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Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review

Received: 18 August 2020     Accepted: 27 August 2020     Published: 3 September 2020
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Abstract

Giant prolactinomas are rare tumors, representing only 2-3% of all prolactin (PRL)-secreting tumors. The management of giant prolactinomas remains a major challenge. Here we report the case of a young man aged 31 years old, followed up for a giant and invasive pituitary macroadenoma of 5cm discovered during a neuro-ophthalmic syndrome. The initial hormonal exploration and follow-up evaluation concluded hyperprolactinemia at 138.5ng/mL, anterior pituitary insufficiency and bilateral blindness related to optic atrophy. Incomplete surgical resection was performed after one month of medical treatment with a dopaminergic agonist. Histologic examination showed an aspect of an invasive macro-adenoma with no signs of malignancy. Despite therapeutic escalation, the evolution was marked by the persistence of resistant hyperprolactinemia. A control MRI was done showing a voluminous extra-axial expansive process measuring 9cm with significant anterolateral and inferior extension in the cavum and nasal fossae. A biopsy of the tumour in the nasal fossae was done showing morphological aspects and an immuno-histochemical profile of an aggressive lactotropic adenoma of the type "Acidophilic stem Cell adenoma" with an over expression of p53. External radiotherapy was indicated, but the patient died rapidly in a brain engagement table. This case illustrates the difficulty in the management and control of invasive and aggressive pituitary adenomas and their unpredictable course.

Published in World Journal of Medical Case Reports (Volume 1, Issue 1)
DOI 10.11648/j.wjmcr.20200101.11
Page(s) 1-4
Creative Commons

This is an Open Access article, distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution and reproduction in any medium or format, provided the original work is properly cited.

Copyright

Copyright © The Author(s), 2020. Published by Science Publishing Group

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Keywords

Giant Prolactinoma, Pituitary Tumor, Cabergoline

References
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[12] Berwaerts J, Verhelst J, Abs R, Appel B, Mahler CH. A giant prolactinoma presenting with unilateral exophthalmos: effect of cabergoline and review of the literature. J Endocrinol Invest. 2000; 23 (6): 393-8.
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[18] Ciric I, Mikhael M, Stafford T, Lawson L, Garces R. Transsphenoidal microsurgery of pituitary macroadenomas with long-term follow-up results. J Neurosurg. 1983; 59 (3): 395-401.
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  • APA Style

    Faten Hadj Kacem, Kawthar El Arbi, Abdalla Mrabet, Khouloud Boujelben, Souha Kallel, et al. (2020). Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review. World Journal of Medical Case Reports, 1(1), 1-4. https://doi.org/10.11648/j.wjmcr.20200101.11

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    ACS Style

    Faten Hadj Kacem; Kawthar El Arbi; Abdalla Mrabet; Khouloud Boujelben; Souha Kallel, et al. Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review. World J. Med. Case Rep. 2020, 1(1), 1-4. doi: 10.11648/j.wjmcr.20200101.11

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    AMA Style

    Faten Hadj Kacem, Kawthar El Arbi, Abdalla Mrabet, Khouloud Boujelben, Souha Kallel, et al. Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review. World J Med Case Rep. 2020;1(1):1-4. doi: 10.11648/j.wjmcr.20200101.11

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  • @article{10.11648/j.wjmcr.20200101.11,
      author = {Faten Hadj Kacem and Kawthar El Arbi and Abdalla Mrabet and Khouloud Boujelben and Souha Kallel and Slim Charfi and Mouna Elleuch and Ilhem Charfeddine and Taheya Boudawara and Mohamed Abid},
      title = {Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review},
      journal = {World Journal of Medical Case Reports},
      volume = {1},
      number = {1},
      pages = {1-4},
      doi = {10.11648/j.wjmcr.20200101.11},
      url = {https://doi.org/10.11648/j.wjmcr.20200101.11},
      eprint = {https://article.sciencepublishinggroup.com/pdf/10.11648.j.wjmcr.20200101.11},
      abstract = {Giant prolactinomas are rare tumors, representing only 2-3% of all prolactin (PRL)-secreting tumors. The management of giant prolactinomas remains a major challenge. Here we report the case of a young man aged 31 years old, followed up for a giant and invasive pituitary macroadenoma of 5cm discovered during a neuro-ophthalmic syndrome. The initial hormonal exploration and follow-up evaluation concluded hyperprolactinemia at 138.5ng/mL, anterior pituitary insufficiency and bilateral blindness related to optic atrophy. Incomplete surgical resection was performed after one month of medical treatment with a dopaminergic agonist. Histologic examination showed an aspect of an invasive macro-adenoma with no signs of malignancy. Despite therapeutic escalation, the evolution was marked by the persistence of resistant hyperprolactinemia. A control MRI was done showing a voluminous extra-axial expansive process measuring 9cm with significant anterolateral and inferior extension in the cavum and nasal fossae. A biopsy of the tumour in the nasal fossae was done showing morphological aspects and an immuno-histochemical profile of an aggressive lactotropic adenoma of the type "Acidophilic stem Cell adenoma" with an over expression of p53. External radiotherapy was indicated, but the patient died rapidly in a brain engagement table. This case illustrates the difficulty in the management and control of invasive and aggressive pituitary adenomas and their unpredictable course.},
     year = {2020}
    }
    

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  • TY  - JOUR
    T1  - Giant Macroadenoma with an Unusual Extension: A Case Report and Literature Review
    AU  - Faten Hadj Kacem
    AU  - Kawthar El Arbi
    AU  - Abdalla Mrabet
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    AU  - Slim Charfi
    AU  - Mouna Elleuch
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    T2  - World Journal of Medical Case Reports
    JF  - World Journal of Medical Case Reports
    JO  - World Journal of Medical Case Reports
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    EP  - 4
    PB  - Science Publishing Group
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    UR  - https://doi.org/10.11648/j.wjmcr.20200101.11
    AB  - Giant prolactinomas are rare tumors, representing only 2-3% of all prolactin (PRL)-secreting tumors. The management of giant prolactinomas remains a major challenge. Here we report the case of a young man aged 31 years old, followed up for a giant and invasive pituitary macroadenoma of 5cm discovered during a neuro-ophthalmic syndrome. The initial hormonal exploration and follow-up evaluation concluded hyperprolactinemia at 138.5ng/mL, anterior pituitary insufficiency and bilateral blindness related to optic atrophy. Incomplete surgical resection was performed after one month of medical treatment with a dopaminergic agonist. Histologic examination showed an aspect of an invasive macro-adenoma with no signs of malignancy. Despite therapeutic escalation, the evolution was marked by the persistence of resistant hyperprolactinemia. A control MRI was done showing a voluminous extra-axial expansive process measuring 9cm with significant anterolateral and inferior extension in the cavum and nasal fossae. A biopsy of the tumour in the nasal fossae was done showing morphological aspects and an immuno-histochemical profile of an aggressive lactotropic adenoma of the type "Acidophilic stem Cell adenoma" with an over expression of p53. External radiotherapy was indicated, but the patient died rapidly in a brain engagement table. This case illustrates the difficulty in the management and control of invasive and aggressive pituitary adenomas and their unpredictable course.
    VL  - 1
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Author Information
  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

  • Department of ORL, Habib Bourguiba Hospital, Sfax, Tunisia

  • Department of Anatomy Pathology, Habib Bourguiba Hospital, Sfax, Tunisia

  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

  • Department of ORL, Habib Bourguiba Hospital, Sfax, Tunisia

  • Department of Anatomy Pathology, Habib Bourguiba Hospital, Sfax, Tunisia

  • Department of Endocrinology, Hedi Chaker Hospital, Sfax, Tunisia

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