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

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 originati...

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Main Authors: Shohei Kishimoto, Shuhei Morita, Chiaki Kurimoto, Chie Kitahara, Tomoya Tsuji, Shinsuke Uraki, Ken Takeshima, Yasushi Furukawa, Hiroshi Iwakura, Hiroto Furuta, Masahiro Nishi, Taka-aki Matsuoka
Format: Article
Language:English
Published: BMC 2022-03-01
Series:BMC Endocrine Disorders
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Online Access:https://doi.org/10.1186/s12902-022-00973-0
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author Shohei Kishimoto
Shuhei Morita
Chiaki Kurimoto
Chie Kitahara
Tomoya Tsuji
Shinsuke Uraki
Ken Takeshima
Yasushi Furukawa
Hiroshi Iwakura
Hiroto Furuta
Masahiro Nishi
Taka-aki Matsuoka
author_facet Shohei Kishimoto
Shuhei Morita
Chiaki Kurimoto
Chie Kitahara
Tomoya Tsuji
Shinsuke Uraki
Ken Takeshima
Yasushi Furukawa
Hiroshi Iwakura
Hiroto Furuta
Masahiro Nishi
Taka-aki Matsuoka
author_sort Shohei Kishimoto
collection DOAJ
description 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.
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spelling doaj.art-c8ccea5497ee403aa257ec4c70d6a47a2022-12-21T22:51:20ZengBMCBMC Endocrine Disorders1472-68232022-03-012211710.1186/s12902-022-00973-0Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case reportShohei Kishimoto0Shuhei Morita1Chiaki Kurimoto2Chie Kitahara3Tomoya Tsuji4Shinsuke Uraki5Ken Takeshima6Yasushi Furukawa7Hiroshi Iwakura8Hiroto Furuta9Masahiro Nishi10Taka-aki Matsuoka11The First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityThe First Department of Medicine, Wakayama Medical UniversityAbstract 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.https://doi.org/10.1186/s12902-022-00973-0HypopituitarismB-cell lymphomaTolosa-Hunt syndrome
spellingShingle Shohei Kishimoto
Shuhei Morita
Chiaki Kurimoto
Chie Kitahara
Tomoya Tsuji
Shinsuke Uraki
Ken Takeshima
Yasushi Furukawa
Hiroshi Iwakura
Hiroto Furuta
Masahiro Nishi
Taka-aki Matsuoka
Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
BMC Endocrine Disorders
Hypopituitarism
B-cell lymphoma
Tolosa-Hunt syndrome
title Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
title_full Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
title_fullStr Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
title_full_unstemmed Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
title_short Hypopituitarism and cranial nerve involvement mimicking Tolosa-Hunt syndrome as the initially presenting feature of diffuse large B-cell lymphoma: a case report
title_sort hypopituitarism and cranial nerve involvement mimicking tolosa hunt syndrome as the initially presenting feature of diffuse large b cell lymphoma a case report
topic Hypopituitarism
B-cell lymphoma
Tolosa-Hunt syndrome
url https://doi.org/10.1186/s12902-022-00973-0
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