Browse

You are looking at 1 - 10 of 39 items

Fumiaki Kawano Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Fumiaki Kawano in
Google Scholar
PubMed
Close
,
Tadato Yonekawa Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Tadato Yonekawa in
Google Scholar
PubMed
Close
,
Hideki Yamaguchi Neurology, Respirology, Endocrinology and Metabolism, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Hideki Yamaguchi in
Google Scholar
PubMed
Close
,
Nobuhiro Shibata Clinical Oncology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Nobuhiro Shibata in
Google Scholar
PubMed
Close
,
Kousei Tashiro Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Kousei Tashiro in
Google Scholar
PubMed
Close
,
Makoto Ikenoue Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Makoto Ikenoue in
Google Scholar
PubMed
Close
,
Shun Munakata Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Shun Munakata in
Google Scholar
PubMed
Close
,
Kazuhiro Higuchi Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Kazuhiro Higuchi in
Google Scholar
PubMed
Close
,
Hiroyuki Tanaka Diagnostic Pathology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Hiroyuki Tanaka in
Google Scholar
PubMed
Close
,
Yuichiro Sato Diagnostic Pathology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Yuichiro Sato in
Google Scholar
PubMed
Close
,
Ayumu Hosokawa Clinical Oncology, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Ayumu Hosokawa in
Google Scholar
PubMed
Close
,
Shinsuke Takeno Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Shinsuke Takeno in
Google Scholar
PubMed
Close
,
Kunihide Nakamura Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Kunihide Nakamura in
Google Scholar
PubMed
Close
, and
Atsushi Nanashima Departments of Surgery, Faculty of Medicine, University of Miyazaki, Miyazaki, Japan

Search for other papers by Atsushi Nanashima in
Google Scholar
PubMed
Close

Summary

A 54-year-old woman was referred to our hospital with a cervical tumor. CT revealed a cervical tumor extending to the upper mediastinum, tracheal deviation and tumor infiltration in the cervical vessels. She was followed-up because no diagnosis of malignancy was made by cytology. However, 2 months later, a CT scan showed enlargement of the tumor and tracheal stenosis, and a surgical biopsy was performed and she was diagnosed with anaplastic thyroid cancer (ATC). The tracheal tube with tracheal stenosis could not be removed due to the rapid growth of the tumor, necessitating management by mechanical ventilation. Due to the difficulty of surgical resection, she was treated with lenvatinib. A lenvatinib solution was made and administered via a nasogastric tube. After lenvatinib treatment, the tumor volume decreased and the tracheal stenosis improved. The tracheal tube was removed and oral intake became possible. She was discharged and received ambulatory lenvatinib therapy. The tumor was significantly reduced in size, but gradually grew and was exposed through the cervical wound 6 months later. Esophageal perforation occurred 10 months after the start of treatment. Lenvatinib was re-administered via a nasogastric tube. Eleven months later, the patient died of massive bleeding from the exposed cervical tumor. Patients with advanced ATC may require management with mechanical ventilation for airway stenosis or with a nasogastric tube for esophageal stenosis and perforation. We experienced a case in which lenvatinib was safely administered via a nasogastric tube while performing mechanical ventilation.

Learning points:

  • An anaplastic thyroid cancer patient under mechanical ventilator management was treated with lenvatinib via a nasogastric tube.

  • The lenvatinib solution can easily be prepared and administered via a nasogastric tube.

  • The lenvatinib solution was effective for a patient with difficulty in oral intake.

  • Lenvatinib could also improve the prognosis of an anaplastic thyroid cancer patient with severe airway and esophageal trouble.

Open access
Anna Luiza Galeazzi Rech Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

Search for other papers by Anna Luiza Galeazzi Rech in
Google Scholar
PubMed
Close
,
Yvon Stüve Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

Search for other papers by Yvon Stüve in
Google Scholar
PubMed
Close
,
Andreas Toepfer Kantonsspital Sankt Gallen, Klinik für Orthopädische Chirurgie und Traumatologie des Bewegungsapparts, Sankt Gallen, Switzerland

Search for other papers by Andreas Toepfer in
Google Scholar
PubMed
Close
, and
Katrin E Schimke Kantonsspital Sankt Gallen, Klinik für Allgemeine Innere Medizin/Hausarztmedizin, Sankt Gallen, Switzerland

Search for other papers by Katrin E Schimke in
Google Scholar
PubMed
Close

Summary

Acute Charcot neuropathic osteoarthropathy (CN) is a clinical entity which can easily go unrecognized in its acute early stages due to lack of awareness and unspecific presentation. However, missing early diagnosis can lead to severe complications. We present the case of a 72-year-old male patient who went through the natural course of the disease unnoticed before the very eyes of his physicians leading to a tragic end. We aim to raise awareness for this rare diabetic complication, emphasizing the necessity of early diagnosis and adequate, interdisciplinary treatment.

Learning points:

  • Clinical signs and symptoms of acute Charcot neuropathic osteoarthropathy (CN).

  • Red flags.

  • Importance of early diagnosis and correct treatment.

  • Diagnostic challenges of acute CN.

  • Awareness of high morbidity and mortality.

Open access
Daramjav Narantsatsral Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Daramjav Narantsatsral in
Google Scholar
PubMed
Close
,
Takagi Junko Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Takagi Junko in
Google Scholar
PubMed
Close
,
Iwayama Hideyuki Department of Pediatrics, Aichi Medical University School of Medicine, Nagakute, Japan

Search for other papers by Iwayama Hideyuki in
Google Scholar
PubMed
Close
,
Inukai Daisuke Department of Otolaryngology, Aichi Medical University School of Medicine, Nagakute, Japan

Search for other papers by Inukai Daisuke in
Google Scholar
PubMed
Close
,
Takama Hiroyuki Department of Dermatology, Aichi Medical University School of Medicine, Nagakute, Japan

Search for other papers by Takama Hiroyuki in
Google Scholar
PubMed
Close
,
Nomura Yuka Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Nomura Yuka in
Google Scholar
PubMed
Close
,
Hirase Syo Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Hirase Syo in
Google Scholar
PubMed
Close
,
Morita Hiroyuki Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Morita Hiroyuki in
Google Scholar
PubMed
Close
,
Otake Kazuo Division of Endocrinology and Metabolism, Department of Internal Medicine

Search for other papers by Otake Kazuo in
Google Scholar
PubMed
Close
,
Ogawa Tetsuya Department of Otolaryngology, Aichi Medical University School of Medicine, Nagakute, Japan

Search for other papers by Ogawa Tetsuya in
Google Scholar
PubMed
Close
, and
Takami Akiyoshi Department of Hematology, Aichi Medical University School of Medicine, Nagakute, Japan

Search for other papers by Takami Akiyoshi in
Google Scholar
PubMed
Close

Summary

Dupilumab an inhibitor of the interleukin (IL)-4R-alpha subunit is used for the treatment of allergic diseases. The patient was a 49-year-old man who received dupilumab for the treatment of severe atopic dermatitis. He presented hyperthyroidism with elevated thyroglobulin and anti-thyroid antibody negativity at 4 months after the initiation of therapy. On scintigraphy, the thyroid radioiodine uptake was low. Ultrasonography showed a diffuse hypoechoic area in the thyroid gland. A pathological study revealed lymphocytic infiltration. The administration of dupilumab was continued because of his atopic dermatitis that showed an excellent response. The patient`s hyperthyroidism changed to hypothyroidism 3 weeks later. Six months later his thyroid function normalized without any treatment. We herein describe the case of a patient with atopic dermatitis who developed painless thyroiditis under treatment with dupilumab. To the best of our knowledge, this is the first report of this event in the literature.

Learning points:

  • Dupilumab, a fully human monoclonal antibody that blocks interleukin-4 and interleukin-13, has been shown to be effective in the treatment atopic dermatitis and asthma with eosinophilia.

  • Painless thyroiditis is characterized by transient hyperthyroidism and hypothyroidism and recovery without anti-thyroid treatment.

  • This is the first report of painless thyroiditis as an adverse effect of dupilumab, although conjunctivitis and nasopharyngitis are the main adverse effects of dupilumab.

Open access
Mariana Barbosa Department of Endocrinology, Hospital de Braga, Braga, Portugal

Search for other papers by Mariana Barbosa in
Google Scholar
PubMed
Close
,
Sílvia Paredes Department of Endocrinology, Hospital de Braga, Braga, Portugal

Search for other papers by Sílvia Paredes in
Google Scholar
PubMed
Close
,
Maria João Machado Department of Neurosurgery, Hospital de Braga, Braga, Portugal

Search for other papers by Maria João Machado in
Google Scholar
PubMed
Close
,
Rui Almeida Department of Neurosurgery, Hospital de Braga, Braga, Portugal
Pituitary Consult, Hospital de Braga, Braga, Portugal

Search for other papers by Rui Almeida in
Google Scholar
PubMed
Close
, and
Olinda Marques Department of Endocrinology, Hospital de Braga, Braga, Portugal
Pituitary Consult, Hospital de Braga, Braga, Portugal

Search for other papers by Olinda Marques in
Google Scholar
PubMed
Close

Summary

Gonadotropin-releasing hormone (GnRH) agonists, currently used in the treatment of advanced prostate cancer, have been described as a rare cause of pituitary apoplexy, a potentially life-threatening clinical condition. We report the case of a 69-year-old man with a known pituitary macroadenoma who was diagnosed with prostate cancer and started treatment with GnRH agonist leuprorelin (other hormones were not tested before treatment). Few minutes after drug administration, the patient presented with acute-onset severe headache, followed by left eye ptosis, diplopia and vomiting. Pituitary MRI revealed tumor enlargement and T1-hyperintense signal, compatible with recent bleeding sellar content. Laboratory endocrine workup was significant for low total testosterone. The patient was managed conservatively with high-dose steroids, and symptoms significantly improved. This case describes a rare phenomenon, pituitary apoplexy induced by GnRH agonist. We review the literature regarding this condition: the pathophysiological mechanism involved is not clearly established and several hypotheses have been proposed. Although uncommon, healthcare professionals and patients should be aware of this complication and recognize the signs, preventing a delay in diagnosis and treatment.

Learning points:

  • Pituitary apoplexy (PA) is a potentially life-threatening complication that can be caused by gonadotropin-releasing hormone agonist (GnRHa) administration for the treatment of advanced prostate cancer.

  • This complication is rare but should be taken into account when using GnRHa, particularly in the setting of a known pre-existing pituitary adenoma.

  • PA presents with classic clinical signs and symptoms that should be promptly recognized.

  • Patients should be instructed to seek medical care if suspicious symptoms occur.

  • Healthcare professionals should be aware of this complication, enabling its early recognition, adequate treatment and favorable outcome.

Open access
Albert S Kim Department of Diabetes and Endocrinology, Westmead Hospital, Westmead, New South Wales, Australia
The University of Sydney, Faculty of Medicine and Health, Sydney, New South Wales, Australia

Search for other papers by Albert S Kim in
Google Scholar
PubMed
Close
,
Rashida Hakeem Department of Maternal-Fetal Medicine, Westmead Institute for Maternal-Fetal Medicine, Westmead Hospital, Westmead, New South Wales, Australia

Search for other papers by Rashida Hakeem in
Google Scholar
PubMed
Close
,
Azaliya Abdullah Department of Maternal-Fetal Medicine, Westmead Institute for Maternal-Fetal Medicine, Westmead Hospital, Westmead, New South Wales, Australia

Search for other papers by Azaliya Abdullah in
Google Scholar
PubMed
Close
,
Amanda J Hooper School of Medicine, Faculty of Health and Medical Sciences, University of Western Australia, Perth, Western Australia, Australia
Department of Clinical Biochemistry, PathWest Laboratory Medicine WA, Royal Perth Hospital and Fiona Stanley Hospital Network, Perth, Western Australia, Australia

Search for other papers by Amanda J Hooper in
Google Scholar
PubMed
Close
,
Michel C Tchan The University of Sydney, Faculty of Medicine and Health, Sydney, New South Wales, Australia
Department of Genetic Medicine, Westmead Hospital, Westmead, New South Wales, Australia

Search for other papers by Michel C Tchan in
Google Scholar
PubMed
Close
,
Thushari I Alahakoon The University of Sydney, Faculty of Medicine and Health, Sydney, New South Wales, Australia
Department of Maternal-Fetal Medicine, Westmead Institute for Maternal-Fetal Medicine, Westmead Hospital, Westmead, New South Wales, Australia

Search for other papers by Thushari I Alahakoon in
Google Scholar
PubMed
Close
, and
Christian M Girgis Department of Diabetes and Endocrinology, Westmead Hospital, Westmead, New South Wales, Australia
The University of Sydney, Faculty of Medicine and Health, Sydney, New South Wales, Australia
Department of Diabetes and Endocrinology, Royal North Shore Hospital, St Leonards, New South Wales, Australia

Search for other papers by Christian M Girgis in
Google Scholar
PubMed
Close

Summary

A 19-year-old female presented at 25-weeks gestation with pancreatitis. She was found to have significant hypertriglyceridaemia in context of an unconfirmed history of familial hypertriglyceridaemia. This was initially managed with fasting and insulin infusion and she was commenced on conventional interventions to lower triglycerides, including a fat-restricted diet, heparin, marine oil and gemfibrozil. Despite these measures, the triglyceride levels continued to increase as she progressed through the pregnancy, and it was postulated that she had an underlying lipoprotein lipase defect. Therefore, a multidisciplinary decision was made to commence therapeutic plasma exchange to prevent further episodes of pancreatitis. She underwent a total of 13 sessions of plasma exchange, and labour was induced at 37-weeks gestation in which a healthy female infant was delivered. There was a rapid and significant reduction in triglycerides in the 48 h post-delivery. Subsequent genetic testing of hypertriglyceridaemia genes revealed a missense mutation of the LPL gene. Fenofibrate and rosuvastatin was commenced to manage her hypertriglyceridaemia postpartum and the importance of preconception counselling for future pregnancies was discussed. Hormonal changes in pregnancy lead to an overall increase in plasma lipids to ensure adequate nutrient delivery to the fetus. These physiological changes become problematic, where a genetic abnormality in lipid metabolism exists and severe complications such as pancreatitis can arise. Available therapies for gestational hypertriglyceridaemia rely on augmentation of LPL activity. Where there is an underlying LPL defect, these therapies are ineffective and removal of triglyceride-rich lipoproteins via plasma exchange should be considered.

Learning points:

  • Hormonal changes in pregnancy, mediated by progesterone,oestrogen and human placental lactogen, lead to a two- to three-fold increase in serum triglyceride levels.

  • Pharmacological intervention for management of gestational hypertriglyceridaemia rely on the augmentation of lipoprotein lipase (LPL) activity to enhance catabolism of triglyceride-rich lipoproteins.

  • Genetic mutations affecting the LPL gene can lead to severe hypertriglyceridaemia.

  • Therapeutic plasma exchange (TPE) is an effective intervention for the management of severe gestational hypertriglyceridaemia and should be considered in cases where there is an underlying LPL defect.

  • Preconception counselling and discussion regarding contraception is of paramount importance in women with familial hypertriglyceridaemia.

Open access
Aishah Ekhzaimy Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Aishah Ekhzaimy in
Google Scholar
PubMed
Close
,
Afshan Masood Obesity Research Center, and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Afshan Masood in
Google Scholar
PubMed
Close
,
Seham Alzahrani Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Seham Alzahrani in
Google Scholar
PubMed
Close
,
Waleed Al-Ghamdi Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Waleed Al-Ghamdi in
Google Scholar
PubMed
Close
,
Daad Alotaibi Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Daad Alotaibi in
Google Scholar
PubMed
Close
, and
Muhammad Mujammami Department of Medicine and College of Medicine, King Saud University, Riyadh, Saudi Arabia

Search for other papers by Muhammad Mujammami in
Google Scholar
PubMed
Close

Summary

Central diabetes insipidus (CDI) and several endocrine disorders previously classified as idiopathic are now considered to be of an autoimmune etiology. Dermatomyositis (DM), a rare autoimmune condition characterized by inflammatory myopathy and skin rashes, is also known to affect the gastrointestinal, pulmonary, and rarely the cardiac systems and the joints. The association of CDI and DM is extremely rare. After an extensive literature search and to the best of our knowledge this is the first reported case in literature, we report the case of a 36-year-old male with a history of CDI, who presented to the hospital’s endocrine outpatient clinic for evaluation of a 3-week history of progressive facial rash accompanied by weakness and aching of the muscles.

Learning points:

  • Accurate biochemical diagnosis should always be followed by etiological investigation.

  • This clinical entity usually constitutes a therapeutic challenge, often requiring a multidisciplinary approach for optimal outcome.

  • Dermatomyositis is an important differential diagnosis in patients presenting with proximal muscle weakness.

  • Associated autoimmune conditions should be considered while evaluating patients with dermatomyositis.

  • Dermatomyositis can relapse at any stage, even following a very long period of remission.

  • Maintenance immunosuppressive therapy should be carefully considered in these patients.

Open access
Maria Tomkins Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

Search for other papers by Maria Tomkins in
Google Scholar
PubMed
Close
,
Roxana Maria Tudor Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

Search for other papers by Roxana Maria Tudor in
Google Scholar
PubMed
Close
,
Diarmuid Smith Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

Search for other papers by Diarmuid Smith in
Google Scholar
PubMed
Close
, and
Amar Agha Department of Endocrinology and Diabetes, Beaumont Hospital Dublin, Dublin, Ireland

Search for other papers by Amar Agha in
Google Scholar
PubMed
Close

Summary

This case is the first to describe a patient who experienced concomitant agranulocytosis and anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis as an adverse effect of propylthiouracil treatment for Graves’ disease. A 42-year-old female with Graves’ disease presented to the emergency department (ED) with a 2-week history of fevers, night sweats, transient lower limb rash, arthralgia, myalgia and fatigue. She had been taking propylthiouracil for 18 months prior to presentation. On admission, agranulocytosis was evident with a neutrophil count of 0.36 × 109/L and immediately propylthiouracil was stopped. There was no evidence of active infection and the patient was treated with broad-spectrum antibodies and one dose of granulocyte colony-stimulation factor, resulting in a satisfactory response. On further investigation, ANCAs were positive with dual positivity for proteinase 3 and myeloperoxidase. There was no evidence of end-organ damage secondary to vasculitis, and the patient’s constitutional symptoms resolved completely on discontinuation of the drug precluding the need for immunosuppressive therapy.

Learning points:

  • Continued vigilance and patient education regarding the risk of antithyroid drug-induced agranulocytosis is vital throughout the course of treatment.

  • ANCA-associated vasculitis is a rare adverse effect of antithyroid drug use.

  • Timely discontinuation of the offending drug is vital in reducing end-organ damage and the need for immunosuppressive therapy in drug-induced ANCA-associated vasculitis.

  • Similarities in the pathogenesis of agranulocytosis and drug-induced ANCA-associated vasculitis may offer insight into an improved understanding of vasculitis and agranulocytosis.

Open access
Karen Decaestecker Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

Search for other papers by Karen Decaestecker in
Google Scholar
PubMed
Close
,
Veerle Wijtvliet Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

Search for other papers by Veerle Wijtvliet in
Google Scholar
PubMed
Close
,
Peter Coremans Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

Search for other papers by Peter Coremans in
Google Scholar
PubMed
Close
, and
Nike Van Doninck Department of Diabetology-Endocrinology, AZ Nikolaas, Sint-Niklaas, Belgium

Search for other papers by Nike Van Doninck in
Google Scholar
PubMed
Close

Summary

ACTH-dependent hypercortisolism is caused by an ectopic ACTH syndrome (EAS) in 20% of cases. We report a rare cause of EAS in a 41-year-old woman, presenting with clinical features of Cushing’s syndrome which developed over several months. Biochemical tests revealed hypokalemic metabolic alkalosis and high morning cortisol and ACTH levels. Further testing, including 24-hour urine analysis, late-night saliva and low-dose dexamethasone suppression test, confirmed hypercortisolism. An MRI of the pituitary gland was normal. Inferior petrosal sinus sampling (IPSS) revealed inconsistent results, with a raised basal gradient but no rise after CRH stimulation. Additional PET-CT showed intense metabolic activity in the left nasal vault. Biopsy of this lesion revealed an unsuspected cause of Cushing’s syndrome: an olfactory neuroblastoma (ONB) with positive immunostaining for ACTH. Our patient underwent transnasal resection of the tumour mass, followed by adjuvant radiotherapy. Normalisation of cortisol and ACTH levels was seen immediately after surgery. Hydrocortisone substitution was started to prevent withdrawal symptoms. As the hypothalamic–pituitary–axis slowly recovered, daily hydrocortisone doses were tapered and stopped 4 months after surgery. Clinical Cushing’s stigmata improved gradually.

Learning points:

  • Ectopic ACTH syndrome can originate from tumours outside the thoracoabdominal region, like the sinonasal cavity.

  • The diagnostic accuracy of IPSS is not 100%: both false positives and false negatives may occur and might be due to a sinonasal tumour with ectopic ACTH secretion.

  • Olfactory neuroblastoma (syn. esthesioneuroblastoma), named because of its sensory (olfactory) and neuroectodermal origin in the upper nasal cavity, is a rare malignant neoplasm. It should not be confused with neuroblastoma, a tumour of the sympathetic nervous system typically occurring in children.

  • If one criticises MRI of the pituitary gland because of ACTH-dependent hypercortisolism, one should take a close look at the sinonasal field as well.

Open access
Misaki Aoshima Departments of Endocrinology Diabetes and Metabolism, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Misaki Aoshima in
Google Scholar
PubMed
Close
,
Koji Nagayama Departments of Endocrinology Diabetes and Metabolism, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Koji Nagayama in
Google Scholar
PubMed
Close
,
Kei Takeshita Departments of Endocrinology Diabetes and Metabolism, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Kei Takeshita in
Google Scholar
PubMed
Close
,
Hiroshi Ajima Departments of Endocrinology Diabetes and Metabolism, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Hiroshi Ajima in
Google Scholar
PubMed
Close
,
Sakurako Orikasa Departments of Endocrinology Diabetes and Metabolism, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Sakurako Orikasa in
Google Scholar
PubMed
Close
,
Ayana Iwazaki ²Departments of Endocrinology Diabetes and Metabolism, Seirei Hamamatsu General Hospital, Hamamatsu, Shizuoka, Japan

Search for other papers by Ayana Iwazaki in
Google Scholar
PubMed
Close
,
Hiroaki Takatori Department of Rheumatology, Hamamatsu Medical Center, Hamamatsu, Shizuoka, Japan

Search for other papers by Hiroaki Takatori in
Google Scholar
PubMed
Close
, and
Yutaka Oki Department of Family and Community Medicine, Hamamatsu University School of Medicine, Hamamatsu, Shizuoka, Japan

Search for other papers by Yutaka Oki in
Google Scholar
PubMed
Close

Summary

Patients treated with immunosuppressive drugs, especially methotrexate (MTX), rarely develop lymphoproliferative disorders (LPDs), known as MTX-related LPD (MTX–LPD). The primary site of MTX–LPD is often extranodal. This is the first reported case of MTX–LPD in the pituitary. A 65-year-old woman was admitted to our hospital with symptoms of oculomotor nerve palsy and multiple subcutaneous nodules. She had been treated with MTX for 11 years for rheumatoid arthritis. Computed tomography showed multiple masses in the orbit, sinuses, lung fields, anterior mediastinum, kidney, and subcutaneous tissue. Brain magnetic resonance imaging revealed a sellar mass. She was diagnosed with hypopituitarism and central diabetes insipidus based on endocrine examination. Although pituitary biopsy could not be performed, we concluded that the pituitary lesion was from MTX–LPD, similar to the lesions in the sinuses, anterior mediastinum, and subcutaneous tissue, which showed polymorphic LPD on biopsy. MTX was discontinued, and methylprednisolone was administered to improve the neurologic symptoms. After several weeks, there was marked improvement of all lesions, including the pituitary lesion, but the pituitary function did not improve. When pituitary lesions are caused by MTX–LPD, the possibility of anterior hypopituitarism and central diabetes insipidus needs to be considered. Further studies are needed to investigate the effectiveness of early diagnosis and treatment of MTX–LPD in restoring pituitary dysfunction.

Learning points

  • Pituitary lesions from MTX–LPD may cause hypopituitarism and central diabetes insipidus.

  • Pituitary metastasis of malignant lymphoma and primary pituitary lymphoma, which have the same tissue types with MTX–LPD, have poor prognosis, but the lesions of MTX–LPD can regress only after MTX discontinuation.

  • In cases of pituitary lesions alone, a diagnosis of MTX–LPD may be difficult, unless pituitary biopsy is performed. This possibility should be considered in patients treated with immunosuppressive drugs.

  • Pituitary hypofunction and diabetes insipidus may persist, even after regression of the lesions on imaging due to MTX discontinuation.

Open access
Punith Kempegowda Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK
University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Punith Kempegowda in
Google Scholar
PubMed
Close
,
Eka Melson Institute of Metabolism and Systems Research, University of Birmingham, Birmingham, UK
University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Eka Melson in
Google Scholar
PubMed
Close
,
Gerald Langman University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Gerald Langman in
Google Scholar
PubMed
Close
,
Fady Khattar University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Fady Khattar in
Google Scholar
PubMed
Close
,
Muhammad Karamat University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Muhammad Karamat in
Google Scholar
PubMed
Close
, and
Quratul-Ain Altaf University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK

Search for other papers by Quratul-Ain Altaf in
Google Scholar
PubMed
Close

Summary

Diabetic myonecrosis, also known as diabetic muscle infarction is a rare complication of diabetes mellitus usually associated with longstanding suboptimal glycaemic control. Although theories of atherosclerosis, diabetic microangiopathy, vasculitis, ischaemia-reperfusion injury and hypercoagulable state have been proposed to explain the pathophysiology, none of these have been able to individually explain the pathophysiology in entirety. Diabetic renal disease is the most common risk factor for developing DMN and its recurrence. The diagnosis is often missed due to lack of awareness and the presentation mimicking other conditions associated with DM. The routine laboratory investigations are often non-specific and do not provide much value in the diagnosis as well. Muscle biopsy can provide a definite diagnosis but is not currently recommended due to its invasiveness and association with prolonged time to symptoms resolution. Magnetic resonance imaging, in combination with classic history and risk factors can clinch the diagnosis. Treatment is generally analgesia and rest, although the former’s use may be limited in the presence of renal disease.

Learning points:

  • Diabetic myonecrosis is a rare complication of diabetes mellitus associated with longstanding suboptimal glycaemic control.

  • Diabetic renal disease is a known risk factor, although the evidence is merely observational.

  • Although muscle biopsy could provide a definite diagnosis, it is not recommended as it can prolong the disease process and should be reserved only for cases not responding to conventional treatment.

  • Typical MRI findings in combination with classic symptoms and risk factors can clinch the diagnosis

  • Current treatment recommendations include NSAIDs and/or aspirin (if not contraindicated) alongside bed rest. Physiotherapy is not recommended in the acute phase but should be started as soon as patient is discharged from hospital.

  • Optimal glycaemic control is key to prevent recurrence.

Open access