Case of minimal deviation adenocarcinoma: Possible clinical link to lobular endocervical glandular hyperplasia as its origin

Sugihara, Takeru; Nakagawa, Shunsuke; Sasajima, Yuko; Matsumoto, Yasuhiro; Takeshita, Shigeki; Ayabe, Tokiyoshi

doi:10.1111/jog.12538

Cited by 10 publications

(6 citation statements)

References 16 publications

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“…10 This is consistent with a previous report. 13 The present study suggests the importance of cytologic change rather than size change. However, it should be noted that two lobular endocervical glandular hyperplasia cases also showed worsening cytology, and some may be associated with an increase in size and worsening cytology, suggesting that the detection of worsening cytology in association with increased lesion size does not necessarily indicate the presence of malignancy.…”

Section: Discussionmentioning

confidence: 56%

Limited frequency of malignant change in lobular endocervical glandular hyperplasia

Kobara¹,

Miyamoto²,

Ando³

et al. 2020

Int J Gynecol Cancer

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IntroductionAlthough lobular endocervical glandular hyperplasia is a benign disorder of the uterine cervix, its potential as a precursor of minimal deviation adenocarcinoma has been reported. However, the natural history of the disease and the frequency of malignant change are not fully understood. We evaluated the frequency of malignant change of clinical lobular endocervical glandular hyperplasia and explored useful parameters indicating malignant change.MethodsThe clinical courses of 175 patients with cervical multi-cystic lesions who visited Shinshu University Hospital between June 1995 and June 2019 were retrospectively analyzed. We examined the results of follow-up and outcomes of the patients diagnosed with lobular endocervical glandular hyperplasia and investigated the frequency of malignant transformation.ResultsOf the 175 patients, 15, 84, and 76 were clinically diagnosed with suspected malignancy, suspected lobular endocervical glandular hyperplasia, and suspected nabothian cyst, respectively. Of these patients, 69 patients with suspected lobular endocervical glandular hyperplasia were followed, and 12 underwent hysterectomy after a mean follow-up of 57.1 (range: 3–154) months due to lesion enlargement (increase in tumor diameter of >20%) and/or worsening cytology. Of these 12 patients, two had lobular endocervical glandular hyperplasia with atypia and one had minimal deviation adenocarcinoma. Of 69 patients, the rate of malignant change was 1.4% (1/69). The growth rates of the lesions for these three patients during follow-up were significantly higher than those of nine patients who underwent surgery with lobular endocervical glandular hyperplasia without atypia and 48 follow-up cases of suspected lobular endocervical glandular hyperplasia. The cut-off value of the growth rate suggesting malignant transformation was 38.1% (84.6% sensitivity and 100% specificity). Tumor size and cytology did not change in the remaining 57 cases continuing follow-up.ConclusionAn increase in tumor size and worsening cytology are important parameters for detecting malignant transformation of lobular endocervical glandular hyperplasia during follow-up. However, the frequency of malignant change of this disease may be limited. These results suggest that conservative management may be an option for clinical lobular endocervical glandular hyperplasia.

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Section: Discussionmentioning

confidence: 56%

Limited frequency of malignant change in lobular endocervical glandular hyperplasia

Kobara¹,

Miyamoto²,

Ando³

et al. 2020

Int J Gynecol Cancer

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“…Sugihara et al reported a case that was initially diagnosed as LEGH, but histologically diagnosed as MDA after a 2-year follow-up, which revealed an increase in the size of the lesion (from 21 mm to 31 mm in diameter), and the deterioration of the Pap smear. 28 They concluded that the deterioration of Pap smear findings and increase in lesion size were important signs of the malignant progression of LEGH lesions to MDA. Therefore, we consider changes in MRI findings and cytology as the hallmarks of malignant transformation.…”

Section: Discussionmentioning

confidence: 99%

Usefulness of a management protocol for patients with cervical multicystic lesions: A retrospective analysis of 94 cases and the significance of GNAS mutation

Ando

Miyamoto

Kashima

et al. 2016

J of Obstet and Gynaecol

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AimThe proper preoperative diagnosis and management of cervical proliferative disorders presenting with multiple cysts, including minimal deviation adenocarcinoma (MDA), lobular endocervical glandular hyperplasia (LEGH), and nabothian cyst (NC), have not been fully established. We previously proposed a management protocol comprising a diagnostic approach using cytology, magnetic resonance imaging, and gastric‐type mucin and subsequent treatment. We herein evaluate the usefulness of this protocol and implications of GNAS mutations in LEGH.MethodsThe clinical courses of 94 patients with cervical multicystic lesions who visited our hospital between June 1995 and September 2014 were retrospectively analyzed. GNAS mutations were investigated in 10 LEGH, five LEGH with atypia, and two MDA cases.ResultsOf the 94 patients, the conditions of 10, 59, and 25 were clinically diagnosed as suspicious of MDA or carcinoma (S/O MDA‐Ca), suspicious of LEGH (S/O LEGH), and NC, respectively. Ten patients each with S/O MDA‐Ca and S/O LEGH underwent hysterectomy, and the correct ratio for diagnosis was 90% (18/20). Of the 42 S/O LEGH cases followed‐up for more than 12 months, three showed an increase in tumor size. After hysterectomy, two were LEGH with atypia while one was NC. The GNAS mutation was detected in two cases of LEGH with atypia, one of which showed an increase in tumor size during follow‐up.ConclusionThe management protocol we propose herein will be useful. An increase in tumor size is important to detect potentially malignant LEGH. GNAS mutations may be involved in the tumorigenesis of potentially malignant LEGH.

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“…However, there are no reliable markers to detect the early stage of malignant transformation. In this regard, an informative report 45 and our report 44 suggested that an increase in lesion size and worsening cytology were important markers to detect malignant change of LEGH. In reference with these reports, we also carefully followed up 69 cases of clinical LEGH, we found one MDA patient.…”

Section: Clinical Managementmentioning

confidence: 68%

“…NC cases generally show NILM, whereas most MDA/GAS shows AGC‐FN, AIS, or adenocarcinoma. Especially, AGC‐NOS suggested benign (nonatypical) LEGH, whereas AGC‐FN suggested atypical LEGH or MDA/GAS 44,45 . Collectively, cervical cytology is useful to distinguish benign LEGH and MDA/GAS/atypical LEGH 14,26,42 .…”

Section: Clinical Managementmentioning

confidence: 99%

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Biology and management of lobular endocervical glandular hyperplasia

Miyamoto

Kobara

Shiozawa

2022

J of Obstet and Gynaecol

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Aim: Lobular endocervical glandular hyperplasia (LEGH) is a multicystic proliferative disorder of the uterine cervix. The aim of this review was to clarify the current understanding of this unique tumor. Method: This article reviews the chronological progress of research regarding clinico-pathological and genetic aspects of LEGH and related cervical cystic diseases such as Nabothian cyst and adenocarcinoma of gastric type (GAS), using the literature and data from our institute. We also describe clinical management including preoperative diagnosis and adequate surgical/expectant treatment based on the biological features.Results: Recent studies revealed several unique aspects of LEGH, that is, (i) production of gastric mucin, (ii) symptomatic and histological similarity with minimal deviation adenocarcinoma (MDA), and (iii) frequent association with GAS, including MDA. These findings indicated that LEGH is a gastric metaplasia, as well as pre-cancerous neoplasia. For the preoperative diagnosis of LEGH, the combination of "cosmos" sign on magnetic resonance imaging, detection of gastric mucin, and lack of nuclear atypia on cytology is important. Cone biopsy is effective for pathological diagnosis. Simple hysterectomy is indicated as surgical treatment for LEGH; however, meticulous follow-up is also an option, especially for young patients, because the rate of malignant transformation was reported to be 1%-2%. For LEGH patients who selected follow-up, a worsening cytology and increase in lesion size were important signs of malignant change of LEGH for safe follow-up. Conclusion: Proper understanding of the characteristics of LEGH is important for adequate management.

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Case of minimal deviation adenocarcinoma: Possible clinical link to lobular endocervical glandular hyperplasia as its origin

Cited by 10 publications

References 16 publications

Limited frequency of malignant change in lobular endocervical glandular hyperplasia

Limited frequency of malignant change in lobular endocervical glandular hyperplasia

Usefulness of a management protocol for patients with cervical multicystic lesions: A retrospective analysis of 94 cases and the significance of GNAS mutation

Biology and management of lobular endocervical glandular hyperplasia

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