Case and Research Letters
Pruritic Primary Nonessential Cutis Verticis Gyrata
Cutis verticis gyrata primario no esencial pruriginoso
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Tomás-Velázquez, E. Moreno-Artero, M. Abengózar, I. Palacios-Álvarez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Tomás-Velázquez" "email" => array:1 [ 0 => "atomasv@unav.es" ] "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" => "E." "apellidos" => "Moreno-Artero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "M." "apellidos" => "Abengózar" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "I." "apellidos" => "Palacios-Álvarez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Departamento de Dermatología Médico-Quirúrgica y Venereología, Clínica Universidad de Navarra, Pamplona, Navarra, España" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Departamento de Anatomía Patológica, Clínica Universidad de Navarra, Pamplona, Navarra, España" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "<span class="elsevierStyleItalic">Cutis verticis gyrata</span> primario no esencial pruriginoso" ] ] "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" => 1059 "Ancho" => 905 "Tamanyo" => 75476 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial magnetic resonance imaging showing slight undulation of the extracranial soft tissue.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The term <span class="elsevierStyleItalic">cutis verticis gyrata</span> (CVG) describes the finding of folds and creases on the top of the scalp, forming patterns reminiscent of those of the surface of the brain.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 25-year-old man who visited our department with pruritis on the scalp that had appeared several months earlier. The patient's past history included immunoglobulin-A (IgA) deficiency, bilateral sensorineural hearing loss, and bilateral diffuse cataracts. Analytical tests and cranial CT scans performed during childhood were normal, and congenital rubella and Alport syndrome were ruled out. The patient's intellectual development and social adaptation were normal.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination at our department revealed hypertrophy and formation of anteroposterior folds on the scalp, forming furrows and ridges (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The patient presented no desquamation, hair loss, or other skin lesions.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Cranial magnetic resonance imaging revealed slight undulation of the extracranial soft tissue, with no associated involvement of the bone or intracranial tissue (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). A skin biopsy of the scalp revealed follicular hypertrophia and hyperplasia, with no other histologic findings of interest (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). Blood tests (blood count, glycemia, lipid, hepatic, renal, and thyroid profiles, cortisol, parathyroid hormone, and acromegaly screening) were normal. In light of these findings, the patient was diagnosed with primary nonessential CVG.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Our patient presented intense pruritis, which had previously been treated with oral antihistamines and topical antipruritics, with no response. Treatment with capsaicin gel 0.075% was instated but the patient's condition worsened; oral gabapentin was then instated with no improvement, and both treatments were therefore suspended. Finally, amitriptyline was prescribed at a dosage of 10<span class="elsevierStyleHsp" style=""></span>mg per day, and the patient remains asymptomatic to date (5 months).</p><p id="par0030" class="elsevierStylePara elsevierViewall">CVG is a rare morphologic syndrome that presents with hypertrophy and folding of the scalp. Incidence in the general population is difficult to estimate, as the disease is generally asymptomatic.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The primary form has no known cause and may occur in isolation (primary essential CVG) or in association with other neurologic (intellectual disability, deafness, microcephaly, or epilepsy) and/or ophthalmologic abnormalities (cataracts, strabismus, blindness, or retinitis pigmentosa).<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a> It is more common in males,<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> with onset usually occurring during puberty, and is usually distributed symmetrically, typically affecting the vertex and occipital region,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> in the form of anteroposterior furrows and ridges that give it the appearance of the surface of the brain.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Secondary CVG is due to the abnormality in the scalp caused by other diseases such as acromegaly, myxedema, fragile X syndrome, Klinefelter syndrome, tuberous sclerosis, psoriasis, eczema, Darier disease, amyloid deposition, extensive nevus or hamartoma, leukemia, Ehlers-Danlos syndrome, or trauma.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1–3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Study of the patient should initially be guided by past history, the physical examination and the biopsy of the affected area, and associated abnormalities and the diseases that cause secondary forms should be ruled out using different tests and imaging studies.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">A typical biopsy of the primary form will reveal hypertrophy and hyperplasia of adnexal structures and increased dermal collagen in skin with no other histologic abnormalities.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Secondary CVG shows abnormalities characteristic of the underlying etiology.</p><p id="par0055" class="elsevierStylePara elsevierViewall">As well as the above-mentioned diseases, the differential diagnosis includes pachydermoperiostosis, Beare-Stevenson cutis gyrata syndrome, and lipedematous scalp. Pachydermoperiostosis is characterized by pachyderma, periostosis, and clubbing of the fingers, and may be associated with CVG.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Beare-Stevenson cutis gyrata syndrome typically presents with craniosynostosis, prominent umbilical cord stump, or acanthosis nigrans, and sometimes presents CVG as a phenotypic trait.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Unlike CVG, lipedematous scalp courses with thickening of the scalp, but this is caused by hyperplasia of the subcutaneous tissue due to proliferation of adipocytes.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Primary CVG is asymptomatic in most cases. If it poses an aesthetic problem, surgical treatment may be performed and different techniques such as partial excision, use of tissue expanders and plasties,<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">7</span></a> or fold subcision<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> have been reported. In secondary CVG, the causal disease is treated.</p><p id="par0065" class="elsevierStylePara elsevierViewall">Higher prevalence of skin disease such as atopic dermatitis has been reported in contexts of IgA deficiency. In the last count, our patient showed IgA levels of 76<span class="elsevierStyleHsp" style=""></span>mg/dL (normal range, 108-325<span class="elsevierStyleHsp" style=""></span>mg/dL) and had no history or clinical signs of atopic dermatitis or other skin disease that explained the pruritis. Furthermore, the IgA deficiency was diagnosed in infancy and the pruritis, located exclusively in the scalp, began when the patient was 25 years old, coinciding with when he first noticed the skin folds at that site.</p><p id="par0070" class="elsevierStylePara elsevierViewall">To date, we have found no cases of pruritic CVG in the literature. We therefore based the treatment of this patient on that described for dysesthesia of the scalp.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> The use of oral antidepressants in patients with chronic pruritis that does not respond to oral antihistamines or topical treatment has been widely reported. While randomized clinical trials are needed, the literature contains case series and open clinical trials in which these drugs have been efficacious.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> Our case joins this group of patients, with an atypical form of CVG that courses with pruritis and responds to amitriptyline – something we have not found in the literature to date.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflicts of Interest</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflicts of Interest" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Tomás-Velázquez A, Moreno-Artero E, Abengózar M, Palacios-Álvarez I. <span class="elsevierStyleItalic">Cutis verticis gyrata</span> primario no esencial pruriginoso. Actas Dermosifiliogr. 2019;110:702–704.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 537 "Ancho" => 1255 "Tamanyo" => 73081 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Anteroposterior folds forming furrows and ridges on the scalp.</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" => 1059 "Ancho" => 905 "Tamanyo" => 75476 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Cranial magnetic resonance imaging showing slight undulation of the extracranial soft tissue.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 933 "Ancho" => 1255 "Tamanyo" => 234832 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Skin biopsy of the scalp showing follicular hypertrophia and hyperplasia, with no other histologic abnormalities of interest.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cutis verticis gyrata" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "D.G. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 11 | 8 | 19 |
| 2024 Octubre | 109 | 53 | 162 |
| 2024 Septiembre | 84 | 23 | 107 |
| 2024 Agosto | 130 | 75 | 205 |
| 2024 Julio | 103 | 27 | 130 |
| 2024 Junio | 137 | 25 | 162 |
| 2024 Mayo | 97 | 28 | 125 |
| 2024 Abril | 96 | 31 | 127 |
| 2024 Marzo | 86 | 27 | 113 |
| 2024 Febrero | 94 | 36 | 130 |
| 2024 Enero | 118 | 30 | 148 |
| 2023 Diciembre | 128 | 19 | 147 |
| 2023 Noviembre | 138 | 54 | 192 |
| 2023 Octubre | 107 | 31 | 138 |
| 2023 Septiembre | 93 | 41 | 134 |
| 2023 Agosto | 86 | 19 | 105 |
| 2023 Julio | 95 | 42 | 137 |
| 2023 Junio | 80 | 35 | 115 |
| 2023 Mayo | 101 | 25 | 126 |
| 2023 Abril | 91 | 26 | 117 |
| 2023 Marzo | 74 | 21 | 95 |
| 2023 Febrero | 112 | 21 | 133 |
| 2023 Enero | 84 | 40 | 124 |
| 2022 Diciembre | 102 | 45 | 147 |
| 2022 Noviembre | 63 | 35 | 98 |
| 2022 Octubre | 51 | 34 | 85 |
| 2022 Septiembre | 23 | 34 | 57 |
| 2022 Agosto | 25 | 38 | 63 |
| 2022 Julio | 19 | 35 | 54 |
| 2022 Junio | 21 | 30 | 51 |
| 2022 Mayo | 104 | 46 | 150 |
| 2022 Abril | 111 | 29 | 140 |
| 2022 Marzo | 92 | 58 | 150 |
| 2022 Febrero | 90 | 35 | 125 |
| 2022 Enero | 91 | 49 | 140 |
| 2021 Diciembre | 78 | 35 | 113 |
| 2021 Noviembre | 58 | 52 | 110 |
| 2021 Octubre | 69 | 48 | 117 |
| 2021 Septiembre | 62 | 35 | 97 |
| 2021 Agosto | 72 | 31 | 103 |
| 2021 Julio | 57 | 42 | 99 |
| 2021 Junio | 64 | 30 | 94 |
| 2021 Mayo | 68 | 35 | 103 |
| 2021 Abril | 166 | 89 | 255 |
| 2021 Marzo | 164 | 30 | 194 |
| 2021 Febrero | 82 | 24 | 106 |
| 2021 Enero | 84 | 13 | 97 |
| 2020 Diciembre | 65 | 12 | 77 |
| 2020 Noviembre | 61 | 15 | 76 |
| 2020 Octubre | 27 | 10 | 37 |
| 2020 Septiembre | 56 | 9 | 65 |
| 2020 Agosto | 56 | 20 | 76 |
| 2020 Julio | 50 | 11 | 61 |
| 2020 Junio | 33 | 24 | 57 |
| 2020 Mayo | 57 | 8 | 65 |
| 2020 Abril | 38 | 9 | 47 |
| 2020 Marzo | 37 | 7 | 44 |
| 2020 Febrero | 7 | 2 | 9 |
| 2019 Octubre | 1 | 0 | 1 |
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