Management of a large uterine fibroid arising from the cervical stump following subtotal hysterectomy causing massive abdominal distension. A rare case report and review of the current literature.

A 45 year-old female, P3L3, presented to our clinic with complaints of abdominal distention. The symptoms started gradually over the course of 1 year and were associated with abdominal pain and bloating, scanty whitish vaginal discharge with a foul smell, heartburn, palpitations, dizziness, generalized body weakness, and bilaterally lower limb swelling. The patient also reported symptoms of anorexia, nausea, vomiting, weight loss and early satiety as well as pressure symptoms such as incomplete rectal emptying, urinary frequency and urgency. Otherwise, she has normal bowel motion and normal urine output. She is known to be hypertensive and has been on regular medications for more than 5 years, but otherwise has a negative personal and family history of ovarian, uterine, bowel, and breast cancers. She had a history of sub-total hysterectomy in 2017, but the reason was unclear. On examination, she appeared unwell with a pale complexion. The patient was afebrile, with a BP of 153/101 mmHg, a PR of 86 bpm, a respiratory rate of 20 bpm, an SP02: 99 % in room air, and a RBG of 6 mmol/L. She weighed 80 kg and was 160 cm in height with an abdominal circumference of 78 cm at the level of the umbilicus. The patient had bilateral lower limb pitting edema and significant abdominal distention, revealing a large mass corresponding with a 30 weeks gravid uterus. The mass was mobile, smooth, and firm in consistency. On vaginal examination, the cervix appeared normal. Both fornices were full with an anterior mass compressing the bladder. There was minimal bleeding with a whitish scanty-foul smelling discharge. On chest examination there was bilaterally decreased air entry. On abdominal examination, there is an extended sub-umbilical incision scar as well as a firm non-tender mass at the level of the umbilicus corresponding to a fundal height of 22/40 cm. Kidneys were ballotable bilaterally. On digital rectal examination, the mass compresses the rectum with free rectal mucosa. The examination of other systems was unremarkable. The leading differential diagnoses at this time included retroperitoneal tumors, ovarian tumors, and uterine tumors. Laboratory workup results highlighted a hemoglobin level of 11.2 g/dL. Serum creatinine was elevated at 274umol/L and glomerular filtration rate was reduced at 17 mL/min. Other basic laboratory parameters, including blood glucose, electrolytes, liver enzymes, and other renal function tests showed no abnormalities. ECG, ECHO and chest x-rays were also all normal. Serum protein was low at 41 g/L (ref range 66–87) and serum albumin was low at 24 g/L (ref range 35–52). Tumor markers CA 125 and CEA were within normal range measuring 19 U/mL (ref range 0.000–35.000) and 2.358 ng/mL (ref range 0.00–5.093) respectively. βHCG and alpha-fetoprotein were within normal limits. Abdominal ultrasound showed a large vascular heterogenous mass with multiple lobulations seen in the pelvic cavity measuring 17x13cm. The uterus could not be identified and neither ovaries were identified. It was observed that the lesion had a mass effect on the intestine, bladder, and both kidneys resulting in bilateral severely dilated renal calyxes with cortical thinning and dilated ureters. MRI showed a well-circumscribed, bilobular-shaped mass inseparable from the posterior urinary bladder wall, measuring 18 × 18 × 14 cm. It displaces and compresses the urinary bladder anteriorly and superiorly. The tumor has an additional mass effect bilaterally on the ureters. The right ureter is displacing laterally and the mass displaces the left ureter pushing the ureter superiorly with subsequent bilateral severe hydronephrosis. The findings from the radiology review were consistent with uterine leiomyoma ( Fig. 1 ). Fig. 1 Pelvic MRI demonstrating a well-circumscribed bi-lobular-shaped mass which is adherent to the posterior urinary bladder wall and measures 18 × 18 × 14 cm. Fig. 1 Pelvic MRI demonstrating a well-circumscribed bi-lobular-shaped mass which is adherent to the posterior urinary bladder wall and measures 18 × 18 × 14 cm. The patient was taken for explorative laparotomy, under general anesthesia, with a left lateral tilt to the operating table. The abdomen was opened in layers with an extended umbilical midline incision. A large vascularized mass was seen to adhere to the posterior bladder wall and the anterior vagina and measured approximately 22 × 17 × 11 cm ( Fig. 2 ). The left ureter was seen crossing through the mass, and the right ureter was normal. Superior mobilization of mass was performed, and anterior mobilizations were performed with the left ureter transected to allow for successful anterior mobilization. The mass was mobilized posteriorly with precaution to spare the rectum and was removed completely with a part of the anterior vaginal wall and the cervix. On inspection no lymph node enlargement was seen. Non-refluxing ureteric implantation was done at the left ureter at the posterior lateral bladder wall. The bladder was then closed in layers. The vaginal vault was closed, and hemostasis was achieved. Abdominal drainage was kept in situ, and the abdomen was washed and closed in layers. The postoperative recovery was uneventful. Histopathological examination showed a capsulated tumor consisting of interlacing fascicles of bland spindle cells without atypia. Small blood vessels were seen. Mitosis and necrosis is not seen. This morphology was consistent with leiomyoma ( Fig. 3 ). Fig. 2 (A and B): Demonstrates a large vascularized mass was seen to adhere to the posterior bladder wall and the anterior vagina measuring approximately 22 × 17 × 11 cm. The mass was completely removed during intraoperative. Fig. 2 Fig. 3 Histopathology of leiomyoma demonstrating a diffuse spindle cell tumor comprised of well defined borders. The cells are normocellular, forming intersecting monotonous fascicles with indistinct borders, eosinophilic cytoplasms, cigar shaped nuclei (with tapered ends), and small nucleoli. Neither mitoses nor necrosis are seen; H&E staining at 40× original magnifications. Fig. 3 (A and B): Demonstrates a large vascularized mass was seen to adhere to the posterior bladder wall and the anterior vagina measuring approximately 22 × 17 × 11 cm. The mass was completely removed during intraoperative. Histopathology of leiomyoma demonstrating a diffuse spindle cell tumor comprised of well defined borders. The cells are normocellular, forming intersecting monotonous fascicles with indistinct borders, eosinophilic cytoplasms, cigar shaped nuclei (with tapered ends), and small nucleoli. Neither mitoses nor necrosis are seen; H&E staining at 40× original magnifications. The patient developed hypotension following the decompression of the mass and she was managed with head tilt and IV fluids. She had received 2 units of blood, 1 unit of fresh frozen plasma, and 3.5 L of Ringer's lactate alternated with normal saline intraoperatively. She received intravenous antibiotics and was put on analgesia. She was discharged on the 15th postoperative day and returned to the clinic after 2 weeks for follow-up with the gynecologist and urologist. She would be followed up every 3 months to monitor her progress. To date, regular follow-up evaluation has shown no evidence of tumor recurrence, with negative radiologic findings, including abdominal ultrasound, chest x-ray, and abdominal pelvic MRI. Additionally her elevated serum creatinine and bilateral lower limb edema have resolved after follow up.

All authors made substantial contributions to this work. John Lugata: Conceptualization, study design, prepared initial manuscript version and approved the final manuscript draft. Baraka Shao: Involved in the patient management, provided supervision and reviewed, and approved the final manuscript draft. Laetitia Makower: Conceptualization, study design, prepared initial manuscript version and approved the final manuscript draft. Ashley Rapheal: Conceptualization, study design, prepared initial manuscript version and approved the final manuscript draft. Alex Mremi: Conceptualization, and also performed histopathological analysis and prepared the final manuscript draft. Bariki Mchome: A lead Obstetrician and Gynaecologist, provided expertise throughout the entire process and revised and approved the final draft.

Written informed consent was obtained from the patient to publish this case report and accompanying images. On request, a copy of the written consent is available for review by the Editor in-Chief of this journal.

This case report was approved by the authors' institution review board committee. The patient's relative provided written informed consent to allow for her de-identified medical information to be used in this publication. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

This work did not receive any fund from any source.

Dr. John Lugata Department of Obstetrics and Gynecology, Kilimanjaro Christian Medical Centre Box 3010, Moshi, Tanzania. Email: [email protected] Tel: +255-767-647-775 ORCID: 0009-0006-9221-0452

This case highlights the importance of long term follow up for patients post hysterectomy as well as contributing to the existing literature and providing insight to the health care providers in handling similar cases. Patients who are affected should receive appropriate counseling on post operative care, recurrence after treatment and the value of early and regular follow up. Given the rarity of the case, more in depth research is required to evaluate whether supra cervical hysterectomies should be avoided in such patients and to discuss the use of better surgical procedures and interventions.

A rare incidence of cervical leiomyoma in a patient who sought care in Northern Tanzania is described in this case report. The initial clinical impression at presentation was a retroperitoneal, ovarian, or uterine tumor, which would be consistent with the clinical history and examination findings. We were able to conclusively establish the diagnosis of cervical leiomyoma following radiological and surgical procedures. In this case report, we discuss the condition's rarity, related reports in the literature, and the numerous difficulties in encountering a patient with cervical leiomyomas. With a prevalence of 20–40 % beyond the age of 35, uterine leiomyomas, also known as fibroids or myomas, are among the most prevalent benign smooth muscle tumors in women [ 11 ]. Among these cervical leiomyomas account for less than 1 % [ 3 ]. Based on the location, they are classified into two categories, intracervical or extracervical. Whilst the majority of the patients with fibroids have no symptoms, about 30 % will display significant symptoms which require treatment. These symptoms can include abnormal uterine bleeding (AUB), pelvic pressure and pain, lower back pain, anemia, constipation, increased frequency of micturition, infertility, and poor obstetric outcomes [ 4 ]. Typically cervical fibroids do not present differently to uterine fibroids. However, due to their large size they can manifest as uterine inversion, incarcerated procidentia or cervical malignancy. Non-modifiable risk factors for leiomyomas involve older age, black race, early age of menarche, nulliparity, and positive family history. Modifiable risk factors include obesity, abnormal levels of vitamin D, high levels of vitamin E, tobacco consumption, and alcohol use. [ 12 , 13 ] The pre-operative diagnosis is crucial in guiding physicians for an appropriate treatment plan. A thorough clinical history and abdominopelvic examination are necessary to inform diagnosis. Additionally the abdominal examination should include palpation for the mass and fundus of the uterus [ 7 , 8 ].On bimanual examination, an enlarged but mobile uterus with an irregular contour can be noted, whilst a fixed enlarged uterus is more suggestive of malignancy or an inflammatory process [ 7 , 8 ]. From a radiological perspective a transvaginal ultrasonography is the initial investigation of choice for the the majority of patients. However, saline infusion sonohysterography, which involves injecting sterile saline into the uterine cavity during transvaginal ultrasonography is more effective in identifying intracavitary lesions [ 14 ]. MRI is an expensive modality in a low resource setting and thus not typically used first line. However, it can be more reliable in assessing for fibroid viability, mapping, and as well as any pathologies existing adjacent, such as adenomyosis, which is a crucial factor when evaluating patients who would benefit from conservative surgical management [ 15 ]. There are various treatment options available for patients with leiomyomas, which can range from medical to surgical based on personal treatment objectives and necessities for interventions. Asymptomatic patients can be managed conservatively with serial monitoring examination and scans. Symptomatic patients might require medical treatment such as oral gonadotropin-releasing hormone antagonist together with estradiol and norethindrone [ 16 ] which reduces the complaints of heavy menstrual bleeding by creating a state of earlier follicular phase [ 15 , 17 ]. Other treatment options include non-hormonal therapies such as increasing vitamin D levels with the therapeutic aim of preventing fibroid growth without impairing ovarian function [ 18 ]. Non steroidal anti-inflammatory medicines (10 % to 52 % reduction in bleeding), and oral tranexamic acid (Lysteda; 26 % to 54 % reduction) are other non-hormonal options, with the tranexamic acid having the added advantage of being safe for use throughout the patient's attempts to conceive; both are only administered whilst the patient is bleeding [ 19 ]. Different surgical techniques can be used in keeping with patient preference for minimally invasive procedures such as myomectomy or uterine artery embolization. The need for a more definitive approach such as hysterectomy as a surgical treatment of cervical leiomyomas can be difficult, requiring a great deal of experience and skill on the part of the surgeon. In fact, the existence of a cervical leiomyoma has been found to be an independent factor impacting the length of time needed for minimally invasive surgery. There are multiple risks involved in the surgery relating to the position of the cervical leiomyomas and their proximity to the pelvic organs such as the bladder, rectum, and ureters, which can cause restrictions in surgical access [ 20 ] as well as chances of injuries to those organs. Numerous techniques were developed to lower the risk of bleeding as it is well-known that intraoperative hemorrhage is a serious concern, particularly during a myomectomy. These techniques include the use of a preoperative GnRH agonist, the tourniquet method, intraoperative injection of vasopressin into the myometrium, and permanent occlusion of the uterine artery [ 21 ]. The patient in this case underwent an exploratory laparotomy. After supracervical hysterectomy, the remnant cervix has the potential for leiomyoma formation. Surgeons performing supracervical hysterectomy should be aware of this possible outcome.

Uterine leiomyomas (fibroids) are the most common pelvic neoplasms among females, resulting in severe symptoms and reduced quality of life for women around the world [ 1 , 2 ]. The incidence of leiomyoma is 20 %–40 % in the reproductive age group, and only 1–2 % are found in the cervix [ 3 ]. After a subtotal hysterectomy procedure, cervical leiomyomas can occur as a rare and delayed complication. These fibroids are unusually found as mesenchymal tumors comprising 0.5–2 % of all uterine leiomyomas. They predominantly present with lower abdominal pain, vaginal bleeding, distension, as well as obstructive and pressure symptoms such as urinary retention and constipation [ 4 ]. Although cervical fibroids are uncommon, their development in the remaining cervical tissue post-hysterectomy highlights the importance of long-term follow-up and awareness among healthcare providers. The exact mechanism of the development of cervical stump fibroids in women who have undergone a subtotal hysterectomy is not well documented; however, these fibroids can arise due to ongoing hormonal stimulation and residual smooth muscle activity in the cervical tissue [ 5 , 6 ]. Diagnosis typically involves a combination of pelvic examination and imaging modalities such as ultrasound, CT scan, and MRI, which help differentiate cervical fibroids from other pelvic pathologies and guide appropriate management [ 7 , 8 , 10 ]. Treatment options range from conservative management with medical therapies to surgical interventions such as myomectomy or completion hysterectomy, especially in cases where symptoms are severe or unresponsive to medical treatment [ 9 ]. Herein, we present a rare case of a 45 year old female in Northern Tanzania who developed a uterine fibroid arising from a cervical stump 7 years after a subtotal hysterectomy. The fibroid was causing significant abdominal distension at presentation. The pertinent literature is discussed. This work has been reported in line with the SCARE criteria [ 22 ].

1. Name of the registry: N/A 2. Unique identifying number or registration ID: N/A 3. Hyperlink to your specific registration (must be publicly accessible and will be checked): N/A Name of the registry: N/A Unique identifying number or registration ID: N/A Hyperlink to your specific registration (must be publicly accessible and will be checked): N/A

All authors have declared that no competing interests exist.