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Intraparenchymal infiltration of Rathke's cleft cysts manifesting as severe neurological deficits and hypopituitarism: 2 case reports.

Ogawa Y, Watanabe M, Tominaga T - BMC Res Notes (2016)

Bottom Line: Most infiltrated cells were lymphocytes and plasma cells, thought to indicate the involvement of long-term underling inflammatory processes in this phenomenon.Long-term subclinical inflammation may be the mechanism of this extraordinary aggressive clinical course.Postoperative steroid administration should be reduced prudently, and careful follow-up imaging is essential in cases of Rathke's cleft cyst with abnormal histological findings.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurosurgery, Kohnan Hospital, 4-20-1 Nagamachiminami, Taihaku-ku, Sendai, Miyagi, 982-8523, Japan. yogawa@kohnan-sendai.or.jp.

ABSTRACT

Background: Rathke's cleft cysts generally remain asymptomatic throughout life, but a few patients may suffer severe neurological and/or endocrinological deficits. The symptoms include visual disturbances caused by compression of the optic chiasm, and severe endocrinological deficits caused by repeated intracystic hemorrhage or leakage of cyst content. However, no case of Rathke's cleft cyst has infiltrated into neuroglial tissue with marked cerebral edema.

Case presentation: Two patients presented with non-infectious re-deterioration of Rathke's cleft cysts with intraparenchymal infiltration and marked cerebral edema, to ipsilateral hypothalamus in one case and to the bilateral frontal lobes in the other. Both patients were surgically treated by extended transsphenoidal surgery, and showed remarkable improvement with postoperative pulse-dose steroid therapy, including disappearance/shrinkage of abnormal enhanced lesion and cerebral edema on magnetic resonance imaging. Histological examination disclosed significant squamous metaplasia in epithelia and marked infiltration of inflammatory cells into the pituitary gland and neuroglial tissues. Most infiltrated cells were lymphocytes and plasma cells, thought to indicate the involvement of long-term underling inflammatory processes in this phenomenon.

Conclusion: Long-term subclinical inflammation may be the mechanism of this extraordinary aggressive clinical course. Postoperative steroid administration should be reduced prudently, and careful follow-up imaging is essential in cases of Rathke's cleft cyst with abnormal histological findings.

No MeSH data available.


Related in: MedlinePlus

Case 1. a Photomicrograph showing smooth transition from single columnar epithelium to stratified squamous epithelia, original magnification ×100. b High-power photomicrograph showing involvement of the pituitary gland in sub-epithelial inflammatory granulation. Most infiltrated cells were lymphocytes and plasma cells, original magnification ×200. c Inflammatory cells had frequently infiltrated into the arachnoid and neuroglial tissues, original magnification ×100. d Significant angiogenesis is present, original magnification ×100. Hematoxylin and eosin staining
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Fig3: Case 1. a Photomicrograph showing smooth transition from single columnar epithelium to stratified squamous epithelia, original magnification ×100. b High-power photomicrograph showing involvement of the pituitary gland in sub-epithelial inflammatory granulation. Most infiltrated cells were lymphocytes and plasma cells, original magnification ×200. c Inflammatory cells had frequently infiltrated into the arachnoid and neuroglial tissues, original magnification ×100. d Significant angiogenesis is present, original magnification ×100. Hematoxylin and eosin staining

Mentions: Surgery was performed through the extended transsphenoidal approach. Intraoperative finding disclosed the tough and thick rugged lesion, which was removed en-bloc together with the infiltrated third ventricle floor. Subtotal removal was achieved. Postoperative histological examination disclosed ciliated and goblet cell columnar epithelium, containing a small component of squamous metaplasia in the lining. The diagnosis was established as Rathke’s cleft cyst. However, significant sub-epithelial infiltration of inflammatory cells was seen, and massive inflammatory granulation had formed. Most infiltrated cells were lymphocytes and plasma cells, frequently seen in neuroglial tissues, and the pituitary gland was isolated in this inflammatory ocean (Fig. 3).Fig. 3


Intraparenchymal infiltration of Rathke's cleft cysts manifesting as severe neurological deficits and hypopituitarism: 2 case reports.

Ogawa Y, Watanabe M, Tominaga T - BMC Res Notes (2016)

Case 1. a Photomicrograph showing smooth transition from single columnar epithelium to stratified squamous epithelia, original magnification ×100. b High-power photomicrograph showing involvement of the pituitary gland in sub-epithelial inflammatory granulation. Most infiltrated cells were lymphocytes and plasma cells, original magnification ×200. c Inflammatory cells had frequently infiltrated into the arachnoid and neuroglial tissues, original magnification ×100. d Significant angiogenesis is present, original magnification ×100. Hematoxylin and eosin staining
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4837620&req=5

Fig3: Case 1. a Photomicrograph showing smooth transition from single columnar epithelium to stratified squamous epithelia, original magnification ×100. b High-power photomicrograph showing involvement of the pituitary gland in sub-epithelial inflammatory granulation. Most infiltrated cells were lymphocytes and plasma cells, original magnification ×200. c Inflammatory cells had frequently infiltrated into the arachnoid and neuroglial tissues, original magnification ×100. d Significant angiogenesis is present, original magnification ×100. Hematoxylin and eosin staining
Mentions: Surgery was performed through the extended transsphenoidal approach. Intraoperative finding disclosed the tough and thick rugged lesion, which was removed en-bloc together with the infiltrated third ventricle floor. Subtotal removal was achieved. Postoperative histological examination disclosed ciliated and goblet cell columnar epithelium, containing a small component of squamous metaplasia in the lining. The diagnosis was established as Rathke’s cleft cyst. However, significant sub-epithelial infiltration of inflammatory cells was seen, and massive inflammatory granulation had formed. Most infiltrated cells were lymphocytes and plasma cells, frequently seen in neuroglial tissues, and the pituitary gland was isolated in this inflammatory ocean (Fig. 3).Fig. 3

Bottom Line: Most infiltrated cells were lymphocytes and plasma cells, thought to indicate the involvement of long-term underling inflammatory processes in this phenomenon.Long-term subclinical inflammation may be the mechanism of this extraordinary aggressive clinical course.Postoperative steroid administration should be reduced prudently, and careful follow-up imaging is essential in cases of Rathke's cleft cyst with abnormal histological findings.

View Article: PubMed Central - PubMed

Affiliation: Department of Neurosurgery, Kohnan Hospital, 4-20-1 Nagamachiminami, Taihaku-ku, Sendai, Miyagi, 982-8523, Japan. yogawa@kohnan-sendai.or.jp.

ABSTRACT

Background: Rathke's cleft cysts generally remain asymptomatic throughout life, but a few patients may suffer severe neurological and/or endocrinological deficits. The symptoms include visual disturbances caused by compression of the optic chiasm, and severe endocrinological deficits caused by repeated intracystic hemorrhage or leakage of cyst content. However, no case of Rathke's cleft cyst has infiltrated into neuroglial tissue with marked cerebral edema.

Case presentation: Two patients presented with non-infectious re-deterioration of Rathke's cleft cysts with intraparenchymal infiltration and marked cerebral edema, to ipsilateral hypothalamus in one case and to the bilateral frontal lobes in the other. Both patients were surgically treated by extended transsphenoidal surgery, and showed remarkable improvement with postoperative pulse-dose steroid therapy, including disappearance/shrinkage of abnormal enhanced lesion and cerebral edema on magnetic resonance imaging. Histological examination disclosed significant squamous metaplasia in epithelia and marked infiltration of inflammatory cells into the pituitary gland and neuroglial tissues. Most infiltrated cells were lymphocytes and plasma cells, thought to indicate the involvement of long-term underling inflammatory processes in this phenomenon.

Conclusion: Long-term subclinical inflammation may be the mechanism of this extraordinary aggressive clinical course. Postoperative steroid administration should be reduced prudently, and careful follow-up imaging is essential in cases of Rathke's cleft cyst with abnormal histological findings.

No MeSH data available.


Related in: MedlinePlus