<!DOCTYPE article PUBLIC "-//NLM//DTD Journal Publishing DTD v2.3 20070202//EN" "../dtd/journalpublishing.dtd">
<article xmlns:xlink="http://www.w3.org/1999/xlink" xmlns:mml="http://www.w3.org/1998/Math/MathML" article-type="case-report" xml:lang="en">
	<front>
		<journal-meta>
			<journal-id journal-id-type="publisher-id">JRI</journal-id>
			<journal-title>Journal of Reproduction and Infertility</journal-title>
			<issn pub-type="ppub">2228-5482</issn>
			<issn pub-type="epub">2251-676X</issn>
			<publisher>
				<publisher-name>Avicenna Research Institute</publisher-name>
			</publisher>
		</journal-meta>
		<article-meta>
			<article-id pub-id-type="publisher-id">JRI-11-59</article-id>
			<article-categories>
				<subj-group subj-group-type="heading">
					<subject>Case Report</subject>
				</subj-group>
			</article-categories>
			<title-group>
				<article-title>Spontaneous Pregnancy in Primary Amenorrhea; a Case Report</article-title>
			</title-group>
			<contrib-group>
				<contrib contrib-type="author" corresp="yes">
					<name>
						<surname>Das</surname>
						<given-names>Ananya</given-names>
					</name>
					<degrees>M.D</degrees>
					<xref ref-type="aff" rid="AF0001">1</xref>
					<xref ref-type="corresp" rid="cor1">&#x002A;</xref>
				</contrib>
				<contrib contrib-type="author">
					<name>
						<surname>Borah</surname>
						<given-names>Tulon</given-names>
					</name>
					<degrees>M.D</degrees>
					<xref ref-type="aff" rid="AF0001">1</xref>
				</contrib>
				<contrib contrib-type="author">
					<name>
						<surname>Panda</surname>
						<given-names>Subrat</given-names>
					</name>
					<degrees>M.D</degrees>
					<xref ref-type="aff" rid="AF0001">1</xref>
				</contrib>
			</contrib-group>
			<aff id="AF0001">
				<label>1</label>Department of Obstetrics &#x0026; Gynecology, North Eastern Indira Gandhi Regional Institute of Health &#x0026; Medical Sciences (NEIGRIHMS), Meghalaya, India</aff>
			<author-notes>
				<corresp id="cor1"><label>&#x002A;</label>
					<italic>Corresponding Author:</italic> Dr. Ananya Das, Department of Obstetrics &#x0026; Gynecology, North Eastern Indira Gandhi Regional Institute of Health &#x0026; Medical Sciences (NEIGRIHMS), Mawdiangdiang, Shillong-793018, Meghalaya, India. <italic>E-mail:</italic> <email xlink:href="mailmedrananyadas@rediffmail.com">mailmedrananyadas@rediffmail.com</email>
				</corresp>
			</author-notes>
			<pub-date pub-type="ppub">
				<season>Apr-Jun</season>
				<year>2010</year>
			</pub-date>
			<volume>11</volume>
			<issue>1</issue>
			<fpage>59</fpage>
			<lpage>60</lpage>
			<history>
				<date date-type="received">
					<day>16</day>
					<month>12</month>
					<year>2009</year>
				</date>
				<date date-type="accepted">
					<day>24</day>
					<month>02</month>
					<year>2010</year>
				</date>
			</history>
			<permissions>
				<copyright-statement>Copyright &#x00A9; 2010 Avicenna Research Institute</copyright-statement>
				<copyright-year>2010</copyright-year>
				<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/">
					<p>This work is licensed under a Creative Commons Attribution-NonCommercial 3.0 Unported License which allows users to read, copy, distribute and make derivative works for non-commercial purposes from the material, as long as the author of the original work is cited properly.</p>
				</license>
			</permissions>
			<abstract>
				<sec id="st1">
					<title>Introduction</title>
					<p>Primary ovarian failure (POF) is a syndrome composed of amenorrhea, estrogen deficiency and Follicular Stimulating Hormone (FSH) of menopausal ranges in young women. In this article, we report a case of primary amenorrhea that presented with full term pregnancy.</p>
				</sec>
				<sec id="st2">
					<title>Case Presentation</title>
					<p>A 29-year old woman with a history of primary amenorrhea attended hospital with full term pregnancy. She had experienced a few episodes of withdrawal bleeding on hormonal treatment initially and she had conceived spontaneously. Subsequently, she had uneventful pregnancy and caesarean delivery on maternal request.</p>
				</sec>
				<sec id="st3">
					<title>Conclusion</title>
					<p>This case was presented to emphasize the real chances of spontaneous conceptions due to intermittent and unpredictable ovarian function in patients with POI. Nevertheless, egg donation is still considered the best option for infertility in such women.</p>
				</sec>
			</abstract>
			<kwd-group>
				<kwd>Estrogen deficiency</kwd>
				<kwd>Follicular stimulating hormone</kwd>
				<kwd>Pregnancy</kwd>
				<kwd>Primary amenorrhea</kwd>
				<kwd>Primary ovarian insufficiency</kwd>
			</kwd-group>
		</article-meta>
		<notes>
		<p>
				<bold>To cite this article:</bold> Das A, Borah T, Panda S. Spontaneous Pregnancy in Primary Amenorrhea; a Case Report. J Reprod Infertil. 2010;11(1):59&#x2013;60.</p></notes>
	</front>
	<body>
		<sec id="S0001" sec-type="intro">
			<title>Introduction</title>
			<p>Fuller Albright first described a condition he termed Primary Ovarian Insufficiency (POI) in 1942 when he reported a syndrome of amenorrhea, estrogen deficiency and menopausal Follicular Stimulating Hormone (FSH) levels in young women (<xref ref-type="bibr" rid="CIT0001">1</xref>). The syndrome affects approximately 1% of the female population.</p>
			<p>Herein, we report such a case that had primary amenorrhea and presented with full term pregnancy.</p>
		</sec>
		<sec id="S0002">
			<title>Case Presentation</title>
			<p>A 29-year-old woman attended antenatal out-patient department of North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences (NEIGRIHMS), Shillong, Meghalaya, India with full term pregnancy in October 2009. She gave a very interesting history of primary amenorrhea with a few episodes of withdrawal bleeding upon hormonal treatment in the past. For the past 3 &#x2013; 4 years she had not been receiving any treatment and had remained amenorrhoeic. After 4 years of marriage, she went to a local doctor for nausea and vomiting and, to her surprise, her urine pregnancy test was positive. Her pregnancy was later confirmed by ultrasound. Her Expected Date of Delivery (EDD) was calculated from the first trimester ultrasound, which confirmed the gestational maturity.</p>
			<p>On examination, her vital signs were within normal limits. On per abdominal examination, uterus was found to be in the form of a full-term pregnancy. The fetus was in cephalic presentation and Fetal Heart Rate (FHR) was 136 beats per minute and regular. Per vaginal examination revealed a closed internal Os with soft, short and central cervix. Pelvis seemed adequate for delivery of the fetus. Caesarean section was performed on maternal request and a healthy male baby weighing 2.5 <italic>kg</italic> was delivered. She had an uneventful puerperium.</p>
		</sec>
		<sec id="S0003" sec-type="discussion">
			<title>Discussion</title>
			<p>Primary Ovarian Insufficiency has varied manifestations of amenorrhea, oligomenorrhea or dysfunctional uterine bleeding. Overall, approximately 10% of women with POI present with primary amenorrhea (<xref ref-type="bibr" rid="CIT0002">2</xref>, <xref ref-type="bibr" rid="CIT0003">3</xref>). Young women who develop POI need special care.</p>
			<p>A similar case of spontaneous pregnancy has been reported in a 27-year old woman with hypergonadotropic ovarian failure (<xref ref-type="bibr" rid="CIT0004">4</xref>).</p>
			<p>We suspected her to be a case of primary ovarian insufficiency on the basis of history alone and in the absence of any laboratory test of increased FSH, since we admitted her for the first time in full term pregnancy. There was also no suggestive family history or any associated medical disorders.</p>
			<p>This case was reported to emphasize the real chances for a spontaneous conception because of the intermittent and unpredictable ovarian function in these patients.</p>
		</sec>
		<sec id="S0004" sec-type="conclusion">
			<title>Conclusion</title>
			<p>Primary ovarian insufficiency occurs in only 1% of women it can still result in spontaneous pregnancy in 5-10% of the cases. Egg donation is still considered the best option for infertility in such women. Women with POI should be educated on the nature of the disease and the current research efforts. It is important to be aware of the condition and the options for future treatment.</p>
		</sec>
	</body>
	<back>
		<ref-list>
			<title>References</title>
			<ref id="CIT0001">
				<label>1</label>
				<nlm-citation citation-type="journal">
					<person-group person-group-type="author">
						<name>
							<surname>Albright</surname>
							<given-names>F</given-names>
						</name>
						<name>
							<surname>Smith</surname>
							<given-names>PH</given-names>
						</name>
						<name>
							<surname>Fraser</surname>
							<given-names>R</given-names>
						</name>
					</person-group>
					<article-title>A syndrome characterized by primary ovarian insufficiency and decreased stature: Report of 11 Cases with a Digression on Hormonal Control of Axillary and Pubic Hair</article-title>
					<source>Am J Med Sci.</source>
					<year>1942</year>
					<volume>204</volume>
					<issue>5</issue>
					<fpage>625</fpage>
					<lpage>48</lpage>
				</nlm-citation>
			</ref>
			<ref id="CIT0002">
				<label>2</label>
				<nlm-citation citation-type="journal">
					<person-group person-group-type="author">
						<name>
							<surname>Rebar</surname>
							<given-names>RW</given-names>
						</name>
						<name>
							<surname>Erickson</surname>
							<given-names>GF</given-names>
						</name>
						<name>
							<surname>Yen</surname>
							<given-names>SS</given-names>
						</name>
					</person-group>
					<article-title>Idiopathic premature ovarian failure: clinical and endocrine characteristics</article-title>
					<source>Fertil Steril.</source>
					<year>1982</year>
					<volume>37</volume>
					<issue>1</issue>
					<fpage>35</fpage>
					<lpage>41</lpage>
				</nlm-citation>
			</ref>
			<ref id="CIT0003">
				<label>3</label>
				<nlm-citation citation-type="journal">
					<person-group person-group-type="author">
						<name>
							<surname>Rebar</surname>
							<given-names>RW</given-names>
						</name>
						<name>
							<surname>Connolly</surname>
							<given-names>HV</given-names>
						</name>
					</person-group>
					<article-title>Clinical features of young women with hypergonadotropic amenorrhea</article-title>
					<source>Fertil Steril.</source>
					<year>1990</year>
					<volume>53</volume>
					<issue>5</issue>
					<fpage>804</fpage>
					<lpage>10</lpage>
				</nlm-citation>
			</ref>
			<ref id="CIT0004">
				<label>4</label>
				<nlm-citation citation-type="journal">
					<person-group person-group-type="author">
						<name>
							<surname>Wright</surname>
							<given-names>CS</given-names>
						</name>
						<name>
							<surname>Jacobs</surname>
							<given-names>HS</given-names>
						</name>
					</person-group>
					<article-title>Spontaneous pregnancy in a patient with hypergonadotrophic ovarian failure</article-title>
					<source>Br J Obstet Gynaecol.</source>
					<year>1979</year>
					<volume>86</volume>
					<issue>5</issue>
					<fpage>389</fpage>
					<lpage>92</lpage>
				</nlm-citation>
			</ref>
		</ref-list>
	</back>
</article>
