Authors: Wael Ferjaoui, Nabil Haloui, Ahmed Omry, Rania Dallagi, Amal Kamtaoui, Med Bachir Khalifa
Categories: Case Report, Case report, Immunohistochemistry, Leiomyoma, Soft tissue, Surgery
Source: International Journal of Surgery Case Reports
Authors: Wael Ferjaoui, Nabil Haloui, Ahmed Omry, Rania Dallagi, Amal Kamtaoui, Med Bachir Khalifa
Leiomyomas, commonly known as fibroids, are benign soft tissue tumors mostly found in the uterus. Although predominantly uterine, they can occur rarely in other areas, notably in the gastrointestinal tract. This article emphasizes an unusual an ileal leiomyoma in a 54-year-old patient, showcasing the distinct diagnostic and management challenges it presents.
A 54-year-old female with no prior medical history presented with a progressively enlarging, painful abdominal mass over six months. Imaging suggested a Gastrointestinal Stromal Tumor (GIST), but guided biopsy yielded an unexpected diagnosis of ileal leiomyoma. Subsequent surgical intervention successfully removed the tumor, leading to an uneventful postoperative recovery.
This case underscores the diagnostic intricacies and clinical manifestations of ileal leiomyomas. While diagnosis can be challenging, timely histopathological evaluation is crucial. Surgical resection guided by pathological findings played a pivotal role in this patient's care, emphasizing the need for a comprehensive approach to diagnosis and treatment in unique medical cases.
The rarity of ileal leiomyomas presents a substantial challenge in the medical field, where expertise can be limited. This case emphasizes the pivotal role of histopathological analysis in confirming the diagnosis and guiding effective surgical management.
Leiomyomas, commonly referred to as fibroids, are benign tumors that can manifest in any soft tissue site [1,2]. Gastrointestinal leiomyomas are infrequent, typically occurring in the esophagus, stomach, and colon [1,3]. Of these, duodenal leiomyomas are the least common, followed by those found in the ileum [1]. Diagnosing leiomyomas can be particularly challenging, given its rarity [4]. Herein, we discuss a rare case of ileal leiomyoma in a 54-year-old female with an abdominal mass, highlighting the unique and significant aspects of this condition. The rarity of this condition presents a challenge, as it hinders any single surgeon from gaining comprehensive expertise. Our analysis provides valuable insights into its clinical presentation, diagnosis, and management, contributing to a better understanding of atypical medical cases. This work has been reported in line with the SCARE 2023 criteria [5].
Case
This is a 54-year-old patient with no prior medical or surgical history who presented to our surgery clinic with the onset of an abdominal swelling that has been developing for the past 6 months. This mass is hypogastric, is painful, and has been gradually increasing in size. The interview did not reveal nausea, vomiting, alterations in bowel habits or traumatism.
Physical examination revealed a body mass index of 23 kg/m^2^, a heart rate of 70 beats per minute, and a blood pressure of 120/75 mmHg. The respiratory rate was within the normal range. In the abdominal examination, a tender, firm, mobile, globular mass measuring 25 × 10 cm was identified in the lower abdomen (hypogastrium), and there were no observable changes in the overlying skin. There was no evidence of lymph node enlargement.
Laboratory data showed no abnormalities. Abdominopelvic computed tomography (CT) revealed a uniform oval-shaped soft tissue mass (with a Hounsfield Unit range of 50–80) originating from the terminal ileum. The mass was 25 × 15 × 10 cm in size with specks of fat within and without calcification. It extended forward and made contact with the rear edge of the right rectus abdominis muscle. The radiological characteristics were suggestive of an ileal GIST (Fig. 1).Fig. 1Axial (A) and sagittal (B) computed tomography sections highlighting the intraperitoneal tumor mass.Fig. 1
A biopsy guided by a CT scan was performed on the lesion. The results of the histopathological examination concluded it was an ileal leiomyoma. Due to the considerable size of the tumor and its adhesions to the abdominal wall, the patient underwent a midline surgical procedure. Intraoperative exploration revealed a sizable ileal tumor mass measuring approximately 30 × 10 cm, adhering to the anterior abdominal wall (Fig. 2). It was located on the anti-mesenteric border of the small intestine, approximately 80 cm from the ileocecal valve and 3 m and 50 cm from the duodenojejunal angle. The rest of the intraoperative exploration did not show any abnormalities. A resection of the small intestine was performed, removing the tumor with a margin of 20 cm on each side of the lesion, and a mechanical side-to-side ileo-ileal anastomosis (Fig. 3). The postoperative recovery was uneventful. The patient was discharged on the 5th day post-surgery.Fig. 2Intraoperative image showing the cystic and tissue-dependent tumor mass originating from a loop of the small intestine.Fig. 2Fig. 3Image of the operative specimen of the tumor mass, weighing 2 kg.Fig. 3
The examination of the resected bowel revealed a hyalinized mass measuring 30 × 20 × 15 cm with irregular and dystrophic calcification. Spindle cells were observed in certain outer areas, and these cells stained positively for actin and desmin, confirming that the lesion was a submucosal leiomyoma. The surgical margins were sound.
By this stage, she could comfortably consume a regular diet and her bowel function was normal. The patient's progress remained positive, and she didn't experience any more episodes of his previous abdominal pain during his one-month follow-up appointment.
Gastrointestinal tract leiomyomas, though uncommon, represent the most prevalent type of benign nonepithelial tumors found in the small intestine [4,6]. Virchow first described these tumors in 1854 [4]. This clinical case presents a compelling scenario, shedding light on a rare cause of a substantial abdominal mass and the intricate challenge of distinguishing between Gastrointestinal Stromal Tumors (GISTs) and leiomyomas without resorting to immunohistochemical analysis [1]. The clinical manifestation depends on the size and location of the lesion [1,7]. Approximately 44–50% of leiomyomas exhibit symptoms such as intestinal obstruction, gastrointestinal bleeding, intussusception or abdominal mass (Table 1) [1,8]. Conversely, smaller lesions are often serendipitously discovered during surgical procedures, radiological examinations, or endoscopic investigations [1]. Diagnosis usually in the fifth decade of life such as our patient [1,6].Table 1Similar reported ileal leiomyoma cases.Table 1AuthorYearAgeSexClinical presentationSizeDiagnosisOGUZHAN SUNAMAK et al.200672FObstruction5 cmPathological specimenLevent Akman et al. (2 cases)201554FAbdominal mass10 cmPathological specimen69F20 cmPathological specimenVuković M et al.202037MMelaena4 cmPathological specimenMichael G et al.202275MObstruction1,4 cmBiopsySyaza Zainudin et al.202030MAbdominal mass10 cmPathological specimenStephanie N. Williams et al.202216MObstruction20 cmPathological specimenAnya Laibangyang et al.202163FPeritonitis17 cmPathological specimenKevin Z Zhou201850FObstruction1,8 cmPathological specimen
Tumors originating from smooth muscle within the gastrointestinal tract can be categorized as either leiomyoma or leiomyosarcoma, based on criteria such as atypical cell characteristics and the mitotic index [1]. Among these, leiomyomas are the most prevalent benign tumors in terms of symptomatic presentation [1].
The diagnostic evaluation involves examining the gastrointestinal tract using endoscopy [9]. Nevertheless, due to the extra-luminal nature of 63% of these tumors, conventional endoscopy may encounter challenges in their assessment [1]. CT scans have demonstrated an 89.5% success rate in identifying leiomyomas, which typically appear as uniform, low-attenuation masses with a growth pattern that is primarily located within the lumen of the affected structure [1,6].
Magnetic resonance imaging enteroclysis and enterography are increasingly being embraced as the primary imaging methods for assessing small bowel diseases, given their superior soft tissue contrast and the capability to acquire both morphological and functional information [1,6]. Distinguishing Gastrointestinal Stromal Tumors (GISTs) from leiomyomas through radiological means is challenging due to overlapping characteristics and their shared anatomical origin [4]. In our clinical case, endoscopic exploration and magnetic resonance imaging were not performed due to the positive histopathological results of the guided biopsy.
The course of treatment should be determined based on the pathological diagnosis, but in practice, obtaining this diagnosis before surgery can be challenging [4]. When the tumor's histology is uncertain, it is advisable to perform an intraoperative pathological diagnosis using a frozen section [1]. Nonetheless, any suspected pathology in the colon or terminal ileum should prompt a surgical resection with consideration for oncological outcomes [4]. Conclusive diagnosis can only be confirmed through histopathological assessment, and the histological features of leiomyomas often resemble those of GISTs [1]. Immunohistochemical (IHC) staining for c-kit (CD 117) expression and other antigenic markers (CD34, SMA, Desmin 2, S 100, Ki 67) can also exhibit varying degrees of positivity [1,4]. The peculiarity of this is case the preoperative histopathological diagnosis. The treatment is surgical and involves the resection of the affected segment with clear margins [6,7].
Differentiating between benign and malignant leiomyomas based solely on imaging can pose challenges [4]. A conclusive diagnosis typically requires histopathological examination and immunohistochemical analysis [6]. Leiomyomas often exhibit similar characteristics to gastrointestinal stromal tumors (GISTs). Immunohistochemical staining for c-kit (CD 117) expression and other antigenic markers (CD34, SMA, Desmin 2, S 100, Ki 67) may also show varying levels of positivity [4,6].
This clinical case not only highlights the rarity of the presented scenario but also underscores the intricate diagnostic and treatment considerations that clinicians face in differentiating and managing leiomyomas within the gastrointestinal tract. The multifaceted approach involving radiological, histopathological, and immunohistochemical analyses is crucial for achieving a comprehensive understanding and ensuring optimal patient outcomes [1].
In conclusion, this case report underscores the diagnostic complexities associated with ileal leiomyomas [6]. The rarity of this condition and its mimicry of GISTs on imaging emphasize the importance of histopathological analysis for definitive diagnosis [1]. Surgical resection, guided by the histopathological findings, proved successful in this case. These insights shed light on the clinical and management challenges posed by leiomyomas, emphasizing the significance of a multidisciplinary approach to effectively address such unique medical cases [1].
Written informed consent was obtained from the patient for the publication of this case report and its accompanying images. A copy of the written consent is available for the Editor-in-Chief of this journal to review upon request.
The author (s) declared that no grants were involved in supporting this work.
AI tools were not used for the elaboration of the manuscript.
Ethical approval is not applicable/waived at our institution.
This research did not receive funding from any specific grant provided by public, commercial, or not-for-profit organizations.
Wael Ferjaoui and Ahmed Omry contributed to manuscript writing and editing, and data collection; Nabil Haloui and Rania Dallagi contributed to data analysis; Amal Kamtaoui and Med Bachir Khalifa contributed to conceptualization and supervision; all authors have read and approved the final manuscript.
Dr. Ahmed Omry.
N/A.
No conflicts of interest.