Carcinoma cuniculatum of the larynx: a rare clinicopathological entity

Puxeddu, Roberto; Cocco, Daniele; Parodo, Giuseppina; Mallarini, G.; Medda, M. P.; Brennan, Peter A.

doi:10.1017/s0022215107000163

Cited by 24 publications

(25 citation statements)

References 18 publications

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“…CC most often occurs in the skin, but have also been described in other sites, including esophagus [2,3], oral cavity [4,5], gingiva [6], tongue [7], penis [8], larynx [9] and nail [10]. In its pure form this neoplasm has only capacity for local infiltrative growth, but not metastasis [3,8].…”

Section: Discussionmentioning

confidence: 99%

“…The very well differentiated light microscopic features of this tumor led the authors to label it as ''epithelioma cuniculatum''. The most commonly involved site for CC is the skin with a predilection for the sole, but noncutaneous sites, such as esophagus [2,3], oral cavity [4,5], gingiva [6], tongue [7], penis [8], larynx [9] and nail [10] are also on record. CC is histologically typified by deeply invaginating, keratin filled, sinus-like structures which have been likened to rabbit burrows (lat.…”

Section: Introductionmentioning

confidence: 99%

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Carcinoma Cuniculatum of the Esophagus and Tongue: Report of Two Cases, Including TP53 Mutational Analysis

Goh

Venkateswaran

Leow

et al. 2014

Head and Neck Pathol

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Carcinoma cuniculatum (CC) is a rare variant of extremely well differentiated squamous cell carcinoma. We present the clinicopathological features of two cases of CC; one lingual and one esophageal case with a molecular genetic study regarding the TP53 gene mutational status. Case 1 was a 62 year old male with enlarging chronic ulcer in the tongue. Case 2 was a 77 year old male with progressive dysphagia and odynophagia. Both patients were treated surgically. Both tumors showed deeply invaginating, keratin-filled, burrowing crypts lined by very well differentiated squamous epithelium. The esophageal tumor showed varying degrees of reactive nuclear atypia largely limited to the areas with dense intratumoral infiltration of neutrophils. No mutation of TP53 was identified in the esophageal case. Cytologic atypia limited to areas of significant acute inflammation may occur in CC and should, in the absence of aggressive stromal invasion, not preclude a diagnosis of CC.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Carcinoma Cuniculatum of the Esophagus and Tongue: Report of Two Cases, Including TP53 Mutational Analysis

Goh

Venkateswaran

Leow

et al. 2014

Head and Neck Pathol

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“…Only two cases of CC have been previously reported in the larynx, both showing locally aggressive disease but a favorable prognosis after surgery. One was a 72-year-old man who interestingly presented similarly to our patient with multiple 'abscesses' of the preepiglottic and prelaryngeal soft tissue, but also extensive thyroid cartilage involvement; this patient was disease free 70 months after surgery [5]. The other was a 63-year-old man with extensive involvement of the right hemilarynx with subglottic extension with no evidence of disease 42 months following surgery [9].…”

Section: Discussionmentioning

confidence: 92%

“…Although similarities exist between CC and VC, and the literature occasionally uses the terms interchangeably, there are some differences, and the 2005 edition of the WHO Pathology and Genetics of Head and Neck Tumours includes CC as a separate variant of squamous cell carcinoma [3]. The etiology of CC is not clear but some of the proposed risks include chronic trauma, Human Papillomavirus infection, alcohol, and tobacco [4,5].…”

Section: Introductionmentioning

confidence: 99%

A Rare Case of Carcinoma Cuniculatum Involving the Larynx in Association with a Saccular Cyst

Griffith

Goodstein

Tuluc

et al. 2015

Head and Neck Pathol

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Carcinoma cuniculatum (CC), a rare, well-differentiated variant of squamous cell carcinoma, is uncommon in head and neck sites but when it does occur is most common in the oral cavity. Here we report a rare case of CC involving the larynx. A 49-year-old man presented with 10 months of worsening hoarseness and, despite multiple biopsies, no diagnosis of malignancy could be established. Eventual partial excision of the lesion and histologic review of prior specimens confirmed the diagnosis of CC. Focally, a transition to respiratory epithelium indicated the presence of an associated saccular cyst. Total laryngectomy was performed and 6 months later the patient is free of disease. Only two prior cases of CC have been reported in the larynx. Diagnosis of CC is challenging given the low grade histologic features and awareness of this entity for both treating physicians and the pathologist is important to reach a diagnosis of malignancy. This case highlights the challenges in diagnosis of CC, especially in unusual locations and when associated with other lesions such as a saccular cyst. Awareness of this rare tumor type combined with close communication between treating clinicians, radiologists and pathologists should allow earlier diagnosis and treatment.

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“…1 Subsequently, it was described in both cutaneous 2,3 and non-cutaneous sites such as the oral mucosa and larynx. 4,5 Carcinoma cuniculatum has a distinctive morphology characterized by burrowing channels lined by extremely well-differentiated squamous epithelium. 3 We previously reported nine cases of esophageal carcinoma cuniculatum diagnosed on esophagectomy specimens in seven men and two women during a 20-year period.…”

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confidence: 99%

Semiquantitative histologic evaluation improves diagnosis of esophageal carcinoma cuniculatum on biopsy

et al. 2013

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Carcinoma cuniculatum, a unique variant of well-differentiated squamous cell carcinoma, has been only rarely reported in the esophagus. The present study was undertaken to determine if a previously observed common histologic pattern for carcinoma cuniculatum is diagnostically useful in esophageal mucosal biopsy specimens. Thirty-five esophageal mucosal biopsies obtained from 25 procedures in 11 patients with a resection-proven diagnosis of carcinoma cuniculatum were compared with 92 esophageal biopsies from 69 patients with benign diagnoses. All biopsies were assessed for the presence of hyperkeratosis, acanthosis, dyskeratosis, deep keratinization, intraepithelial neutrophils, neutrophilic microabscess, focal cytologic atypia, koilocyte-like cells, and keratin-filled cyst/burrows. Each feature, if present, was given one point, and the final histologic score was calculated for each biopsy by summing the points. The mean histologic score was 6.66 (s.d. 1.88) in biopsies from carcinoma cuniculatum vs a mean score of 1.93 (s.d. 1.75) for biopsies with benign diagnoses (Po0.0001). Using a cutoff value of 7 for carcinoma cuniculatum, 57% of biopsies (20/35) from 64% esophagogastroduodenoscopy procedures (16/25) in 91% patients (10/11) would be diagnostic, in comparison to the initial diagnostic rates of carcinoma of 9, 12, and 27%, respectively (Po0.0001 for all). None of the 92 benign biopsies showed a score of Z7. Our results demonstrate that a semiquantitative histologic evaluation of mucosal biopsies taken from an esophageal mass greatly improves the diagnostic sensitivity from patients with carcinoma cuniculatum with 100% specificity. Larger studies are necessary to confirm the current findings.

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Carcinoma cuniculatum of the larynx: a rare clinicopathological entity

Cited by 24 publications

References 18 publications

Carcinoma Cuniculatum of the Esophagus and Tongue: Report of Two Cases, Including TP53 Mutational Analysis

Carcinoma Cuniculatum of the Esophagus and Tongue: Report of Two Cases, Including TP53 Mutational Analysis

A Rare Case of Carcinoma Cuniculatum Involving the Larynx in Association with a Saccular Cyst

Semiquantitative histologic evaluation improves diagnosis of esophageal carcinoma cuniculatum on biopsy

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