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Lymphocytic Hypo- Physitis in a Patient with Systemic Lupus Erythematosus

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Lymphocytic Hypo- Physitis in a Patient with Systemic Lupus Erythematosus CASE REPORT Clinical and Experimental Rheumatology 2000; 18: 78-80. Lymphocytic hypo- ABSTRACT mass and the pathologic findings during A case of lymphocytic hypophysitis in a a transsphenoidal operation were com- physitis in a patient patient with systemic lupus erythema- patible with lymphocytic hypophysitis. tosus is described. A 20-year-old We describe an SLE patient with head- with systemic lupus woman was admitted to our hospital ache and nausea, in whom the presence with generalized myalgia and facial of an intrasellar mass and pathologic erythematosus rash in May 1998. The patient had a findings indicated lymphocytic hypo- medical history, physical examination, physitis. J.D. Ji1, S.Y. Lee2, and laboratory findings compatible 2 2 with systemic lupus erythematosus Case report S.J. Choi , Y.H. Lee , (SLE). Headache and nausea had A 20-year-old woman was admitted to G.G. Song2 developed 3 months previously and the Korea University Anam Hospital in worsened over the following months. May 1998 with a three-month history of 1Hospital for Rheumatic Disease, Hanyang Hormonal investigation showed hypo- facial rash, fever, generalized myalgia, University Medical Center; 2Division of pituitarism except for prolactin. A hair loss and arthralgia. During the three Rheumatology, Department of Internal magnetic resonance image of the brain months before admission, she had noted Medicine, Korea University Hospital, showed a mass lesion in the pituitary intermittent headache and nausea. No Seoul, Korea. fossa. A trans-sphenoidal surgical visual disturbances were present. There Jong Dae Ji, MD; So Young Lee, MD; procedure was performed which was no family history of autoimmune or Seong Jae Choi, MD; Young Ho Lee, MD, revealed a dark-yellowish hematoma. endocrine problems. PhD; Gwan Gyu Song, MD, PhD. Microscopic examination showed On examination, the patient was severely Please address correspondence and reprint diffuse infiltration of lymphocytes and distressed. Her heart rate was 70 beats requests to: Gwan Gyu Song, MD, Division plasma cells with fibrosis in the per minute, her blood pressure 110/70 of Rheumatology, Department of Internal Medicine, Anam Hospital, College of anterior pituitary. Post-operatively the mm Hg, and she had a temperature of Medicine, Korea University, 126-1, patient’s headaches and nausea 36.2°C. Hair loss, malar rash, and a ery- 5-Ka, Anam-Dong, Seongbuk-Gu, Seoul, resolved. This indicates that lympho- thematous rash on the palms and soles 136-705, Korea. cytic hypophysitis may be associated could be seen. Ophthalmological exami- E-mail: [email protected] with SLE. nation showed normal visual fields. The Received on April 29, 1999; remainder of the examination was nor- accepted in revised form on July 20, 1999. Introduction mal. © Copyright CLINICAL AND Lymphocytic hypophysitis is a rare in- Complete blood counts revealed: hemo- EXPERIMENTAL RHEUMATOLOGY 2000. flammatory disorder of the pituitary globin 10.6 g/dl, white blood cell count gland which is characterized by lym- 2,960/mm3, and platelet count 106,000/ Key words: phocytic infiltration and fibrosis of the mm3. Electrolytes and urinalysis were Systemic lupus erythematosus, anterior pituitary (1). Most cases have normal. Alanine aminotransferase was lymphocytic hypophysitis. been diagnosed in women during late 107 IU/l and aspartate aminotransferase pregnancy or the postpartum period. Pa- was 98 IU/l. The results of other labora- tients usually present with symptoms of tory evaluations were: antinuclear anti- an expanding intrasellar mass or vary- body (ANA) titer 1:40 (speckled pat- ing degrees of hypopituitarism. Although tern), anti-dsDNA antibody 20.44 U/ml its causes are not known, it has been re- (normal < 5.3 U/ml), negative anti-RNP ported that lymphocytic hypophysitis is antibody, negative anti-Sm antibody, re- associated with autoimmune thyroiditis active VDRL, negative anti-SS-A and (2, 3), pernicious anemia (4), Grave’s di- anti-SS-B antibodies, erythrocyte sedi- sease (5), systemic lupus erythematosus mentation rate (ESR) 54 mm/hr (normal (SLE) (6), and autoantibodies including < 20 mm/hr), C3 55.5 mg/dl (normal 88 the antipituitary (7), antimitochondrial to 201 mg/dl), C4 15.0 mg/dl (normal (8), antiparietal (4) and antinuclear anti- 16 - 47 mg/dl). SLE was diagnosed based bodies (9). Therefore, lymphocytic hy- on the presence of malar rash, hair loss, pophysitis has been postulated to be an leukopenia, positive ANA, and positive autoimmune disease primarily of preg- anti-dsDNA antibodies. nancy and the puerperium. Lymphocytic Headache and nausea worsened one day hypophysitis commonly manifests as a after admission. Skull x-rays revealed an pituitary mass and pre-operatively has enlarged sella and magnetic resonance been misdiagnosed as a pituitary adeno- imaging (MRI) demonstrated a round ma (7). mass lesion in the sella. T1- and T2- Our patient presented with an intrasellar weighted images revealed high signal 78 Lymphocytic hypophysitis in a patient with SLE / J.D. Ji et al. CASE REPORT (a) (b) Fig. 1. (a) T1-weighted image and (b) gadolinium-enhanced T1-weighted image demonstrating high signal intensity of the pituitary mass. intensity of the pituitary mass (Fig. 1). A trans-sphenoidal surgical procedure toms or signs of hypopituitarism, only A gadolinium-enhanced T1-weighted was performed and revealed a dark liq- headache and nausea. Dynamic pituitary image showed high signal intensity with- uified hematoma. After the hematoma function testing showed absent cortisol in the mass (Fig. 2). Compared with the was aspirated, the pituitary gland could and growth hormone responses to insu- pre-enhanced film, evidence of definite be seen. A biopsy of the pituitary gland lin, impaired TSH response to TRH, and enhancement was not seen. Further in- showed the infiltration of lympocytes hyporesponses of FSH and LH to LHRH. vestigations of pituitary function were and plasma cells with fibrosis in the an- She had an elevated prolactin level. carried out. The basal hormone level re- terior pituitary (Fig. 3). Post-operatively, Many have reported a mild elevation of vealed the following: growth hormone the patient’s headaches and nausea re- prolactin attributed to either the disrup- (GH) 4.12 ng/ml (normal 0-5), cortisol solved. Two months later her hormone tion of hypophyseal transport of prolac- 4.52 mg/ml (normal 5-20), prolactin 90.3 levels were as follows: cortisol 21.04 mg/ tin inhibitory factor by compression of ng/ml (normal 0-20), thyroid stimulat- ml, prolactin 125.74 ng/ml, TSH 1.77 IU/ the pituitary stalk, or to a direct effect of ing hormone (TSH) 1.13 IU/ml (normal ml, LH 1.86 mIU/ml and FSH 2.28 mIU/ the inflammatory process on the prolac- 0-10), lutenizing hormone (LH) 2.86 ml. The patient is currently well and re- tin cells, or to a lactotroph-stimulating mIU/ml (normal 5-25) and follicle stimu- ceiving prednisolone 15 mg and hydrox- antibody arising as a consequence of the lating hormone (FSH) 4.32 mIU/ml (nor- ychloroquine 400 mg daily. inflammatory process (2, 4, 8-10). The mal 5-20). Dynamic tests of pituitary cause of the elevated prolactin level in function were performed. TSH and pro- Discussion our patient is unclear; it could have been lactin responses to thyrotropin releasing Lymphocytic hypophysitis is a rare auto- due to either lymphocytic hypophysitis hormone (TRH) (400 mg), LH and FSH immune disease, diagnosed either by sur- or the SLE itself. Elevated serum prol- responses to lutenizing hormone releas- gical biopsy of the pituitary or at autopsy. actin levels have been reported in SLE ing hormone (LHRH) (100 mg), cortisol Many of the patients reported previously patients (11, 12), and the persistently el- and GH responses to insulin (0.1 unit/ had a large pituitary mass lesion, and evated prolatin levels in our patient af- kg body weight) were evaluated (Table most had various degrees of hypopitui- ter surgery may have been due to dis- I). tarism (7-9). Our patient had no symp- ease activity. While the causes of lymphocytic hypo- Table I. Serum levels of GH, cortisol, prolactin, TSH, LH and FSH after administration of physitis are unclear, several findings sug- TRH, GnRH and insulin. gest that autoimmunity is involved in the pathogenesis. Experimentally, lympho- Minute cytic hypophysitis has been induced by 0 15 30 60 90 120 injecting pituitary tissues and adjuvants GH (ng/ml) 4.12 0.6 0.28 0.32 into footpads of the rat (13). The histo- Cortisol (mg/ml) 4.52 4.35 4.35 3.89 logic features of this lesion were nearly Prolactin (ng/ml) 90.3 100.39 105.5 98.9 identical to that seen in patients with TSH (IU/ml) 1.13 3.31 2.91 3.01 lymphocytic hypophysitis. These results LH (mIU/ml) 2.86 5.78 7.07 7.49 suggest that an autoimmune reaction may be implicated in the genesis of hu- FSH (mIU/ml) 4.32 6.16 5.77 5.95 man lymphocytic hypophysitis. Similar 79 CASE REPORT Lymphocytic hypophysitis in a patient with SLE / J.D. Ji et al. WORTH JMB, EHRLICH EN: Lymphocytic hypophysitis with selective adrenocortico- tropic hormone deficiency. Arch Intern Med 1980; 140: 1243-5. 4. MAZZONE T, KELLY W, ENSINCK J: Lymph- ocytic hypophysitis associated with antiparietal cell antibodies and vitamin B12 deficiency. Arch Intern Med 1983; 143: 1794-5. 5. BAYRAM F, KELESTIMUR F, OZTURK F, SELCUKLU A, PATIROGLU TE, BEYHAN Z: Lymphocytic hypophysitis in a patient with Fig. 3. A biopsy of Grave’s disease. J Endocrinol Invest 1998; 21: the pituitary gland 193-7. showing infiltration 6. HASEGAWA Y, MATSUMOTO M, KAMIMURA of lymphocytes and A, YAMAMOTO M: A case of systemic lupus plasma cells with erythematosus with autoimmune hypophysitis. fibrosis (Hematoxylin Nippon Naika Gakkai Zasshi 1993; 82: 582-3 eosin stain, x100).
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