doi:10.1111/jog.12930
J. Obstet. Gynaecol. Res. Vol. 42, No. 4: 467–470, April 2016
Post-partum hypoglycemia and hypothermia as first manifestations of lymphocytic adenohypophysitis: A case report Shinsuke Koyama, Koichiro Okuno, Hirokazu Naoi, Masahiro Watanabe, Kimiaki Ozaki and Yasuhiko Shiki Department of Obstetrics and Gynecology, Osaka Rosai Hospital, Sakai, Japan
Abstract Lymphocytic adenohypophysitis is a very rare autoimmune disease characterized by an anterior pituitary infiltration of lymphocytes that cause various symptoms, such as headache, visual disturbance, and hypopituitarism. Lymphocytic adenohypophysitis is more common in women and has a remarkable association with the perinatal period. Recently, we experienced a case of lymphocytic adenohypophysitis shortly after delivery. A 37-year-old primiparous woman delivered her baby at 38 weeks’ gestation. The next morning, she presented symptoms of hypoglycemia and hypothermia. Blood testing revealed hypoadrenalism and hypothyroidism. Pituitary magnetic resonance imaging showed characteristic findings consistent with lymphocytic adenohypophysitis. We immediately initiated medication with steroids and levothyroxine. Currently, her pituitary size has normalized and its function has fully recovered. Physical stress resulting from labor and delivery may unmask subclinical lymphocytic adenohypophysitis. Practitioners should keep in mind the possibility of lymphocytic adenohypophysitis in any pregnant women with symptoms of hypoglycemia and hypothermia after delivery. Key words: hypoglycemia, hypopituitarism, hypothermia, lymphocytic adenohypophysitis, lymphocytic hypophysitis.
Introduction Lymphocytic hypophysitis (LH) is a very rare autoimmune disease characterized by lymphocytic infiltration and destruction of the pituitary gland.1 The annual incidence of LH can be estimated at one case per 9 million.2 Based on the affected area of the pituitary gland, LH can be classified as lymphocytic adenohypophysitis (LAH), which involves only the anterior pituitary lobe, lymphocytic infundibuloneurohypophysitis, which involves only the posterior pituitary lobe, and lymphocytic panhypophysitis, which involves both pituitary lobes.3 LAH is predominant in women, with a female-to-male ratio of 6:1.2 In addition, LAH shows a striking association with pregnancy.2 The etiology of this association remains
unexplained, but autoimmune mechanisms are widely believed to be the cause because LH is frequently associated with other autoimmune diseases (about 25–50%), such as Hashimoto disease, systemic lupus erythematosus and so on.4 Clinical presentation of LH is variable and includes symptoms related to pituitary enlargement and various degrees of pituitary hormonal impairment.5 Recently, we experienced a case of LAH that developed with unusual symptoms of hypoglycemia and hypothermia shortly after delivery. Previous reports have shown that the most common and initial complaints of LAH patients are symptoms of sellar compression, represented by headache, vomiting and visual disturbances.2,4,5 We present the clinical course of our case and discuss clinical considerations associated with this rare condition.
Received: September 27 2015. Accepted: November 18 2015. Correspondence: Dr Shinsuke Koyama, Department of Obstetrics and Gynecology, Osaka Rosai Hospital, 1179-3 Nagasone-cho, Kita-ku, Sakai, Osaka 591-8025, Japan. Email: [email protected]
© 2016 Japan Society of Obstetrics and Gynecology
467
S. Koyama et al.
Case Report A healthy 37-year-old woman, gravida 1 para 0, conceived spontaneously. Her clinical course of pregnancy was uneventful until the event. She had no subjective symptoms, such as headache or visual disturbance, throughout her perinatal period. At 38 weeks’ gestation, she delivered her baby by induced labor due to mild preeclampsia. At the time of delivery, vacuum extraction was performed because of non-reassuring fetal status caused by placental abruption. She bled 1800 mL but went into shock (blood pressure, 75/35 mmHg; pulse rate (PR), 130 b.p.m.), so we transfused her with a total of 6 units of red blood cells to stabilize her clinical condition. Circulatory collapse observed was greater than that expected from her blood loss and, thus, we were suspicious of some underlying condition being present. In fact, the next morning (12 h after delivery), she suddenly presented symptoms of hypoglycemia (blood sugar (BS), 35 mg/dL) and hypothermia (body temperature (BT), 35.1 °C). Blood testing showed hypoadrenalism and hypothyroidism; adrenocorticotropic hormone (ACTH),
J. Obstet. Gynaecol. Res. Vol. 42, No. 4: 467–470, April 2016
Post-partum hypoglycemia and hypothermia as first manifestations of lymphocytic adenohypophysitis: A case report Shinsuke Koyama, Koichiro Okuno, Hirokazu Naoi, Masahiro Watanabe, Kimiaki Ozaki and Yasuhiko Shiki Department of Obstetrics and Gynecology, Osaka Rosai Hospital, Sakai, Japan
Abstract Lymphocytic adenohypophysitis is a very rare autoimmune disease characterized by an anterior pituitary infiltration of lymphocytes that cause various symptoms, such as headache, visual disturbance, and hypopituitarism. Lymphocytic adenohypophysitis is more common in women and has a remarkable association with the perinatal period. Recently, we experienced a case of lymphocytic adenohypophysitis shortly after delivery. A 37-year-old primiparous woman delivered her baby at 38 weeks’ gestation. The next morning, she presented symptoms of hypoglycemia and hypothermia. Blood testing revealed hypoadrenalism and hypothyroidism. Pituitary magnetic resonance imaging showed characteristic findings consistent with lymphocytic adenohypophysitis. We immediately initiated medication with steroids and levothyroxine. Currently, her pituitary size has normalized and its function has fully recovered. Physical stress resulting from labor and delivery may unmask subclinical lymphocytic adenohypophysitis. Practitioners should keep in mind the possibility of lymphocytic adenohypophysitis in any pregnant women with symptoms of hypoglycemia and hypothermia after delivery. Key words: hypoglycemia, hypopituitarism, hypothermia, lymphocytic adenohypophysitis, lymphocytic hypophysitis.
Introduction Lymphocytic hypophysitis (LH) is a very rare autoimmune disease characterized by lymphocytic infiltration and destruction of the pituitary gland.1 The annual incidence of LH can be estimated at one case per 9 million.2 Based on the affected area of the pituitary gland, LH can be classified as lymphocytic adenohypophysitis (LAH), which involves only the anterior pituitary lobe, lymphocytic infundibuloneurohypophysitis, which involves only the posterior pituitary lobe, and lymphocytic panhypophysitis, which involves both pituitary lobes.3 LAH is predominant in women, with a female-to-male ratio of 6:1.2 In addition, LAH shows a striking association with pregnancy.2 The etiology of this association remains
unexplained, but autoimmune mechanisms are widely believed to be the cause because LH is frequently associated with other autoimmune diseases (about 25–50%), such as Hashimoto disease, systemic lupus erythematosus and so on.4 Clinical presentation of LH is variable and includes symptoms related to pituitary enlargement and various degrees of pituitary hormonal impairment.5 Recently, we experienced a case of LAH that developed with unusual symptoms of hypoglycemia and hypothermia shortly after delivery. Previous reports have shown that the most common and initial complaints of LAH patients are symptoms of sellar compression, represented by headache, vomiting and visual disturbances.2,4,5 We present the clinical course of our case and discuss clinical considerations associated with this rare condition.
Received: September 27 2015. Accepted: November 18 2015. Correspondence: Dr Shinsuke Koyama, Department of Obstetrics and Gynecology, Osaka Rosai Hospital, 1179-3 Nagasone-cho, Kita-ku, Sakai, Osaka 591-8025, Japan. Email: [email protected]
© 2016 Japan Society of Obstetrics and Gynecology
467
S. Koyama et al.
Case Report A healthy 37-year-old woman, gravida 1 para 0, conceived spontaneously. Her clinical course of pregnancy was uneventful until the event. She had no subjective symptoms, such as headache or visual disturbance, throughout her perinatal period. At 38 weeks’ gestation, she delivered her baby by induced labor due to mild preeclampsia. At the time of delivery, vacuum extraction was performed because of non-reassuring fetal status caused by placental abruption. She bled 1800 mL but went into shock (blood pressure, 75/35 mmHg; pulse rate (PR), 130 b.p.m.), so we transfused her with a total of 6 units of red blood cells to stabilize her clinical condition. Circulatory collapse observed was greater than that expected from her blood loss and, thus, we were suspicious of some underlying condition being present. In fact, the next morning (12 h after delivery), she suddenly presented symptoms of hypoglycemia (blood sugar (BS), 35 mg/dL) and hypothermia (body temperature (BT), 35.1 °C). Blood testing showed hypoadrenalism and hypothyroidism; adrenocorticotropic hormone (ACTH),
