Gastroenterology Research, ISSN 1918-2805 print, 1918-2813 online, Open Access
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Case Report

Volume 13, Number 1, February 2020, pages 44-51

Esophageal Carcinoma Cuniculatum Diagnosed on Mucosal Biopsies Using a Semiquantitative Histologic Schema: Report of Two Esophagectomy-Confirmed Cases

Figures

Figure 1.
Figure 1. (a) Endoscopic finding of an esophageal mass on esophagogastroduodenoscopy (case 1). (b) Mural destruction by the mass on endoscopic ultrasound (EUS) examination. (c) Enlarged hypoechoic nodes, suspicious for metastases on EUS examination. (d) Positron emission tomography (PET) revealed a hypermetabolic mass in the distal esophagus (axial view).
Figure 2.
Figure 2. Histologic features of mucosal biopsy from the mass (case 1). Squamous epithelium shows hyperkeratosis and acanthosis ((a) hematoxylin and eosin stain, original magnification × 40), focal atypia ((b) hematoxylin and eosin stain, original magnification × 200), intraepithelial neutrophilic inflammation and microabscesses and dyskeratosis ((c) hematoxylin and eosin stain, original magnification × 200), koilocyte-like cells ((d) hematoxylin and eosin stain, original magnification × 200), deep keratinization ((e) hematoxylin and eosin stain, original magnification × 100), and furrow ((f) hematoxylin and eosin stain, original magnification × 20).
Figure 3.
Figure 3. Esophagectomy (case 1) shows mural fibrosis and inflammation ((a) hematoxylin and eosin stain, original magnification × 20), few residual cyst lined with well-differentiated bland squamous epithelium ((b) hematoxylin and eosin stain, original magnification × 100), and histiocytic and giant cell response to keratinous material ((c) hematoxylin and eosin stain, original magnification × 200) and residual squamous carcinomatous cells ((d) hematoxylin and eosin stain, original magnification × 200).
Figure 4.
Figure 4. Histologic features of mucosal biopsy from the mass (case 2). Squamous epithelium shows hyperkeratosis and acanthosis ((a) hematoxylin and eosin stain, original magnification × 40), focal atypia ((b) hematoxylin and eosin stain, original magnification × 400), intraepithelial neutrophilic inflammation and microabscesses and dyskeratosis ((c) hematoxylin and eosin stain, original magnification × 200), koilocyte-like cells ((d) hematoxylin and eosin stain, original magnification × 200), deep keratinization ((e) hematoxylin and eosin stain, original magnification × 200), and furrow ((f) hematoxylin and eosin stain, original magnification × 40).
Figure 5.
Figure 5. Esophagectomy (case 2) shows histiocytic and giant cell response to keratinous material and residual terminally differentiated squamous carcinomatous cells ((a) and (b) hematoxylin and eosin stain, original magnification × 100 and × 200, respectively).

Tables

Table 1. Clinical, Radiologic, and Endoscopic Findings in These Two Patients With Esophageal Carcinoma Cuniculatum
 
PatientAgeGenderSymptomsChest CTPET/CTEGDEUS
CT: computed tomography; PET/CT: positron emission tomography-computed tomography; EGD: esophagogastroduodenoscopy; EUS: endoscopic ultrasound.
167MaleProgressive dysphagia (4 - 5 months) and 40-lb weight lossEsophageal wall thickeningAvid esophageal mass (4 cm × 6 cm)Partially obstructive mass (4 cm) in the distal esophagus and gastroesophageal junctionHypoechoic mass, extending beyond the muscularis propria and five malignant appearing lymph nodes (EUS staging of T3N2Mx)
262MaleProgressive dysphagia (5 months) and 30-lb weight lossEsophageal wall thickeningAvid esophageal massFirm, friable, partially obstructive and ulcerated mass (4 cm) in the distal esophagus, extending to the cardiaHypoechoic mass, extending beyond the muscularis propria and one 6 mm hypoechoic lymph node (EUS staging of T3N1Mx)

 

Table 2. Histopathologic Findings and Clinical Follow-Up in These Two Patients With Esophageal Carcinoma Cuniculatum
 
PatientNumber of biopsy setsHistology features on biopsyPreoperative diagnosisNeoadjuvant treatmentFinal histopathologic diagnosis on esophagectomyFollow-up
5-FU: 5-fluorouracil.
12Hyperkeratosis, acanthosis, dyskeratosis, deep keratinization, intraepithelial neutrophils, neutrophilic microabscess, focal cytologic atypia, koilocyte-like cells, and keratin-filled cyst/burrows.Carcinoma cuniculatumPaclitaxel/carboplatinResidual carcinoma cuniculatum with marked tumor regressionPatient was well 1 month after esophagectomy.
Ulceration
RadiationMural fibrosis
Histiocytic and giant cell response
15 negative lymph nodes
ypT3N0Mx
23Hyperkeratosis, acanthosis, dyskeratosis, deep keratinization, intraepithelial neutrophils, neutrophilic microabscess, focal cytologic atypia, koilocyte-like cells, and keratin-filled cyst/burrows.Carcinoma cuniculatum5-FU and cisplatinResidual carcinoma cuniculatum with marked tumor regressionNo recurrence or metastases were detected during his follow-up. Patient died from mixed obstructive-restrictive lung disease 36 months after the esophagectomy.
RadiationUlceration
Mural fibrosis
Histiocytic and giant cell response
28 negative lymph nodes
ypT3N0Mx