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ISSN: 2643-6760

Surgery & Case Studies: Open Access Journal

Case Report(ISSN: 2643-6760)

A Unique Challenge: Laparoscopic Management of an Adenomyotic Cyst Volume 4 - Issue 4

Mohammad Jaffal1, Ruchi Bhandari1, Pratima Poddar1, Shashi-Lata Tripathi1, Anastasia Salame2 and Rekha Bhandari1*

  • 1Obstetrics and Gynecology Department - Division of Minimally Invasive Surgery Bhandari hospital and Research Center, Jaipur, India
  • 2Fakih Fertility Center - Al Ain City, Abu Dhabi, United Arab Emirates

Received: February 05, 2020;   Published: February 21, 2020

Corresponding author: Rekha Bhandari, Bhandari hospital and Research Center, Jaipur, 138-A, Vasundhra Colony, Gopalpura Bypass, Tonk Road, Jaipur - 302018, India

DOI: 10.32474/SCSOAJ.2020.04.000192

Abstract PDF

Abstract

Introduction: dysmenorrhea is a common gynecological complaint, which can be primary or secondary.

Case: 19-year-old patient with severe secondary dysmenorrhea, operated for an adenomyotic cyst.

Discussion: rare causes of secondary dysmenorrhea include mullerian malformations and adenomyotic cysts. The diagnosis and management is a challenge. Laparoscopic approach in tackling those pathologies is an acceptable treatment modality.

Conclusion: Laparoscopy is a safe and adequate, diagnostic and therapeutic method in the management of adenomyotic cysts.

Dysmenorrhea is a common gynecological complaint, which can be primary or secondary. Diagnosis and management of rare cases of dysmenorrhea is a challenge for clinicians. We present a case of a 19 year old patient with severe secondary dysmenorrhea, operated for an adenomyotic cyst with a laparoscopic approach. Rare causes of secondary dysmenorrhea include mullerian malformations and adenomyotic cysts. Laparoscopic approach in tackling those pathologies is an acceptable treatment modality. In conclusion laparoscopy is a safe and adequate, diagnostic and therapeutic method in the management of adenomyotic cysts.

Keywords: Dysmenorrhea, adenomyotic cyst, laparoscopy, hysteroscopy

Introduction

The most common gynecologic complaint faced by clinicians is dysmenorrhea. Characterized by painful cramps during menses, dysmenorrhea affects reproductive age women of all races. Dysemorrhea is divided into primary dysmenorrhea where a pelvic pathology is absent and secondary dysmenorrhea with endometriosis being the most common cause [1]. Rare causes of secondary dysmenorrhea include cavitary uterine horns and adenomyotic cysts [2,3].

Case

A 19-year-old nulliparous, not sexually active lady presented to our institution because of severe worsening dysmenorrhea and abdominal cramping. The patient had no medical or surgical history. Her first menses occurred at the age of 6 years, which was never investigated. Her menstrual cycle was regular. Dysmenorrhea began to worsen during the past few months, being insufficiently relieved by analgesics. The patient denied menorrhagia or dyschezia. Physical examination in the clinic showed a soft abdomen with no palpable masses or lymphadenopathy. Inspection of the vulva revealed normal looking introits with a patent vagina. Transabdominal ultrasound performed showed an anteverted uterus with a cystic lesion 2.4 x 2.4 centimeters (cm) near the left cornu of the uterus with hemtometra, raising suspicion for a noncommunicating cavitary uterine horn. Ovaries were of normal size and appearance.
Laboratory findings including complete blood cell count, complete metabolic panel, and urinalysis were within normal limits. Pelvic magnetic resonance imaging (MRI) showed a suspected unicornuate uterus with a noncommunicating left rudimentary horn measuring 2.5cm in diameter with blood accumulation inside (as shown in Figure 1). Patient was scheduled for laparoscopic excision of a suspected left uterine horn, aiming to alleviate the patient’s pain and eliminate the chances for future pregnancy in the horn. An informed consent was obtained from the patient. Operative laparoscopy was performed with the patient under general anesthesia. With the patient in a lithotomy position, pneumoperitoneum was induced using a 10 mm umbilical trocar with direct entry, and 3 trocars were inserted (as shown in Figure 2). At laparoscopic observation, the abdominal cavity was inspected and a normal appearing uterus with a small bulge on the left cornual side below the insertion of the round ligament anteriorly was seen with bilateral normal adnexa and few peritoneal endometriotic implants. Gentle diagnostic hysteroscopy was performed, and it disclosed a normal appearing uterine cavity with two tubal ostia, thus excluding any uterine malformation.

Figure 1: Port position during the surgery.

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Figure 2: MRI images with a left sided uterine cystic lesion misinterpreted as a rudimentary horn.

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The MRI images were reviewed intraoperatively. And an adenomyotic cyst was suspected the situation, and the area of the cyst was palpated with graspers and it showed a softer texture compared to the fundus. Thus, the decision was to excise this area. Diluted vasopressin was injected at the surgical site to decrease bleeding. Then with the help of harmonic scalpel the overlying serosa and myomectium were removed. After reaching the cyst wall it was punctured and it revealed chocolate colored blood. A corkscrew was inserted into the lesion for traction and myometrial fibers dissected similarly to a myomectomy with the exception of an absent clear plane, until the whole lesion was removed measuring about 4cm in diameter. The endometrial cavity was not entered. Gentle coagulation of the bed was done and the defect was closed in 3 layers using a barbed suture, to obliterate any dead space thus enhancing proper healing. The specimen was removed with an endoscopic bag through dilated lateral port incision. Hemostasis was secured.
Operative time was 90 minutes, and blood loss was 50mL. The postoperative course was uneventful, and the patient was discharged 24 hours after the intervention. At macroscopic examination, the lesion measured 4 cm in diameter. with final pathology showing an adenomyotic lesion with areas of internal bleeding diagnostic of an adenomyotic cyst.

Video 1:

Discussion

Pain is a very distressing feeling that might affect a patient’s life. Menstrual pain or dysmenorrhea affects a wide range of women worldwide with a prevalence of 16.8% to 81% [1]. Social and academic withdrawal as a consequence of this condition places a major responsibility on physicians to diagnose and treat this condition. Various causes of secondary dysmenorrhea have been described in the literature, yet there are few rare causes that necessitate the attentiveness of the treating physician, which might include rudimentary horns and adenomyotic cysts [2,3]. Uterine cystic lesions are rare entities that are usually benign and can be divided into congenital or acquired. Noncommunicating rudimentary horn with a unicornuate uterus is one of the causes of congenital uterine cystic lesions. While adenomyotic cysts are considered among acquired lesions with a differential diagnosis which also includes cystic degeneration of uterine myomas and serosal cysts [4]. The rarity of those conditions is a major cause for misinterpretation or misdiagnosis. The diagnosis is best established by magnetic resonance imaging (MRI) [3,5], read by an experienced radiologist. When ultrasound is performed it is best done in the second half of the cycle, when the endometrium is thick and echogenic [3].
Partial development of the mullerian duct during the embryogenesis results in a rudimentary horn and unicornuate uterus. There is no communication between the uterine cavity and the cavity of the rudimentary horn in most of the cases, and the two bodies are only connected with fibrous tissue bands. Non cavitated rudimentary uterine horns are usually asymptomatic and thus can be observed with conservative management. On the other hand, cavitated uterine horns result in hematometra or blood accumulation within the horn, causing cyclic abdominal pain [3]. Moreover, it also increases the predisposion to endometriosis with 25% of cases [6]. In addition to that, uterine rupture following a horn pregnancy as a result of transperitoneal migration of a fertilized ovum or spermatozoa is a rare yet life threatening condition [7]. Given all this data, surgical excision of a cavitated uterine horn is recommended after diagnosis [3].
The adenomyotic cyst is a rare category of adenomyosis, a benign condition characterized by the invasion of ectopic tissue from the basal layer of the endometrium into the myometrium, the pathophysiology of which is not clear, with theories including endomyometrial invagination, estrogen stimulation of mullerian ectopic cells, or iatrogenic implantation during uterine surgery [8]. Adenomyotic cysts are rarely larger than 1 cm [4] and might cause pain due to distention, internal bleeding, and rarely rupture [2,3]. Despite the lack of a uniform definition and classification, some authors have classified these lesions into 2 categories: the adult and the juvenile forms. Trauma at the endometrium-myometrium interface during instrumentation is the main proposed cause of the adult type, which involves mainly the posterior uterine wall. While the juvenile form develops from duplication and persistence of ductal mullerian tissue close to the root of the round ligament at the time of embryogenesis and development [6].
The differential diagnosis for cystic adenomyosis is extensive, including hemorrhagic leiomyoma and subserosal cysts; which need an experienced radiologist to detect minor findings on MRI for diagnosis. In the case of degenerating leiomyoma, methemoglobin accumulates in obstructed veins at the periphery, producing a rim that is T1-hyperintense and T2- hypointense. This is distinct from the T1- and T2- hypointense rim of hemosiderin in cystic adenomyosis. On the other hand, subserosal cysts contain simple fluid and lack a hemosiderin rim [5]. At histological analysis, adenomyotic cysts are lined by endometrial epithelium with stromal red blood cells and hemosiderin-laden macrophages similar to those of the endometrium [6]. The presence of ectopic endometrial tissue on pathological studies aids the diagnosis of an adenomyotic cyst that is easily misdiagnosed as a leiomyoma due to the presence of smooth muscle cells and hyaline degeneration which is considered almost pathognomonic for a leiomyoma [9].
Surgery is the treatment of choice in symptomatic lesions. Adenomyotic cysts are more clearly delineated from the normal myometrium than uterine adenomyosis, but less than uterine myomas. Varies sizes and approaches have been reported in literature. Moussaoui et al reported a case of a large 20 cm × 12 cm adenomyotic cyst in a 35-year-old patient with multiple previous surgeries at the posterior uterine wall, which was treated with open surgery [8]. Similarly, Bhalla et al reported a surgical excision of adenomyotic cyst in a 16-year-old girl [5]. Perino et al performed a laparoscopic excision of a cyst in a 25-year-old patient with no major complications [4]. Adenomyotic cysts diagnosed during pregnancy have been reported. Lim et al described a laparoscopic drainage of an adenomyotic cyst during the first trimester, with a subsequent normal pregnancy course and no recurrence of the cyst [10].
In our case, the diagnosis of the patient’s condition was driven by the MRI report indicating a rudimentary horn. Upon surgical exploration, and after the revision of the images, the diagnosis was shifted towards an adenomyotic cyst. The excision was completed laparoscopically with no major complications. This highlights the difficulty and challenge of diagnosing rare cases of dysmenorrhea, and the importance of surgical interventions in both diagnoses and treatment of those conditions.

Conclusion

Diagnosis of adenomyotic cysts is a challenging task that faces treating physicians. The rarity of the condition that can mimic other disorders including mullerian malformations, in addition to the absence of clear clinical and radiological criteria makes the diagnosis a difficult one. Management of symptomatic adenomyotic cysts is usually surgical, including both laparoscopic and open surgery.

Disclosure Statement

The authors declare that they have no conflicts of interest and nothing to disclose.

References

  1. Osayande Amimi S, Suarna Mehulic (2014) "Diagnosis and initial management of dysmenorrhea." Am Fam Physician 89(5): 341-346.
  2. Dilbaz, Berna (2014) Adenomyotic cyst of the uterus associated with pelvic pain: A case report. J Clin Exp Vol 5: 1.
  3. Camran Nezhat, Farr Nezhat (2014) Ceana Nezhat. Nezhat’s Operative Gynecologic Laparoscopy and Hysteroscopy Third Edition.
  4. Cucinella Gaspare (2013) "Adenomyotic cyst in a 25-year-old woman: case report." Journal of minimally invasive gynecology 20(6): 894-898.
  5. Ho, Mai-Lan (2008) "Adenomyotic cyst of the uterus in an adolescent." Pediatric radiology 38(1): 1239-1242.
  6. Fedele Luigi (2005) "Laparoscopic removal of the cavitated noncommunicating rudimentary uterine horn: surgical aspects in 10 cases." Fertility and sterility 83(2): 432-436.
  7. Heinonen Pentti K (1997) "Unicornuate uterus and rudimentary horn." Fertility and sterility 68(2): 224-230.
  8. El Mehdi E, Mounir M, Abdellah B, Jaouad K, Driss M (2018) Large Adenomyotic Cyst in A 35-Year-Old Woman. European Journal of Pharmaceutical and Medical Research - epmjr 5(9): 150-151.
  9. Tamai Ken (2005) "MR imaging findings of adenomyosis: correlation with histopathologic features and diagnostic pitfalls." Radiographics 25(1): 21-40.
  10. Raimi ZH (2016) "Huge Adenomyotic Cyst in Pregnancy." Journal of Surgical Academia 6(2): 40-42.
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