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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 11 | 8 | 19 |
| 2024 October | 109 | 53 | 162 |
| 2024 September | 84 | 23 | 107 |
| 2024 August | 130 | 75 | 205 |
| 2024 July | 103 | 27 | 130 |
| 2024 June | 137 | 25 | 162 |
| 2024 May | 97 | 28 | 125 |
| 2024 April | 96 | 31 | 127 |
| 2024 March | 86 | 27 | 113 |
| 2024 February | 94 | 36 | 130 |
| 2024 January | 118 | 30 | 148 |
| 2023 December | 128 | 19 | 147 |
| 2023 November | 138 | 54 | 192 |
| 2023 October | 107 | 31 | 138 |
| 2023 September | 93 | 41 | 134 |
| 2023 August | 86 | 19 | 105 |
| 2023 July | 95 | 42 | 137 |
| 2023 June | 80 | 35 | 115 |
| 2023 May | 101 | 25 | 126 |
| 2023 April | 91 | 26 | 117 |
| 2023 March | 74 | 21 | 95 |
| 2023 February | 112 | 21 | 133 |
| 2023 January | 84 | 40 | 124 |
| 2022 December | 102 | 45 | 147 |
| 2022 November | 63 | 35 | 98 |
| 2022 October | 51 | 34 | 85 |
| 2022 September | 23 | 34 | 57 |
| 2022 August | 25 | 38 | 63 |
| 2022 July | 19 | 35 | 54 |
| 2022 June | 21 | 30 | 51 |
| 2022 May | 104 | 46 | 150 |
| 2022 April | 111 | 29 | 140 |
| 2022 March | 92 | 58 | 150 |
| 2022 February | 90 | 35 | 125 |
| 2022 January | 91 | 49 | 140 |
| 2021 December | 78 | 35 | 113 |
| 2021 November | 58 | 52 | 110 |
| 2021 October | 69 | 48 | 117 |
| 2021 September | 62 | 35 | 97 |
| 2021 August | 72 | 31 | 103 |
| 2021 July | 57 | 42 | 99 |
| 2021 June | 64 | 30 | 94 |
| 2021 May | 68 | 35 | 103 |
| 2021 April | 166 | 89 | 255 |
| 2021 March | 164 | 30 | 194 |
| 2021 February | 82 | 24 | 106 |
| 2021 January | 84 | 13 | 97 |
| 2020 December | 65 | 12 | 77 |
| 2020 November | 61 | 15 | 76 |
| 2020 October | 27 | 10 | 37 |
| 2020 September | 56 | 9 | 65 |
| 2020 August | 56 | 20 | 76 |
| 2020 July | 50 | 11 | 61 |
| 2020 June | 33 | 24 | 57 |
| 2020 May | 57 | 8 | 65 |
| 2020 April | 38 | 9 | 47 |
| 2020 March | 37 | 7 | 44 |
| 2020 February | 7 | 2 | 9 |
| 2019 October | 1 | 0 | 1 |
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