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Case report of Pierre Robin sequence with severe upper airway obstruction who was
rescued by fiberoptic nasotracheal intubation
#MMPMID28288578
Takeshita S
; Ueda H
; Goto T
; Muto D
; Kakita H
; Oshima K
; Tainaka T
; Ono T
; Kazaoka Y
; Yamada Y
BMC Anesthesiol
2017[Mar]; 17
(1
): 43
PMID28288578
show ga
BACKGROUND: Pierre Robin sequence (PRS) refers to the association of
micrognathia, glossoptosis, and airway obstruction. Cases with severe dyspnea due
to upper airway obstruction immediately after birth are very rare. We here report
two cases with PRS who developed severe dyspnea due to morphological abnormality
immediately after birth and were rescued by fiberoptic nasotracheal intubation.
CASE PRESENTATION: The patient in case 1 had micrognathia and cleft palate, and
his tongue protruded into the nasal cavity via a cleft palate. His invaginated
tongue was considered an extreme type of glossoptosis and he was diagnosed as
Pierre Robin sequence. The patient in case 2 also had micrognathia and cleft
palate same as case 1 and accompanied some anomalad. Her chromosome analysis
confirmed a diagnosis of 1p36 deletion syndrome and she diagnosed as 1p36
deletion syndrome complicated with Pierre Robin sequence. In both cases, tongue
protruded into the nasal cavity via a cleft palate occupied pharynx and nasal
cavity, resulting in severe dyspnea. Only the backside of the tongue was visible
by laryngoscopy and oropharyngeal intubation was impossible. Therefore,
fiberoptic nasotracheal intubation was done to secure the airway for
resuscitation. CONCLUSION: We conclude that extreme type of glossoptosis in PRS
concludes tongue invaginated into nasal cavity which have not reported before and
that such cases require resuscitation by fiberoptic intubation immediately after
birth. As such, neonatologists should obtain the skill of fiberoptic intubation.
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