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Case report
Scaphoid–trapezium hereditary coalition. A case report
Coalición carpiana escafotrapezoidea hereditaria. A propósito de un caso
L. García-Lamas
Corresponding author
lorenylla@hotmail.com

Corresponding author.
, M.Á. Porras-Moreno, V. Jiménez-Díaz
Servicio de Cirugía Ortopédica y Traumatología, Hospital Universitario 12 de Octubre, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Carpal coalitions are caused by synostosis or synchondrosis of 2 or more carpal bones&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Their incidence in the adult population varies from 0&#46;1&#37; to 9&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The few references to this condition show that complete coalitions are more common between the semilunate and pyramidal bones &#40;SP&#41;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> and the capitate and hamate bones&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Coalitions between the scaphoid and trapezium bones &#40;ST&#41; are very rare&#46; The majority of series contain one or no cases&#44; and when they do contain one&#44; they are often associated with hereditary syndromes or other coalitions&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">We present the case of a patient with asymptomatic bilateral hereditary ST diagnosed following fracture of the distal radius&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 17 year-old man &#40;patient A&#41; with a history of asthma visited the Emergency Department of our hospital to evaluate a distal radius fracture caused by a chance fall 2 weeks beforehand&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The first X-ray images showed a 12&#176; dorsal angle and an 18&#176; radial inclination &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>a&#41; of the distal end of the radius&#46; Open physes were observed&#44; as well as an increase in the scapholunate space&#46; Given the time that had passed since the fracture&#44; computed tomographic imaging was used to evaluate the existence of a fracture callus prior to attempting the closed reduction of the same &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>b&#41;&#46; The report confirmed the diagnosis of type II epiphysiolysis according to Salter&#39;s classification&#46; No signs of consolidation were detected and a 3<span class="elsevierStyleHsp" style=""></span>mm scapholunate space was found&#44; together with a dorsal angle of 11&#176; and a 17&#176; radial inclination and extension of the semilunate bone respecting the scaphoid&#46; No liquid was found in the joint and no other findings are mentioned&#46; An attempt at closed reduction of the fracture was made on the basis of this information&#44; without achieving any clear improvement in radiological measurements&#46; The possibility of surgery was then raised&#44; together with a fluoroscopic test to evaluate the scapholunate space&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">After reviewing the tomographic images during preoperative planning a coalition was detected between the scaphoid and trapezium bones&#46; Taking this finding into account it was decided to take a comparative X-ray image &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>c&#41; of the contralateral wrist&#44; which showed the same findings&#58; ST coalition with an increase in the scapholunate space and semilunate bone in extension&#46; The patient was completely asymptomatic in the contralateral wrist and had no symptoms prior to the fall&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Given these findings and the lack of improvement in radiological parameters after reduction&#44; the decision was taken to apply orthopaedic treatment of the fracture&#44; removing the plaster cast after 5 weeks &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>d&#41;&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Results</span><p id="par0040" class="elsevierStylePara elsevierViewall">After removing the plaster cast the patient had an immediate range of movement of 60&#176; dorsal flexion&#44; 40&#176; volar flexion and complete pronosupination&#46; The patient had no pain in the site of the fracture and the external appearance of the wrist was very similar to that of the contralateral one&#44; with no visible deformity&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Taking into account the possibility that this is a hereditary coalition the decision was taken to take X-ray images of both hands of the patient&#39;s parents&#46; In the case of the mother &#40;patient B&#41; the same type of coalition was found in different degrees&#46; On the right side it was a synchondriosis and on the left side it consisted of a synostosis without an increased scapholunate space &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; The patient was clinically asymptomatic&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Discussion</span><p id="par0050" class="elsevierStylePara elsevierViewall">Carpal coalitions are relatively common entities&#44; and in the bibliography their incidence is said to vary from 0&#46;1&#37; to 9&#37;&#44; and they are more frequent in individuals of black races&#46;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#8211;4</span></a> They may be associated with syndromes such as multiple congenital arthrogryposis&#44; foetal alcohol syndrome or Turner&#39;s syndrome&#44;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> among others&#46; In these cases coalitions may occur between more than 2 carpal bones&#46; Cases associated with other bone malformations have been described&#44; such as Madelung&#39;s disease or proximal radiocubital synostosis&#44; although in the majority of cases it occurs without any association to other complaints&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">The most widely accepted aetiology for the origin of the abnormality is a fault in differentiation during weeks 4&#8211;8 of pregnancy&#44;<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1&#44;2</span></a> giving rise to incomplete separation of the cartilaginous precursors&#46; Most authors agree that the term &#8220;fusion&#8221; should be abolished from the attempt to explain the origin of this complaint&#44; given that it may only be used in acquired cases&#46; In the case we present&#44; an X-ray image taken 3 years before the trauma shows 2 differentiated ossification nuclei for the scaphoid and trapezium which subsequently fused&#44; giving rise to this ST coalition&#46; Other possible etiologies are trauma&#44; inflammatory arthropathies and even the metaplastic conversion of mesoderm derivatives&#44; as in the case of coalition between the pyramidal and pisiform bones&#46; All of these causes are less frequent in this type of anatomical abnormality&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Carpal coalitions were described for the first time by Sandifort in 1779&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> There are not many references in the bibliography to this entity&#46; The longest series published to date is the one by DeFazio et al&#46;&#44;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> with a total of 103 cases in 85 individuals&#44; 18 of which are bilateral&#46; The most frequent coalition is SP&#44; with 92 cases&#44; while only 2 were between the scaphoid and trapezium&#44; neither of which was bilateral&#46; Van Hoorns et al&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> described a total of 98 cases&#44; 85 between the capitate and hamate bones&#59; none of the coalitions described in this study were of the ST&#44; as occurred in our clinical case&#46; Delaney and Estar<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> mention a total of 36 cases in which the most frequent coalition was the SP&#44; with no case of ST coalition&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Coalition between the scaphoid and trapezium is of the intercarpal type&#44; given that it occurs between bones in both rows of carpal bones&#46; This is far less common than coalition between bones in the same row&#46; We only found 2 other references&#44; apart from those cited&#44; to this type of coalition in the bibliography&#58; the first of these refers to a unilateral case&#59;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> the second describes a bilateral case<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> with increased scapholunate space&#46; In this second case the mother was also affected&#44; with the same bilateral coalition but without increased scapholunate space&#46; This agrees with data that show intercarpal fusion is genetically transmissible&#44; dominant autosomic Mendelian inheritance unlinked to sex having been proven&#46;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">The most widely used classification of carpal coalitions in the bibliography is the one described by DeVilliers Minnaar<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> for the SP coalition&#44; and this has been extended to cover other types&#46; This classification divides them into 4 types&#44; depending on the type of fusion between the bones and other associated carpal abnormalities&#46; Although this classification has been widely used&#44; it is not easy to apply because of the difficulty in detecting the degree of ossification and associated alterations&#46; Due to these problems other classifications have been created&#44; such as the one by Sing et al&#46;&#44;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> which is very descriptive but contains many subtypes&#44; making it hard to use&#46; Lastly&#44; the classification described by Burnett<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> divides coalitions into bony and non-bony&#44; making it simpler to use in clinical practice&#46; According to this classification&#44; patient B has a bilateral ST carpal coalition of two different types&#46; Her right hand has a non-bone coalition that is detectable in oblique projection due to the irregular margin separating both bones&#44; with the chief differentiating diagnosis of arthrosis at this level&#46; Her left hand has a bone coalition with clear continuity between both bones&#46; There is no other published case in the bibliography of bilateral ST coalition in different degrees in the same patient&#46;</p><p id="par0075" class="elsevierStylePara elsevierViewall">To conclude&#44; it is essential to underline the importance of taking comparative X-ray images when we detect rare findings in a unilateral X-ray image or when&#44; as in this case&#44; a distance is detected between the semilunate and scaphoid bones which seems abnormal&#46; Performing studies of this type helps us to reach a correct diagnosis and therefore select the most suitable treatment for our patients&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Level of evidence</span><p id="par0080" class="elsevierStylePara elsevierViewall">Level of evidence <span class="elsevierStyleSmallCaps">v</span>&#46;</p></span></span>"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Carpal coalitions are relatively frequent between bones in the same carpal row&#46; Coalitions between bones of different rows&#44; however&#44; are very uncommon&#46; We present a case of bilateral intercarpal coalition between the trapezium and scaphoid as an incidental finding&#44; confirming its hereditary origin since a parent has the same bilateral coalition&#46;</p></span>"
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        "titulo" => "Resumen"
        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Las coaliciones carpianas son relativamente frecuentes entre huesos de la misma hilera del carpo&#46; Son mucho m&#225;s infrecuentes las coaliciones entre huesos de diferente hilera&#46; Presentamos un caso de coalici&#243;n intercarpiana entre trapecio y escafoides bilateral como hallazgo casual&#44; confirmando su origen hereditario ya que uno de los progenitores presenta la misma coalici&#243;n&#46;</p></span>"
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        "etiqueta" => "&#9734;"
        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Garc&#237;a-Lamas L&#44; Porras-Moreno M&#193;&#44; Jim&#233;nez-D&#237;az V&#46; Coalici&#243;n carpiana escafotrapezoidea hereditaria&#46; A prop&#243;sito de un caso&#46; Rev Esp Cir Ortop Traumatol&#46; 2018&#59;62&#58;467&#8211;470&#46;</p>"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Patient A&#58; &#40;a&#41; initial X-ray images showing the fracture and increased scapholunate space&#59; &#40;b&#41; computed tomography image showing the scaphoid&#8211;trapezium coalition&#59; &#40;c&#41; comparative X-ray images of the right wrist&#59; &#40;d&#41; final X-ray images of the patient&#46;</p>"
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                            0 => "T&#46;J&#46; Delaney"
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                      "titulo" => "Carpal coalitions on radiographs&#58; prevalence and association with ordering indication"
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                      "titulo" => "Bilateral scaphotrapezium&#8211;trapezoid coalition with bipartite scaphoid&#46; Case report"
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                            0 => "D&#46;A&#46; Stewart"
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                            0 => "H&#46;W&#46; Hey"
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                          ]
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                      "autores" => array:1 [
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Article information
ISSN: 19888856
Original language: English
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos