Author Information
Sathe
Pragati A*, Patil Lalita Y**, Vaideeswar Pradeep***, Mayadeo Niranjan M****
(*
Associate Professor, ** Assistant Professor,
***Additional Professor, Department of Pathology, **** Professor,
Department of Obstetrics and Gynecology, Seth GS Medical
College & KEM Hospital , Mumbai ,
India
Abstract
Ichthyosis
uteri, a rare condition, is called so when the entire surface of the
endometrium is replaced by keratinized stratified squamous epithelium. It is a
benign lesion and its association with benign and malignant conditions has been
reported in the literature. If widespread keratinization of endometrial surface
is detected in the curettage or biopsy then an advanced examination should be
done to rule out an underlying malignancy.
Introduction
The term ichthyosis
uteri is used when there is widespread replacement of the surface endometrium
by keratinized stratified squamous epithelium.[1] It is considered a
benign lesion but its association with malignancy has been reported in the
literature. [2] We report a case of ichthyosis uteri detected on endometrial
curettage in an elderly postmenopausal woman.
Case Report
A
60-year-old woman, gravida 9, para 9 with eight living issues came with
complaints of foul smelling white discharge of three months duration. Per
vaginal examination showed a normal sized anteverted uterus. Bilateral fornices
were free. On per speculum examination, foul smelling cervical discharge was
noted. The vagina was normal. No growth was appreciated in the cervix.
Ultrasonography of the pelvis showed 3x2x1.5 centimeters sized collection
inside the uterine cavity. Both ovaries were normal. Clinical impression was
endometritis with pyometra. The hematological and routine biochemical
investigations were within normal range. The patient underwent cervical
dilatation along with drainage of 50 ml of profuse greenish foul smelling
purulent discharge from the uterine cavity. Papanicolau stain of the cervical
discharge showed mature keratinized squamous epithelial cells on a background
of neutrophils. Fractional endometrial curettage was performed after six weeks
and the material was sent for histopathological examination. The microscopy
showed prominent neutrophilic infiltrate with necrosis. Bathing in this
exudates, were seen strips of keratinized squamous epithelium. The normal endometrial
glands were covered by this epithelium (Figure 1a). There was low grade
dysplasia of the squamous epithelium (Fig 1b). There was no evidence of associated
endometrial malignancy. A diagnosis of ichthyosis uteri was given. The patient
was administered oral antibiotics and discharged with an advice for a
hysterectomy. She refused further treatment and was lost to follow-up, despite
explanation of the associated risks.
Figure 1. Ichthyosis Uteri. 1a. Keratinized
squamous epithelium with endometrial glands beneath. (Hematoxylin and Eosin,
100x); 1b. Low grade dysplasia seen in the squamous epithelium. (Hematoxylin
and Eosin, 400x)
Discussion
First coined by Zeller in 1885, the term ‘ichthyosis uteri’
means widespread keratinization of the endometrium. [3] It is rarer compared to squamous metaplasia. Chronic
trauma, repair, irritation, inflammation, foreign material, and estrogenic
effects have all been implicated.[3] The
etiology of endometrial keratinization is not well understood. This rare
pathology has been seen in association with benign conditions like tuberculous
endometritis, puerperal endometritis, endometrial polyps, hyperplasias,
squamous papilloma and with pyometra as a result of cervical stenosis.[1,3] Hence, when ichthyosis is seen on endometrial curettage,
one has to look for associated benign pathologies especially tuberculosis,
endometrial polyps and hyperplasia.
According to some investigators, ichthyosis lacks malignant
potential.[1] However, dysplastic and anaplastic changes in the squamous
epithelium have been reported, which may predispose to the rare endometrial squamous carcinoma in
postmenopausal women.[1,2]
The main differential diagnosis to be considered before
making a diagnosis of pure ichthyosis is the extension of squamous carcinoma of the
cervix into the endometrial cavity. The primary tumor in such a case would have
an infiltrating rather than a polypoid morphology. The extension of well
differentiated squamous carcinoma from the cervix can be distinguished from
ichthyosis by detailed examination of the lower genital tract, by the presence
of koilocytic changes and the presence of dysplastic changes in the squamous
epithelium which favors a diagnosis of squamous carcinoma extension from cervix.
[1,3]
In our case, the predisposing factor seems to be repeated
episodes of pyometra which is suggested by strips of squamous epithelium embedded
within abundant neutrophilic exudates as seen on endometrial curettage. The low
grade dysplasia seen in the epithelium could suggest the likelihood of associated
squamous carcinoma cervix. Unfortunately, our patient declined any further
treatment and hence the cause of ichthyosis could not be ascertained
To conclude,
ichthyosis is a benign condition but can lead to or be associated with
endometrial malignancy, hence the need for timely recognition of this
condition.[2] The clinical diagnosis in such cases is most
frequently pyometra which has to be thoroughly investigated, especially when it
occurs in postmenopausal women. If widespread keratinization of endometrial
surface is detected in the curettage or biopsy then an advanced examination
should be done to rule out an underlying malignancy.[1]
References
1.
Fadare O. Dysplastic Ichthyosis uteri-like
changes of the entire endometrium associated with a squamous cell carcinoma of
the uterine cervix. Diagn Pathol 2006;1:8-11.
2.
Bagga
PK Mahajan
NC
3.
Bewtra C, Xie QM, Hunter WJ, Jurgensen
W.Ichthyosis uteri: a case report and review of literature. Arch Pathol Lab Med
2005;129:e124-e125.
Citation
Sathe PA,
Patil LY, Vaideeswar P, Mayadeo
NM