examination showed a marital atrophic introitus . The uterus was small and anteflexed . Results of laboratory studies were within normal limits . The diagnosis of fecal impaction was made, and because of the patient's age it was decided to remove this obstruction under general anesthesia . Operation was performed the same day . The rectal mass was grasped with a sponge forceps,
899
brought into view and carefully dissected from the bowel wall to which it was attached . A small hole observed in the anterior rectal wall was closed . The patient's postoperative course was uneventful and she was discharged on the eighth day . The specimen removed was an irregular, yellowish gray fragment of firm, osseous tissue in which two small spicules of bone could be seen . These fragments of bone measured approximately 12 mm . in length and i mm . in diameter ; their surface was irregular and there were numerous small projections . In one area of the specimen there were several laminated fragments of bony tissue which suggested the thoracic cage in outline . Cut section disclosed some yellowish cheesy material and small particles of bone . Microscopic examination revealed multiple fragments of partially calcified, degenerated hone, hyaline cartilage and necrotic tissue intermingled with old blood . Along one edge portions of tubal wall with preserved epithelium were present . CASE 11 . A twenty-one year old married colored woman was admitted to the Millard Fillmore Hospital on October 29, 1953, with a history of having dull pain in the suprapubic area and in the right lower quadrant of the abdomen of about one month's duration . Following onset of this pain she said she had noticed a whitish mucoid discharge from her rectum ; one week prior to hospitalization she had a rather profuse rectal discharge of a yellow, purulent fluid, after which the pain had disappeared . A whitish discharge, however, continued . Examination by her physician prior to admission revealed a marital introitus, bilateral adnexal tenderness and a fullness in the cul-de-sac . The uterus was not enlarged or remarkable in any way . Proctoscopy showed a fistula draining into the rectum . Her past history was non-contributory and included no surgery or serious illnesses . Her menstrual periods had always been regular with the exception of two pregnancies, one at the age of fifteen and the other at the age of eighteen . Both terminated by spontaneous abortion . Subsequent to the first abortion she had had a vaginal discharge for which she received oral medication . Physical examination showed no abnormalities except for slight protuberance of the abdomen and mild right lower quadrant tender-
Lithopedion
I
2
Lateral view of gross specimen (lithopedion) removed from abscess cavity in Case u . FIG. 2 . Contralateral view of same specimen . The formation of the rib cage, femur, tibia, fibula and other bones can be seen . FIG . I .
4 3 In the roentgenogram of the specimen, same view as Figure 2, the bony parts can be further identified . FIG . 4 . This view also clearly demonstrates calcium deposition in soft tissues . FIG . 3 .
goo
Lithopedion ness . Sigmoidoscopy revealed an anorectal fistula which was surrounded by an area of inflammation and induration . Roentgenographic studies included a scout film of the abdomen, an upper gastrointestinal tract series and a mucosal relief roentgenogram . These were negative . Results of laboratory studies were within normal limits . On November loth a laparotomy was performed . Lysis of adhesions between the right fallopian tube and ovary and the sigmoid colon revealed an abscess cavity containing a hard calcified mass about 41.2 cm . in its longest axis . This mass had eroded into the rectum . The mass was removed and a right salpingooophorectomy and a sigmoidal loop colostomy were performed . Three ,Weeks later an end-toend anastomosis of the sigmoid colon was effected . The patient's postoperative course Was uneventful and she was discharged on December 12, 1953 . The specimen was a partially calcified bone structure approximately 4t°2 e m . i n length and 2 1 9 cm . in its widest diameter . This possessed the anatomic configuration of a fetus (Figs . i and 2) and consisted of multiple bones, including ribs, scapula, femur and spine . Roentgenogranms of the specimen clearly defined the fetal structure. (Figs . 3 and 4 .) SUMMARY 1 . The incidence and the classification of Iithopedion formation are reviewed . 2 . Two cases of lithotecnons or lithopedions are presented, bringing the total number of reported cases to 258 . 3 . In one case the patient apparently had carried her calcified fetus as an asymptomatic foreign body for over thirty years .
4 . In both cases the presence of the lithopedion resulted in rectal symptoms and lesions. REFERENCES
C . B . and KIMBROUGH, R . A . Clinical Obstetrics . Philadelphia, 1953 . J . B . Lippincott Co . 2 . TM S, P . Management of Obstetrical Difficulties, 4th ed . St . Louis, 1940 . C. V . Mosby Co. 3 . EASTMAN, N . J . Williams' Obstetrics, Loth ed . New York, 1950 . Appleton-Century-Crofts, Inc . 4- GREENHILL, J . P . Principles and Practices of Obstetrics, loth ed . Philadelphia, 1951 . AV . B . Saunders Co . i- MASSON, J . C. and SID1oN, FL E . Extra-uterine pregnancy ; lithopedion, Surg ., Gvnec . e'- Obst ., 46 : 500, 1028 . 6 . ANDERSON, J . R ., COUNSELOR, V . S . and WOOLNER, L . B . Lithopedion, report of an unusual case and notes of two other cases . Am . J. Obst . e'-° Cvnec ., 62 :439, 1 951 . KUCHENx1EISTER . Arcb . f. Gvndk ., 17 : 1 53, 1881 . Quoted by Brandman, L ." 8 . SCHt .MANN, E . A . Extra-uterine Pregnancy . New York, 1921 . D . Appleton Co . Quoted by Anderson, Counselor and Woolner." 9 . A1ADERN4, N . A rare case of lithopedion diagnosed radiologically . Arcb . Radiol., 21 : 121, 1947 . Quoted by Anderson, Counselor and \\•'oolner ." 10 .\IIRGON, B . Two cases of extra-uterine pregnancy at term : the first, lithopedion at term, the second, macerated fetus at terns . Afrique franc . chit ., 3 : 65, 1952 . 1 1 . \MCC1 .i RE, J . 11 . and EPPERSON, J . \V . NN' . Lithopedion pregnancy . Ohio State M . J., 48 : 3, 1952 . 12 . DANIS, J . E . Lithopedion . Am . J. Ob.ct . e- Cvnec ., 65 : 673, 19i313 . GLASS, B . A . and ABRAMSON, P . D . Volvulus of cecum due to lithopedion . Ain . J . Surg ., 86 : 348, 1 . LL 1.1,
1953 . 14 . BRANDMAN,
15 .
16 .
L . True lithopedion ; incidental finding at necropsy and review of the literature . Am . J . Obst . c'" Gy-nec ., 54 : 887, 1947 . BACON, II . E . Anus, Rectum, and Sigmoid Colon, vol . 2, 3rd ed . Philadelphia, 1049-J . B . Lippincott
CO . FIIRSCHMAN, L . J .
Synopsis of Ano-rectal Diseases, 2nd ed . St . Louis, 1942 . C . V . Mosby Co .