22 October 2021
: Case report 
Vulvar Extramammary Paget Disease Detected by Cytology for Cervical Cancer Screening: A Case Report and Literature Review
Challenging differential diagnosis, Rare disease
Kotaro Takahashi1ACDEF*, Akira Kikuchi1BDEF, Ayano Horiuchi2DEF, Ryo Tamura1DEF, Kazufumi Haino3DEF, Masayuki Yamaguchi1DEF, Takashi Kawasaki4BDEFDOI: 10.12659/AJCR.933655
Am J Case Rep 2021; 22:e933655
Table 1. Reported cases of primary or secondary extramammary Paget disease with abnormal cervical cytology.
| Author | Patient age, y | Primary Lesion | Treatment history of primary lesion | Clinical presentation | Cervical cytology | Cervical histology |
|---|---|---|---|---|---|---|
| Costello et al []3 | 78 | PEMPD | Yes | Itching of vulva and vagina | Adenocarcinoma | EMPD |
| Arnould et al []4 | 72 | UC | Yes | Cervical cancer screening | Malignant urothelial cells | EMPD |
| Hendsch et al []5 | 53 | UC | No | Cervical cancer screening | Adenocarcinoma | No cervical neoplasm in hysterectomy specimen |
| Gu et al []6 | 78 | PEMPD | Yes | Cervical cancer screening | Adenocarcinoma | EMPD |
| Gu et al []6 | 87 | PEMPD | No | Cervical cancer screening | SCC | EMPD |
| Reyes et al []7 | 58 | UC | Yes | Cervical cancer screening | ASC-H | EMPD; She was initially diagnosed with HSIL/CIN3 |
| Westacott et al []8 | 81 | PEMPD | No | Vulvar eczema and itching | Abnormal cells | EMPD |
| Clayton et al []9 | 73 | PEMPD | Yes | Postmenopausal bleeding | Abnormal cells | EMPD |
| Davis et al []10 | 66 | PEMPD | Yes | Cervical cancer screening | SCC | EMPD |
| van der Linden et al []11 | 66 | CRC | No | Postmenopausal bleeding | Adenocarcinoma | EMPD |
| Koyanagi et al []12 | 68 | UC | Yes | Postmenopausal bleeding | Adenocarcinoma | No cervical neoplasm in conization specimen |
| This case | 77 | UC | Yes | Cervical cancer screening | Adenocarcinoma | Atypical glandular cells |
| ASC-H – atypical squamous cells-cannot exclude high-grade squamous intraepithelial lesion; CRC – colorectal carcinoma; EMPD – extramammary Paget disease; HSIL/CIN3 – high-grade squamous intraepithelial lesion/cervical intraepithelial neoplasia 3; PEMPD – primary extramammary Paget disease; SCC – squamous cell carcinoma; UC – urothelial carcinoma. | ||||||