A retrospective study on imperforate hymen and haematometrocolpos in a regional hospital

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1 Hong Kong Journal of Emergency Medicine A retrospective study on imperforate hymen and haematometrocolpos in a regional hospital CT Lui, TWT Chan, HT Fung, SYH Tang Objective: To find out the characteristics and presentations of imperforate hymen in the local population in Hong Kong and to assess if diagnosis of imperforate hymen made in the emergency department can reduce time to operation and length of hospital stay. Design: Retrospective study. Setting: A regional public hospital in Hong Kong. Patients: We retrospectively collected data of all patients with the diagnosis of imperforate hymen (ICD 9 coding ) from the period of January 1999 to June Demographics of the patients, their presenting symptoms and signs, the diagnostic process, investigation findings and time of operation were recorded and analysed. Results: Fifteen cases of imperforate hymen were reported during that period. All were adolescent girls aged from 10 to 15 years. A total of 13 patients presented to the accident and emergency department (AED). Seven patients had the diagnosis made in the AED and 2 patients received bedside pelvic ultrasound. More than half of them (8 patients) presented with acute retention of urine. Other presentations included lower abdominal pain, constipation, lower abdominal mass, and protruding introital mass. Most cases diagnosed in the AED (5 out of 7) were admitted to the gynaecology ward while the others were admitted to the surgical or urology wards. Five out of the 7 cases diagnosed in the AED received operation within 24 hours; whereas only 1 out of the 6 cases with the diagnosis made after admission had operation within 24 hours. The difference was statistically significant (p=0.035, Fisher's exact test). The mean length of stay of the group diagnosed in the AED was 1.9 days while the mean length of stay for the group diagnosed after admission was 4.2 days. Conclusion: Early diagnosis of imperforate hymen and haematometrocolpos in the AED for adolescent girls with primary amenorrhoea could have positive impact on proper admission to the gynaecology ward, prompt operation and shorter length of stay in hospital. (Hong Kong j.emerg.med. 2010;17: ) Correspondence to: Lui Chun Tat, MBBS(HK), MRCSEd Tuen Mun Hospital, Accident and Emergency Department, Tsing Chung Koon Road, Tuen Mun, N.T., Hong Kong ectlui@yahoo.com.hk Chan Wai To, Total, MBBS(HK) Fung Hin Tat, FHKAM(Emergency Medicine) Tang Yiu Hang, Simon, FHKAM(Emergency Medicine)

2 436 Hong Kong j. emerg. med. Vol. 17(5) Nov p= Keywords: Hematocolpos, hematometra, hymen, ultrasonography, urinary retention Introduction Imperforate hymen is a rare congenital anomaly, with an incidence of about 1 in 2000 female births. 1 It creates a challenge for diagnosis by emergency physicians as the presentations are usually non-specific with lower abdominal pain or retention of urine in pubescent girls. We reviewed the presentations of the cases of imperforate hymen and analysed on whether correct diagnosis in the emergency department was associated with earlier operation, shorter length of stay and probably less morbidity. Methods This was a single-centre retrospective study on imperforate hymen in a regional pubic hospital in the recent 10 years. The intra-hospital electronic database was searched using the diagnosis of imperforate hymen (International Classification of Diseases, Ninth edition [ICD-9] coding ) and cases with such diagnosis made in the hospital from January 1999 to June 2009 were identified. Fifteen relevant cases were retrieved. Clinical details were retrieved from both electronic medical records and written case records. We collected the demographics of these patients, their presenting symptoms and signs as well as details including the diagnostic process, investigation findings, time of operation and length of hospital stay. Fisher's exact test and Mann Whitney U test were used for binary and continuous variables respectively in the statistical analysis. A p-value less than 0.05 would be regarded as significant. Results The age and clinical presentations of the cases of imperforate hymen are shown in Table 1. All of them were adolescent girls. The age of presentation ranged from 10 to 15 years with mean age of 12. Thirteen out of the 15 cases presented to the accident and emergency department (AED). For the presentation symptoms and signs, 8 (53%) patients presented as acute retention of urine. Other presentations included lower abdominal pain (9 patients) and constipation (3 patients). Physical examination findings included lower abdominal mass (3 patients), but more than half were noted to have a protruding introital mass (9 patients). In other words, acute urinary retention, lower abdominal pain and protruding introital mass were the most common presentations of patients with imperforate hymen attending the emergency department. Table 2 showed the assessment in the AED and wards, whether operation was performed within 24 hours, the length of stay as well as other findings during investigation. Among the 13 patients who presented to the AED, 7 patients (54%) had the diagnosis made in the AED with 2 of them received bedside pelvic ultrasound examination. Most of the cases with the diagnosis made in the AED were admitted to gynaecology wards (5 out of 7) while most of the cases diagnosed after admission were initially admitted to surgical wards. Among the 7 cases with the diagnosis made in the AED, 5 of them received hymenotomy within 24 hours; whereas only 1 out of the 6 cases with the diagnosis made after admission had operation done within the same time frame. The difference was

3 Lui et al./imperforate hymen and haematometrocolpos 437 Table 1. Cases of imperforate hymen and their clinical presentations Case no. Age Attended AROU Abdominal Protruding Lower abdominal Constipation AED pain introital mass mass 1 11 Yes Yes No Yes No No 2 11 Yes No Yes Yes No No 3 12 Yes Yes No No No No 4 11 Yes Yes No No No No 5 13 Yes No Yes No No No 6 12 Yes Yes No Yes No No 7 13 No No No No No No 8 10 Yes No Yes Yes No No 9 12 Yes Yes Yes No No Yes No No No Yes No No Yes Yes Yes No Yes No Yes No Yes Yes Yes Yes Yes Yes Yes Yes No Yes Yes No Yes Yes No No Yes Yes Yes Yes Yes No AED=accident and emergency department; AROU=acute retention of urine. Table 2. Cases of imperforate hymen with management parameters and associated urogenital anomalies Case no. Diagnosis USG in Ward Operation LOS Associated Remarks made in AED initially within (days) urogenital AED admitted 24 hours anormalies 1 No No Surgery No 2 2 No No Surgery No 2 Septated vagina 3 No No Surgery No 11 Bifid left Delayed diagnosis with pelvicalyceal system cystoscopy and IVU for AROU 4 No No Surgery Yes 1 Recurrent AROU 5 No No Surgery No 6 Laparoscopy done for abdominal pain to review haematometrocolpos 6 Yes No Gynaecology Yes Bilateral vaginal Presented with vaginal cysts discharge. Found to have imperforate hymen with pyometra 8 Yes No Surgery No 1 Right renal agenesis 9 No No Urology No Presented with hypo/amenorrhoea 11 Yes Yes Gynaecology Yes 3 12 Yes No Gynaecology Yes 1 13 Yes No Urology No 1 Transferral to another hospital 14 Yes No Gynaecology Yes 2 15 Yes Yes Gynaecology Yes 3 AED=accident and emergency department; AROU=acute retention of urine; IVU=intravenous urography; LOS=length of stay; USG=ultrasonography of abdomen and pelvis.

438 Hong Kong j. emerg. med. Vol. 17(5) Nov 2010 statistically significant (p=0.035, Fisher's exact test). Case 4 presented with recurrent urinary retention before the diagnosis was made.

Shorter hospital stay was also observed in the group with the diagnosis made in the AED (1 to 3 days, mean length of stay 1.

4 438 Hong Kong j. emerg. med. Vol. 17(5) Nov 2010 statistically significant (p=0.035, Fisher's exact test). Case 4 presented with recurrent urinary retention before the diagnosis was made. Case 3 had delayed diagnosis after investigations including intravenous urography and cystoscopy. Case 5 even received laparoscopy, which is invasive and probably unnecessary. Shorter hospital stay was also observed in the group with the diagnosis made in the AED (1 to 3 days, mean length of stay 1.9 days) compared to the group with the diagnosis made after admission (1 to 11 days, mean length of stay 4.2 days). With the small sample size, statistical significance could not be demonstrated due to lack of power (p=0.25, Mann Whitney U test). All cases presenting to the emergency department were operated within the same admission except case 13 who was transferred to another hospital for operation. Therefore, successful diagnosis in the AED was highly correlated with correct admission to gynaecology wards instead of surgical (including urology) wards, and subsequently resulting in earlier operation and shorter length of hospital stay. could be made on clinical grounds, assisted by bedside ultrasonography. Imperforate hymen is the most common genital tract malformation in female, with an estimated incidence of about 1 in 2000 female births. 1 However, the exact incidence and prevalence remained uncertain. McCann et al reported an incidence up to 1.2% in a descriptive study. 2 The hymen is a mesodermal remnant at the confluence of the urogenital sinus and the Müller's duct, that normally perforates during the embryological development. Failure of the hymen to rupture during the perinatal period results in For the 2 cases that had bedside pelvic sonogram performed in the AED, both presented with acute urinary retention and lower abdominal mass. Ultrasonography found a huge cystic echogenic pelvic mass besides the distended bladder (Figures 1 and 2) which represented the fluid-distended uterus and vagina (haematometrocolpos). Bedside renal ultrasound was grossly unremarkable. Both cases were admitted to the gynaecology ward with operation done on the same day. The length of stay in hospital for them was 3 days. Figure 1. Pelvic ultrasound prior to urinary catheterization (transverse view). Discussion Our study was the first published local report to correlate the diagnosis of imperforate hymen in the AED with the destination of admission, time to operation and length of stay in hospital. We demonstrated that correct diagnosis in the AED was associated with admission to the gynaecology ward that subsequently could lead to earlier operation, reduction of unnecessary invasive investigations and shorter length of stay in hospital. The diagnosis Figure 2. Pelvic ultrasound after urinary catheterization (transverse view).

5 Lui et al./imperforate hymen and haematometrocolpos 439 imperforate hymen. The girl usually remains clinically quiescent until the age of puberty. When the girl starts to menstruate, the menstrual blood is collected inside the vagina (haematocolpos), the uterus (haematometra), or up to the fallopian tube (haematosalpinx), to cause pressure effects and obstructive symptoms. There have been rare occurrences of familial clusters 3-5 though most of the cases are sporadic. The inheritance of mullerian defects is likely polygenic and multifactorial. No specific mode of inheritance has been found in familial clusters and both recessive and dominant inheritances have been reported. Imperforate hymen can be diagnosed at any age by inspection of the external genitalia. In some parts of the world there is bimodal distribution of age at presentation. Most cases of imperforate hymen are symptomatic only at the pubertal age and more morbidity would be noted in the group with delayed diagnosis. 6 The clinical presentation can be primary amenorrhoea, recurrent periodic abdominal pain, 7 acute urinary retention, 8-11 or constipation. 12 Obstructive and irritative urinary symptoms such as urinary hesitancy and dysuria are common and occur in up to 58% of haematocolpos. 8 Sciatica, low back pain and lumbar radiculopathy have been documented in case reports and irritation of the sacral plexus or nerve roots is postulated as the mechanism. The presentation can also be acute abdomen caused by rupture of a haematosalpinx and haemoperitoneum. 17 Imperforate hymen presenting as acute urinary retention is well reported in the literature, and in a study on the causes of acute urinary retention in children, the incidence of imperforate hymen was found to be 10.7%. 11 Therefore, emergency physicians should have a high index of suspicion of imperforate hymen as a cause for pubescent girls presenting with acute retention of urine. On physical examination, the classical findings are lower abdominal tenderness with a pelvic mass, associated with a bluish bulging mass through the introitus. Digital rectal examination may reveal an extrinsic mass corresponding to the vagina. The girl usually has attained Tanner stage 3 pubertal development with absence of menarche. 18 The differential diagnoses of a pubescent girl presenting with primary amenorrhea, lower abdominal pain and a pelvic mass include transverse vaginal septum, longitudinal vaginal septum, vaginal agenesis and cervical atresia. 19 Imperforate hymen can be differentiated from low type transverse vaginal septum by the Valsalva manoeuvre, in which bulging of the hymen should occur with imperforate hymen but not with septated vagina. The diagnosis can be aided by imaging such as ultrasonography and magnetic resonance imaging (MRI). The diagnosis of haematometrocolpos can be assisted by transabdominal, transvaginal or transperineal sonography. Transrectal sonography has been advocated by some gynaecologists as an alternative route for the diagnosis. 20,21 Associated mullerian malformations such as mullerian agenesis, obstructing vaginal septa, unicornuate uterus, bicornuate uterus, uterus didelphys and septated uterus should be looked for during sonography and further imaging such as MRI may be necessary. Screening renal sonography should be performed to detect any associated renal malformation such as renal agenesis or duplication of the renal system. With the increasing availability and expertise of ultrasonography in emergency departments, diagnosis of imperforate hymen can be made promptly by the findings of a cystic pelvic mass representing fluiddistended vagina and uterus (haematometrocolpos). The content of the cystic mass may be homogeneous dense fluid or heterogeneous content with blood clots or infection. There may also be associated hydrosalpinx. In the same session, examination of the kidneys should also be performed to screen for hydronephrosis, agenesis or other obvious associated urological tract malformation. Conclusion Imperforate hymen and haematocolpos should be suspected in adolescent girls with primary amenorrhoea presenting with lower abdominal pain, acute urinary

6 440 Hong Kong j. emerg. med. Vol. 17(5) Nov 2010 retention, pelvic mass, constipation or low back pain. Physical examination and bedside ultrasonography is the key to correct diagnosis in the emergency department that bears positive impacts on the patient including proper admission to gynaecology wards, prompt operation and shorter length of hospital stay. References 1. Parazzini F, Cecchetti G. The frequency of imperforate hymen in northern Italy. Int J Epidemiol 1990;19(3): McCann J, Wells R, Simon M, Voris J. Genital findings in prepubertal girls selected for nonabuse: a descriptive study. Pediatrics 1990;86(3): Lim YH, Ng SP, Jamil MA. Imperforate hymen: report of an unusual familial occurrence. J Obstet Gynaecol Res 2003;29(6): Stelling JR, Gray MR, Davis AJ, Cowan JM, Reindollar RH. Dominant transmission of imperforate hymen. Fertil Steril 2000;74(6): Usta IM, Awwad JT, Usta JA, Makarem MM, Karam KS. Imperforate hymen: report of an unusual familial occurrence. Obstet Gynecol 1993;82(4 Pt 2 Suppl): Posner JC, Spandorfer PR. Early detection of imperforate hymen prevents morbidity from delays in diagnosis. Pediatrics 2005;115(4): Kumar K, Waseem M. An uncommon cause of abdominal pain in an adolescent. South Med J 2008; 101(10): Chircop R. A case of retention of urine and haematocolpometra. Eur J Emerg Med 2003;10(3): Adali E, Kurdoglu M, Yildizhan R, Kolusari A. An overlooked cause of acute urinary retention in an adolescent girl: a case report. Arch Gynecol Obstet 2009;279(5): Chang JW, Yang LY, Wang HH, Wang JK, Tiu CM. Acute urinary retention as the presentation of imperforate hymen. J Chin Med Assoc 2007;70(12): Asgari SA, Mansour Ghanaie M, Simforoosh N, Kajbafzadeh A, Zare A. Acute urinary retention in children. Urol J 2005;2(1): Wang W, Chen MH, Yang W, Hwang DL. Imperforate hymen presenting with chronic constipation and lumbago: report of one case. Acta Paediatr Taiwan 2004; 45(6): Letts M, Haasbeek J. Hematocolpos as a cause of back pain in premenarchal adolescents. J Pediatr Orthop 1990;10(6): London NJ, Sefton GK. Hematocolpos. An unusual cause of sciatica in an adolescent girl. Spine 1996;21 (11): Deathe AB. Hematometra as a cause of lumbar radiculopathy. A case report. Spine 1993;18(13): Harrison CS. Hematocolpos as a cause of low-back pain. A case report. Spine 1991;16(8): Bakos O, Berglund L. Imperforate hymen and ruptured hematosalpinx: a case report with a review of the literature. J Adolesc Health 1999;24(3): Master-Hunter T, Heiman DL. Amenorrhea: evaluation and treatment. Am Fam Physician 2006;73(8): Burgis J. Obstructive Müllerian anomalies: case report, diagnosis, and management. Am J Obstet Gynecol 2001;185(2): Kushnir O, Garde K, Blankstein J. Rectal sonography for diagnosing hematocolpometra. A case report. J Reprod Med 1997;42(8): Anguenot JL, Ibecheole V, Salvat J, Campana A. Hematocolpos secondary to imperforate hymen, contribution of transrectal echography. Acta Obstet Gynecol Scand 2000;79(7):614-5.

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