Fetal and Pediatric Pathology, 34:103–107, 2015 C Informa Healthcare USA, Inc. Copyright ISSN: 1551-3815 print / 1551-3823 online DOI: 10.3109/15513815.2014.970263
ARTICLE
Passage of Decidual Cast Following Poor Compliance with Oral Contraceptive Pill Mokerrum F. Malik,1 Henry Adekola,1 William Porter,2 and Janet M. Poulik2 1 Department of Obstetrics and Gynecology, Hutzel Womens Hospital, Wayne State University School of Medicine, Detroit, MI, USA; 2 Department of Pediatric Pathology, Children’s Hospital of Michigan/Wayne State University School of Medicine, Detroit, MI, USA
Background: Decidual cast describes the spontaneous sloughing of endometrium as an entire piece while retaining the shape of the endometrial cavity. It may be associated with increased serum progesterone levels and must be considered as a differential diagnosis in a patient who passes tissue per vagina while on progesterone containing hormonal contraception. Case: A 13year-old adolescent with a history of menorrhagia since menarche, presented to the pediatric emergency room with worsening abdominal pain and heavy vaginal bleeding stopping her oral contraceptive pill 10 days prior to presentation. Her symptoms resolved spontaneously following passage of tissue per vagina which was later by histopathology to be a decidual cast. Conclusion: Decidual cast is a rare pathological entity that may be a side effect of progesterone-containing hormonal contraceptives, requiring patient education before use. Keywords: adolescent, menorrhagia, progesterone-containing hormonal contraception
INTRODUCTION Menstrual disorders constitute a significant fraction of gynecological morbidity affecting postmenarchal adolescents [1–3]. Menorrhagia accounted for between 5–18% of menstrual disorders in a particular series [3]. Menorrhagia is a manifestation of a myriad of disorders in an adolescent. This includes; hypothalamic-pituitaryovarian(HPO) axis immaturity, HPO axis malfunction secondary to eating disorders, stress, excessive physical activities, drug abuse and tumors of the central nervous system. In addition, hematological disorders, e.g. von Willebrand factor deficiency, hemophilia, Glanzmann thrombasthenia, leukemia, etc. Endocrine disorders, e.g. thyroid disease and congenital adrenal hyperplasia may also present with menorrhagia. Hormonal contraception has been utilized to attenuate and regulate menstrual bleeding in most of these cases to avert anemia. Pregnancy must always be suspected and ruled out whenever an adolescent presents with menorrhagia or any other form of menstrual disorder.
Received 23 August 2014; Revised 22 September 2014; accepted 24 September 2014. Address correspondence to Dr Henry Adekola, Department of Obstetrics and Gynecology, Hutzel Womens Hospital,Wayne State University School of Medicine, 3990 John R Street, Detroit, MI, USA. E-mail: [email protected]
M. F. Malik et al.
Figure 1. Whole fragment of sloughed endometrium.
We report the case of a 13-year-old on oral contraceptive pill due to a history of menorrhagia who presented with a rare side effect of this form of medical management of menorrhagia. CASE A 13-year-old female presented to the Children’s Hospital of Michigan emergency room with heavy vaginal bleeding and abdominal cramping since stopping oral contraceptive pills (OCP) 10 days prior to presentation. She had been on OCP and oral iron supplementation for the last 10 months due to menorrhagia complicated by anemia which was diagnosed a few months after attaining menarche. Hematologic work up prior to commencement of OCP revealed no evidence of hematological disorder. Prior to evaluation by the gynecology team, she passed fragments of “large tissue from her vagina” (Figures 1 and 2) which lead to immediate resolution of presenting symptoms. Physical examination revealed adequate pubertal development for age. She also had normal cardiopulmonary examination findings. Perineum was blood stained, but no active bleeding vaginally. Her hymen was intact. Transabdominal ultrasound showed a revealed no uterine anomaly. Laboratory findings included hemoglobin of 10.8 g/dL, platelet count of 375 000/mm3 and urine pregnancy test was negative. Submitted to pathology were two pink-tan membranous portions of tissues measuring 10.4 × 6.3 × 1.7 cm and 6.8 × 5.7 × 2.3 cm, respectively. Microscopically, the tissue
Figure 2. Micrograph of decidua-like stroma with non-proliferating glands (H&E × 4). Fetal and Pediatric Pathology
Passage of Decidual Cast Following Poor Compliance
consisted of endometrium. The endometrial stroma was partially necrotic but where well preserved and showed sheets of cells with abundant pale cytoplasm with welldefined cell borders. In addition to the presence of non-proliferating glands, these findings were consistent with decidualized endometrium. No fetal parts or chorionic villi were identified DISCUSSION A decidual cast is spontaneously sloughed endometrium, usually whole, which retains the shape of the endometrial cavity [4]. This clinical entity was first described by the Italian anatomic pathologist Giovanni Bauttista Morgagni in the eighteenth century and was originally termed “dysmenorrhea membranacea” [5]. It is a rare clinical presentation with about 20 cases reported in the literature [6–18]. Mostly reported as a side effect of progesterone containing hormonal contraception [6, 12, 14–16, 18], it is also a potential cause of antepartum bleeding in pregnancies with Mullerian anomalies [11, 17, 19, 20]. It has also been described in ectopic pregnancies with the grave potential of mimicking an intrauterine pregnancy in these cases [8–10, 13]. Other potential causes of passage of tissue per vagina in the pediatric age group include fibroepithelial polyp and sarcoma botryroides. Although these two lesions may be distinguished from a decidual cast by gross examination, a histopathology examination is required. The exact etiology of decidual cast is unknown. However, several hypotheses have been proffered. These include: (1) an increase in estrogen and progesterone production, leading to incomplete disintegration of the endometrium, which becomes thickened [21]; (2) abnormal cell–to-cell adhesion activity [5]; (3) inhibition of relaxin by hyperprogestogenic state which in turn prevents the fragmentation of the endometrium [22]; (4) intrauterine infection [23] and (5) excessive decidualization of endometrial stromal cells via transcriptional activation of corticotrophin-releasing hormone in the presence of high progesterone levels [24, 25]. The presence of progesterone is the common denominator in most of the aforementioned hypotheses. This is consistent with all reported cases where the use of hormonal contraception or pregnancy (which also increases serum progesterone significantly) was associated with this clinical finding [6–19]. The literature reports an association between decidual casts and depo medroxyprogesterone acetate (DMPA) exposure in teenagers [12, 16]. The case series by Omar et al. reports three out of six patients were on DMPA prior to expulsion of decidual casts [12]. Also in that case series and in other reports, decidual casts can result from transdermal patch and oral contraception pill use [6, 12, 14, 18] like in our case. The difference here is that unlike other studies where a decidual cast occurred while using progesteronecontaining hormonal contraception; ours was associated with brief stoppage of said medication. Whether this was coincidental is uncertain. It is interesting to note that all reports of decidual cast in association with exposure to progesterone-containing hormonal contraception have been restricted to postmenarchal adolescents [6, 12, 14, 16, 18]. Further research is needed to investigate the restriction of this complication to this age group. Some investigators have recommended discontinuation of the offending drug as passage of decidual casts seems to be an iatrogenic pathology [5]. It is accepted practice to start oral contraceptive therapy in an adolescent with abnormal uterine bleeding if hemoglobin level is
ARTICLE
Passage of Decidual Cast Following Poor Compliance with Oral Contraceptive Pill Mokerrum F. Malik,1 Henry Adekola,1 William Porter,2 and Janet M. Poulik2 1 Department of Obstetrics and Gynecology, Hutzel Womens Hospital, Wayne State University School of Medicine, Detroit, MI, USA; 2 Department of Pediatric Pathology, Children’s Hospital of Michigan/Wayne State University School of Medicine, Detroit, MI, USA
Background: Decidual cast describes the spontaneous sloughing of endometrium as an entire piece while retaining the shape of the endometrial cavity. It may be associated with increased serum progesterone levels and must be considered as a differential diagnosis in a patient who passes tissue per vagina while on progesterone containing hormonal contraception. Case: A 13year-old adolescent with a history of menorrhagia since menarche, presented to the pediatric emergency room with worsening abdominal pain and heavy vaginal bleeding stopping her oral contraceptive pill 10 days prior to presentation. Her symptoms resolved spontaneously following passage of tissue per vagina which was later by histopathology to be a decidual cast. Conclusion: Decidual cast is a rare pathological entity that may be a side effect of progesterone-containing hormonal contraceptives, requiring patient education before use. Keywords: adolescent, menorrhagia, progesterone-containing hormonal contraception
INTRODUCTION Menstrual disorders constitute a significant fraction of gynecological morbidity affecting postmenarchal adolescents [1–3]. Menorrhagia accounted for between 5–18% of menstrual disorders in a particular series [3]. Menorrhagia is a manifestation of a myriad of disorders in an adolescent. This includes; hypothalamic-pituitaryovarian(HPO) axis immaturity, HPO axis malfunction secondary to eating disorders, stress, excessive physical activities, drug abuse and tumors of the central nervous system. In addition, hematological disorders, e.g. von Willebrand factor deficiency, hemophilia, Glanzmann thrombasthenia, leukemia, etc. Endocrine disorders, e.g. thyroid disease and congenital adrenal hyperplasia may also present with menorrhagia. Hormonal contraception has been utilized to attenuate and regulate menstrual bleeding in most of these cases to avert anemia. Pregnancy must always be suspected and ruled out whenever an adolescent presents with menorrhagia or any other form of menstrual disorder.
Received 23 August 2014; Revised 22 September 2014; accepted 24 September 2014. Address correspondence to Dr Henry Adekola, Department of Obstetrics and Gynecology, Hutzel Womens Hospital,Wayne State University School of Medicine, 3990 John R Street, Detroit, MI, USA. E-mail: [email protected]
M. F. Malik et al.
Figure 1. Whole fragment of sloughed endometrium.
We report the case of a 13-year-old on oral contraceptive pill due to a history of menorrhagia who presented with a rare side effect of this form of medical management of menorrhagia. CASE A 13-year-old female presented to the Children’s Hospital of Michigan emergency room with heavy vaginal bleeding and abdominal cramping since stopping oral contraceptive pills (OCP) 10 days prior to presentation. She had been on OCP and oral iron supplementation for the last 10 months due to menorrhagia complicated by anemia which was diagnosed a few months after attaining menarche. Hematologic work up prior to commencement of OCP revealed no evidence of hematological disorder. Prior to evaluation by the gynecology team, she passed fragments of “large tissue from her vagina” (Figures 1 and 2) which lead to immediate resolution of presenting symptoms. Physical examination revealed adequate pubertal development for age. She also had normal cardiopulmonary examination findings. Perineum was blood stained, but no active bleeding vaginally. Her hymen was intact. Transabdominal ultrasound showed a revealed no uterine anomaly. Laboratory findings included hemoglobin of 10.8 g/dL, platelet count of 375 000/mm3 and urine pregnancy test was negative. Submitted to pathology were two pink-tan membranous portions of tissues measuring 10.4 × 6.3 × 1.7 cm and 6.8 × 5.7 × 2.3 cm, respectively. Microscopically, the tissue
Figure 2. Micrograph of decidua-like stroma with non-proliferating glands (H&E × 4). Fetal and Pediatric Pathology
Passage of Decidual Cast Following Poor Compliance
consisted of endometrium. The endometrial stroma was partially necrotic but where well preserved and showed sheets of cells with abundant pale cytoplasm with welldefined cell borders. In addition to the presence of non-proliferating glands, these findings were consistent with decidualized endometrium. No fetal parts or chorionic villi were identified DISCUSSION A decidual cast is spontaneously sloughed endometrium, usually whole, which retains the shape of the endometrial cavity [4]. This clinical entity was first described by the Italian anatomic pathologist Giovanni Bauttista Morgagni in the eighteenth century and was originally termed “dysmenorrhea membranacea” [5]. It is a rare clinical presentation with about 20 cases reported in the literature [6–18]. Mostly reported as a side effect of progesterone containing hormonal contraception [6, 12, 14–16, 18], it is also a potential cause of antepartum bleeding in pregnancies with Mullerian anomalies [11, 17, 19, 20]. It has also been described in ectopic pregnancies with the grave potential of mimicking an intrauterine pregnancy in these cases [8–10, 13]. Other potential causes of passage of tissue per vagina in the pediatric age group include fibroepithelial polyp and sarcoma botryroides. Although these two lesions may be distinguished from a decidual cast by gross examination, a histopathology examination is required. The exact etiology of decidual cast is unknown. However, several hypotheses have been proffered. These include: (1) an increase in estrogen and progesterone production, leading to incomplete disintegration of the endometrium, which becomes thickened [21]; (2) abnormal cell–to-cell adhesion activity [5]; (3) inhibition of relaxin by hyperprogestogenic state which in turn prevents the fragmentation of the endometrium [22]; (4) intrauterine infection [23] and (5) excessive decidualization of endometrial stromal cells via transcriptional activation of corticotrophin-releasing hormone in the presence of high progesterone levels [24, 25]. The presence of progesterone is the common denominator in most of the aforementioned hypotheses. This is consistent with all reported cases where the use of hormonal contraception or pregnancy (which also increases serum progesterone significantly) was associated with this clinical finding [6–19]. The literature reports an association between decidual casts and depo medroxyprogesterone acetate (DMPA) exposure in teenagers [12, 16]. The case series by Omar et al. reports three out of six patients were on DMPA prior to expulsion of decidual casts [12]. Also in that case series and in other reports, decidual casts can result from transdermal patch and oral contraception pill use [6, 12, 14, 18] like in our case. The difference here is that unlike other studies where a decidual cast occurred while using progesteronecontaining hormonal contraception; ours was associated with brief stoppage of said medication. Whether this was coincidental is uncertain. It is interesting to note that all reports of decidual cast in association with exposure to progesterone-containing hormonal contraception have been restricted to postmenarchal adolescents [6, 12, 14, 16, 18]. Further research is needed to investigate the restriction of this complication to this age group. Some investigators have recommended discontinuation of the offending drug as passage of decidual casts seems to be an iatrogenic pathology [5]. It is accepted practice to start oral contraceptive therapy in an adolescent with abnormal uterine bleeding if hemoglobin level is
