Twin pregnancy: Live fetus with complete hydatidiform molar—A case report

Abstract

Gestational trophoblastic disease encompasses a range of lesions, each culminating in varied clinical manifestations. Among these, the simultaneous occurrence of molar changes within the placenta and a viable fetus remains a particularly elusive and challenging presentation, posing intricate dilemmas for obstetricians. In this article, we delineate a distinct case involving a 21-year-old woman with a history of consanguineous marriage. She was referred from Médecins Sans Frontières to the Rafic Hariri University Hospital during her 19th week of gestation. With an unremarkable medical history and uncomplicated previous pregnancies, her presentation to the Rafic Hariri University Hospital was punctuated by symptoms of premature labor. Clinical evaluations revealed ruptured membranes, a fetus-positioned breech, and discernible complications tied to the placental morphology. Given these clinical findings, a decision was made in favor of a Cesarean delivery. Prior to this, the patient had been administered corticosteroids and tocolytics. Subsequent delivery produced a live male infant at 34 + 5 gestational days, and further assessment unveiled a complete molar pregnancy. The coexistence of a viable fetus alongside a complete molar pregnancy is an infrequently documented phenomenon, underlining the clinical significance of this case. A comprehensive literature survey reinforced the exceptional nature of our findings. Postpartum, both the mother and neonate were monitored for a month, ensuring their well-being. This case accentuates the paramountcy of prompt and precise diagnostic interventions when faced with atypical presentations. It further underscores the imperative for obstetricians to possess nuanced expertise in confronting the intricate challenges posed by gestational trophoblastic disease, ensuring optimal maternal and neonatal outcomes.

Introduction

Twin gestations, characterized by the simultaneous presence of a viable fetus and a molar pregnancy, manifest as a particularly exceptional subset of molar pregnancies in Lebanon. Their incidence, ranging between 5 and 10 in every 100,000 pregnancies, underscores their rarity. Deriving from the dispermic fertilization of a standard haploid oocyte, these cases result in a triploid zygote containing a trio of chromosomal sets, meriting profound medical scrutiny.^1,2

We present an intriguing case centered on a 21-year-old gravida, referred to Rafic Hariri University Hospital (RHUH) during her 19th gestational week. Diagnostic evaluations ascertained the presence of a hydatidiform mole concomitant with a live fetus. The salience of this presentation is amplified by factors including the patient’s relatively young age, the strategic clinical management employed, and the auspicious outcome circumventing the need for intensive care nursery intervention.

This account serves as an instructive paradigm, shedding light on the judicious management of analogous clinical scenarios. It accentuates the pivotal role of meticulous prenatal care coupled with astute diagnostic acumen in orchestrating triumphant clinical outcomes. Furthermore, it advocates for an economically judicious approach, emphasizing the viability of alternative therapeutic strategies in addressing the complexities inherent in such pregnancies.

Through this exposition, our intent is to augment awareness and comprehension of this uncommon condition within the clinical realm. A heightened cognizance can expedite timely diagnostic endeavors and interventions, optimizing the prognostic trajectory for both maternal and neonatal outcomes. We delve deeply into the nuances of this case, juxtaposing our findings with prevailing literature, and elucidating pivotal takeaways instrumental in enriching the comprehension of such esoteric twin gestations.

To encapsulate, while instances of twin gestations featuring a coexistent molar pregnancy and a live fetus are not entirely unprecedented, each elucidation enriches our collective clinical repository. Our discourse augments the existing literature corpus, championing the tenets of bespoke patient care, avant-garde management paradigms, and an elevated cognizance of such rarified clinical presentations.

Case report

A 21-year-old woman, Gravida 4, Para 1, Abortion 2, spontaneously conceived within a consanguineous marriage. Apart from her two abortions, she did not report other obstetric complications in her past pregnancies, and there was no history of twin or molar pregnancies.

Initial diagnosis and referral

The patient was directed to our esteemed center at RHUH from Medecins Sans Frontieres (MSF) during her 19th week of gestation. At this juncture, a diagnostic lapse transpired: the concurrent molar pregnancy was overlooked, resulting in an erroneous interpretation as a twin gestation. During her first trimester, she manifested symptoms congruent with hyperemesis gravidarum, notably pronounced nausea and vomiting, but no episodes of vaginal hemorrhage were reported (Figure 1).

Figure 1.

Abdominal circumference of the fetus at 19 + 5 weeks.

Clinical findings

Physical examination

• Vital Signs:

• • Temperature: 37°C

• • Pulse rate: 89 bpm

HEENT:

• Head: Normocephalic, atraumatic. Absence of trauma or neoplastic masses.

• Eyes: Pupils symmetrically round, reactive to both light and accommodation. Conjunctivae pristine; sclerae unblemished. Absence of icterus.

• Ears: Unblemished tympanic membranes. No exudate or inflammatory signs.

• Nose: Absence of nasal discharge; septum intact.

• Throat: Oropharynx moist without erythema or exudates. Non-hypertrophied tonsils.

Thyroid Gland:

• Inspection: Absence of overt enlargement or neoplastic masses.

• Palpation: Gland exhibited no tenderness, smooth in texture, devoid of nodules or masses. Dimensions are within standard parameters for age and gender.

Chest: We cannot do X-RAY because she is a pregnant patient.

Cardiovascular System: (physical exam)

• Inspection: Absence of visible cardiac pulsations or heaves.

• Palpation: Regular cardiac rhythm; absence of thrills.

• Auscultation: Heartbeat regular in rate and rhythm. No auscultated murmurs or extraneous sounds.

Abdomen:

Inspection:

The abdomen prominently features a gravid uterus, consistent with the stated gestational age.

• Palpation:

The abdominal contour is supple and non-tender. The presence of the fetus is palpable, with no other masses discerned.

• Leopold’s Maneuver:

The fetus is identified in a cephalic presentation, with no additional unusual findings noted.

• Auscultation:

Bowel sounds are normoactive, and the fetal heart tones are distinct and regular.

• Genitourinary System with Digital Vaginal Examination:

• • Vaginal Examination: Intact cervix, absence of active hemorrhage or aberrant discharge. Uterine dimensions are in line with gestational age.

Clinical impression

Upon evaluation, the patient exudes a state of general well-being without visible signs of distress. The thyroid gland, both upon inspection and palpation, did not yield any abnormalities. Given her gravid state, radiological imaging for thyroid assessment remains contraindicated, owing to potential fetal risks. The findings from the physical examination are corroborated by laboratory assessments that indicated thyroid function within standard parameters. While continuous monitoring is imperative, especially in light of the ultrasonographic findings, the thyroid currently does not pose any immediate concerns.

Multicystic placenta

• By the 26th week, she showed signs of premature labor. Nifedipine 10 mg was prescribed, and regular blood pressure monitoring was initiated. After the 28th week, corticosteroids were administered to aid fetal lung maturity.

• At 34 + 4 weeks of gestation, preterm labor was induced due to an untreated urinary tract infection (UTI). A pelvic ultrasound revealed two fused placentas: one multicystic and another thinner placental tissue, with indications of intrauterine growth retardation (IUGR). The serum β-HCG level at 34 weeks was 26,500 mIU/ml.

Management

Post the 28-week gestation, the patient was administered corticosteroids to enhance fetal lung maturation, foreseeing a potential premature delivery. By 34+4 weeks of gestation, owing to an unresolved UTI, induction of preterm labor became necessary. For expert management, she was subsequently admitted to our esteemed institution. A pivotal pelvic ultrasound at this time depicted a convoluted picture: a potential confluence of two placentas—one conspicuously thick and multicystic, spanning 10.7 cm, punctuated with cystic spaces and a venous lake, juxtaposed against another more svelte placental tissue (kindly refer to Figure 2(a) and (b)). In addition, this imaging intimated compromised fetal dimensions, suggestive of IUGR. Concurrently, the serum β-HCG level, recorded at 34 weeks, stood at 26,500 mIU/ml.

Figure 2.

(a) Anterior nonprevia, breech presentation, fetus with normal amniotic fluid. (b) Two fused placentas: one normal and one molar.

Intervention and outcomes

In light of the neonate’s breech presentation, an elective cesarean section was adjudged as the optimal mode of delivery. The procedure culminated in the birth of a male neonate, weighing 2100 g, boasting an APGAR (Appearance, Pulse, Grimace response, Activity, Respiration) score of 9/10 at the inaugural minute, which marginally tapered to 8/10 at the 5-minute mark. ³ After his birth, he was promptly relocated to the neonatal unit for surveillance. Histological examination of the surgically retrieved placenta unveiled a dual confluence of structures, manifesting discrete attributes; notably, one exhibited vesicles teeming with myriad grape-like cystic formations (kindly refer to Figure 2(a) and (b)). Consistently across her gestational journey, the patient’s hemoglobin indices remained stable, registering a modest post-surgical uptick from 7.9 to 8.2. Therapeutically, she has prescribed a regimen encompassing three ampules of Venofer.

One fused normal placenta with molar

After the delivery, the patient’s recuperation trajectory was uncomplicated, devoid of any manifestations of postpartum hemorrhage. The immediate post-surgical β-hCG concentration registered a value of 167,230 mIU/ml. Propitiously, the neonate’s clinical status remained steadfastly stable, obviating the requirement for neonatal intensive care unit (NICU) intervention. Sonographic evaluation, undertaken 24 h postpartum, authenticated the absence of residual placental or molar tissue (Figure 5).

Histopathological examination

Placental histology delineated two juxtaposed disks, each elucidating distinct morphological attributes. The inaugural disk, congruent with third-trimester morphological norms, manifested well-defined chorionic villi, an impeccable membrane, complemented by a robust umbilical cord. Conversely, the secondary disk evinced a pronounced macro-vesicular configuration, emblematic of a quintessential complete molar pattern. This disk’s villi were conspicuously hypertrophied, engendering a salient “bunch of grapes” visual cue, attributable to edematous changes. Pronounced trophoblastic hyperplasia was evident, with trophoblasts delineating a lucid circumferential contour around the vesicles. A supranormal trophoblastic cell count was also discerned. The conspicuous void of a vascular core in numerous villi, combined with cisternogenesis and the disproportionate trophoblastic proliferation devoid of concomitant fetal vasculature, reinforced the diagnosis of a definitive complete molar gestation (Figures 3 and 4).

Figure 3.

Two placentas one normal and other molar.

Figure 4.

Two placentas one normal and other molar.

Figure 5.

The newborn after 20 days from delivery.

Throughout the postnatal tenure, the patient was subjected to meticulous surveillance, chiefly facilitated via weekly urinary β-hCG evaluations, all of which transpired uneventfully. During the subsequent postnatal review, the patient was subjected to an array of diagnostic assays, encompassing renal function tests, liver function tests, complete blood count, chest radiography, and a coagulation profile, all of which yielded results within normalcy parameters. A β-hCG assay, executed a week post-delivery, delineated a concentration of 3572 mIU/ml.

Management oversights and derived insights

1. Elusive IUGR diagnosis: Despite hints of suboptimal fetal dimensions during the pelvic ultrasonography at 34 + 4 gestational weeks, the accurate identification of IUGR remained evasive. Such diagnostic imprecision could gravely influence clinical approaches, accentuating the paramountcy of assiduous evaluations for early IUGR identification.

2. Anemia oversight: Notwithstanding regular hemoglobin monitoring during gestation, a postoperative ascent from 7.9 to 8.2 underscores a potential deficiency in anemia management. A more assertive iron supplementation strategy or alternative therapeutic avenues might have further harmonized her hemoglobin concentrations, amplifying her physiological well-being throughout gestation.

3. Preliminary misreading: An initial discernment erroneously conflated a coexistent molar pregnancy with a twin gestation. Such diagnostic ambiguity underscores the imperative for scrupulous differentiation between molar and twin pregnancies, ensuring tailored clinical management.

4. Unaddressed UTI: The precipitous induction of preterm labor at 34 + 4 gestational weeks was consequential to an unmitigated UTI, underscoring the indispensability of prompt infectious ailment diagnosis and intervention, particularly to avert complications like preterm labor onset.

5. Surveillance and postpartum oversight: While the consistent β-hCG level monitoring and exhaustive postpartum review were praiseworthy, this scenario underscores the requisites for an exhaustive approach to preemptively detect and address potential gestational complications.

Central to navigating this multifaceted case was the pivotal role of patient autonomy and preference. The patient’s proclivity for a more conservative trajectory, coupled with her aversion to aggressive interventions, necessitated clinical adaptability, particularly when she demurred from certain advised diagnostic procedures.

Centrality of patient-oriented care

The need to recalibrate the therapeutic strategy based on the patient’s clinical choices underscored the imperativeness of adaptable, patient-centric care. This emphasized the essentiality of fostering an environment of transparent communication, ensuring that the patient remained apprised of potential clinical ramifications. Striving to synchronize medical advisories with the patient’s informed choices facilitated a synergistic decision-making paradigm.

Balancing the patient’s conservative inclinations against the intricate case complexity was a challenging endeavor. Nevertheless, it imparted salient lessons on the quintessence of personalized, patient-centric care, particularly when navigating intricate and atypical clinical scenarios.

Discussion

Gestational trophoblastic disease encompasses an expansive array of pathologies, dichotomized into benign entities and malignant perturbations. The benign milieu incorporates both partial and complete moles. While molar pregnancies are infrequent, they present distinct clinical conundrums. They can culminate in vesicular proliferation within the uterus, potentially impeding fetal maturation and compromising endometrial and placental structures. In divergence from triploid partial molar pregnancies, which emanate from dispermy, complete molar pregnancies predominantly exhibit a diploid nature. Deriving from the fecundation of an enucleated oocyte by a spermatozoon, they often culminate in gestational attrition. The present case under scrutiny delineates the synchronous presence of a complete mole alongside a viable fetus—a phenomenon with an incidence oscillating between 1 in 22,000 and 1 in 100,000 gestations.^1,4

Historically, gestations with molar pathology were routinely curtailed due to inherent risks. Nonetheless, contemporary medical innovations have broadened therapeutic avenues, especially when conjoined with a living fetus. Employing ultrasonography—a linchpin for prodromal detection—our patient’s anomaly was discerned at the 19th gestational week. Disregarding the pronounced hyperemesis gravidarum and elevated β-hCG titers, she was devoid of quintessential molar gestational manifestations.

Hemida et al. ⁵ delved into prospective repercussions, underscoring perils such as hemorrhagic episodes, pre-eclampsia, spontaneous gestational termination, and fetal aberrations. Their elucidations, showcasing a pronounced 71.4% prematurity incidence, dovetail with our clinical narrative, culminating in delivery at the 34th gestational week.

Giorgione et al. ⁶ unraveled ultrasonographic misinterpretations, elucidating that a significant cohort initially diagnosed with subchorionic hematoma subsequently exhibited a complete mole in tandem with a live fetus—paralleling the inferences drawn by Piura et al. ⁷

Lin et al. ⁸ articulated on postpartum emergence of gestational trophoblastic neoplasia, linking its manifestation more intrinsically with gestational complications and heightened β-hCG titers than elective gestational cessation. In juxtaposition, Yayna et al. ⁹ delineated a case besieged by hyperthyroidism—a stark divergence from our episode where neonatal NICU intervention was superfluous, and maternal thyroidal equilibrium persisted.

Zilberman Sharon et al. ¹⁰ and Farazestanian et al. ¹¹ proffered insights resonating with our observations, while Uemura et al. ¹² and Loza et al. ¹³ extolled the virtues of avant-garde diagnostic modalities.

Cases as delineated by Krishnamoorthy et al. ¹⁴ and Tariq et al. ¹⁵ further enrich the compendium of gestational trophoblastic anomalies.

Sheik et al. ¹⁶ delved deeper into the complexities surrounding twin gestations wherein a complete hydatidiform mole coexists with a viable fetus, highlighting the inherent rarity and concomitant challenges of such presentations.

Conclusion

This singular report—centering on a 21-year-old female presenting with an amalgamation of a complete molar gestation and a concomitant viable fetus—underscores the labyrinthine intricacies intrinsic to the management of such esoteric obstetric phenomena. The juxtaposition of two placental disks of disparate nature accentuates the intricate duality of this ailment. Favorable prognoses in such instances, as evinced here, vindicate the primacy of prompt clinical interventions, assiduous oversight, and bespoke care regimens that resonate with the patient’s unique requisites.

Considering the preliminary diagnostic oversight and the intricate nature of such gestational presentations, we promulgate an intensified focus on sophisticated ultrasonographic tutelage. This is paramount for the nascent detection and differentiation of molar gestations vis-à-vis twin gestations. Discerning the nuanced differentials between these conditions is instrumental in fortifying optimal maternal–fetal prognoses. By tackling these diagnostic conundrums and instituting tailored clinical blueprints, we aspire to augment the extant literary corpus on managing coexisting molar gestations and viable fetuses.