CPQ Women and Child Health (2020) 3:1
Case Report

Utero-Cutaneous Fistula After Open Abdominal Myomectomy: A Case Report


Georges KOZAH*, Labib RIACHI, Paul MOARBES, Souzane ISKANDAR, Ihsan SAYOUR, Rawane EL ASSAAD, Rim KARAKI, Naameh MOUSSAOUMAY, Wouroude ELMOSTAFA, Hrant GHAZELIAN, Toni AZZI & Saad EL DAOU

Department of Obstetrics and Gynecology, Levant Hospital, Beirut, Lebanon

*Correspondence to: Dr. Georges KOZAH, Department of Obstetrics and Gynecology, Levant Hospital, Beirut, Lebanon

Copyright © 2020 Dr. Georges KOZAH, et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: 28 May 2020
Published: 04 June 2020

Keywords: Abdominal Myomectomy; Post-Operative Complication; Uterocutaneous Fistula


Abstract

Novel minimally invasive surgery technique is a new option for the treatment of this condition. We hereby, are reporting a case of a 42 years old woman who developed an uterocutaneous fistula 3 months after myomectomy and was treated by minimally invasive supracervical hysterectomy and fistulectomy

Introduction
A fistula is a communication between two epithelium-lined surfaces. It may occur after traumas, radiation, injuries, infections, and surgeries involving the abdominal or pelvic organs. The inflammatory process caused will develop an adherence between the tissues, and will chronically lead to the abnormal connection between them [1]. Most uterine fistulas are vesico-uterine, recto- uterine or cervico-vaginal. An uterocutaneous fistula (communication between the uterus and the skin) is a very rare condition, and there are only few cases reported in the existing literature. The causes include multiple surgeries, incomplete closure of the incisions, anemia and diabetes [2]. We describe a patient with this kind of rare fistulas after multiple surgeries. Eventually, she had a successful repair using for the first time a robotic assisted approach with a fistulectomy and a supracervical hysterectomy in Lebanon.

Case Presentation
A fistula is a communication between two epithelium-lined surfaces. It may occur after traumas, radiation, injuries, infections, and surgeries involving the abdominal or pelvic organs. The inflammatory process caused will develop an adherence between the tissues, and will chronically lead to the abnormal connection between them [1]. Most uterine fistulas are vesico-uterine, recto- uterine or cervico-vaginal. An uterocutaneous fistula (communication between the uterus and the skin) is a very rare condition, and there are only few cases reported in the existing literature. The causes include multiple surgeries, incomplete closure of the incisions, anemia and diabetes [2]. We describe a patient with this kind of rare fistulas after multiple surgeries. Eventually, she had a successful repair using for the first time a robotic assisted approach with a fistulectomy and a supracervical hysterectomy in Lebanon.

A third ultrasound was done showing the same findings. The patient was started on Ertepenem (Invanz). A CT scan showed a hypodense area in the left aspect of the uterine body, measuring (4.9*4.8cm), with an enhancing tract starting from the anterior aspect of the uterus coursing anteriorly through the lower anterior abdominal wall and reaching the skin. The patient had no wishes for future childbearing (she had 7 children, 6 NSVD and one C-section) and underwent a Robotic supracervical hysterectomy with a fistulectomy and an incisional repair. We noted intraoperatively, extensive adhesions of the bowel with omental tissues wrapped around the fundus of the uterus all the way extending to the anterior abdominal wall covering the fistula. A small catheter was introduced from the external opening of the fistula at the skin level, and travelled through the tract into the right corneal part of the uterus. Endometrial epithelialization of the fistulous tract was grossly visible during the procedure. This was facilitated by the 3D, high definition, and up to 10 times magnification of the robotic platform. Extensive lysis of adhesions separating the uterus from the abdominal wall, and a supracervical hysterectomy were successfully completed. The left ovary was preserved, the right one was removed during her previous C-section.


Figure 1: Dissection of the fistula from the anterior abdominal wall


Figure 2: The external opening of the fistula.


Figure 3: The catheter introduced from the external opening of the fistula reached the uterus

Discussion
Uterocutaneous fistulas are the rarest form of all fistulas, and whose pathophysiology is not fully understood. Their diagnosis and treatment may be difficult and challenging.

Blood leakage from the incision scar during menstruation is present in 65% of the cases of UCF, and malodorous discharge with a constant fever is present in 80% of the cases [3]. This kind of fistulas has various etiologies that can be categorized into 2 big sections [4]:

• Surgical Conditions: drain use, iatrogenic trauma, multiple abdominal surgeries, and incomplete closure of uterine wounds.

• Maternel Conditions : endometriosis, diabetes, anemia. Intra-abdominal sepsis or infectious causes.

Dragoumis et al [5] , reports have shown fistulas presentation after septic abortion [6], pelvic abscesses removal. Infection with actinomycosis due to intrauterine devices [7], curettage, difficult vaginal delivery, and the use of forceps. Our patient had a fistula following an open abdominal myomectomy

In a recent case series by Rezaei et al. reported a co-existence of a uterocutaneous fistula and a vesicocutaneous fistula, in a multiparous lady with two previous cesarean sections [8].

The time of presentation for this kind of fistula is highly variable. Our patient presented 3 months after her myomectomy, however in the literature this can vary from months to 6 years after the last surgery [3,4].

Treatment for that kind of fistula is usually surgical with or without a hysterectomy depending on the age of the patient and her fertility expectations. Seyhan et al [9] reported a patient treated with gonadotropin releasing hormone agonist (GnRH) alone: The GnRH agonist induces atrophic changes in the epithelium, and assists in the closure of the fistula. This treatment can only be used for smaller fistulas, a large fistula will not respond to the GnRH agonist treatment and the treatment of choice will only be a surgical excision of the fistulous tract.

Surgical excision can be achieved via an open or a laparoscopic approach. We performed the first robotic assisted fistulectomy excision reported in the literature. Thubert et al [3] used medical treatment and minimally invasive surgery (laparoscopy) for the excision of a fistula tract. They injected methylene blue through the external opening of the patient’s skin to find the tract and excise the fistula. We used a small flexible catheter that was introduced from the external opening of the fistula and we dissected all along the tract of the catheter in order to identify and excise the whole fistulous tract. Also Sonmezer et al [6], used another method by injecting the blue dye from the cervix and internal opening and successfully treated their patients.

Conclusion
This case report highlights the very rare possibility of an uterocutaneous fistula occurring in a woman following an open abdominal myomectomy, and the development of an abscess after her surgery. Successful excision of the tract using a minimal invasive surgical approach was done robotically for the first time in the literature using a small catheter inserted in the external opening of the fistula for proper and precise identification.

Bibliography

  1. Gupta, S., Shukla, V., Varma, D., et al. (1993). Uterocutaneous fistula. Postgraduate Medical Journal, 69(816), 822-823.
  2. Arteaga, J. D. R., Murillo, A. N. V. & Hernández Trejo, M. C. (2012). Utero-cutaneous fistula: a case report and literature review. Ginecologia y obstetricia de Mexico., 80(02), 95-98.
  3. Thubert, T., Denoiseux, C., Faivre, E., et al. (2012). Combined conservative surgical and medical treatment of a uterocutaneous fistula. Journal of Minimally Invasive Gynecology, 19(2), 244-247.
  4. Okoro, O. & Onwere, S. (2008). Retained products of conception in a utero cutaneous fistula: a case report. Nigerian Journal of Clinical Practice, 11(2), 170-171.
  5. Dragoumis, K., Mikos, T., Zafrakas, M., et al. (2004). Endometriotic uterocutaneous fistula after cesarean section. Gynecologic and Obstetric Investigation, 57(2), 90-92.
  6. Sönmezer, M., Şahincioğlu, Ö. & Çetinkaya, E. (2009). Uterocutaneous fistula after surgical treatment of an incomplete abortion: methylene blue test to verify the diagnosis. Archives of Gynecology and Obstetrics, 279(2), 225.
  7. Tedeschi, A., Di Mezza, G., D’Amico, O., et al. (2003). A case of pelvic actinomycosis presenting as cutaneous fistula. European Journal of Obstetrics & Gynecology and Reproductive Biology, 108(1), 103-105.
  8. Rezaei, Z., Shahraki, Z. & Shirazi, M. (2017). Utero-cutaneous Fistula as a rare complication after cesarean delivery: case series. J Obstet Gynecol Cancer Res., 2, e14071.
  9. Seyhan, A., Ata, B., Sidal, B., et al. (2008). Medical treatment of uterocutaneous fistula with gonadotropin-releasing hormone agonist administration. Obstetrics & Gynecology, 111(2), 526-528.

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