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Abdominal Wall Endometriosis Suspected as Uterine Myoma and Soft Tissue Tumor: A Case Report and Literature Review

I Wayan Agus Surya Pradnyana*1, Kadek Agus Wijaya2, Ni Luh Putu Primadi3

¹Faculty of Medicine, Udayana University, Prof. dr. I.G.N.G Ngoerah General Hospital, Denpasar, Bali, Indonesia
²Fertility and Reproductive Endocrinologist, Department of Obstetrics and Gynaecology, Tabanan General Hospital, Bali, Indonesia
³Department of Pathology Anatomy, Tabanan General Hospital, Bali, Indonesia

*Corresponding author

Wayan Agus Surya Pradnyana, 1Faculty of Medicine, Udayana University, Prof. dr. I.G.N.G Ngoerah General Hospital, Denpasar, Bali, Indonesia

Abstract

Background: Endometriosis refers to the presence of ectopic endometrial tissue. Extra pelvic endometriosis can manifest as distinct mass known as abdominal wall endometrioma (AWE). Diagnosis can be challenging due to nonspecific and variable nature of clinical symptoms. Awareness of this entity can help make early diagnosis and treatment.

Clinical Presentation: 29-year-old woman presented to obstetrics and gynecology clinic with lower abdominal pain for weeks and palpable mass, intense pain during menstruation for last two months. Ultrasonography showed mass with suspicion of uterine myoma and supported by multi-slice CT-Scan of the abdomen. The patient underwent a laparoscopic myomectomy, but the uterus appeared to be normal. Consultation with general surgery during the operation suspected as soft tissue tumor. During surgery, mass was found to have infiltrated the abdominal wall muscle, which later identified as AWE by histopathological examination. The patient's condition improved postoperatively, and pathology results showed no malignancy.

Discussion: Scar endometriosis is an entirely iatrogenic disease with an incidence of 0.03–1% in women with history of caesarean births. There are situations when surgery can be avoided, especially in cases of endometriomas or recurrent endometriomas that can be medically managed by hormonal therapy or minimally invasive procedures. Surgical intervention becomes imperative for patients that report perpetual pain, structures that are possibly malignant, infertility, or gradual increases in size.

Conclusion: The gold standard treatment in such cases is surgical, consisting of the excision of mass within safe margins. This also allows the specimen to be histopathologically analyzed to confirm the diagnosis and excluding malignancy.

Keywords: Abdominal wall, Endometriosis, Cesarean sca

Introduction

Abdominal wall endometriosis (AWE) is defined as the presence of endometriotic infiltration in any segment or depth of the abdominal wall.1 In most cases, endometriosis is located within the pelvis. Although, ectopic endometrial tissue can also be found outside the pelvis, affecting different organs with a variety of symptoms. The intestine, urinary tract (kidney, ureter, bladder), lymph nodes, and abdominal wall are the main sites of extrapelvic endometriosis.2 Spontaneous abdominal wall endometriosis usually occurs in the abdomen without scars and accounts for about 20% of all AWE cases.3 On the other hand, secondary AWE develops in surgical scars from obstetric or gynecological operations and is more common. The etiology of endometriosis is complex and multifactorial, with theories explaining its pathogenesis have not been fully confirmed. However, the etiology of secondary AWE is probably due to direct implantation of endometrial cells into the soft tissues of the abdominal wall, through an iatrogenic process during abdominal-pelvic surgery.4–6

The incidence of AWE is estimated at 0.03 to 3.5% of all endometriosis cases, affecting approximately 6 to 10% of women of childbearing age worldwide.1,7,8  Cases of AWE are considered rare globally, with most cases reported in women who have undergone a Caesarean section, with AWE appearing on the surgical scar reaching 57 to 92%.1,4,9 However, this disease is often undiagnosed. The average age at diagnosis is 33.2 to 35 years,10,11 and the time span between previous surgery and diagnosis of secondary AWE can vary from three months to two decades.8 The prevalence of this case in Indonesia is still not available.

The typical clinical triad includes an abdominal wall mass or nodule at the site of a previous scar, cyclic pain associated with menstruation, and a history of previous abdominal surgery.12 Depending on the layer of the abdominal wall involved, skin changes (ecchymosis or hyperpigmentation), swelling, and bruising at the surgical site associated with the menstrual cycle may be observed.2 Although it is considered as a benign condition, endometriosis is a highly debilitating disease exacerbated by its association with dysmenorrhea, dyspareunia, menstrual irregularities, and infertility, with significant detrimental impact on social, occupational, and psychological functioning.13,14

Ultrasonography (USG), computed tomography (CT) scan, and/or magnetic resonance imaging (MRI) of the pelvis and abdomen, including abdominal wall examination, play a critical role in the diagnosis.1,12 Wide surgical excision is the only curative therapy for AWE.1,9 Preoperative evaluation of any invasive therapy is essential since the involvement of a general surgeon with all its logistical implications is required, especially in the case of gross muscle invasion or deep invasion into the omentum or bowel. The final and definitive diagnosis can be decided by histopathological analysis of the nodule.2

This case report aims to present an investigation of the clinical course and management of secondary AWE, including diagnostic steps and surgical management.

Case Presentation

A 29-year-old woman came to the Obstetrics and Gynecology Department of Tabanan Regional General Hospital (RSUD) with complaints of lower abdominal pain since a week ago. The patient has a history of menarche at the age of 10 with a regular cycle of 28 days, a menstrual period of 3 days, and a volume of 2 to 3 changes of pads per day, in which the patient feels pain every time she menstruates. The patient's marital history is that she was legally married. The patient's history of pregnancy, childbirth, and postpartum, giving birth to and breastfeeding a normal term boy by caesarean section (CS) 5 years prior, with a birth weight of 3400 grams, and delivery assisted by health workers. The patient has a history of using an intrauterine contraceptive device (IUD) for one year. Family history of disease is denied. History of drug and food allergies is denied. There was no history of other diseases and gynecological history. There were no complaints of urination and defecation, the patient did not report any difficulty sleeping.

On physical examination, the patient was found to have a general condition of pain in the symphysis, compos mentis consciousness with Glasgow Coma Scale (GCS) 15, and vital signs: blood pressure 94/70 mmHg, pulse 80 times/minute, respiration 18 times/minute, temperature 36.2°C. Body weight 67 kg and height 157 cm, body mass index (BMI) 27.2 kg/m2. Normal conjunctiva, white sclera, symmetrical mammary glands without discharge, adequate cleanliness. The nipples appear protruding without discharge. Examination of the legs was found to be symmetrical, without edema, with positive physiological reflexes. Obstetric examination of the abdominal region showed a surgical scar. There was no discharge on anogenital examination, no abnormalities on vaginal speculum. Based on the results of anamnesis and physical examination, the patient had a work-up diagnosis of abdominal pain et causa suspected adenomyosis and was given management by a collaboration of obstetrician and gynecologist, planned radiological examination of abdominal computed tomography (CT) scan with contrast, laboratory examination (complete blood count, random blood sugar, blood urea nitrogen (BUN), and serum creatinine/SC), and consultation with a cardiologist. AP chest X-ray examination showed no cardiomegaly and no active lung process. The results of the ultrasound examination (USG) showed a solid round mass with ill-defined margin (Figure 1). The hematology laboratory examination was carried out with the results described in Table 1.

Figure 1: An Ill-defined margins, solid round lesion, hypohyper echoic mass was found during TAS on the anterior of the uterus(A-B), left and right ovarium appears to be normal (C)

Figure 2: Abdominal Multi-Slice Computerized Tomography Showing an Isodense Lesion with Firm Margins Suspected as Lower Uterine Segment Mass

According to the anamnesis, physical and supporting examination, the patient was diagnosed with abdominal pain et causa suspected adenomyosis. The patient was given IEC and then sent home. However, the patient returned to Tabanan Hospital with the same complaint. The results of the consultation with a cardiology specialist obtained a patient with Lee Cardiac Risk Index Class I with MCE 0.4% or no contraindications for pro laparoscopic myomectomy.

The results of the Multi-slice Computerized Tomography (MSCT) Abdomen examination of axial reformatted coronal and sagittal slices with and without contrast, the patient had the impression of a large uterus with a round isodense lesion with relatively clear boundaries measuring 5.23×5.79 cm which was enhanced after contrast administration from 26-35 HU to 72-85 HU, in the lower uterine segment region according to the image suggested as lower uterine segment mass. While other intra-abdominal organs have a good impression (Figure 2).

In pre-operative, the patient returned to the obstetrics and gynecology department of Tabanan Hospital with complaints of lower abdominal pain. Physical examination showed good general condition, compos mentis consciousness with GCS 15, and vital signs: blood pressure 105/80 mmHg, pulse 66 times/minute, respiration 20 times/minute. Body weight 70 kg and height 157 cm (BMI: 28.4 kg/m2). Normal sclera, symmetrical mammary without discharge, adequate cleanliness. Symmetrical legs. Obstetric examination of the abdominal region found surgical scars. There was no discharge on anogenital examination, no abnormalities on vaginal speculum. The patient was then scheduled to undergo laparoscopic adhesiolysis, subserous myomectomy, and abdominal wall repair, and undergo delegative therapy as recommended by the gynecologist. The patient was also given drug therapy in the form of prophylactic antibiotics Cefotaxime 2 grams pre-operatively, packed red blood cells (PRC) 1 cup of blood (blood type O). After the procedure, the patient was said to be able to mobilize and with no complaints.

During surgery, the patient underwent general anesthesia and was positioned in a lithotomy position. During laparoscopy, the liver appeared normal, the uterus was anteflexed, and omental adhesions with the uterus and peritoneal wall were visible (Figure 3). Furthermore, adhesiolysis was performed, myoma in the fundus with a diameter of 2 cm (Figure 4). During myomectomy, both tubes and ovaries were normal in size. A tubal patency test was performed, and bilateral tubal patency was obtained. A solid mass was felt under the abdominal skin, differentially diagnosed with a suspected soft tissue tumor. The duration of surgery was 30 minutes with a bleeding of 110 cc.

Table 1: Laboratory Result Prior to Operation 

Figure 3: Uterus and Adnexa Appears to be Normal During Laparoscopy (A), Both Right and Left Ovaries are Normal and Both Tubal Appears to be Patent (B, C)

Figure 4: An Adhesion to The Pelvic Wall 

Figure 5. Midline excision was done above the mass, (A – C) brown discharge was found during the excision suggestive of endometriosis, (D) resected mass, the specimen consists of rubbery tissue with brownies discharge

Figure 6: Haematoxylin and Eosin Stain with Magnification 10X: Endometrial glands surrounded by endometrial stroma embedded in dense abdominal fibrous tissues.

Figure 7: Haematoxylin and Eosin Stain with Magnification 40X. Endometrial cells were seen without signs of malignancy. (A) Endometrial glands and stroma in between fibrotic tissue. (B) A histological slice of the mass showing scarce endometrial glands and stroma

The surgery was continued by a general surgeon, the patient was positioned supine with GAOTT. The fascia was opened and a mass measuring 5 cm × 4 cm × 3 cm was found, with brownish fluid in the abdominal mass. Figure 5 shows the excision procedure on the mass for pathological anatomy (PA) examination. Furthermore, abdominal wall repair and surgical wound suturing were performed. The duration of the operation was 75 minutes.

On physical examination of the patient after laparoscopic adhesiolysis, subserous myomectomy day 0, excision biopsy, abdominal wall repair, and PA examination, the patient was found to be in good general condition, compos mentis with GCS 15, and vital signs of blood pressure 100/59 mmHg, pulse 78 times/minute, respiration 20 times/minute, temperature 36.5°C. Body weight 67 kg and height 157 cm (BMI 27.2 kg/m2). The patient was then observed while continuing the delegated actions of the Obstetrics and Gynecology specialist, in the form of administering Cefotaxime 1 gram every 8 hours intravenously, Tranexamic Acid 500 grams every 8 hours intravenously, Mefenamic Acid 500 mg every 8 hours orally, Paracetamol 500 mg every 8 hours orally, Antacid 1 tablet every 8 hours orally, and sending the PA results. The patient was also given Fentanyl and Ketorolac drips in D5% 20 to 30 micro drops, and Ondansetron 4 mg every 8 hours orally by an anesthesiologist.

During the follow-up on a day after surgery, the patient complained of minimal pain in the surgical wound, the patient had flatus, spontaneous urination, and the patient was able to walk. The patient appeared moderately ill, GCS 15, with vital signs within normal limits. General status examination showed that the conjunctiva of both eyes appeared anemic, the abdomen did not appear distended, bowel sounds were normal, and the surgical wound was well cared for. Meanwhile, the general status of the thorax, extremities and genitals was within normal limits. PA examination were conducted to an abdominal wall sample (1cc irregular tissue) and showed microscopic results of connective tissue stroma with foci of stroma and endometrial glands, no signs of malignancy. Figure 6 shows a histological picture of endometrial gland tissue that is more purple than the surrounding area, encircled by brighter abdominal fibrous tissue. Figure 7 shows a closer picture of the endometrial glands with no signs of malignancy.

For outpatient treatment, the patient was given Cefadroxil therapy 500 mg every 12 hours orally, Mefenamic Acid 500 mg every 8 hours orally, Vitamin B Complex 1 tablet every 12 hours orally. The patient was then given IEC before going home and was advised to consult and control as well one week later.

Discussion

Endometriosis is the presence of endometrial tissue, both glands and stroma, outside the uterus. It is commonly seen in women of reproductive age and occurs in the ovaries, fallopian tubes, and pelvic peritoneum. Extrapelvic locations including the respiratory, gastrointestinal, and urinary tracts have been previously reported.15-17 Abdominal wall endometriosis accounts for up to 3.5% and even reached 75.9% in a recent study of extrapelvic endometriosis, with most cases arising from previous abdominal surgical scars due to previous cesarean section or abdominal hysterectomy.1,18,19 Although the exact pathogenesis of abdominal wall endometriosis is unclear, various theories have been proposed. The metaplastic theory proposes that cells in the mesothelial lining of the abdominal peritoneum that have undergone differentiation and metaplasia can differentiate into endometrial cells. The direct transplantation theory states that endometrial cells are implanted directly into the surgical scar or surrounding tissue during surgery. Finally, the lymphatic and vascular metastasis theory states that endometrial cells enter the lymphatic or vascular circulation system and invade the abdominal wall.6,17,20

The most common location of extra pelvic endometriosis is the abdominal wall (4%) and is associated with previous surgical scars after abdominal gynecologic procedures such as cesarean section, hysterotomy, hysterectomy, tubaligation or myomectomy.1 The clinical presentation of AWE can vary as reported by previous studies. The most common complaint is severe cyclic pain at the site of the abdominal wall mass associated with menstruation in 57% of cases. Other symptoms include tenderness at the site of the lesion, a painful mass, variation in the size of the mass following the menstrual cycle, discomfort around the mass or lesion, skin discoloration, and bleeding. Cyclic pelvic pain and subfertility may also occur.1,21-23 In this case, a 29-year-old female patient was reported with complaints of lower abdominal pain since the past week, and felt pain every menstruation. This patient's complaints were nonspecific, there was no change in the size of the mass during the menstrual cycle, no tenderness and no change in skin color. With such broad clinical symptoms, the diagnosis of abdominal wall endometriosis before surgery is very difficult to decide.

According to Wang and Lam, the typical presentation of AWE usually includes three things, namely a history of previous cesarean section, cyclical pain localized in the lesion area related to menstruation, and a mass near the surgical scar. However, only 60% of patients showed all three symptoms. Abdominal mass (96%) and pain in the mass (87%) were the most common symptoms, while cyclical pain occurred in only 57% of patients. Patients may also complain of an increase in size, bleeding, and skin discoloration of the mass associated with menstruation.22 While Piriyev et al., found that the clinical picture of umbilical endometriosis from 22 women, including cyclic bleeding from the nodule (60%) and was observed more frequently than cyclic bleeding from nodules in other locations (17.5%), and only 9% had undergone cesarean section.24

The main risk factors for AWE were a history of previous cesarean section (1.8%), followed by other abdominal and pelvic surgical procedures, either by laparoscopy or laparotomy.1 Benedetto et al., reported eight patients (9.6%) who experienced secondary AWE after laparoscopic surgery. Six of them developed umbilical scar endometriosis (75%), one developed from an implant at the site of secondary trocar placement in the left iliac fossa (12.5%), and one developed from an implant in the Pfannenstiel incision performed for bowel extraction during segmental bowel resection (12.5%).5 Abdominal wall endometriosis commonly affects women in the reproductive age group. In previous literature, the average age of individuals diagnosed with AWE ranged from 33.2 to 35 years. Most AWE cases can be diagnosed around 5 to 8.6 years, later than other types of endometriosis.9,10 Benedetto et al., showed the time span between previous surgery and the diagnosis of secondary AWE reached 5.2 years.5 Increased body weight (BMI ≥25 kg/m2) seems to be associated with the occurrence of AWE. Several studies have shown a predominance of increased body mass in patients diagnosed with AWE (25.5 to 29.2 kg/m2) and justify this association due to technical difficulties in operating on obese patients, possibly related to inadequate hysterorrhaphy.5,11 While in this case report, AWE was diagnosed 7 years after cesarean section and when the patient was 29 years old with a BMI of 27.2 kg/m2.

Diagnosis is made preoperatively through anamnesis, physical examination, and supporting examinations. Supporting examinations include non-pathognomonic radiographic techniques, such as USG, CT scan and MRI. Normally, the first-line radiographic modality is abdominal USG which will show a round/oval mass or lesion with ill-defined borders found at the scar site. The lesion usually appears as a heterogeneous hypoechoic area surrounded by a hyperechoic ring, with low blood flow (containing internal vascularization) or even absent when viewed in Doppler mode.2,25,26 Computed tomography scan with contrast can provide a better picture of the location, size, characteristics, and whether the muscle or subcutaneous layer are involved, while MRI is more suitable for smaller lesions and can also help identify associated blood vessels; Both of these modalities can be considered if the USG findings are inconclusive.1,19,27 The USG examination results in this case showed uterine myoma, but were reconfirmed by MSCT examination of the abdomen with axial reformatted coronal and sagittal slices with and without contrast, with findings of a large uterine impression with a relatively well-defined isodense round lesion measuring 5.23×5.79 cm with enhancement after contrast administration from 26-35 HU to 72-85 HU, in the lower uterine segment region according to the image of a lower uterine segment mass/cervical mass.

There are several differential diagnoses that resemble abdominal wall endometriosis, including sarcoma, metastatic malignant tumors, granulomas, abscesses, sediments, incisional hernias, hematomas, desmoid fibromatosis, and lipomas. Therefore, histological examination of the tumor is needed to state the correct diagnosis and exclude malignancy.2,11 Ultrasound-guided fine needle aspiration cytology (FNAC) is a simple, non-invasive, inexpensive, and easy procedure that can help confirm the diagnosis of AWE. However, FNAC has a risk of secondary spread by placing new implants at the puncture site. Hence, histopathological evaluation of the resected mass is often performed to make an accurate diagnosis.3,28 The patient was previously diagnosed with abdominal pain et causa suspected adenomyosis after anamnesis, physical examination, and supporting examinations. However, after laparoscopic surgery, no abnormalities were found in the gynecological organs, but omentum adhesions were found with the uterus and peritoneal wall. A solid mass was felt under the abdominal skin, bringing this case into a different diagnosis of suspected soft tissue tumor, which then excision and histopathological examination were performed. The results of the examination showed an impression of the endometrium of the abdominal wall.

The primary treatment for AWE is surgical procedure, which are a complete excision of the endometriotic nodule or wide local excision to achieve a clear margin of at least 1 cm to prevent recurrence or malignant transformation.20,29 However, some other researchers recommend a margin-free excision of 5 to 10 mm to prevent recurrence. In cases of nonpalpable nodules, preoperative ultrasound-guided demarcation of the lesion can help determine the exact location of the nodule during surgery.4 The patient in this case was given surgical management of laparoscopic adhesiolysis, subserosal myomectomy, and abdominal wall repair.

Previous studies have found that malignant transformation of endometriosis is rare, with a reported incidence of 1%. If the underlying abdominal wall fascia and muscles are involved or the mass is 5 cm or larger in diameter, reconstruction of the defect will be necessary to prevent future hernia formation. Many studies have found that the reported recurrence rate is 5 to 9% and there is a higher risk of recurrence in large masses and lesions involving the rectus muscle.5,20 Similar to pelvic endometriosis, the fibrotic tissue around the nodule may contain endometrial tissue, that possibly will be the starting point of nodule recurrence. Surgical site complications (hematoma and/or seroma) are more common in nodules measuring ≥30 mm compared to nodules <30 mm (35.3% and 4.5%, respectively).5

During gynecologic/obstetric surgical procedures, there are several recommendations that can be useful to avoid endometriosis implantation in the abdominal wall. One of them is the use of wound protectors/retractors in all cesarean sections, which can also be considered in patients undergoing segmental bowel resection, as this reduces the rate of surgical site infection and avoids contamination of the abdominal wall with endometriosis cells. If the use of retractors/protectors is not possible, the abdominal wall wound should be intensively irrigated and thoroughly cleaned with saline solution. Through all laparoscopic surgeries, especially in the cases of endometriosis, the use of an endobag to remove the surgical specimen is recommended. Lastly, for procedures requiring suturing of the uterus and abdominal wall, it is important to emphasize the use of a new needle/suture to close the abdominal wall plane (different from the one used to close the uterus) in an effort to prevent iatrogenic implantation of endometriosis in the abdominal wall.5,30

Conclusions

A case of a 29-year-old woman with lower abdominal pain since a week ago, pain during menstruation, and a history of cesarean section 7 years ago has been reported. Based on the results of anamnesis, physical examination and supporting examination, the patient was diagnosed with abdominal pain et causa suspected adenomyosis, then laparoscopic adhesiolysis, subserous myomectomy, and abdominal wall repair were performed. However, after laparoscopic surgery, no abnormalities were found in the gynecological organs, but omentum adhesions were found within the uterus and peritoneal wall. Moreover, a solid mass was felt under the abdominal skin, bringing this case into a different diagnosis of suspected soft tissue tumor, which then excision and histopathological examination were performed. The results of the examination showed an impression of the endometrium of the abdominal wall. Further monitoring and evaluation are still needed to determine whether there are complications, recurrence, or even transformation of the case into malignancy.

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