Journal of Medical Sciences and Health

Introduction

Majority of ovarian cystic lesions are benign and represent a clinically significant subset, among which mature cystic teratomas account for approximately 10-20% of all ovarian tumors[1]. Ovarian autoamputation, a rare complication primarily resulting from adnexal torsion, occurs in an estimated 0.01% to 0.5% of ovarian torsion cases, with mature cystic teratomas being the cyst type most frequently involved[2, 3]. Due to the nonspecific clinical presentation and imaging limitations, ovarian autoamputation is usually diagnosed incidentally during surgery. Delayed diagnosis and management of ovarian torsion may result in irreversible ovarian damage and rare complications such as autoamputation[4].

Case Presentation

A 34-year-old multiparous woman (P2L2), with a history of two prior lower segment cesarean deliveries, 

presented with a three-day history of progressively worsening pain in the right iliac and suprapubic regions. The pain was initially dull in nature, gradually increased in intensity, was non-radiating in nature, and did not respond to analgesics. Ultrasonography and computed tomography abdomen and pelvis performed at an outside facility (done 8 months ago) revealed a right ovarian dermoid cyst measuring 4.5 × 3.3 cm, for which laparoscopic dermoid cyst excision was advised. Patient had history of ovarian cystectomy done on the same side 14 years back, further details were not available. 

On admission, the patient was hemodynamically stable, except for tachycardia (pulse 104 beats/min). General and systemic examinations were unremarkable. Abdominal examination revealed localized tenderness in the right iliac fossa and suprapubic region along with guarding and rigidity. Per speculum examination was unremarkable. On bimanual examination, a tender, firm solid cystic mass measuring approximately 5 × 5 cm was palpated in the right adnexa, restricted mobility and above the uterus. 

An urgent pelvic ultrasonography demonstrated a well-defined heterogeneous isoechoic lesion measuring 5.4 × 3.7 cm in the right adnexa, with absence of intralesional vascularity [Fig. 1]. The right ovary was not visualized separately, and mild free fluid was noted in the pouch of Douglas. Given persistent acute pain and imaging findings, ovarian torsion was strongly suspected. All routine hematological, biochemical investigations  

along with tumor markers were done [Table. 1].

Emergency exploratory laparotomy revealed a solid, well-defined, unruptured mass measuring approximately 4 × 4 cm was found lying free in the pouch of Douglas, retrieved free of adhesions [Fig. 2]. The base of the mass appeared necrotic and blackened, suggestive of an autoamputated dermoid cyst. The right ovary was absent, consistent with chronic torsion and autoamputation, and the right fallopian tube terminated abruptly, possibly consistent with intermittent torsion and chronic ischemia [Fig. 3]. Opportunistic left salpingectomy was performed. The excised mass was removed intact along with left fallopian tube sent for histopathological examination. The postoperative course was uneventful, and the patient was discharged in stable condition on postoperative day 4. On follow up at 3 months, the patient is symptom-free. Histopathological examination confirmed the presence of a dermoid cyst with surrounding severe necrosis (consistent with torsion) and no evidence of malignancy. The timeline of events has been briefly tabulated in [Table. 2].

Discussion

This case highlights the consequences of delay in intervention for a diagnosed case of ovarian torsion, particularly in the context of mature cystic teratomas. 

Autoamputation of the ovarian cyst is defined as cystic masses that are free from tubo-ovarian pedicle and freely mobile within the peritoneal cavity. It is a rare phenomenon and Kennedy et al. were the first to report it[5]. This clinical condition represents the end stage of prolonged or unrecognized adnexal torsion[6].

It is most often identified incidentally during surgical exploration rather than being diagnosed preoperatively. Among the various ovarian pathologies associated with this phenomenon, mature cystic teratoma is the most reported lesion[7]. This is attributed to the high prevalence of dermoid cysts among benign ovarian tumors, slow growing nature and their increased tendency to undergo torsion due to their size, weight, and heterogeneous contents.

The pathogenesis of ovarian autoamputation begins with adnexal torsion, leading initially to venous and lymphatic obstruction followed by arterial compromise. In cases of chronic or intermittent torsion, the necrotic adnexa may gradually detach from its pedicle and become autoamputated. The detached mass may remain free within the peritoneal cavity or adhere to adjacent structures such as the omentum or bowel, occasionally developing a secondary blood supply and behaving as a parasitic mass[8]. This chronic evolution explains the absence of acute symptoms in many patients and contributes to delayed or missed diagnosis.

Preoperative diagnosis of an autoamputated ovarian cyst remains challenging. Ultrasonography may demonstrate a free-floating, partially calcified cystic mass with a fluid debris level, a feature commonly described in autoamputated ovarian cysts[8]. In our case, it demonstrated the absence of blood flow to the mass raising the suspicion of ovarian torsion. The mass was not freely mobile unlike the usual expected radiological finding. However, Doppler ultrasound findings may be misleading, as vascular proliferation within surrounding fibrotic or inflammatory tissue can create a false appearance of vascularity. Consequently, the presence of Doppler flow does not reliably exclude torsion, particularly in cases of chronic or intermittent torsion, contributing to diagnostic delay and progression to autoamputation. In our case, the diagnosis of dermoid cyst was made 8 months back (outside facility) and was advised surgery, but patient did not follow up despite intermittent abdominal pain episodes.

Management depends on the timing of diagnosis and clinical presentation. Once autoamputation has occurred, surgical excision of the detached mass is the treatment of choice. Removal is recommended to prevent potential complications such as infection, adhesion formation, intestinal obstruction, or, rarely, malignant transformation[9]. Histopathological examination is essential to confirm the diagnosis and exclude malignancy. 

Laparoscopy is the preferred approach when feasible due to its minimally invasive nature and superior visualization, although laparotomy may be required in cases of large masses or extensive adhesions. In the current study, the patient opted to undergo laparotomy and denied minimally invasive procedure. [Table. 3] mentions the recent case reports highlighting similar presentations with varied symptomatology and lines of management.

Conclusion

In conclusion, mature cystic teratoma is the ovarian cyst most associated with autoamputation, primarily due to its high propensity for torsion. Ovarian autoamputation represents a late complication of chronic or missed torsion and is rarely diagnosed preoperatively. Preoperative diagnosis of an auto-amputated ovarian cyst is difficult due to nonspecific clinical and imaging signs. Ultrasound may suggest suspicion if a free-floating cyst or absent ovary is seen, while MRI can better define the lesion. Without a clear diagnosis before surgery, minimally invasive planning is limited, often requiring open surgery. 

Patient Consent:

Written informed consent was obtained from the patient for publication of this case report.