Letters to the Editor
Annals of Indian Academy of Neurology ¦ Volume 22 ¦ Issue 2 ¦ April-June 2019
248
Sir,
A 24 year old women with Parkinson’s disease (PD) was admitted to give her first birth. The patient’s history revealed that the patient had been suffering tremor of the right hand and slowness of movement due to a head trauma that she experienced at the age of 9 years. The patient presented to our polyclinic at the gestational week of 37 according to the last menstrual period. The patient was still using levodopa– benserazide 250 mg/day, and the neurological examination was normal except for bradymimia, bradykinesia. The patient was spontaneously pregnant after 5 years of infertility and had no clinical problems during the prenatal follow-up. The ultrasonographic measurements were consistent with 34 weeks of gestation. The patient had a spontaneous vaginal birth and delivered a live female baby weighing 2.050 g with an APGAR score of 7/9. The baby had a heart rate of 130/min, respiratory rate of 60/min, and oxygen saturation of 95%. On serial follow-up, the baby had excess secretions in the airway and developed tachypnea. An arterial blood gas revealed pH: 7.13,
PCO2: 60.5 mmHg, PO2: 47 mmHg, and HCO3: 17.3 mEq/l.
Depending on these outcomes, the baby was intubated, an umbilical vein catheter was inserted, and fluid resuscitation was started with 10% dextrose 80 cc/kg/day. Surfactant was given since the complete atelectasis of the left hemothorax was seen on the chest X-ray. The baby was monitored in the incubator and an orogastric tube was inserted since the excess secretions in the airway were still continuing. The patient was considered to have esophageal atresia since the orogastric tube did not progress and was seen to curl up at the level of upper-middle esophagus in the chest X-ray. A consultation was made with the ear–nose–throat clinic regarding the facial anomalies, but no pathology was detected. The baby was then transferred to a tertiary-level neonatal intensive care unit and was operatively treated with a diagnosis of esophageal atresia. The baby was discharged after the feeding problem was treated.
PD is a common late-life neurodegenerative disorder. About 5% of PD patients have an onset younger than 40 years of age. The young-onset below the age of 20 is known as juvenile PD
and is more common in men than in women.[1] The coexistence
of PD and pregnancy is rare. To our knowledge, about thirty cases have been reported in the literature.[2]
In this report, we present a case with the coexistence of PD and pregnancy who used levodopa–benserazide during pregnancy and whose neonate was detected with esophageal atresia, which has never been reported in the literature.
In patients with severe and advanced-stage PD, performing daily activities and maintaining a proper posture may become difficult during pregnancy.[2,3]
The combination of carbidopa or benserazide with levodopa appears to be safe, and no teratogenic effect of these combinations has been reported. Studies have shown that levodopa crosses the placenta, but carbidopa does not cross the placenta. In a previous study, an infant with prior exposure to levodopa was detected with osteomalacia, but no causal relationship was established.[4,5]
In the case presented, although the levodopa–benserazide therapy caused no complication during pregnancy, esophageal atresia occurred in the neonate, which could not be completely attributed to the therapy. Accordingly, this is the first study to report the presence of esophageal atresia in the neonate of a mother with PD who used levodopa–benserazide during pregnancy. Nevertheless, there has been no case report regarding the use of ropinirole or apomorphine, and there is scarce information in the literature regarding the use of selegiline and rasagiline.[4,5]
In the case presented, although the levodopa–benserazide therapy caused no complication during pregnancy, esophageal atresia occurred in the neonate, which could not be completely attributed to the therapy. Accordingly, this is the first study to report the presence of esophageal atresia in the neonate of a mother with PD who used levodopa–benserazide during pregnancy. Nevertheless, further studies are needed to investigate the relationship between neonatal esophageal atresia and the coexistence of PD and pregnancy.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
Mehmet Hamamcı, Kaya Yücesoy1, Nur Pekmezci2,
Zeynep Erdoğan Çetin3, Refah Sayın4 Departments of Neurology, 1Gynecology and Obstetrics, 2Pediatric Diseases and 3Otorhinolaryngology, Ardahan State Hospital,
Ardahan, 4Department of Neurology, Medical School,
Ufuk University, Ankara, Turkey
Address for correspondence: Dr. Mehmet Hamamcı,
Department of Neurology, Bozok University Medical School, Yozgat, Turkey. Email: [email protected]
Esophageal Atresia in a Newborn Born to a Parkinson’s Mother
Letters to the Editor
Annals of Indian Academy of Neurology ¦ Volume 22 ¦ Issue 2 ¦ April-June 2019 249
R
efeRences1. Emre M, Hanağası HA, Şahin HA, Yazıcı J. Movement Disorders. In: Öge AE, Baykan B, editors. Nöroloji. İstanbul: Nobel Tıp Kitapevleri; 2011. p. 513-38.
2. Asha B, Hansali N, Apoorva P. Successful birth of an IVF baby in a patient with Parkinson’s disease. J Hum Reprod Sci 2010;3:42-3. 3. Jacquemard F, Palaric JC, Allain H, Giraud JR. Parkinson disease and
pregnancy. Apropos of a case. J Gynecol Obstet Biol Reprod (Paris) 1990;19:461-3.
4. Lamichhane D, Narayanan NS, Gonzalez-Alegre P. Two cases of pregnancy in Parkinson’s disease. Parkinsonism Relat Disord
This is an open access journal, and articles are distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as appropriate credit is given and the new creations are licensed under the identical terms.
DOI: 10.4103/aian.AIAN_356_18
2014;20:239-40.
5. Robottom BJ, Mullins RJ, Shulman LM. Pregnancy in Parkinson’s disease: Case report and discussion. Expert Rev Neurother 2008;8:1799-805.
Comments on “Short‑lasting, unilateral, neuralgiform headache
attacks”
Sir,
We have a few comments to some statements in a recent article in your journal,[1] largely dealing with SUNCT.
SUNCT was originally described by our group.[2] It is an
acronym: “Shortlasting, Unilateral, Neuralgiform Headache Attacks with Conjunctival Injection, Tearing, Sweating, and Rhinorrhea.”[2]
Comment I: The authors[1] do not cite the acronym[2] correctly,
in that they have left out the three last words of the title: “-sweating, and rhinorrhea” (two first lines, ”Introduction”[1]).
In that way, the readers’ insight into the full breadth of the autonomic involvement in SUNCT is hindered. This involvement does not only consist of conjunctival injection and tearing but also of forehead sweating and rhinorrhea and, besides, a multitude of other autonomic abnormalities.[3] These
facts are essential for the understanding of SUNCT and for the understanding of the disagreement between the authors and the commentators. The same misinformation is found several times later in the article. This tends to show that we are not faced with a casual mishap, but with a systematic trend. We certainly hope that similar citation blunders will not happen again.
Comment II: SUNCT is characterized by unilateral, short-lasting spikes of pain in the ocular/periocular area. A perhaps even more characteristic trait of SUNCT is the coexistence of ipsilateral, cranial autonomic disturbances. They are invariably/close-to-invariably present. The ocular pulse amplitudes start increasing a couple seconds prior to the pain during attack, and the amplitude increase outlasts the pain by a couple seconds.[3] This observation contributes
to emphasizing the importance of the autonomic aberration in SUNCT. The amplitude increase does not seem to be secondary to the pain – it may even be vice versa. SUNCT with this unique, almost diagnostic combination, must not be linked with headache disorders having minimal autonomic disturbances.
Short-lasting unilateral neuralgiform headache attacks [SUNA] with cranial autonomic symptoms [e.g., 1] is a more recent invention. The inventors try to link it with SUNCT. SUNA is, if anything, rare. In a recent review,[4] there were about six
times as many SUNCT as SUNA cases.
In this situation, the present authors come up with a proposal: In this situation, the present authors[1] come up with a
proposal for a superstructure, even with a new confusing abbreviation, that is, SUNHA, which should comprise many (all?) short-lasting, unilateral headaches, including trigeminal neuralgia (TN). It is stated that the clinical picture of SUNCT is rather similar to that of TN. Indeed, duration of pain attacks of SUNCT and TN may overlap. But, and this is tremendously important – the autonomic involvement differs vastly. The pure number of solitary autonomic phenomena, that is, conjunctival injection, lacrimation, and rhinorrhea, was counted in two adequately large series of TN, first branch, and SUNCT. In SUNCT, autonomic phenomena were much more prevalent than in TN (P < 0.0000005; Chi-square test). And, when present, such phenomena are much more profuse in SUNCT. Although the last variables have not been quantified, this difference is obvious, with massive abnormalities in SUNCT, and mostly hardly noticeable
abnormalities in TN:[5] This difference in appearance
tells about the fundamental differences in underlying mechanisms. The main trait of SUNCT is probably the autonomic aberrations. It can probably be stated as firmly as this: Without these autonomic abnormalities: No SUNCT. SUNCT can thus not, just like that, be lumped with any short-lasting, unilateral headache.
Moreover, in SUNCT, there is a poor effect of carbamazepine and oxcarbazepine,[4] whereas in TN there is an excellent
effect.[6] Nocturnal attacks are more frequent in TN than in
SUNCT. Sex preponderance differs clearly in the two disorders. These are all significant differences. Trying to lump SUNCT, which is characterized by heavy autonomic involvement with
