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| CASE REPORT |
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| Year : 2018 | Volume
: 7
| Issue : 11 | Page : 35-37 |
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Uterocutaneous fistula: A rare clinical entity
Richard Aniediabasi Offiong1, Nathaniel David Adewole2, Maryam Muhammad Zakari1, Daniel Owoicho Okochi1
1 Deprtment of Obstetrics and Gynecology, University of Abuja Teaching Hospital, Gwagwalada, Nigeria
2 Department of Obstetrics and Gynecology, University of Abuja Teaching Hospital, Gwagwalada; Department of Obstetrics and Gynecology, College of Medical Sciences, University of Abuja, Abuja, Nigeria
| Date of Web Publication | 3-Jul-2018 |
Correspondence Address:
Richard Aniediabasi Offiong
Department of Obstetrics and Gynecology, University of Abuja Teaching Hospital, Gwagwalada, Abuja
Nigeria
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/nnjcr.nnjcr_24_17
Uterocutaneous fistula that was present in a 36-year-old para 1+0 alive is reported. The presenting complaint was of simultaneous retrograde cyclical effluent of menstrual blood through an abdominal sinus that developed after cesarean section. Diagnosis was confirmed by hysterosalpingogram. Surgical excision of the fistula was carried out successfully.
Keywords: Cesarean section, fistulectomy, uterocutaneous fistula
How to cite this article:
Offiong RA, Adewole ND, Zakari MM, Okochi DO. Uterocutaneous fistula: A rare clinical entity. N Niger J Clin Res 2018;7:35-7 |
| Introduction | |  |
A fistula is an abnormal communication between two epithelial surfaces.[1],[2] This communication may be between two hollow organs or between a hollow organ and the surface epithelium. They are usually lined by granulation tissue but can become epithelized and commonly are associated with distressing symptomatology.[3],[4] Gynecologists are quite familiar with urinary and fecal fistulae some of which involve the uterus (uterovesical, uterocolonic, and ureterouterine)[1] and less common, compared to vesicovaginal, ureterovaginal, and rectovaginal fistulae.
Uterocutaneous fistula although not exceptional in character is a much rarer clinical entity and does give rise to the alarming and embarrassing retrograde effluent of menstrual blood concomitantly with normal menstruation.[4],[5] Approximately 120 cases have been reported over the past 200 years, 25 cases in the past 50 years, and 15 cases in the past 20 years.[6],[7] We found only a few cases in the literature reported from Nigeria.[8],[9],[10]
Usually, uterocutaneous fistula results from postpartum or postoperative complications, but other recognized risk factors include trauma, pelvic abscess, intrauterine device-associated actinomycosis, abdominal drains, pelvic malignancies, endometriosis, and chronic granulomatous infections such as tuberculosis and schistosomiasis.[1],[9]
| Case Report | | |
Mrs. A. F was a 36-year-old para 1+0, alive, whose last childbirth was 11 months before presentation. She presented to our gynecological clinic with a 7-month history of cyclical bleeding from an opening midway between her pubis and umbilicus. She had first noticed a persistently painful nodule at the same point 2 weeks after she had an elective cesarean section through a Pfannenstiel incision. An incision was made on the indurated skin to “let out blood” at a general hospital and wound dressing instituted, but healing failed to occur after several weeks. About 4 months after her delivery, she resumed her menstrual period and noticed simultaneous cyclical effluent of blood from the wound on her anterior abdominal wall. It was for this reason she was referred to our facility.
Her pregnancy was reported in a private clinic and the antenatal period was uneventful. An elective cesarean section was done at the same clinic with prolonged pregnancy as the indication. Details of the surgery could not be ascertained. She was, however, discharged on the 6th postoperative day with no history of postoperative sepsis, wound breakdown, or any other complication.
Her past medical and surgical histories were not contributory. She attained menarche at 14 years and had menstruated normally for 5 days in regular 30-day cycles until her first pregnancy. She was married in a monogamous setting and her husband had been very supportive.
On physical examination, she was a healthy-looking young woman. She was not febrile, pale, or jaundiced.
The cardiovascular and respiratory systems were normal. The abdomen revealed a healed Pfannenstiel scar and a 0.5 mm sinus midway between the umbilicus and the pubis discharging bloody effluent. There was no organomegaly, and no abdominopelvic mass was palpable. Pelvic examination confirmed menstrual effluent from the cervical os, the uterus was bulky, both uterine adnexae were free, and there was no cervical motion tenderness.
Our working diagnosis was uterocutaneous fistula with a differential diagnosis of an endometriotic nodule. Pelvic ultrasound scan was suggestive and hysterosalpingography more specifically confirmed the clinical suspicion of uterocutaneous fistula [Figure 1]. Consent for surgery was obtained after appropriately counseling the patient, and preoperative workup included her hematocrit, blood counts, blood group urinalysis, and screening for the human immunodeficiency virus and hepatitis; all of which were within limits of normal. | Figure 1: Hysterosalpingography showing contrast-filled fistulous tract at the cervicouterine junction
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At surgery done under regional anesthesia, the fistulous tract was cannulated with a size 6 pediatric feeding tube [Figure 2], which provided a useful guide as the 6 cm fistula [Figure 3] was being dissected. Moderate adhesions around the fistulous tract which involved the dome of the bladder were divided and excised. Around the point of entry to the uterus, there was an area of granulation tissue which was dissected out leaving a defect that was subsequently repaired in layers, using polyglactin (vicryl) sutures [Figure 4]. Postoperative antibiotics and analgesia were administered. She was discharged from hospital on the 5th postoperative day.
At her second follow-up visit, 6-week postsurgery, she had no complaints. She was having her first menstrual period postoperatively which was confined to the vagina. The midline subumbilical skin incision had healed. There was no sinus on the abdomen discharging blood. Histology of the excised fistulous tract was unremarkable. She was counseled on the safe mode of delivery in her future pregnancy and also advised on contraception for at least 1-year postoperative.
| Discussion | | |
This case is being reported to heighten the levels of suspicion among clinicians when confronted with abdominal or operation wound sinuses that develop following uterine surgery. Uterocutaneous fistula, even though rare, should be considered in the differential diagnosis. In the index case, which followed a cesarean section, an abdominal sinus developed and was worrisome to the patient as it failed to heal. Following the period of lactational amenorrhea, she noticed blood issuing from the abdominal opening when her menstrual period resumed.
While other reported cases presented between 6 months and 3 years after the initial surgical procedure, our case presented to hospital barely 2 months following cesarean section. However, it took a further 9 months for an accurate diagnosis to be made. It was also observed that in our case the resultant fistula opened unto the intact skin at a point remote from the initial surgical scar unlike many of the other cases reported.[1],[8],[9],[10] Improper closure of the uterine and anterior abdominal wall incisions may be responsible for this unusual site of presentation. Infection has been implicated in its etiology due to disruption in the continuity of tissue planes. However, the postoperative period in the index case appeared devoid of such complication.
The clinical diagnosis of uterocutaneous fistula was made based on the presence of abdominal effluent of blood occurring simultaneously with the normal menstruation. This was confirmed through the performance of a hysterosalpingogram. Other reports have observed that successful demonstration of the fistulous tract by this procedure is dependent on its diameter.[8],[10] Other diagnostic modalities include a fistulography, hysteroscopy, contrast-guided tomography (computed tomography), or magnetic contrast imaging (magnetic resonance imaging).[1],[2],[7]
Obliteration of the fistulous tract by surgical method is the standard of care. The extent of surgery can range from simple fistulectomy to hysterectomy depending on the fertility desires of the patient. Fistulectomy was carried out on our patient to preserve fertility. She had a definite resolution of her problem and returned to work free of the embarrassment of being stained unusually with menstrual blood.
| Conclusion | | |
Uterocutaneous fistula is rare and early diagnosis could be elusive. Suspicion must, therefore, be heightened in women who complain of an abdominal or abdominal wound sinus, especially following surgical procedures on the uterus.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Maddah G, Fattahi AS, Rahnama A, Jamshidi ST. Uterocutaneous fistula following cesarean section: Successful management of a case. Iran J Med Sci 2016;41:157-60.  |
| 2. | Thubert T, Denoiseux C, Faivre E, Naveau A, Trichot C, Deffieux X, et al. Combined conservative surgical and medical treatment of a uterocutaneous fistula. J Minim Invasive Gynecol 2012;19:244-7. |
| 3. | Dragoumis K, Mikos T, Zafrakas M, Assimakopoulos E, Stamatopoulos P, Bontis J, et al. Endometriotic uterocutaneous fistula after cesarean section. A case report. Gynecol Obstet Invest 2004;57:90-2. |
| 4. | Yadav R, Rami PR, Jayalakshmi D. Utero-cutaneous fistula. J Obstet Gynecol India 2006;56:81-2. |
| 5. | |
| 6. | Shukla D, Pandey S, Pandey LK, Shukla VK. Repair of uterocutaneous fistula. Obstet Gynecol 2006;108:732-3. |
| 7. | Vellanki VS, Goginemi S, Jahnavi KS. Case report of utero-cutaneous fistula. J Womens Health Care 2015;4:231. |
| 8. | Taingson MC, Adze JA, Bature SB, Durosilorun AM, Caleb M, Amina A. Utero-cutaneous fistula following Caesarean section. Niger J Surg Res 2016;17:58-60. [Full text] |
| 9. | Okoro O, Onwere S. Retained products of conception in a utero cutaneous fistula: A case report. Niger J Clin Pract 2008;11:170-1. [ PUBMED] |
| 10. | Abasiattai AM, Ibanga GH, Akpan A, Ume KU. Post caesarean section utero-cutaneous fistula: A case report. J Womens Health Issues 2014;3:5. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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