J Pediatr Endocr Met 2015; 28(3-4): 457–462

Patient report Ricardo Zegarra-Linares, Kathleen C. Moltz and Nahed Abdel-Haq*

Pituitary abscess in an adolescent girl: a case report and review of the literature Abstract: We report the case of a 15-year-old girl who presented with a history of recurrent bitemporal headaches for the last 2 months. In the prior few days, she complained of neck pain, emesis, phonophobia and photophobia, but no fever. Additional symptoms included polydipsia, polyuria and weight gain in the last year. Magnetic resonance imaging (MRI) of the brain demonstrated a cystic sellar and suprasellar mass with peripheral enhancement. Cerebrospinal fluid studies showed pleocytosis. Serum hormone levels were consistent with panhypopituitarism. Transnasal sphenoidotomy was performed, and 2  mL of purulent material was drained, confirming the diagnosis of pituitary abscess. The patient completed 6 weeks of parenteral antibiotics. She improved but continued to require home hormonal replacement therapy. A repeated MRI 3  months later showed abscess resolution. In addition to tumors, pituitary abscess should be considered in children who present with headache and panhypopituitarism, particularly in those who present with signs of meningeal inflammation. Prolonged parenteral antibiotics and surgical drainage are effective. Keywords: abscess.

children;

panhypopituitarism;

pituitary

DOI 10.1515/jpem-2014-0112 Received March 5, 2014; accepted July 14, 2014; previously published online August 12, 2014 *Corresponding author: Nahed Abdel-Haq, MD, Division of Pediatric Infectious Diseases, Children’s Hospital of Michigan, 3901 Beaubien Boulevard, Detroit, MI 48201, USA, Phone: +1 3137455862, Fax: +1 3139938846, E-mail: [email protected]; and Children’s Hospital of Michigan, Division of Infectious Diseases, the Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, MI, USA Ricardo Zegarra-Linares: Children’s Hospital of Michigan, Division of Infectious Diseases, the Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, MI, USA Kathleen C. Moltz: Children’s Hospital of Michigan, Division of Endocrinology, the Carman and Ann Adams Department of Pediatrics, Wayne State University School of Medicine, Detroit, MI, USA

Introduction Pituitary abscess is a rare disease, and it is most frequently reported in the adult population. Clinical presentation is variable, making a definitive preoperative diagnosis very challenging despite improvement in the imaging technology. We describe the case of a teenage girl with pituitary abscess who presented with chronic headache, visual problems and panhypopituitarism in addition to symptoms of meningeal inflammation. We also reviewed the literature, with special attention to pediatric cases.

Case report This is the case of a 15-year-old Caucasian girl with a history of hypothyroidism with decreasing thyroid hormone replacement requirement who presented to the emergency room with headaches for about 2 months. Symptoms occurred once to twice a week, lasting for a few hours. She also had a history of blurred vision, and an initial evaluation by an ophthalmologist noted right-sided papilledema. However, reevaluation by a neuro-ophthalmologist revealed normal fundoscopic examination. During the week prior to presentation, her headaches increased in frequency and severity, and were associated with neck pain, nausea, vomiting, phonophobia and photophobia. No fever had been reported, no respiratory symptoms and no sick contacts. Her review of systems was remarkable for polyuria and polydipsia, weight gain despite healthy diet and amenorrhea during the last year. Her past medical history is significant for acquired hypothyroidism due to Hashimoto’s thyroiditis diagnosed at 8 years of age; she is currently on treatment with levothyroxine. She was hospitalized 1 year prior to presentation due to pneumonia; she had no other hospitalizations. She was initially seen in a community emergency room where she was alert but complained of severe bitemporal headache. A computed tomography (CT) scan of

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458      Zegarra-Linares et al.: Pituitary abscess in an adolescent girl: a case report and review of the literature the head showed a pituitary mass measuring 1.2 × 1.1  cm with no mass effect. A lumbar puncture was performed with an opening pressure of 29  mm Hg. Cerebrospinal fluid (CSF) analysis revealed 234/mm3 of nucleated cells (neutrophils 4% and lymphocytes 86%), red cell count of 19/mm3, glucose level of 38 mg/dL and protein level of 87.7 mg/dL. Her headache improved partially after lumbar puncture. Electrolyte levels and renal function test results were unremarkable, and the pregnancy test result was negative. She was transferred to our hospital for further evaluation and management. At our emergency department, her temperature was 37.1°C; pulse, 74/min; respiratory rate, 20/min; blood pressure, 120/76 mm Hg; and weight, 104 kg. She was alert and oriented. She had normal sensory and motor function and no meningeal signs. Her cranial nerves were intact. Photophobia was present. No papilledema was noted on fundoscopic exam. The rest of the physical examination results were unremarkable. Laboratory findings revealed a white blood cell count of 13,400/mm3 (neutrophils 62% and lymphocytes 28%), hemoglobin level of 13.8 g/dL, hematocrit level of 39.3% and platelet level of 261,000/mm3. Serum pituitary hormones were checked (see Table 1). Low cortisol was verified on low-dose adrenocorticotropic hormone (ACTH) testing, which confirmed insufficient cortisol response consistent with secondary adrenal insufficiency. Low free thyroxine (fT4) and thyroid-stimulating hormone (TSH) were consistent with secondary hypothyroidism, and low folliclestimulating hormone (FSH), luteinizing hormone (LH) and estradiol levels (along with secondary amenorrhea) were consistent with secondary gonadotropin deficiency. Stress dosing of hydrocortisone was initiated, and levothyroxine was restarted after 48 h of hydrocortisone replacement.

The patient was started on intravenous ceftriaxone at meningitic doses. Brain magnetic resonance imaging (MRI) demonstrated a 1.1 × 1.4 × 1.3-cm centrally necrotic, sellar and suprasellar cystic mass lesion with peripheral enhancement that extended into the infundibulum and floor of the third ventricle (Figure 1, left). Diffusion restriction in the center of the cystic necrotic portion of the mass was noted. The suprasellar component of the mass demonstrated an obvious indentation on optic chiasm. Based on imaging, pituitary abscess was suspected. The patient underwent a transnasal sphenoidotomy, and 2  mL of purulent material was drained, confirming the diagnosis. Intravenous metronidazole and vancomycin meningitic doses were added. Bacterial cultures grew viridans streptococci susceptible to vancomycin but moderately susceptible to cephalosporins and also grew Corynebacterium argentoratense susceptible to vancomycin. Her therapy was switched to intravenous meropenem and vancomycin. Wound anaerobic bacteria, fungal and mycobacterial cultures showed no growth. CSF culture showed no growth as well. Her visual problems and headaches resolved shortly after surgery. She completed 6 weeks of parenteral antibiotic treatment. A repeated MRI of the brain approximately 3 months after surgery showed no evidence of fluid collections within the pituitary gland (Figure 1, right). While in the preoperative period, the patient complained of polydipsia; postoperatively, she developed polyuria, which was responsive to intravenous vasopressin and then transitioned to oral desmopressin. A formal water deprivation test was not performed; however, holding a dose of desmopressin led to more than 5 L of dilute urine output accompanied by increasing serum

Table 1 Initial endocrine laboratory results, 6 a.m. fasting (normal range). Cortisol Esradiol Follicle-stimulating hormone Luteinizing hormone Free thyroxine Thyroid-stimulating hormone Prolactin Insulin-like growth factor 1 Sodium Potassium Chloride Carbon dioxide

                       

Cortrosyn (synacthen) 1–μg stimulation test –– 0-min adrenocorticotropic hormone   –– 0-min cortisol   –– 50-min cortisol  

2.2 μg/dL ( > 10 μg/dL) 17 pg/mL (follicular: 20–196 pg/mL; luteal 27–304 pg/mL) 2.4 mIU/mL (follicular: 2.5–10.2 mIU/mL; luteal: 1.5–9.1 mIU/mL) 0.4 mIU/mL (follicular: 1.9–12.5 mIU/mL; luteal: 0.5–16.9 mIU/mL) 0.7 ng/dL (0.8–1.8 ng/dL) 0.200 μIU/mL (0.210–4.940 μIU/mL) 10.3 ng/mL (2.8–29.2 ng/mL) 271 ng/mL (94–765 ng/mL) 141 mmol/L 4.3 mmol/L 107 mmol/L 25 mmol/L 9 pg/mL (6–55 pg/mL) 2.8 μg/dL 6.9 μg/dL

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Zegarra-Linares et al.: Pituitary abscess in an adolescent girl: a case report and review of the literature      459

Figure 1 (Left) Preoperative MRI: Post-contrast sagittal image showing a sellar cystic mass lesion with peripheral enhancement and central diffusion restriction. (Right) Similar sagittal view obtained after transsphenoidal drainage and 6 weeks of parenteral antibiotic treatment, showing no evidence of fluid collection within the pituitary gland.

osmolality (287–304 mOsm/kg) and serum sodium level (136–142 mmol/L). Urine osmolality increased only from 121 to 234 mOsm/kg during the same 12 h. This was considered sufficient to confirm central diabetes insipidus, and oral desmopressin was resumed. Additionally, in the postoperative period, thyroid hormone replacement was increased from 0.05 mg daily to 0.075 mg daily, with a corresponding increase in fT4 level from 0.7 to 1.2 ng/dL and a return to normal of TSH level (0.200 to 1.772 μIU/mL) by 1 month postoperative. A year after her surgical intervention, she continued to have evidence of partial hypopituitarism and requires hormone replacement therapy with desmopressin for central diabetes insipidus as well as levothyroxine for her preexisting acquired primary hypothyroidism (see Table 2). Her adrenal function has returned to normal.

After a few episodes of spotting and normalization of gonadotropin levels, persistent secondary amenorrhea is being treated with medroxyprogesterone every 3 months. This allows for normal menstruation without complete suppression of her own hypothalamic-pituitary-gonadal axis, which may recover with weight loss. No pelvic radiography or gonadotropin stimulation testing has been pursued by the family’s preference.

Discussion Pituitary abscess is an uncommon clinical entity that accounts for   10 μg/dL) 8 pg/mL (6–55 pg/mL) 49 pg/mL (follicular: 20–196 pg/mL; luteal: 27–304 pg/mL) 5.5 mIU/mL (follicular: 2.5–10.2 mIU/mL; luteal: 1.5–9.1 mIU/mL) 5.1 mIU/mL (follicular: 1.9–12.5 mIU/mL; luteal: 0.5–16.9 mIU/mL) 1.2 ng/dL (0.8–1.8 ng/dL) 1.726 μIU/mL (0.210–4.940 μIU/mL) 139 mmol/L 4.6 mmol/L 105 mmol/L 23 mmol/L

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  12, M

  16, F

  19, M

  17, F

  12, F

  12, F

Vates et al. (1)

Vates et al. (1)

Vates et al. (1)

Vates et al. (1)

Dutta et al. (3)

Kaur et al. (4)

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Our case

  15, F

Hazra et al. (7)   14, M

Zhang et al. (6)   19, F

Thiryayi et al. (5)  21, F

  12, M

Vates et al. (1)

  None

  Endocrine findings   Cultures

  Antibiotic treatment

  Retro-orbital headache, visual   field defects, diplopia, fever, CSF pleocytosis   Headache, primary   amenorrhea, decreased visual acuity   Fever, gait imbalance, slurring   speech, amnesia, leukocytosis   Headache, meningismus,   blurred vision, CSF pleocytosis, elevated opening pressure

  None

  Negative

None

None

  Staphylococcus aureus   Panhypopituitarism  S. viridans group, Corynebacterium argentoratense

  None

Pituitary adenoma   Panhypopituitarism  Toxoplasma gondii cysts and prolactinemia

None

  Transient diabetes insipidus, full recovery   No recovery of endocrine function

  Growth hormone dysfunction   Full recovery

  Hypothyroidism

  Recurrent craniopharyngioma

  No recovery of endocrine function   Full recovery

  Full recovery

  Outcome

  Frontal lobe syndrome   Vancomycin+meropenem  ×   Partial recovery of 6 weeks endocrine function

  No data

  No data

Cefazolin+amikacin+chl oramphenicol  × 4 weeks, followed by cefadroxil  × 1 month   No data

Antibiotics  × 6 weeks

Ceftriaxone  × 4 weeks

Ampicillin  × 10 days

Penicillin  × 6 weeks

  α-Hemolytic streptoccoci,   Penicillin  × 6 weeks Neisseria spp., Micrococcus spp., S. Epidermidis   Inflamed pituitary   Panhypopituitarism  Negative   Penicillin  × 6 weeks

  None

  Underlying condition of pituitary gland

  Headache, bitemporal hemianopsia   Headache, meningismus, fever,  Inflamed pituitary   Panhypopituitarism  Negative   elevated opening pressure   Retro orbital headache   Craniopharyngioma  None   α-Hemolytic streptoccoci,   Neisseria spp., Micrococcus spp., S. Epidermidis   Headache, meningismus, fever,  Fibrous tissue with   Panhypopituitarism  Mycobacterium fortuitum   leukocytosis, CSF pleocytosis, inflammation elevated opening pressure   Headache, fever, bitemporal   Rathke’s cleft cyst   Prolactinemia   Acinetobacter,   hemianopsia Staphylococcus spp.   Headache, decreased visual   None   None   No surgical drainage   acuity, leukocytosis

  Headache

  Age (years)/  Clinical findings gender

References

Table 3 Summary of pediatric cases of pituitary abscess reported in the literature (  ≤  21 years of age).

460      Zegarra-Linares et al.: Pituitary abscess in an adolescent girl: a case report and review of the literature

Zegarra-Linares et al.: Pituitary abscess in an adolescent girl: a case report and review of the literature      461

mostly published as case reports and series of cases (2). Vates et al. (1) described one of the largest series including 24 patients, five of whom were  

Pituitary abscess in an adolescent girl: a case report and review of the literature.

We report the case of a 15-year-old girl who presented with a history of recurrent bitemporal headaches for the last 2 months. In the prior few days, ...
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