Neurocutaneous melanosis in association with encephalocraniocutaneous lipomatosis
We describe a white female infant with neurocutaneous melanosis (NCM) and encephalocraniocutaneous lipomatosis (ECCL). Multiple, giant and small congenital melanocytic nevi (CMN) were observed on the head, neck and trunk and involved 70% of body surface area. Histologic examination of several CMN re...
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| Veröffentlicht in: | Journal of the American Academy of Dermatology 2002-08, Vol.47 (2), p.S196-S200 |
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| container_title | Journal of the American Academy of Dermatology |
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| creator | Ahmed, Iftikhar Tope, Whitney D. Young, Terri L. Miller, Danielle M. Bloom, Kenneth E. |
| description | We describe a white female infant with neurocutaneous melanosis (NCM) and encephalocraniocutaneous lipomatosis (ECCL). Multiple, giant and small congenital melanocytic nevi (CMN) were observed on the head, neck and trunk and involved 70% of body surface area. Histologic examination of several CMN revealed atypical nodular proliferations of dermal nevomelanocytes. In a small ( |
| doi_str_mv | 10.1067/mjd.2002.110073 |
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Multiple, giant and small congenital melanocytic nevi (CMN) were observed on the head, neck and trunk and involved 70% of body surface area. Histologic examination of several CMN revealed atypical nodular proliferations of dermal nevomelanocytes. In a small (<1 cm) truncal CMN, single and dyscohesive intraepidermal nests of atypical nevomelanocytes simulating a superficial spreading melanoma, were observed. The placenta was grossly normal and histologically demonstrated multiple banal appearing nevomelanocytes within the stroma of its villi. At the 17-month follow-up no evidence of primary or metastatic melanoma was present. This previously undescribed association of NCM, ECCL and placental nevomelanocytes provides strong support for the hypothesized causal role of anomalous neural crest morphogenesis and migration in the development of all three disorders. The genetic mechanism underlying these complex birth defects has been hypothesized to result from the action of lethal autosomal dominant genes surviving by mosaicism. 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Multiple, giant and small congenital melanocytic nevi (CMN) were observed on the head, neck and trunk and involved 70% of body surface area. Histologic examination of several CMN revealed atypical nodular proliferations of dermal nevomelanocytes. In a small (<1 cm) truncal CMN, single and dyscohesive intraepidermal nests of atypical nevomelanocytes simulating a superficial spreading melanoma, were observed. The placenta was grossly normal and histologically demonstrated multiple banal appearing nevomelanocytes within the stroma of its villi. At the 17-month follow-up no evidence of primary or metastatic melanoma was present. This previously undescribed association of NCM, ECCL and placental nevomelanocytes provides strong support for the hypothesized causal role of anomalous neural crest morphogenesis and migration in the development of all three disorders. The genetic mechanism underlying these complex birth defects has been hypothesized to result from the action of lethal autosomal dominant genes surviving by mosaicism. (J Am Acad Dermatol 2002;47:S196-200.)</description><subject>Biological and medical sciences</subject><subject>Dermatology</subject><subject>Eye Diseases - complications</subject><subject>Eye Diseases - pathology</subject><subject>Female</subject><subject>Humans</subject><subject>Infant, Newborn</subject><subject>Lipomatosis - complications</subject><subject>Lipomatosis - pathology</subject><subject>Medical sciences</subject><subject>Melanosis - complications</subject><subject>Melanosis - pathology</subject><subject>Neurocutaneous Syndromes - complications</subject><subject>Neurocutaneous Syndromes - pathology</subject><subject>Pigmentary diseases of the skin</subject><subject>Skin - pathology</subject><subject>Skin Diseases - complications</subject><subject>Skin Diseases - pathology</subject><issn>0190-9622</issn><issn>1097-6787</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2002</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp1kM9LwzAcxYMobk7P3qQXvXX7JmmT5ijiLxiKsHtIk5RltM1MWsX_3o4W5sXTu3ze4_FB6BrDEgPjq2ZnlgSALDEG4PQEzTEInjJe8FM0BywgFYyQGbqIcQcAIqP8HM0wwRlkDM_Rx5vtg9d9p1rr-5g0tlatjy4mrk1UjF471TnfJt-u2ya21Xa_VbXXQbXuT612e9-o7lC8RGeVqqO9mnKBNk-Pm4eXdP3-_Ppwv0415bRLs1zpzPAi14ZUAkOZFZzyitqqwmVBQWAiWEGsMUIXOadlCdaUhrG8hNIAXaC7cXYf_GdvYycbF7Wt6_GR5FhkBBgbwNUI6uBjDLaS--AaFX4kBnmQKAeJ8iBRjhKHxs003ZeNNUd-sjYAtxOgolZ1NcjQLh45WuCcUjJwYuTs4OHL2SCjdgeHxgWrO2m8-_fEL85nj24</recordid><startdate>20020801</startdate><enddate>20020801</enddate><creator>Ahmed, Iftikhar</creator><creator>Tope, Whitney D.</creator><creator>Young, Terri L.</creator><creator>Miller, Danielle M.</creator><creator>Bloom, Kenneth E.</creator><general>Mosby, Inc</general><general>Elsevier</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20020801</creationdate><title>Neurocutaneous melanosis in association with encephalocraniocutaneous lipomatosis</title><author>Ahmed, Iftikhar ; Tope, Whitney D. ; Young, Terri L. ; Miller, Danielle M. ; Bloom, Kenneth E.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c373t-45ac4d785cd2f910b48737f3eff1b8309129682edd9c8573bb0edbd665b0bd03</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2002</creationdate><topic>Biological and medical sciences</topic><topic>Dermatology</topic><topic>Eye Diseases - complications</topic><topic>Eye Diseases - pathology</topic><topic>Female</topic><topic>Humans</topic><topic>Infant, Newborn</topic><topic>Lipomatosis - complications</topic><topic>Lipomatosis - pathology</topic><topic>Medical sciences</topic><topic>Melanosis - complications</topic><topic>Melanosis - pathology</topic><topic>Neurocutaneous Syndromes - complications</topic><topic>Neurocutaneous Syndromes - pathology</topic><topic>Pigmentary diseases of the skin</topic><topic>Skin - pathology</topic><topic>Skin Diseases - complications</topic><topic>Skin Diseases - pathology</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Ahmed, Iftikhar</creatorcontrib><creatorcontrib>Tope, Whitney D.</creatorcontrib><creatorcontrib>Young, Terri L.</creatorcontrib><creatorcontrib>Miller, Danielle M.</creatorcontrib><creatorcontrib>Bloom, Kenneth E.</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of the American Academy of Dermatology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Ahmed, Iftikhar</au><au>Tope, Whitney D.</au><au>Young, Terri L.</au><au>Miller, Danielle M.</au><au>Bloom, Kenneth E.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Neurocutaneous melanosis in association with encephalocraniocutaneous lipomatosis</atitle><jtitle>Journal of the American Academy of Dermatology</jtitle><addtitle>J Am Acad Dermatol</addtitle><date>2002-08-01</date><risdate>2002</risdate><volume>47</volume><issue>2</issue><spage>S196</spage><epage>S200</epage><pages>S196-S200</pages><issn>0190-9622</issn><eissn>1097-6787</eissn><coden>JAADDB</coden><abstract>We describe a white female infant with neurocutaneous melanosis (NCM) and encephalocraniocutaneous lipomatosis (ECCL). Multiple, giant and small congenital melanocytic nevi (CMN) were observed on the head, neck and trunk and involved 70% of body surface area. Histologic examination of several CMN revealed atypical nodular proliferations of dermal nevomelanocytes. In a small (<1 cm) truncal CMN, single and dyscohesive intraepidermal nests of atypical nevomelanocytes simulating a superficial spreading melanoma, were observed. The placenta was grossly normal and histologically demonstrated multiple banal appearing nevomelanocytes within the stroma of its villi. At the 17-month follow-up no evidence of primary or metastatic melanoma was present. This previously undescribed association of NCM, ECCL and placental nevomelanocytes provides strong support for the hypothesized causal role of anomalous neural crest morphogenesis and migration in the development of all three disorders. The genetic mechanism underlying these complex birth defects has been hypothesized to result from the action of lethal autosomal dominant genes surviving by mosaicism. (J Am Acad Dermatol 2002;47:S196-200.)</abstract><cop>New York, NY</cop><pub>Mosby, Inc</pub><pmid>12140461</pmid><doi>10.1067/mjd.2002.110073</doi></addata></record> |
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| source | MEDLINE; ScienceDirect Journals (5 years ago - present) |
| subjects | Biological and medical sciences Dermatology Eye Diseases - complications Eye Diseases - pathology Female Humans Infant, Newborn Lipomatosis - complications Lipomatosis - pathology Medical sciences Melanosis - complications Melanosis - pathology Neurocutaneous Syndromes - complications Neurocutaneous Syndromes - pathology Pigmentary diseases of the skin Skin - pathology Skin Diseases - complications Skin Diseases - pathology |
| title | Neurocutaneous melanosis in association with encephalocraniocutaneous lipomatosis |
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