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Case Reports
. 2022 Mar 14;22(1):65.
doi: 10.1186/s12902-022-00973-0.

Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report

Shohei Kishimoto #  1 , Shuhei Morita #  2 , Chiaki Kurimoto  1 , Chie Kitahara  1 , Tomoya Tsuji  1 , Shinsuke Uraki  1 , Ken Takeshima  1 , Yasushi Furukawa  1 , Hiroshi Iwakura  1 , Hiroto Furuta  1 , Masahiro Nishi  1 , Taka-Aki Matsuoka  1
Affiliations
Case Reports

Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report

Shohei Kishimoto et al. BMC Endocr Disord. .

Abstract

Background: Early diagnosis of lymphoma involving the central nervous system is sometimes difficult but emergent to avoid the delay of therapeutic initiation. Pituitary insufficiencies are usually associated with lymphoma in the pituitary gland. There have been no cases of lymphoma originating from extra pituitary gland with hypopituitarism that simultaneously presenting unilateral upper cranial nerve palsies and ophthalmalgia. These symptoms are mostly caused by neoplastic involvement of the skull base or benign diseases such as Tolosa-Hunt syndrome (THS). We report a case of lymphoma with unique clinical courses initially presenting hypopituitarism and symptoms mimicking THS with a mass in sphenoidal and cavernous sinuses accompanying sphenoidal bone erosion.

Case presentation: A 71-year-old woman visited our hospital with left ophthalmalgia, ptosis, and diplopia. Neurological findings revealed left oculomotor, trochlear and abducent nerve palsies. Endocrine tests indicated partial hypopituitarism. Initial CT and MRI revealed that a mass in sphenoidal and cavernous sinuses had invaded the sella with osteolysis of the sphenoid bone. At around four weeks, almost all the symptoms of cranial nerve palsies were relieved. Seven weeks later, she had a high fever and cervical lymph node (CLN) swellings. CLN biopsy revealed CD20-positive B-cells. She was diagnosed with diffuse large B-cell lymphoma (DLBCL). 18F-fluorodeoxyglucose positron emission tomography/computed tomography (PET/CT) revealed elevated uptake at the erosion lesion of the sphenoidal bone, but not the pituitary gland. After chemotherapy, all the symptoms related to systemic lymphoma were relieved, but partial hypopituitarism remained. The mass in sphenoidal and cavernous sinuses and elevated uptake by PET/CT were dissolved.

Conclusion: This case of DLBCL had a unique clinical course; initial presentation of hypopituitarism and symptoms mimicking THS. There was also rare demonstration of mass lesions related to DLBCL in the sphenoidal and cavernous sinuses compressing the pituitary gland through an eroded area of the sphenoidal bone. It should be clinically cautioned that DLBCL can be associated with erosion of the sphenoidal bone and cause both hypopituitarism and THS-mimicking symptoms.

Keywords: B-cell lymphoma; Hypopituitarism; Tolosa-Hunt syndrome.

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Conflict of interest statement

The authors declare that they have no competing interests.

Figures

Fig. 1
Fig. 1
Clinical course
Fig. 2
Fig. 2
Pituitary function test at the first admission. A, B, C, D represent the results after CRH, GnRH, TRH, GHRP-2 load, respectively
Fig. 3
Fig. 3
MRI A-D and CT E images at the first admission. C, D MRI performed after injection of gadolinium. E Space between arrows indicates the erosion of the sphenoidal bone
Fig. 4
Fig. 4
18F-fluorodeoxyglucose positron emission tomography images at the second admission A and B and after six courses of treatment with R-CHOP C and D. Arrowheads in A and B indicate elevated uptake at left sphenoidal bone lesion
See this image and copyright information in PMC

References

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