Successful <i>ex utero</i> intrapartum treatment procedure for prenatally diagnosed severe micrognathia: A case report

Suenaga, Masahiko; Hidaka, Noriaki; Kido, Saki; Otera, Yuka; Fukushima, Kotaro; Kato, Kiyoko

doi:10.1111/jog.12423

Cited by 9 publications

(11 citation statements)

References 11 publications

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“…Clinicians should consider the ex utero intrapartum treatment (EXIT) when a severely hypoplastic mandible accompanied by polyhydramnios and absent stomach visualization is noted on ultrasound. A multidisciplinary team with experienced members should be assembled to perform the procedure [20].…”

Section: Management-outcome-prognosismentioning

confidence: 99%

Focus on Prenatal Detection of Micrognathia

Antonakopoulos

Bhide

2019

Journal of Fetal Medicine

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Fetal micrognathia involves abnormal or arrested development of the fetal mandible. Till recently, the prenatal diagnosis was subjective, based on the evaluation of the fetal profile and assessment of the relationship between the maxilla and the mandible. Recently objective sonographic methods have been utilized for diagnosing micrognathia such as the inferior facial angle, the jaw index, the frontal nasomental angle, the mandible width/maxilla width ratio and the mandibular length. Another useful sonographic sign, the mandibular gap in the retronasal triangle view, increases the accuracy of the diagnosis early in the first trimester. 3D sonographic views can add to the diagnosis and prenatal MRI is a useful adjunct to ultrasound in cases of limited acoustic window, maternal obesity, oligohydramnios and anterior spine position. The identification of micrognathia should prompt karyotyping and sonographic investigation for other abnormalities. The outcome of fetuses with this seemingly isolated finding is more guarded than one would intuitively believe, and the parents should be counseled accordingly. Postnatal complications including mild to severe upper airway obstruction leading to respiratory distress, feeding difficulties and mild to severe long-term developmental delay are common. One should be careful in pronouncing a fetus having 'micrognathia', especially on subjective evaluation, as this term implies that the fetus is abnormal with presence of significant pathology. There is no 'gold standard' for a definitive diagnosis of micrognathia on post-natal evaluation. Using a combination of objective sonographic markers as well as follow-up ultrasound assessments can significantly reduce the risk of a false diagnosis. Follow-up scans should be arranged, and neonatal service should be alerted in cases of ongoing pregnancies.Keywords Fetal mandible Á Micrognathia Á Retrognathia Á Inferior facial angle Á Frontal nasomental angle Á Jaw index Á Mandibular gap Á Chromosomal abnormalities Á Genetic syndromes Á Pierre Robin sequence

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Section: Management-outcome-prognosismentioning

confidence: 99%

Focus on Prenatal Detection of Micrognathia

Antonakopoulos

Bhide

2019

Journal of Fetal Medicine

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“…They can occur in isolation or as part of a sequence such as Pierre Robin sequence or syndromes such as Cornelia de Lange, Stickler, or Treacher-Collins. 10 The airway management associated with micrognathia depends on the severity of obstruction. In mild cases, it can be managed with positional therapy and a nasal airway.…”

Section: Discussionmentioning

confidence: 99%

“…7,8 To date, most of the studies were case reports or small case series with few patients. [9][10][11] With this study, we hope to identify factors that predict a difficult airway at birth and review the perinatal outcomes of patients with antenatally diagnosed micrognathia on fetal imaging.…”

Section: Introductionmentioning

confidence: 99%

Predictive Factors for Perinatal Outcomes of Infants Diagnosed With Micrognathia Antenatally

et al. 2019

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Introduction: Advances in fetal imaging have allowed us to identify abnormalities previously not appreciated. With this study, we hope to identify factors predicting a difficult airway at birth and review the perinatal outcomes of these patients. Methods: Sixteen patients with antenatally diagnosed micrognathia were reviewed from a tertiary care hospital database from 2011 to 2016. Jaw index (JI), amniotic fluid index (AFI), glossoptosis, gastric size, and oropharynx obliteration were assessed. The airway support required at birth, specialist team involvement, and outcomes were evaluated. Results: Nine (56.3%) of 16 patients had JI <5th percentile, 3 (33.3%) of 9 had difficult intubation, 2 (22.2%) of 9 needed an emergency tracheostomy, and 1 (11.1%) of 9 died. Seven patients had polyhydramnios, 2 (28.6%) of 7 had difficult intubation, 2 (28.6%) of 7 required tracheostomy, and 1 (14.3%) of 7 died. Twelve patients had either JI <5th percentile or abnormal AFI, 5 (41.7%) of 12 had difficult intubation, 2 (16.7%) of 12 required tracheostomy, and 1 (8.33%) of 12 died. For the group without otolaryngology consultation, 8 (50%) of 16, 1 (12.5%) of 8 had difficult intubation and 1 (12.5%) of 8 died because airway was not secured after 45 minutes of resuscitation. Conclusion: Jaw index <5th percentile or abnormal AFI predicts a difficult airway. A multidisciplinary approach with otolaryngology involvement for airway intervention may be required at birth.

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“…Forty-one of the included studies, accounting for 129 patients, described the EXIT procedure (12, [160][161][162]. Six studies provided cumulative data on the anesthesia modality and therefore were excluded from the subgroup analysis (21,38,62,68,124,129).…”

Section: Ex-utero Intrapartum Treatmentmentioning

confidence: 99%

“…Fifty-six studies, with a total of 1,127 patients, specified the drugs used to provide general maternal anesthesia (11, 13 [160][161][162]173). The extreme variability between the studies did not allow a specific analysis.…”

Section: Anesthetic Agents For General Maternal Anesthesiamentioning

confidence: 99%

Anesthesia for fetal operative procedures: A systematic review

et al. 2022

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ObjectiveThe anesthetic management of fetal operative procedures (FOP) is a highly debated topic. Literature on fetal pain perception and response to external stimuli is rapidly expanding. Nonetheless, there is no consensus on the fetal consciousness nor on the instruments to measure pain levels. As a result, no guidelines or clinical recommendations on anesthesia modality during FOP are available. This systematic literature review aimed to collect the available knowledge on the most common fetal interventions, and summarize the reported outcomes for each anesthetic approach. Additional aim was to provide an overall evaluation of the most commonly used anesthetic agents.MethodsTwo systematic literature searches were performed in Embase, Medline, Web of Science Core Collection and Cochrane Central Register of Controlled Trials up to December 2021. To best cover the available evidence, one literature search was mostly focused on fetal surgical procedures; while anesthesia during FOP was the main target for the second search. The following fetal procedures were included: fetal transfusion, laser ablation of placental anastomosis, twin-reversed arterial perfusion treatment, fetoscopic endoluminal tracheal occlusion, thoraco-amniotic shunt, vesico-amniotic shunt, myelomeningocele repair, resection of sacrococcygeal teratoma, ligation of amniotic bands, balloon valvuloplasty/septoplasty, ex-utero intrapartum treatment, and ovarian cyst resection/aspiration. Yielded articles were screened against the same inclusion criteria. Studies reporting anesthesia details and procedures’ outcomes were considered. Descriptive statistical analysis was performed and findings were reported in a narrative manner.ResultsThe literature searches yielded 1,679 articles, with 429 being selected for full-text evaluation. A total of 168 articles were included. Overall, no significant differences were found among procedures performed under maternal anesthesia or maternal-fetal anesthesia. Procedures requiring invasive fetal manipulation resulted to be more effective when performed under maternal anesthesia only. Based on the available data, a wide range of anesthetic agents are currently deployed and no consistency has been found neither between centers nor procedures.ConclusionsThis systematic review shows great variance in the anesthetic management during FOP. Further studies, systematically reporting intraoperative fetal monitoring and fetal hormonal responses to external stimuli, are necessary to identify the best anesthetic approach. Additional investigations on pain pathways and fetal pain perception are advisable.

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Successful ex utero intrapartum treatment procedure for prenatally diagnosed severe micrognathia: A case report

Cited by 9 publications

References 11 publications

Focus on Prenatal Detection of Micrognathia

Focus on Prenatal Detection of Micrognathia

Predictive Factors for Perinatal Outcomes of Infants Diagnosed With Micrognathia Antenatally

Anesthesia for fetal operative procedures: A systematic review

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