Iatrogenic cloaca: A case report

Abstract

BACKGROUND

Traumatic cloacal deformities are a result of major obstetric injury and usually happens after a fourth-degree perineal laceration. This is characterized by complete disruption of the perineal body, anterior defect of the internal and external anal sphincter, and loss of the distal rectovaginal and/or anovaginal septum. The common chamber incorporating vagina and recto anal outlet ensues as cloaca.

CASE SUMMARY

We present a case of unmarried 15-year female, who developed hematometra and was diagnosed as cervical ostia obstruction at the age of 12 year. She underwent drainage of hematometra, and later some abdominal and perineal procedures elsewhere and subsequently developed fecal discharge from the vagina. Examination revealed absence of posterior wall of vagina and anterior wall of anorectum. In addition, the anterior half of the anal sphincter was also absent. She underwent vaginoplasty, anorectoplasty, sphincteroplasty with levatorplasty with creation of rectovaginal septum.

CONCLUSION

This case report highlights expert management of a case of mismanaged benign gynaecological condition in a young female that resulted in lifelong impairment of the patient's quality of life following hysterectomy and salpingo-oophorectomy, leading to the development of a common cloaca.

Key Words: Iatrogenic cloaca; Hematocolpometra mismanagement; Reconstruction; Sphincteroplasty; Vaginoplasty; Complications; Case report

Core Tip: This rare and unusual case highlights the mismanagement of hematocolpometra in a child and its subsequent complication in the form of iatrogenic cloaca. Here the authors discuss the management of this patients in details, and review the literature on surgical management of iatrogenic cloaca.

INTRODUCTION

Iatrogenic cloacal deformities represent a severe yet rare clinical challenge. Post intervention formation of cloaca is similar as common cloacal deformity of infancy. It is demonstrated clinically as a common channel incorporating the genitourinary and recto-anal openings with loss of anovaginal septum. These deformities, often arising from obstetric injury or secondary to prior perineal surgical interventions, leading to significant anatomical disruption of sphincter complex and pelvic floor muscles as well as functional impairments. The reported incidence of traumatic cloacal deformities following obstetric injury is approximately 3 cases per 100000 vaginal deliveries[1]. They are frequently associated with fourth-degree perineal tears with loss of vaginal mucosa and wall, rectovaginal septum, perineal body, sphincter complex along with anorectal mucosa resulting in formation of a common channel. All these anatomical deformities leading to faecal functional impairment, recurrent urinary and vaginal infections. Management includes careful assessment of all the anatomical deformities by an expert followed by surgical correction by using native tissue, autologous tissue or synthetic grafts, the first one described by Sieber and Klein for congenital cloacal deformity[2].

CASE PRESENTATION

Chief complaints

A 15-year-old female patient presented with passage of mucous discharge per vagina for one year duration following drainage of colpohematometra and multiple surgeries elsewhere.

History of present illness

At 12 years of age, a female patient had complaints of lower abdomen pain and amenorrhoea. On evaluation with USG abdomen, she was found to have cervical ostia obstruction and hematometra. She then underwent perineal evacuation of blood clots. A few days later, following surgery, she started passing faeces and flatus per vagina. This was managed by proximal diversion colostomy. For persistent menstrual symptoms, she underwent multiple sessions of drainage of hematometra followed by total abdominal hysterectomy and bilateral salpingo-oopherectomy and attempted repair of perineal deformity elsewhere before being referred to us. The main presenting symptom was passage of mucous discharge per vagina with stoma in situ in left iliac fossa.

History of past illness

No significant history of chronic illness.

Personal and family history

Not contributory.

Physical examination

Perineal examination revealed absent anterior wall of anorectum and rectovaginal septum with single cavity of vagina and anal canal (common cloaca, Figure 1). The urethral opening was present anteriorly in the vaginal wall.

Figure 1 Common cloaca, absence of uterus, intact urethral opening in anterior vaginal wall. A: Sketch diagram at presentation-absence of uterus and common cloaca; B: Common Cloaca with intact urethra.

Laboratory examinations

All required laboratory investigations done were within normal limits.

Imaging examinations

Magnetic resonance imaging (MRI) pelvis revealed absence of uterus and bilateral ovaries with defect in anterior wall of anal canal and lower rectum involving half circumference of the internal and external sphincter anteriorly. Posterior wall of vagina was absent suggestive of common cloaca (Figure 2).

Figure 2  Magnetic resonance imaging pelvis.

FINAL DIAGNOSIS

Iatrogenic common cloaca.

TREATMENT

She was taken up for surgery after discussion and counselling regarding the surgical procedure with the patient and her parents. Surgical procedure involved anorectoplasty, vaginoplasty, sphincteroplasty and levatorplasty. Surgery was performed in lithotomy position in regional anaesthesia under antibiotic coverage (Injection Cefuroxime and metronidazole). Posterior wall of vagina along with anterior wall of anal canal and part of lower rectum were absent. External sphincter was absent from 9 o' clock to 3 o' clock. Ends of sphincter were retracted and thinned out. The retracted ends of the posterior wall of vagina were separated from the retracted ends of the anorectum. The anorectum was repaired with 4/0 interrupted Vicryl suture. Levator ani muscle was loosely approximated with PDS 2/0 followed by sphincteroplasty (End to End – using interrupted 1/0 proline). Posterior vaginal wall was reconstructed with 3/0 interrupted polydioxanone suture. Rectovaginal septum was created by putting Vicryl mesh between repaired posterior wall of vagina and anterior wall of anorectum. Corrugated drain was placed behind the repaired posterior wall of vagina and the mesh. Twenty-eight (No.28) French, 5 cm length of polyvinyl chloride drain was placed in the reconstructed rectum to avoid the collapse and also for rectal drainage Figure 3. Earlier created sigmoid colostomy was retained. Antibiotic was continued for 5 days.

Figure 3 Post repair appearance. PVC: polyvinyl chloride.

OUTCOME AND FOLLOW-UP

Postoperative course was uneventful. Drain was removed on POD5 and patient was discharged on POD7 without any complication. On two months follow up she is asymptomatic with well healed anorectum (Figures 4 and 5).

Figure 4 Surgical site on follow up. Appearance of wound 2 month after surgery.

Figure 5  Contrast study from sigmoid colostomy.

DISCUSSION

Iatrogenic cloacal deformities typically result from obstetric injuries. The high-risk factors reported are operative vaginal deliveries with use of forceps, primiparity, large foetal weight, and median episiotomies. Other cause of such deformity occurs during any perineal procedure for gynaecological pathology as was evident in this case[3].

Accurate diagnosis involves physical examination and imaging modalities. Most commonly used modality is MRI, anorectal ultrasound which provides valuable insights into structural abnormalities of anal sphincter complex, assisting in preoperative planning. Another novel modality used in complex cloacal malformations is rotational fluoroscopy with 3D reconstruction facilitates diagnostic accuracy of complex anatomy and preoperative surgical planning[4].

Functional assessment of anal sphincter is assessed with anorectal manometry. This is not mandatory preoperatively but helps in assessing the faecal continence functional outcome in postoperative period. Further, Clinical scoring systems like the Wexner incontinence score and quality-of-life assessments are crucial for evaluating symptom severity and tracking postoperative improvements.

Management of iatrogenic cloacal deformity is found to be more challenging compared to traumatic due to multiple attempted interventions followed by traumatic inflammatory changes leading to fibrosis of the tissues and disturbed anatomical planes. Therefore, attempt to definitive repair should be postponed to at least 3 months or when the local inflammation subsides[5]. Approaches for reconstruction can be perineal, abdominal or posterior sagittal, but the most preferred in iatrogenic deformity have been reported to be perineal approach. Pena and Hendren were the two pioneers who had reported multiple management strategies for congenital common cloaca according to the length of common cloaca and associated anomalies[6,7].

Since, there is no standard procedure described yet for management of iatrogenic cloaca, its management principles are followed similar to the congenital cloacal malformation which is based on length of common cloaca[8]. He has described posterior sagittal approach for mobilizing anterior rectal wall and posterior vaginal wall followed by vaginoplasty and anoplasty for common channel shorter than 1 cm. For common channel length between 1 and 3 cm, he added the need of total urogenital mobilisation before repair via posterior sagittal approach. With increase in length of common channel, he advised for combined posterior sagittal and transabdominal approach for adequate mobilisation. In case of inadequate tissue for vaginal repair, vaginal switch[9] or vaginal replacement with rectum, sigmoid or small bowel was suggested. Similar approach can be applied for management of iatrogenic cloaca considering the extent of injury to rectovaginal septum.

The repair process described, consists of three major steps. The first step, as described by Penna[6] for the correction of congenital cloaca, involves the dissection of the rectovaginal septum to separate the rectal and vaginal walls, along with the reinforcement of the sphincters. This step begins with the injection of adrenaline saline into the rectovaginal plane, followed by a transverse, curvilinear, or X-shaped incision on the perineal skin at the anovaginal septum[10]. The dissection plane is then deepened between the vaginal and rectal walls, extending laterally into the ischiorectal fossa and cranially up to the levator ani. Care must be taken to avoid the inadvertent creation of buttonholes in either mucosa.

The second step focuses on the identification and mobilization of the sphincter complex muscles, which include the internal and external anal sphincters and the levator ani muscle. Mobilization is performed from lateral to medial, beginning in the para-rectal space. The sphincter complex is then approximated or overlapped, with concurrent reconstruction of the rectovaginal space. This step helps obliterate the space created in the first step, thereby reducing the risk of hematoma or abscess formation. Additionally, it enhances the continence function of the sphincter.

The third and final step involves the formation of the perineal body by approximating the puborectalis and transverse perineal muscles, along with the repair of the anorectal and vaginal mucosa. This strengthens the pelvic floor and reconstructs the neovagina and anal canal. Various methods for vaginal reconstructed for congenital cloacal deformity are tabulated in Table 1, as described by Peña[6], which can be applied equally in iatrogenic cloaca also. If the puborectalis muscle is inadequate for approximation, a bulbocavernosus (Martius) or gracilis muscle flap can be used for perineal body reconstruction. Vaginal mucosal replacement may also be performed using rectal, colonic, or small bowel tissue in cases where native mucosa is insufficient. Skin closure can be achieved through simple approximation or the use of skin flaps (e.g., island or lotus petal flaps)[11,12]. Alternatively, partial wound closure may be employed to allow fluid drainage and secondary intention healing, which has been associated with a reduced risk of wound infection.

Table 1 Various methods of vaginal reconstruction[6].

1. For vaginal pull through	Total urogenital mobilisation
	Vagino-urethroplasty
2. For vaginal replacement	Rectum
	Small bowel
	Colon
3. Flaps	Vaginal flap
	Skin/Labial flap
4. Vaginal switch

The role of a Defunctioning stoma remains controversial. Proponents argue that diverting faecal matter from the suture site reduces the risk of infection, wound dehiscence and allows optimal healing of the sutured muscles by preventing stool passage through the anal canal. However, there is no evidence of benefit of routine defunctioning stoma. In a retrospective study[13], author has shown similar rates of perineal wound dehiscence in patient with and without defunctioning stoma with higher rates of stoma related complications along with need of second surgery in patients with defuncting stoma. Rather, most of the surgeons have used alternative options for avoiding perineal wound dehiscence than stoma, like partial closure of wound with healing by secondary intent. Defunctioning stoma should be selectively used in case of complex cloacal deformity with multiple attempts of repair or significant faecal contamination.

Postoperative management includes adequate pain control with analgesics, prophylactic antibiotics, and restrictions on sitting or squatting to minimize strain on the surgical site. Regular inspection of the wound is essential to detect early signs of infection or dehiscence. Patients are typically discharged after 3 to 4 days, with a follow-up scheduled within 7 to 14 days to assess suture site healing, the presence of rectovaginal fistula, and faecal incontinence. Sexual activity should be avoided for at least three months to ensure proper healing.

The surgical repair/ reconstruction of cloacal type injury is not free from complications. The early complications include urinary retention, wound infection, wound dehiscence and surgical site hematoma. The most common early complication is wound infection, which can be managed conservatively with drainage and regular dressing. Similarly, surgical site hematoma also needed to be drained followed by regular dressing.

The late surgical complications include vaginal stricture/shortening, dyspareunia, anal stricture, fecal incontinence, development of rectovaginal or rectocutaneous fistula. Some patient may land up with complete failure of the reconstruction. Patients who develop rectovaginal or cutaneous fistulas, a subset of them heal spontaneously, while others require a defunctioning stoma followed by fistula repair. Anal stricture can be managed with serial dilations. In cases where reconstruction fails, reattempting the procedure by an experienced surgeon is an option, or alternatively, a permanent diversion stoma may be considered. Overall functional outcomes of the surgery largely depend on the length of the common channel[6] and surgical expertise. The various reported series on management of iatrogenic cloaca and their outcomes have been highlighted in Table 2.

Table 2 Published case series/reports on iatrogenic cloaca: Aetiology, presentation, management, and outcome.

Ref.	Age, gender/No. of patients	Aetiology	Clinical presentation	Examination	Evaluation	Operative procedure	Follow up
Ibrahim et al[14]	45, F	Remote vaginal delivery	Postcoital bleed, faecal urgency, NO faecal incontinence	Grade 4 perineal tear, Absent perineal body, receding post vaginal wall, no anterior anal sphincter	Manometry- diminished IAS and EAS pressure, hypersensitivity of rectal sensation, RAIR+	Sphincteroplasty (overlapping), levatoroplasty, vaginoplasty, perineal body reconstruction, rectocele repair	6 months- no faecal incontinence
Balmaceda et al[1]	17, F	Vaginal delivery-7 days (high BW, forcep usage)	Anal incontinence, vaginal discharge	Hypotonic sphincter, disruption of anterior rectoanal wall and perineal body, lower middle third of rectovaginal septum, vaginal mucosa	EUS- anterior defect of IAS+EAS	Sphincteroplasty (overlapping), anoplasty, perineal body repair, vaginoplasty	Postop- perianal subcutaneous fistula- healed spontaneously. Six months- good continence
Stavros et al[15]	39, F	3 years after vaginal delivery	Common cloaca	Absent perineal body, absent resting and squeeze pressure.	-	-	-
Venkatesh et al[16]	44	Obstetric injury (39), Others (5)	Faecal incontinence, recurrent infections	Anterior defect of IAS+EAS	Manometry and imaging	Sphincteroplasty, levatorplasty, and flaps	Improved continence in 39, incontinence to flatus in 4, incontinence to liquid and flatus in 1
Haitham et al[17]	30, F	Post primary repair of genital injury due to sexual assault	Stools per vagina	Rectovaginal fistula at 2 cm from anal verge. EAS intact		Fistula repair with diversion colotomy	Asymptomatic after 1 year of stoma reversal
Spelzini et al[18]	27, F	2 year post vaginal delivery	Stool per vagina	Common cloaca		Rectovaginal septum repair, perineal body reconstruction	2 years, asymptomatic
Present case	15, F	Mismanaged calpohematometra	Stool per vagina	Common cloaca	MRI	Anorectoplasty, vaginoplasty, sphincterplasty with levaterplasty and creation of rectovaginal septum	2 months asymptomatic, waiting for colostomy closure

F: Female; IAS: Internal anal sphincter; EAS: External anal sphincter; EUS: Endoscopic ultrasonography; RAIR: Rectoanal inhibitory reflex; MRI: Magnetic resonance imaging.

In rare cases, the loss of the uterus and ovaries, as happened in our patient, have a significant impact on sexual and reproductive health, as well as psychosocial well-being. Even patients with intact reproductive organs may experience long-term issues such as irregular menstrual cycles, infertility, dyspareunia, and early menopause. This hormonal disruption may further increase the risk of cardiovascular disease, osteoporosis, and other hormone-related changes. To prevent the effect of hormonal disruption, expert opinions and follow up is advised regarding use of hormonal therapy and counselling.

CONCLUSION

Iatrogenic cloacal deformities are rare but debilitating conditions. This case highlights the mismanagement of hematocolpometra leading to iatrogenic cloaca and later attempted failed repair. It is better not to attempt repair if one is inexperienced but refer the patient to the expert centre for better outcome and quality of life. Since there is no standard protocol for its management, further research and collaboration are needed to develop a standardized protocols to improve long-term outcomes for affected patients.