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All Journal International Journal of Electrical and Computer Engineering Medical Journal of Indonesia Journal of the Indonesian Medical Association : Majalah Kedokteran Indonesia World Nutrition Journal eJournal Kedokteran Indonesia Jurnal Penyakit Dalam Indonesia Indonesian Journal of Endocrinology Metabolism and Diabetes (InaJEMD)
Tri Juli Edi Tarigan
Division Of Endocrinology, Metabolic, And Diabetes, Department Of Internal Medicine, Faculty Of Medicine, Universitas Indonesia/Cipto Mangun Kusumo General Hospital, Jakarta, Indonesia
Biochemistry, Genetics & Molecular Biology Computer Science & IT Education Electrical & Electronics Engineering Health Professions Immunology & microbiology Medicine & Pharmacology Neuroscience Public Health

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Endocrine Complications in Langerhans Cell Histiocytosis: A Case of Empty Sella Syndrome with Hypopituitarism Rizqi Rifani; Tri Juli Edi Tarigan
‎ InaJEMD - Indonesian Journal of Endocrinology Metabolic and Diabetes Vol. 2 No. 2 (2025): InaJEMD Vol. 2, No. 2
Publisher : PP PERKENI

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.66266/inajemd.v2i2.61

Abstract

Langerhans cell histiocytosis (LCH) is a rare clonal myeloid neoplasia characterized by infiltration of CD1a+/CD207+ dendritic cells, frequently affecting bones, skin, and the central nervous system. Endocrine complications, particularly involving the hypothalamic-pituitary axis, are common in multisystem disease and may result in irreversible dysfunction, impacting growth, development, and overall quality of life. Case Presentation: An 18-year-old male diagnosed with LCH at age 10 presenting with polyuria, polydipsia, and delayed puberty. He was found to have central diabetes insipidus, central hypothyroidism, hypogonadotropic hypogonadism, and a grade IV empty sella on brain MRI. The patient had a history of bone and soft tissue involvement and underwent chemotherapy for multisystem LCH. His current therapy includes desmopressin, levothyroxine, vitamin D, calcium supplementation, and planned to given testosterone replacement. This case illustrates the classic progression of endocrine complications in LCH with hypothalamic-pituitary axis involvement. Central diabetes insipidus is often the first manifestation followed by anterior pituitary hormone deficiencies. The finding of total empty sella on MRI likely reflects chronic inflammatory damage or pituitary atrophy. The combination of central diabetes insipidus (CDI), central hypothyroidism, and delayed puberty requires lifelong hormonal replacement and regular endocrine follow-up. The case also highlights the importance of addressing bone health and growth delays secondary to hormonal deficiencies and previous glucocorticoid therapy. Early identification and management of endocrine complications in LCH, particularly those involving the pituitary are essential to reduce morbidity and improve patient outcomes. Lifelong monitoring and multidisciplinary care are necessary for optimal management of LCH survivors.
Mitigating Hungry Bone Syndrome: Case Reports on Best Practices After Parathyroidectomy Sarah Firdausa; Luki Kusumaningtyas; Dicky L Tahapary; Wismandari Wisnu; Tri Juli Edi Tarigan
‎ InaJEMD - Indonesian Journal of Endocrinology Metabolic and Diabetes Vol. 2 No. 2 (2025): InaJEMD Vol. 2, No. 2
Publisher : PP PERKENI

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.66266/inajemd.v2i2.65

Abstract

Severe hyperparathyroidism caused by prolonged high levels of parathyroid hormone (PTH) can be managed by removing the gland. One of the critical complications related to parathyroidectomy is hungry bone syndrome (HBS), an emergency morbidity which may be fatal if not promptly and adequately managed. HBS is defined by a rapid and profound decline in serum calcium levels following surgery, as the bones avidly uptake calcium and phosphate in the absence of high PTH levels. It may present as worsened bone pain, carpopedal spasm, severe hypocalcemia, hypophosphatemia, and hypomagnesemia. This report highlights two patients who underwent parathyroidectomy and had different postoperative outcomes for HBS. The first case involves a 19-year-old male who had a history of recurrent fractures and bone pain. In 2019, he got a fracture from a fall, and in 2020, he experienced another fall leading to shoulder dislocation and further fractures. By late 2021, he was diagnosed with severe hyperparathyroidism due to parathyroid adenoma and the gland was removed. Two days post parathyroidectomy, he developed HBS. He was treated with calcium and vitamin D supplementation. Over two years of follow-ups, his bone density and mobility improved significantly. The second case involves a 46-year-old male with uncontrolled hypertension and chronic kidney disease stage 5 on hemodialysis, presenting with bone pain and deformities. This patient had a long-standing history of bone pain and fractures. He underwent a similar surgical intervention for tertiary hyperparathyroidism but did not develop HBS postoperatively. Careful perioperative monitoring of electrolytes, vigorous supplementation of calcium and vitamin D, and the use of antiresorptive therapies before surgery had been employed. These cases underline the variety of postoperative outcomes and the importance of tailored management strategies. Early intervention, appropriate surgical management, and aggressive postoperative supplementation are crucial to prevent and manage HBS in patients with severe hyperparathyroidism. Multidisciplinary approach and the utilization of various imaging modalities and intraoperative PTH monitoring are mandatory in managing such complex cases. Applying these approaches will reduce the risk of HBS while guaranteeing excellent postoperative care for individuals following parathyroidectomy.
Detection of Small Lesion Insulinoma with 68Ga-DOTATATE PET/CT scan: A Case Report Irma Wahyuni; Tri Juli Edi Tarigan; Hendra Budiawan; Alvita Dewi; Rian Hidayatullah; Hasan Maulahela
‎ InaJEMD - Indonesian Journal of Endocrinology Metabolism and Diabetes Vol. 2 No. 1 (2025): InaJEMD Vol. 2, No. 1
Publisher : PP PERKENI

Show Abstract | Download Original | Original Source | Check in Google Scholar | DOI: 10.66266/inajemd.v2i1.40

Abstract

Endogenous hyperinsulinism is a rare condition characterized by inappropriate insulin secretion, with insulinoma accounting for approximately 55% of cases. A 36-year-old woman presented with recurrent hypoglycemic episodes for 4.5 years, particularly in the morning, relieved by glucose intake. Laboratory findings during a supervised fasting test revealed a blood glucose of 44 mg/dL, elevated insulin (15 μIU/mL), C-peptide (2.27 ng/mL), proinsulin (53.1 pmol/L), and suppressed beta-hydroxybutyrate (0.1 mmol/L), indicating endogenous hyperinsulinemia. Initial imaging modalities including CT, MRI, and EUS failed to localize the lesion. The patient discontinued investigation but resumed a year later with nuclear imaging studies. A 99mTc-HYNIC-TOC scan was inconclusive, but subsequent ^68Ga-DOTATATE PET/CT at Hasan Sadikin Hospital revealed a somatostatin receptor-expressing nodule at the pancreatic head, consistent with insulinoma. Given the lesion's location and imaging findings, treatment options were discussed, including Whipple’s procedure versus radiofrequency ablation. Insulinomas are typically small, with 80% under 2 cm and 40% under 1 cm, often making localization challenging. While CT and MRI have detection rates of 70% and 86% respectively, the combination of CT and EUS may reach 100% sensitivity. In cases where conventional imaging is inconclusive, ^68Ga-DOTATATE PET/CT provides a valuable alternative, capable of detecting neuroendocrine tumors as small as 6 mm. This case highlights the critical role of nuclear medicine in localizing elusive insulinomas and guiding definitive treatment.
Co-Authors Adeputri Tanesha Idhayu Adhiatma, Kresna Agnes Stephanie Harahap Alvita Dewi Arif Mansjoer Assyarify, Hanif Cleopas Martin Rumende Dewi Friska Dicky L Tahapary E. Mudjaddid A. Siswanto Deddy N.W.Achadiono Hamzah Shatri Eka Susanto Em Yunir, Em Fatya, Atikah Isna Hadi, Dwi Rendra Hardigaloeh, Amanda Trixie Hasan Maulahela Hendra Budiawan Ida Gunawan Ikhwan Rinaldi Ilham Utama Surya Ira Laurentika Iris Rengganis Irma Wahyuni Iswati, Eni Kanadi Sumapradja Kuntjoro Harimurti Luki Kusumaningtyas Lusiani Lusiani, Lusiani Maruhum B.H. Marbun Muhammad Hanif Nadhif Muhammad Irsyad Nugroho, Setyo Widi Nury Nusdwinuringtya Nury Nusdwinuringtya Nyoman Murdana Nyoman Murdana Pangestuti, Mugi Rahayu Pringgodigdo Nugroho, Pringgodigdo Purwitasari Darmaputri Purwitasari Darmaputri Rian Hidayatullah Rizqi Rifani Robert Shinto Rudi Putranto Sarah Firdausa Sarumpaet, Angela Tahapary, Dicky Taufik Agung Trismiyanti Trismiyanti Wismandari Wisnu