<?xml version="1.0" encoding="UTF-8"?><!DOCTYPE article  PUBLIC "-//NLM//DTD Journal Publishing DTD v3.0 20080202//EN" "http://dtd.nlm.nih.gov/publishing/3.0/journalpublishing3.dtd"><article xmlns:mml="http://www.w3.org/1998/Math/MathML" xmlns:xlink="http://www.w3.org/1999/xlink" dtd-version="3.0" xml:lang="en" article-type="research article"><front><journal-meta><journal-id journal-id-type="publisher-id">OJU</journal-id><journal-title-group><journal-title>Open Journal of Urology</journal-title></journal-title-group><issn pub-type="epub">2160-5440</issn><publisher><publisher-name>Scientific Research Publishing</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="doi">10.4236/oju.2023.133010</article-id><article-id pub-id-type="publisher-id">OJU-123530</article-id><article-categories><subj-group subj-group-type="heading"><subject>Articles</subject></subj-group><subj-group subj-group-type="Discipline-v2"><subject>Medicine&amp;Healthcare</subject></subj-group></article-categories><title-group><article-title>
 
 
  Persistent Congenital Paraurethral Cyst, a Rare Cause of Dysuria in a Girl
 
</article-title></title-group><contrib-group><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Doudou</surname><given-names>Gueye</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref><xref ref-type="corresp" rid="cor1"><sup>*</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Ndeye</surname><given-names>Fatou Seck</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Florent</surname><given-names>Tshibwid A. Zeng</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Papa</surname><given-names>Alassane Mbaye</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Ibrahima</surname><given-names>Bocar Wellé</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Lissoune</surname><given-names>Cissé</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Ndeye</surname><given-names>Aby Ndoye</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib><contrib contrib-type="author" xlink:type="simple"><name name-style="western"><surname>Alo&amp;#239;se</surname><given-names>Sagna</given-names></name><xref ref-type="aff" rid="aff1"><sup>1</sup></xref></contrib></contrib-group><aff id="aff1"><addr-line>Department of Pediatric Surgery, Albert Royer National Children’s Hospital Centre, Université Cheikh Anta Diop, Dakar, Senegal</addr-line></aff><pub-date pub-type="epub"><day>06</day><month>03</month><year>2023</year></pub-date><volume>13</volume><issue>03</issue><fpage>83</fpage><lpage>87</lpage><history><date date-type="received"><day>8,</day>	<month>January</month>	<year>2023</year></date><date date-type="rev-recd"><day>4,</day>	<month>March</month>	<year>2023</year>	</date><date date-type="accepted"><day>7,</day>	<month>March</month>	<year>2023</year></date></history><permissions><copyright-statement>&#169; Copyright  2014 by authors and Scientific Research Publishing Inc. </copyright-statement><copyright-year>2014</copyright-year><license><license-p>This work is licensed under the Creative Commons Attribution International License (CC BY). http://creativecommons.org/licenses/by/4.0/</license-p></license></permissions><abstract><p>
 
 
  Congenital paraurethral cyst rarely occurs. Its natural history is spontaneous regression, leading to conservative management adoption. We report an exceptional case of a 13-year-old female with a persistent congenital paraurethral cyst, leading to dysuria. We surgically excised the cyst, and the outcomes were unremarkable 20 months postoperatively.
 
</p></abstract><kwd-group><kwd>Congenital Paraurethral Cyst</kwd><kwd> Persistent</kwd><kwd> Dysuria</kwd><kwd> Girl</kwd><kwd> Surgical Excision</kwd></kwd-group></article-meta></front><body><sec id="s1"><title>1. Introduction</title><p>Congenital paraurethral cyst (CPUC) is a rare condition, and its incidence is reported to range from one in 1038 to 7246 live female births [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] . It is due to ductal obstruction of paraurethral glands, Skene’s glands [<xref ref-type="bibr" rid="scirp.123530-ref2">2</xref>] . The diagnosis is usually made in neonate, during the physical examination, with investigations needed only for atypical presentation to exclude differentials [<xref ref-type="bibr" rid="scirp.123530-ref3">3</xref>] . The natural history of CPUC is regression, as influence of maternal hormones decreases. Its persistence is exceptional [<xref ref-type="bibr" rid="scirp.123530-ref4">4</xref>] [<xref ref-type="bibr" rid="scirp.123530-ref5">5</xref>] . Management of CPUC during the neonatal period is conservative, when surgery (excision or drainage) is indicated for persistent or lately diagnosed CPUC [<xref ref-type="bibr" rid="scirp.123530-ref3">3</xref>] . We report a girl thirteen years old who presented dysuria due to a persistent CPUC.</p></sec><sec id="s2"><title>2. Case Presentation</title><p>A thirteen-year-old girl was received in our department for dysuria and interlabial mass. The mother reported that soon after birth, a vulvar mass was identified in the patient. At the maternity, she was reassured that the mass would soon regress, and no investigations were requested. However, the mass persisted, and since infancy, it progressively increased its size, but no medical nor traditional management was sook. During the past months, the patient experienced progressive dysuria, which worsened two weeks before the consultation in our department. Then parents consulted at a district medical center, where she was referred. The patient had no medical history; she had her menarche a year ago.</p><p>On physical examination, vitals were within normal ranges. An interlabial globular pink mass was identified. It was soft, not painful, and was located on the right side of the urethral meatus, distorted, and deviated on the left side. Its largest diameter was 20 mm (<xref ref-type="fig" rid="fig1">Figure 1</xref>). Assumed diagnoses were CPUC or prolapsed megaureter. An abdominopelvic ultrasound (US) was ordered and did not identify any urinary tract anomaly. A bacteriological examination of urines did not show any bacteria.</p><p>The patient was hospitalized at our department. Initial management included parenteral paracetamol (15 mg/kg/6hours) as analgesic, and placement of a Foley catheter to allow better emptying of the bladder. The same day, she underwent surgical excision of the cyst under general anesthesia, using cautery. The implantation of the cyst was sutured with 4/0 Vicryl using interrupted sutures (<xref ref-type="fig" rid="fig2">Figure 2</xref>). Postoperatively, intravenous paracetamol was continued, along with ibuprofen (10 mg/kg/8hours). The immediate postoperative course was uneventful, and she was discharged 24 hours later, with per oral paracetamol and ibuprofen for 7 days. Histology of the excised tissue revealed presence of a urothelial tissue with Malpighian metaplasia and unspecific sclero-inflammatory reaction. After 20 months of follow-up, no recurrence nor complication was noted, the physical examination was normal.</p></sec><sec id="s3"><title>3. Discussion</title><p>Congenital paraurethral cyst is a rare congenital malformation of the urinary</p><p>tract and an exceptional etiology of dysuria in girls [<xref ref-type="bibr" rid="scirp.123530-ref4">4</xref>] . It is secondary to obstruction of paraurethral glands, homologous to the male prostate, which drains into the female urethra through their ducts. Skene’s glands are the largest two glands of 6 to 30 that secrete a mucoid material on sexual stimulation into the distal two-thirds of the female urethra [<xref ref-type="bibr" rid="scirp.123530-ref6">6</xref>] . Etiology of the ductal obstruction is still controversial. Advocated hypothesis include: influence of maternal estrogen, stenotic or inflammatory obstruction, and urothelial dislocation [<xref ref-type="bibr" rid="scirp.123530-ref5">5</xref>] .</p><p>During the newborn examination, genitalia examination is mandatory. When properly performed, it allows the diagnosis of this congenital lesion which presents as an interlabial golden mass (due to its milky content), lateral to the urethral meatus, and non-painful [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] . The natural history of CPUC is regression as the child grows due to the reduction of maternal hormones impregnation, as suggested by authors supporting the hormonal hypothesis [<xref ref-type="bibr" rid="scirp.123530-ref7">7</xref>] . No additional investigations are necessary unless the patient presents atypically, such as in our case, where persistence up to 13 years old is exceptional. In such cases, differentials should formally be excluded before attempting any surgical option [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] . One of the possible differentials is prolapsed ectopic uretereterocele, which must be ruled out by urinary tract (UT) US [<xref ref-type="bibr" rid="scirp.123530-ref8">8</xref>] . In our case, no anomaly of the UT was detected by the US. Another possible differential is urethral prolapse, frequently encountered in African and African descent girls, as well as in our context [<xref ref-type="bibr" rid="scirp.123530-ref9">9</xref>] . In our patient, it was ruled out after clinical examination, as the mas was lateral to the urethral meatus. Other causes of interlabial mass must be ruled out: botryoidal rhabdomyosarcoma, imperforated hymen with subsequent hydrocolpos, hymenal cyst, Gartner duct cyst, and urethral polyp or diverticulum [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] [<xref ref-type="bibr" rid="scirp.123530-ref10">10</xref>] .</p><p>Management of CPUC is controversial [<xref ref-type="bibr" rid="scirp.123530-ref7">7</xref>] . Considering its natural history and absence of symptoms, many authors favor conservative management with close monitoring, especially in newborns. In a series of five patients, Japanese authors reported spontaneous regression from 76 to 304 days [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] . However, other authors praise operative management, arguing that interlabial mass in the neonate can be a stressful situation for parents, and its surgical removal guarantees the absence of recurrence [<xref ref-type="bibr" rid="scirp.123530-ref7">7</xref>] . Several operative options include needle aspiration, incisional drainage, marsupialization or surgical excision of the lesion [<xref ref-type="bibr" rid="scirp.123530-ref3">3</xref>] [<xref ref-type="bibr" rid="scirp.123530-ref11">11</xref>] . Optimal management of CPUP should consider both natural history and the possibility of persistence or complications, such as meatal obstruction, which occurred in our patient. Therefore, neonatal and asymptomatic CPUC should be managed conservatively, and persistent or symptomatic lesions should be operatively managed [<xref ref-type="bibr" rid="scirp.123530-ref1">1</xref>] . In our case, persistence until 13 years old and dysuria due to mass effect on the distal urethra and urethral meatus, we chose surgical excision. Outcome of CPUC is excellent, so that no recurrence was reported in the literature, neither after conservative nor surgical management [<xref ref-type="bibr" rid="scirp.123530-ref12">12</xref>] .</p></sec><sec id="s4"><title>4. Conclusion</title><p>Congenital paraurethral cyst is a rare condition. Usually asymptomatic and spontaneously resolving, it can be persistent and lead to dysuria, as shown in our patient. In such cases, surgical excision is the cornerstone of management, with excellent outcomes.</p></sec><sec id="s5"><title>Consent for Publication</title><p>A written consent for publication was obtained from the patient’s parents.</p></sec><sec id="s6"><title>Acknowledgements</title><p>The author FTAZ thanks the NGO F&#246;rderverein Uni Kinshasa (fUNIKIN), which funds his pediatric surgery specialization through the BEBUC excellence scholarship.</p></sec><sec id="s7"><title>Conflicts of Interest</title><p>The authors declare no conflicts of interest regarding the publication of this paper.</p></sec><sec id="s8"><title>Cite this paper</title><p>Gueye, D., Seck, N.F., Zeng, F.T.A., Mbaye, P.A., Well&#233;Ibrahima, B., Ciss&#233;, L., Ndoye, N.A., Sagna, A. and Ngom, G. (2023) Persistent Congenital Paraurethral Cyst, a Rare Cause of Dysuria in a Girl. Open Journal of Urology, 13, 83-87. https://doi.org/10.4236/oju.2023.133010<sup> </sup></p></sec></body><back><ref-list><title>References</title><ref id="scirp.123530-ref1"><label>1</label><mixed-citation publication-type="other" xlink:type="simple">Herek, O., Ergin, H., Karaduman, D., Cetin, O. and Aksit, M.A. (2000) Paraurethral Cysts in Newborn: A Case Report and Review of Literature. European Journal of Pediatric Surgery, 10, 65-67. https://doi.org/10.1055/s-2008-1072327</mixed-citation></ref><ref id="scirp.123530-ref2"><label>2</label><mixed-citation publication-type="other" xlink:type="simple">Soyer, T., Aydemir, E. and Atmaca, E. (2007) Paraurethral Cysts in Female Newborns: Role of Maternal Estrogens. Journal of Pediatric and Adolescent Gynecology, 20, 249-251. https://doi.org/10.1016/j.jpag.2007.04.007</mixed-citation></ref><ref id="scirp.123530-ref3"><label>3</label><mixed-citation publication-type="other" xlink:type="simple">Fathi, K. and Pinter, A. (2003) Paraurethral Cysts in Female Neonates. Case Reports. Acta Paediatrica, 92, 758-759. https://doi.org/10.1111/j.1651-2227.2003.tb00615.x</mixed-citation></ref><ref id="scirp.123530-ref4"><label>4</label><mixed-citation publication-type="other" xlink:type="simple">Centoze, A., Salerno, D., Capillo, S., Mazzei, A. and Stranieri, G. (2019) Skene Gland Cyst in Post Pubertal Girl. Journal of Pediatric Surgery Case Reports, 42, 9-11. https://doi.org/10.1016/j.epsc.2018.12.014</mixed-citation></ref><ref id="scirp.123530-ref5"><label>5</label><mixed-citation publication-type="other" xlink:type="simple">Yayla, D., Demitas, G., Tagci, S., Karabulut, B. and Tiryaki, H.T. (2021) Paraurethral Skene’s Cyst Case in Puberty. Journal of Pediatric Surgery Case Reports, 68, Article ID: 101788. https://doi.org/10.1016/j.epsc.2021.101788</mixed-citation></ref><ref id="scirp.123530-ref6"><label>6</label><mixed-citation publication-type="other" xlink:type="simple">Tseng, M.H. and Lin, S.H. (2016) Interlabial Mass in a Neonate with Paraurethral Cyst. World Journal of Pediatrics, 1, Article No. 121. https://doi.org/10.1007/s12519-015-0051-1</mixed-citation></ref><ref id="scirp.123530-ref7"><label>7</label><mixed-citation publication-type="other" xlink:type="simple">Konecki, T., Salagierski, M. and Sosnowski, M. (2009) Treatment of Paraurethral Cysts in Female Patients-Description of Tree Cases. Central European Journal of Urology, 62, 111-113. https://doi.org/10.5173/ceju.2009.02.art13</mixed-citation></ref><ref id="scirp.123530-ref8"><label>8</label><mixed-citation publication-type="other" xlink:type="simple">Diabate, I. and Sow, I. (2011) Diverticule de l’urètre féminin contenant de multiples calculs. Progrès en Urologie, 21, 229-232. https://doi.org/10.1016/j.purol.2010.04.002</mixed-citation></ref><ref id="scirp.123530-ref9"><label>9</label><mixed-citation publication-type="other" xlink:type="simple">Seck, N.F., Ndoye, N.A., Zeng, F.T.A., Diop, M.B., Cissé, L., Wellé, I.B., et al. (2022) Conservative Management of Urethral Prolapse in African Girls: A Report of 15 Cases. African Journal of Urology, 28, Article No. 26. https://doi.org/10.1186/s12301-022-00293-8</mixed-citation></ref><ref id="scirp.123530-ref10"><label>10</label><mixed-citation publication-type="other" xlink:type="simple">Mekhimer, M.I., Mandour, A.H., Selim, S.S. and Wahba, A.H. (2021) A Rare Interlabial Mass in a 9-Month-Old Infant: A Case Report and Review of Literature. Journal of Pediatric and Adolescent Gynecology, 34, 415-417. https://doi.org/10.1016/j.jpag.2021.01.008</mixed-citation></ref><ref id="scirp.123530-ref11"><label>11</label><mixed-citation publication-type="other" xlink:type="simple">Mi&amp;#241;o, A. and Rodríguez, J. (2014) Skene Duct Cyst in Female Newborn: Case Reports. Revista Chilena de Pediatria, 85, 584-587.</mixed-citation></ref><ref id="scirp.123530-ref12"><label>12</label><mixed-citation publication-type="other" xlink:type="simple">Vatopoulou, A. and Roos, E. (2019) Paraurethral Cyst in a Newborn: Case Report and Discussion. Journal of Pediatric and Adolescent Gynecology, 32, 639-640.</mixed-citation></ref></ref-list></back></article>