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Spontaneous rupture of the spleen in third trimester of pregnancy. Report of a case and review of the literature
Eur. J. Obstet. Gynecol. Reprod. Biol., 25 (1987) 243-247
243
Elsevier EJO 00479
Spontaneous rupture of the spleen in third trimester of pregnancy. Report of a case and review of the literature Jan de Graaff
and Pierre M. Pijpers
Department of Obstetrics and Gynecology and Department of Pathology, St. Elisabeth Ziekenhuis, Tilburg The Nether/an&
Accepted for publication 3 February 1987
A case of idiopathic spontaneous rupture of the spleen in the third trimester of pregnancy is presented. The cause remains an enigma; the diagnosis is difficult, but echography may prove to be helpful. The treatment requires early exploration to enhance fetal as well as maternal survival. In this case mother and child did not survive. The etiology and clinical picture are discussed. The literature relating to rupture of the spleen is reviewed, including one particular case of a rupture in the third trimester of pregnancy. Rupture of the spleen; Echography; Third trimester
Introduction Rupture of the spleen is a rare and unpredictable cause of collapse in pregnancy. It is associated with appreciable maternal and fetal mortality. Celsus reported in the 15th century the first case of splenic rupture, and Saxtorph in 1803 the first case of splenic rupture in pregnancy [l]. In 1958 Sparkman collected 42 cases of rupture of the spleen in pregnancy from the literature, and reported two cases of his own [2]. Buchsbaum summarized the literature from 1958 till 1967 and reported 26 cases, including one of his own [l]. A review of the last 16 years (1969-1984) reveals 9 additional cases [3-91. Rupture of the spleen in Correspondence: J. de Graaff, M.D., Department of Obstetrics and Gynecology, St. Elisabeth Ziekenhuis, Tilburg, The Netherlands.
0028-2243/87/$03.50
0 1987 Elsevier Science Publishers B.V. (Biomedical Division)
244
pregnancy can be classified as traumatic or spontaneous. A spontaneous rupture may occur in a previously diseased spleen, i.e. in associated diseases such as mononucleosis infectiosa, leukemia sarcoidosis, amyloidosis, malaria etc. [lo]. It is doubtful whether idiopathic spontaneous rupture occurs at all. Many authorities believe that the patient is unable to recollect trivial trauma [ll]. Orloff and Peskin [12] applied a set of rigid criteria to fulfill the diagnosis ‘spontaneous splenic rupture’, viz. there must be no history whatsoever of trauma on thorough prospective and retrospective questioning, nor evidence of a systemic disease that could be associated with splenic pathology. Finally, there should be no evidence of local disease or previous trauma in the spleen at surgery or histologic examination. The clinical picture may be immediate or delayed (biphasic rupture) with a latent period of some days. During the first phase there is a short period of upper abdominal pain, and/or back-ache across the subscapular region of the back. Most patients also complain of acute (subacute) pain in the left hypochondrium, or diffuse abdominal or lower quadrant pain. Left shoulder pain is often present. In cases where continuous intraperitoneal bleeding occurs, the abdominal tenderness becomes diffuse, the abdomen rigid, and the patient shows the clinical picture of shock. A progressive enlargement of the abdomen may be noticed. Physical examination reveals mostly marked pallor, hypotensive blood pressure, tachycardia, a distended abdomen showing guarding and rigidity. The uterine fundus can hardly be felt, and fetal heart tones are no longer audible. Pelvic examination reveals a bulging posterior fornix. Treatment consists of blood replacement and splenectomy [lo]. A preoperative diagnosis is rarely made and even less frequently carried out in cases of spontaneous rupture of the spleen. The clinical picture mimics a ruptured ectopic pregnancy during the first trimester of pregnancy, and in second and third trimester abruptio placentae, uterine rupture and rupture of uterine or other intra-abdominal vessels such as aneurysms [ 131. Rupture of the spleen during delivery and in the early post-partum period is generally classified as traumatic. Maternal mortality is high. Figures vary from 30 to 70%. Fetal mortality is even higher (80-908) [5,10]. Case report A 25-yr-old Caucasian woman, para II, gravida III, was admitted to the St. Elisabeth Hospital by her general practitioner, in the 30th week of pregnancy, with a history of collapse for a short time, and abdominal pain for several hours following. The course of her current pregnancy was uneventful until that moment. On admission the patient looked quite well, but complained about abdominal pain. There was no vaginal bleeding. Physical examination revealed rigidity of the uterus and tenderness of the abdomen. The blood pressure was 90/60 mmHg, the pulse rate 100 beats per min. Fetal heart tones were not audible but echographical examination showed 60 beats per min. Some minutes later the fetal heart tones were absent. Echographic examination did not show intra-abdominal fluid. The provisional diagnosis was abruptio placentae. Intravenous access was established, and plasma expanders and whole blood were given. Within 30 min after admission the
Fig. 1. Intact part of the capsule of the spleen with fresh bleedings in the parenchymous tissue. (H&E, x 150)
Fig. 2. Ruptured part of the capsule of the spleen. (H&E, x150).
246
patient’s condition worsened. She went into deep shock and had a cardiac and respiratory arrest. She was put on artificial endotrachial oxygenation and an external heart massage was performed. At that time an enlargement of the abdomen was noticed. A second echography showed a lot of fluid outside the uterus in the abdomen. Laboratory findings showed an Hb of 3.4 mmol/l. Signs of diffuse intravascular coagulation were lacking. After several electric defibrillations, external heart massage, oxygenation and intravenous administration of plasma expanders and whole blood, it was decided to perform an explorative laparotomy. Three and a half hours after admission the abdomen was opened by a lower midline incision. It contained more than 4 liters of blood. No abnormality of the uterus was detected. On further inspection a large tear in the spleen was seen. Splenectomy was performed followed by a hysterectomy. During the post-operative course, it was impossible to obtain a normal blood pressure. Laboratory findings showed a severe DIC; the EEG was nearly flat, and the ECG was pathological. It was decided to stop further treatment. The patient died 11 h after admission. Post-mortem findings of the mother showed signs of ischaemia in several organs, and decompensatio cordis. The removed spleen weighed 258 g, measured 14 x 11 x 2.2 cm, and showed a tear of 5 cm at the hilus. Microscopic examination of the spleen only revealed some small fresh bleedings (Figs. 1 and 2). The structure was quite normal; there were no abnormalities in the parenchymous tissue. Iron staining was negative. Discussion Idiopathic spontaneous rupture of the spleen in pregnancy is a very rare complication. In the early literature rupture of the spleen during pregnancy was thought to be due to changes in organ position caused by the growing uterus, and the hypervolaemia during pregnancy [lo]. In that case one would expect the highest incidence during the last trimester of pregnancy. However, after excluding traumatically and pathologically changed spleens, no data have demonstrated that ruptures occur more frequently in early as opposed to late pregnancy. Our case was diagnosed as abruptio placentae. Surgical intervention was therefore delayed until a stage of irreversible shock. We believed that in this case the criteria of Orloff and Peskin were fulfilled and therefore this case must be classified as an idiopathic spontaneous rupture of the spleen in the third trimester of pregnancy. Research carried out by Sparkman showed 3 cases of third-trimester spontaneous ruptures of the spleen, whereas Buchsbaum reported 2 cases. Henderson and Keeping [7] described another 2 cases, one during the third stage of labour, the other in the 37th week of gestation. In both cases the mothers and babies survived. Epstein et al. [5] described one case of biphasic rupture in a patient in late pregnancy. In this case mother and child survived, due to rapid surgical intervention. Studies carried out by Sparkman, Buchsbaum, Henderson and Epstein indicate that rapid surgical intervention might save the lives of mothers and babies. Our case
241
illustrates the importance of an intraperitoneal haemorrhage as a cause of collapse in pregnancy. Careful echographic examination should lead to an early diagnosis of abdominal bleeding, requiring immediate surgical intervention. References 1 Buchsbaum HJ. Splenic rupture in pregnancy. Report of a case and review of the literature. Obstet Gynecol Surv 1967;22:381-395. 2 Sparkman RS. Rupture of the spleen in pregnancy. Report of two cases and review of the literature. Am J Obstet Gynecol 1958;76:587-598. 3 Christau S, Klebe JG. Rupture of the spleen during delivery. Acta Obstet Gynecol Stand 1978;57: 187-188. 4 Ellis GF, Cantor, B. Splenic rupture in pregnancy. Am J Obstet Gynecol 1978;132:220-221. 5 Epstein M, King R, Kenney D. Splenic rupture at term. Case report. Missouri Med 1983;80:83-84. 6 Gudgeon, CW. Splenic complicating pregnancy. Aust NZJ Obstet Gynecol 1967;7:99-100. 7 Henderson PR, Keeping JD. Spontaneous rupture of the spleen in late pregnancy. Aust NZJ Obstet Gynecol. 1979;19:116-118. 8 Holt S. Spontaneous rupture of a normal spleen diagnosed as ruptured ectopic pregnancy. Two case reports. Br J Obstet Gynaecol 1982;89:1062-1063. 9 Smith EB. Rupture of the spleen in pregnancy. J Nat1 Med Assoc 1971;65:281-282. 10 Hoffman RL. Rupture of the spleen, A review and report of a case following abdominal hysterectomy. Am J Obstet Gynecol 1972;113:524-530. 11 Bankole, MA, Kent SW. Spontaneous rupture of spleen in pregnancy. Arch Surg 1966;92:120-122. 12 Orloff MJ, Peskin GW. Spontaneous rupture of the normal spleen- a surgical enigma. Int Abst Surg 1958;106:1-5. 13 Furler IK, Robertson DNS. Spontaneous rupture of the splenic artery in pregnancy. Lancet 1962;i:588-590.
243
Elsevier EJO 00479
Spontaneous rupture of the spleen in third trimester of pregnancy. Report of a case and review of the literature Jan de Graaff
and Pierre M. Pijpers
Department of Obstetrics and Gynecology and Department of Pathology, St. Elisabeth Ziekenhuis, Tilburg The Nether/an&
Accepted for publication 3 February 1987
A case of idiopathic spontaneous rupture of the spleen in the third trimester of pregnancy is presented. The cause remains an enigma; the diagnosis is difficult, but echography may prove to be helpful. The treatment requires early exploration to enhance fetal as well as maternal survival. In this case mother and child did not survive. The etiology and clinical picture are discussed. The literature relating to rupture of the spleen is reviewed, including one particular case of a rupture in the third trimester of pregnancy. Rupture of the spleen; Echography; Third trimester
Introduction Rupture of the spleen is a rare and unpredictable cause of collapse in pregnancy. It is associated with appreciable maternal and fetal mortality. Celsus reported in the 15th century the first case of splenic rupture, and Saxtorph in 1803 the first case of splenic rupture in pregnancy [l]. In 1958 Sparkman collected 42 cases of rupture of the spleen in pregnancy from the literature, and reported two cases of his own [2]. Buchsbaum summarized the literature from 1958 till 1967 and reported 26 cases, including one of his own [l]. A review of the last 16 years (1969-1984) reveals 9 additional cases [3-91. Rupture of the spleen in Correspondence: J. de Graaff, M.D., Department of Obstetrics and Gynecology, St. Elisabeth Ziekenhuis, Tilburg, The Netherlands.
0028-2243/87/$03.50
0 1987 Elsevier Science Publishers B.V. (Biomedical Division)
244
pregnancy can be classified as traumatic or spontaneous. A spontaneous rupture may occur in a previously diseased spleen, i.e. in associated diseases such as mononucleosis infectiosa, leukemia sarcoidosis, amyloidosis, malaria etc. [lo]. It is doubtful whether idiopathic spontaneous rupture occurs at all. Many authorities believe that the patient is unable to recollect trivial trauma [ll]. Orloff and Peskin [12] applied a set of rigid criteria to fulfill the diagnosis ‘spontaneous splenic rupture’, viz. there must be no history whatsoever of trauma on thorough prospective and retrospective questioning, nor evidence of a systemic disease that could be associated with splenic pathology. Finally, there should be no evidence of local disease or previous trauma in the spleen at surgery or histologic examination. The clinical picture may be immediate or delayed (biphasic rupture) with a latent period of some days. During the first phase there is a short period of upper abdominal pain, and/or back-ache across the subscapular region of the back. Most patients also complain of acute (subacute) pain in the left hypochondrium, or diffuse abdominal or lower quadrant pain. Left shoulder pain is often present. In cases where continuous intraperitoneal bleeding occurs, the abdominal tenderness becomes diffuse, the abdomen rigid, and the patient shows the clinical picture of shock. A progressive enlargement of the abdomen may be noticed. Physical examination reveals mostly marked pallor, hypotensive blood pressure, tachycardia, a distended abdomen showing guarding and rigidity. The uterine fundus can hardly be felt, and fetal heart tones are no longer audible. Pelvic examination reveals a bulging posterior fornix. Treatment consists of blood replacement and splenectomy [lo]. A preoperative diagnosis is rarely made and even less frequently carried out in cases of spontaneous rupture of the spleen. The clinical picture mimics a ruptured ectopic pregnancy during the first trimester of pregnancy, and in second and third trimester abruptio placentae, uterine rupture and rupture of uterine or other intra-abdominal vessels such as aneurysms [ 131. Rupture of the spleen during delivery and in the early post-partum period is generally classified as traumatic. Maternal mortality is high. Figures vary from 30 to 70%. Fetal mortality is even higher (80-908) [5,10]. Case report A 25-yr-old Caucasian woman, para II, gravida III, was admitted to the St. Elisabeth Hospital by her general practitioner, in the 30th week of pregnancy, with a history of collapse for a short time, and abdominal pain for several hours following. The course of her current pregnancy was uneventful until that moment. On admission the patient looked quite well, but complained about abdominal pain. There was no vaginal bleeding. Physical examination revealed rigidity of the uterus and tenderness of the abdomen. The blood pressure was 90/60 mmHg, the pulse rate 100 beats per min. Fetal heart tones were not audible but echographical examination showed 60 beats per min. Some minutes later the fetal heart tones were absent. Echographic examination did not show intra-abdominal fluid. The provisional diagnosis was abruptio placentae. Intravenous access was established, and plasma expanders and whole blood were given. Within 30 min after admission the
Fig. 1. Intact part of the capsule of the spleen with fresh bleedings in the parenchymous tissue. (H&E, x 150)
Fig. 2. Ruptured part of the capsule of the spleen. (H&E, x150).
246
patient’s condition worsened. She went into deep shock and had a cardiac and respiratory arrest. She was put on artificial endotrachial oxygenation and an external heart massage was performed. At that time an enlargement of the abdomen was noticed. A second echography showed a lot of fluid outside the uterus in the abdomen. Laboratory findings showed an Hb of 3.4 mmol/l. Signs of diffuse intravascular coagulation were lacking. After several electric defibrillations, external heart massage, oxygenation and intravenous administration of plasma expanders and whole blood, it was decided to perform an explorative laparotomy. Three and a half hours after admission the abdomen was opened by a lower midline incision. It contained more than 4 liters of blood. No abnormality of the uterus was detected. On further inspection a large tear in the spleen was seen. Splenectomy was performed followed by a hysterectomy. During the post-operative course, it was impossible to obtain a normal blood pressure. Laboratory findings showed a severe DIC; the EEG was nearly flat, and the ECG was pathological. It was decided to stop further treatment. The patient died 11 h after admission. Post-mortem findings of the mother showed signs of ischaemia in several organs, and decompensatio cordis. The removed spleen weighed 258 g, measured 14 x 11 x 2.2 cm, and showed a tear of 5 cm at the hilus. Microscopic examination of the spleen only revealed some small fresh bleedings (Figs. 1 and 2). The structure was quite normal; there were no abnormalities in the parenchymous tissue. Iron staining was negative. Discussion Idiopathic spontaneous rupture of the spleen in pregnancy is a very rare complication. In the early literature rupture of the spleen during pregnancy was thought to be due to changes in organ position caused by the growing uterus, and the hypervolaemia during pregnancy [lo]. In that case one would expect the highest incidence during the last trimester of pregnancy. However, after excluding traumatically and pathologically changed spleens, no data have demonstrated that ruptures occur more frequently in early as opposed to late pregnancy. Our case was diagnosed as abruptio placentae. Surgical intervention was therefore delayed until a stage of irreversible shock. We believed that in this case the criteria of Orloff and Peskin were fulfilled and therefore this case must be classified as an idiopathic spontaneous rupture of the spleen in the third trimester of pregnancy. Research carried out by Sparkman showed 3 cases of third-trimester spontaneous ruptures of the spleen, whereas Buchsbaum reported 2 cases. Henderson and Keeping [7] described another 2 cases, one during the third stage of labour, the other in the 37th week of gestation. In both cases the mothers and babies survived. Epstein et al. [5] described one case of biphasic rupture in a patient in late pregnancy. In this case mother and child survived, due to rapid surgical intervention. Studies carried out by Sparkman, Buchsbaum, Henderson and Epstein indicate that rapid surgical intervention might save the lives of mothers and babies. Our case
241
illustrates the importance of an intraperitoneal haemorrhage as a cause of collapse in pregnancy. Careful echographic examination should lead to an early diagnosis of abdominal bleeding, requiring immediate surgical intervention. References 1 Buchsbaum HJ. Splenic rupture in pregnancy. Report of a case and review of the literature. Obstet Gynecol Surv 1967;22:381-395. 2 Sparkman RS. Rupture of the spleen in pregnancy. Report of two cases and review of the literature. Am J Obstet Gynecol 1958;76:587-598. 3 Christau S, Klebe JG. Rupture of the spleen during delivery. Acta Obstet Gynecol Stand 1978;57: 187-188. 4 Ellis GF, Cantor, B. Splenic rupture in pregnancy. Am J Obstet Gynecol 1978;132:220-221. 5 Epstein M, King R, Kenney D. Splenic rupture at term. Case report. Missouri Med 1983;80:83-84. 6 Gudgeon, CW. Splenic complicating pregnancy. Aust NZJ Obstet Gynecol 1967;7:99-100. 7 Henderson PR, Keeping JD. Spontaneous rupture of the spleen in late pregnancy. Aust NZJ Obstet Gynecol. 1979;19:116-118. 8 Holt S. Spontaneous rupture of a normal spleen diagnosed as ruptured ectopic pregnancy. Two case reports. Br J Obstet Gynaecol 1982;89:1062-1063. 9 Smith EB. Rupture of the spleen in pregnancy. J Nat1 Med Assoc 1971;65:281-282. 10 Hoffman RL. Rupture of the spleen, A review and report of a case following abdominal hysterectomy. Am J Obstet Gynecol 1972;113:524-530. 11 Bankole, MA, Kent SW. Spontaneous rupture of spleen in pregnancy. Arch Surg 1966;92:120-122. 12 Orloff MJ, Peskin GW. Spontaneous rupture of the normal spleen- a surgical enigma. Int Abst Surg 1958;106:1-5. 13 Furler IK, Robertson DNS. Spontaneous rupture of the splenic artery in pregnancy. Lancet 1962;i:588-590.