Analysis of Characteristics and Survival of Primary Cutaneous Adenoid Cystic Carcinoma of the Head and Neck

Behbahani, Sara; Wassef, David W.; Povolotskiy, Roman; Pinto, Jessica; Joseph, Nia; Paskhover, Boris

doi:10.1177/0003489420936719

Cited by 6 publications

(9 citation statements)

References 21 publications

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“…Reviewing the literature, only about 450 cases of PCACC have been reported [ 1 , 2 , 3 , 4 , 5 , 6 , 7 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 ], with only two young patients (14 years of age) recorded [ 1 ].…”

Section: Discussionmentioning

confidence: 99%

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A Unique Case of Primary Cutaneous Adenoid Cystic Carcinoma Associated with Aplasia Cutis Congenita in a Four-Year-Old Female: A Case Report

et al. 2022

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Introduction: Primary cutaneous adenoid-cystic carcinoma (PCACC) is a rare malignant tumour reported in only about 450 cases in the literature, with only two adolescent cases reported. PCACC seems to occur between the fifth and seventh decade of life, and the most frequent regions involved are head and neck (46%). Aplasia cutis congenita (ACC) has an incidence of 1:10,000, and it seems to be rarely associated with neoplastic lesions. Interestingly, the association between PCACC and ACC has, so far, never been described. Methods: We report a case of PCACC in the scalp associated with ACC in a four-year-old patient. Discussion: The patient was under follow-up at the dermatology unit, but suddenly a red lesion appeared within the ACC. This red, ulcerated area increased rapidly over six months, so it was surgically removed, and the pathological examination results were suggestive for cribriform PCACC. According to the guidelines for skin tumours, the patient underwent widening resection, and an advancement-sliding skin flap was performed to recreate the scalp. After one year of follow-up, the patient has no local or widespread recurrence of the PCACC, and the surgical scar appears to have healed well. Conclusions: This clinical case is the first known patient with PCACC associated with ACC. A skin excision biopsy should be performed with wide margins to avoid a second widening resection of skin in a similar scenario. Genetic studies may help to identify the origin of this rare association.

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Section: Discussionmentioning

confidence: 99%

“…The standard treatment of PCACC described in the literature is wide surgical excision with at least a 2 cm safety margin from the tumour. Wide excision has the purpose of avoiding frequent recurrence [ 27 ].…”

Section: Discussionmentioning

confidence: 99%

A Unique Case of Primary Cutaneous Adenoid Cystic Carcinoma Associated with Aplasia Cutis Congenita in a Four-Year-Old Female: A Case Report

et al. 2022

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“…Currently, there is no consensus about the appropriate treatment for ACC of the head and neck. The preferred treatment modalities for patients with ACC of the head and neck is surgical excision ( 22 ). Surgery has been widely accepted as a prognostic factor for ACC patients, and the overall survival of ACC patients who receive surgery has been shown to be improved ( 2 , 16 ).…”

Section: Discussionmentioning

confidence: 99%

Prognostic effects of surgery or radiotherapy on adenoid cystic carcinoma of the head and neck: a retrospective cohort study

Zhang¹,

Huang²,

Liu³

et al. 2022

Gland Surg

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Background: There is little evidence exploring prognostic effects of surgery and radiotherapy on adenoid cystic carcinoma (ACC) of the head and neck. This study sought to evaluate the prognostic effects of surgery or radiotherapy on ACC of the salivary gland, oropharynx, and nose, nasal cavity, and middle ear. Methods:In this cohort study, the data of 2,392 participants with ACC of the head and neck were extracted from the Surveillance, Epidemiology and End Results (SEER) database. Participants were divided into the salivary gland group (n=1,351), the mouth and oropharynx group (n=563), and the nose, nasal cavity, and middle ear group (n=478). Baseline characteristics were assessed via questionnaires or laboratory analysis and outcome variables were all-cause death and cancer-specific death of patients. Baseline data were collected in 2004, and patients were followed-up to 2016. The survival time of patients were recorded. Univariate and multivariate Cox regression analyses explored the effects of surgery and radiotherapy on overall prognosis of ACC patients. Fine-Gray test assessed the effects of surgery and radiotherapy on cancer-specific mortality of ACC patients.Results: In total, 766 died and 1,626 survived with a median survival time of 9.92 years. After adjusting for confounders, patients with ACC of the salivary gland who underwent surgery had a decreased risk of all-cause mortality [hazard ratio (HR) =0.51, 95% confidence interval (CI): 0.36-0.71] and cancer-specific mortality (HR =0.57, 95% CI: 0.34-0.97). Surgery was found to be a protective factor for the risk of allcause mortality (HR =0.47, 95% CI: 0.28-0.78) and cancer-specific mortality (HR =0.35, 95% CI: 0.2-0.61) of patients with ACC of the mouth and oropharynx after adjusting for confounders. After adjusting for confounders, patients with ACC of the nose, nasal cavity, and middle ear who underwent surgery had a decreased risk of all-cause mortality (HR =0.46, 95% CI: 0.30-0.70) and cancer-specific mortality (HR =0.35, 95% CI: 0.20-0.61).Conclusions: Surgery was associated with a decreased risk of mortality in patients with ACC of the salivary gland, oropharynx, and nose, nasal cavity, and middle ear, which suggested the value of surgery for improving their prognosis.

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“…Unfortunately, efficacy of this particular technique is uncertain due to paucity of information within the medical literature and low patient numbers. Likewise, there is conflicting advice and evidence in the literature regarding use of adjuvant chemoradiotherapy, with more research needed to clarify whether there is consistent benefit of radiotherapy 16–18…”

Section: Discussionmentioning

confidence: 99%

Primary cutaneous adenoid cystic carcinoma of the neck presenting as a rapidly growing posterior triangle neck mass

2022

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A woman in her 70s presented to a tertiary otorhinolaryngology outpatient department with a 25-year history of right-sided subcutaneous neck lesion that had steadily grown over the preceding 6 months, now with skin involvement. The patient was asymptomatic except for some mild tenderness. The 3 × 3 cm mass lay fixed to deep tissues adjacent to the sternocleidomastoid muscle, though no associated lymphadenopathy was found on palpation, with imaging confirming no regional or distant metastases. Biopsy confirmed the lesion to be primary cutaneous adenoid cystic carcinoma, a malignancy not previously described as a primary on the neck, which was treated by wide local excision after multidisciplinary team discussion. The lesion was completely excised with negative margins, and after surveillance over 3 years, the patient is still well with no signs of recurrence.

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Analysis of Characteristics and Survival of Primary Cutaneous Adenoid Cystic Carcinoma of the Head and Neck

Cited by 6 publications

References 21 publications

A Unique Case of Primary Cutaneous Adenoid Cystic Carcinoma Associated with Aplasia Cutis Congenita in a Four-Year-Old Female: A Case Report

A Unique Case of Primary Cutaneous Adenoid Cystic Carcinoma Associated with Aplasia Cutis Congenita in a Four-Year-Old Female: A Case Report

Prognostic effects of surgery or radiotherapy on adenoid cystic carcinoma of the head and neck: a retrospective cohort study

Primary cutaneous adenoid cystic carcinoma of the neck presenting as a rapidly growing posterior triangle neck mass

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