Primary cutaneous adenoid carcinoma of the scalp

iprs000004 10.3205/iprs000004 urn:nbn:de:0183-iprs0000049 Case Report Primary cutaneous adenoid carcinoma of the scalp Primäres kutanes adenoidzystisches Karzinom der Kopfhaut Keck Keck Meike M

Plastic Surgery Center, Birkenwerder, Germany

author Ueberreiter Ueberreiter Klaus K

Plastic Surgery Center, Birkenwerder, Germany

author Tanzella Tanzella Ursula U

Plastic Surgery Center, Birkenwerder, Germany

author Doll Doll Dietrich D

German Armed Forces Hospital of Berlin, Plastic and Hand Surgery, Department of Surgery, Berlin, Germany

author Krapohl Krapohl Björn Dirk BD Prof. Dr. Dr. MD

German Armed Forces Hospital of Berlin, Plastic and Hand Surgery, Department of Surgery, Scharnhorststraße 13, D-10115 Berlin, Germany, Phone: +49(0)30/2841-1205, Fax: +49(0)30/2841-1299German Armed Forces Hospital of Berlin, Plastic and Hand Surgery, Department of Surgery, Berlin, Germany

PlastChirBWBerlin@web.de author German Medical Science GMS Publishing House

Düsseldorf

610 adenoid carcinoma skin cancer tumor of the scalp adenoidzystisches Karzinom Hauttumor Tumor der Kopfhaut 2012010920160310 engl This is an Open Access article distributed under the terms of the Creative Commons Attribution License. You are free: to Share - to copy, distribute and transmit the work, provided the original author and source are credited. Dieser Artikel ist ein Open Access-Artikel und steht unter den Creative Commons Lizenzbedingungen. Er darf vervielfältigt, verbreitet und öffentlich zugänglich gemacht werden, vorausgesetzt dass Autor und Quelle genannt werden. 2193-8091 1 GMS Interdisciplinary Plastic and Reconstructive Surgery DGPW GMS Interdiscip Plast Reconstr Surg DGPW 04 20160309ISSN added Primäre adenoidzystische Karzinome sind seltene Hauttumoren. Wir stellen eine 75-jährige Patientin mit einem derartigen Tumor der Kopfhaut vor. Weite Exzision unter Einschluss von Teilen der knöchernen Kalotte, Defektdeckung mit einem freien Latissimus-dorsi-Muskellappen und nachfolgende Bestrahlung führten zu einer Verzögerung des Tumorwachstums, konnten jedoch letztlich die weitere Schädelinfiltration nicht verhindern. Primary adenoid carcinoma are rare skin tumors. We present a 75-year-old female with this primary cutaneous tumor of the scalp with additional bone involvement. Wide scalp excision with bone enclosure, latissimus-dorsi-free-flap defect overage, and subsequent radiation slowed down the disease but could not prevent further skull infiltration. Case descriptionA 75-year-old female presented with two nodules of 1.5 cm</PlainText></TextGroup> and 1.0 cm on the scalp in an area of previous excision. A year before the patient’s physician had excised a mass without taking samples for histology (initial situation: Figure 1 <ImgLink imgNo="1" imgType="figure"/>; preoperative state one year after first excision: Figure 2 <ImgLink imgNo="2" imgType="figure"/>). The tumour was indolent and accompanied by focal alopecia. After tumour excision (intraoperative situs: Figure 3 <ImgLink imgNo="3" imgType="figure"/>) histopathological findings showed a firm tumour involving the dermis and subcutaneous fat, consisting of basaloid epithelial cells arranged in cribriform and tubular pattern as well as some focal solid pattern. No periodic acid-Schiff-positive material was found. The resection margins were infiltrated. Perineural invasion was not seen. It was discussed with the pathologist whether the tumour was most likely a distant metastasis or a rare primary cutaneous tumour. </Pgraph><Pgraph>A primary salivary gland origin as well as any other primary origin like breast, thyroid gland, lungs, lacrimal, and mucosal glands was excluded by detailed clinical and radiological investigation (magnet resonance imaging, computer tomography). The remaining tumour was excised with 3 cm margins in width. Considering the clinical background, histological investigations were compatible with a primary cutaneous adenoid cystic carcinoma. Intraoperatively there were signs of cranial bone involvement. Histological investigations showed tumour free margins except for the involvement of the external cortical wall. Another postoperative CT scan showed no further infiltration of the skull. The external cortical wall of the cranial bone was removed (Figure 4 <ImgLink imgNo="4" imgType="figure"/>). A lateral neck dissection was performed and the surgical defect (10x14 cm) was covered with a free latissimus dorsi flap (Figure 5 <ImgLink imgNo="5" imgType="figure"/>) with a split-thickness skin graft (postoperative result: <TextGroup><PlainText>Figure 6 </PlainText></TextGroup><ImgLink imgNo="6" imgType="figure"/>).</Pgraph></TextBlock> <TextBlock linked="yes" name="Discussion"> <MainHeadline>Discussion</MainHeadline><Pgraph>Adenoid cystic carcinoma mainly occurs as a neoplasm of the salivary glands. Other less-frequent primary locations described have been the lacrimal glands, mucosal glands of the upper respiratory tract, external auditory canal, breast, Bartholini’s glands of the vulva, uterine cervix, prostate gland, and the esophagus <TextLink reference="1"></TextLink>. Primary cutaneous adenoid cystic carcinoma (PCACC) is a rare entity with less than 50 cases reported in the literature <TextLink reference="2"></TextLink>. In approximately 40% primary cutaneous adenoid cystic carcinoma arises on the scalp, approximately 18% are found on the skin of the breast <TextLink reference="3"></TextLink>, <TextLink reference="4"></TextLink>, <TextLink reference="5"></TextLink>. The male to female ratio is 1:1.2 <TextLink reference="6"></TextLink>. The natural history of PCACC is a long, indolent course characterized by progressive local recurrence. PCACC is characterized by an aggressive infiltrative growth into the reticular dermis and subcutis with frequent perineural invasion. This leads to local recurrence after tumour excision in more than half of the patients <TextLink reference="6"></TextLink>. Adenoid cystic carcinoma of all sites can invade lymph nodes by direct extension. Embolic metastases are rare <TextLink reference="1"></TextLink>. In PCACC only 3 cases with distant metastasis have been reported. Metastatic focus was the lungs. Histolopathologically, PCACC resembles adenoid castic carcinoma (ACC) of the salivary glands and consists of basaloid neoplastic cells that have inconspicuous cytoplasm and round hyperchromatic nuclei without nuclear atypia <TextLink reference="7"></TextLink>, <TextLink reference="8"></TextLink>. It exhibits cribiform, tubular, cystic, and solid patterns and mainly occupies mid and reticular dermis. The lumina of tubular structures and the surrounding stroma may contain mucin or eosinophilic necrotic cells. A hyaline membrane, which probably represents redublicated basal lamina, may surround the cell islands and tubules and is periodic acid-Schiff-positive <TextLink reference="8"></TextLink>. In our case, no periodic acid-Schiff-positive material was found. In our patient perineural invasion was not seen although described in more than 50% of the previously published cases <TextLink reference="3"></TextLink>, <TextLink reference="6"></TextLink>, <TextLink reference="8"></TextLink>, <TextLink reference="9"></TextLink>. Golden standard for PCACC treatment is still wide local excision with tumour-free margins established by permanent section. It has also been reported that Moh’s surgery is a good opportunity for the excision of PCACC. Lateral neck dissection is recommended <TextLink reference="1"></TextLink>. However, perineural extension may be discontinuous and may lead to false-negative margins with a higher recurrence rate <TextLink reference="3"></TextLink>, <TextLink reference="6"></TextLink>, <TextLink reference="9"></TextLink>, <TextLink reference="10"></TextLink>. Therefore radiation therapy is recommended in addition to the surgical treatment. If subsequent radiation is considered, coverage of the defect with stabile and well vascularised tissue is obligatory. The best way to achieve this is the coverage with a muscle flap. Defects of the scalp can be successfully covered by a free M. latissimus dorsi flap as presented in our case. The pedicle may be anastomosed with temporal vessels or in case of insufficient vessel diameter, as in our case, bigger branches of the external carotoid artery and the jugular veins may be used for flap vascularisation. The muscle flap will then be covered by a split thickness skin graft. After approximately one month when wound healing is completed radiation therapy can be commenced. </Pgraph></TextBlock> <TextBlock linked="yes" name="Notes"> <MainHeadline>Notes</MainHeadline><SubHeadline>Competing interests</SubHeadline><Pgraph>The authors declare that they have no competing interests.</Pgraph></TextBlock> <References linked="yes"> <Reference refNo="1"> <RefAuthor>Weekly M</RefAuthor> <RefAuthor>Lydiatt DD</RefAuthor> <RefAuthor>Lydiatt WM</RefAuthor> <RefAuthor>Baker SC</RefAuthor> <RefAuthor>Johansson SL</RefAuthor> <RefTitle>Primary cutaneous adenoid cystic carcinoma metastatic to cervical lymph nodes</RefTitle> <RefYear>2000</RefYear> <RefJournal>Head Neck</RefJournal> <RefPage>84-6</RefPage> <RefTotal>Weekly M, Lydiatt DD, Lydiatt WM, Baker SC, Johansson SL. Primary cutaneous adenoid cystic carcinoma metastatic to cervical lymph nodes. 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Cutis. 2011;87(5):237-9.</RefTotal> </Reference> </References> <Media> <Tables> <NoOfTables>0</NoOfTables> </Tables> <Figures> <Figure format="png" height="715" width="617"> <MediaNo>1</MediaNo> <MediaID>1</MediaID> <Caption><Pgraph><Mark1>Figure 1: Preoperative finding</Mark1></Pgraph></Caption> </Figure> <Figure format="png" height="715" width="576"> <MediaNo>2</MediaNo> <MediaID>2</MediaID> <Caption><Pgraph><Mark1>Figure 2: Local recurrence</Mark1></Pgraph></Caption> </Figure> <Figure format="png" height="715" width="609"> <MediaNo>3</MediaNo> <MediaID>3</MediaID> <Caption><Pgraph><Mark1>Figure 3: Intraoperativ findings: involvement of the skull</Mark1></Pgraph></Caption> </Figure> <Figure format="png" height="715" width="554"> <MediaNo>4</MediaNo> <MediaID>4</MediaID> <Caption><Pgraph><Mark1>Figure 4: After abrasion of the external cortical wall</Mark1></Pgraph></Caption> </Figure> <Figure format="png" height="715" width="570"> <MediaNo>5</MediaNo> <MediaID>5</MediaID> <Caption><Pgraph><Mark1>Figure 5: Coverage with M. latissimus dorsi free flap</Mark1></Pgraph></Caption> </Figure> <Figure format="png" height="503" width="939"> <MediaNo>6</MediaNo> <MediaID>6</MediaID> <Caption><Pgraph><Mark1>Figure 6: One week and six months after surgery</Mark1></Pgraph></Caption> </Figure> <NoOfPictures>6</NoOfPictures> </Figures> <InlineFigures> <NoOfPictures>0</NoOfPictures> </InlineFigures> <Attachments> <NoOfAttachments>0</NoOfAttachments> </Attachments> </Media> </OrigData> </GmsArticle>