CLINICAL CASE LETTERS

perpetual remodelling which allows lead to be regularly re- introduced into the blood stream [6]. Chelation therapy brings down the blood lead levels acutely only to rebound within weeks to months after treatment. Often, repeated courses of chelation are required [5]. This case report emphasizes the need for long term follow- up with periodic monitoring of lead levels in children with chronic lead poisoning to assess the need for repeated chelation therapy. Blood lead concentration may rise to toxic levels even after removal of exposure due to constant re-distribution in a growing child.

SAIDALIKUTTY FOUZEAMOL,* SHEIK SHAHANA PARVIN AND RAMALINGAM RAMAKRISHNAN TIRUPPUR CHINNAPPAN Department of Pediatrics, G Kuppuswamy Naidu Memorial Hospital, Coimbatore, Tamil Nadu, India. Fig. 1 X-ray left knee of the index patient showing lead lines *[email protected] (arrows) over lower end of femur and upper end of tibia. REFERENCES 1. Wani AL, Ara A, Usmani JA. Lead toxicity: A review. Interdiscip Toxicol. 2015;8:55-64. has been stable except for a few bouts of anger outbursts for 2. Keosaian J, Venkatesh T, D’Amico S, Gardiner P, Saper R. which he is on follow up with child psychiatrist. He is on close Blood lead levels of children using traditional Indian follow-up and may require further chelation. medicine and cosmetics: A feasibility study. Glob Adv Lead is not known to serve any significant biological Health Med. 2019;8:1-6. function and deposition does not spare any organ in the body 3. Krishnaswamy K, kumar BD. Lead toxicity. Indian Pediatr. [1]. It has high affinity for the skeleton and chronic exposure 1998;35:209-16. often sequesters large proportion in the bones followed by the 4. Pounds JG, Long GJ, Rosen JF. Cellular and molecular kidneys [4]. After a period of initial exposure lead is toxicity of lead in bone. Environ Health Perspect. 1991;91: redistributed to the soft tissues. If cessation of exposure occurs 17-32. at this juncture, there is a decrease in the blood lead levels post 5. Hu H, Pepper L, Goldman R. Effect of repeated the initial rise [5]. Bone, being a dynamic tissue, undergoes occupational exposure to lead, cessation of exposure and remodelling throughout life which is regulated by a wide range of chelation on levels of lead in bone. Am J Ind Med. and local availability factors. Prolonged exposure also 1991;20:723-35. results in slow release of lead from the bone stores over a 6. Barbosa Jr F, Tanus- Santos JE, Gerlach RF, Parsons PJ. A protracted period of time [4]. Children are at high risk of lead critical review of biomarkers used for monitoring human poisoning as they are in a state of constant growth and exposure to lead: Advantages, limitations, and future needs. development. Moreover, the growing bones in children undergo Environ Health Perspect. 2005;113:1669-74.

Autoimmune in Systemic Herein, we report a rare case of in a male child who presented with features of central and diabetes Lupus Erythematosus insipidus that was diagnosed as SLE-associated AH. He was treated with pulse methylpre-dnisolone and cyclophosphamide with replacement. A 14-year-old male child, fourth issue of a non- (AH) is a rare consanguineous marriage was admitted with history of fever, that occurs when the is infiltrated with weight loss, pallor and generalized weakness since one month. lymphocytes and plasma cells, leading to impaired hormonal There was no history of rash, bleeding manifestations, secretion. Rare cases of association of systemic lupus erythe- abdominal distension, night sweats, oral ulcers, icterus, matosus (SLE) with AH have been reported in literature but headaches, visual disturbances or joint swelling. He had received mainly in adult population. AH commonly involves anterior multiple oral antibiotics with no improvement. In the past, he pituitary; labelled as lymphocytic adenohypophysitis (LAH) had suffered a stroke at ten years of age with MRI brain showing but it can also involve which is called acute lacunar infarct in right corona radiata. Birth history was lymphocytic infundibulo-neurohypophysitis (LINH) [1-4]. uneventful and he was immunized as per schedule.

INDIAN PEDIATRICS 84 VOLUME 58__JANUARY 15, 2021 CLINICAL CASE LETTERS

Anthropometric parameters and vitals, including blood posterior pituitary bright spot on T1-weighted MRI. The gold pressure, were normal. Clinical examination revealed malar rash, standard of diagnosis is pituitary biopsy which reveals massive oral ulcers, severe pallor with moderate hepato-splenomegaly. infiltration of lymphocytes and plasma cells in the pituitary Laboratory investigations revealed anemia (Hb, 6.4 g/dL), direct gland but is usually denied by patients due to its invasive nature. Coombs test (DCT) positive, normal white blood cell (WBC) Our patient is similar to the case reported by Jing, et al. [4] in counts (WBC, 7.9×109/L, Neutrophils 49%, Lymphocyctes which a 15 year lupus child revealed low levels of sex hormones, 51%), thrombocytopenia (Platelet count, 30,000/cmm), raised hormones and serum cortisol with MRI of pituitary ESR (81 mm at end of one hour), prolonged activated partial region demonstrating an enlargement of the pituitary stalk. She thromboplastin time (APTT) [Test, 54 sec (26.9-36.3)], high was diagnosed as LINH associated with SLE and responded spot urine protein creatinine ratio (0.9, normal <0.2) with well to glucocorticoids and cyclophosphamide [4]. We treated normal liver enzymes, serum electrolytes and X-Ray chest. our patient with steroids, cyclophosphamide, hydroxy-chloro- Immunological investigations showed strongly positive anti- quine, hormone replacement and warfarin with significant nuclear antibody (ANA titres 1:2560, speckled pattern), low clinical improvement in constitutional symptoms, normali- serum complement C3 (C3-60 mg/dL; normal range 82-173 mg/ zation of acute phase reactants, complement levels, thyroid dL) and C4 (C4 11.2 mg/dL; normal range: 13-46 mg/dL), function and urine proteinuria at 3 months of follow up. positive anti-cardiolipin IgM antibody, beta-2 glycoprotein Warfarin was added to treatment protocol considering positive IgM antibody and lupus anti-coagulant. Anti-double stranded antiphospholipid antibody and history of prior lacunar infarct. DNA antibody and anti-Smith (anti-Sm) antibody were To the best of our knowledge this is the first case reported from negative. Ophthalmology examination showed retinal India with features of AH in a case of juvenile SLE. hemorrhages. Thyroid function test revealed central Our case demonstrates association of SLE and AH in hypothyroidism [low free T3 (<1 pg/mL), low free T4 (0.46 ng/ children. In the presence of central hypothyroidism and dL), and low TSH (<0.01uIU/mL)], with positive anti-thyroid in a lupus patient, endocrine hormonal peroxidase (anti-TPO) antibodies. During the hospital stay, evaluation and an MRI of pituitary gland is warranted to rule child started developing delirium and agitation along with out AH. polyuria. Serum osmolality was high (320 mOsm/kg). Magnetic resonance imaging (MRI) showed absence of posterior pituitary SUNIL KAPUR,1 JITENDRA OSWAL,1* RAHUL JAHAGIRDAR2 bright spot in T1-weighted imaging consistent with diabetes AND VIJAY VISWANATHAN1 insipidus. Serum cortisol, , and sex hormone levels 1Pediatric Rheumatology and 2Endocrinology, were within normal limit. Parents did not consent for renal Department of Pediatrics, biopsy. Although desirable, IgG4-related disease (IgG4-RD) Bharati Vidyapeeth University Medical College Hospital and workup could not be done due to financial constraints. A Research Centre, Pune, Maharashtra, India. diagnosis of SLE with multiple organ involvement with AH was *[email protected] made. He was initiated on intravenous pulse REFERENCES methylprednisolone and cyclophosphamide followed by oral steroids, monthly cyclophosphamide, hydroxychloroquine, 1. Kawano T, Kobayashi S, Ebizuka T, et al. warfarin, thyroid replacement and oral desmopressin, with a associated with empty sella after steroid pulse therapy in a close follow up. patient with SLE. Ryumachi. 1994;34:54-8. 2. Katano H, Umemura A, Kamiya K, Kanai H, Yamada K. In our patient, SLE was diagnosed based on constitutional Visual disturbance by lymphocytic hypophysitis in a non- symptoms, malar rash, oral ulcers, thrombocytopenia with pregnant woman with systemic lupus erythematosus. Lu- auto-immune hemolytic anemia (Evans syndrome), low WBC pus. 1998;7:554-6. counts, high ESR, ANA positivity, low complement levels, 3. Hashimoto K, Asaba K, Tamura K, Takao T, Nakamura T. positive antiphospholipid antibody, and high urine protein A case of lymphocytic hypophysitis associated with sys- creatinine ratio. AH is a rare disease, mainly affecting females temic lupus erythematousus. Endocr J. 2002;49:605-10. though in our case it was a male. It has incidence reported to be 4. Jing E, Li Cao, Hui Wang, et al. A teenager diagnosed as lym- 1 in 9 million [5] but the actual number may be more, phocytic hypophysitis associated with systemic lupus particularly as IgG4-RD and involvement of the hypophysis by erythematosus (SLE). J Blood Disorders Transf. systemic pathologies has increasingly been recognised [6]. AH 2014;5:221. can be primary (idiopathic) or secondary to sella and parasella 5. Caturegli P, Newschaffer C, Olivi A, Pomper MG, Burger lesions, systemic diseases, or drugs (mainly immune checkpoint PC, Rose NR. Autoimmune hypophysitis. Endocr Rev. inhibitors). The predominant feature of LINH is central diabetes 2005;26:599-614. insipidus which was present in our patient. The diagnosis of AH 6. Joshi MN, Whitelaw BC, Carroll PV. Mechanisms in endo- was made when our patient showed features of central crinology: Hypophysitis: Diagnosis and treatment. Eur J hypothyroidism and diabetes insipidus with loss of normal Endocrinol. 2018;179:151-63.

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