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doi:10.1111/jog.12378

J. Obstet. Gynaecol. Res. Vol. 40, No. 6: 1814–1818, June 2014

Pelvic tuberculous granuloma successfully treated with laparoscopy to preserve fertility: A case report and review of the published work Tatsuo Nakahara, Akira Iwase, Masahiko Mori, Mika Kondo, Maki Goto and Fumitaka Kikkawa Department of Obstetrics and Gynecology, Nagoya University, Graduate School of Medicine, Nagoya, Japan

Abstract Tuberculous granuloma must be considered in the differential diagnosis of pelvic masses in women of reproductive age because the major sequela of pelvic tuberculosis is infertility; however, currently there is very little information about its fertility-preserving treatment. We report the case of a woman with a history of tuberculous peritonitis who referred to our hospital for evaluation of an adnexal mass and primary infertility. The patient underwent excision of pelvic tuberculous granuloma with fertility-preserving laparoscopic surgery. We resected as much of the tuberculous granuloma as possible using the laparoscopic technique without causing damage to the uterus or ovaries. In particular, we report for the first time in the published work the laparoscopic removal of tuberculous granuloma without causing damage to the uterus or ovaries. Our experience from this case suggests that laparoscopic diagnosis and treatment of tuberculous granuloma is a feasible procedure in a patient who wants to conceive. Key words: female genital tuberculosis, infertility, laparoscopic surgery, tuberculous granuloma.

Introduction Although tuberculosis has become less common since the advent of antituberculous chemotherapy and improved standards of living, the worldwide incidence of tuberculosis, particularly the extrapulmonary form, is increasing. The genital tract is one of the most common sites of involvement of extrapulmonary tuberculosis among women, accounting for 5–13% of all infections occurring outside the lungs.1 Female genital tuberculosis is often diagnosed incidentally. Female patients with genital tuberculosis typically exhibit menstrual irregularity, abnormal vaginal bleeding, infertility and/or pelvic pain.2 Tuberculosis is the cause of approximately 10% of cases of sterility in women worldwide and approximately 1% of cases of sterility in women in industrialized countries.2 As the

clinical manifestations of abdominal tuberculous granuloma are often similar to those of ovarian carcinoma or other types of pelvic masses, making a differential diagnosis is often difficult. We herein report the case of a woman of reproductive age with pelvic tuberculous granuloma who underwent laparoscopic surgery.

Case Report A 27-year-old, null gravida woman was referred to the gynecology department of our university hospital for evaluation of an adnexal mass and primary infertility. She had been admitted due to a complaint of abdominal distention 5 years previously and had been given quadruple drug therapy (isoniazid, rifampicin, ethambutol and pyrazinamide) after being diagnosed with

Received: January 17 2013. Accepted: December 10 2013. Reprint request to: Mr Tatsuo Nakahara, Department of Obstetrics and Gynecology, Nagoya University, Graduate School of Medicine, 65 Tsuruma-cho, Showa-ku, Nagoya 466-8550, Japan. Email: [email protected]

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tuberculous peritonitis. She had not experienced any gynecological complaints or bodyweight loss; however, she did develop infertility. Transvaginal ultrasonography revealed an unidentified mass measuring approximately 8 × 7 × 6 cm in diameter in the left adnexal region (Fig. 1a). Abdominopelvic computed tomography (CT) demonstrated the presence of a calcified solid mass (Fig. 1b). Magnetic resonance imaging (MRI) showed a well-defined mass with a mix of high and low signal intensities on T2-weighted images (Fig. 1c and d). On the basis of these findings and the patient’s history, degenerating uterine leiomyoma and tuberculous granuloma were considered in the differential diagnosis, and laparoscopic surgery was performed in order to verify the diagnosis and resect the mass. During laparoscopy, we found ‘violin string’ fibrinous strands extending from the surface of the liver, gallbladder, omentum and abdominal wall, which may have occurred as a result of the previous tuberculous peritonitis infection. After gently removing the

adhesions, we found a soft mass approximately 7 cm in diameter located on and firmly adhered to the posterior surface of the uterus. Fallopian tubes and fimbrias could not be identified. The mass was also adhered to the sigmoid colon, appendix and pelvic peritoneum. On macroscopic examination, the mass was found to have a yellow–gray surface (Fig. 2a and b). Rupture of the mass was inevitable in spite of gentle traction of the mass and careful separation of the severe adhesions from the surrounding tissue (Fig. 2c). The wall of the mass cavity measured approximately 5 mm in thickness, and the cavity contained thick, yellowish and foamy tubercles (Fig. 2d). We also found that both fallopian tubes and fimbrias were completely buried in the mass. After adhesiolysis around both fallopian tubes and fimbrias, we injected indigo carmine dye through the uterine manipulator; however, we could not confirm both fallopian tubes were patent. The wall and contents of the mass were removed without damaging the surrounding organs and collected via a laparoscopic bag taking care to avoid spillage of the

(a)

(b)

(c)

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Figure 1 (a) Transvaginal ultrasound showing irregular borders with speckled calcifications. (b) Computed tomography showing an adnexal mass with flecked calcifications. (c,d) T2weighted magnetic resonance imaging revealed a well-defined mass with a mix of high and low signal intensities.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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(a)

(b)

(d)

(c)

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contents into the abdominal cavity. The patient recovered without developing any postoperative complications. Histopathology demonstrated the presence of multiple granulomatous reactions formed of epithelioid cells, Langerhans giant cells and caseous necrosis, without any evidence of malignant tumors. A polymerase chain reaction test for Mycobacterium tuberculosis was positive. Ziehl–Neelsen staining and periodic acid-Schiff staining yielded negative results. After consulting with the hospital infection control team regarding the treatment course for the patient, we chose to administer no additional antibiotic therapies against tuberculosis. Hysterosalpingogram was performed at 6 months after surgery, which showed both fallopian tubes were open and the shape of the uterine cavity was normal.

Discussion Female genital tuberculosis is a rare disease in developed countries; however, it often leads to problems, such as pelvic pain and infertility in developing countries.2 In this case, we were aware of the possibility for uterine tuberculosis because the patient had a history of tuberculosis peritonitis. It might be difficult to con-

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Figure 2 (a–c) Laparoscopic images. (a) The mass measured approximately 7 cm in diameter and had a yellow–gray surface. (b) The mass was found to adhere to the surface of the uterus. (c) Rupture of the mass was inevitable in spite of careful division of the severe adhesions on the appendix. (d) The gross appearance of the tumor, which contained thick, yellowish and foamy tubercles. (e) The histopathological study demonstrated the presence of epithelioid granulomas with central caseous necrosis (Scale bar: 1000 μm; hematoxylin–eosin stain).

sider the tuberculous granuloma as a differential diagnosis without previous history of tuberculous peritonitis. As the clinical manifestations and gross appearance of tuberculous granuloma are often similar to those of ovarian carcinoma, making a preoperative differential diagnosis between peritoneal tuberculosis and ovarian carcinoma can be difficult. Table 1 summarizes five previously reported cases of patients suspected as having ovarian carcinoma and finally diagnosed with tuberculosis between 2006 and 2012.3–7 Twenty-six cases described by Sharma et al. showed that eight patients had a history of tuberculous treatment.7 To make early diagnosis and timely management possible, the possibility for peritoneal tuberculosis should be kept in mind while treating female patients with pelvic masses of unknown origin, especially those with history of tuberculosis. Methods of treatment were variable depending on patient backgrounds. Hysterectomies were carried out on the parous women in these studies. However, these studies did not address operative methods for fertility preservation. Imaging methods, such as ultrasonography, CT and MRI, may be helpful for localizing lesions and determining the differential diagnosis. Ultrasonography may be useful for evaluating the size, number, position and relation of the uterus and echogenic

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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Table 1 Recently published reports of suspected ovarian carcinoma finally diagnosed as tuberculosis Author (year)Ref.

Case(s)

TB history

Methods of treatment

Operative findings of tuberculous granuloma

Boss et al. (2012)3

27 years old

None

TAHBSO and appendectomy followed by ATT

Patel et al. (2012)6 Oge et al. (2012)5

15 cases, mean age 39 years (range 19–65) 20 cases, mean age 37.5 years (range 16–70)

ND

Exploratory laparotomy followed by ATT Oophorectomy (63%), followed by ATT

Sharma et al. (2010)7

26 cases, mean age 34.7 years (range 15–65)

8 cases (30.7%)

Bilaterally enlarged ovaries, inflamed fallopian tubes adherent to the pelvic sidewalls, a large amount of ascetic fluid Ill-defined, irregular, nodular pelvic mass (100%), omental thickening (5%) Widespread miliary nodules (81%), adhesions (45%), adnexal masses (72%), caseous necrotic substances (36%) Adhesions (100%), caseation (57.7%), ascites (46.2%)

Koc et al. (2006)4

22 cases, mean age 36.9 years (range 21–68)

None

None

TAH (15.4%), ovariotomy (42.3%), followed by ATT Laparotomy (55%), laparoscopic evaluation (40%), followed by ATT

Ascites, multiple white colored tubercules and adhesion

ATT, antitubercular treatment; ND, no data available; TAHBSO, total abdominal hysterectomy, bilateral salpingo-oophorectomy; TB, tuberculosis.

structures.8 CT is the most sensitive modality for identifying calcifications, which occur in over 50% of cases of genitourinary tuberculosis.9 MRI can help to identify the location and origin of masses more accurately than ultrasonography or CT. Sharma et al. reported that, on MRI, tubercular tubo-ovarian masses usually present as complex adnexal masses with large amounts of ascites and may demonstrate thickened tubes or nodularities along the tubo-ovarian surface with or without lymphadenopathy.10 Clinicians should not rely solely on MRI findings to make a diagnosis of genital tuberculosis when ovarian cancer is possible in order to avoid missing the correct diagnosis. A tissue biopsy for histology and culture growth of Mycobacterium is the most sensitive and specific diagnostic procedure for detecting tuberculous peritonitis. There is no consensus regarding the optimal method for making a preoperative diagnosis of pelvic tuberculosis. Oge et al. reported that ultrasound-guided biopsies are useful in selected patients.5 However, in this case, we did not perform a biopsy of the mass for several reasons. First, the mass could not be approached transvaginally due to the long distance from the periphery to the mass. Second, we were concerned that the mass may rupture and potentially spread. Third, there were risks associated with the adhesions caused by the previous tuberculous peritonitis infection.

Granuloma formation, caseous necrosis and cavitation are stages of progressive tuberculous infection that can eventually destroy the entire uterus and ultimately cause infertility. Because the patient in this case had hoped for pregnancy, we determined that surgical intervention was needed for diagnostic and therapeutic management. Less invasive laparoscopic surgery may be considered as the first choice of therapy for the diagnosis and treatment of tuberculous granuloma resembling ovarian tumors or degenerative leiomyoma of the uterus. Genital tuberculosis is infrequent, usually asymptomatic and is often incidentally detected in female patients presenting with pelvic masses. However, tuberculous granuloma must be considered in the differential diagnosis of pelvic masses in women of reproductive age because the major sequela of pelvic tuberculosis is infertility. A distinctive feature of our case was the use of the laparoscopic technique not only in diagnosis but also for treatment of tuberculous granuloma. In the modern age, the laparoscopic diagnosis and treatment of tuberculous granuloma may therefore be a feasible procedure in the patient who wants to conceive.

Disclosure None of the authors has anything to disclose.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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References 1. Chow TW, Lim BK, Vallipuram S. The masquerades of female pelvic tuberculosis: Case reports and review of literature on clinical presentations and diagnosis. J Obstet Gynaecol Res 2002; 28: 203–210. 2. Parikh FR, Nadkarni SG, Kamat SA, Naik N, Soonawala SB, Parikh RM. Genital tuberculosis – A major pelvic factor causing infertility in Indian women. Fertil Steril 1997; 67: 497– 500. 3. Boss JD, Shah CT, Oluwole O, Sheagren JN. TB peritonitis mistaken for ovarian carcinomatosis based on an elevated CA-125. Case Rep Med 2012; 2012: 215293– 215295. 4. Koc S, Beydilli G, Tulunay G et al. Peritoneal tuberculosis mimicking advanced ovarian cancer: A retrospective review of 22 cases. Gynecol Oncol 2006; 103: 565– 569.

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5. Oge T, Ozalp SS, Yalcin OT et al. Peritoneal tuberculosis mimicking ovarian cancer. Eur J Obstet Gynecol Reprod Biol 2012; 162: 105–108. 6. Patel SM, Lahamge KK, Desai AD, Dave KS. Ovarian carcinoma or abdominal tuberculosis? A diagnostic dilemma: Study of fifteen cases. J Obstet Gynaecol India 2012; 62: 176– 178. 7. Sharma JB, Jain SK, Pushparaj M et al. Abdomino-peritoneal tuberculosis masquerading as ovarian cancer: A retrospective study of 26 cases. Arch Gynecol Obstet 2010; 282: 643–648. 8. Das KM, Indudhara R, Vaidyanathan S. Sonographic features of genitourinary tuberculosis. AJR Am J Roentgenol 1992; 158: 327–329. 9. Lee WK, Van Tonder F, Tartaglia CJ et al. CT appearances of abdominal tuberculosis. Clin Radiol 2012; 67: 596–604. 10. Sharma JB, Karmakar D, Hari S et al. Magnetic resonance imaging findings among women with tubercular tuboovarian masses. Int J Gynaecol Obstet 2011; 113: 76–80.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Pelvic tuberculous granuloma successfully treated with laparoscopy to preserve fertility: a case report and review of the published work.

Tuberculous granuloma must be considered in the differential diagnosis of pelvic masses in women of reproductive age because the major sequela of pelv...
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