British Journal of Radiology (2003) 76, 704-711
© 2003 British Institute of Radiology
doi: 10.1259/bjr/69247894
Spontaneous rupture of the spleen: ultrasound patterns, diagnosis and follow-up
C Görg, MD1,
J Cölle1,
K Görg, MD1,
H Prinz, PhD2 and
G Zugmaier, MD1
1 Zentrum für Innere Medizin, Klinikum der Philipps-Universität, Baldingerstraße, 35043 Marburg and 2 Koordinationszentrum für klinische Studien, Klinikum der Philipps-Universität, Bunsenstraße, Marburg, Germany
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Abstract
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Spontaneous rupture of the spleen is an extremely rare complication usually of infectious diseases or disorders of the haematopoietic system and has been described mostly in case reports. The incidence, symptoms, causes, therapy, and prognosis are poorly defined. From July 1985 to January 2000 41 patients with spontaneous splenic rupture were diagnosed by abdominal ultrasound and confirmed by splenectomy (n=12), CT (n=15), and ultrasound follow up (n=26). An ultrasound grading system was retrospectively established based on the degree of splenic injury (grade 0–2=low grade injury, grade 3=high grade injury) and correlated with surgical procedures. 30 day mortality rate was studied in relation to underlying disorders, ultrasound grades and treatment decisions. 21 patients had underlying malignant disorders (group I) and 20 patients had benign diseases (group II). Between group I and II we observed a highly significant difference in 30 day mortality rates (n=7; 38.1% vs n=1; 5%, p<0.01), but no significant difference in high grade injury rate (n=3; 14.3% vs n=2; 10.0%; p=ns) and surgical treatment rate (n=5; 23.8% vs n=7; 35.0%; p=ns). Depending on ultrasound grades the surgical procedures were 0% for grade 0, 16.7% for grade 1, 30.4% for grade 2, and 60% for grade 3. There were no significant differences between patients, who died within the first 30 days (n=9) and those who survived more than 30 days (n=32) regarding high grade splenic injury rate (n=0; 0% vs n=5; 15.6%; p=ns), and surgical treatment rate (n=2; 22.2% vs n=10; 31.2%; p=ns). Spontaneous rupture of the spleen is an extremely rare event. It is associated with a high mortality rate within 30 days in patients with malignant disease. Sonomorphologic grading is helpful for treatment decisions. 30 day mortality rate is correlated with neither ultrasound grades, nor surgical treatment rates.
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Introduction
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Rupture of the spleen is a potentially life threatening complication usually occurring after blunt abdominal trauma with a frequency up to 40% [1]. Unlike traumatic splenic rupture spontaneous non-traumatic splenic rupture is extremely rare. The term spontaneous splenic rupture is poorly defined [2]. Wiedemann first defined the term as resulting from an "incident without external force", Knoblich distinguished the non-traumatic rupture of a pathological spleen from the extremely rare non-traumatic splenic rupture of unknown origin [3].
Ultrasound performed by experienced investigators is regarded as a valid and reliable method for diagnosis of splenic lesions [4, 5]. Because of its availability, low cost, and non-invasiveness ultrasound has been accepted as a diagnostic method in patients with abdominal trauma [6]. Several grading systems based on ultrasound and CT have been established for traumatic splenic ruptures and have been shown to be reliable and helpful for therapeutic decisions [7, 8].
Conversely there are no valid data on incidence rates, symptoms, causes, therapy and prognosis of spontaneous splenic rupture. Numerous case reports have been published, but a comprehensive assessment is still missing.
The goal of this retrospective study was to describe the clinical and ultrasound spectrum of spontaneous rupture of the spleen and to determine, whether an ultrasound grading system, and therapeutic procedures, are correlated with the 30 day mortality rates.
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Patients and methods
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From July 1985 to January 2000 670 patients with focal splenic lesions were documented by abdominal ultrasound at the department of sonography in a university medical centre. 41 patients were included in this study. The inclusion criteria for this retrospective evaluation were:
1. no clinical history of trauma
2. ultrasound evidence of splenic rupture.
The ultrasound criteria for diagnosis of splenic rupture have been described elsewhere, i.e. abdominal free blood, subcapsular haematoma, intraparenchymal bleeding [4, 5]. The ultrasound findings of splenic rupture in study patients were confirmed by surgery in 12 cases, by CT in 15 cases, and by ultrasound follow-up in 26 cases.
Through a standardized evaluation form the clinical data and the ultrasound findings were retrospectively collected from all patients included in the study.
Clinical evaluation
The following clinical findings were recorded: age; sex; underlying diseases; symptoms of pain at the time of diagnosed splenic rupture (no pain, diffuse pain, localized pain in the left upper abdomen).
Ultrasound findings
The size of the spleen was defined by two parameters (length=largest craniocaudal extension, width=axial diameter in the hilus region) [9]. A splenic size of 
5 x 11 cm was defined as normal. A size of >5 x 11 cm was defined as moderate splenomegaly, size of >6 x 16 cm: high grade splenomegaly, size of >8 x 20 cm: massive splenomegaly [10]. Any one measurement over the sizes given would put that spleen into the higher category.
The findings of other focal splenic lesions were documented. The ultrasound criteria for diagnosis of this have been described elsewhere, e.g. infiltration by lymphoma [10], infarction [11], metastasis [12], cyst [13], or abscess [14, 15]. All patients were investigated by colour Doppler ultrasound (CDS) for detection of arterial flow signals in liquid intraparenchymatous parts.
For the evaluation of splenic rupture the following grading system was used [4, 16]:
Low grade splenic injury:
Grade 0: - perisplenic blood without subcapsular splenic hematoma and without intraparenchymal bleeding ("sentinel clot" [17]).
Grade 1: - subcapsular haematoma: diameter 3 cm, or
- intraparenchymal bleeding/laceration: diameter 3 cm with sonomorphologically intact splenic capsule, or
- minimal free intra-abdominal pool of blood.
Grade 2: - subcapsular haematoma: diameter >3 cm, or
- intraparenchymal bleeding/laceration: diameter >3 cm with sonomorphologically intact splenic capsule, or
- moderate free intra-abdominal pool of blood.
High grade splenic injury:
Grade 3: - fragmentation of the spleen with the capsule torn apart, or
- lacking evidence of intraparenchymal blood flow in parts of the spleen, or
- detection of arterial flow signals in liquid intraparenchymatous parts by CDS (intraparenchymatous pseudoaneurysm [18]), or
- high grade free intra-abdominal blood.
The diagnosis of haematoperitoneum was confirmed by ultrasound guided puncture in all patients. The amount of free blood was determined semiquantitatively: minimal <500 ml, moderate <1000 ml, high grade >1000 ml [19].
The ultrasound examinations were performed with a LCS 7000 Picker International (Highland Heights, OH) with 3.5 MHz and 5 MHz sector transducers.
The CDS studies were performed with the ultrasonographs Acuson 128 and Acuson Sequoia (Mountain View, CA) with variable 3.5 MHz to 8 MHz sector transducers ultrasound studies were performed by CG and KG. The wall filter was kept at its lowest value, and the pulse-repetition frequency was adjusted manually to its lowest setting without aliasing.
The correlation between qualitative variables was studied with the Fisher's exact test, for quantitative parameters the t-test for paired samples was used.
Clinical and ultrasound course
The clinical and ultrasound courses were evaluated according to the following criteria:
- Surgical treatment: (early splenectomy 24 h after diagnosis, delayed splenectomy >24 h after diagnosis, splenorrhaphy).
- Conservative treatment: ultrasound follow-up with evaluation of the final state of the spleen [20].
- Overall survival: (death within the first 30 days after the diagnosis of splenic rupture, survival longer than 30 days after the diagnosis of splenic rupture).
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Results
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Clinical evaluation
The mean age was 54.2 years (range 20–82 years). 12 patients (29.3%) were female, 29 patients (70%) were male. The main underlying diseases are summarized in Table 1
. 8 of 41 patients (19%) did not have pain at the onset of splenic ruptures (Figure 1).
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Figure 1. Distribution of symptoms of pain at the time of diagnosed splenic rupture in 41 study patients.
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Ultrasound evaluation
The size of the spleen was normal in 8 patients (19.5%). Moderate splenomegaly was observed in 12 patients (29.3%), high grade splenomegaly was detected in 10 patients (24.4%), 11 patients (26.8%) had massive spleno-megaly.
Additional focal splenic lesions were found in 22 patients (53.6%). In 17 patients (41.5%) splenic infarction was diagnosed (Figure 2), 4 patients had splenic metastases (Figure 3), 1 patient had nodular lymphoma involvement of the spleen.
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Figure 2. 59-year-old patient with chronic myeloid leukaemia in the chronic phase and pain in the left upper abdomen. (a) Ultrasound of the spleen showed irregular delineated wedge shaped intrasplenic lesion (arrowheads) suggesting splenic infarction (INF), with a small echofree area (arrow). (b) On colour Doppler ultrasound a swirl flow was seen in the liquid intraparenchymatous parts suggesting a intrasplenic pseudoaneurysm, classified for grade 3 splenic injury (from reference [5] with the permission of the publisher).
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Figure 3. 38-year-old patient with metastatic malignant melanoma and pain in the left upper abdomen. (a) Ultrasound of the spleen showed a focal hypoechoic splenic lesions suggesting splenic metastases (M) with hyperechoic subcapsular haematoma (H) classified for a grade 2 splenic injury. (b) On ultrasound follow-up 6 weeks later a hypoechoic transformation of the haematoma was seen.
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Ultrasound grading
The initial ultrasound findings of the 41 study patients showed free intra-abdominal blood in 13 cases (31.7%), which was confirmed by ultrasound guided diagnostic puncture, subcapsular haematoma in 20 cases (48.8%) and intraparenchymal bleeding in 23 cases (56.1%). 5 patients (12.2%) had intrasplenic pseudoaneurysms. The individual classification of splenic rupture in different grades was carried out according to the criteria described above. If several lesions were present, the classification was based on the lesion with the highest ultrasound grade. 36 patients (87.8%) presented with low grade splenic injury (grade 0–2), and 5 patients (12.2%) had high grade splenic injury (grade 3) (Figure 4). The high grade injury rates in relation to the underlying diseases are summarized in Table 2.
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Table 2. Ultrasound grade, surgical treatment and 30 day mortality rates in relation to underlying diseases in 41 patients with spontaneous splenic rupture
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Clinical and ultrasound course
Surgical treatment
12 patients (29.2%) were treated surgically (Table 2
). Depending on ultrasound grades the surgical procedures were 0% for grade 0, 16.7% for grade 1, 30.4% for grade 2, and 60% for grade 3 splenic injury (Figure 5). Surgeon's decisions for surgical treatment were supported by the following ultrasound findings, singularly or in combination: secondary delayed splenic rupture (n=2) [21], increasing amounts of free intra-abdominal blood (n=4), increasing subcapsular haematoma (n=6), increasing intraparenchymal bleeding (n=7), non-traumatic intrasplenic pseudoaneurysms (n=3). Intrasplenic pseudoaneurysms were observed in 5 patients (12.2%) by CDS. In two cases surgery was rejected by the patients. In both cases spontaneous thrombosis of the aneurysms occurred. In 4 cases (9.8%) an early splenectomy was performed, a delayed splenectomy was performed in 7 patients (17.1%). One patient underwent surgery resulting in preservation of the spleen (delayed operation). The surgical treatment rate was 23.8% (n=5) for patients with malignant diseases and 35.6% (n=7) for patients with benign diseases (p=ns) (Table 2).
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Figure 5. Therapeutic procedures in relation to ultrasound grades in 41 patients with spontaneous splenic rupture.
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Conservative treatment
29 patients (70.8%) were treated conservatively (Figure 6). Ultrasound follow-up was performed in 26 out of 29 patients. The median time for the follow-up was 402 days (range 13.5 years–7 days). Follow-up abnormal findings of intrasplenic pseudocysts (n=2) and functional hyposplenia/asplenia (n=3) (7%) were observed (Figure 7). The diagnosis of functional hyposplenia/asplenia was confirmed by scintigraphy.
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Figure 6. 58-year-old patient with lower limb vein thrombosis. Coumarin therapy and pain in the left upper abdomen (from reference [5] with the permission of the publisher). (a) Ultrasound of the spleen showed a irregular delineated subcapsular haematoma (H) classified for grade 2 splenic injury. (b) On ultrasound follow-up 2 weeks later a hypoechoic transformation of the haematoma was seen. (c) On ultrasound follow up 6 weeks later a nearly complete healing was seen with a inhomogeneity of splenic texture. (d) CT at the time of primary diagnosis of spontaneous splenic rupture confirmed subcapsular haematoma of the spleen.
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Figure 7. 29-year-old patient with varicella sepsis and pain in the left upper abdomen. (a) Ultrasound of the spleen showed diffuse inhomogeneity of the splenic parenchyma (s). The spleen is covered by a hypoechoic mass suggesting subcapsular haematoma (H), classified for grade 2 splenic injury, (from reference [5] with the permission of the publisher). (b) On ultrasound follow-up 1 year later a small spleen was seen without flow signals by colour Doppler ultrasound (from Görg C, Schwerk WB (2000: Milz. In: Braun B, Günther R, Schwerk WB (Hrsg.). Ultraschalldiagnostik, Ecomed Verlagsgesellschaft, with the permission of the publisher). (c) Splenic scintigraphy using 270mbq 99Tcm-MDP. Physiological concentrations in the liver and an absent nuclide concentration in the spleen 20 min after injection, indicating functional asplenia.
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Overall survival
Within the first month after ultrasound diagnosis of spontaneous splenic ruptures nine of the patients (21.9%) included in the study died (Table 2). Eight of the nine patients had underlying malignant disorders (Table 3). One patient died of endocarditis. One patient with malignant disease died of complications due to the operation. The 30 day mortality rate was 38.1% (n=8) for patients with malignant diseases and 5.0% (n=1) for patients with benign diseases (p=<0.01) (Table 2).
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Table 3. Clinical data and causes of death in patients with spontaneous splenic rupture and death within 30 day at the onset of splenic ruptures diagnosed
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Discussion
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Spontaneous rupture of the spleen has been described mainly as case reports. It may be caused by either intrinsic or extrinsic factors. Infectious diseases especially mononucleosis, induce a swelling of the spleen due to hyperplasia of the pulpa and hyperaemia of the sinus and mantle plexus [22]. In malignant disorders the swelling of the spleen is caused by infiltrating malignant cells and extramedullary haematopoiesis [23]. Case reports have described spontaneous splenic rupture in splenic infarctions [24], coagulation disorders [23], thrombocytopenia [25], portal hypertension [26], vasculitis [27], venous thrombosis in the spleen [28], and focal splenic lesions [29]. In a review of pathologic rupture of the spleen in lymphoma and leukaemia, splenic infarction was present in 50% of patients with leukaemia and 20% of patients with lymphoma [30].
To date there have been no published data on the incidence rates of spontaneous ruptures of the spleen. Although the sensitivity of ultrasound for the detection of free intra-abdominal fluid is 98–100% [31], the sensitivity of ultrasound for the detection of acute splenic parenchymal lesions is 72–78% [32]. The specifity of ultrasound for the detection of splenic rupture is at a rate of 91–100% [31, 32]. The incidence resulting from this study is based on ultrasound screening, thus the true incidence may be higher.
As an additional factor for low incidence rates of spontaneous splenic rupture, clinically silent splenic pathology has to be mentioned. In the present study splenic rupture without clinical symptoms was observed by chance in 20% of the cases (Figure 4). The phenomenon of silent splenic pathology has been observed in patients with splenic infarction at a similar rate [10]. One may speculate that clinically silent spontaneous rupture of the spleen is frequently not diagnosed.
It has been well known that traumatic splenic ruptures predominantly occur in males. Interestingly also in the present study spontaneous splenic ruptures were observed more commenly in males than in females. We have found no explanation for this phenomenon.
After the first reports on the overwhelming post-splenectomy infection in children in 1952 [33], the indication for splenectomy has been viewed critically and a conservative approach have been applied whoever possible [34]. Several studies on traumatic splenic rupture and infarction of the spleen [7, 10] have shown, that splenic lesions can heal spontaneously. Thus, the decision to wait and watch may be an alternative for patients with splenic rupture. In the present study 71% of patients with non-traumatic splenic rupture were conservatively treated.
The rate of splenectomy was considerably lower for spontaneous splenic rupture compared with that of traumatic splenic rupture which has been described to be up to 50% [35]. One reason for this might be the different frequency of high grade splenic injury in traumatic and spontaneous splenic rupture. Our study has demonstrated high grade splenic injury in 12.2% of patients with spontaneous splenic rupture (Table 2). The rate of high grade splenic injury after blunt abdominal trauma is considerably higher and has been described to be up to 60% [16]. On the other hand indications for surgical treatment of splenic rupture depend on the underlying diseases and on the prognosis of the patients. In patients with end stage malignant disease and spontaneous splenic rupture a conservative approach is often warranted.
Several ultrasound and CT grading systems developed in retrospective and prospective studies on traumatic splenic rupture have been shown to be valuable assets for therapeutic decisions [4, 34, 35]. As shown in the present study ultrasound grades do correlate with the therapeutic decisions in patients with spontaneous splenic rupture (Figure 5).
Although 71% of the patients with spontaneous splenic rupture were treated conservatively, they showed a remarkable high mortality rate of 22% after 30 days. Patients with malignant disorders showed an even higher mortality rate of 38% in contrast to patients with benign diseases having a 30 day mortality rate of 5% (Table 2). One may speculate, that in patients with malignant disorders the progressing neoplastic disease is responsible for the high 30 day mortality rate (Table 3). Studies on traumatic splenic rupture report a (perioperative) mortality rate of 18% [36]. Additional abdominal complications are responsible for the high rate of mortality in these patients.
The intrasplenic pseudoaneurysm is a major complication of spontaneous splenic rupture (Figure 2) [18]. It was observed in our study by CDS in 12.2% of the cases investigated and was the main cause for classification of high grade splenic injury (Figure 4). Traumatic splenic ruptures may be followed by pseudoaneurysms at a similar rate [37]. Pseudoaneurysms usually have to be treated surgically, because they may cause secondary delayed splenic rupture [38] although in our study two of five spontaneously thrombosed.
In addition, functional asplenia was observed in 7% of the cases after spontaneous splenic ruptures. Functional asplenia has been reported for patients with sickle cell anaemia [39] and also in case reports with spontaneous splenic rupture [40]. Up to date functional asplenia has not been reported as a complication of traumatic splenic rupture. The ultrasound picture is characterized by an increasing reduction of the splenic size on follow up (Figure 3) [20]. The diagnosis should be confirmed by technetium-99m colloidscintigraphy or by the finding of Howell-Jolly bodies in the erythrocytes [41]. For these patients a triple vaccination is recommended, in order to avoid post-splenectomy infection [39].
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Conclusion
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Spontaneous splenic rupture is a rare event in patients with various underlying diseases. It is mostly associated with varying degrees of splenomegaly and has a male predominance. In this series about a fifth of patients did not have any abdominal pains and a half had additional focal splenic lesions. There is an increased mortality in patients with malignant disease.
Received for publication October 31, 2002.
Revision received June 11, 2003.
Accepted for publication July 14, 2003.
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Abstract
Introduction
