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Diagnosis and Treatment > Medication

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Taisuke Uchida Department of Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Hideki Yamaguchi Department of Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Kazuhiro Nagamine Department of Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Tadato Yonekawa Department of Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Eriko Nakamura Department of Diagnostic Pathology, University of Miyazaki Hospital, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Nobuhiro Shibata Department of Clinical Oncology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Fumiaki Kawano Division of the Gastrointestinal, Endocrine and Pediatric Surgery, Department of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Yujiro Asada Department of Diagnostic Pathology, University of Miyazaki Hospital, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Masamitsu Nakazato Department of Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

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Summary

We report a case of rapid pleural effusion after discontinuation of lenvatinib. A 73-year-old woman was diagnosed with poorly differentiated thyroid cancer with right pleural metastasis. Weekly paclitaxel treatment was performed for 18 weeks, but it was not effective. Oral administration of lenvatinib, a multi-target tyrosine kinase inhibitor, reduced the size of cervical and thoracic tumors and lowered serum thyroglobulin levels. Lenvatinib was discontinued on day 28 because of Grade 2 thrombocytopenia and Grade 3 petechiae. Seven days after discontinuation of lenvatinib, the patient was hospitalized because of dyspnea and right pleural effusion. Pleural effusion rapidly improved with drainage and re-initiation of lenvatinib and did not recur. Anorexia caused by lenvatinib led to undernutrition, which resulted in death 13 months after initiation of lenvatinib. Autopsy revealed extensive necrosis with primary and metastatic lesions, suggesting that the patient responded to lenvatinib. Physicians should be aware of the possibility of flare-up in patients with thyroid cancer treated with lenvatinib.

Learning points:

  • Autopsy findings revealed that lenvatinib was efficacious in treating poorly differentiated thyroid cancer without primary lesion resection.

  • Flare-up phenomenon may occur in thyroid cancer treated with lenvatinib.

  • Attention should be paid to flare-up phenomenon within a few days of discontinuing lenvatinib.

Taxonomy:
Clinical Overview, Gland/Organ, Thyroid, Topic, Thyroid, Hormone, Thyroxine (T4), Triiodothyronine (T3), TSH, Condition/ Syndrome, Anaplastic thyroid cancer, Hypothyroidism, Lung metastases, Patient Demographics, Age, Geriatric, Gender, Female, Ethnicity, Asian - Japanese, Country of Treatment, Japan, Diagnosis and Treatment, Signs and Symptoms, Anorexia, Dyspnoea, Hypothyroidism, Hypoxia, Petechiae, Pneumonia, Thrombocytopenia, Weight loss, Investigation, CT scan, FT3, FT4, Histopathology, PET scan, Thyroglobulin, Thyroid function, TSH, X-ray, Medication, Dexamethasone, Glucocorticoids, Levatinib, Levothyroxine, Paclitaxel, Tyrosine-kinase inhibitors, Related Disciplines, Oncology, Publication Details, Case Report Type, Unusual effects of medical treatment
DOI:
https://doi.org/10.1530/EDM-18-0158
Volume/Issue:
Volume 2019: Issue 1
Open access
G K Dimitriadis Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK
Division of Experimental Medicine, Faculty of Medicine, Imperial College London, Hammersmith Campus, London, UK
Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK

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K Gopalakrishnan Department of Histopathology, Coventry and Warwickshire, Pathology Service, UHCW NHS Trust, Coventry, UK

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R Rao Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK

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D K Grammatopoulos Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK
Department of Clinical Biochemistry and Histopathology, Coventry and Warwickshire, Pathology Service, UHCW NHS Trust, Coventry, UK

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H S Randeva Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK
Division of Translational and Systems Medicine, Warwick Medical School, University of Warwick, Coventry, UK

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M O Weickert Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK

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N Murthy Warwick Institute for the Study of Endocrinology Diabetes and Metabolism (WISDEM Centre), The Arden NET Centre, University Hospitals of Coventry and Warwickshire, UHCW NHS Trust, ENETS CoE, Coventry, UK

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Summary

We report the case of a 70-year-old previously healthy female who presented acutely to the Accident and Emergency department with left-sided vasomotor symptoms including reduced muscle tone, weakness upon walking and slurred speech. Physical examination confirmed hemiparesis with VIIth nerve palsy and profound hepatomegaly. A random glucose was low at 1.7 mmol/l, which upon correction resolved her symptoms. In hindsight, the patient recalled having had similar episodes periodically over the past 3 months to which she did not give much attention. While hospitalized, she continued having episodes of symptomatic hypoglycaemia during most nights, requiring treatment with i.v. dextrose and/or glucagon. Blood tests including insulin and C-peptide were invariably suppressed, in correlation with low glucose. A Synacthen stimulation test was normal (Cort (0′) 390 nmol/l, Cort (30′) 773 nmol/l). A computed tomography scan showed multiple lobulated masses in the abdomen, liver and pelvis. An ultrasound guided biopsy of one of the pelvic masses was performed. Immunohistochemistry supported the diagnosis of a gastrointestinal stromal tumour (GIST) positive for CD34 and CD117. A diagnosis of a non islet cell tumour hypoglycaemia (NICTH) secondary to an IGF2 secreting GIST was confirmed with further biochemical investigations (IGF2=96.5 nmol/l; IGF2:IGF1 ratio 18.9, ULN <10). Treatment with growth hormone resolved the patient's hypoglycaemic symptoms and subsequent targeted therapy with Imatinib was successful in controlling disease progression over an 8-year observation period.

Learning points

  • NICTH can be a rare complication of GISTs that may manifest with severe hypoglycaemia and neuroglucopenic symptoms.

  • NICTH can masquerade as other pathologies thus causing diagnostic confusion.

  • Histological confirmation of GIST induced NICTH and exclusion of other conditions causing hypoglycaemia is essential.

  • Mutational analysis of GISTs should be carried out in all cases as it guides treatment decision.

  • Tailored management of hypoglycaemia, in this case using growth hormone and targeted cyto-reductive therapy, minimizes the risk of possible life-threatening complications.

Taxonomy:
Clinical Overview, Gland/Organ, Duodenum, Stomach, Topic, Endocrine-related cancer, Hormone, IGF2, Insulin, Condition/ Syndrome, Gastrointestinal stromal tumour, Hypoglycaemia, Non-islet-cell tumour hypoglycaemia, Patient Demographics, Age, Geriatric, Gender, Female, Ethnicity, White, Country of Treatment, United Kingdom, Diagnosis and Treatment, Signs and Symptoms, Facial palsy, Headache, Hepatomegaly, Hypoglycaemia, Myasthaenia, Paraesthesia, Pelvic mass, Ptosis, Investigation, Biopsy, C-peptide (blood), CT scan, Glucose (blood), IGF1, Immunohistochemistry, Insulin, Ultrasound-guided biopsy, Medication, GH, Glucocorticoids, Glucagon, Glucose, Imatinib, Prednisolone, Tyrosine-kinase inhibitors, Related Disciplines, Gastroenterology, Publication Details, Case Report Type, Unique/unexpected symptoms or presentations of a disease
DOI:
https://doi.org/10.1530/EDM-15-0062
Volume/Issue:
Volume 2015: Issue 1
Open access