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Heliyon
Heliyon
Heliyon
2405-8440
Elsevier

S2405-8440(24)12106-8
10.1016/j.heliyon.2024.e36075
e36075
Case Report
Retroperitoneal leiomyomas with intratumoral bleeding: Presentation with multiple-organ dysfunction syndrome - A case report
Wang Shishi a1
Li Lihua ab1
Lin Ying ac
Su Jiawei ad
Shen Minghong fjslyyshenmh@fjmu.edu.cn
ab⁎
Lin Tan ab⁎⁎
a Shengli Clinical Medical College of Fujian Medical University, Fuzhou, Fujian Province, 350001, China
b Department of Gynecology, Fujian Provincial Hospital, Fuzhou, Fujian Province, 350001, China
c Department of Pathology, Fujian Provincial Hospital, Fuzhou, Fujian Province, 350001, China
d Department of Radiology, Fujian Provincial Hospital, Fuzhou, Fujian Province, 350001, China
⁎ Corresponding author. Department of Gynecology, Fujian Provincial Hospital, 134 East Street, Gulou District, Fuzhou, Fujian Province, 350001, China. fjslyyshenmh@fjmu.edu.cn
⁎⁎ Corresponding author. Department of Gynecology, Fujian Provincial Hospital, 134 East Street, Gulou District, Fuzhou, Fujian Province, 350001, China.
1 These authors have contributed equally to this work.

09 8 2024
30 8 2024
09 8 2024
10 16 e3607524 3 2024
19 6 2024
8 8 2024
© 2024 The Authors. Published by Elsevier Ltd.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Background

Retroperitoneal leiomyomas are rare benign smooth muscle tumours. Diagnosing these tumours is often challenging due to their unique growth site and nonspecific clinical manifestations. There are a few reports of leiomyomas with intratumoral bleeding.

Case presentation

A patient with a giant retroperitoneal leiomyoma presented with multiple-organ dysfunction syndrome accompanied by a progressive decrease in haemoglobin. Computed tomography (CT) revealed two cystic tumours in the abdominal cavity. The patient was underwent transabdominal retroperitoneal tumour resection. During surgery, we found two retroperitoneal tumours—one contained approximately 9000 mL of dark red fluid and the other contained 1000 mL of light brown fluid. She has recovered well without any complications.

Conclusions

There have been only a few reports of retroperitoneal leiomyomas with intratumoral bleeding. This case highlights the importance of recognizing intratumoral haemorrhage in patients with large leiomyomas and a progressive decrease in haemoglobin after ruling out external haemorrhage. If necessary, dynamic monitoring via CT may help clarify the diagnosis.

Keywords

Retroperitoneal leiomyomas
Haemorrhage
Multiple organ failure
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pmc1 Introduction

Leiomyomas account for a majority of benign tumours in women, with a cumulative incidence of 70–80 % in patients at the age of 45 years [1]. Most leiomyomas are located in the uterus, while extrauterine leiomyomas are relatively rare, especially those located behind the peritoneum [2,3]. The diagnosis of retroperitoneal leiomyomas is often challenging due to their unique growth site. In addition, leiomyomas may undergo several types of degeneration, such as hyaline degeneration, cystic degeneration, red degeneration, calcification, or sarcomatous degeneration. However, few studies have reported on leiomyomas with intratumoral bleeding. This study aimed to report a case of retroperitoneal leiomyoma accompanied by intratumoral bleeding with multiple organ dysfunction syndrome as the initial manifestation (see Table 1).Table 1 Summarized timeline of the case.

Table 1Past medical history	● a history of hypertension

	
Symptom	● abdominal distension since 6 days ago

● chest distress, asthma since 3 days ago

● anuria since 1 day ago

	
Diagnosis	● Computed tomography (CT): Two cystic tumours in the abdominal cavity, with compression of the ureter leading to double renal pelvis effusion and enlargement and double lung infection

● Echocardiogram: Atrial fibrillation and left ventricular diastolic dysfunction

● Blood gas analysis: Type II respiratory failure

● Laboratory tests: white blood cells, neutrophils, serum pre-B-type natriuretic peptide, high-sensitivity troponin, and creatinine levels were elevated, accompanied by a decrease in hemoglobin and a normal coagulation function

	
Treatment	● correction of heart failure, assisted ventilation, anti-infection therapy, diuresis, and bilateral ureteral stent placement

● transabdominal retroperitoneal tumor resection

	
Follow-up	● She has recovered well without any complications

	

2 Case presentation

A 69-year-old woman presented with a 6-day history of abdominal distension, chest pain, a 3-day history of asthma and a 1-day history of anuria. She had a history of hypertension and no history of surgery. She was gravida 2, para 2 and had been menopausal for 20 years. Her family members did not have a similar medical history. Physical examination revealed a normal pulse (88 times/minute) and blood pressure (105/70 mmHg) and a body mass index of 37.6 kg/m2, with orthopnea. Computed tomography (CT) revealed two cystic tumours in the abdominal cavity (Fig. 1A–B) in addition to compression of the ureter leading to double renal pelvis effusion and enlargement and double lung infection. Her echocardiogram revealed atrial fibrillation and left ventricular diastolic dysfunction. Blood gas analysis suggested type II respiratory failure. According to laboratory tests, her white blood cell count (21.0 × 10^9/L; normal value [3.5–9.5] × 10^9/L), neutrophil count (87.0 %; normal value 40.0–75.0 %), serum pre-B-type natriuretic peptide level (3127 pg/mL; normal value < 900 pg/mL), high-sensitivity troponin level (607 ng/L; normal value < 45 ng/L), and creatinine level (250 μmol/L; normal value < 81 μmol/L) were elevated, but her haemoglobin level (99-77 g/L; normal value ≥ 115 g/L) was decreased and her coagulation function was normal. After she was admitted to the intensive care unit, she underwent heart failure treatment, assisted ventilation, anti-infection therapy, diuresis, and bilateral ureteral stent placement. After multidisciplinary discussions, transabdominal retroperitoneal tumour resection was performed. During surgery, we found that a large retroperitoneal tumour contained approximately 9000 mL of dark red fluid (Fig. 1C), while another retroperitoneal tumour, which was connected to the left sacral ligament, contained approximately 1000 mL of light brown fluid. The surgery went smoothly, with 600 mL of intraoperative bleeding. After the surgery, she received a blood transfusion, anti-infection therapy, thrombus prevention treatment, and nutritional support. After a few days, the patient's heart and kidney functions, as well as her lung condition, returned to normal. Postoperative histopathological analysis confirmed that the larger tumour was a leiomyoma with localized bleeding (Fig. 1D–E), and the other tumour connected to the sacral ligament was a leiomyoma, which was followed by the development of a multifocal infarction (desmin (+++) and SMA (+++) (Fig. 1F–H).Fig. 1 Retroperitoneal leiomyoma accompanied by intratumoral bleeding. (A) Computed tomography revealed two cystic tumours in the abdominal cavity, the larger of which were approximately 28.9 × 9.5 × 28.9 cm3 in size. The red arrow indicates two cystic masses, while the blue arrow indicates a normal uterus. (B) Computed tomography revealed signs of bleeding in the larger tumor (indicated by the red arrows). (C) Gross image of the larger tumor. (D and E) Histopathologic examination confirmed a leiomyoma with localized bleeding (red arrow). (G) Immunohistochemical staining for Desmin (+++). (H) Immunohistochemical staining revealed SMA (+++).

Fig. 1

3 Discussion

The clinical manifestations of retroperitoneal smooth muscle tumours include a wide range of symptoms, primarily due to the pressure exerted on adjacent organs. Diagnosing these tumours based only on clinical manifestations is challenging due to their lack of specificity [[4], [5], [6]]. Imaging plays a crucial role in the diagnosis of retroperitoneal smooth muscle tumours. This approach not only helps in understanding the anatomical relationship between tumours and neighbouring organs but also aids in identifying any degeneration of tumours and associated complications [2,5,7,8]. A previous study revealed significant differences in the clinical manifestations of retroperitoneal leiomyomas, including abdominal distension, urinary system symptoms, weight loss, and pelvic pain [9]. Approximately 90 % of patients may present with large pelvic masses [10]. In this case, the patient initially presented with multiorgan failure, including heart and kidney failure, along with pulmonary infection, which is a rare occurrence. This may be attributed to giant retroperitoneal smooth muscle tumours causing diaphragmatic elevation and increased intrathoracic pressure, leading to bilateral lung infection; at the same time, tumour compression leads to ureteral obstruction and secondary renal failure. Additionally, the patient exhibited a progressive decrease in haemoglobin despite the absence of trauma, vaginal bleeding, and intra-abdominal haemorrhage, thus intratumoural haemorrhage was strongly suspected by CT imaging before surgery and subsequently confirmed by surgery. According to published case reports, the incidence of uterine fibroids with intratumoral bleeding but no intra-abdominal bleeding is approximately 4 % (5/125); however, there have been few reports of retroperitoneal leiomyomas with intratumoral bleeding [11]. In this case, the patient experienced subacute intratumoral haemorrhage, which led to cardiac and renal failure accompanied by pulmonary infection within a few days, with a relatively insidious clinical presentation. After actively improving the condition of the heart, lungs, and kidneys, surgery was performed to remove the tumours. After the tumours were resected, the patient's intrathoracic pressure returned to normal, ureteral compression was relieved, and with active anti-infection and supportive treatments, the patient's heart function, kidney function, and lung condition quickly recovered after surgery. Therefore, we believe that her case of multiple-organ failure may have been related to the presence of retroperitoneal leiomyomas with intratumoral bleeding.

4 Conclusion

This article presents a rare case of a patient who initially presented with multiple-organ failure and was diagnosed with a giant retroperitoneal leiomyoma complicated by subacute intratumoral haemorrhage. This highlights the importance of recognizing intratumoral haemorrhage in patients with large leiomyomas and a progressive decrease in haemoglobin after ruling out external haemorrhage. If necessary, dynamic monitoring via CT may help clarify the diagnosis.

Ethics statement

This study was reviewed and approved by the Institutional Review Board of Fujian Provincial Hospital with the approval number: 2023-031-01, dated April 26th, 2023. The patient provided informed consent for the publication of their anonymized case details and images.

Data availability statement

Data associated with this study has not been deposited into a publicly available repository and will be available upon reasonable request after the approval of the corresponding author （Minghong Shen, email: ）fjslyyshenmh@fjmu.edu.cn）.

Funding

None.

Conﬂicts of interest

None reported.

CRediT authorship contribution statement

Shishi Wang: Writing – review & editing, Writing – original draft. Lihua Li: Writing – original draft. Ying Lin: Resources. Jiawei Su: Resources. Minghong Shen: Writing – review & editing, Writing – original draft, Supervision. Tan Lin: Supervision.

Declaration of competing interest

We declare that we have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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