scholarly journals Remitting Seronegative Symmetrical Synovitis With Subsequent IgA Hypergammaglobulinemia. A Case of Chronic Inflammation Causing Macroprolactinemia and a Misdiagnosis

2021 ◽  
Vol 5 (Supplement_1) ◽  
pp. A607-A608
Author(s):  
Paria Zarghamravanbakhsh ◽  
Jose Gonzalo Sanchez

Abstract Prolactin is a pituitary hormone that functions in breast development and milk production in women and also plays an important role in immunoregulation and human immune responses, including autoimmune diseases. Macroprolactin, known as “big-big prolactin”, is due to the presence of marked hyperprolactinemia associated with evidence of prolactin-Ig (typically IgG4 or less frequently IgA) circulating complexes. We describes a case of 51 year-old female, with more than a 4 year history of reported hyperprolcatinemia who was treated with Cabergoline 0.5 mg weekly. Prior to treatment, she reported menses every 30-40 days, but denied galactorrhea or symptoms of sellar mass effect. Our patient had mildly elevated prolactin levels of 40-50 ng/dl. Her Thyroid function test were within normal limits. Patient had two pituitary MRIs in 2017 and 2019 which did not show sellar abnormalities. Prior to cabergolibe initiation, she was diagnosed with RS3PE syndrome (Remitting seronegative symmetrical synovitis with pitting edema) due to bilateral swelling in the dorsum of her hands. She was found to have hypergammaglobulinemia which was related to IgA elevation from chronic inflammation. Further investigation showed actual bio-active monomeric prolactin level was normal (4.8 ng/dl) and macroprolactin elevation from hypergammagolbulinemia. Before diagnosing her paraproteinemia and her macroprolactin predominance, she had received years of dopamine agonist therapy which was discontinued after diagnosis. We report a novel association of IgA predominant hypergammaglobulinemia from a chronic rheumatologic condition, leading to a misdiagnosed hyperprolcatinemia. Care should be taken to determine monomeric Prolactin levels prior to treatment, specially when symptoms are equivocal and/or imaging studies are negative.

2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Varshini Chakravarthy ◽  
Sehar Ejaz

Abstract Background: Swyer Syndrome is an extremely rare disorder of sexual development. These patients often present with primary amenorrhea during adolescence and are phenotypically female with 46 XY chromosomes. Given the association of invasive gonadal malignancies with this disorder, suspicion should be high in patients who present with a stagnant or decreased rate of pubertal progression. We present a case of Swyer Syndrome in a 14-year-old female with primary amenorrhea in the setting of decreased pubertal progression. Case: A 14-year-old female presents with a chief complaint of primary amenorrhea. She first noticed breast budding 2 years prior but reports no significant increase in breast tissue over the last 2 years. She does not appreciate any other signs of puberty. She denies any acne, body odor, hirsutism, hair loss, or abdominal/pelvic pain. She denies any changes in her diet or physical activity and is not on any medication. No history of cancer, surgeries, or radiation exposure. There is no family history of infertility or delayed puberty. Her vitals on presentation are within normal limits. Her growth parameters are the following: weight-69.9 kilos, height-163 cm, and BMI-26.3. Physical exam shows a well-appearing adolescent with grossly female external genitalia and the breast exam is SMR II. No pubic or axillary hair appreciated on the exam. Although our patient did not meet the traditional definition of primary amenorrhea, a workup was started due to the slow progression of puberty. Initial blood testing shows normal blood count, electrolytes and thyroid levels. DHEA-S androstenedione, free and total testosterone were all within normal limits. Further results such as LH (25.4 uIU/mL), FSH (56.5 mIU/mL) and estradiol (22 pg/mL) along with low levels of AMH (0.52 ng/mL) and inhibin A (1pg/mL) confirms suspicion for ovarian insufficiency. Chromosomal analysis and pelvic ultrasound findings of a small uterus and ovaries led to our diagnosis of Swyer syndrome. Our patient had surgical resection of both ovaries and fallopian tubes and the ovarian pathology showed gonadoblastoma with invasive dysgerminoma in both gonads. She was started on hormone replacement after gonadectomy. Conclusion: Although Swyer syndrome is uncommon with an incidence of 1 in 80,000, this case illustrates that suspicion for Swyer Syndrome should be high in patients with slow progression of puberty and primary amenorrhea (1). Early diagnosis is critical, as patients with gonadal dysgenesis are at great risk for germ cell cancers. Though most of these patients have an identifiable genetic mutation, we were unable to elicit the exact mutation in our patient despite whole-genome sequencing. References: Jaideep Khare, Prasun Deb, Prachi Srivastava & Babul H. Reddy (2017) Swyer syndrome: The gender swayer?, Alexandria Journal of Medicine, 53:2, 197–200, DOI: 10.1016/j.ajme.2016.05.006 Varshini Chakravarthy, Sehar Ejaz. A 16-Year-Old With Amenorrhea and Delayed Breast Development - Medscape - Jan 14, 2020


1960 ◽  
Vol 04 (03) ◽  
pp. 369-375 ◽  
Author(s):  
E. H Braun ◽  
David B. Stollar

SummaryA case of haemophilia in a young white girl is described. There was a history of bleeding from birth. The thromboplastin generation test was grossly abnormal and A. H. G. levels were below 1%. Bleeding time and capillary morphology was within normal limits. Dental extraction after transfusion caused almost uncontrollable haemorrhage.A complete family history was obtained for four generations. There was no case of a “bleeder” amongst these.The girl’s apparent sex was confirmed by sex chromatin studies.


2017 ◽  
Vol 53 (1) ◽  
pp. 59-63
Author(s):  
Kathleen Ann Bonawandt ◽  
Jason M. Berg ◽  
Richard J. Joseph ◽  
Joseph D. Stefanacci

ABSTRACT A 7 yr old female spayed Yorkshire terrier was referred to the author's institute for a 5 mo history of recurrent cervical spinal pain. Neurologic examination did not reveal any deficits. Hematologic and serum analyses were within normal limits. Thoracic radiographs that incorporated the cervical spine did not show structural abnormalities. Magnetic resonance imaging of the cervical spine demonstrated a contrast enhancing, intradural extramedullary lesion at the level of the C2 vertebra. Hemilaminectomy was performed, during which a long, narrow nematode was visualized upon opening of the dura mater. The parasite was alive when removed during surgery, and the dog recovered with complete resolution of symptoms. The parasite was submitted and confirmed as a male adult Dirofilaria immitis. This is a novel case of an intradural D. immitis infection in the dog with a magnetic resonance imaging description of spinal D. immitis.


2014 ◽  
Vol 7 ◽  
pp. CCRep.S20086 ◽  
Author(s):  
Marilyn N. Bulloch

Background Iodine is a naturally occurring element commercially available alone or in a multitude of products. Iodine crystals and iodine tincture are used in the production of methamphetamine. Although rarely fatal, iodine toxicity from oral ingestion can produce distressing gastrointestinal symptoms and systemic symptoms, such as hypotension and tachycardia, from subsequent hypovolemia. Objective The objective of this case report is to describe a case of iodine toxicity from suspected oral methamphetamine ingestion. Case Report A male in his early 20′s presented with gastrointestinal symptoms, chills, fever, tachycardia, and tachypnea after orally ingesting a substance suspected to be methamphetamine. The patient had elevated levels of serum creatinine, liver function tests, and bands on arrival, which returned to within normal limits by day 4 of admission. Based on the patient's narrow anion gap, halogen levels were ordered on day 3 and indicated iodine toxicity. This is thought to be the first documented case of iodine toxicity secondary to suspected oral methamphetamine abuse. Conclusion Considering that the incidence of methamphetamine abuse is expected to continue to rise, clinicians should be aware of potential iodine toxicity in a patient with a history of methamphetamine abuse.


2016 ◽  
Vol 33 (S1) ◽  
pp. S147-S147
Author(s):  
M.D.C. Ferreira ◽  
S. Varanda ◽  
G. Carneiro ◽  
B. Santos ◽  
Á. Machado

IntroductionPsychosis is one of the most prevalent non-motor complications in Parkinson's disease (PD). Risk factors for PD psychosis are advancing age, longer disease duration, severe motor symptoms, presence of dementia, sleep disorders, depression and autonomic dysfunction. Treatment is challenging in this setting because antipsychotic medications are known to worse motor symptoms.ObjectivesTo highlight the therapeutic difficulties in PD-related psychosis.MethodsCase description and literature review.ResultsWe report a case of a 74-year-old woman with a 9-year history of PD, who presented a complex psychotic disorder consisting in auditory, olfactory and visual (gulliverian and lilliputian) hallucinations, persecutory and sexual delusions. Additionally, the patient presented euthymic mood, without evidence of cognitive impairment or impulse-control disorder. These symptoms began after dopamine agonist therapy (ropinirole 4 mg/day). Other medical conditions that could justify these symptoms were excluded. Initially, ropinirole was removed, but without psychotic remission. Then, she was treated with antipsychotic medication (clozapine 25 mg/day) with full psychotic remission and without significant worsening of motor symptoms.ConclusionsClozapine treatment is frequently delayed, mainly for fear of its side effects, particularly agranulocytosis. However, this antipsychotic drug presents many benefits regarding the management of PD-related psychosis, namely few motor effects and even improvement of motor fluctuations.Disclosure of interestThe authors have not supplied their declaration of competing interest.


Author(s):  
Juliane Léger ◽  
Clemence Delcour ◽  
Jean-Claude Carel

Abstract Fetal and neonatal dysfunctions include rare serious disorders involving abnormal thyroid function during the second half of gestation, which may persist throughout life, as for most congenital thyroid disorders, or be transient, resolving in the first few weeks of life, as in autoimmune hyperthyroidism or hypothyroidism and some cases of congenital hypothyroidism (CH) with the thyroid gland in situ. Primary CH is diagnosed by neonatal screening, which has been implemented for 40 years in developed countries and should be introduced worldwide, as early treatment prevents irreversible neurodevelopmental delay.Central CH is a rarer entity occurring mostly in association with multiple pituitary hormone deficiencies. Other rare disorders impair the action of thyroid hormones. Neonatal Grave’s disease (GD) results from the passage of thyrotropin receptor antibodies (TRAb) across the placenta, from mother to fetus. It may affect the fetuses and neonates of mothers with a history of current or past GD, but hyperthyroidism develops only in those with high levels of stimulatory TRAb activity. The presence of antibodies predominantly blocking TSH receptors may result in transient hypothyroidism, possibly followed by neonatal hyperthyroidism, depending on the balance between the antibodies present. Antithyroid drugs taken by the mother cross the placenta, treating potential fetal hyperthyroidism,but they may also cause transient fetal and neonatal hypothyroidism. Early diagnosis and treatment are key to optimizing the child’s prognosis. This review focuses on the diagnosis and management of these patients during the fetal and neonatal periods. It includes the description of a case of fetal and neonatal autoimmune hyperthyroidism.


2020 ◽  
Vol 79 (Suppl 1) ◽  
pp. 842-843
Author(s):  
M. Higashida-Konishi ◽  
K. Izumi ◽  
S. Hama ◽  
Y. Hayashi ◽  
Y. Okano ◽  
...  

Background:In the case of seronegative arthritis, it was difficult to make a differential diagnosis between remitting seronegative symmetrical synovitis with pitting edema syndrome (RS3PE) and seronegative rheumatoid arthritis (seronegative RA) because the distribution of affected joints was similar and the patients with RS3PE or seronegative RA may have edema.Objectives:To compare the clinical characteristics of RS3PE and seronegative RAMethods:We retrospectively examine consecutive patients diagnosed with RS3PE or seronegative RA in our hospital from 2007 to 2019. Patients in whom both ACPA and RF were negative were included. The patients with RS3PE met the criteria of McCarty et al.: (1) pitting edema of the dorsum of both hands and both feet, (2) sudden onset of polyarthritis, (3) seronegative for ACPA and RF. (4)no radiologically evident erosions developed. The patients with seronegative RA met the EULAR/ACR 2010 criteria. The patients who were diagnosed with RS3PE at first and then diagnosed with seronegative RA afterward were included in seronegative RA group. The first analysis was performed on the affected joints, CRP, ESR, Hb, LDH, edema, the history of malignancy 2 years before and after the diagnosis, treatment, and the history of infection requiring hospitalization after the start of treatment. The affected joints were shoulders, elbows, wrists, finger joints (the MCP, and PIP joints), hips, knees, ankles, and toe joints (the MTP and PIP joints). The secondary analysis was performed on the above evaluations with a propensity score (PS) matching for age.Results:In the first analysis, 20 patients with RS3PE and 122 patients with seronegative RA were enrolled. The mean ages (RS3PE, seronegative RA) were 81.1, 67.4 years old. Females were 60.0%, 63.1%. The mean observation period was 25.4, 63.6 months. The proportion of affected joints were shoulders (25.0%, 42.6%), elbows (10.0%, 29.5%: p=0.06), wrists (85.0%, 73.8%), finger joints (80.0%, 95.1%: p=0.01), hips (0%, 9.8%), knees (40.0%, 37.7%), ankles (65.0%, 39.3%: p=0.03) and toe joints (40.0%, 32.8%). Edema at diganosis was observed in 100%, 17.21% (p <0.0001). The mean levels of the following blood tests at diagnosis were noted: CRP, 9.0 and 4.8 mg/dL (p=0.02); ESR, 87.6 and 60.7 mm/1h (p=0.003); Hb, 10.4 and 11.8 mg/dl (p=0.001); LDH, 198.3 and 177.9 U/L (p = 0.12); MMP-3, 742.5 and 633.8 ng/mL (p = 0.14). The proportion of patients with high LDH levels (>222 U/L) was 13.6% and 9.0% (p=0.0269). The proportion of patients having the history of malignancy was 20.0%, 8.2% (p=0.10). The patient treated with prednisolone as the initial treatment was 100% and 41.0%; the mean dose was 14.3 and 9.9 mg/d. After the start of treatment, the proportion of infection requiring hospitalization was 20.0 and 3.28% (p=0.002).In the secondary analysis with PS, 17 patients with RS3PE and 17 patients with seronegative RA were enrolled. The mean ages were 80.4, 78.9 years old. Females were 52.9, 76.4%. The affected joints with difference were elbows (11.8, 35.3%: p=0.10), wrists (82.4, 100%: p=0.06), and finger joints (82.4, 100%: p=0.06). The mean levels of Hb at diagnosis was 10.4, 11.4 mg/dL (p=0.01). The proportion of patients having the history of malignancy was 23.5% and 0% (p=0.03). After the start of treatment, the proportion of infection requiring hospitalization was 23.5% and 0% (p=0.03).Conclusion:When the ankles are affected and edema is observed, RS3PE is more likely than seronegative RA. RS3PE had higher levels of CRP, ESR, and LDH. The proportion of anemia was higher in RS3PE. The proportions of infection requiring hospitalization and the history of malignancy were higher in RS3PE.References:[1]McCarty DJ, O’Duffy JD et al. Remitting Seronegative Symmetrical Synovitis with Pitting Edema (RS3PE Syndrome). JAMA 1985; 254: 2763–2767. DOI:10.1001/jama.1985.03360190069027Disclosure of Interests:Misako Higashida-Konishi: None declared, Keisuke Izumi Grant/research support from: Asahi Kasei Pharma, Takeda Pharmaceutical Co., Ltd., Speakers bureau: Asahi Kasei Pharma Corp, Astellas Pharma Inc., Bristol Myers Squibb, Chugai Pharmaceutical Co., Ltd., Eli Lilly Japan K.K., Mitsubishi Tanabe Pharma Co., Satoshi Hama: None declared, Yutaro Hayashi: None declared, Yutaka Okano: None declared, Hisaji Oshima: None declared


2021 ◽  
Vol 11 (6) ◽  
pp. 231-233
Author(s):  
Nimitha K J ◽  
Porimita Chutia ◽  
Pooja Misal

Constipation is one major complaint in elderly population. It may be due to physiological and anatomical reasons of aging, but it can be also due chronic medical and mental illnesses and due to use of multiple medications. Constipation itself is a precipitating factor for delirium. Drugs used for constipation can also be the culprit. A 64-year-old female who had a history of hypertension and chronic constipation presented with symptoms of confused and altered behavior, decreased oral intake, decreased sleep. On history taking it was known that she was using Herbal medication containing senna glycoside and other compounds since 8-9months. On examination she had signs of dehydration, disoriented and attention was impaired. On investigation her serum sodium was 122.6 mmol/ and other investigations were within normal limits. She was diagnosed as a case of Delirium according to ICD-10 criteria. Her dehydration was corrected by giving intravenous fluids and serum sodium level was corrected using salt capsules 2 tablets thrice daily. For disturbed sleep she was prescribed Tab Melatonin 10mg at bedtime and constipation was treated with per rectal enema and syrup lactulose 30ml at bedtime. Patient improved in 1 week time. Senna a herbal laxative used to treat constipation. It can alter intestinal electrolyte transportation and irritates intestinal mucosa. It is due to increased peristalsis and increased defecation and even diarrhea Prolonged use can cause dehydration, electrolyte imbalance and delirium. Senna, an over-the-counter laxative with FDA approval is a matter of concern in the current scenario. This case report warns into the judicious use of laxatives containing senna in elderly population. Key words: Delirium, Herbal Laxative, Senna, Constipation.


2017 ◽  
Vol 12 (2) ◽  
pp. 86-87
Author(s):  
Shahana Ahmed ◽  
Dipti Rani Shaha

Invasive mole is a condition where a molar pregnancy, such as a partial hydatidiform mole or complete hydatidiform mole, invades the wall of the uterus, potentially spreading and metastasizing to other parts of the body. Here is a case who presented with history of evacuation for molar pregnancy. She presented with irregular P/V bleeding on and off and after admission silent perforation with massive haemoperitoneum was detected for which emergency laparotomy was done. She recovered and was followed up till her b-hCG levels were within normal limits. As patient presented to us with haemoperitoneum and on laparotomy, there was invasion into whole of the uterus, it could not be saved and hysterectomy was done.Faridpur Med. Coll. J. Jul 2017;12(2): 86-87


2014 ◽  
Vol 2014 ◽  
pp. 1-7 ◽  
Author(s):  
Ehud Lebel ◽  
Yuri Mishukov ◽  
Liana Babchenko ◽  
Arnon Samueloff ◽  
Ari Zimran ◽  
...  

Changes of bone during pregnancy and during lactation evaluated by bone mineral density (BMD) may have implications for risk of osteoporosis and fractures. We studied BMD in women of differing ages, parity, and lactation histories immediately postpartum for BMD,T-scores, andZ-scores. Institutional Review Board approval was received. All women while still in hospital postpartum were asked to participate. BMD was performed by dual-energy X-ray absorptiometry (DXA) machine at femoral neck (FN) and lumbar spine (LS) by a single technician. Of 132 participants, 73 (55.3%) were ≤30 years; 27 (20.5%) were primiparous; 36 (27.3%) were grand multiparous; 35 (26.5%) never breast fed. Mean FNT-scores andZ-scores were higher than respective mean LS scores, but all means were within the normal limits. Mean LST-scores andZ-scores were highest in the grand multiparas. There were only 2 (1.5%) outliers with lowZ-scores. We conclude that, in a large cohort of Israeli women with BMD parameters assessed by DXA within two days postpartum, meanT-scores andZ-scores at both the LS and FN were within normal limits regardless of age (20–46 years), parity (1–13 viable births), and history of either no or prolonged months of lactation (up to 11.25 years).


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