Letter to the Editor
First case of Moebius–Poland syndrome in child prenatally exposed to misoprostol
Primer caso de síndrome de Moebius-Poland en niño expuesto prenatalmente a misoprostol
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Pachajoa, C. Isaza" "autores" => array:2 [ 0 => array:4 [ "nombre" => "H." "apellidos" => "Pachajoa" "email" => array:1 [ 0 => "harrympl@yahoo.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "C." "apellidos" => "Isaza" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Departamento de Ciencias Básicas Médicas, Programa de Medicina, Universidad Icesi, Cali, Colombia" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Facultad de Salud, Universidad del Valle, Cali, Colombia" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Primer caso de síndrome de Moebius-Poland en niño expuesto prenatalmente a misoprostol" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 877 "Ancho" => 600 "Tamanyo" => 63759 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Note the bilateral facial paralysis, a typical characteristic of Moebius syndrome.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Moebius syndrome (OMIM <a href="omim:157900">157900</a>) is characterised by facial paralysis with impairment of ocular abduction. The facial nerve (cranial nerve VII) and the abducens nerve (cranial nerve VI) are the ones most commonly involved, but other cranial nerves can also be affected. The phenotype is variable and may include congenital, orofacial and limb defects.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">The Poland syndrome (OMIM <a href="omim:173800">173800</a>) is characterised by the presence of unilateral brachysyndactyly and ipsilateral aplasia of the sternal portion of the pectoralis major muscle. It is sometimes called Poland sequence, as it was initially described in Poland.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The combination of Moebius and Poland syndrome is rarely seen. It has been estimated to occur in 1 in 500,000 people; in the literature we reviewed, we did not find this association with cases of prenatal exposition to misoprostol.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">We present a report on Moebius and Poland syndrome in a newborn prenatally exposed to misoprostol.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient, who was the son of an 18-year-old mother, was brought to consult for facial dysmorphism. However, on examination he was found to have bilateral facial paralysis, a rounded face, narrow palpebral fissures, cupid's bow lips, arched palate, micrognathia and hypoplasia with absence of pectoralis major nipple (Atelier), as well as proximal syndactyly of second and third fingers and bilateral clubfoot (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). As an important background fact, we found that his mother had used 400<span class="elsevierStyleHsp" style=""></span>g of misoprostol orally and vaginally at 5 weeks of gestation to bring about an abortion, but had had only slight bleeding.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">Misoprostol is a synthetic analogue of prostaglandin E1 approved by drug regulatory agencies in many countries for the prevention and treatment of gastric ulcers associated to the use of non-steroidal anti-inflammatories, due to its gastric acid antisecretory effect. Prenatal exposure to misoprostol has been associated to vascular disruption defects, mainly Moebius sequence and terminal transverse type limb defects.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">The abnormalities in the vascular structure could be secondary to teratogenic effects. Teratogens can act directly by reducing blood flow or blood vessel development, changing anatomy and/or structure.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a> The vascular abnormalities of the right subclavian artery observed in Poland syndrome could be related to a vascular disruption caused by misoprostol during a critical period.<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,8</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">The presentation of a case of Moebius and Poland syndromes associated to prenatal exposure to misoprostol has not been reported. However, this combination of two congenital defects has been reported as being associated to another vascular disruptor (such as cocaine).<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The presentation of these two defects in a same patient provides evidence that both pathologies present similar mechanisms, which are probably those of a vascular disruption.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Defects due to a vascular disruption are structural disturbances in development produced by vascular problems, such as severe intermittent vasoconstriction, abnormal vessel regression during the remodelling of the vascular system, arterial thrombosis or any other phenomenon that produces a lack of O<span class="elsevierStyleInf">2</span>.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The aetiology of Moebius and Poland syndromes is unknown; a possible vascular origin has been suggested for the two pathologies. Hypotheses suggest a transient ischemia, particularly in the vertebral arteries.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">10,11</span></a> Premature obstruction or regression of the terminal arteries of the trigeminal (V) nerve and/or delayed formation of the vertebral basilar system could lead to anomalies in cranial nerve development.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">12</span></a> In children with Moebius syndrome, cerebral necrosis has been reported together with capillary telangiectasia in the midbrain and bridge.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">13</span></a> Disruption of the subclavian artery occurs around week 6 of gestation and is related to Moebius–Poland syndrome; this same phenomenon is related to transversal limb defects and arthrogryposis.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">14</span></a> The disruption phenomenon can be secondary to a blood flow interruption secondary to an arterial spasm during the sensitive embryonic phase.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">15,16</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Pachajoa H, Isaza C. Primer caso de síndrome de Moebius-Poland en niño expuesto prenatalmente a misoprostol. Neurología. 2011;26:502-3.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 875 "Ancho" => 600 "Tamanyo" => 105334 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Note the typical phenotype of Moebius–Poland syndrome, hypoplasia of the chest with clubfoot Atelier.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 877 "Ancho" => 600 "Tamanyo" => 63759 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Note the bilateral facial paralysis, a typical characteristic of Moebius syndrome.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:16 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The spectrum of Moebius syndrome: an electrophysiological study" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "H.T. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 October | 58 | 12 | 70 |
| 2024 September | 75 | 24 | 99 |
| 2024 August | 63 | 12 | 75 |
| 2024 July | 64 | 17 | 81 |
| 2024 June | 64 | 18 | 82 |
| 2024 May | 57 | 29 | 86 |
| 2024 April | 48 | 10 | 58 |
| 2024 March | 86 | 20 | 106 |
| 2024 February | 114 | 47 | 161 |
| 2024 January | 161 | 47 | 208 |
| 2023 December | 174 | 48 | 222 |
| 2023 November | 208 | 46 | 254 |
| 2023 October | 314 | 25 | 339 |
| 2023 September | 103 | 5 | 108 |
| 2023 August | 93 | 13 | 106 |
| 2023 July | 122 | 20 | 142 |
| 2023 June | 117 | 21 | 138 |
| 2023 May | 181 | 23 | 204 |
| 2023 April | 200 | 19 | 219 |
| 2023 March | 213 | 6 | 219 |
| 2023 February | 195 | 8 | 203 |
| 2023 January | 203 | 13 | 216 |
| 2022 December | 149 | 11 | 160 |
| 2022 November | 155 | 27 | 182 |
| 2022 October | 149 | 19 | 168 |
| 2022 September | 172 | 28 | 200 |
| 2022 August | 194 | 29 | 223 |
| 2022 July | 141 | 23 | 164 |
| 2022 June | 144 | 16 | 160 |
| 2022 May | 337 | 16 | 353 |
| 2022 April | 860 | 14 | 874 |
| 2022 March | 941 | 8 | 949 |
| 2022 February | 969 | 5 | 974 |
| 2022 January | 1035 | 9 | 1044 |
| 2021 December | 867 | 16 | 883 |
| 2021 November | 354 | 18 | 372 |
| 2021 October | 146 | 32 | 178 |
| 2021 September | 137 | 26 | 163 |
| 2021 August | 128 | 21 | 149 |
| 2021 July | 111 | 9 | 120 |
| 2021 June | 83 | 15 | 98 |
| 2021 May | 141 | 17 | 158 |
| 2021 April | 308 | 46 | 354 |
| 2021 March | 215 | 13 | 228 |
| 2021 February | 168 | 21 | 189 |
| 2021 January | 258 | 18 | 276 |
| 2020 December | 172 | 13 | 185 |
| 2020 November | 197 | 10 | 207 |
| 2020 October | 142 | 10 | 152 |
| 2020 September | 178 | 9 | 187 |
| 2020 August | 207 | 22 | 229 |
| 2020 July | 175 | 13 | 188 |
| 2020 June | 142 | 16 | 158 |
| 2020 May | 141 | 25 | 166 |
| 2020 April | 142 | 15 | 157 |
| 2020 March | 176 | 14 | 190 |
| 2020 February | 183 | 16 | 199 |
| 2020 January | 173 | 20 | 193 |
| 2019 December | 89 | 20 | 109 |
| 2019 November | 98 | 21 | 119 |
| 2019 October | 109 | 14 | 123 |
| 2019 September | 112 | 15 | 127 |
| 2019 August | 86 | 11 | 97 |
| 2019 July | 206 | 16 | 222 |
| 2019 June | 269 | 32 | 301 |
| 2019 May | 382 | 67 | 449 |
| 2019 April | 238 | 54 | 292 |
| 2019 March | 67 | 14 | 81 |
| 2019 February | 107 | 20 | 127 |
| 2019 January | 147 | 8 | 155 |
| 2018 December | 86 | 8 | 94 |
| 2018 November | 94 | 16 | 110 |
| 2018 October | 91 | 21 | 112 |
| 2018 September | 179 | 31 | 210 |
| 2018 August | 115 | 20 | 135 |
| 2018 July | 123 | 5 | 128 |
| 2018 June | 126 | 15 | 141 |
| 2018 May | 102 | 5 | 107 |
| 2018 April | 87 | 4 | 91 |
| 2018 March | 68 | 10 | 78 |
| 2018 February | 105 | 13 | 118 |
| 2018 January | 61 | 2 | 63 |
| 2017 December | 63 | 4 | 67 |
| 2017 November | 98 | 12 | 110 |
| 2017 October | 108 | 2 | 110 |
| 2017 September | 91 | 16 | 107 |
| 2017 August | 45 | 4 | 49 |
| 2017 July | 72 | 8 | 80 |
| 2017 June | 77 | 23 | 100 |
| 2017 May | 84 | 20 | 104 |
| 2017 April | 58 | 15 | 73 |
| 2017 March | 58 | 20 | 78 |
| 2017 February | 139 | 15 | 154 |
| 2017 January | 41 | 13 | 54 |
| 2016 December | 50 | 15 | 65 |
| 2016 November | 80 | 14 | 94 |
| 2016 October | 106 | 15 | 121 |
| 2016 September | 156 | 9 | 165 |
| 2016 August | 84 | 8 | 92 |
| 2016 July | 89 | 7 | 96 |
| 2016 June | 150 | 27 | 177 |
| 2016 May | 139 | 29 | 168 |
| 2016 April | 117 | 42 | 159 |
| 2016 March | 136 | 40 | 176 |
| 2016 February | 117 | 37 | 154 |
| 2016 January | 76 | 36 | 112 |
| 2015 December | 113 | 32 | 145 |
| 2015 November | 101 | 30 | 131 |
| 2015 October | 112 | 27 | 139 |
| 2015 September | 114 | 49 | 163 |
| 2015 August | 93 | 10 | 103 |
| 2015 July | 126 | 12 | 138 |
| 2015 June | 147 | 3 | 150 |
| 2015 May | 96 | 15 | 111 |
| 2015 April | 70 | 22 | 92 |
| 2015 March | 75 | 13 | 88 |
| 2015 February | 47 | 5 | 52 |
| 2015 January | 26 | 7 | 33 |
| 2014 December | 39 | 11 | 50 |
| 2014 November | 23 | 4 | 27 |
| 2014 October | 33 | 8 | 41 |
| 2014 September | 25 | 7 | 32 |
| 2014 August | 51 | 8 | 59 |
| 2014 July | 54 | 13 | 67 |
| 2014 June | 31 | 2 | 33 |
| 2014 May | 24 | 2 | 26 |
| 2014 April | 17 | 5 | 22 |
| 2014 March | 22 | 7 | 29 |
| 2014 February | 18 | 4 | 22 |
| 2014 January | 27 | 7 | 34 |
| 2013 December | 17 | 2 | 19 |
| 2013 November | 29 | 7 | 36 |
| 2013 October | 54 | 13 | 67 |
| 2013 September | 29 | 6 | 35 |
| 2013 August | 40 | 10 | 50 |
| 2013 July | 13 | 2 | 15 |
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