Human Reproduction, Vol. 15, No. 8, 1873-1874,
August 2000
© 2000 European Society of Human Reproduction and Embryology
Letters to the editor |
Stanford University Endoscopy Center for Training and Technology, Medical School Office Building, Suite X340, Stanford CA 94305, USA
Dear Sir
We are pleased that Drs Nisolle and Donnez have found this case as unique and perplexing as we did when preparing our report (Nezhat and Smith, 1999
). In the absence of a previously reported case with the same anatomy we set out to describe our findings and, using the current understanding of Müllerian embryology, hypothesize their evolution.
While we acknowledge the contributions of Drs Donnez and Nisolle, we roundly reject their theory that our patient had an `inflammatory reaction between an endocervical canal and uterine cavity'. Indeed, final histopathologic diagnosis revealed endometrium in both rudimentary cavities. Additionally, the proposed area of `stricture' produced endometrial polyps which projected into the inferior endometrial cavity (the proposed atretic cervix).
Given that a lack of lateral fusion of the Müllerian ducts was at least in part responsible for our patient's anatomy, this case may represent a variant of double uterus. However, no vaginal cavity of even the smallest calibre was encountered to support the suggestion of a blind hemivagina. Additionally, with the clear presence of a well-developed unicornuate uterus, we stand behind the hypothesis presented.
We have adopted the designation of a second rudimentary horn in view of the current variations of unicornuate uterus described by the American Fertility Society (AFS) classification (American Fertility Society, 1998). Accordingly, our illustration was in the manner of those provided by the AFS classification, which does not include such sequelae of anomalies as haematosalpinx, as described in the text.
Regarding the management of this case, while creation of a fistula from the uterine cavity to the vagina has been described, (Spence, 1998), most cases have led to disappointing results and some have led to sepsis-related deaths. We submit however that the surgical approach to Müllerian anomalies is varied and should be determined on a case-by-case basis. We echo the concern to preserve the patient's ovaries when possible. As described in our original article, the attenuated vascular supply and severe involvement by endometriosis warranted the unilateral oophorectomy.
The 1997 report by Drs Donnez and Nisolle on 14 cases were not discussed by Amara et al. (1997) because it was not yet published at that time. The focus of the article by Donnez and Nisolle was hysteroscopic treatment of uterine septa, not laparoscopic management of Müllerian anomalies. In the last paragraph of the article it is simply stated that 14 cases `of laparoscopic resection of rudimentary horns', were performed. The cases themselves are not featured in the report at all.
Finally we are delighted that our report has encouraged discussion regarding Müllerian embryology; that was exactly the purpose. The penultimate statement by Drs Donnez and Nisolle is that our denomination `may not be the right one'. The last sentence however suggests the `impossibility' of our hypothesis. In an era when cloning is fast upon us and laparoscopic advances are continuing to be made, may we suggest that `impossible is impossible'?
References
Amara, D.P., Nezhat, F., Giudice, L. et al. (1997) Laparoscopic management of a non-communicating uterine horn in a patient with an acute abdomen. Surg. Laparosc. Endosc., 7, 56–59.[Web of Science][Medline]
American Fertility Society (1988) The AFS classification of adnexal adhesions, distal tubal occlusion, tubal occlusion secondary to tubal ligation, tubal pregnancies, Mullerian anomalies and intrauterine adhesions. Fertil. Steril., 49, 944–955.[Web of Science][Medline]
Donnez, J. and Nisolle, M. (1997) Endoscopic laser treatment of uterine malformations. Hum. Reprod., 12, 1381–1387.[Abstract]
Nezhat, C.R. and Smith, K.S. (1999) Laparoscopic management of a unicornuate uterus with two rudimentary horns. Hum. Reprod., 14, 1965–1968
Spence, J.E.H. (1998) Vaginal and uterine anomalies in pediatric and adolescent patient. J. Pediatr. Adolesc. Gynecol., 11, 3–11.[Medline]
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