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Case 94: Uterus Didelphys with Obstructing Hemivaginal Septum and Ipsilateral Renal Agenesis¹

¹ From the Hospital de S. João and Faculdade de Medicina do Porto, Rua S. João Bosco, 305-8C, 4100-531 Porto, Portugal. Received July 21, 2003; revision requested October 13; revision received March 30, 2004; accepted April 16; final version accepted May 13.

Correspondence: Address correspondence to: A.J.M. (e-mail: ajbmadureira{at}clix.pt).

	History

TOP History Imaging Findings Discussion References

 
A 12-year-old girl was admitted to the hospital because of lower abdominal pain. She reported previous episodes of similar, but less severe, pain. Physical examination did not reveal any abnormal findings, and there was only mild tenderness at palpation of the middle lower abdomen. Findings of laboratory tests were unremarkable, with a total white blood cell count of 7.16 x 10⁹/L (normal range, [5.5–11.5] x 10⁹/L), a differential cell count of 64.8% neutrophils (normal range, 40.9%–64.9%), and a hemoglobin level of 14.2 g/dL (normal range, 14.0–16.0 g/dL). The patient's medical history was noncontributory. Menstruation first occurred in this patient at 11 years of age; at admission, she was on the 3rd day of her menstrual cycle.

The patient underwent ultrasonography (images not shown). Magnetic resonance (MR) imaging of the pelvis was recommended and performed to enable further evaluation.

	Imaging Findings

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MR images show a large mass (8 x 11-cm) occupying the lower pelvis with high signal intensity on T1-weighted images (Fig 1a) and low signal intensity on T2-weighted images (Figs 1b, 1c); these signal characteristics indicate that the mass has a hematic nature (ie, composed of blood). The T1-weighted high-signal-intensity area and the T2-weighted low-signal-intensity area indicate the presence of intracellular methemoglobin from recent bleeding and rebleeding, and they cannot be caused by subacute extracellular methemoglobin. The mass is well defined and has regular borders. Two elongated structures with a muscular wall can be identified in contact with the upper portion of the mass: The structure on the left is in direct communication with the mass, and it is filled with the same blood products as those that fill the mass (Figs 1b, 1d, 1f). On the other hand, the structure on the right has high signal intensity; however, no direct communication between the structures can be established (Figs 1c, 1d, 1f). These three structures represent two diverging uterine horns and a massively dilated blood-filled left hemivagina. The right cervix is not clearly identified, as it is markedly compressed by the left cervix. On the coronal T2-weighted image, the right kidney is in its usual location and appears unremarkable, while the left kidney cannot be identified (Fig 1e).

View larger version (128K): [in this window] [in a new window] [Download PPT slide]   Figure 1a: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

View larger version (130K): [in this window] [in a new window] [Download PPT slide]   Figure 1b: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

View larger version (148K): [in this window] [in a new window] [Download PPT slide]   Figure 1c: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

View larger version (131K): [in this window] [in a new window] [Download PPT slide]   Figure 1d: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

View larger version (146K): [in this window] [in a new window] [Download PPT slide]   Figure 1e: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

View larger version (123K): [in this window] [in a new window] [Download PPT slide]   Figure 1f: Selected MR images of the pelvis and abdomen. (a) Transverse T1-weighted image (repetition time msec/echo time msec, 500/15; two signals acquired) shows an 8-cm-diameter pelvic mass with high signal intensity and a muscular wall (arrows), representing a massively distended and blood-filled left hemivagina. (b) Transverse T2-weighted fast spin-echo image (2100/100 [effective]; echo train length, 19; six signals acquired). Fluid in the central part of the vagina has low signal intensity and is surrounded by concentric T2 hyperintense and hypointense rings, thus indicating the presence of blood in various stages of evolution. Note the communication through the narrow neck (arrowhead) with the mildly dilated and blood-filled left uterine horn (arrows). (c) Sagittal T2-weighted fast spin-echo image obtained slightly to the right of the midline (2100/100 [effective]; echo train length, 19; six signals acquired). The right uterine horn (arrows) does not communicate with the 11-cm-long vagina. (d, e) Coronal T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; eight signals acquired). In a more anterior image (d), the left uterine horn (arrows) is dilated and filled with blood, whereas the right uterine horn (arrowhead) has a normal appearance. The right kidney (arrowhead in e) is seen, and the left renal fossa is occupied by small-bowel loops (arrow in e). (f) Transverse oblique T2-weighted fast spin-echo image obtained with fat suppression (2100/100 [effective], echo train length, 19; eight signals acquired). Communication of the left uterine horn (arrows) with the dilated left hemivagina is demonstrated; the appearance of the right uterine horn (arrowheads) is normal.

	Discussion

TOP History Imaging Findings Discussion References

At cystoscopy, an impression on the posterior vesical wall was noted, and the left ureteral meatus was not identified; the position of the right ureteral meatus was normal. The child also underwent vaginoscopy and hysteroscopy at the same time; these evaluations revealed a marked impression on the left anterolateral wall of the vagina that extended from the lower third of the vagina to the dome of the vagina. The right cervix and endocervical canal were evaluated, and the findings were unremarkable. A 2-cm-long vaginal septotomy was performed, and 150 mL of fluid was drained. After this procedure, the left cervix was evaluated; it appeared normal, with the usual and characteristic cryptae. The left cervix was in direct communication with the left uterine horn and ostium of the left fallopian tube, both of which were also identified. No communication was found between the two endometrial cavities.

Since being discharged, this patient has been having normal menstrual cycles. Follow-up MR images (Fig 2) showed the two uterine cervices were clearly separated from each other, and this allowed us to confirm the diagnosis of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis.

View larger version (137K): [in this window] [in a new window] [Download PPT slide]   Figure 2a: Follow-up MR images obtained after surgery. (a) Coronal and (b) transverse oblique T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; six signals acquired; field of view, 350 mm; relative field of view, 80%; matrix, 191 x 256) clearly depict the two cervices (arrows) separated by a band of fibrous tissue (arrowhead). A hyperintense right ovarian cyst can be seen.

View larger version (109K): [in this window] [in a new window] [Download PPT slide]   Figure 2b: Follow-up MR images obtained after surgery. (a) Coronal and (b) transverse oblique T2-weighted fast spin-echo images (2100/100 [effective]; echo train length, 19; six signals acquired; field of view, 350 mm; relative field of view, 80%; matrix, 191 x 256) clearly depict the two cervices (arrows) separated by a band of fibrous tissue (arrowhead). A hyperintense right ovarian cyst can be seen.

It is fundamental to have a basic knowledge of the embryology of the female genital tract to understand this group of congenital uterine anomalies. The uterus, fallopian tubes, and upper two-thirds of the vagina originate from paired müllerian ducts, whereas the lower third of the vagina arises separately from the urogenital sinus (6). This explains the occurrence of cases of complete uterine agenesis with normal external vaginal development. The first stage of müllerian duct development begins at approximately 6 weeks gestational age when the paired müllerian ducts invaginate and then grow caudally and cross over the wolffian ducts to meet at the midline. The subsequent three phases (fusion, resorption, and vaginal induction) proceed in an orderly fashion from the 9th to the 22nd gestational week. Müllerian duct development occurs in close association with the development of the urinary system, and this explains the frequent association of anomalies of these two systems.

The most frequently used system for classification of MDAs was proposed by Buttram and Gibbons (7); this system is used to organize these anomalies into six categories. Class I anomalies are associated with hypoplasia or agenesis of part or all of the tubes, uterus, cervix, or vagina (Mayer-Rokitansky-Küster-Hauser syndrome). Class II anomalies comprise a partial or complete unilateral agenesis (unicornuate uterus) and account for approximately 15% of MDAs (2). There are four subtypes of unicornuate uterus: simple unicornuate uterus (35%), unicornuate uterus with noncavitary rudimentary horn (33%), unicornuate uterus with noncommunicating cavitary horn (22%), and unicornuate uterus with communicating cavitary horn (10%). The characteristic MR appearance is that of a banana-shaped uterine cavity, with or without a rudimentary horn. In patients with uterus didelphys (class III anomaly), two separate, widely divergent uterine horns and two cervices are present. A vertical septum extending into the upper vagina can be identified with MR imaging in up to 75% of patients (8). In a small number of patients, a transverse vaginal septum obstructs one of the uterine canals, thus leading to hematometrocolpos. Bicornuate uterus (class IV anomaly) results from partial failure of müllerian duct fusion and is characterized by a concave external fundal contour with a fundal cleft larger than 1 cm separating the two uterine horns above a single cervix and increased intercornual distance (more than 4 cm). Occasionally, two cervices may be present. Arcuate uterus is the mildest partial fusion anomaly and is considered a normal variant. The endometrial cavity is not divided, and the external contour of the uterus is flattened or minimally concave. Septate uterus (class V anomaly) results from failed resorption of the midline septum after normal müllerian duct fusion. The key feature that allows the differentiation of this anomaly from bicornuate uterus is the presence of a normal or minimally concave (<1 cm) external uterine contour; MR imaging is the best noninvasive modality for assessment of this feature. Class VI anomaly is a separate anomaly that results from in utero exposure to diethylstilbestrol. The classic and most common uterine abnormality is a T-shaped uterus.

The specific association between uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis has been recognized (9). As of 1998, more than 180 cases had been reported in the literature (10). The usual clinical presentation of this syndrome is abdominal pain that starts right after menarche and is caused by hematocolpos.

The association of renal agenesis and MDA is derived from the close embryologic development of the urinary and genital systems. In a study of 57 patients with MDAs, Li et al (1) found an incidence of renal agenesis of 30%. Renal agenesis was detected more frequently in patients with obstructed MDAs (85.6%) than in patients with unobstructed MDAs (13.6%); this difference was statistically significant (P < .05). An increased association of renal agenesis in patients with uterus didelphys compared with other types of MDA was also reported. In fact, among the 17 patients with renal agenesis, there were two cases of uterine and vaginal agenesis, two cases of unicornuate uterus, and 13 cases of uterus didelphys. Unlike the current case, however, most of the anomalies were located on the right side; this finding was in accordance with findings of previous reports (11). An important point is that in all of the patients with obstructed uterus didelphys, renal agenesis was located on the same side as the obstruction. It is worth mentioning that in cases of unicornuate uteri, the renal anomalies that may be associated are also always ipsilateral to the rudimentary or absent horn (2).

MR imaging is the most accurate noninvasive modality available for classification of MDA (12,13) because it provides information about the external fundal contour, shape of the uterine cavity, and tissue characterization of the septa (when present).

	FOOTNOTES

 

Part one of this case appeared 4 months previously and may contain larger images.

 

	References

TOP History Imaging Findings Discussion References

 

1. Li S, Qayyum A, Coakley FV, Hricak H. Association of renal agenesis and müllerian duct anomalies. J Comput Assist Tomogr 2000;24:829–834.[CrossRef][Medline]
2. Brody JM, Koelliker SL, Frishman GN. Unicornuate uterus: imaging appearance, associated anomalies, and clinical implications. AJR Am J Roentgenol 1998;171:1341–1347.[Free Full Text]
3. Nahum GG. Uterine anomalies: how common are they and what is their distribution among subtypes? J Reprod Med 1998;43:877–887.[Medline]
4. Stampe Sorensen S. Estimated prevalence of müllerian anomalies. Acta Obstet Gynecol Scand 1988;67:441–445.[Medline]
5. Shenker L, Brickam FE. Bicornuate uterus with incomplete vaginal septum and unilateral renal agenesis. Radiology 1979;133:455–457.[Abstract]
6. Woodward PJ, Gilfeather M. Magnetic resonance imaging of the female pelvis. Semin Ultrasound CT MR 1998;19:90–103.[CrossRef][Medline]
7. Buttram VC Jr, Gibbons WE. Müllerian anomalies: a proposed classification (an analysis of 144 cases). Fertil Steril 1979;32:40–46.[Medline]
8. O'Neill MJ, Yoder IC, Connolly SA, Mueller PR. Imaging evaluation and classification of developmental anomalies of the female reproductive system with an emphasis on MR imaging. AJR Am J Roentgenol 1999;173:407–416.[Free Full Text]
9. Purslow CE. A case of unilateral hematocolpos, hematometra, and hematosalpinx [letter]. J Obstet Gynecol Br Emp 1922;29:643.
10. Tanaka YO, Kurosaki Y, Kobayashi T, et al. Uterus didelphys associated with obstructed hemivagina and ipsilateral renal agenesis: MR findings in seven cases. Abdom Imaging 1998;23:437–441.[CrossRef][Medline]
11. Yoder IC, Pfister RC. Unilateral hematocolpos and ipsilateral renal agenesis: report of two cases and review of the literature. AJR Am J Roentgenol 1976;127:303–308.[Abstract]
12. Carrington BM, Hricak H, Nuruddin RN, Secaf E, Laros RK Jr, Hill EC. Müllerian duct anomalies: MR imaging evaluation. Radiology 1990;176:715–720.[Abstract/Free Full Text]
13. Troiano RN, McCarthy SM. Müllerian duct anomalies: imaging and clinical issues. Radiology 2004;233:19–74.[Abstract/Free Full Text]

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